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[[Translated article]]Nadolol for Infantile Hemangiomas Previously Treated with Propranolol

Fetched: December 5th, 2023, 5:01am GMT by M Colmenero Sendra

Actas Dermosifiliogr. 2023 Dec 2:S0001-7310(23)00957-2. doi: 10.1016/j.ad.2023.11.015. Online ahead of print.

NO ABSTRACT

PMID:38048940 | DOI:10.1016/j.ad.2023.11.015

Intense 18F-PSMA-1007 Uptake of Splenic Hemangioma

Fetched: December 5th, 2023, 5:01am GMT by Guorong Jia

Clin Nucl Med. 2023 Dec 1. doi: 10.1097/RLU.0000000000004995. Online ahead of print.

ABSTRACT

A 77-year-old man was referred for 18F-PSMA-1007 PET/CT scan for initial staging of biopsy-proved prostate adenocarcinoma. 18F-PSMA-1007 PET/CT showed focal intense 18F-PSMA-1007 of the prostate adenocarcinoma and a focal intense activity (SUVmax, 27) in the spleen. The 18F-PSMA-1007-avid splenic lesion corresponded to a splenic hemangioma, which was initially detected on contrast-enhanced CT 7 months ago and unchanged in size and enhancement pattern on follow-up contrast-enhanced CT. This case indicates that splenic hemangioma should be included in the differential diagnosis of PSMA-avid splenic lesions.

PMID:38048527 | DOI:10.1097/RLU.0000000000004995

Permalink for '18F-Fluorodeoxyglucose Positron Emission Tomography/Computed Tomography in a Rare Case of Epithelioid Hemangioendothelioma - A Diagnostic Challenge'

18F-Fluorodeoxyglucose Positron Emission Tomography/Computed Tomography in a Rare Case of Epithelioid Hemangioendothelioma - A Diagnostic Challenge

Fetched: December 5th, 2023, 5:01am GMT by Fahad Nisamudeen

Indian J Nucl Med. 2023 Jul-Sep;38(3):282-285. doi: 10.4103/ijnm.ijnm_171_22. Epub 2023 Oct 10.

ABSTRACT

Our case highlights the 18F-fluorodeoxyglucose positron emission tomography/computed tomography (18F-FDG PET/CT) scan findings in a rare case of biopsy-proven epithelioid hemangioendothelioma (EHE) in a 66-year-old woman with multi-organ involvement (lung, liver, and bone) who was subsequently treated with palliative radiation therapy and oral pazopanib. Furthermore, follow-up 18F-FDG PET/CT findings are detailed. EHE is a rare malignant vascular neoplasm (<1% of all vascular tumors) with an epithelioid and histiocytoid appearance arising from the vascular endothelial and preendothelial cells.

PMID:38046956 | PMC:PMC10693370 | DOI:10.4103/ijnm.ijnm_171_22

Benign Orofacial Vascular Anomalies: Review of 47 Cases in Enugu, Nigeria

Fetched: December 5th, 2023, 5:01am GMT by M C Nwoga

Niger J Clin Pract. 2023 Nov 1;26(11):1723-1727. doi: 10.4103/njcp.njcp_332_23. Epub 2023 Dec 4.

ABSTRACT

BACKGROUND: A study of oral vascular anomalies has not been conducted in Nigeria to provide baseline data for comparison with reports in the literature.

AIMS: To study the prevalence and distribution of benign orofacial vascular anomalies at a tertiary hospital in Enugu.

MATERIALS AND METHODS: This is a 10-year retrospective observational study of consecutive patients with orofacial vascular anomalies, diagnosed by histology. The clinic-pathologic information was obtained from records archived in the department, and descriptive analysis was used to determine the frequency, tables for categorical variables, and a Chi-square test to determine the statistical significance.

RESULT: There were 47 cases of benign vascular anomalies out of 897 orofacial lesions giving a prevalence of 5.2%. There were 35.4% (17) male and 64.6% (31) female patients. The mean age in this series was 37.4 ± 19.8 (range: 1 to 76 years). Pyogenic granuloma was the most common vascular lesion 78.7% (37), followed by hemangioma 14.9% (7) and lymphangioma 6.4% (3). The gingiva was the most frequent site of oral occurrence 65.9% (31), especially maxillary gingivae 48.9% (23). The type of orofacial vascular anomalies was significantly associated with the anatomical site of occurrence, P = 0.00. The mean ages for the occurrence of pyogenic granuloma, hemangioma, and lymphangioma were 37.7 ± 18.3, 50.7 ± 16.9 years, and 3.3 ± 3.2 years, respectively. Pain was a frequent occurrence in 36.2% (17) of anomalies.

CONCLUSION: Oral vascular anomalies predominantly presented as pyogenic granuloma on the gingivae, while oral hemangioma was observed in adults, and lymphangioma was infrequent.

PMID:38044779 | DOI:10.4103/njcp.njcp_332_23

Modified orbitozygomatic craniotomy with a single burr hole in the alternative sphenoid ridge keyhole

Fetched: December 4th, 2023, 5:01am GMT by Semyon A Melchenko

Neurochirurgie. 2023 Dec 2;70(1):101514. doi: 10.1016/j.neuchi.2023.101514. Online ahead of print.

ABSTRACT

BACKGROUND: One-piece modified orbitozygomatic approach (OZA) is an extended version of the pterional approach that also includes orbital walls and frontal process of the zygomatic bone. For this craniotomy one burr hole must be placed in MacCarty keyhole and another - in the temporal region.

OBJECTIVE: To develop a technique of the one-piece modified OZA with single a burr hole in the alternative sphenoid ridge keyhole that allows access to orbit, anterior cranial fossa and middle cranial fossa and apply it intraoperatively.

METHODS: A single human head specimen was used. The dissection was performed using standard surgical instruments high-speed Stryker drill. Every stage of the approach was photographed. We also report a surgical case of a patient with orbital cavernous hemangioma that was resected using the described technique.

RESULTS: The technique of the one-piece modified OZA with a single burr hole in the alternative sphenoid ridge keyhole is described, and its advantages and limitations are analyzed. The technique is used to totally resect an orbital cavernous hemangioma with good functional and cosmetic result.

CONCLUSION: Modified OZA with a single burr hole in the sphenoid ridge keyhole is possible and may be an alternative to the classic technique. The advantages of this variation are the placement of just one burr hole and the preservation of a larger portion of the orbital roof. The latter facilitates better bone reconstruction and better cosmetic outcome. Disadvantages are the difficulty of identifying the location of the sphenoid ridge keyhole and risk of damaging the dura.

PMID:38043139 | DOI:10.1016/j.neuchi.2023.101514

Application of Lasers in Vascular Anomalies

Fetched: December 1st, 2023, 5:02am GMT by Uddhav Anandrao Patil

Indian J Plast Surg. 2023 Nov 3;56(5):395-404. doi: 10.1055/s-0043-1775871. eCollection 2023 Oct.

ABSTRACT

Laser technology has significantly improved giving better results, which in turn has led to an increase in the indications for laser therapy. Vascular anomalies comprise vascular tumors and malformations. They are classified according to the type of vessels involved including arteries, capillaries, postcapillary venules, veins, lymphatic vessels, and a combination of two or more of these. Laser needs a chromophore to get absorbed and act. Hemoglobin, both oxy and deoxy, is one of the naturally occurring chromophore that is abundant in vascular lesions. Therefore, in most of the vascular lesions, lasers can bring improvement of a varying degree, while for superficial hemangiomas and port wine stain (PWS) laser treatment is now the standard of care. However, even though there is increase in the use of lasers in clinical practice, many surgeons are still unaware of its versatility and they are unsure about its safety. This article provides a brief overview of laser and intense pulsed light (IPL) technology, and describes the key principles in using these energy sources in vascular malformations. Readers are also familiarized with possible adverse effects and measures to prevent and treat them.

PMID:38026771 | PMC:PMC10663075 | DOI:10.1055/s-0043-1775871

Rare adrenal cavernous hemangioma: a case report highlighting diagnostic challenges

Fetched: December 1st, 2023, 5:02am GMT by Ryan Michael Antar

Front Surg. 2023 Nov 10;10:1293925. doi: 10.3389/fsurg.2023.1293925. eCollection 2023.

ABSTRACT

INTRODUCTION: Adrenal cavernous hemangiomas are rare benign vascular tumors that pose significant diagnostic challenges. Despite their benign nature, features overlapping with malignancies often complicate management decisions.

CASE PRESENTATION: A 64-year-old male presented with a 4.4 cm necrotic left adrenal mass discovered incidentally on imaging. His medical history included papillary thyroid carcinoma, with subsequent thyroidectomy and radioactive iodine ablation. Evaluations for hiccups revealed multiple lung nodules, hypertrophic cardiomyopathy, and anemia. Given the patient's previous cancer history, elevated aldosterone/renin ratio, and mass size, our multidisciplinary tumor board decided to proceed with a left adrenalectomy. Post-surgical pathology confirmed a diagnosis of adrenal cavernous hemangioma.

CONCLUSION: The occurrence of ambiguous adrenal mass with other pathologies, such as our patient's papillary thyroid carcinoma, complicates the diagnostic and therapeutic landscape. As demonstrated in our case, opting for surgery remains a viable solution for adrenal cavernous hemangiomas, especially for masses greater than 4 cm. Interdisciplinary collaboration, exemplified by our tumor board's decision-making process, is crucial for optimal management. This case underscores the need for a multifaceted approach when confronting adrenal masses with such diagnostic ambiguity.

PMID:38026486 | PMC:PMC10667707 | DOI:10.3389/fsurg.2023.1293925

Unique Cobb syndrome with Kaposi hemangioendothelioma/tufted angioma as dominant phenotype: a case report

Fetched: December 1st, 2023, 5:02am GMT by Na Lin

World J Pediatr Surg. 2023 Nov 17;6(4):e000695. doi: 10.1136/wjps-2023-000695. eCollection 2023.

NO ABSTRACT

PMID:38025901 | PMC:PMC10660422 | DOI:10.1136/wjps-2023-000695

Two Discrete Intra-articular Synovial Hemangiomas of a Knee Joint Presenting as Chronic Knee Pain

Fetched: December 1st, 2023, 5:02am GMT by Mantu Jain

J Orthop Case Rep. 2023 Nov;13(11):13-17. doi: 10.13107/jocr.2023.v13.i11.3988.

ABSTRACT

INTRODUCTION: Synovial hemangioma (SH) is a rare benign soft-tissue tumor of vascular origin. That can be extraarticular, juxta-articular, or intraarticular. The knee joint is the most common joint involved. Symptoms can be variable and diagnosis can be made using magnetic resonance imaging. Differentials can be ruled out by biopsy and the gold standard treatment is complete excision of the lesion.

CASE REPORT: A 20-year-old male presented to outpatient clinic with a chief complaint of right-sided anterior knee pain for 2 years aggravated for 2 months. Ultrasonography and magnetic resonance imaging showed two discrete intra-articular lesions. The lesion was hypointense on T1W and hyperintense on T2W and STIR sequence that was enhancing on contrast. There were adjoining dilated vessels. With a provisional diagnosis of hemangioma, FNAC was done and then complete excision was done using a medial parapatellar arthrotomy. The patient is doing well at 1-year follow-up.

CONCLUSION: Knee joint SH is a rare presentation to orthopedic outdoors and has slight female predominance with pre-existing history of trauma. In the present study, both cases were of patella-femoral type (anterior and infra-patellar fat pad). For such lesions, en bloc excision is the gold standard procedure to prevent recurrence, same procedure was followed in our study, and good functional outcome was achieved.

PMID:38025371 | PMC:PMC10664226 | DOI:10.13107/jocr.2023.v13.i11.3988

Penile vascular anomalies: A retrospective single center study and cumulative analysis of studies from China

Fetched: December 1st, 2023, 5:02am GMT by Chong Ma

Asian J Urol. 2023 Oct;10(4):555-562. doi: 10.1016/j.ajur.2023.03.006. Epub 2023 Aug 10.

ABSTRACT

OBJECTIVE: Penile vascular anomalies (PVAs) or hemangioma can arouse patient concern about aesthetics and cause symptoms like bleeding and sexual dysfunction. However, its low incidence and the deficiency of large-volume studies hinder urologists from making informed decisions. This study aimed to investigate the clinical features and treatment experience of PVAs at the Seventh Medical Center of PLA General Hospital, Beijing, China. Furthermore, by systematically analysis of studies on PVAs in Chinese people, we aimed to provide novel insights on the management of this condition.

METHODS: We retrospectively investigated clinical features and pathology of surgery-treated PVAs at our center. Moreover, by systemically reviewing the literature from PubMed and the three largest medical databases (China National Knowledge Infrastructure, Wan Fang, and Chinese Medical Journal Database) in China, we analyzed the clinical features and various therapies of PVAs in Chinese people.

RESULTS: Between March 1, 2018 and March 1, 2023, a total of 356 cases with vascular anomalies were treated with surgery at out center. Only seven (2.0%) cases had lesions involving the perineum and external genitalia. All the seven cases were pathologically benign and demonstrated no recurrence over a follow-up period of 1-52 months (median 14 months). A total of 410 cases from 44 studies were selected in the cumulative analysis. Most patients (92.4%) diagnosed with PVAs were asymptomatic, and 68.8% of the patients were treated with sclerotherapy. As to the pathology, 57.1% were venous malformation.

CONCLUSION: The most common PVA is venous malformation and the majority of patients are asymptomatic. Sclerotherapy and laser have emerged as viable options for treating small lesions. Surgery still has its role in treating large lesions and obtaining pathology. Although PVAs often relapse or demand multiple treatments, the prognosis is favorable.

PMID:38024431 | PMC:PMC10659982 | DOI:10.1016/j.ajur.2023.03.006

A Liver-Derived Vascular Lesion: Hepatic Hemangioma or Hepatic Epithelioid Hemangioendothelioma?

Fetched: December 1st, 2023, 5:02am GMT by Roham Gholami

Middle East J Dig Dis. 2023 Jul;15(3):210-213. doi: 10.34172/mejdd.2023.347. Epub 2023 Jul 30.

ABSTRACT

Hepatic epithelioid hemangioendothelioma (EHE) is an uncommon vascular endothelial cell tumor of the liver with numerous symptoms and features. The median affected age is 41, and females are more frequently affected than men. In the following article, a 37-year-old nurse is presented who was referred to the hospital with severe right upper quadrant pain. She had been misdiagnosed with hepatic hemangioma for years, which finally turned out to be hepatic EHE. Liver transplantation has been recognized as the therapeutic method of choice due to the considerable extent of liver involvement and nonresponse to medications.

PMID:38023470 | PMC:PMC10660322 | DOI:10.34172/mejdd.2023.347

Permalink for 'Corrigendum: Case Report: Surgical treatment of a primary giant epithelioid hemangioendothelioma of the spine with total en-bloc spondylectomy'

Corrigendum: Case Report: Surgical treatment of a primary giant epithelioid hemangioendothelioma of the spine with total en-bloc spondylectomy

Fetched: December 1st, 2023, 5:02am GMT by Wanbao Ge

Front Oncol. 2023 Nov 9;13:1308503. doi: 10.3389/fonc.2023.1308503. eCollection 2023.

ABSTRACT

[This corrects the article DOI: 10.3389/fonc.2023.1109643.].

PMID:38023208 | PMC:PMC10666915 | DOI:10.3389/fonc.2023.1308503

Scar Endometriosis vs Hemangioma: A Diagnostic Conundrum

Fetched: December 1st, 2023, 5:02am GMT by Hrithik Dakssesh Putta Nagarajan

Cureus. 2023 Oct 25;15(10):e47674. doi: 10.7759/cureus.47674. eCollection 2023 Oct.

ABSTRACT

Scar endometriosis refers to the presence of endometrial glands and stroma at the site of a scar. Hemangiomas, on the other hand, are benign vascular tumors. In this case report, we unravel the clinical enigma around a patient who presented with a painful mass at the previous cesarean section scar site. Initially, we were confident that this was ectopic endometrium presenting as scar endometriosis. However, our journey took an unexpected turn when histopathological findings contradicted our clinical suspicions. Here, we delve into the intricate details of this captivating case, shedding light on the complexities of the diagnosis we faced.

PMID:38021669 | PMC:PMC10672459 | DOI:10.7759/cureus.47674

Cavernous Sinus capillary hemangioma: Case report and literature review

Fetched: December 1st, 2023, 5:02am GMT by Mustafa Caglar Sahin

Brain Spine. 2023 Jul 1;3:101776. doi: 10.1016/j.bas.2023.101776. eCollection 2023.

ABSTRACT

INTRODUCTION: Capillary hemangioma of the cavernous sinus is considered a benign vascular skull base tumor. In this case report, a patient with capillary hemangioma located in the cavernous sinus who was operated on with endoscopic endonasal approach is presented.

RESEARCH QUESTION: What should be considered when planning the treatment for capillary hemangiomas located in the cavernous sinus?

MATERIAL AND METHODS: The treatment approaches applied in 12 patients with a diagnosis of capillary hemangioma located in the cavernous sinus, reported in the English literature, are reviewed in this article. We presented the clinical and radiological results of the treatments.

RESULTS: Cavernous sinus capillary hemangiomas are considered benign tumors. However, surgical treatment is preferred in the foreground because of the neurological deficits due to the compression effect caused by the mass. In the case reported by Masman et al. for the first time in the literature in 2021, they provided total excision with the endoscopic endonasal approach in the patient they first diagnosed by taking a biopsy with the endoscopic endonasal approach. In our case report, we provided total excision with endoscopic endonasal transsphenoidal approach.

DISCUSSION AND CONCLUSION: Many factors should be considered when deciding on the treatment method in these patients. Although radiotherapy is quite effective in the treatment of capillary hemangioma, surgery should be preferred, especially in symptomatic patients. In patients who cannot undergo surgery, radiotherapy is very valuable, although the long-term results are not yet known.

PMID:38021022 | PMC:PMC10668058 | DOI:10.1016/j.bas.2023.101776

Intramuscular cavernous malformation in the temporalis muscle: Diagnosis and treatment of a rare tumor in a rare site

Fetched: December 1st, 2023, 5:02am GMT by Mohammad Jafarian

Clin Case Rep. 2023 Nov 27;11(12):e8267. doi: 10.1002/ccr3.8267. eCollection 2023 Dec.

ABSTRACT

In this case report we present a very rare case of intramuscular cavernous hemangioma in the temporalis muscle which was successfully managed with surgical excision with no evidence of recurrence in follow-up.

PMID:38033698 | PMC:PMC10682246 | DOI:10.1002/ccr3.8267

Lipid-poor vertebral hemangioma mimicking a vertebral metastasis of cervical carcinoma

Fetched: December 1st, 2023, 5:02am GMT by Wend-Yam Mohamed Traore

Clin Case Rep. 2023 Nov 27;11(12):e8195. doi: 10.1002/ccr3.8195. eCollection 2023 Dec.

ABSTRACT

Vertebral hemangiomas are classified into typical and atypical forms. On imaging, atypical forms may look like malignant lesions. The confrontation of data from different imaging modalities (MRI; CT; scintigraphy) facilitates the diagnosis.

PMID:38033692 | PMC:PMC10682230 | DOI:10.1002/ccr3.8195

Female splenogonadal fusion: A PLEA for conservative management

Fetched: December 1st, 2023, 5:02am GMT by Silvia Ceccanti

Urol Case Rep. 2023 Nov 16;51:102620. doi: 10.1016/j.eucr.2023.102620. eCollection 2023 Nov.

ABSTRACT

Splenogonadal fusion in female patients is seldom reported. We describe a 6-month-old girl who represents the youngest living female with splenogonadal fusion reported to date. The lesion was diagnosed as an incidental finding during screening abdominal ultrasonography performed for a vulvar infantile hemangioma. A tail-like structure with splenic echotexture connecting a normally located spleen and the left ovary was detected and better characterized by MRI. We also reviewed the pertinent literature on managing this usually asymptomatic condition, especially in female patients. Greater professionals' awareness of this benign anomaly is paramount to avoid the unnecessary removal of an otherwise normal gonad.

PMID:38033426 | PMC:PMC10686837 | DOI:10.1016/j.eucr.2023.102620

Intralesional radiofrequency ablation followed by lip reduction surgery for infantile haemangioma of the lip

Fetched: December 1st, 2023, 5:02am GMT by Rhea Ahuja

Indian J Dermatol Venereol Leprol. 2023 Nov 23:1-3. doi: 10.25259/IJDVL_394_2023. Online ahead of print.

NO ABSTRACT

PMID:38031675 | DOI:10.25259/IJDVL_394_2023

Spinal epidural cavernous hemangiomas in the lumbar spine: A case report

Fetched: December 1st, 2023, 5:02am GMT by Dingyan Zhao

Int J Surg Case Rep. 2023 Nov 22;114:109040. doi: 10.1016/j.ijscr.2023.109040. Online ahead of print.

ABSTRACT

INTRODUCTION: Spinal epidural cavernous hemangiomas (SECHs) are relatively rare intradural epidural lesions of the spinal canal, and those occurring in the lumbar spine are even rarer.

CASE PRESENTATION: A 60-year-old man presented for low back pain with right leg pain. His pee and feces were both normal but symptoms were very similar to a typical lumbar disc herniation. The MRI findings suggest an epidural space of unknown nature in the spinal cord at the L2-3 level and a definite diagnosis of SECHs was made by postoperative pathological examination.

CLINICAL DISCUSSION: Patients who are suspected of having SECHs should undergo initial classification and differential diagnosis based on MRI imaging features. It is crucial to identify the responsible segment in correlation with the presenting symptoms. During surgery, the primary objective should be the complete removal of the mass, while taking utmost care to protect the nerves. Dynamic stabilization systems, utilizing pedicle rods, can be considered as one of the treatment options for such patients.

CONCLUSION: Patients presenting with low back pain and neurological symptoms should undergo MRI, and diagnosed with SECHs should undergo early surgical intervention. For patients with an intradural mass in the spinal canal, complete resection should be performed while prioritizing nerve protection.

PMID:38029575 | DOI:10.1016/j.ijscr.2023.109040

Capillary Hemangioma Managed With Laser Ablation: A Case Report

Fetched: December 1st, 2023, 5:02am GMT by Aravind Warrier S

J Lasers Med Sci. 2023 Sep 30;14:e39. doi: 10.34172/jlms.2023.39. eCollection 2023.

ABSTRACT

Introduction: Hemangiomas are hamartomatous benign tumours of the vascular tissue, which are common among infants and adolescents and are found in the head and neck region. Oral hemangiomas are infrequently encountered in the palate. Baseline soft tissue imaging must be considered for an appropriate diagnosis and treatment approach. Case Report: A 68-year-old female presented with a painless growth in the roof of her mouth that gradually increased in its size. She was subjected to intraoral ultrasonography and was diagnosed with capillary hemangioma. In this instance, laser ablation was chosen as the preferred treatment due to the complications associated with the conventional surgery. Conclusion: This case report enlightens the systematic diagnostic approach and treatment plan.

PMID:38028886 | PMC:PMC10658107 | DOI:10.34172/jlms.2023.39

A rare cause of rectal bleeding in a 48-year-old lupus patient

Fetched: December 1st, 2023, 5:02am GMT by Oumayma El Eulj

Radiol Case Rep. 2023 Nov 8;19(1):315-318. doi: 10.1016/j.radcr.2023.10.029. eCollection 2024 Jan.

ABSTRACT

Cavernous hemangiomas represent a rare benign cause of rectal bleeding. It corresponds to a vascular malformation that can be located anywhere in the gastrointestinal tract. Our paper reports the case of a 48-year-old woman treated for cutaneous lupus who presented to our department with intermittent moderate rectal bleeding. The Hemoglobin level was normal. A colonoscopy showed a congestive nodular red-purple vascular formation. At Computerized tomography (CT)-Scan, the lesion appeared as an irregular thickening of the posterior rectal wall, invading the meso-rectum. Magnetic resonance imaging (MRI) showed a submucosal mass of the rectum containing phleboliths with progressive enhancement in the T2 sequence. Diagnosis of rectal cavernous hemangioma was confirmed. Surgery is the most appropriate treatment for this condition, but since bleeding was not important and had no biological repercussions, multidisciplinary experts meeting decided not to operate on the patient and continue monitoring.

PMID:38028309 | PMC:PMC10661552 | DOI:10.1016/j.radcr.2023.10.029

Intramedullary spinal cord hemangioma: A rare case report

Fetched: December 1st, 2023, 5:02am GMT by Tam Truong Faulds

Radiol Case Rep. 2023 Nov 3;19(1):223-226. doi: 10.1016/j.radcr.2023.10.006. eCollection 2024 Jan.

ABSTRACT

Spinal cord hemangiomas are rare vascular malformations that can cause neurological deficits. We report a case of a 57-year-old male who previously underwent spondylolisthesis reduction for treatment of a 30% (1 cm) anterolisthesis of L4-L5 with continued neurological deficit post-operatively. The patient still reported bilateral lower extremity weakness, and he was found to have an additional ovoid intramedullary lesion at the T2-T3 level, pathologically consistent with a hemangioma. The patient underwent a T2-T3 laminectomy, and the lesion was resected without any complications. Histopathological examination confirmed the diagnosis of hemangioma. Postoperatively, the patient showed significant improvement in his lower extremity weakness and back pain. Hemangiomas are rare lesions that account for less than 5% of all spinal cord tumors. They consist of vessels similar to those of embryonic capillaries and can cause pressure on the surrounding neural tissue, leading to neurological deficits. MRI is the imaging modality of choice for diagnosing spinal cord hemangiomas. Hemangiomas typically demonstrate mixed signal intensities on T1-weighted images, depending on the fat content of the lesion. They usually demonstrate high T2 signal intensity due to high water content, and avid contrast enhancement due to high vascularity. Surgical resection is the treatment of choice for symptomatic hemangiomas, and complete resection is associated with a good prognosis.

PMID:38028300 | PMC:PMC10630760 | DOI:10.1016/j.radcr.2023.10.006

Elevated 18F-AIF-FAPI-04 Uptake in Hepatic Hemangioma

Fetched: November 29th, 2023, 5:02am GMT by Yuyun Sun

Clin Nucl Med. 2023 Nov 27. doi: 10.1097/RLU.0000000000004965. Online ahead of print.

ABSTRACT

A 67-year-old woman who was diagnosed with intrahepatic cholangiocellular carcinoma (CCC) by biopsy underwent 18F-FDG and 18F-AIF-FAPI-04 PET/CT for initial and treatment assessment. In addition to CCC, she had a history of hepatic hemangioma for 3 years. 18F-FDG PET/CT images showed increased uptake in CCC, but no uptake in hemangiomas. However, images on 18F-AIF-FAPI-04 PET/CT indicated negative 18F-AIF-FAPI-04 uptake in CCC, but intense activity in hemangiomas. Our case illustrates that hepatic hemangioma demonstrated intense 18F-AIF-FAPI-04 uptake, and final diagnosis should be made with caution.

PMID:38015630 | DOI:10.1097/RLU.0000000000004965

Transpedicular Onyx embolization of a thoracic hemangioma with robotic assistance: illustrative case

Fetched: November 29th, 2023, 5:02am GMT by Andrew M Hersh

J Neurosurg Case Lessons. 2023 Jun 26;5(26):CASE2348. doi: 10.3171/CASE2348. Print 2023 Jun 26.

ABSTRACT

BACKGROUND: Hemangiomas are common benign vascular lesions that rarely present with pain and neurological deficits. Symptomatic lesions are often treated with endovascular embolization. However, transarterial embolization can be technically challenging depending on the size and caliber of the vessels. Moreover, embolization can result in osteonecrosis and vertebral collapse.

OBSERVATIONS: Here the authors report the first case of a T10 vertebral hemangioma treated with transpedicular Onyx embolization aided by a robotic platform that guided pedicle cannulation and Craig needle placement. An intravenous catheter was attached to the needle and dimethylsulfoxide was infused, followed by Onyx under real-time fluoroscopy. Repeat angiography demonstrated significantly reduced contrast opacification of the vertebral body without compromise of the segmental artery. A T9-11 pedicle screw fixation was performed to optimize long-term stability. The patient's symptoms improved and was stable at the 6-month follow-up.

LESSONS: Transpedicular embolization of vertebral hemangiomas can be performed successfully under robotic navigation guidance, avoiding complications seen with the intra-arterial approach and allowing for simultaneous pedicle screw fixation to prevent collapse and delayed kyphotic deformity. During the same procedure, a biopsy specimen can be collected for pathology. This technique can help to alleviate patient symptoms while avoiding complications associated with transarterial embolization or open resection.

PMID:38015021 | PMC:PMC10550547 | DOI:10.3171/CASE2348

A giant head and neck hemangioma of the fetus: A case report

Fetched: November 29th, 2023, 5:02am GMT by Xueying Wang

Medicine (Baltimore). 2023 Nov 24;102(47):e35855. doi: 10.1097/MD.0000000000035855.

ABSTRACT

RATIONALE: Hemangioma is a common benign disease in clinical practice, but it is rare to find a giant hemangioma in the fetal period.

PATIENT CONCERNS: Here, we report a case of a giant hemangioma of the fetal head and neck measuring approximately 10.1 × 6.5 cm.

DIAGNOSES: At first, only ultrasonography was used to diagnose the suspected hemangioma. The pregnant woman refused to undergo further testing and requested induction of labor, after which the tumor was finally sent for pathological examination to confirm hemangioma.

INTERVENTIONS AND OUTCOMES: Additionally, the fetus developed severe edema (fluid accumulation in the thoracic, abdominal, and pericardial cavities), which can be fatal to the fetus. Finally, the mother refused to continue the pregnancy and underwent induction of labor with rivanol.

LESSONS: Most hemangiomas are small and asymptomatic. Giant hemangiomas are rare and associated with a variety of maternal and fetal complications. Therefore, this article aims to summarize the knowledge related to hemangioma through this case, strengthen doctors' understanding of this disease, and bring the attention of pregnant women to this disease to ensure early diagnosis and treatment and prevent a poor prognosis.

PMID:38013270 | PMC:PMC10681613 | DOI:10.1097/MD.0000000000035855

Pulmonary sclerosing pneumocytoma - approaching a solitary pulmonary nodule and the limitations of risk prediction models

Fetched: November 28th, 2023, 5:01am GMT by Kundan Reddy Saripalli

BMJ Case Rep. 2023 Nov 17;16(11):e257208. doi: 10.1136/bcr-2023-257208.

ABSTRACT

Our case is an asymptomatic, non-smoking, East Asian woman in her 40s presenting with a solitary pulmonary nodule (SPN). On imaging, the 1.7 cm solid SPN located in the left upper lobe, was rounded in morphology and moderately fluorodeoxyglucose avid. The clinical pretest probability of malignancy assessed by risk prediction models such as Brock (19.1%), Mayo Clinic (56.2%) and Herder (51.4%) was discordant. She underwent a percutaneous CT-guided needle biopsy, establishing a diagnosis of pulmonary sclerosing pneumocytoma (PSP). PSP is a rare benign lung neoplasm with indolent growth characteristics that has been described predominantly in non-smoking women. Our case illustrates the limitations of applying existing risk prediction models in Asia where the epidemiology and biology of lung cancer differ significantly from the Caucasian derivation cohorts. Additionally, the risk models do not account for tuberculosis, which is endemic in Asia and can mimic malignancy. Non-surgical lung biopsy remains useful in minimising unnecessary thoracotomy.

PMID:37977835 | PMC:PMC10660428 | DOI:10.1136/bcr-2023-257208

Diverse clinical presentations of pseudomyogenic hemangioendothelioma associated with EGFL7::FOSB fusion: a second case

Fetched: November 28th, 2023, 5:01am GMT by Bin Li

Histopathology. 2023 Nov 27. doi: 10.1111/his.15108. Online ahead of print.

NO ABSTRACT

PMID:38012540 | DOI:10.1111/his.15108

Yttrium-90 Radioembolization of a Large Hepatic Hemangioma

Fetched: November 28th, 2023, 5:01am GMT by Vikram F Gupta

Cardiovasc Intervent Radiol. 2023 Nov 27. doi: 10.1007/s00270-023-03615-7. Online ahead of print.

NO ABSTRACT

PMID:38010506 | DOI:10.1007/s00270-023-03615-7

Primary intraosseous cavernous hemangioma of the cranium: a case report

Fetched: November 28th, 2023, 5:01am GMT by E A Nenashev

Khirurgiia (Mosk). 2023;(11):113-117. doi: 10.17116/hirurgia2023111113.

ABSTRACT

Primary intraosseous cavernous hemangioma (PICH) is a rare benign vascular tumor. This neoplasm is common in the spine and less common in skull. Toynbee J. first described this tumor in 1845. PICH of the cranium does not always have typical X-ray features and should be always differentiated with other more common skull lesions. Surgical resection is preferable since total resection is followed by favorable prognosis. We present a 65-year-old patient with asymptomatic tumor of the right parietal bone. CT revealed osteolytic lesion that required total resection and skull repair. Histopathological analysis revealed intraosseous cavernous hemangioma.

PMID:38010025 | DOI:10.17116/hirurgia2023111113

Rapid low-cost hyperspectral imaging system for quantitative assessment of infantile hemangioma

Fetched: November 28th, 2023, 5:01am GMT by Sergei Perkov

J Biophotonics. 2023 Nov 27:e202300375. doi: 10.1002/jbio.202300375. Online ahead of print.

ABSTRACT

Hemangioma, the predominant benign tumor occurring in infancy, exhibits a wide range of prognoses and associated outcomes. The accurate determination of prognosis through noninvasive imaging modalities holds essential importance in enabling effective personalized treatment strategies and minimizing unnecessary surgical interventions for individual patients. The present study focuses on advancing the personalized prognosis of hemangioma by leveraging noninvasive optical sensing technologies by the development of a novel rapid hyperspectral sensor (image collection in 5 seconds, lateral resolution of 10 μm) that is capable of quantifying hemoglobin oxygenation and vascularization dynamics during the course of tumor evolution. We have developed a quantitative parameter for hemangioma assessment, that demonstrated agreement with the clinician's conclusion in 90% among all cases during clinical studies on 6 patients, who visited clinician from 2 to 4 times. The presented methodology have potential to be implemented as a supportive tool for accurate hemangioma diagnostics in clinics. This article is protected by copyright. All rights reserved.

PMID:38009761 | DOI:10.1002/jbio.202300375

Excessively High Chronic Propranolol Overdose in Infantile Hemangioma: A Case Report

Fetched: November 28th, 2023, 5:01am GMT by Heba Alshammari

Am J Case Rep. 2023 Nov 27;24:e941765. doi: 10.12659/AJCR.941765.

ABSTRACT

BACKGROUND Infantile hemangiomas are the most common benign tumors of childhood, occurring in approximately 5% of infants. Oral propranolol at 2 to 3 mg/kg daily is recommended for systemic treatment of high-risk infantile hemangiomas. Multiple propranolol formulations exist, and propranolol overdose can occur due to improper patient counseling. Propranolol acute toxicity in the pediatric population and its management are well described in the literature. However, data are lacking on chronic propranolol overdose and how to manage it, with the awareness that abrupt discontinuation of therapeutic doses of propranolol can lead to rebound sinus tachycardia. CASE REPORT A 7-month-old girl was prescribed a therapeutic dose of propranolol (1 mg/kg/day) to treat infantile hemangioma. However, due to an administration error, the patient received approximately 8 times the recommended dose (7.6 mg/kg/day for 2 months, then increased to 15.5 mg/kg/day for 2 weeks) and, surprisingly, remained asymptomatic. Her electrocardiogram was normal, and all routine laboratory tests were within the reference range. Propranolol was successfully tapered over 3 weeks by reducing the dose by 50% weekly until it reached the therapeutic dose. After tapering, the patient was asymptomatic, with a mild increase in hemangioma size. After 6 weeks of the therapeutic dose, the hemangioma was fading away. CONCLUSIONS This case is one of the few cases reported in the literature of high, chronic propranolol overdose in pediatric patients. The patient remained asymptomatic, and the overdose was successfully managed with gradual tapering over several weeks. This case report can serve as a guide in managing subsequent cases.

PMID:38008932 | DOI:10.12659/AJCR.941765

Bilateral circumscribed choroidal hemangiomas in 2 patients: a rare finding

Fetched: November 27th, 2023, 5:02am GMT by Joseph D DeSimone

Can J Ophthalmol. 2023 Nov 23:S0008-4182(23)00343-5. doi: 10.1016/j.jcjo.2023.11.001. Online ahead of print.

NO ABSTRACT

PMID:38008126 | DOI:10.1016/j.jcjo.2023.11.001

Importance of Determining Vascular Endothelial Growth Factor Serum Levels in Children with Infantile Hemangioma

Fetched: November 26th, 2023, 5:02am GMT by Arnes Rešić

Medicina (Kaunas). 2023 Oct 29;59(11):1914. doi: 10.3390/medicina59111914.

ABSTRACT

Background and Objectives: A potential role of vascular endothelial growth factor (VEGF) in the pathophysiology of infantile hemangiomas (IH) is thought to be plausible. The primary objective of this study was to investigate the importance of determining VEGF serum levels at various stages of IH growth in children. Materials and Methods: A nested case-control study was conducted. For the purposes of the researched target group, samples of fifty (N = 50) children with IH without associated diseases at different stages of hemangioma growth (proliferative and involutional stages) were used. The control group consisted of one hundred (N = 100) healthy children comparable in terms of age and sex, in whom the existence of IH and vascular malformations was ruled out via clinical examination. An immunoassay (ELISA) was used to determine VEGF serum levels in hemangioma growth's proliferation and involution phases. Results: A comparison of serum levels of VEGF in the phases of proliferation and involution in the group of patients with IH did not show a statistically significant difference (p = 0.171). The control group had significantly higher serum VEGF levels than the patient group in both the proliferation phase (p = 0.009) and the involution phase (p = 0.019). In the proliferation phase, a multivariate regression model explained 15% of the variance in the dependent variable, without significant predictor variables, while in the involution phase, it explained 21% of the variance in the dependent variable, and the history of invasive prenatal procedures stood out as a significant predictor variable positively associated with serum VEGF levels (beta coefficient = 0.33; p = 0.043). Conclusions: Although IH is thought to be the result of the dysregulation of angiogenesis and vasculogenesis under the influence of angiogenic factors, especially VEGF, this study did not demonstrate that VEGF serum levels in the proliferation phase of hemangioma growth were higher than those in the involution phase, or in relation to the control group.

PMID:38003963 | PMC:PMC10673278 | DOI:10.3390/medicina59111914

Right Parapharyngeal Cavernous Hemangioma, A Rare Entity: Case Report and Literature Review

Fetched: November 25th, 2023, 5:01am GMT by S Emelda Rose

Ear Nose Throat J. 2023 Nov 24:1455613231212597. doi: 10.1177/01455613231212597. Online ahead of print.

ABSTRACT

Cavernous hemangioma is a noncancerous vascular growth that arises from different parts of the head and neck region. However, parapharyngeal space contributes a very small percentage for its occurrence. We present a case of right parapharyngeal cavernous hemangioma, a very rare clinical presentation. This is a 57-year-old female presented with throat discomfort for 3 months. Examination finding showed a soft, diffuse, and non-pulsating mass over the right upper jugulodigastric region. A contrasted computed topographic scan revealed multiple calcifications in right parapharyngeal space. T2-weighted magnetic resonance imaging showed right parapharyngeal space mass with high signal and multiple phleboliths and dynamic angiogram unremarkable. Surgical resection done via transcervical approach and histopathological report revealed cavernous hemangioma with calcified thrombi. In conclusion, surgical intervention is the mainstay treatment and transcervical approach which is adopted in this case is the commonest approach used in surgical resection of cavernous hemangioma.

PMID:37997797 | DOI:10.1177/01455613231212597

Diagnosis of focal liver lesions from ultrasound images using a pretrained residual neural network

Fetched: November 23rd, 2023, 5:02am GMT by Sutthirak Tangruangkiat

J Appl Clin Med Phys. 2023 Nov 22:e14210. doi: 10.1002/acm2.14210. Online ahead of print.

ABSTRACT

OBJECTIVE: This study aims to develop a ResNet50-based deep learning model for focal liver lesion (FLL) classification in ultrasound images, comparing its performance with other models and prior research.

METHODOLOGY: We retrospectively collected 581 ultrasound images from the Chulabhorn Hospital's HCC surveillance and screening project (2010-2018). The dataset comprised five classes: non-FLL, hepatic cyst (Cyst), hemangioma (HMG), focal fatty sparing (FFS), and hepatocellular carcinoma (HCC). We conducted 5-fold cross-validation after random dataset partitioning, enhancing training data with data augmentation. Our models used modified pre-trained ResNet50, GGN, ResNet18, and VGG16 architectures. Model performance, assessed via confusion matrices for sensitivity, specificity, and accuracy, was compared across models and with prior studies.

RESULTS: ResNet50 outperformed other models, achieving a 5-fold cross-validation accuracy of 87 ± 2.2%. While VGG16 showed similar performance, it exhibited higher uncertainty. In the testing phase, the pretrained ResNet50 excelled in classifying non-FLL, cysts, and FFS. To compare with other research, ResNet50 surpassed the prior methods like two-layered feed-forward neural networks (FFNN) and CNN+ReLU in FLL diagnosis.

CONCLUSION: ResNet50 exhibited good performance in FLL diagnosis, especially for HCC classification, suggesting its potential for developing computer-aided FLL diagnosis. However, further refinement is required for HCC and HMG classification in future studies.

PMID:37991141 | DOI:10.1002/acm2.14210

Eccrine Angiomatous Neavus With Verrucous Haemangioma-Like Characteristics: A Rare Case

Fetched: November 23rd, 2023, 5:02am GMT by Prabin P Dev

Dermatol Pract Concept. 2023 Oct 1;13(4):e2023205. doi: 10.5826/dpc.1304a205.

NO ABSTRACT

PMID:37992343 | PMC:PMC10656125 | DOI:10.5826/dpc.1304a205

Unilateral chorea linked to cavernous haemangioma involving the putamen improved by surgery

Fetched: November 22nd, 2023, 5:02am GMT by Yosuke Ito

BMJ Case Rep. 2023 Nov 21;16(11):e257218. doi: 10.1136/bcr-2023-257218.

ABSTRACT

Unilateral chorea movements caused by cavernous haemangioma in the putamen are extremely rare. We report a case with chorea movements linked to cavernous haemangioma, localised to an area including the putamen in which pharmacotherapy was found to be ineffective. Symptoms were, however, improved by resection of the cavernous haemangioma. In cases where chorea movements linked to cavernous haemangioma, involving the putamen, prove intractable with watchful waiting or pharmacotherapy, improvement can be expected with surgical removal of the cavernous haemangioma. It is also possible to reduce the risk of complications through the use of intraoperative navigation and monitoring.

PMID:37989329 | PMC:PMC10668196 | DOI:10.1136/bcr-2023-257218

Cavernous Hemangioma of the Retina and Brain

Fetched: November 22nd, 2023, 5:02am GMT by Kayo Sugiura

Ophthalmology. 2023 Nov 21:S0161-6420(23)00793-5. doi: 10.1016/j.ophtha.2023.10.024. Online ahead of print.

NO ABSTRACT

PMID:37988043 | DOI:10.1016/j.ophtha.2023.10.024

Isolated bronchial hemangioma causing recurrent hemoptysis: A case report

Fetched: November 22nd, 2023, 5:02am GMT by Zhipeng Lin

Medicine (Baltimore). 2023 Nov 17;102(46):e36135. doi: 10.1097/MD.0000000000036135.

ABSTRACT

RATIONAL: The development of bronchial hemangioma in adults is rare, and massive hemoptysis due to diffuse vascular proliferation of bronchial hemangioma is fatal.

PATIENT CONCERNS: A case of a 29-year-old woman kept massive hemoptysis even after being underwent repeated interventional embolization for recurrent massive hemoptysis. Eventually, the patient was performed the operation of right upper lung lobectomy and bronchial hemangioma with extracorporeal membrane oxygenation support and was followed up for 4 years without recurrent hemoptysis.

DIAGNOSES: Bronchial hemangioma.

CONCLUSION: For patients with bronchial angiomas bonded with bronchial artery-pulmonary arteriovenous fistulae, the early surgical resection is recommended if bronchial artery embolization (BAE) is considered ineffective.

PMID:37986342 | PMC:PMC10659665 | DOI:10.1097/MD.0000000000036135

Permalink for 'Exploration of predictors associated with bleeding in computed tomography-guided core needle splenic biopsy: A retrospective study'

Exploration of predictors associated with bleeding in computed tomography-guided core needle splenic biopsy: A retrospective study

Fetched: November 22nd, 2023, 5:02am GMT by Sakue Masuda

Medicine (Baltimore). 2023 Nov 17;102(46):e34951. doi: 10.1097/MD.0000000000034951.

ABSTRACT

Splenic diseases may be caused by infections and can be either malignant, such as lymphoma and lung cancer, or benign, such as hemangioma. In some cases, diagnostic uncertainty of imaging persists, and image-guided splenic needle biopsy is a useful diagnostic tool to avoid the disadvantages of incorrect diagnosis, including performing unnecessary splenectomy or not giving the necessary treatment. Splenic biopsies can be divided into ultrasound-guided, computed tomography (CT)-guided fine-needle aspiration, or core needle biopsy (CNB). However, few studies have focused exclusively on complications associated with CT-guided CNB of the spleen. Therefore, we assessed bleeding, the most common complication of CT-guided CNB of the spleen, and evaluated factors associated with the bleeding. Using the biopsy database maintained at the institution, all patients who underwent CT-guided CNB of the spleen between May 2012 and September 2022 were identified retrospectively. The 18 identified patients were divided into post-biopsy bleeding and non-bleeding groups for analysis. In total, 17 patients (94.4%) could be diagnosed accurately with CT-guided CNB. Bleeding complications occurred in 7 cases of CT-guided CNB; of these, 2 patients with Common Terminology Criteria for Adverse Events grade 4 disease required transcatheter arterial embolization. The bleeding group was characterized by diffuse spleen tumors in all cases, with significantly more diffuse spleen tumors than the non-bleeding group. CT-guided CNB is a useful option for neoplastic lesions of the spleen that are difficult to diagnose using imaging alone. However, consideration should be given to post-biopsy bleeding in patients with diffuse splenic tumors.

PMID:37986279 | PMC:PMC10659597 | DOI:10.1097/MD.0000000000034951

Endoscopic submucosal dissection for oesophageal cavernous haemangioma accompanied by squamous epithelial dysplasia

Fetched: November 22nd, 2023, 5:02am GMT by Chen Wu

Dig Liver Dis. 2023 Nov 18:S1590-8658(23)01011-3. doi: 10.1016/j.dld.2023.10.022. Online ahead of print.

NO ABSTRACT

PMID:37985249 | DOI:10.1016/j.dld.2023.10.022

Mesenteric cystic lymphangioma in an adolescent male; a diagnostic dilemma: A case report

Fetched: November 22nd, 2023, 5:02am GMT by Asmita Bhusal

Int J Surg Case Rep. 2023 Dec;113:109042. doi: 10.1016/j.ijscr.2023.109042. Epub 2023 Nov 14.

ABSTRACT

INTRODUCTION AND IMPORTANCE: Hemolymphangioma, a rare type of lymphangioma, is a benign hamartoma of the blood vessels and lymphatic system. Considered to be extremely rare among adults with infrequent occurrence in abdominal regions, lymphangioma involving mesentery accounts for <1 % of cases and only 0.05 % involve the GI tract. Due to the absence of typical clinical presentation, making a confirmatory preoperative diagnosis is challenging.

CASE PRESENTATION: An 18-year-old Bangladeshi man visited the hospital with the complaints of epigastric and umbilical pain for 15 days which was insidious in onset. Physical examination revealed an ill defined lump that was palpable on the right side of the abdomen. Ultrasonography showed multiloculated cystic mass having septation approximately 13 × 6 cm in size. An abdominal CT scan showed cystic lesion with septations measuring about 14.5 × 12.3 cm, compressing the bowel loops towards left, that was suggestive of mesenteric lymphangioma. The patient underwent partial surgical resection. The excised mass was sent for histopathology. Histopathology disclosed a benign tumor composed of proliferation of blood vessels of different sizes lined by endothelium in a fibromuscular stroma.

CLINICAL DISCUSSION: Mesenteric lymphangiomas are infrequent malformations and very few cases of mesenteric hemolymphangioma have been reported in adults. Histopathology is required for confirmatory diagnosis and immunohistochemistry is required to differentiate the tumor subtype. Surgical resection is deemed a standard treatment modality.

CONCLUSION: We report an extremely rare case of mesenteric hemolymphangioma to bring it to concern that even with the vague clinical presentations and involvement of age groups beyond the status quo, surgeons must be vigilant about abdominal MCL/hemolymphangioma and proceed accordingly.

PMID:37984261 | DOI:10.1016/j.ijscr.2023.109042

Unusual False Positive on 99mTc-Tektrotyd Scintigraphy Mimicking Bone Metastasis

Fetched: November 22nd, 2023, 5:02am GMT by Chaimae Sebbar

Nucl Med Mol Imaging. 2023 Dec;57(6):295-297. doi: 10.1007/s13139-023-00803-8. Epub 2023 Apr 27.

ABSTRACT

Somatostatin receptor scintigraphy is a key diagnostic tool in the initial staging and therapeutic evaluation of neuroendocrine tumors. Its specificity can be compromised by the presence of false positives. We illustrate here the case of a vertebral hemangioma detected on ^99mTc-Tektrotyd scintigraphy as an incidental finding in a 34-year-old man referred for the staging of a well-differentiated neuroendocrine tumor of the ampulla of Vater (Fig. 1).

PMID:37982104 | PMC:PMC10654281 | DOI:10.1007/s13139-023-00803-8

A Case of Phlebolith in the Tip of Tongue

Fetched: November 22nd, 2023, 5:02am GMT by Hisashi Kato

Tokai J Exp Clin Med. 2023 Dec 20;48(4):139-143.

ABSTRACT

The differential diagnosis of a tongue mass containing calcified bodies includes a phlebolith associated with vascular lesions, such as hemangioma and vascular malformation, and diseases such as neoplasm, osseous choristoma and hypercalcemic states, including hyperparathyroidism. The appearance of the calcified bodies on plain radiographs may help to differentiate these entities. Computed tomography, magnetic resonance imaging, and ultrasonography are also useful for differentiating these soft tissue lesions. We report a 40-year-old man with a small mass containing a calcified body in the tip of tongue. The mass was surgically resected and histologically evaluated, confirming the diagnosis of phlebolith. Our case was a rare phlebolith that did not involve a vascular lesion.

PMID:37981851

Vascular Lesions

Fetched: November 20th, 2023, 5:02am GMT by Matthew J Urban

Facial Plast Surg Clin North Am. 2024 Feb;32(1):13-25. doi: 10.1016/j.fsc.2023.09.003. Epub 2023 Oct 21.

ABSTRACT

Vascular lesions impact up to 5% of children and range in clinical impact from minor cutaneous aberrations to large masses impacting both form and function. Vascular lesions may be characterized as tumors or malformations. Establishing a clear diagnosis is imperative to understanding the natural history of a vascular lesion and developing a treatment plan. Medical, surgical, intralesional, and laser therapy are all effective and indicated on a case-by-case basis. There are a number of important surgical considerations for operative management of these lesions.

PMID:37981409 | DOI:10.1016/j.fsc.2023.09.003

Terminology and Classifications of Vascular Lesions Based on Molecular Identification

Fetched: November 20th, 2023, 5:02am GMT by Srinivasa R Chandra

Oral Maxillofac Surg Clin North Am. 2024 Feb;36(1):35-48. doi: 10.1016/j.coms.2023.09.010.

ABSTRACT

The majority of the vascular anomalies are seen in the head and neck region. Even though the incidence of this anomaly could be construed as a rare disease entity, with only 5% of overall affliction, the lack of knowledgeable management has disfigured many. A comprehensive understanding of this benign yet complex life-changing entity is essential. A historical perspective, pathophysiology-logical evolution, and the current knowledge of management modalities are essential for rendering clinical care in this subspecialty care. We propose a very succint Head and Neck Surgical classification for vascular anamolies, which has been a good guide for surgical approaches.

PMID:37981344 | DOI:10.1016/j.coms.2023.09.010

Medical Management and Therapeutic Updates on Vascular Anomalies of the Head and Neck: Part 2

Fetched: November 20th, 2023, 5:02am GMT by Jorie Gatts

Oral Maxillofac Surg Clin North Am. 2024 Feb;36(1):115-123. doi: 10.1016/j.coms.2023.09.012.

ABSTRACT

Discovery of inherited and somatic genetic mutations, along with advancements in clinical and scientific research, has improved understanding of vascular anomalies and changed the treatment paradigm. With aim of minimizing need for invasive procedures and improving disease outcomes, molecularly targeted medications and anti-angiogenesis agents have become important as both adjuncts to surgery, and increasingly, as the primary treatment of vascular anomalies. This article highlights the commonly used and emerging therapeutic medications for non-malignant vascular tumors and vascular malformations in addition to medical management of associated hematologic abnormalities.

PMID:37981343 | DOI:10.1016/j.coms.2023.09.012

The oncogenic fusion protein TAZ::CAMTA1 promotes genomic instability and senescence through hypertranscription

Fetched: November 20th, 2023, 5:02am GMT by Emily Neil

Commun Biol. 2023 Nov 18;6(1):1174. doi: 10.1038/s42003-023-05540-4.

ABSTRACT

TAZ::CAMTA1 is a fusion protein found in over 90% of Epithelioid Hemangioendothelioma (EHE), a rare vascular sarcoma with an unpredictable disease course. To date, how TAZ::CAMTA1 initiates tumour formation remains unexplained. To study the oncogenic mechanism leading to EHE initiation, we developed a model system whereby TAZ::CAMTA1 expression is induced by doxycycline in primary endothelial cells. Using this model, we establish that upon TAZ::CAMTA1 expression endothelial cells rapidly enter a hypertranscription state, triggering considerable DNA damage. As a result, TC-expressing cells become trapped in S phase. Additionally, TAZ::CAMTA1-expressing endothelial cells have impaired homologous recombination, as shown by reduced BRCA1 and RAD51 foci formation. Consequently, the DNA damage remains unrepaired and TAZ::CAMTA1-expressing cells enter senescence. Knockout of Cdkn2a, the most common secondary mutation found in EHE, allows senescence bypass and uncontrolled growth. Together, this provides a mechanistic explanation for the clinical course of EHE and offers novel insight into therapeutic options.

PMID:37980390 | PMC:PMC10657451 | DOI:10.1038/s42003-023-05540-4

Cavernous hemangioma of the lip: combined treatment with intralesional laser and surgery

Fetched: November 19th, 2023, 5:01am GMT by Alberto Goldman

Wien Med Wochenschr. 2023 Nov 17. doi: 10.1007/s10354-023-01028-2. Online ahead of print.

ABSTRACT

Infantile cavernous hemangioma is a benign vascular tumor in childhood. Cavernous hemangiomas have a tendency of slow enlargement with subsequent complications such as ulceration and bleeding. We report a case of a large cavernous hemangioma affecting the median upper lip in an 11-year-old girl. The lesion was growing and painful on pressure. The patient felt stigmatized. Treatment was wished for by patient and parents. After careful examination including Doppler ultrasound, we suggested a combined approach. In the first step the lesion was coagulated with an intralesional neodymium-YAG laser using a blunt tip. In the second step, immediately after the action of the laser, the lesion was removed completely by surgery. Defect closure was realized with bilateral tissue-expanding vermillion myocutaneous flaps. The patient was followed-up to 5 months. Disfigurement and functional impairment were eliminated. The result was satisfying for both patient and parents. Vascular lesions of the upper lip-particularly when close to Cupid's bow-can become disfiguring and cause functional impairment. We propose a combined approach with intralesional neodymium-YAG laser photocoagulation to avoid bleeding and to shrink the lesion before complete surgical removal. For defect closure, bilateral tissue-expanding vermillion myocutaneous flaps (Goldstein technique modified by Sawada) were used.

PMID:37975966 | DOI:10.1007/s10354-023-01028-2

Epithelioid Hemangioma of Tongue a Case Report

Fetched: November 19th, 2023, 5:01am GMT by S Prabakaran

Indian J Otolaryngol Head Neck Surg. 2023 Dec;75(4):3912-3916. doi: 10.1007/s12070-023-03969-x. Epub 2023 Jun 17.

ABSTRACT

Epithelioid hemangioma is an uncommon benign vascular lesion which most commonly appears as a nodular lesion in the head and neck area. Here, we present a rare case of nodular swelling in the dorsal aspect of the tongue in a 30 year old male for the past 3 months. Excision of growth was done and histopathological examination revealed epithelioid hemangioma of the tongue. Patient is on regular follow up and there is no evidence of recurrence.

SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1007/s12070-023-03969-x.

PMID:37974871 | PMC:PMC10646121 | DOI:10.1007/s12070-023-03969-x

Intraconal Retroorbital Cavernous Hemangioma - A Case Report

Fetched: November 19th, 2023, 5:01am GMT by Jitendra Singh Yadav

Indian J Otolaryngol Head Neck Surg. 2023 Dec;75(4):3925-3928. doi: 10.1007/s12070-023-03984-y. Epub 2023 Jun 19.

ABSTRACT

Cavernous hemangioma of orbit is a benign, noninfiltrative, slowly progressive vascular neoplasm. It is usually asymptomatic but patients may present with proptosis and diminished vision due to compression of second cranial nerve, optic nerve. This can be usually diagnosed with the help of clinical examination and computed tomography (CT) or magnetic resonance imaging (MRI). Small sized tumours are worth wait and watch while large ones need surgical excision. In our case report, A 65-year-old male patient presented to the head and neck surgery with proptosis of left eye since 5 years along with decreased vision since 4 years. MDCT scan (orbits plain) suggestive of large solid retroocular, intraconal mass in left orbit leading to proptosis of left eyeball. The patient underwent excision of tumour through a transnasal endoscopic approach. Histopathological examination of the tumour identified as cavernous hemangioma. It is safe and effective way to access and excise the orbital tumours through the transnasal endoscopic approach. It is essential to have experienced surgeon in endoscopic procedures. The patient had satisfactory results at three months follow up and showed no symptoms or relapse on CT scans of orbital region.

SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1007/s12070-023-03984-y.

PMID:37974857 | PMC:PMC10645695 | DOI:10.1007/s12070-023-03984-y

Epithelioid Hemangioendothelioma of the Mastoid Bone with Extension of Middle Cranial Fossa: A Case Report

Fetched: November 19th, 2023, 5:01am GMT by Hatice Secil Akyildiz

Indian J Otolaryngol Head Neck Surg. 2023 Dec;75(4):4050-4053. doi: 10.1007/s12070-023-03954-4. Epub 2023 Jul 5.

ABSTRACT

Epithelioid hemangioendothelioma (EHE) is a rare sarcoma of vascular origin. It is frequently seen in the liver, lungs and bones. Cases with mastoid bone and intracranial extension are rarer. A 40 year old male, presented with a history of progressive headaches and a mass in the postauricular region for a duration of three months. Computer tomography of the temporal bone showed an approximately 40 × 30 mm soft tissue with osteolytic bony changes located in the mastoid bone. Epithelioid hemangioendothelioma is a rare sarcoma of vascular origin. Surgery is the first line of therapy. Prognosis is generally good, and may be better for primary intracranial disease than that for EHE originating elsewhere.

PMID:37974704 | PMC:PMC10645722 | DOI:10.1007/s12070-023-03954-4

Capillary Hemangioma of the Maxillary Sinus: A Rare Cause of Massive Life-Threatening Epistaxis

Fetched: November 19th, 2023, 5:01am GMT by Brihaspati Sigdel

Indian J Otolaryngol Head Neck Surg. 2023 Dec;75(4):4071-4074. doi: 10.1007/s12070-023-04046-z. Epub 2023 Jul 12.

ABSTRACT

Hemangioma of the paranasal sinus is an infrequent condition. Two types of hemangioma are present at the nose; cavernous and capillary. Capillary hemangioma is rare in the sinonasal tract. Patients presented with massive epistaxis should think of nasal hemangioma. Resuscitation of patients is important and urgent Computerized Tomography scans should be performed which will help character and extension of any mass present at the nasal cavity. Initial nasal packing may help to prevent bleeding. We report a case of capillary hemangioma of a 30-year lady present with massive epistaxis initially controlled by Nasal packing. Her CT scan shows a homogenous mass at left maxillary sinuses and underwent left endoscopic sphenopalatine artery ligation and removal of the mass.

SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1007/s12070-023-04046-z.

PMID:37974689 | PMC:PMC10645818 | DOI:10.1007/s12070-023-04046-z

Atypical Cutaneous Lymphoid Hyperplasia of Nose: A Pathological Puzzle

Fetched: November 19th, 2023, 5:01am GMT by Lakshmi Krishnakumar

Indian J Otolaryngol Head Neck Surg. 2023 Dec;75(4):3980-3983. doi: 10.1007/s12070-023-03960-6. Epub 2023 Jun 29.

ABSTRACT

Atypical cutaneous lymphoid hyperplasia is a rare tumour of the head and neck which represents a lymphoproliferative continuum and mimics cutaneous lymphoma. Hereby reporting the case of a 40-year-old gentleman who presented with a swelling over dorsum of nose. On evaluation, Fine needle aspiration cytology was inconclusive with only inflammatory cells. Contrast Enhanced CT Nose and PNS suggested a possibility of a haemangioma. Patient underwent laser assisted excision of mass and frozen section showed sheets of basaloid cells suggestive of basal cell carcinoma. Wide local excision and primary paramedian forehead flap closure was done. Histopathology was suggestive of CD30 + atypical cutaneous lymphoid hyperplasia which has potential for malignant transformation into overt lymphoma. Patient underwent pedicle division of flap and is on regular follow up. In this case, aspiration cytology and frozen section reports were discordant with the histopathology, and thereby enlightens us of a rare differential diagnosis for head and neck tumours.

PMID:37974678 | PMC:PMC10646108 | DOI:10.1007/s12070-023-03960-6

Intraosseous Hemangioma of Zygoma: A Case Report with Review of Literature

Fetched: November 19th, 2023, 5:01am GMT by Mangesh M Londhe

Indian J Otolaryngol Head Neck Surg. 2023 Dec;75(4):4024-4027. doi: 10.1007/s12070-023-03982-0. Epub 2023 Jul 4.

ABSTRACT

Intraosseous hemangiomas are uncommon and account to less than 1% of all osseous tumors. Vertebral body and skull are the most common sites involved. However involvement of facial bones is rare with zygoma being even rarer site. Due to its rarity it creates diagnostic dilemma clinically and radiologically. Its vascular nature carries the risk of intraoperative bleeding hence an accurate preoperative diagnosis with arterial embolization helps to avoid the dire consequences. Fine needle aspiration cytology along with radiological correlation is extremely helpful in such cases. We report such rare case in forty eight years female patient with brief review of literature.

PMID:37974668 | PMC:PMC10646063 | DOI:10.1007/s12070-023-03982-0

Vascular malformation and tumors

Fetched: November 19th, 2023, 5:01am GMT by Alexandra Hartel

MMW Fortschr Med. 2023 Nov;165(20):53-55. doi: 10.1007/s15006-023-3097-3.

ABSTRACT

Vascular anomalies can be categorized into two groups: on one side the congenital vascular malformations, on the other hand vascular tumours. The most common benign vascular tumour are infantile haemangiomas. Those occur in the first few weeks of life and then show a characteristic pattern of growth and regression. Vascular malformations on the other hand are abnormal vessels in addition to the normal vascular system. They can occur in all systems, capillary, venous, arterial and lymphatic.

PMID:37973754 | DOI:10.1007/s15006-023-3097-3

LUMBAR Syndrome and Bladder Exstrophy - a case report

Fetched: November 19th, 2023, 5:01am GMT by Catherine Nguyen

Urology. 2023 Nov 14:S0090-4295(23)00953-6. doi: 10.1016/j.urology.2023.10.016. Online ahead of print.

ABSTRACT

LUMBAR syndrome is rare with a multitude of features that requires a high index of suspicion for timely diagnosis and appropriate management. We present on a newborn female whose untreated segmental infantile hemangioma (IH) lead to poor healing of her bladder exstrophy closure. The objective of this report is to describe bladder exstrophy as a urogenital anomaly in patients with LUMBAR syndrome and the importance of balancing management of IH and time to surgery.

PMID:37972897 | DOI:10.1016/j.urology.2023.10.016

Kaposiform hemangioendothelioma presented with raynaud phenomenon: a case report

Fetched: November 19th, 2023, 5:01am GMT by Lingke Liu

BMC Pediatr. 2023 Nov 17;23(1):574. doi: 10.1186/s12887-023-04407-1.

ABSTRACT

BACKGROUND: Kaposiform hemangioendothelioma (KHE) is a rare vascular neoplasm affecting infants or young children. KHE includes a spectrum of lesions, ranging from small and superficial tumors to large and invasive lesions with Kasabach-Merritt phenomenon (KMP). Currently, no published studies have reported a KHE presenting as thrombocytopenia and Raynaud phenomenon.

CASE PRESENTATION: A 2-year-old boy with right hand swelling and thrombocytopenia was admitted to our hospital. His right hand turned swelling and red, even occasionally cyanotic. This condition became worse in response to cool environments and improved with warming, and platelet counts were between 50 ~ 80 × 10^9/L. Physical examination on admission revealed the swelling and frostbite-like rash of the right-hand fingers, and the skin temperature of the right hand was lower than the left. On day 3 of admission, chest CT results showed an irregular mass on the right side of the spine. The puncture biopsy demonstrated positive CD31, D2-40, and FLI1 immunohistochemical staining, but negative GLUT1 staining, confirming the diagnosis of KHE. Furthermore, endothelin-1 (ET1) expression levels significantly increased, and eNOS and A20 expression levels significantly decreased comparing with control patients. The patient received methylprednisolone and sirolimus treatments, and his condition gradually improved during the follow-up.

CONCLUSIONS: We reported the first case of KHE presenting with thrombocytopenia and Raynaud phenomenon. The development of Raynaud phenomenon could be associated with increased ET-1 and reduced eNOS and A20 expressions. Careful differential diagnosis of hidden KHE should be considered in children with thrombocytopenia and Raynaud phenomenon.

PMID:37978357 | PMC:PMC10655467 | DOI:10.1186/s12887-023-04407-1

Diagnostic efficiency of transcutaneous ultrasound scanning and Dopplerography in laryngeal pathology

Fetched: November 17th, 2023, 5:02am GMT by M V Subbotina

Vestn Otorinolaringol. 2023;88(5):27-33. doi: 10.17116/otorino20238805127.

ABSTRACT

OBJECTIVE: To assess the efficiency and the place of grey scale ultrasound and color Doppler sonography of the larynx in the diagnosis of laryngeal pathology.

MATERIAL AND METHODS: A prospective blind cohort examination in B-mode laryngeal ultrasound (LUS) and color Doppler imaging (CDI) with linear scanning transducer 7-15 MHz was performed in 120 patients aged from 6 months to 52 years (average age 7.6±5.8 years, Me 6 year) and in 40 patients without laryngeal pathology (average age 7.0±5.0 years). The patients presented with complaints of voice and/or stridor. The diagnosis was verified by followed laryngoscopy.

RESULTS: Laryngeal papillomas, hemangiomas, scarring and vocal fold's nodules were identified as hyperechoic formations. Color Doppler sonography made it possible to visualize them better: small formations were highlighted in color and the space around the large ones was colored. There were paradoxical movements of the hyperechoic arytenoid cartilages during inspiration to the anterior commissure in patients with laryngomalacia. Color Doppler ultrasonography revealed changes during phonation in patients with functional dysphonia. The sensitivity and specificity of LUS were 58% (95% CI 48-66) and 98% (95% CI 87-99) compared with laryngoscopy in the detection of laryngeal pathology, but laryngeal CDI - 81% (95% CI 72-87) and 98% (95% CI 87-99) respectively.

CONCLUSION: Ultrasound of the larynx in B-mode has a diagnostic efficiency of 67.5%, and in CDI mode - 85% for ruling in laryngeal pathologies compared to laryngoscopy. So, this method is a modern affordable, non-invasive and informative diagnostic tool for the detection of laryngeal diseases, especially in those cases, when it is impossible to carry out a laryngoscopy.

PMID:37970767 | DOI:10.17116/otorino20238805127

Prenatal ultrasound diagnosis of congenital infantile fibrosarcoma and congenital hemangioma: Three case reports

Fetched: November 17th, 2023, 5:02am GMT by Ru-Na Liang

World J Clin Cases. 2023 Oct 26;11(30):7403-7412. doi: 10.12998/wjcc.v11.i30.7403.

ABSTRACT

BACKGROUND: Congenital infantile fibrosarcoma (CIF) and congenital hemangioma (CH) have similarities on prenatal ultrasound and are rare.

CASE SUMMARY: We report 3 cases of fetuses with superficial hypervascular tumors, confirmed by postnatal pathology as CIF (1 case) and CH (2 cases, including 1 in a twin fetus). In Case 1, a mass with a rich blood supply in the fetal axilla was discovered by prenatal ultrasound at 28+0 wk of gestation. The postpartum pathological diagnosis was CIF, the mass was surgically removed, and the prognosis of the child was good. In Case 2, at 23+1 wk of gestation, a mass was discovered at the base of the fetus's thigh on prenatal ultrasound. The postpartum pathological diagnosis was CH. After conservative treatment, the mass shrank significantly. Case 3 occurred in a twin fetus. At 30+0 wk of gestation, prenatal ultrasound revealed a bulging mass with a rich blood supply on the abdominal wall of one of the fetuses. Three weeks later, the affected fetus died, and the unaffected baby was successfully delivered by emergency cesarean section. The affected fetus was pathologically diagnosed with CH.

CONCLUSION: Prenatal ultrasound can provide accurate information, such as the location, size and blood supply of a surface mass in a fetus. We found similarities between CIF and CH in prenatal ultrasound findings. Although it is difficult to distinguish these conditions by prenatal ultrasound alone, for superficial hypervascular tumors that rapidly increase in size in a short period, close ultrasound monitoring of the fetus is required to quickly address possible adverse outcomes.

PMID:37969437 | PMC:PMC10643080 | DOI:10.12998/wjcc.v11.i30.7403

A rare case of adult pancreatic cavernous hemangioma

Fetched: November 16th, 2023, 5:02am GMT by Xinyu Lei

Asian J Surg. 2023 Nov 13:S1015-9584(23)01755-4. doi: 10.1016/j.asjsur.2023.11.024. Online ahead of print.

NO ABSTRACT

PMID:37968210 | DOI:10.1016/j.asjsur.2023.11.024

Ophthalmological and Interdisciplinary Surgical Approaches to the Orbit

Fetched: November 16th, 2023, 5:02am GMT by Eckart Bertelmann

Klin Monbl Augenheilkd. 2023 Nov 15. doi: 10.1055/a-2149-0911. Online ahead of print.

ABSTRACT

The treatment of orbital masses often requires an interdisciplinary approach. Ophthalmological surgical approaches include the anterior transcutaneous approach for removal of dermoids or taking biopsies. Anterior transconjunctival orbitotomy is suitable for masses of the inferior orbit or the intraconal compartment, but is inappropriate for masses of the orbital apex. The lateral transosseous orbitotomy often fits for masses of the lateral or intraconal compartment, especially for neoplasms of the lacrimal gland e.g. pleomorphic adenoma. Cryoextraction is often the technique of first choice for cavernous haemangiomas of all localisations. Transfrontal approaches are suitable for the resection of tumours of the optic nerve and of the orbital apex.

PMID:37967818 | DOI:10.1055/a-2149-0911

Osmundacetone Inhibits Angiogenesis of Infantile Hemangiomas through Inducing Caspases and Reducing VEGFR2/MMP2

Fetched: November 15th, 2023, 5:02am GMT by Chen Ke

Anticancer Agents Med Chem. 2023 Nov 8. doi: 10.2174/0118715206273410231103100600. Online ahead of print.

ABSTRACT

AIM: This study aims to explore the potential of Osmundacetone (OSC) as a new treatment for infantile hemangiomas (IH), the most common benign tumors in infancy. Currently, propranolol serves as the primary treatment for IH, but its effectiveness is limited, and it poses challenges of drug resistance and side effects. Therefore, there is a pressing need to identify alternative therapies for IH.

METHODS: The effects of OSC on the proliferation and apoptosis of HemECs (endothelial cells from hemangiomas) were assessed using CCK-8 assay, colony formation assay, HOCHEST 33342 staining, and flow cytometry. Western blot analysis was performed to investigate OSC's influence on Caspases and angiogenesis-related proteins. Animal models were established using HemECs and BALB/c mice, and histological and immunohistochemical staining were conducted to evaluate the impact of OSC on mouse hemangiomas, VEGFR2, and MMP9 expression.

RESULTS: OSC treatment significantly reduced HemECs' viability and colony-forming ability, while promoting apoptosis, as indicated by increased HOCHEST 33342 staining. OSC upregulated the protein expression of Bax, PARP, Caspase9, Caspase3, AIF, Cyto C, FADD, and Caspase8 in HemECs. In animal models, OSC treatment effectively reduced hemangioma size and improved histopathological changes. OSC also suppressed VEGFR2 and MMP9 expression while elevating Caspase3 levels in mouse hemangiomas.

CONCLUSION: OSC demonstrated promising results in inhibiting HemECs' proliferation, inducing apoptosis, and ameliorating pathological changes in hemangiomas in mice. Moreover, it influenced the expression of crucial caspases and angiogenesis-related proteins. These findings suggest that OSC holds potential as a novel drug for clinical treatment of IH.

PMID:37957872 | DOI:10.2174/0118715206273410231103100600

Oncogenic Gαq activates RhoJ through PDZ-RhoGEF

Fetched: November 15th, 2023, 5:02am GMT by Rodolfo Daniel Cervantes-Villagrana

Int J Mol Sci. 2023 Oct 29;24(21):15734. doi: 10.3390/ijms242115734.

ABSTRACT

Oncogenic Gαq causes uveal melanoma via non-canonical signaling pathways. This constitutively active mutant GTPase is also found in cutaneous melanoma, lung adenocarcinoma, and seminoma, as well as in benign vascular tumors, such as congenital hemangiomas. We recently described that PDZ-RhoGEF (also known as ARHGEF11), a canonical Gα12/13 effector, is enabled by Gαs Q227L to activate CdcIn addition, and we demonstrated that constitutively active Gαq interacts with the PDZ-RhoGEF DH-PH catalytic module, but does not affect its binding to RhoA or Cdc. This suggests that it guides this RhoGEF to gain affinity for other GTPases. Since RhoJ, a small GTPase of the Cdc42 subfamily, has been involved in tumor-induced angiogenesis and the metastatic dissemination of cancer cells, we hypothesized that it might be a target of oncogenic Gαq signaling via PDZ-RhoGEF. Consistent with this possibility, we found that Gαq Q209L drives full-length PDZ-RhoGEF and a DH-PH construct to interact with nucleotide-free RhoJ-G33A, a mutant with affinity for active RhoJ-GEFs. Gαq Q209L binding to PDZ-RhoGEF was mapped to the PH domain, which, as an isolated construct, attenuated the interaction of this mutant GTPase with PDZ-RhoGEF's catalytic module (DH-PH domains). Expression of these catalytic domains caused contraction of endothelial cells and generated fine cell sprouts that were inhibited by co-expression of dominant negative RhoJ. Using relational data mining of uveal melanoma patient TCGA datasets, we got an insight into the signaling landscape that accompanies the Gαq/PDZ-RhoGEF/RhoJ axis. We identified three transcriptional signatures statistically linked with shorter patient survival, including GPCRs and signaling effectors that are recognized as vulnerabilities in cancer cell synthetic lethality datasets. In conclusion, we demonstrated that an oncogenic Gαq mutant enables the PDZ-RhoGEF DH-PH module to recognize RhoJ, suggesting an allosteric mechanism by which this constitutively active GTPase stimulates RhoJ via PDZ-RhoGEF. These findings highlight PDZ-RhoGEF and RhoJ as potential targets in tumors driven by mutant Gαq.

PMID:37958718 | PMC:PMC10647656 | DOI:10.3390/ijms242115734

Permalink for 'The Mechanism of Oxymatrine Targeting miR-27a-3p/PPAR-γ Signaling Pathway through m6A Modification to Regulate the Influence on Hemangioma Stem Cells on Propranolol Resistance'

The Mechanism of Oxymatrine Targeting miR-27a-3p/PPAR-γ Signaling Pathway through m6A Modification to Regulate the Influence on Hemangioma Stem Cells on Propranolol Resistance

Fetched: November 15th, 2023, 5:02am GMT by Yuxin Dai

Cancers (Basel). 2023 Oct 30;15(21):5213. doi: 10.3390/cancers15215213.

ABSTRACT

OBJECTIVE: The proliferation and migration of hemangioma stem cells (HemSCs) induced apoptosis and adipose differentiation as well as increased the sensitivity of HemSCs to propranolol (PPNL). MiR-27a-3p negatively controlled the peroxisome-proliferator-activated receptor γ (PPAR-γ) level, counteracting the effect of PPAR-γ on HemSC progression and PPNL resistance. OMT accelerated HemSC progression and adipocyte differentiation via modulating the miR-27a-3p/PPAR-γ axis, inhibiting HemSC resistance to PPNL. In tumor-forming experiments, OMT exhibited a dose-dependent inhibitory effect on the volume of IH PPNL-resistant tumors, which was partially dependent on the regulation of m6A methylation transfer enzyme METTL3 and the miR-27a-3p/PPAR-γ axis, thereby inducing apoptosis.

CONCLUSIONS: We conclude that OMT regulates IH and influences PPNL resistance via targeting the miR-27a-3p/PPAR-γ signaling pathway through m6A modification.

PMID:37958388 | PMC:PMC10649746 | DOI:10.3390/cancers15215213

Effects of propranolol on glucose metabolism in hemangioma-derived endothelial cells

Fetched: November 14th, 2023, 5:02am GMT by Kaiying Yang

Biochem Pharmacol. 2023 Nov 11;218:115922. doi: 10.1016/j.bcp.2023.115922. Online ahead of print.

ABSTRACT

Infantile hemangioma (IH) is the most common benign tumor in children. Propranolol is the first-line treatment for IH, but the underlying mechanism of propranolol treatment in IH is not completely understood. Integrated transcriptional and metabolic analyses were performed to investigate the metabolic changes in hemangioma-derived endothelial cells (HemECs) after propranolol treatment. The findings were then further validated through independent cell experiments using a Seahorse XFp analyzer, Western blotting, immunohistochemistry and mitochondrial functional assays. Thirty-four differentially expressed metabolites, including the glycolysis metabolites glucose 6-phosphate, fructose 6-phosphate and fructose 1,6-bisphosphate, were identified by targeted metabolomics. A KEGG pathway enrichment analysis showed that the disturbances in these metabolites were highly related to glucose metabolism-related pathways, including the pentose phosphate pathway, the Warburg effect, glycolysis and the citric acid cycle. Transcriptional analysis revealed that metabolism-related pathways, including glycine, serine and threonine metabolism, tyrosine metabolism, and glutathione metabolism, were highly enriched. Moreover, integration of the metabolomic and transcriptomic data revealed that glucose metabolism-related pathways, particularly glycolysis, were altered after propranolol treatment. Cell experiments demonstrated that HemECs exhibited higher levels of glycolysis than human umbilical vein ECs (HUVECs) and that propranolol suppressed glycolysis in HemECs. In conclusion, propranolol inhibited glucose metabolism in HemECs by suppressing glucose metabolic pathways, particularly glycolysis.

PMID:37956892 | DOI:10.1016/j.bcp.2023.115922

Permalink for 'A Patient With Lobular Capillary Hemangioma Originating From the Inferior Turbinate Following Cauterization With Silver Nitrate'

A Patient With Lobular Capillary Hemangioma Originating From the Inferior Turbinate Following Cauterization With Silver Nitrate

Fetched: November 14th, 2023, 5:02am GMT by Masahiro Nakamura

J Craniofac Surg. 2023 Nov 13. doi: 10.1097/SCS.0000000000009882. Online ahead of print.

ABSTRACT

Lobular capillary hemangiomas (LCH), which usually originate in the skin and mucous membranes of the oral cavity, are uncommon from the posterior portion of the inferior turbinate. Although the exact cause of LCH in the nasal cavity has not been elucidated, trauma, caused by factors such as intranasal packing and habitual nose-picking, has been reported as one of the causes. In addition, 2 cases of LCH caused by submucosal resection with powered instrumentation to the inferior turbinate have been reported, suggesting that various types of traumas to the nasal mucosa can cause LCH. The authors report the first case of LCH formation in the posterior portion of the inferior turbinate after cauterization with silver nitrate.

PMID:37955465 | DOI:10.1097/SCS.0000000000009882

Infantile Scalp Hemangiomas: Surgical Management and Lessons Learned

Fetched: November 14th, 2023, 5:02am GMT by Nicole DiDonna

J Craniofac Surg. 2023 Nov 13. doi: 10.1097/SCS.0000000000009816. Online ahead of print.

ABSTRACT

Infantile hemangiomas (IH) are common benign vascular tumors in pediatric patients, often found on the scalp. While most IH naturally regress without intervention, surgical excision becomes necessary when severe anatomic or physiological complications arise. This review examines previously published case studies on pediatric scalp IH excisions, focusing on surgical outcomes and complications. A comprehensive search of the US National Library of Medicine National Institutes of Health (PubMed) database identified 19 relevant case studies. Most patients were female, with a wide age range at the time of excision. Various anatomic locations and sizes of the hemangiomas were observed. The reviewed literature demonstrates that surgical excision of scalp IH can be performed with minimal complications, even for sizable tumors. Successful outcomes and low complication rates highlight the effectiveness of surgical intervention in cases of life-threatening IH sequelae. Further research is warranted to explore optimal timing, techniques, and adjunct therapies for surgical management of scalp IH.

PMID:37955432 | DOI:10.1097/SCS.0000000000009816

Multimodal imaging features of retroperitoneal anastomosing hemangioma: a case report and literature review

Fetched: November 14th, 2023, 5:02am GMT by Liqing Zhang

Front Oncol. 2023 Oct 25;13:1269631. doi: 10.3389/fonc.2023.1269631. eCollection 2023.

ABSTRACT

BACKGROUND: Anastomotic hemangioma is a rare subtype of capillary hemangioma primarily found in the genitourinary tract. We present a case of a patient with an anastomotic hemangioma located in the retroperitoneal space; then, we explore and summarize the imaging features from previously reported cases for accurate diagnosis.

CASE PRESENTATION: A 57-year-old woman complained of left lower back pain. Contrast-enhanced ultrasound revealed a hypoechoic mass with "slow-in and slow-out" enhancement. Abdominal CT scan displayed a well-defined, round soft tissue mass in the right retroperitoneal region with obvious enhancement. MRI indicated low signal on T1-weighted imaging, high signal on T2-weighted imaging and diffusion-weighted imaging, and progressive enhancement after enhancement. Surgical removal of the tumor was performed. Histopathological examination exhibited a distinct tumor border with interconnected blood vessels and a cavity lined by a single layer of cubic endothelial cells. Immunohistochemistry confirmed the presence of CD31[+] and CD34[+]. The final pathological diagnosis was anastomotic hemangioma. No recurrence was observed during a 40-month follow-up.

CONCLUSION: Retroperitoneal anastomotic hemangioma is a rare and benign neoplasm that may be misdiagnosed as ectopic pheochromocytoma or angiosarcoma. This case report presents and analyzes the imaging characteristics of a series of retroperitoneal anastomotic hemangiomas, which can be valuable for future diagnoses and help prevent unnecessary surgeries.

PMID:37954079 | PMC:PMC10634416 | DOI:10.3389/fonc.2023.1269631

Vascular tumors of the liver: A brief review

Fetched: November 14th, 2023, 5:02am GMT by Sujata Sarangi

Ann Hepatobiliary Pancreat Surg. 2023 Nov 30;27(4):329-341. doi: 10.14701/ahbps.23-046. Epub 2023 Nov 13.

ABSTRACT

Vascular tumors of the liver are mesenchymal lesions from endothelial cells. They range from common benign lesions such as haemangioma, intermediate tumors like Kaposi sarcoma, and perivascular epithelioid cell tumor to malignant tumors such as hepatic epithelioid hemangioendothelioma and hepatic angiosarcoma in adults. Pediatric vascular tumors of the liver also include benign, locally aggressive, borderline, and malignant masses with haemangiomas being the most common benign tumors and epithelioid hemangioendothelioma being an uncommon pediatric malignancy. The list of these lesions is completed by nodular regenerative hyperplasia, solitary fibrous tumour, and hepatic small vessel neoplasms (HSVN). Some of these tumors are uncommon and rare. This review article aimed to enumerate hepatic vascular tumors along with their imaging, histopathology, molecular findings for accurate diagnosis that can result in better management.

PMID:37953709 | DOI:10.14701/ahbps.23-046

Permalink for 'Efficacy of timolol in the treatment of facial hemangioma and its effect on the proliferation and apoptosis of hemangioma stem cells'

Efficacy of timolol in the treatment of facial hemangioma and its effect on the proliferation and apoptosis of hemangioma stem cells

Fetched: November 14th, 2023, 5:02am GMT by Hongjian Zhu

Cell Mol Biol (Noisy-le-grand). 2023 Oct 31;69(10):115-120. doi: 10.14715/cmb/2023.69.10.16.

ABSTRACT

To investigate the efficacy of timolol in the treatment of facial hemangioma and the effect on the proliferation and apoptosis of hemangioma stem cells, 60 cases of children with IHs admitted to our hospital between 2020 and 2021 were selected and divided into two groups. The grouping was according to the lottery method, with 30 cases in each group. In the observation group, 0.5% timolol maleate eye drops were applied topically, and in the control group, propranolol hydrochloride tablets were administered orally to observe the efficacy of hemangioma, changes in hemangioma stem cells and the incidence of adverse reactions in both groups. Results showed that combined with the four-level score and ultrasound results, the number of effective treatment cases in the observation group was 28, which was higher than that in the control group, (P<0.05). The total number of adverse reactions in the observation group was 2, with an incidence rate. Under the intervention conditions of timolol, the proliferation level of hemangioma stem cells was inhibited, and the apoptosis rate of hemangioma stem cells increased with the increase of culture time (P<0.05). Among them, the apoptosis rate of the timolol group was higher than that of the blank control group at the same time point (P<0.05), and the difference was most significant at 48h (P<0.001). In conclusion, Timolol can effectively treat facial hemangioma in children, inhibit the proliferation of hemangioma stem cells and promote their apoptosis, with good curative effect, short treatment time and no obvious adverse reactions and it is economical and easy to accept.

PMID:37953576 | DOI:10.14715/cmb/2023.69.10.16

An Illustrative Review of the Pathomechanisms of Symptomatic Developmental Venous Anomalies

Fetched: November 14th, 2023, 5:02am GMT by Sabha Ahmed

J Comput Assist Tomogr. 2023 Nov-Dec 01;47(6):940-950. doi: 10.1097/RCT.0000000000001508. Epub 2023 Jul 22.

ABSTRACT

OBJECTIVE: Symptomatic developmental venous anomalies (DVAs) are rare. Here, we illustrate the varied clinicoradiologic profiles of symptomatic DVAs and contemplate the mechanisms that render these (allegedly) benign entities symptomatic supported by a review of literature.

METHODS: Institutional databases were searched to identify cases of symptomatic DVAs. Clinical and imaging (angiographic and cross-sectional) data of 9 cases with 11 neurovascular symptoms consequent to inflow/outflow perturbations and mechanical obstruction that manifested because of the strategic topography of underlying DVAs were analyzed. A review of the existing literature on DVAs in agreement with our case series was performed on publications retrieved from the PubMed database.

RESULTS: Symptoms secondary to venous hypertension arising from flow-related perturbations were broadly divided into those arising from restricted outflow and increased inflow. Restricted outflow occurred because of collector vein stenosis (n = 2) and collector vein/DVA thrombosis (n = 3), whereas the latter pathomechanism was initiated by arterialized/transitional DVAs (n = 2). A mechanical/obstructive pathomechanism culminating in moderate supratentorial ventriculomegaly was noted in 1 case. One patient was given a diagnosis of hemorrhage associated with a cavernoma.

CONCLUSIONS: Awareness and contextualization of potential flow-related perturbations and mechanical insults that render DVAs symptomatic aid in accurate diagnosis, management, and prognostication.

PMID:37948370 | DOI:10.1097/RCT.0000000000001508

Multimodal imaging of a macular retinal capillary hemangioma

Fetched: November 12th, 2023, 5:02am GMT by J Almeida

Arch Soc Esp Oftalmol (Engl Ed). 2023 Nov 10:S2173-5794(23)00182-2. doi: 10.1016/j.oftale.2023.11.006. Online ahead of print.

NO ABSTRACT

PMID:37951355 | DOI:10.1016/j.oftale.2023.11.006

Surgery for primary splenic tumors and metastases of the spleen

Fetched: November 10th, 2023, 5:02am GMT by F Huettl

Chirurgie (Heidelb). 2023 Nov 9. doi: 10.1007/s00104-023-01978-8. Online ahead of print.

ABSTRACT

The correct indications for surgical treatment of primary splenic tumors as well as metastases of the spleen are challenging due to the rarity of the various entities. Primary solid splenic tumors include benign lesions, such as hemangiomas, hamartomas and sclerosing angiomatous nodular transformation (SANT) of the spleen. In these cases, surgical treatment is indicated only in the case of inconclusive imaging and after careful consideration of the risk-benefit ratio, even in the case of pronounced symptoms. In contrast, primary angiosarcoma or undifferentiated pleomorphic sarcoma as highly malignant tumors represent an urgent indication for surgery. Although more frequent than primary splenic malignancies, secondary splenic tumors are also not that frequent. Solitary splenic metastases are rare; however, from an oncological point of view they can be treated by resection. In the case of oligometastasis with splenic involvement, splenectomy is used only as part of a palliative concept in cases of pronounced symptoms or in the context of cytoreductive surgery. In general, the laparoscopic approach is to be preferred when the operation is technically feasible as it is associated with fewer pulmonary and infectious complications and a shorter hospital stay. In addition, to reduce the risk of severe infections after splenectomy, the option of partial splenectomy should be considered, especially for benign lesions. A thorough informing of the patient regarding both intraoperative and perioperative risks as well as potential long-term sequelae, especially severe infectious diseases, is an essential component of informed consent before surgery.

PMID:37946024 | DOI:10.1007/s00104-023-01978-8

Permalink for 'Inhibition of infantile hemangioma growth and promotion of apoptosis via VEGF/PI3K/Akt axis by 755-nm long-pulse alexandrite laser'

Inhibition of infantile hemangioma growth and promotion of apoptosis via VEGF/PI3K/Akt axis by 755-nm long-pulse alexandrite laser

Fetched: November 10th, 2023, 5:02am GMT by Chen Ke

Biomed J. 2023 Nov 7:100675. doi: 10.1016/j.bj.2023.100675. Online ahead of print.

ABSTRACT

BACKGROUND: Infantile hemangioma (IH) is a common vascular tumor in female infants, which can lead to aesthetic issues and facial scarring. This study aimed to investigate the inhibitory effects and underlying mechanisms of 755 nm long-pulsed alexandrite laser on IH.

METHODS: Hemangioma endothelial cells (HemECs) were exposed to 755 nm long-pulsed alexandrite laser to evaluate its impact on cell proliferation and apoptosis. A patient-derived xenograft model was established to assess the inhibitory effects of laser treatment on IH in vivo.

RESULTS: In vitro, 755 nm long-pulsed alexandrite laser effectively suppressed the proliferation of HemECs and induced cell apoptosis. Laser treatment significantly inhibited the volume and weight of tumors, accompanied by significant downregulation of vascular endothelial growth factor A (VEGFA), vascular endothelial growth factor receptor 2 (VEGFR2), phosphatidylinositol 3-kinase (PI3K), and protein kinase B (Akt) expression levels in both hemangioma cells and tumors. Additionally, laser treatment resulted in the conversion of VEGFA165a to VEGFA165b. TUNEL staining demonstrated increased apoptosis in tumor cells after laser treatment, along with upregulation of cleaved caspase-3 and Bax, and downregulation of Bcl-2.

CONCLUSION: In addition to the principle of selective photothermal decomposition, modulation of the VEGF/PI3K/Akt axis may serve as a potential mechanism for IH treatment using a long pulse-width 755 nm laser. This sheds valuable light on the molecular mechanisms underlying IH pathogenesis and potential therapeutic targets while providing a theoretical basis for the safe and efficient management of proliferative IH using laser therapy.

PMID:37944864 | DOI:10.1016/j.bj.2023.100675

Generalized cystic lymphangiomatosis incidentally diagnosed in an asymptomatic adult: Imaging findings of a very rare case

Fetched: November 10th, 2023, 5:02am GMT by Zhale Tabrizi

Radiol Case Rep. 2023 Oct 28;19(1):130-135. doi: 10.1016/j.radcr.2023.10.017. eCollection 2024 Jan.

ABSTRACT

Lymphangiomas are benign lesions of vascular origin with lymphatic differentiation, most commonly found in the head and neck. Generalized lymphangiomatosis is a very rare condition in adults, which is characterized by a diffuse proliferation of lymphatic vessels. The lymphangioma is composed of lymphatic endothelium-lined cystic spaces. This condition can be histologically differentiated from other vascular disorders such as cavernous or capillary hemangioma. However, many cases of lymphangioma can be confused with other vascular disorders, because of overlapping histologic findings. radiologic examinations, such as CT scan and MR imaging, are useful for assessing the morphologic feature and also the extent of disease, it is important to know the radiologic findings of generalized lymphangiomatosis. In this paper, we report a case of generalized lymphangiomatosis in a 42-year-old male who presented with left flank pain and hematuria. The first differential diagnosis was renal colic; hence he underwent an abdominopelvic computed tomography scan (CT scan). In the performed CT scan multiple cystic lesions were seen in the liver and spleen. Also, lytic lesions were seen in bones. CT-guided biopsy was performed and the result was compatible with generalized lymphangiomatosis, confirmed by cytology. Generalized lymphangiomatosis is a rarely reported disease in children and young adults. Delayed diagnosis in older patients or misdiagnosis is common due to its rarity and nonspecific clinical presentation. Different imaging modalities can incidentally diagnose the disease in asymptomatic patients. So radiologists should be aware of the disease manifestations in imaging modalities to diagnose the disease sooner and help the clinician start the therapy if needed.

PMID:37941984 | PMC:PMC10628799 | DOI:10.1016/j.radcr.2023.10.017

Hepatic hemangioma: proportion and predictor of surgical treatment with emphasis on its growth rate

Fetched: November 9th, 2023, 5:02am GMT by Young Cheol Shin

Korean J Intern Med. 2023 Nov;38(6):818-830. doi: 10.3904/kjim.2023.062. Epub 2023 Nov 1.

ABSTRACT

BACKGROUND/AIMS: There are limited studies on the management of hepatic hemangiomas (HHs). We investigated the proportion and predictors of surgical resection and analyzed HH growth rates in addition to associated factors.

METHODS: A retrospective case-control study of patients treated in 2 centers was conducted. Thirty-six patients who underwent surgical resection were assigned to the case group. Patients who did not undergo surgical treatment were randomly sigselected at a 1:10 ratio and assigned to the control group (n = 360). Baseline characteristics, clinical course and surgical outcomes were analyzed.

RESULTS: The proportion of surgically treated HH patients was 0.3% (36 per 11,049). The longest diameter at diagnosis (mean ± standard deviation) was 7.7 ± 5.2 cm in the case group and 2.4 ± 1.8 cm in the control group (p < 0.001). In the multivariate analysis, the presence of more than 2 HHs (odds ratio [OR] 7.64, 95% confidence interval [CI] 1.40-41.72) and a growth rate of more than 4.8%/year (OR 30.73, 95% CI 4.86-194.51) were independently associated with surgical treatment. Symptom development during follow-up was related to HH size > 10 cm (OR 10.50, 95% CI 1.06-103.77, p = 0.04). The subgroup analysis showed substantial growth in 41.3% with an overall mean annual growth rate of 0.14 cm.

CONCLUSION: Approximately one in 300 patients with an HH underwent surgical treatment. Multiple HHs and a growth rate of more than 4.8%/year were indications for surgical treatment. Nearly half of the HHs showed growing pattern in our study.

PMID:37939665 | PMC:PMC10636540 | DOI:10.3904/kjim.2023.062

Anastomosing haemangioma of the mediastinum: Clinicopathological series with radiological and genetic characterisation

Fetched: November 9th, 2023, 5:02am GMT by Nicholas J Caldwell

Histopathology. 2023 Nov 7. doi: 10.1111/his.15085. Online ahead of print.

ABSTRACT

AIMS: Anastomosing haemangiomas are benign tumours with anastomosing vascular channels that may mimic angiosarcoma. While anastomosing haemangiomas have been described in diverse locations, particularly the abdominal/paraspinal region, data on anastomosing haemangiomas in the mediastinum remain limited. We report the clinicopathological, radiological and molecular characteristics of the largest single-institutional series of mediastinal anastomosing haemangiomas.

METHODS AND RESULTS: In our pathology archives in 2011-23, we reviewed all vascular lesions involving the mediastinum and identified seven anastomosing haemangiomas. Clinical information was abstracted from medical charts; available radiological imaging was reviewed. Targeted DNA-based next-generation sequencing (447 genes, including GNAQ and GNA11) was performed on five cases. The seven patients included five women and two men, with an age range of 55-77 (median = 72) years. Of the six tumours with available radiology, two each were in the prevascular, visceral and paravertebral mediastinum, with lobulated peripheral enhancement in all tumours examined with contrast enhancement. Six patients underwent tumour resection; one patient received proton radiotherapy. Microscopically, each tumour was solitary and characterised by anastomosing capillary-sized vessels lined by hobnail endothelial cells. Fibrin microthrombi, hyaline globules and extramedullary haematopoiesis were common. In the five tumours analysed by next-generation sequencing, GNAQ p.Q209P was identified in one tumour; no additional reportable alterations were identified in the remaining cases. No recurrence was noted in the four patients with available follow-up of 3-58 (median = 9.5) months after resection.

CONCLUSION: While mediastinal anastomosing haemangiomas can microscopically mimic angiosarcoma, awareness of this entity and radiological correlation may help to circumvent this diagnostic pitfall.

PMID:37936489 | DOI:10.1111/his.15085

Infectious complications of vascular anomalies treated with sirolimus: A systematic review

Fetched: November 8th, 2023, 5:01am GMT by Rachel Kalbfell

Pediatr Blood Cancer. 2024 Jan;71(1):e30758. doi: 10.1002/pbc.30758. Epub 2023 Nov 6.

ABSTRACT

BACKGROUND AND OBJECTIVES: Initially developed as immunosuppressive agents, mammalian target of rapamycin (mTOR) inhibitors are currently used widely in the management of vascular malformations and tumors. The incidence of infectious complications in the vascular anomalies (VA) population is not well defined. The goal of this systematic review was to better define the types and severity of reported infectious complications in patients with VAs treated with mTOR inhibition.

METHODS: This was a systematic review conducted following PRISMA guidelines evaluating all research articles focused on infectious complications in patients with VAs treated with sirolimus or everolimus. Thirty articles including 1182 total patients and 316 infections (in 291 unique patients) were ultimately included.

RESULTS: The majority of infections were viral upper respiratory (n = 137, 54%), followed by pneumonia (n = 53, 20%), and cutaneous infections (n = 20, 8%). There were six total infection-related fatalities, which all occurred in patients younger than 2 years. Two cases of Pneumocystis jirovecii pneumonia (PJP) were reported. These were infants with kaposiform hemangioendothelioma (KHE) who were also treated with steroids and did not receive PJP prophylaxis. Almost one-third (n = 96, 32%) of infectious complications were graded 3-4 according to Common Terminology Criteria for Adverse Events (CTCAE) criteria. Details of patient age, subtype of VA, and timing of infection were lacking from many reports.

CONCLUSIONS: Most infectious complications reported in patients with VA on mTOR inhibitors were viral respiratory infections and non-severe. Bacteremia, infectious fatalities, and PJP are exceedingly rare. Future studies are needed to clarify the spectrum of infectious risks in VA patients and to provide guidance for infection prevention.

PMID:37933207 | DOI:10.1002/pbc.30758

Sturge-Weber Syndrome and Hippocampal Sclerosis: Two Epileptogenic Conditions in One Patient

Fetched: November 8th, 2023, 5:01am GMT by João Vitor Gerdulli Tamanini

Neurol India. 2023 Sep-Oct;71(5):1090-1091. doi: 10.4103/0028-3886.388109.

NO ABSTRACT

PMID:37929486 | DOI:10.4103/0028-3886.388109

Urethral hemangioma: How to investigate as a cause of hematuria after male sexual activities

Fetched: November 7th, 2023, 5:02am GMT by Kimiyasu Ishikawa

IJU Case Rep. 2023 Sep 24;6(6):454-457. doi: 10.1002/iju5.12646. eCollection 2023 Nov.

ABSTRACT

INTRODUCTION: Urethral hemangioma is an extremely rare occurrence and is not typically considered a common cause of hematuria. Since 2000, only 22 male cases have been reported.

CASE PRESENTATION: A 45-year-old man presented with recurrent painless gross hematuria and the passage of blood clots after ejaculation. The patient underwent a transurethral resection of a 6-mm hemangioma. This isolated sessile lesion was situated between the distal end of the verumontanum and the external sphincter, following an induced erection. The patient remained asymptomatic during the 1-month follow-up visit.

CONCLUSION: This study included the assessment of patient symptoms, diagnoses, and treatments and the literature review of 22 patients. We propose that relaxation of the external urethral sphincter muscle under general anesthesia and artificially inducing an erection can aid in the identification of urethral hemangiomas near the verumontanum during cystourethroscopy.

PMID:37928305 | PMC:PMC10622190 | DOI:10.1002/iju5.12646

Retroperitoneal capillary arteriovenous malformation mimicking a malignant neoplasm

Fetched: November 7th, 2023, 5:02am GMT by Kazuki Yanagida

IJU Case Rep. 2023 Aug 28;6(6):398-401. doi: 10.1002/iju5.12632. eCollection 2023 Nov.

ABSTRACT

INTRODUCTION: Retroperitoneal tumors account for 0.2% of all neoplasms. Among these tumors, retroperitoneal vascular malformations are particularly rare, with most previously reported cases being venous malformations.

CASE PRESENTATION: A 72-year-old woman was diagnosed with a retroperitoneal tumor on abdominal computed tomography. The 27-mm diameter tumor was located away from the right kidney and major vessels in the right perirenal adipose tissue. Contrast-enhanced computed tomography revealed a heterogeneously enhanced tumor with well-defined borders. Dynamic contrast-enhanced magnetic resonance imaging revealed rapid enhancement in the arterial phase and a progressive filling-in pattern in the delayed phase. Although vascular malformation was suspected, a definitive diagnosis could not be established. The retroperitoneal tumor was excised laparoscopically for therapeutic and diagnostic purposes, and the histopathological diagnosis confirmed it as a capillary arteriovenous malformation.

CONCLUSION: Herein, we presented a rare case of retroperitoneal capillary arteriovenous malformation that was difficult to definitively diagnose preoperatively.

PMID:37928304 | PMC:PMC10622200 | DOI:10.1002/iju5.12632

Endoscopic removal of sinonasal hemangioma using vessel sealing device without tumor embolization

Fetched: November 7th, 2023, 5:02am GMT by Hong Jin Kim

Clin Case Rep. 2023 Nov 2;11(11):e8148. doi: 10.1002/ccr3.8148. eCollection 2023 Nov.

ABSTRACT

When massive bleeding is anticipated during endoscopic sinonasal tumor removal, a vessel sealing device is useful for successful tumor removal.

PMID:37927978 | PMC:PMC10622400 | DOI:10.1002/ccr3.8148

Mechanism of LEF1-AS1 regulating HUVEC cells by targeting miR-489-3p/S100A11 axis

Fetched: November 7th, 2023, 5:02am GMT by Haoran Zhang

PeerJ. 2023 Nov 1;11:e16128. doi: 10.7717/peerj.16128. eCollection 2023.

ABSTRACT

BACKGROUND: The venous malformation is the most common congenital vascular malformation and exhibits the characteristics of local invasion and lifelong progressive development. Long noncoding RNA (lncRNA) regulates endothelial cells, vascular smooth muscle cells, macrophages, vascular inflammation, and metabolism and also affects the development of venous malformations. This study aimed to elucidate the role of the lncRNA LEF1-AS1 in the development of venous malformations and examine the interaction among LEF1-AS1, miR-489-3p, and S100A11 in HUVEC cells.

METHODS: Venous malformation tissues, corresponding normal venous tissues, and HUVEC cells were used. Agilent human lncRNA microarray gene chip was used to screen differential genes, RNA expression was detected using quantitative reverse transcription PCR, and protein expression was detected using Western blotting. The proliferation, migration, and angiogenesis of HUVEC cells were assessed using CCK8, transwell, and in vitro angiogenesis tests.

RESULTS: A total of 1,651 lncRNAs were screened using gene chip analysis, of which 1015 were upregulated and 636 were downregulated. The lncRNA LEF1-AS1 was upregulated with an obvious difference multiple, and the fold-change value was 11.03273. The results of the analysis performed using the StarBase bioinformatics prediction website showed that LEF1-AS1 and miR-489-3p possessed complementary binding sites and that miR-489-3p and S100A11 also had complementary binding sites. The findings of tissue experiments revealed that the expressions of LEF1-AS1 and S100A11 were higher in tissues with venous malformations than in normal tissues, whereas the expression of miR-489-3p was lower in venous malformations than in normal tissues. Cell culture experiments indicated that LEF1-AS1 promoted the proliferation, migration, and angiogenesis of HUVEC cells. In these cells, LEF1-AS1 targeted miR-489-3p, which in turn targeted S100A11. LEF1-AS1 acted as a competitive endogenous RNA and promoted the expression of S100A11 by competitively binding to miR-489-3p and enhancing the proliferation, migration, and angiogenesis of HUVEC cells. Thus, LEF1-AS1 participated in the occurrence and development of venous malformation.

CONCLUSIONS: The expression of LEF1-AS1 was upregulated in venous malformations, and the expression of S100A11 was increased by the adsorption of miR-489-3p to venous endothelial cells, thus enhancing the proliferation, migration, and angiogenesis of HUVEC cells. In conclusion, LEF1-AS1 is involved in the occurrence and development of venous malformations by regulating the miR-489-3p/S100A11 axis, which provides valuable insights into the pathogenesis of this disease and opens new avenues for its treatment.

PMID:37927791 | PMC:PMC10625350 | DOI:10.7717/peerj.16128

Single-nucleus DNA sequencing reveals hidden somatic loss-of-heterozygosity in Cerebral Cavernous Malformations

Fetched: November 7th, 2023, 5:02am GMT by Andrew K Ressler

Nat Commun. 2023 Nov 2;14(1):7009. doi: 10.1038/s41467-023-42908-w.

ABSTRACT

Cerebral Cavernous Malformations (CCMs) are vascular malformations of the central nervous system which can lead to moderate to severe neurological phenotypes in patients. A majority of CCM lesions are driven by a cancer-like three-hit mutational mechanism, including a somatic, activating mutation in the oncogene PIK3CA, as well as biallelic loss-of-function mutations in a CCM gene. However, standard sequencing approaches often fail to yield a full complement of pathogenic mutations in many CCMs. We suggest this reality reflects the limited sensitivity to identify low-frequency variants and the presence of mutations undetectable with bulk short-read sequencing. Here we report a single-nucleus DNA-sequencing approach that leverages the underlying biology of CCMs to identify lesions with somatic loss-of-heterozygosity, a class of such hidden mutations. We identify an alternative genetic mechanism for CCM pathogenesis and establish a method that can be repurposed to investigate the genetic underpinning of other disorders with multiple somatic mutations.

PMID:37919320 | PMC:PMC10622526 | DOI:10.1038/s41467-023-42908-w

Global research trends of infantile hemangioma: A bibliometric and visualization analysis from 2000 to 2022

Fetched: November 4th, 2023, 5:02am GMT by Qian Lin

Heliyon. 2023 Oct 21;9(11):e21300. doi: 10.1016/j.heliyon.2023.e21300. eCollection 2023 Nov.

ABSTRACT

BACKGROUND: Infantile hemangioma (IH) has received global attention, resulting in a significant volume of literature. However, there is a lack of bibliometric analyses specifically focusing on IH publications. This study aims to fill this gap by conducting a comprehensive analysis of IH publications, investigating their characteristics, contribution distribution, and developmental trends. By enhancing our understanding of IH and identifying potential research topics and collaborators, this study will contribute to the advancement of the field.

METHODS: A total of 4333 articles and reviews on IH were collected from the Web of Science (WoS) database, spanning the years 2000-2022. The study encompassed a comprehensive analysis of IH publications, evaluating their quantity and quality. Additionally, we profiled publishing groups based on country, institution, author publication records, and collaboration networks. Lastly, we identified and summarized the prominent research topics.

RESULTS: Annual publications on IH have increased over the past 20 years. The United States has the highest number of publications and the highest total number of citations. Pediatric Dermatology was the most influential journal in the IH field. The citation analysis indicated that the articles published by Léauté-Labrèze in 2008 had the highest number of citations. The articles published by North PE in 2000 and Boye E in 2001 laid a certain research foundation for this field. Concerning institutions, most of the cooperative relationships were established in the same country/region. The United States has the largest number of scientific research institutions and IH researchers, leading most of the cross-country collaboration. The University of California, San Francisco, Medical College of Wisconsin, Harvard University, and Shanghai Jiaotong University were the research centers that published the most IH-related research. Frieden IJ, Mulliken JB, and Drolet BA were the top three most influential authors. Frieden IJ, Garzon MC, and Mulliken JB were the top three authors with the most cited frequency. In addition, keywords and keyword co-occurrence networks prompted that the pathological mechanism of IH, clinical analysis, and other vascular anomalies are research hotspots. Analysis of trending topics suggests that research on IH has evolved from treatment-focused studies towards investigations of other vascular diseases and a series of clinical case studies. Currently, clinical case studies receive the most attention in the field.

CONCLUSIONS: This comprehensive bibliometric study provides a thorough analysis of post-2000 publications in the field of IH, offering insights into current research trends for the first time. The findings suggest that future investigations will continue to prioritize understanding IH mechanisms, treatment approaches, and treatment evaluation. Furthermore, the exploration of other vascular diseases and the inclusion of clinical case studies are expected to contribute to advancements in IH clinical practice. By identifying potential collaborators, partner institutions, and new research avenues, this study offers valuable guidance for future in-depth research on IH.

PMID:37920523 | PMC:PMC10618776 | DOI:10.1016/j.heliyon.2023.e21300

Regorafenib in advanced solitary fibrous tumour: Results from an exploratory phase II clinical study

Fetched: November 3rd, 2023, 5:02am GMT by Silvia Stacchiotti

Eur J Cancer. 2023 Oct 18;195:113391. doi: 10.1016/j.ejca.2023.113391. Online ahead of print.

ABSTRACT

BACKGROUND: To investigate the activity of regorafenib in advanced solitary fibrous tumour (SFT).

METHODS: An Italian monocentric investigator-initiated exploratory single-arm Phase II trial was conducted of regorafenib in adult patients with advanced and progressive SFT, until progression or limiting toxicity. Prior treatment with antiangiogenics was allowed. Primary and secondary end-points were: overall response rate (ORR) by Choi criteria, and ORR by RECIST, progression-free survival (PFS), overall survival (OS).

RESULTS: From January 2016 to February 2021, 18 patients were enroled [malignant-SFT = 13; dedifferentiated-SFT (D-SFT) = 4; typical-SFT (T-SFT) = 1]. Fourteen patients were pre-treated, in 12 cases with antiangiogenics (median [m-] lines of treatment = 3). Sixteen patients were evaluable for response (one screening failure; one early discontinuation). Six/16 (35.2%) required a definitive dose reduction. ORR by Choi was 37.5% (95% CI: 15.2-64.6), with 6/16 (37.5%) partial responses (PR), 6/16 (37.5%) stable disease (SD) and 4/16 (25%) progressions; 5/6 responses occurred in patients pre-treated with antiangiogenics. No responses were detected in D-SFT. Best RECIST responses were: 1/16 (6.2%) PR, 12/16 (75%) SD, 3/16 (18.8%) progressions. At 48.4 month m-FU, m-PFS by Choi was 4.7 (inter-quartile range: 2.4-13.1) months, with 31.2% patients progression-free at 1 year.

CONCLUSION: Regorafenib showed activity in SFT, with 30% patients free-from-progression at one year. Responses were observed also in patients pretreated and refractory to another antiangiogenic agents. However, ORR and m-PFS were lower than reported with other antiangiogenics, and this was possibly due to discrepancies in the patient population and the high-rate of dose reductions.

PMID:37918286 | DOI:10.1016/j.ejca.2023.113391

Permalink for 'A retrospective analysis of infantile haemangioma referrals: a comparison with the IH European Task Force 'Infantile Haemangioma Referral Score''

A retrospective analysis of infantile haemangioma referrals: a comparison with the IH European Task Force 'Infantile Haemangioma Referral Score'

Fetched: November 3rd, 2023, 5:02am GMT by Nicole Fagan

Ir J Med Sci. 2023 Nov 2. doi: 10.1007/s11845-023-03556-7. Online ahead of print.

ABSTRACT

BACKGROUND: Infantile haemangiomas (IH) are common vascular tumours of infancy that can have significant complications. The IH European Task Force developed the IH Referral Score (IHReS) to help non-specialists quickly identify IH that need to be referred to optimise outcomes in high-risk lesions.

AIMS: The aim of this study was to assess the quality of IH referrals to a national care centre and compare the IH referred to the IHReS tool.

METHODS: This was a retrospective cross-sectional study that collected data from a random sample of 56 IH patients over a 13-month period.

RESULTS: Less than 10% included a photograph (8.93%) and a minority of referrals included if there were complications including ulceration (1.79%) or function threatening (8.93%). Three-quarters of patients satisfied the IHReS criteria.

CONCLUSION: The IHReS screening tool is a convenient and efficient resource for non-experts to identify children who require referral to a specialist centre.

PMID:37917305 | DOI:10.1007/s11845-023-03556-7

Endoscopic approach to geniculate ganglion: a multicentric experience

Fetched: November 3rd, 2023, 5:02am GMT by Giulia Molinari

Eur Arch Otorhinolaryngol. 2023 Nov 2. doi: 10.1007/s00405-023-08294-1. Online ahead of print.

ABSTRACT

PURPOSE: A variety of lesions could arise from the GG area, or extend into this region from adjacent sites. The management of perigeniculate lesions includes observation, surgery, and radiation, according to the nature, the size of the lesion, and the accompanying symptoms. Preliminary experiences on the exclusive transcanal endoscopic approach to the GG area have shown safety and feasibility avoiding of any postauricular incision, or brain manipulation. The experience from two referral centers on patients treated for a GG lesion with a totally endoscopic approach is herein reported.

METHODS: Data about patients who underwent exclusive endoscopic approach to the GG area at the Otolaryngology Departments of the University Hospitals of Modena and Bologna between May 2017 and February 2022 were retrospectively collected.

RESULTS: The total number of patients included in our study was 11. 10 patients (91%) had progressive unilateral facial paralysis and 1 patient (11%) presented with chronic otorrhea. The mean largest diameter of the treated lesions was of 8 mm. The resection was extended to the fundus of the IAC in 2 patients (expanded approach). The remaining 9 patients (82%) underwent partial ossicular replacement prosthesis (PORP). No major complications occurred. Facial nerve outcomes were good in all patients and the mean ABG worsened from 12 dB pre-operatively to 22 dB post-operatively.

CONCLUSIONS: The exclusively endoscopic approach to GG lesions represents a viable alternative to traditional microscopic approaches and may be included in the armamentarium of ear surgeons.

PMID:37917164 | DOI:10.1007/s00405-023-08294-1

Giant papillary hemangioma-A rare tumor with an exceptional size

Fetched: November 3rd, 2023, 5:02am GMT by Maryam Abdo

J Cutan Pathol. 2023 Nov 2. doi: 10.1111/cup.14554. Online ahead of print.

ABSTRACT

Papillary hemangioma (PH) is a recently described vascular tumor with a predilection for the skin of the head and neck. Histopathologically, it is characterized by a bland endothelial proliferation arranged in a papillary configuration, bearing resemblance to glomeruloid hemangioma seen in the context of polyneuropathy, organomegaly, endocrinopathy, monoclonal protein, skin changes syndrome. The largest cutaneous PH reported to date measured 1.5 cm in greatest dimension. Here, we report a case of PH with an alarming size of 10 cm. We present this case to add to the limited literature on this rare tumor, highlight the histopathologic differences between PH and its mimics, and emphasize the variable nature of PH tumor size.

PMID:37916657 | DOI:10.1111/cup.14554

Permalink for 'Intraoral epithelioid and spindle cell hemangioma associated with pseudoepitheliomatous hyperplasia: An immunohistochemical study'

Intraoral epithelioid and spindle cell hemangioma associated with pseudoepitheliomatous hyperplasia: An immunohistochemical study

Fetched: November 3rd, 2023, 5:02am GMT by Evânio Vilela Silva

J Cutan Pathol. 2023 Nov 2. doi: 10.1111/cup.14555. Online ahead of print.

NO ABSTRACT

PMID:37916647 | DOI:10.1111/cup.14555

A case report on enormous haemangioma of liver

Fetched: November 3rd, 2023, 5:02am GMT by Pratik Baral

Ann Med Surg (Lond). 2023 Oct 4;85(11):5782-5784. doi: 10.1097/MS9.0000000000001360. eCollection 2023 Nov.

ABSTRACT

INTRODUCTION: Hemangiomas are the most common benign liver tumour. These tumours arise from the proliferation of vascular endothelial cells and increase in size owing to dilation. If their diameter exceeds 5 cm, they are classified as giant hemangiomas, while those surpassing 15 cm are considered enormous hemangiomas.

CASE PRESENTATION: A 38-year-old female patient presented with complaints of abdominal fullness for 18 months. Contrast-enhanced computed tomography was performed and two hemangiomas were diagnosed; the , larger one was an enormous hemangioma of size 20 × 16 cm. Non-anatomical hepatic resection was performed to remove the hemangiomas. The patient recovered well, without any complications.

CLINICAL DISCUSSION: Hepatic hemangiomas are common, but hemangiomas greater than 15 cm in size are rare. They usually require no treatment unless the patient is symptomatic. Hepatectomy and enucleation of hemangioma are the most common surgical procedure for such hemangioma.

CONCLUSIONS: Rarely, large hepatic hemangioma can be the cause of abdominal fullness lasting for months. Often, surgical intervention is required.

PMID:37915671 | PMC:PMC10617932 | DOI:10.1097/MS9.0000000000001360

Supratentorial hemangioblastoma: correlation between phenotype, gender and vascular territory affected

Fetched: November 3rd, 2023, 5:02am GMT by Yosef Laviv

Neurosurg Rev. 2023 Oct 25;46(1):281. doi: 10.1007/s10143-023-02194-y.

ABSTRACT

Supratentorial hemangioblastomas are rare, vascular lesions. The presence of peri-tumoral cysts and edema has meaningful clinical, diagnostic and therapeutic implications. Nevertheless, the pathogenesis of both cyst and edema formation is not fully understood. This study sought to determine if the radiologic phenotype of supratentorial hemangioblastoma is affected by the different cerebral arterial circulations. Review of the English-language literature from 1973 to 2023 yielded 53 cases of parenchymal supratentorial hemangioblastomas eligible for analysis. Patients were divided by the vascular territorial distribution of the lesions: anterior circulation (n = 36) or posterior circulation (n = 17), and the groups were compared for demographic, clinical, radiologic and molecular variables. Univariate analyses yielded a significant difference between the groups in five variables. Cystic changes and "classic" radiological phenotype were associated with hemangioblastomas of the posterior circulation (OR = 0.19, p = 0.045 and OR = 0.287, p = 0.048, respectively), while female gender, significant peritumoral edema and purely solid phenotype were associated with hemangioblastomas of the anterior circulation (OR = 3.384, p = 0.045 and OR = 5.25, p = 0.05 and OR = 14.0, p = 0.015; respectively). On multivariate analysis, solid phenotype and female gender remained significantly associated with the anterior circulation (OR = 36.04, p = 0.014 and OR = 4.45, p = 0.045). The incidence of von-Hippel Lindau disease was higher in the anterior-circulation group. Cystic tumors were present in all females in the posterior-circulation group compared to 43.4% in the anterior-circulation group (OR = 20.714, 95% CI 1.061 to 404.122; p = 0.045). Based on historical cases of supratentorial hemangioblastoma, this study shows that different tumor phenotypes are associated with the different cerebral circulations. Gender was also associated with differences in tumor distribution and radiologic phenotype. These novel data may improve our understanding of unique vascular diseases of the central nervous system.

PMID:37875641 | DOI:10.1007/s10143-023-02194-y

Surgical and visual outcomes after vitreoretinal surgery for complex retinal capillary hemangioblastoma

Fetched: November 3rd, 2023, 5:02am GMT by Vishal R Raval

Indian J Ophthalmol. 2023 Nov;71(11):3544-3551. doi: 10.4103/IJO.IJO_3325_22.

ABSTRACT

PURPOSE: We evaluated the anatomical and functional outcomes after vitreoretinal surgery for complex retinal capillary hemangioblastoma (RCH).

METHODS: This was a retrospective case series of 15 patients (16 eyes) with tractional or combined retinal detachment (RD) managed with pars plana vitrectomy and tumor endoresection (ER) with/without feeder vessel ligation.

RESULTS: The mean age at the time of surgery was 30 years (range, 14-46 years). The most common tumor locations were inferotemporal (six eyes) and temporal (six eyes) quadrants. Indications for surgery included exudative RD with fibrovascular proliferation (eight eyes), combined RD (five eyes), vitreous hemorrhage (four eyes), and rhegmatogenous RD (two eyes). Tumor destruction was performed with laser and/or cryotherapy in nine eyes (57%) and ER in seven eyes (43%). Feeder vessel was ligated and cauterized in 10 (63%) and six eyes (37%), respectively. Anatomical success after initial surgery was 50% (eight eyes), which improved to 88% (14 eyes) after they underwent a repeat procedure for recurrent RD (eight eyes). At the last visit, visual acuity improved in seven eyes (44%), was stable in four eyes (25%), and worsened in five eyes (31%) with a mean follow-up of 29 months (6-79 months). Comparison between the ER group and the laser/cryotherapy group revealed no significant difference in final retinal reattachment rate (89% vs. 86%, P > 0.05), with better visual outcomes in laser/cryotherapy group (57% vs. 78%, P < 0.05).

CONCLUSION: Pars plana vitrectomy with/without tumor endoresection can be a safe and effective treatment option for complex RCHs, achieving good tumor control and anatomical success with limited functional success.

PMID:37870022 | DOI:10.4103/IJO.IJO_3325_22

False-positive Imaging for Papillary Thyroid Cancer Caused by Intraosseous Hemangiomas

Fetched: November 2nd, 2023, 5:02am GMT by Heejoo Kang

JCEM Case Rep. 2023 Sep 27;1(5):luad102. doi: 10.1210/jcemcr/luad102. eCollection 2023 Sep.

ABSTRACT

Two patients with papillary thyroid carcinoma and an elevated thyroglobulin had false-positive imaging studies from intraosseous hemangiomas (IH). A 62-year-old man presented with a palpable lytic skull mass suspicious for a bone metastasis after computed tomography (CT) and magnetic resonance imaging (MRI) scans. Surgical excision confirmed an IH. The second patient is a 64-year-old woman whose I-123 whole-body scan with single photon emission computed tomography/CT demonstrated radioiodine uptake in the right frontal bone. Her MRI and CT scans were also consistent with an IH. These cases reveal the limitations of nuclear imaging and of CT and MRI scans in distinguishing metastatic differentiated thyroid cancer from IH in patients with lytic bone lesions. Because no imaging studies are definitive for an IH, bone cranial lesions may warrant resection to establish a diagnosis and avoid potential brain invasion by a malignancy or unnecessary radioiodine treatment.

PMID:37908210 | PMC:PMC10612475 | DOI:10.1210/jcemcr/luad102

Permalink for 'Late third trimester diagnosis of congenital giant hemangioma complicated by the Kasabach-Merritt phenomen: a case report and literature review'

Late third trimester diagnosis of congenital giant hemangioma complicated by the Kasabach-Merritt phenomen: a case report and literature review

Fetched: November 2nd, 2023, 5:02am GMT by I Fantasia

J Matern Fetal Neonatal Med. 2023 Dec;36(2):2274803. doi: 10.1080/14767058.2023.2274803. Epub 2023 Oct 31.

ABSTRACT

Objective. To describe the case of a large cervical mass diagnosed in the late third trimester with development of Kasabach-Merritt phenomenon (KMP) in the immediate postnatal period, along with a literature review.Methods. Description of case-report and literature search through Medline/Pubmed, performed from inception to December 2022 for articles relating to the pre and postnatal diagnosis of KMP.Results. A 36-year-old multiparous woman was admitted to hospital for contractions at 40 weeks of gestation, in an otherwise uneventful pregnancy. Admission's ultrasound showed the presence of a voluminous mass of 14x15 cm of the posterior side of the neck, highly vascularized, and no signs of hemodynamic imbalance. Postnatally, blood tests showed the presence of severe anemia and thrombocytopenia requiring several transfusions of blood, plasma, platelets and clotting factors. Due to the association of congenital hemangioma and thrombocytopenia a diagnosis of KMP was made. After attempts of conservative treatment, surgical removal was needed to stop the hematological cascade with regression of symptoms. The review of the literature identified 14 articles including 9 cases of prenatally suspected KMP and 6 diagnosed in the immediate postnatal period and without signs of fetal hydrops. Adverse perinatal outcome, in terms of postnatal death/termination of pregnancy, was observed in 67% of cases (6/9) in the prenatally suspected group and 33% of cases in those with a postnatal diagnosis of KMP. Fetal hydrops was present in 83% of cases with adverse perinatal outcome.Conclusions. The Kasabach-Merrit syndrome is a rare condition, which can have a dangerous evolution when it develops in utero or in the immediate postnatal period carrying a risk of perinatal mortality of approximately 50%. Even if the fetus shows no signs of anemia or heart failure, the risk of developing it in the immediate postnatal period is high and should be mentioned to the couple.

PMID:37908053 | DOI:10.1080/14767058.2023.2274803

Updates to the Management of Gorham-Stout Disease and Osseous Vascular Lesions in the Head and Neck

Fetched: November 1st, 2023, 5:01am GMT by Andrea B Burke

Oral Maxillofac Surg Clin North Am. 2023 Oct 28:S1042-3699(23)00058-4. doi: 10.1016/j.coms.2023.09.004. Online ahead of print.

ABSTRACT

Osseous vascular anomalies can be characterized as vascular tumors or malformations. Classification is vital for prognosis and treatment. Much remains unknown about conditions such as Gorham-Stout disease. Treatments target the proposed genetic pathways such as PI3KCA/AKT/mTOR pathway.

PMID:37903662 | DOI:10.1016/j.coms.2023.09.004

Orbital fibrous capsule formation with abscess found 3 years after penetrating injury from a wooden chopstick

Fetched: November 1st, 2023, 5:01am GMT by Saori Kawamoto

Orbit. 2023 Oct 30:1-6. doi: 10.1080/01676830.2023.2269237. Online ahead of print.

ABSTRACT

Intraorbital wooden foreign bodies are sometimes difficult to diagnose because of nonspecific clinical manifestations and diversity of imaging characteristics. We herein report a case involving a 72-year-old woman with a history of trauma induced by a coated wooden chopstick 3 years prior. Two years after the incident, computed tomography (CT) scan revealed an intraorbital mass that was initially diagnosed as an intraorbital hemangioma. The patient presented with hyperemia, impairment of ocular movement, and optic neuropathy in her right eye. Magnetic resonance imaging (MRI) showed granulation tissue and an abscess around a foreign body, which was compressing the eyeball. Surgical extraction of the foreign body was performed, leading to resolution of symptoms. The depiction of wooden foreign bodies by imaging is complicated and affected by several factors, increasing the risk of delayed diagnosis. To avoid permanent sequelae, MRI might be helpful because its imaging capabilities are superior to those of CT.

PMID:37902564 | DOI:10.1080/01676830.2023.2269237

PHACE(S) Syndrome with Ocular Involvements and No Periocular Hemangioma

Fetched: November 1st, 2023, 5:01am GMT by Aisha Sheriff Kalambe

Case Rep Ophthalmol. 2023 Sep 22;14(1):477-483. doi: 10.1159/000533887. eCollection 2023 Jan-Dec.

ABSTRACT

PHACE(S) syndrome is a neurocutaneous disorder with a hallmark finding of an infantile facial hemangioma (IFH) >5 cm. Eye examination of patients with PHACE(S) syndrome with no IFH at periorbital region is reported to be of low yield. We report a unique case of the syndrome with ocular manifestations without periorbital IFH or systemic findings. A 3-week-old female infant with right periauricular IFH >5 cm, extending to the neck and cheek and lower lip IFH was presented. Examination revealed pseudoptosis due to microphthalmia with esotropia and hypertropia. Both corneas were clear with diameters of 5 mm and 10 mm, right eye (RE) and left eye (LE), respectively. There was a posterior polar cataract with a poor view of the fundus RE. Ocular B-scan and magnetic resonance imaging (MRI) findings were suggestive of a dysmorphic globe, vitreous hemorrhage, spherophakia and persistent fetal vasculature RE and normal findings LE. Clinical evaluation, MRI, and MR angiography revealed no other systemic abnormalities. Subsequent follow-up visits revealed progressive clouding of the cornea with neovascularization and the development of phthisis bulbi RE at which point an ocular prosthesis was placed. The IFH was managed with dye laser and with oral propranolol. At 1 year, the patient has remained stable with no development of new local or systemic anomalies, regression of the periauricular and lip IFH, and normal development of the orbital structure RE with an ocular prosthesis in situ. Ocular involvement in patients with PHACE(S) syndrome may be present without periorbital IFH. Regardless of the location of the IFH and the presence or absence of a periocular component, it is recommended that they receive a full initial ophthalmological assessment.

PMID:37901638 | PMC:PMC10601854 | DOI:10.1159/000533887

Rotationplasty type BIIIb as an effective alternative to limb salvage procedure in adults: Two case reports

Fetched: November 1st, 2023, 5:01am GMT by Zhang-Xin Chen

World J Clin Cases. 2023 Oct 6;11(28):6877-6888. doi: 10.12998/wjcc.v11.i28.6877.

ABSTRACT

BACKGROUND: Rotationplasty is often performed for malignant tumors, but type BIIIb rotationplasty is rarely reported, and there needs to be more evidence of the procedure and treatment. The purpose of this case study was to report a new direction in the use of type BIIIb rotationplasty in treating patients with limb salvage and long-term non-healing infections.

CASE SUMMARY: Case 1: A 47-year-old man underwent radiotherapy for hemangioendothelioma in his left thigh, resulting in a femoral fracture. Despite the use of plates, intramedullary nailing, and external fixators, the femoral bone failed to unite due to infectious nonunion. Multiple operations were unable to control the infection, leaving the patient immobile. We performed a modified tibia-pelvic-constrained hip rotationplasty, utilizing a constrained prosthetic hip between the tibia and pelvis following a femur resection. Two years post-surgery, the patient was able to walk with the prosthetic device without any signs of recurring infection. The corresponding functional scores were 72 points for the Musculoskeletal Tumor Society (MSTS), 53 for the Functional Mobility Assessment (FMA), 93 for the Toronto Extremity Salvage Score (TESS), and 56 for the MOS 36-item short form health survey (SF-36). Case 2: A 59-year-old woman presented with liposarcoma in her left thigh. Magnetic resonance imaging revealed tumors in the medial, anterior, and posterior femur muscles, encircling the femoral vessels and nerves. Fortunately, there were no symptoms of sciatic dysfunction, and the tumor had not invaded the sciatic nucleus. After one year of follow-up, the patient expressed satisfaction with limb preservation post-type BIIIb rotationplasty. The corresponding functional scores were 63 points for the MSTS, 47 for the FMA, 88 for the TESS, and 52 for the SF-36.

CONCLUSION: Our study suggests that type BIIIb rotationplasty may be an alternative to amputation in patients with incurable infections. For malignant tumors of the lower extremities without invasion of the sciatic nerve, type BIIIb rotationplasty remains an excellent alternative to amputation. This surgical method may prevent amputation, improve functional outcomes, and facilitate biological reconstruction.

PMID:37901010 | PMC:PMC10600845 | DOI:10.12998/wjcc.v11.i28.6877

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