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Cureus. 2024 Oct 3;16(10):e70751. doi: 10.7759/cureus.70751. eCollection 2024 Oct.
ABSTRACT
There are many benefits to early detection of prostate cancer, including improved survival rates, less invasive treatment options, and better quality of life. Apart from traditional tests and imaging (including the prostate-specific antigen (PSA) test, digital rectal exam, biopsy, computed tomography (CT), and magnetic resonance imaging (MRI)), an imaging technique called prostate-specific membrane antigen positron emission tomography (PSMA PET) is more specific and sensitive, targeting PSMA expressed by prostate cancer cells on their surface. This case report describes a 62-year-old male with metastatic prostate adenocarcinoma and unusual PSMA uptake in the spleen. Elevated PSA levels and MRI indicated aggressive prostate cancer, confirmed by a Gleason score of 7 from biopsies. A PSMA PET/CT scan showed intense activity in both the prostate and spleen, initially suggesting metastasis. However, further imaging identified the splenic lesion as a benign hemangioma. This case showcases the importance of thorough diagnostic evaluations for distinguishing metastatic disease from benign conditions to ensure accurate diagnosis and appropriate treatment.
PMID:39364177 | PMC:PMC11448008 | DOI:10.7759/cureus.70751
Front Med (Lausanne). 2024 Sep 19;11:1399913. doi: 10.3389/fmed.2024.1399913. eCollection 2024.
ABSTRACT
INTRODUCTION: Adult diffuse hepatic hemangiomatosis (DHH) is an extremely rare disease. Consequently, its characteristics are poorly understood. Herein, we report a case of adult DHH involving both liver lobes but without extrahepatic involvement. To the best of our knowledge, this the largest reported adult DHH to date.
CASE PRESENTATION: A 51-year-old man was admitted due to abdominal distension and dyspnea. Physical examination revealed marked liver enlargement. Color Doppler, plain and contrast-enhanced computed tomography, and contrast-enhanced magnetic resonance imaging revealed a hepatic lesion sized 35.1 × 32.1 × 14.1 cm occupying nearly the entire abdominal and pelvic cavities. Diagnosis was established by liver puncture biopsy. The patient exhibited clinical signs of portal hypertension and hypersplenism, but remains free of serious DHH-related complications. He is followed up regularly, with proactive evaluation for future liver transplantation.
CONCLUSION: This case will contribute to the current knowledge on the clinical and imaging features of this rare entity.
PMID:39364018 | PMC:PMC11447642 | DOI:10.3389/fmed.2024.1399913
Front Genet. 2024 Sep 19;15:1436462. doi: 10.3389/fgene.2024.1436462. eCollection 2024.
ABSTRACT
Objective: To investigate a case of neurodevelopmental disorder caused by mutation of FBXW7. Methods: Clinical data were collected from the patient, trio-WES (whole-exome sequencing) was performed on the patient and his parents (trio), and the results were verified by Sanger sequencing. RESULTS: The patient was a 2-year and 1-month old male who presented with facial dysmorphism (prominent forehead, ocular hypertelorism, and low nasal bridge), global developmental delay, language impairment, hypertonia, labial hemangioma, hydrocele, and overgrowth. The trio-WES confirmed that the child had a pathogenic de novo FBXW7 gene variant, c.1612C>T (p.G1n538*), a heretofore unreported locus. Conclusion: This case of developmental delay, hypotonia, and impaired language (OMIM: #620012) related to a mutation in FBXW7, is a rare genetic disorder, newly identified in recent years, and seldom reported. The presence of hypertonia, labial hemangioma, and hydrocele in this child suggests significant phenotypic heterogeneity of the disease, and the discovery of new mutant loci enriches the spectrum of pathogenic variants of the disease.
PMID:39364007 | PMC:PMC11446863 | DOI:10.3389/fgene.2024.1436462
Cureus. 2024 Sep 2;16(9):e68446. doi: 10.7759/cureus.68446. eCollection 2024 Sep.
ABSTRACT
Thrombotic hemangioma with organizing/anastomosing features (THOA) is a newly identified variant within the spectrum of hemangiomas that harbor mutations in the guanine nucleotide-binding protein alpha subunit (GNA) genes (like GNAQ or GNA11). While THOA shares similarities with anastomosing hemangioma, it possesses distinct clinical and morphological characteristics that make it a separate entity. All reported cases of THOA have demonstrated benign behavior. However, histologic features such as anastomosing vascular growth, mitotic figures, and endothelial hobnailing may raise concerns for a low-grade malignant vascular neoplasm. We report the case of a 74-year-old female with an unremarkable medical history who presented with a vascular lesion on her upper torso. The lesion persisted after the initial biopsy and was re-excised, displaying similar histologic characteristics. Next-generation sequencing (NGS) revealed a GNAQ mutation (p.Q209H) in both samples. Notably, a TP53 mutation (p.R273H) was detected in the first specimen but was absent in the subsequent excision. The lesion was diagnosed as persistent THOA. This case report discusses the salient features, genetic profile, and prognosis of this uncommon lesion.
PMID:39360118 | PMC:PMC11446179 | DOI:10.7759/cureus.68446
JNMA J Nepal Med Assoc. 2024 Feb 29;62(271):211-213. doi: 10.31729/jnma.8500.
ABSTRACT
Subglottic hemangiomas are rare benign vascular tumors of infancy which involve the airway. It is a subtype of infantile hemangiomas and is a potentially life-threatening condition with a mortality rate of 50% if left untreated. Hence, early intervention in this condition is essential. Here we present a case of a 4-month-old infant, a male infant with a history of cough and noisy breathing requiring multiple hospital visits before eventually being diagnosed with subglottic hemangioma. Due to its similar presentation with other more common respiratory illnesses, the diagnosis can be missed. Oral propranolol is the first-line therapy, which was used successfully in our case.
PMID:39356782 | PMC:PMC10924481 | DOI:10.31729/jnma.8500
BMJ Case Rep. 2024 Oct 1;17(10):e259086. doi: 10.1136/bcr-2023-259086.
ABSTRACT
We present our single-centre experience of two cases of lipochoristoma, a rare lesion causing unilateral hearing loss. Differential diagnoses include the more common vestibular schwannoma, haemangioma and meningioma. Diagnosis was confirmed with fat suppression sequences on MRI. Management of both patients was with serial imaging, under the guidance of neuro-otology multidisciplinary team, due to the slow and typically non-aggressive progression of this lesion.
PMID:39353668 | DOI:10.1136/bcr-2023-259086
Int J Surg Pathol. 2024 Oct 1:10668969241286068. doi: 10.1177/10668969241286068. Online ahead of print.
ABSTRACT
Pseudomyogenic hemangioendothelioma (PMHE), a rare soft tissue tumor predominantly affecting young adults, often presents as multiple nodules in various tissue planes of a limb. Malignant transformation and metastatic disease are unusual and pose diagnostic and therapeutic challenges. A 17-year-old patient from Western India, with a history of recurrent excisions for a toe swelling presented to our center for evaluation and management. A below-knee amputation was performed, and histopathology revealed PMHE. Adjuvant therapy was deemed unnecessary given the borderline nature of the tumor. Shortly thereafter, he developed features of local recurrence and underwent above-knee amputation. An expert histopathological review confirmed the diagnosis and noted features of malignant transformation-progression to a higher grade with greater cytological atypia, confluent growth, and increased mitotic activity over time. Upon further distant progression in the lung, he was started on a palliative regimen of weekly paclitaxel, vinblastine, and propranolol but eventually succumbed to his illness. In contrast to conventional descriptions of low mitotic activity, minimal nuclear atypia, and absence of necrosis, our patient exhibited increased mitotic rates, nuclear atypia, and evolving necrosis in serial histopathological evaluations. The fulminant clinical progression within a short interval was also atypical. Our patient's clinical course underscores the need for meticulous histopathological and molecular characterization and vigilant clinical surveillance after resection in patients with PMHE. Providing the standard of care for malignant disease in the adjuvant setting is challenging owing to the rarity and the lack of treatment guidelines.
PMID:39350761 | DOI:10.1177/10668969241286068
Anticancer Res. 2024 Oct;44(10):4147-4153. doi: 10.21873/anticanres.17245.
ABSTRACT
Epithelioid hemangioendothelioma (EHE) is a rare malignant vascular tumor arising from vascular endothelial cells. This study delves into the molecular mechanisms underlying EHE, with a specific focus on the Hippo-YAP/TAZ pathway. EHE is characterized molecularly by transcriptional co-activator with a PDZ-motif (TAZ)-calmodulin binding transcription activator 1 (CAMTA1) or Yes-associated protein (YAP)-transcription factor E3 (TFE3) fusions. YAP/TAZ, a transcription co-activator, binds to transcription factors and regulates gene expression. The YAP/TAZ and its upstream Hippo pathway are involved in cell proliferation and cell contact inhibition, regulating organ size and carcinogenesis. In addition to oncogenic effects, dysfunction or gene duplication of the Hippo pathway results in a poor prognosis due to epithelial-mesenchymal transformation of epithelial cells, stem cell transformation, and increased drug resistance. Notably, the TAZ-CAMTA1 fusion is specific to EHE, and genetic alterations in the Hippo pathway other than this fusion gene are absent in EHE. The TAZ-CAMTA1 fusion is a promising therapeutic target. This review summarizes recent advances in EHE, focusing on the role of the Hippo-YAP/TAZ pathway in EHE and its potential as a therapeutic target for drug development.
PMID:39348982 | DOI:10.21873/anticanres.17245
Liver Transpl. 2024 Oct 1. doi: 10.1097/LVT.0000000000000499. Online ahead of print.
ABSTRACT
The evolving field of liver transplant (LT) oncology calls for tailored immunosuppression protocols to minimize the risk of tumor recurrence. We systematically reviewed the available evidence from inception to May 2023 regarding immunosuppression protocols used in patients undergoing LT for cholangiocarcinoma, neuroendocrine tumors (NET), hepatic-endothelial hemangioendothelioma (HEHE) and colorectal liver metastases (CRLM), to identify common practices and to evaluate their association with oncological outcomes. Studies not involving humans, case reports and short case series (i.e., n < 10) were excluded. Among 3,374 screened references, we included 117 studies involving 6,797 patients distributed as follows: cholangiocarcinoma (58.1%), NETs (18.8%), HEHE (7.7%), CRLM (6.8%), mixed neoplasms (6.8%) or others (1.7%). Only 41% of the studies disclosed details of the immunosuppression protocol, and 20.8% of studies provided drug trough concentrations during follow-up. The immunosuppression protocols described were heterogeneous and broadly mirrored routine practices for non-tumoral indications. The only exception was CRLM where tacrolimus minimization -or even withdrawal- in combination with inhibitors of the mammalian target of rapamycin (mTORi) were consistently reported. None of the studies evaluated the relationship between the immunosuppression protocol and oncological outcomes. In conclusion, based on low-quality and indirect scientific evidence, patients with tumoral indications for LT should receive the lowest tacrolimus level tolerated under close surveillance. The combination with mTORi titrated to achieve the top therapeutic range of trough concentrations could allow complete tacrolimus withdrawal. This approach may be particularly useful in patients with cholangiocarcinoma and CRLM, in whom tumor recurrence is the main cause of death. We propose a tool for reporting immunosuppression protocols which could be implemented in future transplant oncology studies.
PMID:39347698 | DOI:10.1097/LVT.0000000000000499
Front Med (Lausanne). 2024 Sep 13;11:1439449. doi: 10.3389/fmed.2024.1439449. eCollection 2024.
ABSTRACT
OBJECTIVES: To investigate the safety and cost analysis of oral propranolol treatment for high-risk infantile hemangiomas starting from the outpatient setting.
METHODS: A total of 41 high-risk infantile hemangioma patients from outpatient settings and 43 from inpatient settings were selected for the study. After routine pre-treatment examinations, patients were administered propranolol in a stepwise incremental dosing regimen over three consecutive days in the outpatient clinic. Changes in heart rate, blood pressure and PR interval before and after medication were compared. On the 10th day post-medication, liver and kidney functions, fasting blood glucose, tumor ultrasonography, and electrocardiogram were re-evaluated. The costs of treatment starting from the outpatient clinic (including pre-treatment examinations and the first three days of treatment) were calculated and compared with those of similarly managed inpatient cases.
RESULTS: The majority of patients exhibited a reduction in heart rate and blood pressure, as well as an extended PR interval after treatment of medication (P < 0.05), which remained within normal limits without clinical symptoms. On the 10th day post-medication, statistical differences in blood biochemistry and electrocardiograms were observed when compared to pre-treatment values (P < 0.05), but all values remained within normal ranges. No severe adverse reactions such as hypoglycemia occurred. Additionally, the cost of treatment from the outpatient clinic was significantly lower than that of inpatient care.
CONCLUSION: Oral propranolol treatment for high-risk infantile hemangiomas starting from the outpatient setting is associated with few adverse reactions and significantly reduced treatment costs. It is worthy of broader application in hospitals without dermatology wards.
PMID:39346938 | PMC:PMC11437523 | DOI:10.3389/fmed.2024.1439449
J Craniofac Surg. 2024 Sep 30. doi: 10.1097/SCS.0000000000010708. Online ahead of print.
ABSTRACT
Multiple primary cavernous hemangiomas of the skull are exceedingly rare, with surgery often being the treatment of choice. The complexity of radiologic diagnosis means that the identification of these hemangiomas still largely depends on pathologic analysis. This article outlines the diagnostic and therapeutic journey of a 52-year-old female patient afflicted with multiple primary cavernous hemangiomas of the skull. Although the occurrence of multiple cavernous hemangiomas in this patient may seem fortuitous, the authors aim to contribute to understanding the pathogenesis of such conditions through this case report.
PMID:39345146 | DOI:10.1097/SCS.0000000000010708
West Afr J Med. 2024 Jun 28;41(6):714-720.
ABSTRACT
INTRODUCTION: Coronavirus disease (COVID-19) quickly became a pandemic causing a global concern affecting public health directly and indirectly. Dermatology outpatient services were not an exception to the impact of COVID-19, especially in Sub-Saharan Africa. To the best of our knowledge, no similar study has been done in this region, and identifying patterns of dermatological diseases would help prepare adequately for future crises.
OBJECTIVE: This study examined the changes in diagnostic patterns at the dermatology clinic before the COVID-19 pandemic and during the pandemic.
METHODS: This retrospective study was conducted on dermatology outpatients seen at Nnamdi Azikiwe University Teaching Hospital, Nnewi, Nigeria. Diagnoses were compared during the pandemic (March to August 2020) with the corresponding period of the previous year.
RESULTS: A total of 534 patients with 559 diagnoses were included. The average weekly dermatology clinic visits remained unchanged during the pandemic, although there was a 25.8% reduction in dermatology outpatients. The most common reasons for visiting the outpatient clinic during the COVID-19 pandemic were acne vulgaris, scabies, atopic dermatitis, dermatophytosis, and pityriasis versicolor. While the percentage of scabies significantly decreased during the pandemic; the percentage of allergic contact dermatitis, prurigo nodularis, candidiasis, hemangioma, and lupus erythematosus increased respectively.
CONCLUSION: Although the COVID-19 pandemic had a negligible impact on the average weekly number of skin clinic visits, it caused a change in diagnostic patterns. Understanding the pattern of skin diagnoses may help hospitals better prepare for future pandemics.
PMID:39342417
Jpn J Ophthalmol. 2024 Sep 28. doi: 10.1007/s10384-024-01102-1. Online ahead of print.
ABSTRACT
PURPOSE: To clarify the clinical features including multimodal imaging of primary choroidal lymphoma.
STUDY DESIGN: Retrospective, observational case series METHODS: Six consecutive patients with primary choroidal lymphoma diagnosed at Tokyo Medical University Hospital were studied. Demographic profile, clinical features and multimodal imaging were reviewed retrospectively.
RESULTS: The patients comprised 3 men and 3 women with average age of 68.2 ± 12.4 years. Clinical diagnosis before referring to our hospital included Vogt-Koyanagi-Harada disease, posterior scleritis, and choroidal hemangioma, and no patients had been diagnosed with choroidal lymphoma. Average duration from onset of ocular symptoms to establishing a diagnosis was 14.2 months. Ocular fundus examination revealed yellowish orange nummular infiltration in all six cases, and granular pigment deposits at the posterior pole in four cases. Optical coherence tomography showed increased choroidal thickness and waving of the retinal pigment epithelium in all cases. Ultrasonography depicted choroidal thickening and extraocular nodular lesions adjacent to the posterior sclera in all cases. None of the cases showed elevated interleukin-10 in the aqueous humor. Ocular fundus manifestations and visual acuity improved after external beam radiation therapy, except one case with bullous retinal detachment from the initial visit.
CONCLUSIONS: Characteristic ocular fundus manifestation together with multimodal imaging findings are useful for the clinical diagnosis of primary choroidal lymphoma.
PMID:39340726 | DOI:10.1007/s10384-024-01102-1
Int J Mol Sci. 2024 Sep 10;25(18):9760. doi: 10.3390/ijms25189760.
ABSTRACT
Infantile hemangiomas (IHs) are benign vascular neoplasms of childhood (prevalence 5-10%) due to the abnormal proliferation of endothelial cells. IHs are characterized by a peculiar natural life cycle enclosing three phases: proliferative (≤12 months), involuting (≥13 months), and involuted (up to 4-7 years). The mechanisms underlying this neoplastic disease still remain uncovered. Twenty-seven IH tissue specimens (15 proliferative and 12 involuting) were subjected to hematoxylin and eosin staining and a panel of diagnostic markers by immunohistochemistry. WT1, nestin, CD133, and CD26 were also analyzed. Moreover, CD31pos/CD26pos proliferative hemangioma-derived endothelial cells (Hem-ECs) were freshly isolated, exposed to vildagliptin (a DPP-IV/CD26 inhibitor), and tested for cell survival and proliferation by MTT assay, FACS analysis, and Western blot assay. All IHs displayed positive CD31, GLUT1, WT1, and nestin immunostaining but were negative for D2-40. Increased endothelial cell proliferation in IH samples was documented by ki67 labeling. All endothelia of proliferative IHs were positive for CD26 (100%), while only 10 expressed CD133 (66.6%). Surprisingly, seven involuting IH samples (58.3%) exhibited coexisting proliferative and involuting aspects in the same hemangiomatous lesion. Importantly, proliferative areas were characterized by CD26 immunolabeling, at variance from involuting sites that were always CD26 negative. Finally, in vitro DPP-IV pharmacological inhibition by vildagliptin significantly reduced Hem-ECs proliferation through the modulation of ki67 and induced cell cycle arrest associated with the upregulation of p21 protein expression. Taken together, our findings suggest that CD26 might represent a reliable biomarker to detect proliferative sites and unveil non-regressive IHs after a 12-month life cycle.
PMID:39337249 | PMC:PMC11432178 | DOI:10.3390/ijms25189760
J Clin Med. 2024 Sep 20;13(18):5586. doi: 10.3390/jcm13185586.
ABSTRACT
Background: Bone hemangiomas are rare benign vascular tumors, comprising less than 1% of all bone tumors. They are predominantly found in the vertebral body or skull; rib hemangiomas are particularly rare and are often misdiagnosed as malignant tumors. Given the high malignancy rate of primary rib tumors, understanding rib hemangiomas is crucial to avoid misdiagnosis. Methods: A systematic review was conducted according to PRISMA standards. A comprehensive literature search was performed in PubMed, EMBASE, Web of Science, and Scopus. Data on patient demographics, tumor characteristics, and clinical presentation were analyzed using STATA/SE 17. Results: From 306 articles, 40 studies including 43 patients met the inclusion criteria. Rib hemangiomas showed a bimodal age distribution, with peaks in patients younger than 30 years (mean age 21.43 ± 5.60 years) and ≥30 years (mean age 59.96 ± 9.70 years). Females were more affected (62.79%) than males (37.21%), with a ratio of 1.69:1. The tumors were most frequently located in mid-thoracic ribs (4-8) and predominantly on the left side of the thorax. The mean tumor size was 7.27 cm, with 76.19% exhibiting osteolytic changes. Clinically, 63.41% of cases were asymptomatic, while symptomatic cases mainly presented with pain. Conclusions: Rib hemangiomas, though rare, should be considered in the differential diagnosis of thoracic tumors. They present as well-demarcated lytic lesions with distinct imaging features, and they typically require complete surgical excision, which may be aided with preoperative embolization. Their accurate diagnosis involves a combination of radiologic and clinical evaluation. Further studies are needed to understand the disease's pathophysiology and to refine diagnostic and treatment protocols.
PMID:39337072 | PMC:PMC11433624 | DOI:10.3390/jcm13185586
Genes (Basel). 2024 Sep 11;15(9):1192. doi: 10.3390/genes15091192.
ABSTRACT
Purpose: To delineate the genotype and phenotype of RH in a Chinese cohort. Methods: A group of 51 Chinese probands with RH across 76 eyes was assembled and underwent complete retinal imaging examinations. Sanger sequencing and universal primer quantitative fluorescent multiplex-polymerase chain reaction (UPQFM-PCR) were employed for mutation detection in the coding region of the Von Hippel-Lindal (VHL) gene. For frequency calculation, our series was combined with three large cohorts of East Asian descent through a literature review. Results: The Von Hippel-Lindal (VHL) syndrome was excluded in fifteen patients (median age: 32.00 years) with unilateral solitary RH. Thirty-six patients of younger ages (median: 22.00 years, p = 0.008, Mann-Whitney test) conformed to the diagnostic criteria of the VHL syndrome, and thirty-four patients were genetically confirmed. There were four novel variants identified in the VHL gene. Codons 167, 161 and 86 exhibited a mutation occurrence of more than 5% after pooling with literature data, and the large genomic deletion demonstrated a frequency of 17.65%. The RHs were classified as "extrapapillary", "juxtapapillary" and "mixed" types in 53, 7 and 5 eyes, respectively. Almost all extrapapillary RH lesions were found in the peripheral retina. Hemangioblastomas in the central nervous system (CNS) were observed in 25 out of 31 kindreds (80.65%) with full systemic evaluation data. Conclusions: VHL-associated RH might exhibit earlier onset than non-VHL RH. Large genomic deletions were observed at a notably high frequency in the Chinese series with VHL-associated RH, which might be associated with East Asian ethnicity background. RH could potentially serve as an early indicator of CNS hemangioblastoma.
PMID:39336783 | PMC:PMC11431690 | DOI:10.3390/genes15091192
Children (Basel). 2024 Sep 4;11(9):1086. doi: 10.3390/children11091086.
ABSTRACT
BACKGROUND: Infantile haemangiomas (IHs) sometimes require treatment with propranolol. Sleep disturbances are the most frequently reported side effects. Monitoring adverse drug events necessitates repeated hospital visits, which can be challenging during a pandemic.
OBJECTIVES: To explore the effectiveness of a new electronic questionnaire in identifying sleep disturbances related to treatment with propranolol and potential confounding factors. To evaluate the response rate to the questionnaire. To report the proportion of patients on propranolol with sleep disturbances.
METHODS: In an observational, prospective cohort study, caregivers provided clinical information during ambulatory visits and via an electronic questionnaire after an 8-week treatment course with propranolol and at the time of treatment interruption. Adverse drug reaction reporting forms were assessed for causality.
RESULTS: The questionnaire response rate was 91%, and the completion rate was 100%. A total of 59% of patients experienced sleep disturbances during propranolol treatment, which were considered adverse reactions. Sleep disorders were frequent during sleep regression phases and in subjects who fell asleep during physical contact with caregivers or bed-sharing with parents.
CONCLUSION: The application of this questionnaire allows for identifying adverse sleep events associated with propranolol in IHs and potential confounders. Counselling on sleep hygiene is recommended before treatment onset.
PMID:39334619 | PMC:PMC11430174 | DOI:10.3390/children11091086
Medicine (Baltimore). 2024 Sep 27;103(39):e39800. doi: 10.1097/MD.0000000000039800.
ABSTRACT
RATIONALE: Hepatic angiosarcoma (HSA) has a poor prognosis. Our understanding of its clinical features, diagnosis, treatment, and prognosis remains limited. In certain cases, vascular tumors such as HSA can induce bleeding complications due to thrombocytopenia, known as Kasabach-Merritt syndrome (KMS). When KMS symptoms occur in the lungs, its clinical manifestations closely resemble those of ARDS, leading to misdiagnosis and poor outcomes. Unfortunately, this condition is extremely rare and there is a lack of relevant case reports, which further adds to the difficulty of its diagnosis and treatment.
PATIENT CONCERNS: This case report describes a patient who initially presented with symptoms of ARDS. Due to the unique nature of these symptoms, the patient underwent a complex diagnostic and treatment process before finally being diagnosed with HSA complicated by KMS through pathological examination.
DIAGNOSES: The patient was eventually diagnosed with HSA by pathology and KMS with multiorgan hemorrhage.
INTERVENTIONS: Highly misleading clinical manifestations were recorded during the diagnosis and treatment, which, to our knowledge, have not been previously reported.
OUTCOMES: The patient died from a massive pulmonary hemorrhage.
LESSONS: Dysfunction of a single organ or system may be the external manifestation of a multi-system clinical disease. Therefore, in the clinical diagnosis and treatment process, especially during early diagnosis, while it is important to focus on the primary or typical clinical symptoms, it is equally crucial not to underestimate or ignore accompanying symptoms that lack specificity. When diagnosis and treatment reach an impasse, these "atypical" symptoms often prove to be key in solving the puzzle.
PMID:39331928 | PMC:PMC11441907 | DOI:10.1097/MD.0000000000039800
Neurosurg Rev. 2024 Sep 27;47(1):695. doi: 10.1007/s10143-024-02919-7.
ABSTRACT
The study titled "Long-term outcome and quality of life after CNS cavernoma resection: eloquent vs. non-eloquent areas," by Shoubash et al. (2022) provides crucial insights into the long-term neurological outcomes and quality of life (QoL) in patients following CNS cavernoma resection. Differentiating between eloquent and non-eloquent areas, the study shows that patients generally experience non-inferior QoL, with some differences in physical role functioning. Utilizing the Short Form-12 (SF12) questionnaire at a mean follow-up of 6.5 years, the study's findings are significant for clinical decision-making and patient counseling. However, the study's small sample size and retrospective design limit its generalizability and introduce potential biases. The lack of preoperative QoL assessments further constrains its conclusions. Future research should focus on larger, prospective studies with comprehensive QoL metrics and longitudinal follow-up to better understand the impact of surgery on patient outcomes and improve clinical strategies.
PMID:39327346 | DOI:10.1007/s10143-024-02919-7
Cardiovasc Intervent Radiol. 2024 Oct 15. doi: 10.1007/s00270-024-03876-w. Online ahead of print.
ABSTRACT
PURPOSE: To evaluate the safety, efficacy, and long-term outcomes of transarterial chemoembolization (TACE) with bleomycin-Lipiodol for giant liver hemangiomas.
MATERIALS AND METHODS: Single-center retrospective study from 1998 to January 2020, including patients with giant liver hemangiomas treated with bleomycin-Lipiodol TACE and followed up >36 months. The exclusion criteria were defined as patients who had been treated but had no available follow-up above 3 years and patients who had previously been treated with any other treatment method. Clinical success was defined as the disappearance of symptoms and radiological success (responded vs. non-responded groups) as a more than 50% decrease in the volume of the giant hemangioma in follow-up CT or MRI compared to the baseline images.
RESULTS: A total of 121 patients were included. The mean maximum diameter of the hemangiomas decreased from 122 (range: 40-300) to 73 mm (range: 15-240), and the mean volume reduced from 984.4 (range: 30-7312) to 286.6 cm3 (range: 1-3835). There were 106 patients in the responded group, while only 15 patients were in the non-responded group. No significant difference was found in size and volume change percentages across these two groups based on gender, age, lesion size, lesion volume, lesion number, and second TACE. When the follow-up period was stratified in 5-year periods, the maximum volume decrease was observed in the first 5-year period and then remained constant up to > 15 years.
CONCLUSION: TACE with bleomycin-Lipiodol is safe, reducing the size and volume of giant liver hemangiomas with stable results in the long-term follow-up.
PMID:39406873 | DOI:10.1007/s00270-024-03876-w
Ultraschall Med. 2024 Oct 15. doi: 10.1055/a-2421-6047. Online ahead of print.
ABSTRACT
Kaposiform hemangioendothelioma (KHE) is a rare, locally aggressive vascular tumor with high morbidity and mortality. The aim of this study was to evaluate ultrasonographic findings associated with KHE.The clinical and ultrasonographic findings of a cohort of 64 cases with pathologically proven KHE were retrospectively reviewed and analyzed between November 2014 and February 2021. Two subtypes were divided according to the presence or absence of the Kasabach-Merritt phenomenon (KMP). The KMP risk factors in patients with KHE were analyzed statistically.Among the 64 cases of KHE, 43 (67.2%) were accompanied by KMP. There was a positive correlation between the appearance of KMP and tumor size. KHEs had an increased risk of developing KMP if the lesions measured were >6 cm and if they belonged to the deep or mixed subtype. On ultrasonography, all KHE lesions were heterogeneous, and 81.3% were hypoechoic; 93.8% of KHEs exhibited ill-defined margins, 68.7% had strands branching into the adjacent tissue, and 84.4% presented marked hypervascularity. Elastography showed that central hypoechogenic lesion areas were hard, and surrounding hyperechogenic lesion areas were soft.KHEs can occur in different parts of childrens' bodies. On ultrasonography, the main findings are heterogeneous low erosions, indistinct margins, branching strangulation into adjacent tissues, and obvious hypervascularity. Patients with lesions larger than 6 cm or belonging to deep or mixed subtypes (musculoskeletal infiltrates) are at risk for developing KMP, and clinicians should be vigilant.
PMID:39406368 | DOI:10.1055/a-2421-6047
Clin Cancer Res. 2024 Oct 15. doi: 10.1158/1078-0432.CCR-24-2519. Online ahead of print.
ABSTRACT
PURPOSE: Survival of patients with metastatic sarcoma remains poor, and there is pressing need for new therapies. Most sarcoma subtypes are not responsive to immune checkpoint inhibition alone. Lenvatinib, a multi-receptor tyrosine kinase inhibitor targeting tumor vasculature, has immunomodulatory activity that contributes to its antitumor effects. Therefore we hypothesized that combination of lenvatinib and pembrolizumab would lead to improved clinical outcomes in patients with sarcoma.
METHODS: This was an open-label, single-arm study of lenvatinib and pembrolizumab in the following cohorts A: leiomyosarcoma, B: undifferentiated pleomorphic sarcoma (UPS), C: vascular sarcomas (angiosarcoma and epithelioid hemangioendothelioma), D: synovial sarcoma or malignant peripheral nerve sheath tumor (MPNST), and E: bone sarcomas (osteosarcoma and chondrosarcoma). The primary endpoint was best overall response (BOR) rate documented by RECIST v1.1 by 27 weeks in each cohort, with a threshold of ≥2 responses among 10 patients. Secondary endpoints included progression-free survival, overall survival, duration of response and safety.
RESULTS: Forty-six patients were evaluable for the primary endpoint which was met in the UPS and MPNST/synovial cohorts (BOR rates by 27 weeks of 25% and 30%,respectively). There were 7 partial responses overall with additional responses noted in angiosarcoma and osteosarcoma. Treatment-related adverse events of any grade, and Grade 3 or higher, occurred in 50/51 (98%) and 29/51 (57%) of patients respectively.
CONCLUSIONS: We observed durable responses in MPNST, synovial sarcoma and osteosarcoma. Patients with UPS and angiosarcoma also responded. Further exploration of this approach is warranted to confirm activity and determine optimal dosing schedules.
PMID:39405335 | DOI:10.1158/1078-0432.CCR-24-2519
J Craniofac Surg. 2024 Oct 15. doi: 10.1097/SCS.0000000000010671. Online ahead of print.
ABSTRACT
BACKGROUND: Treating extensive benign tumors of the head and face presents a longstanding challenge, necessitating efficacy at the lesion site, and postoperative esthetic considerations for the donor area.
OBJECTIVE: This study aims to explore the clinical outcomes of pre-expanded local flap reconstruction for extensive benign tumors of the head and face post resection.
METHOD: From March 2018 to March 2023, a total of 18 patients with extensive benign tumors of the head and face were admitted, including 13 cases of nevus, 2 cases of hemangioma, 2 cases of neurofibroma, and 1 case of verruca. Based on the location and size of the lesions, suitable local areas were selected for tissue expansion, and expanders were implanted for regular saline injections over 8 to 16 weeks. After reaching the desired expansion, resection of the head and facial tumors and local flap reconstruction were performed. Postoperatively, data on patients' information, tumor types, tumor area, expansion volume, postexpander complications, vascular condition after flap transfer, and donor site condition were collected.
RESULT: In this series of 18 patients, benign tumors of the head and face were completely repaired through 1 to 2 stages of tissue expansion surgeries. Postimplantation complications included hematoma in 1 case and infections in 2 cases, with one instance of expander infection leading to surgical failure. However, all other patients achieved adequate expansion, successful flap survival post-transfer, and experienced no other complications. Follow-up over 6 to 24 months showed no recurrence at the lesion sites, with flaps maintaining consistent color, texture, and thickness matching surrounding skin tissue. In addition, donor site healing was excellent, with no obvious surgical scars.
CONCLUSIONS: Pre-expanded local flap reconstruction is an ideal method for repairing extensive benign tumors of the head and face postresection.
PMID:39404590 | DOI:10.1097/SCS.0000000000010671
Heliyon. 2024 Sep 28;10(19):e38704. doi: 10.1016/j.heliyon.2024.e38704. eCollection 2024 Oct 15.
ABSTRACT
BACKGROUND: Epithelioid hemangioendothelioma (EHE) is an extremely rare vascular tumor that accounts for less than 1 % of all vascular tumors. The diagnosis of primary hepatic epithelioid hemangioendothelioma (HEHE) can be challenging because it may be mistaken for other liver lesions. This has led to misdiagnosis in many cases. This study aimed to present a rare case of HEHE that was diagnosed in a female patient with familial Mediterranean fever (FMF) and hypothyroidism who received pazopanib treatment and demonstrated a long-term response to it.
CASE PRESENTATION: A 43-year-old Syrian woman with FMF who was treated with colchicine for 5 years and hypothyroidism treated with thyroxin was diagnosed with HEHE. She was treated with a tyrosine kinase inhibitor (pazopanib) for over two years and showed a durable response. Although the patient stopped taking colchicine, the episodes of abdominal pain improved after the initiation of pazopanib administration. Pazopanib was well tolerated, and the patient is still alive and doing well.
CONCLUSIONS: This study highlights a case of chronic abdominal pain in a patient with multiple comorbidities that resulted in the diagnosis of a rare case of HEHE and prolonged response to pazopanib. In addition, we discuss the effects of pazopanib on FMF symptoms.
PMID:39403458 | PMC:PMC11471504 | DOI:10.1016/j.heliyon.2024.e38704
J Clin Exp Dent. 2024 Sep 1;16(9):e1151-e1156. doi: 10.4317/jced.61925. eCollection 2024 Sep.
ABSTRACT
Epithelioid hemangioendothelioma (EHE) is a rare neoplasm derived from the vascular endothelium. Although it can occur anywhere in the body, few cases have been described in the oral cavity. We report a 47-year-old woman presenting with a painful ulcerated lesion on the mandibular gingiva, suggestive of a traumatic decubitus ulcer. Histology and immunohistochemistry confirmed the diagnosis of epithelioid hemangioendothelioma. A literature review of EHE of mandibular gingiva was done. Pubmed were searched from 1975 through June 2024 using the following search terms: epithelioid hemangioentothelioma, vascular tumor, oral cavity and mandibular gingiva. Relevant manuscripts were selected and the results were used to update a narrative overview of the diagnosis and management of this entity. We found 38 cases of EHE in the oral cavity, of which 16 were located on the gingiva. Most of them were located on the mandibular gingiva as painless swelling, unlike our case. 70 % of the cases presenting in the mandibular gingiva had bone resorption on imaging. However, only half of those located in the maxillary gingiva had this bone resorption. Only 2 cases located in the mandibular gingiva presented recurrence and 1 of them debuted with nodal metastases after a 7-year follow-up. The clinical and histological diagnosis of EHE is complex and must be confirmed by immunohistochemistry. Upon diagnosing this entity, we should perform an excision with clear margins and conduct long-term follow-up due to the risk of local and distant recurrence. Key words:Epithelioid hemangioendothelioma, Gingival pathologies, Oral cavity, Mandibular Diseases, CD31, Immmunohistochemical markers, Vascular tumor.
PMID:39399857 | PMC:PMC11470459 | DOI:10.4317/jced.61925
Spine Surg Relat Res. 2024 May 10;8(5):548-551. doi: 10.22603/ssrr.2024-0023. eCollection 2024 Sep 27.
NO ABSTRACT
PMID:39399455 | PMC:PMC11464831 | DOI:10.22603/ssrr.2024-0023
Ultrastruct Pathol. 2024 Oct 13:1-12. doi: 10.1080/01913123.2024.2415608. Online ahead of print.
ABSTRACT
Telocytes (TCs) are a distinctive cell entity of the stromal microenvironment of multiple tumors; to date, their existence in infantile hemangioma (IH) remains almost unexplored. This study was therefore undertaken to characterize the immunophenotype, location, morphology, and ultrastructure of telocytes in the IH by means of immunohistochemistry, immunofluorescence confocal microscopy, and transmission electron microscopy. Telocytes were initially identified by CD34, PDGFR-α, Vimentin, and AQP-1 immunostaining. Analyzing the spatial relationship among telocytes, stem cells, endothelial cells, pericytes in the IH with AQP-1/CD31, AQP-1/Glut-1, AQP-1/α-SMA, AQP-1/CD146 and AQP-1/CD133 double immunofluorescence. TCs were immunonegative for CD31, Glut-1, CD146, α-SMA, CD133, and C-kit in the IH. The ultrastructural examination confirmed the presence of TCs, namely stromal cells with characteristic cytoplasmic processes (i.e. telopodes) forming labyrinthine networks around microvessels and releasing extracellular vesicles. Our study provides evidence that telocytes are present and PDGFR-α and AQP-1 are specific antigenic markers in the IH.
PMID:39397344 | DOI:10.1080/01913123.2024.2415608
J Vasc Surg Venous Lymphat Disord. 2024 Oct 10:101989. doi: 10.1016/j.jvsv.2024.101989. Online ahead of print.
ABSTRACT
BACKGROUND: Intravenous leiomyomatosis (IVL) is a rare neoplasm, the accumulated knowledge about the characteristics and prognosis of this tumor has been derived mainly from isolated case reports with no comprehensive research. In this study, we reviewed our institution's experience with IVL over a 20-year period and developed a classification system that can be used to guide surgical management.
MATERIALS AND METHODS: The study had a retrospective cohort design and included patients who underwent resection of IVL at our institution between January 2002 and December 2022. Perioperative parameters were then collected among 4 stages of our proposed classiï¬cation. The long-term outcomes, oncologic prognosis, and factors associated with recurrence were analyzed.
RESULTS: A total of 216 patients were included (stage 1, n=92; stage 2, n=39; stage 3, n=76; stage 4, n=9). The mean follow-up duration was 26.34 months, during which 18 patients (9.7%) in the complete resection group had recurrence, and 12 (39.0%) in the incomplete resection group showed disease progression. Recurrence or progression of residual disease was associated with adjuvant aromatase inhibitor therapy and maximum tumor thrombus diameter but not with total hysterectomy and bilateral salpingo-oophorectomy, age, or postoperative treatment with a gonadotropin-releasing hormone agonist therapy.
CONCLUSIONS: This is the largest single-center report on IVL published to date and provides valuable information on its clinical features and long-term outcomes as well as surgical technique. Our classiï¬cation system can be used to evaluate the extent of lesion involvement and guide surgical management.
PMID:39395469 | DOI:10.1016/j.jvsv.2024.101989
Egypt Heart J. 2024 Oct 10;76(1):139. doi: 10.1186/s43044-024-00571-6.
ABSTRACT
BACKGROUND: Cardiac myxomas are the most common primary benign tumors of the heart usually presenting as an oval mass attached to the interatrial septum. Mild heterogenous enhancement is typically seen upon contrast administration. Myxomas are benign, however can present with embolic episodes necessitating prompt diagnosis and treatment.
CASE PRESENTATION: A middle-aged woman presenting with acute ischemic stroke was detected to have a complex cystic mass in the left atrium on echocardiography. Further evaluation with cardiac magnetic resonance imaging narrowed a differential diagnosis to hemangioma, hemangioendothelioma, sarcoma, hydatid cysts, bronchogenic cysts, and cystic variant of cardiac myxoma. CTA demonstrated arterial supply from the left circumflex artery supplying the lesion. Following surgical excision, histopathology analysis suggested a cystic cardiac myxoma with secondary degeneration.
CONCLUSION: Cystic masses in the heart are challenging to diagnose due to similar imaging characteristics, and misdiagnosis may lead to further major downstream complications such as stroke, hemorrhage, and hemodynamic alterations causing syncope. Therefore, surgical excision of a cystic cardiac mass is justified to establish a definitive histopathological diagnosis and prevent further downstream complications. We hereby report a rare case of cystic cardiac myxoma in the left atrium showing intense progressive enhancement in CMR and CTA in a middle-aged woman presenting with acute stroke. This case highlights an exceedingly rare variant of cystic cardiac myxoma.
PMID:39390292 | PMC:PMC11467151 | DOI:10.1186/s43044-024-00571-6
J Pediatr Surg. 2024 Sep 28:161932. doi: 10.1016/j.jpedsurg.2024.161932. Online ahead of print.
ABSTRACT
BACKGROUND: This study generalized and analyzed the clinical attributes observed in patients afflicted with Kaposiform hemangioendothelioma (KHE) with the aim of elucidating the risk factors contributing to the manifestation of Kasabach-Merritt phenomenon (KMP).
METHODS: We retrospectively analyzed 96 pediatric cases diagnosed with KHE at the Children's Hospital of Fudan University from January 2013 to December 2021. Among them, 62 patients (65%) showed KMP (KHE + KMP group), while 34 patients (35%) did not (KHE-KMP group). The risk factors for KMP associated with KHE were analyzed using univariate analysis and binary logistic regression analysis, comparing the differences between KHE + KMP group and KHE-KMP group.
RESULTS: Univariate analysis indicated no statistically significant differences between the two groups in gender, prematurity, family history, or color of involved skin. However, statistically significant differences were observed in age of onset, lesion site, and lesion depth. Multivariate analysis revealed significant associations: children with onset age ≤1 month had a 51-fold increased risk of KMP compared to those with onset age >1 month (95% CI 5.238-501.663); non-extremity lesion sites exhibited a 21-fold higher risk of KMP compared to extremity sites (95% CI 3.970-105.958); deeper lesions conferred a 5-fold higher risk of KMP compared to superficial lesions (95% CI 1.073-21.005); lesions >60 mm carried a 17-fold higher risk of KMP compared to lesions ≤60 mm (95% CI 2.999-96.157). A comprehensive predictive model was developed using the fitting formula: Logit (P) = 3.937∗(age at onset) + 1.558∗(lesion depth) + 3.021∗(lesion site) + 2.832∗(lesion size), demonstrating an accuracy of 82.9%. Furthermore, a scoring system was established to assess the likelihood of KMP occurrence. Children diagnosed with KHE were likely to have KMP if their score exceeded 72.5, as determined by Receiver Operating Characteristic (ROC) curve analysis.
CONCLUSION: Age of onset ≤1 month, deeper lesions, non-extremity sites, and lesions >60 mm are independent risk factors for KMP in children with KHE. The cumulative presence of these factors escalates the likelihood of KMP development. Additionally, the identification of these factors allows for the early recognition of potential KMP cases among children with KHE, facilitating prompt therapeutic intervention.
CATEGORY OF THE MANUSCRIPT: Clinical Research article.
LEVEL OF EVIDENCE: LEVEL â…¢.
PMID:39389877 | DOI:10.1016/j.jpedsurg.2024.161932
Semin Pediatr Neurol. 2024 Oct;51:101152. doi: 10.1016/j.spen.2024.101152. Epub 2024 Sep 11.
ABSTRACT
PHACE syndrome is a neurocutaneous disorder consisting of posterior fossa brain abnormalities, facial infantile hemangioma, arterial anomalies, cardiac anomalies (most commonly coarctation of the aorta), and eye anomalies. While the cutaneous finding of infantile hemangioma represents the most clinically apparent feature, extracutaneous findings-specifically, developmental abnormalities of the aorta and medium-sized thoracic, cervical, and cerebral arteries-are common and pose significant potential morbidity and mortality. Cerebral arteriopathy can be progressive and lead to arterial ischemic stroke in childhood and increased stroke risk in adulthood; headache and neurodevelopmental symptoms are also common. Thus, it is important for pediatric neurologists to be familiar with this disorder and its potential structural and functional neurological sequelae. This review article summarizes the clinical features, diagnostic considerations, epidemiology, and management of this condition with an emphasis on features most pertinent to the practicing pediatric neurologist.
PMID:39389654 | DOI:10.1016/j.spen.2024.101152
Semin Pediatr Neurol. 2024 Oct;51:101151. doi: 10.1016/j.spen.2024.101151. Epub 2024 Sep 7.
ABSTRACT
First described in the late 1800's, Sturge-Weber syndrome is one of the more common neurocutaneous disorders. In most cases, it is caused by a somatic mosaic variant in the GNAQ gene driving aberrant overgrowth in endothelial cells which leads to capillary-venous malformations. Characteristic findings are unilateral facial port-wine stain, ipsilateral parieto-occipital leptomeningeal angioma with calcifications and atrophy, and ipsilateral glaucoma, though there is significant variability. The predilection for facial skin and brain is likely due to common embryologic progenitors. The risk of brain involvement is increased with a hemifacial, forehead, or medial facial port-wine stain. Neurologic features include epilepsy, stroke-like episodes, transient or permanent hemiparesis and visual field deficit, headaches, and cognitive and behavioral impairment. Magnetic resonance imaging reveals contrast-enhancing leptomeningeal angiomatosis, progressive atrophy, calcifications, and ipsilateral dilated choroid plexus. The treatment of glaucoma typically requires surgery and port-wine stains are treated with laser therapy. Retrospective data from small cohorts show potential benefits of presymptomatic treatment with anti-seizure medications and/or low dose aspirin. Epilepsy surgery can benefit those with a greater degree of hemiparesis and intractable seizures. Low-dose aspirin has proven effective in lowering the frequency and severity of recoverable stroke-like events. Sirolimus has been reported preliminarily to have satisfactory results regarding cognitive function in pediatric patients, but is not a mainstay of treatment to date. Quality of life is often negatively affected by port-wine stain appearance, intractable seizures, headaches, and mood disorders. Future studies are warranted assessing medication and surgery outcomes, quality of life measures, and timing of imaging and treatment initiation.
PMID:39389653 | DOI:10.1016/j.spen.2024.101151
Clin Nucl Med. 2024 Oct 10. doi: 10.1097/RLU.0000000000005484. Online ahead of print.
ABSTRACT
68Ga-pentixafor uptake in hemangioma is rare. We report 68Ga-pentixafor PET/MRI findings of a hepatic hemangioma in a 54-year-old woman. The images revealed a hyperintense lesion in the right posterior liver on T2-weighted images, with mild pentixafor uptake. The lesion was consistent with typical presentations of a hemangioma on MRI and contrast-enhanced CT.
PMID:39385358 | DOI:10.1097/RLU.0000000000005484
J Orthop Case Rep. 2024 Oct;14(10):184-187. doi: 10.13107/jocr.2024.v14.i10.4856.
ABSTRACT
INTRODUCTION: Synovial hemangioma of the knee is a benign vascular malformation that is often misdiagnosed due to its non-specific symptoms.
CASE REPORT: We present a case involving a 7-year-old male child with chronic knee pain and swelling in the left knee, which remained undiagnosed for over 3 years. Magnetic resonance imaging revealed a synovial vascular malformation. Surgical excision of the lesion and partial synovectomy were performed, and histopathological analysis confirmed the diagnosis of synovial hemangioma. After 18 months of follow-up, there were no signs of recurrence or recurrent joint effusion.
CONCLUSION: Synovial hemangioma of the knee, although uncommon, should be considered as a differential diagnosis in cases of chronic knee pain and swelling. En-bloc excision is the treatment of choice to prevent recurrence, yielding good clinical outcomes.
PMID:39381297 | PMC:PMC11458251 | DOI:10.13107/jocr.2024.v14.i10.4856
Pediatr Dermatol. 2024 Oct 8. doi: 10.1111/pde.15753. Online ahead of print.
ABSTRACT
Propranolol has been the primary treatment for infantile hemangioma (IH) since 2008. Prior studies have investigated the effects in late childhood of propranolol therapy given in infancy for IH, including neurocognitive dysfunction, sleep disorders, and hypoglycemia. However, few studies have determined the risk of these adverse effects later in life. Using the TrinetX database, we studied the risk of growth impairment, sleep disorders, learning disabilities, and diabetes mellitus in children aged 10-17 years who had received propranolol for IH in infancy. The maximum age of 17 years was chosen for the study, as propranolol was established as a treatment for IH in 2008. The results showed no statistically significant risk of growth impairment, sleep disorders, learning disabilities, or diabetes mellitus in IH patients treated with propranolol. These findings support existing evidence that propranolol therapy given in infancy for IH is not associated with long-term adverse effects up to age 17 years in the studied patient population.
PMID:39379322 | DOI:10.1111/pde.15753
Anatol J Cardiol. 2024 Oct 8. doi: 10.14744/AnatolJCardiol.2024.4860. Online ahead of print.
NO ABSTRACT
PMID:39378325 | DOI:10.14744/AnatolJCardiol.2024.4860
Pathologica. 2024 Aug;116(4):258-266. doi: 10.32074/1591-951X-1003.
ABSTRACT
Chorioangiomas are benign angiomas arising from chorionic tissue and they are the most common non-trophoblastic tumors of the placenta, as they are observed in 1% of all placentas examined. Most chorioangiomas are small and asymptomatic, often undetected during a prenatal ultrasound, and their clinical significance is still unknown. Large chorioangiomas, measuring more than 4-5 cm in diameter, can usually be detected prenatally by gray-scale or color Doppler sonography, and may be associated with maternal or fetal complications, such as preeclampsia, maternal mirror syndrome, preterm delivery, nonimmune fetal hydrops, fetal growth restriction and fetal demise. We herein describe the clinical-pathological features of a monocentric series of 30 placental chorioangiomas and discuss their clinical-pathological features and possible molecular mechanisms underlying their development.
PMID:39377509 | PMC:PMC11460147 | DOI:10.32074/1591-951X-1003
Cureus. 2024 Sep 6;16(9):e68814. doi: 10.7759/cureus.68814. eCollection 2024 Sep.
ABSTRACT
A hepatic hemangioma is a benign liver tumor made up of a number of blood-filled chambers surrounded by liver-supplied endothelial cells. Most liver hemangiomas are asymptomatic and are only discovered during imaging studies for other conditions. Ultrasound is used for initial screening followed by a computed tomography scan, which shows slow enhancement due to small vessels and can be used to diagnose the location, number, and size of a hepatic hemangioma. A large liver hemangioma can range in size from 10 centimeters to more than 20 centimeters and can cause symptoms and complications that require prompt intervention. Hepatic hemangiomas can co-occur with other localized hepatic lesions; a careful diagnosis is necessary to distinguish them. In this case study, a 48-year-old woman complained of a stomachache that had persisted for three months. Following an initial clinical evaluation, hepatomegaly was found, and contrast-enhanced computed tomography (CECT) abdomen and pelvis was performed, revealing numerous giant hepatic hemangiomas. Significant improvements were noted in the patient's condition with tumor embolization.
PMID:39376836 | PMC:PMC11458059 | DOI:10.7759/cureus.68814
J Maxillofac Oral Surg. 2024 Oct;23(5):1282-1288. doi: 10.1007/s12663-024-02138-z. Epub 2024 Jun 1.
ABSTRACT
BACKGROUND: Cryotherapy was reported in the treatment of infantile hemangiomas by many studies using liquid nitrogen as a cryogen.
OBJECTIVE: To evaluate the outcome of cryo-carboxy surgery in treatment of infantile lip hemangiomas.
METHODS: In this study, we present the use of carbon dioxide as the cryogen in 50 patients with infantile hemangiomas of the lips with successful results. The patient evaluation was done including the improvement in color, texture, and volume of the lesion and each of the three parameters was given a score on a 4-point scale; excellent, good, fair, or poor.
RESULTS: The average evaluation score of our patients was excellent in 37 (74%) cases and good in 13 (26%) cases. We had no fair or poor results. There was no postprocedural hypopigmentation in any of the patients with lesions limited to the mucosa and Vermillion. In contrast, all cases with lesions extending to the lip skin (10 cases) experienced hypopigmentation which resolved spontaneously within 3 months. No complications were observed in our cases during the follow-up period and no cases, during the follow-up period, showed regrowth of the treated lesions.
CONCLUSION: We can conclude that, cryo-carboxy surgery is an effective safe new tool in the armamentarium of the treatment of infantile hemangiomas of the lips with successful results.
PMID:39376749 | PMC:PMC11456146 | DOI:10.1007/s12663-024-02138-z
J Surg Case Rep. 2024 Oct 5;2024(10):rjae624. doi: 10.1093/jscr/rjae624. eCollection 2024 Oct.
ABSTRACT
Anastomosing hemangioma (AH) is an exceedingly rare benign vascular tumor, often mistaken for malignant neoplasms due to its histological features. First described in 2009, AH has been documented in various sites, including the kidney, liver, and adrenal gland. This report presents a 64-year-old man who, following a bicycle accident, underwent imaging that revealed a nodule suspicious for a paraganglioma in the right para-caval region. Despite the high radiotracer uptake on 68Ga-DOTANOC PET scan, which suggested paraganglioma, the patient remained asymptomatic and laboratory tests ruled out excessive catecholamine secretion. Surgical excision led to a diagnosis of AH, distinguished by its unique anastomosing vascular pattern and histological similarity to angiosarcoma. This case underlines the diagnostic challenges and potential for overtreatment of AH, highlighting the need for heightened awareness and careful histopathological and imaging evaluation to avoid misdiagnosis and ensure appropriate management.
PMID:39376719 | PMC:PMC11456863 | DOI:10.1093/jscr/rjae624
Indian J Otolaryngol Head Neck Surg. 2024 Oct;76(5):3956-3961. doi: 10.1007/s12070-024-04752-2. Epub 2024 Jun 6.
ABSTRACT
Epithelioid haemangioendothelioma (EHE) is a rare vascular tumour that primarily affects the liver, lungs and bone. It is very rarely described in the head and neck region, and is exceptionally uncommon within the submandibular region. We report a very rare case of EHE originating in a lymph node within the submandibular salivary gland of a 54-year-old female patient. The tumour was resected and the patient was regularly followed up, with no recurrence of disease at 24 months postoperatively. A review of existing literature, clinical and immunohistopathological features are discussed, which highlight the diagnostic dilemma, absence of consensus for management and appropriate surveillance method associated with EHE.
PMID:39376453 | PMC:PMC11455700 | DOI:10.1007/s12070-024-04752-2
Indian J Otolaryngol Head Neck Surg. 2024 Oct;76(5):4626-4629. doi: 10.1007/s12070-024-04651-6. Epub 2024 May 31.
ABSTRACT
This case report details the management of orbital cavernous hemangioma in a middle-aged female. The treatment involved a medial orbitotomy combined with an innovative application of a cryoprobe. This approach underscores the benefits of open orbitotomy, particularly when combined with cryoextraction. This combination provides improved control over bleeding and results in a substantial reduction in surgical time, offering valuable insights in the context of the contemporary trend towards endoscopic surgeries.
PMID:39376425 | PMC:PMC11455788 | DOI:10.1007/s12070-024-04651-6
PLoS One. 2024 Oct 7;19(10):e0311665. doi: 10.1371/journal.pone.0311665. eCollection 2024.
ABSTRACT
This scientific report aims to investigate the genotype-phenotype correlations of retinal hemangioblastoma (RH) in von Hippel-Lindau (VHL) disease. The study included 77 patients with genetically confirmed VHL disease who visited an ophthalmology clinic for the evaluation of RH. The presence, location, and size of RH were evaluated, Patients were categorized into three groups based on variants: HIF-1α binding site missense (HM), non-HIF-1α binding site missense (nHM), and truncating (TR) mutations. Fifty-six patients (72.7%) had RH in either eye, and 24 had bilateral RH. Sixteen patients (20.8%) had juxtapapillary RH in either eye. Nine patients had RH ≥ 2.0 disc diameters in size. VHL c.208G>A variant was the most frequent single mutation. Compared with patients having nHM mutations (15 patients) in VHL gene, patients with HM mutations (33 patients) or TR mutations (26 patients) presented a greater number of eyes affected (p = 0.007 and 0.004, respectively), a greater number of RH (p = 0.012 and 0.003, respectively), and more frequent presentation of large RH ≥ 2.0 disc diameters (p = 0.012, and 0.013, respectively). In conclusion, this study provides a deeper understanding of the genetic spectrum of VHL disease in Korean VHL disease and highlights the importance of the location of missense mutations regarding the risk of RH.
PMID:39374255 | PMC:PMC11458008 | DOI:10.1371/journal.pone.0311665
Cureus. 2024 Oct 4;16(10):e70853. doi: 10.7759/cureus.70853. eCollection 2024 Oct.
ABSTRACT
Intravascular lobular capillary hemangioma (ILCH), also known as intravenous pyogenic granuloma, is a benign vascular lesion with a distinctive lobular arrangement of capillaries. It is typically confined to the head, neck, and upper extremities, and its occurrence in the testicular region is exceedingly rare. Here, we present a case of a 68-year-old male who was initially diagnosed with a testicular tumor based on imaging studies but was later found to have an ILCH located in the epididymis. This case underscores the diagnostic challenges posed by this rare entity due to its unusual presentation and mimicking of malignant conditions.
PMID:39372378 | PMC:PMC11452083 | DOI:10.7759/cureus.70853
Cureus. 2024 Sep 5;16(9):e68711. doi: 10.7759/cureus.68711. eCollection 2024 Sep.
ABSTRACT
Hemangiomas are benign tumors characterized by an abnormal proliferation of blood vessels, which can be particularly challenging to diagnose and manage when located in unusual sites such as the foot. Herein, we report a case of a 36-year-old woman with a plantar hemangioma on the right foot, characterized by a long-standing, periodically changing subcutaneous lump. Clinical examination and magnetic resonance imaging revealed a hyperintense mass involving the musculus flexor digitorum brevis. The patient underwent surgical excision, which was complicated by intraoperative rupture of the mass but ultimately resulted in complete removal. Histopathological analysis confirmed the diagnosis of an intramuscular hemangioma. Postoperative recovery was uneventful, and follow-up showed no recurrence after six months. This case highlights the critical role of accurate diagnosis through physical examination and imaging, particularly magnetic resonance imaging, to differentiate benign hemangiomas from malignant tumors and guide treatment. While surgical excision is the primary treatment for symptomatic or cosmetically concerning hemangiomas, less invasive alternatives like sclerotherapy may be appropriate for superficial lesions. Effective management requires precise diagnostic imaging and a tailored therapeutic approach.
PMID:39371750 | PMC:PMC11453145 | DOI:10.7759/cureus.68711
Nat Commun. 2024 Oct 6;15(1):8660. doi: 10.1038/s41467-024-52866-6.
ABSTRACT
Force-driven cellular interactions are crucial for cancer cell invasion but remain underexplored in vascular abnormalities. Cerebral cavernous malformations (CCM), a vascular abnormality characterized by leaky vessels, involves CCM mutant cells recruiting wild-type endothelial cells to form and expand mosaic lesions. The mechanisms behind this recruitment remain poorly understood. Here, we use an in-vitro model of angiogenic invasion with traction force microscopy to reveal that hyper-angiogenic Ccm2-silenced endothelial cells enhance angiogenic invasion of neighboring wild-type cells through force and extracellular matrix-guided mechanisms. We demonstrate that mechanically hyperactive CCM2-silenced tips guide wild-type cells by transmitting pulling forces and by creating paths in the matrix, in a ROCKs-dependent manner. This is associated with reinforcement of β1 integrin and actin cytoskeleton in wild-type cells. Further, wild-type cells are reprogrammed into stalk cells and activate matrisome and DNA replication programs, thereby initiating proliferation. Our findings reveal how CCM2 mutants hijack wild-type cell functions to fuel lesion growth, providing insight into the etiology of vascular malformations. By integrating biophysical and molecular techniques, we offer tools for studying cell mechanics in tissue heterogeneity and disease progression.
PMID:39370485 | PMC:PMC11456588 | DOI:10.1038/s41467-024-52866-6
Int J Cancer. 2024 Oct 6. doi: 10.1002/ijc.35207. Online ahead of print.
ABSTRACT
Kaposiform hemangioendotheliomas (KHEs) are vascular tumors that are considered borderline or locally aggressive and may lead to lethal outcomes. Traditional therapies, such as surgery and embolization, may be insufficient or technically impossible for patients with KHE. Sirolimus (or rapamycin), a specific inhibitor of mechanistic target of rapamycin, has recently been demonstrated to be very useful in the treatment of KHEs. Here, we highlight recent substantial progress regarding the effects of sirolimus on KHEs and discuss the potential mechanisms of action of sirolimus in treating this disease. The prevention of platelet activation and inflammation, along with antiangiogenic effects, the inhibition of lymphangiogenesis, the attenuation of fibrosis, or a combination of all these effects, may be responsible for the therapeutic effects of sirolimus. In addition, the mechanism of sirolimus resistance in some KHE patients is discussed. Finally, we review the somatic mutations that have recently been identified in KEH lesions, and discuss the potential of novel therapeutic targets based on these further understandings of the cellular and molecular pathogenesis of KHE.
PMID:39369447 | DOI:10.1002/ijc.35207
J Fr Ophtalmol. 2024 Oct 3:104311. doi: 10.1016/j.jfo.2024.104311. Online ahead of print.
NO ABSTRACT
PMID:39366821 | DOI:10.1016/j.jfo.2024.104311
Clin Exp Dermatol. 2024 Oct 4:llae401. doi: 10.1093/ced/llae401. Online ahead of print.
ABSTRACT
Infantile haemangiomas (IH) remain the most common benign vascular tumours in childhood. While most IH can be managed conservatively, a proportion of these lesions can cause disfigurement, ulceration or functional impairment, requiring prompt intervention. Propranolol, a lipophilic non-selective beta blocker, has been regarded as first-line therapy, following a serendipitous discovery of its use for IH in 2008. While efficacious, it has been associated with adverse effects such as hypoglycaemia, bronchospasm, sleep disturbances and agitation in infant trials. Hence, atenolol, a hydrophilic beta-1 selective blocker, has demonstrated similar efficacy and potentially greater tolerability, being less likely to cause sleep disturbances given its inability to cross the blood brain barrier, and a decrease in bronchial reactivity. The purpose of this review is to explore and critique the current knowledge on the efficacy and safety of propranolol against atenolol in children with IH. A total of seven studies comparing the two beta-blockers were identified in our search. Atenolol appeared to be as efficacious as propranolol and was associated with fewer central nervous system and bronchial-related adverse events. Further research exploring the optimal dosing for atenolol, particularly for ulcerated or syndromic IH, as well as incidence and management of rebound growth would be beneficial.
PMID:39365598 | DOI:10.1093/ced/llae401
Ann Plast Surg. 2024 Sep 3. doi: 10.1097/SAP.0000000000004094. Online ahead of print.
ABSTRACT
STUDY DESIGN: Systematic, scoping literature review and case presentation.
OBJECTIVE: The goal of this study is to review current literature on management trends and outcomes of pediatric intranasal lobular capillary hemangioma (ILCH).
CASE PRESENTATION: A 14-year-old male patient presented with a 15-month history of unilateral epistaxis, nasal congestion, and mouth breathing. Further workup revealed a pale nasal mass obstructing the right nasal cavity. The patient was successfully treated using a minimally invasive endoscopic endonasal approach for mass resection and nasal floor free mucosal graft for septal reconstruction.
METHODS: Independent queries of the PubMed Central, MEDLINE, and Bookshelf databases were performed; articles from 1990 to 2023 were abstracted. All studies that described pediatric ILCH using the Boolean method and relevant search term combinations, including "Intranasal," "Lobular capillary hemangioma," "Pyogenic granuloma," "Pediatric," "Outcome," and "Management," were collected for subsequent analysis.
RESULTS: A total of 407 relevant unique articles were identified for analysis. Of these, 19 articles were deemed appropriate for inclusion in this literature review. Twenty-two pediatric ILCH cases were identified with a mean age at diagnosis of 10.5 years. The majority of cases occurred in males and presented with recurrent epistaxis and nasal obstruction. Most lesions originated from the anterior nasal septum and were resected using an endoscopic endonasal approach with no recurrence at last follow-up.
CONCLUSIONS: Pediatric ILCH, a benign vascular neoplasm, often presents with unilateral nasal obstruction and severe, refractory epistaxis. This comprehensive review aims to highlight the importance of including this lesion in the differential diagnosis for unilateral nasal obstruction and epistaxis in young children.
PMID:39227566 | DOI:10.1097/SAP.0000000000004094
Eur J Nucl Med Mol Imaging. 2024 Sep 4. doi: 10.1007/s00259-024-06909-1. Online ahead of print.
NO ABSTRACT
PMID:39227425 | DOI:10.1007/s00259-024-06909-1
Asian J Surg. 2024 Sep 3:S1015-9584(24)01835-9. doi: 10.1016/j.asjsur.2024.08.095. Online ahead of print.
NO ABSTRACT
PMID:39227298 | DOI:10.1016/j.asjsur.2024.08.095
IJU Case Rep. 2024 Aug 5;7(5):404-407. doi: 10.1002/iju5.12768. eCollection 2024 Sep.
ABSTRACT
INTRODUCTION: Hemiscrotal agenesis is a rare scrotal developmental disorder. Orchiopexy on the opposite side of the scrotum with rugae or scrotoplasty combined with orchiopexy is usually performed as a surgical treatment for hemiscrotal agenesis with cryptorchidism. Till date, there are only eight published case studies of hemiscrotal agenesis.
CASE PRESENTATION: A 6-year-old boy, who had been previously treated for infantile hemangiomas, was referred by a pediatrician to our hospital for the follow-up of hemiscrotal agenesis without cryptorchidism. Thermography demonstrated that the temperature of the right scrotal skin with no rugae was higher than that of the left scrotal skin with rugae. The patient's parent declined scrotoplasty because the hemiscrotal agenesis was to be followed up without scrotoplasty and scrotal ultrasonography revealed no abnormal findings in both testes.
CONCLUSION: High-temperature environment may not impair the testicular growth in prepubertal hemiscrotal agenesis without cryptorchidism.
PMID:39224679 | PMC:PMC11366427 | DOI:10.1002/iju5.12768
IJU Case Rep. 2024 Jul 9;7(5):379-382. doi: 10.1002/iju5.12760. eCollection 2024 Sep.
ABSTRACT
INTRODUCTION: We describe a case of an adrenal cavernous hemangioma that was surgically resected because of tumor growth and intratumoral hemorrhage.
CASE PRESENTATION: A 73-year-old woman presented with an enlarged adrenal tumor and intratumoral hemorrhage during the follow-up of an incidental adrenal tumor. A computed tomography showed that the left adrenal tumor had grown from 23 to 44 mm over 1 year. Blood tests revealed a normal metabolic profile. Paragangliomas and metastatic tumors were suspected on imaging. Laparoscopic adrenalectomy was performed to prevent tumor rupture due to further bleeding. No adhesions or bleeding were observed around the tumor during surgery. Pathological diagnosis was adrenal cavernous hemangioma.
CONCLUSION: Adrenal cavernous hemangioma is difficult to distinguish preoperatively from other adrenal tumors, including malignant tumors. The intraoperative findings of this case suggest that laparoscopic adrenalectomy is a safe treatment option for relatively small adrenal cavernous hemangioma.
PMID:39224674 | PMC:PMC11366426 | DOI:10.1002/iju5.12760
Ocul Oncol Pathol. 2024 Sep;10(3):146-153. doi: 10.1159/000539384. Epub 2024 May 22.
ABSTRACT
INTRODUCTION: We aim to report the anatomical and functional outcomes of ruthenium-106 brachytherapy in the management of circumscribed choroidal hemangiomas (CCH).
METHODS: This is a single-center, retrospective case series including patients with unilateral symptomatic CCH treated with ruthenium-106 brachytherapy at the Cairo University Ocular Oncology Service. Patient records were analyzed for patients' demographics, best corrected visual acuity (BCVA), tumor dimensions (thickness and largest base diameter), foveal subretinal fluid, radiation-related complications, and recurrence.
RESULTS: Seven patients were included in the study (including 6 males) with a mean age of 39.3 ± 15.4 years; ruthenium-106 plaque was used to deliver 50 Gray to the tumor apex. After a mean follow-up duration of 12.5 months, all patients had significant improvement in BCVA after treatment, mean tumor height decreased significantly from 4.76 ± 1.76 mm to 1.70 ± 1.2 mm (p value 0.01). The largest tumor base diameter also decreased significantly from 9.13 ± 2.68 mm to 4.65 ± 3.75 mm (p value 0.05). Subretinal fluid and exudative retinal detachment resolved in all patients, and no significant radiation-related complications were observed in any patient. None of the patients needed any further treatment or experienced recurrence within the follow-up period.
CONCLUSION: Ruthenium-106 brachytherapy is an effective tool in the management of symptomatic CCH with a good visual prognosis and safety profile.
PMID:39224529 | PMC:PMC11368397 | DOI:10.1159/000539384
J Neurosurg Case Lessons. 2024 Sep 2;8(10):CASE24227. doi: 10.3171/CASE24227. Print 2024 Sep 2.
ABSTRACT
BACKGROUND: Cerebral cavernous hemangiomas (CHs) are the most common vascular malformations and can be found in many locations in the brain. The most common extra-axial locations are the intrasellar and parasellar regions. Both locations are rare for CH.
OBSERVATIONS: A 41-year-old female who had experienced headaches for about a year presented to the authors' institution because of a mass located in the left cavernous sinus (CS). It was confirmed that it was not causing any loss in the visual field and that there was no pituitary irregularity. Surgery was planned with a preliminary diagnosis of CH. An endoscopic endonasal approach (EEA) was undertaken with the otorhinolaryngology team. The dura mater was opened following excision of the back wall of the sphenoid sinus. A blue-purple vascular lesion was observed, filling the left CS. Gross-total resection (GTR) was achieved. In the literature, there were 10 cases in which GTR had been performed using the EEA.
LESSONS: Patients with CHs located in the sellar region present with various complaints. The preference for endoscopic surgery over cranial surgery for such lesions requires more comprehensive studies, but it is thought that this approach can reduce surgical complications and the time to discharge for the patient. [https:]
PMID:39222542 | DOI:10.3171/CASE24227
Cureus. 2024 Aug 1;16(8):e65927. doi: 10.7759/cureus.65927. eCollection 2024 Aug.
ABSTRACT
When treating patients with a massive cavernous hemangioma of the liver that requires nonsurgical therapy, transcatheter arterial embolization has proven to be an effective technique. Significant advantages include the ability to obliterate the vascular supply of these lesions and the minimally invasive nature compared to surgery. A 65-year-old woman arrived at our hospital complaining primarily of stomach pain that had been there for six months. The patient had a hard lump in the right hypochondrium on clinical examination. Ultrasound showed a large, well-defined, heterogeneous lesion with central necrotic areas, with the rest of the liver parenchyma having normal echotexture and flow in the portal vein. The 65-year-old woman's primary complaint upon arrival at our hospital was a stomach ache that had been there for six months. The results of the liver function test were normal. Upon presumptive identification of a significant hepatic hemangioma, the patient was brought to the angio-suite for angiography and proper hepatic artery embolization. Considering the patient's age, the severity of the lesion, and its highly vascular character, endovascular embolization of the proper hepatic artery using lipidol and bleomycin was performed. The patient was discharged after two days in the hospital, administered antibiotics, and advised to follow up after 15 days. Liver function after embolization was within normal limits. The patient had no symptoms after a follow-up at three months. Therefore, endovascular embolization with lipidol and bleomycin is a safe and effective method to obliterate the vascular supply to the lesion, prevent catastrophic bleeding, and provide symptomatic relief to the patient.
PMID:39221374 | PMC:PMC11365718 | DOI:10.7759/cureus.65927
Cureus. 2024 Aug 2;16(8):e66020. doi: 10.7759/cureus.66020. eCollection 2024 Aug.
ABSTRACT
Ramsay Hunt syndrome is a rare viral condition that develops from the varicella zoster virus that affects the face's geniculate ganglion. It has been defined by a herpes zoster oticus, which can be associated with further cranial nerve lesions and acute peripheral facial nerve palsies. In this case, we present a 73-year-old female patient who presented to the outpatient department (OPD) with unbearable pain in the lower left cheek that she had been experiencing for the last four days. The reported pain was continuous and could be described as deep-aching and burning. Facial swelling was observed in relation to the lower lip, especially in the vermillion area; there was ulceration, paralysis seen on the left face, and swelling on the submandibular region. Intravenous corticosteroids and antiviral drugs were administered to her for seven days as an association therapy. In this report, the authors want to stress the necessity of using adequate clinical examination and early intervention in the case of the Ramsay Hunt syndrome.
PMID:39221360 | PMC:PMC11366263 | DOI:10.7759/cureus.66020
J Surg Case Rep. 2024 Aug 30;2024(8):rjae536. doi: 10.1093/jscr/rjae536. eCollection 2024 Aug.
ABSTRACT
Pediatric hemangiomas are benign vascular malformations or angiodysplasias characterized by the proliferation of endothelial cells in blood vessels. Their incidence is 4%-10% in children under 1 year of age, and they most frequently occur on the head and neck [1, 2]. In addition to causing facial deformities, facial hemangiomas can lead to both functional and aesthetic issues, often resulting in significant complications. These complications negatively impact the quality of life for patients and can also affect the psychological well-being and self-esteem of their families [3]. With the expected global smartphone penetration reaching 70% [4], this case report explores the use of smartphones integrated with thermographic cameras to monitor the involution of hemangiomas during sclerosing treatment. This approach represents a promising advancement in managing and assessing hemangioma treatment through accessible, real-time monitoring technology.
PMID:39220167 | PMC:PMC11364451 | DOI:10.1093/jscr/rjae536
Oncol Res. 2024 Aug 23;32(9):1453-1465. doi: 10.32604/or.2024.048315. eCollection 2024.
ABSTRACT
BACKGROUND: Interleukin 13 receptor subunit alpha 2 (IL13RA2) plays an essential role in the progression of many cancers. However, the role of IL13RA2 in infantile haemangioma (IH) is still unknown.
MATERIALS AND METHODS: IL13RA2 expression in IH tissues was analyzed using western blot, qRT-PCR, and immunofluorescence. The role of IL13RA2 in haemangioma-derived endothelial cells (HemECs) was determined following knockdown or overexpression of IL13RA2 using CCK-8, colony formation, apoptosis, wound healing, tubule formation, Transwell, and western blot.
RESULTS: IL13RA2 expression was upregulated in IH tissues. IL13RA2 overexpression promoted proliferation, migration, and invasion of HemECs and induced glycolysis, which was confirmed with a glycolysis inhibitor. Specifically, IL13RA2 interacted with β-catenin and activated the Wnt/β-catenin pathway in HemECs, which were involved in the above-mentioned effects of IL13RA2.
CONCLUSIONS: These findings revealed that targeting IL13RA2 is a potential therapeutic approach for IH.
PMID:39220137 | PMC:PMC11361910 | DOI:10.32604/or.2024.048315
World J Gastrointest Surg. 2024 Aug 27;16(8):2735-2741. doi: 10.4240/wjgs.v16.i8.2735.
ABSTRACT
BACKGROUND: Anorectal hemangioma is a rare and frequently misdiagnosed cause of lower gastrointestinal (GI) bleeding. Here, we present a minimally invasive therapy with selective embolization.
CASE SUMMARY: A 21-year-old male patient experienced painless rectal bleeding since childhood and was treated for ulcerative colitis. Diagnostic studies later revealed specific characteristics for vascular lesions-anorectal hemangiomas. The severity of rectal bleeding caused symptomatic anemia and possible surgical treatment was associated with a high risk of fecal incontinence. Here, we present selective embolization, a minimally invasive therapeutic approach that is proven as an alternative therapeutic method of choice. The patient significantly improved temporarily and had a small ischemic ulcer, which healed with a control colonoscopy and developed no stenosis.
CONCLUSION: Awareness of the clinical and radiological features of GI hemangiomas may help improve diagnostics and avoid inappropriate therapeutic procedures.
PMID:39220052 | PMC:PMC11362948 | DOI:10.4240/wjgs.v16.i8.2735
Cureus. 2024 Jul 31;16(7):e65904. doi: 10.7759/cureus.65904. eCollection 2024 Jul.
ABSTRACT
Hemangiomas are benign vascular tumours of the head and neck. Lobular capillary hemangioma (LCH) is a common, acquired proliferative reaction in vascular tissue. It has female predilection, and peak incidence occurs in adolescents and young adults. Histopathologically, it is characterised by nodular proliferation of capillary-sized vessels lined by endothelial cells with plump nuclei. The capillary lumen shows the presence of numerous erythrocytes. To distinguish this lesion from other vascular lesions, a precise diagnosis is required. The majority of oral hemangiomas will regress without any treatment. If these tumours continue into adulthood, it may lead to difficulty in speech and swallowing. This case report presents an atypical manifestation of LCH of the buccal mucosa in a 51-year-old male patient.
PMID:39219971 | PMC:PMC11364916 | DOI:10.7759/cureus.65904
Ear Nose Throat J. 2024 Sep 1:1455613241279716. doi: 10.1177/01455613241279716. Online ahead of print.
NO ABSTRACT
PMID:39219233 | DOI:10.1177/01455613241279716
Ear Nose Throat J. 2024 Aug 30:1455613241276391. doi: 10.1177/01455613241276391. Online ahead of print.
ABSTRACT
Laryngeal hemangiomas are rare vascular tumors, mainly in children, and less common in adults. Giant lesions typically require multiple endoscopic procedures and temporary tracheostomy. Open surgery allows single-procedure removal with fewer complications. CO2 laser excision controls bleeding and minimizes tissue damage. This study evaluates this hybrid approach to manage adult giant supraglottic hemangiomas. A 54-year-old woman with a giant supraglottic hemangioma was successfully treated using a combination of open surgery and CO2 laser. Limited data on adult laryngeal hemangiomas result in a lack of established treatment protocols. For giant supraglottic hemangiomas, combining open surgery with CO2 laser resection offers distinct advantages: enhanced lesion visibility, optimal airway control, effective hemostasis, reduced tissue damage, and lower recurrence rates. This hybrid approach also supports rapid recovery and favorable clinical outcomes. Combining open surgical excision with CO2 laser is effective for managing giant supraglottic hemangiomas in adults.
PMID:39215488 | DOI:10.1177/01455613241276391
BMJ Case Rep. 2024 Aug 30;17(8):e255434. doi: 10.1136/bcr-2023-255434.
ABSTRACT
A girl in middle childhood presented with glaucoma in her right eye along with segmental haemangiomas on the right side of the face and neck. Magnetic resonance angiography of the brain showed hypoplasia of the right internal carotid artery, leading to the diagnosis of posterior fossa malformations, haemangioma, arterial anomalies, cardiac defects and eye abnormalities (PHACE) syndrome. High-definition anterior segment ocular coherence tomography (AS-OCT) of the right eye showed an absence of Schlemm's canal and a hyperreflective membrane over the trabecular meshwork. The presence of this angle dysgenesis on AS-OCT, a novel finding in this disease, explained the elevated intraocular pressure in the right eye. The embryological basis for the development of angle dysgenesis might help better understand the pathogenesis of PHACE syndrome.
PMID:39214587 | DOI:10.1136/bcr-2023-255434
CJC Open. 2024 May 28;6(8):1028-1031. doi: 10.1016/j.cjco.2024.05.009. eCollection 2024 Aug.
NO ABSTRACT
PMID:39211752 | PMC:PMC11357761 | DOI:10.1016/j.cjco.2024.05.009
Prog Rehabil Med. 2024 Aug 29;9:20240027. doi: 10.2490/prm.20240027. eCollection 2024.
ABSTRACT
BACKGROUND: : COVID-19 can cause respiratory symptoms, as well as various complications and sequelae. This report describes a patient with worsening neurological symptoms caused by a spinal cavernous hemangioma after infection with COVID-19. Cavernous hemangioma usually occurs in the upper part of the brain (70%-90%) and rarely occurs in the spinal cord (5%-7%). Approximately 65% of cases of intramedullary spinal cavernous hemangioma present with neurological symptoms, and more than half of these cases show a slow worsening of symptoms. This is a rare case of intramedullary spinal cavernous hemangioma with cysto-rectal involvement in which neurological symptoms rapidly worsened following COVID-19 infection.
CASE: : A woman in her 30s was admitted to the hospital because of the sudden onset of muscle weakness in both lower limbs and cysto-rectal disturbances after COVID-19 infection. She was diagnosed with a hemorrhage from a spinal cord tumor and underwent emergency resection. The pathological diagnosis was a spinal cavernous hemangioma. At first, she had a spinal cord injury (third thoracic vertebrae; American Spinal Injury Association Impairment Scale, C; Frankel classification, B; with cysto-rectal impairment), but 2 months later, she started walking with knee-ankle-foot orthoses and parallel bars. After 3 months, she could move independently around the ward using a wheelchair. Upon discharge, the patient could walk with ankle-foot orthoses and Lofstrand crutches.
DISCUSSION: : COVID-19 is associated with various extrapulmonary manifestations and may increase the risk of hemorrhage in cases of intramedullary spinal cavernous hemangioma.
PMID:39211535 | PMC:PMC11350290 | DOI:10.2490/prm.20240027
Clin Case Rep. 2024 Aug 29;12(9):e9362. doi: 10.1002/ccr3.9362. eCollection 2024 Sep.
ABSTRACT
Ovarian hemangioma, though rare and asymptomatic, can mimic malignant ovarian tumors, thus it is necessary for comprehensive histopathological and immunohistochemical evaluation for accurate diagnosis and appropriate management.
PMID:39210922 | PMC:PMC11358698 | DOI:10.1002/ccr3.9362
Clin Gastroenterol Hepatol. 2024 Aug 27:S1542-3565(24)00785-7. doi: 10.1016/j.cgh.2024.08.016. Online ahead of print.
NO ABSTRACT
PMID:39209189 | DOI:10.1016/j.cgh.2024.08.016
Cancers (Basel). 2024 Aug 21;16(16):2908. doi: 10.3390/cancers16162908.
ABSTRACT
An incidental focal liver lesion (IFLL) is defined as a hepatic lesion identified in a patient imaged for an unrelated reason. They are frequently encountered in daily practice, sometimes leading to unnecessary, invasive and potentially harmful follow-up investigations. The clinical presentation and the imaging aspects play an important role in deciding if, and what further evaluation, is needed. In low-risk patients (i.e., without a history of malignant or chronic liver disease or related symptoms), especially in those younger than 40 years old, more than 95% of IFLLs are likely benign. Shear Wave liver Elastography (SWE) of the surrounding liver parenchyma should be considered to exclude liver cirrhosis and for further risk stratification. If an IFLL in a low-risk patient has a typical appearance on B-mode ultrasound of a benign lesion (e.g., simple cyst, calcification, focal fatty change, typical hemangioma), no further imaging is needed. Contrast-Enhanced Ultrasound (CEUS) should be considered as the first-line contrast imaging modality to differentiate benign from malignant IFLLs, since it has a similar accuracy to contrast-enhanced (CE)-MRI. On CEUS, hypoenhancement of a lesion in the late vascular phase is characteristic for malignancy. CE-CT should be avoided for characterizing probable benign FLL and reserved for staging once a lesion is proven malignant. In high-risk patients (i.e., with chronic liver disease or an oncological history), each IFLL should initially be considered as potentially malignant, and every effort should be made to confirm or exclude malignancy. US-guided biopsy should be considered in those with unresectable malignant lesions, particularly if the diagnosis remains unclear, or when a specific tissue diagnosis is needed.
PMID:39199678 | PMC:PMC11352778 | DOI:10.3390/cancers16162908
Colloids Surf B Biointerfaces. 2024 Aug 24;244:114174. doi: 10.1016/j.colsurfb.2024.114174. Online ahead of print.
ABSTRACT
Hemangiomas are superficial tumors characterized by dense vascular structures that often affect the patient's aesthetic appearance due to the obvious red appearance on the skin. Current treatments, especially timolol maleate in the form of eye drops and hydrogels, suffer from low transdermal drug delivery rates, resulting in prolonged treatment time. To address this challenge, our study introduced a soluble microneedle patch with dextran as the main material to form microcatheters for sustained delivery of timolol maleate. In addition, we proposed a vascular embolization strategy to disrupt the blood supply in hemangiomas. Oxidized cellulose (C-cellulose) was selected for its excellent hemostatic properties. We incorporated C-cellulose into dextran microneedles to facilitate thrombosis in the vascular-rich areas of hemangiomas. The innovative microneedle patch we developed can penetrate the skin to a depth of 430 μm and dissolve rapidly within 3 minutes, ensuring direct drug delivery to the subcutaneous layer. Notably, the treated skin area regained its original appearance within two hours after treatment. In addition to excellent skin permeability and rapid dissolution, these patches significantly promoted apoptosis and inhibited cell migration in mouse hemangioendothelioma EOMA cells. Our results demonstrate that this approach not only achieves significant tumor inhibition in a mouse hemangioma model, but also represents a more effective, convenient, and non-invasive treatment option. Therefore, dextran/C-cellulose/timolol maleate microneedle patch (MNs/Timolol) has broad clinical application prospects in the treatment of hemangiomas, minimizing the risk of additional damage and improving treatment efficacy.
PMID:39197327 | DOI:10.1016/j.colsurfb.2024.114174
JMIR Dermatol. 2024 Aug 28;7:e52493. doi: 10.2196/52493.
ABSTRACT
Epithelioid hemangioendothelioma (EHE) is a rare vascular tumor with metastatic potential. EHE can have single- or multiorgan involvement, with presentations ranging from asymptomatic disease to pain and systemic symptoms. The extremely heterogeneous clinical presentation and disease progression complicates EHE diagnosis and management. We present the case of a 24-year-old woman with two periauricular erythematous papules, leading to the discovery of metastatic EHE through routine biopsy, despite a noncontributory medical history. Histology revealed the dermal proliferation of epithelioid cells and vacuoles containing red blood cells. Immunohistochemistry markers consistent with EHE solidified the diagnosis. Although extremely rare, prompt diagnosis of EHE is essential for informed decision-making and favorable outcomes. Key clinical and histopathological findings are highlighted to aid dermatologists in diagnosing and managing this uncommon condition.
PMID:39196615 | DOI:10.2196/52493
Nat Cardiovasc Res. 2024 Jul;3(7):771-773. doi: 10.1038/s44161-024-00504-1.
NO ABSTRACT
PMID:39196172 | DOI:10.1038/s44161-024-00504-1
Balkan Med J. 2024 Aug 28. doi: 10.4274/balkanmedj.galenos.2024.2024-5-86. Online ahead of print.
NO ABSTRACT
PMID:39192587 | DOI:10.4274/balkanmedj.galenos.2024.2024-5-86
Clin Nucl Med. 2024 Aug 28. doi: 10.1097/RLU.0000000000005416. Online ahead of print.
ABSTRACT
Cavernous hemangioma of mediastinum is a rare, benign tumor originating from vascular endothelial cells. It typically arises in the anterior mediastinum but has also been reported in the posterior mediastinum or the middle mediastinum, and occasionally involves both the anterior and middle compartments. We present the case of a 20-year-old man with a cavernous hemangioma of mediastinum initially misdiagnosed as a reproductive tumor by 18F-FDG PET/CT.
PMID:39192500 | DOI:10.1097/RLU.0000000000005416
Cureus. 2024 Jul 27;16(7):e65502. doi: 10.7759/cureus.65502. eCollection 2024 Jul.
ABSTRACT
This case report describes the successful surgical removal of a giant hemangioma in a 41-year-old female with hepatitis B. The patient came with stomach distension, right upper quadrant, and right lumbar region pain. Imaging studies showed a mass measuring 12x7.6x11 cm emerging from the left lobe of the liver, causing compression of surrounding structures. The patient has undergone a laparotomy with successful anatomical resection of the hemangioma. Postoperative recovery was uneventful, and the patient was discharged on the fifth postoperative day. This case highlights the significance of considering surgery for symptomatic giant hemangiomas and normal follow-up to screen for recurrence and aims to present the successful surgical management of a giant hemangioma.
PMID:39188480 | PMC:PMC11346131 | DOI:10.7759/cureus.65502
Cureus. 2024 Jul 26;16(7):e65471. doi: 10.7759/cureus.65471. eCollection 2024 Jul.
ABSTRACT
Subglottic hemangiomas are uncommon forms of infantile vascular tumors often misdiagnosed due to symptom overlap with other conditions like laryngomalacia, bronchiolitis, and asthma. Early and accurate diagnosis is vital for effective management. This case report discusses a unique presentation of subglottic hemangioma in a three-month-old infant, highlighting its diagnostic challenge and management. It adds valuable insights into the differentiation of subglottic hemangioma from other common causes of respiratory distress in infants. The infant presented with severe respiratory distress since birth, worsening over the last four weeks, accompanied by gastroesophageal reflux and poor weight gain. Initially, the case was suspected and treated as croup and laryngomalacia. A CT angiogram revealed a vascular lesion in the subglottic area, confirmed by flexible bronchoscopy as a hemangioma. Treatment with propranolol led to significant improvement. Early diagnosis and treatment of subglottic hemangioma are crucial for a good prognosis. This case emphasizes the importance of considering subglottic hemangioma in infants with unresolved airway distress.
PMID:39188454 | PMC:PMC11345464 | DOI:10.7759/cureus.65471
In Vivo. 2024 Sep-Oct;38(5):2545-2549. doi: 10.21873/invivo.13728.
ABSTRACT
BACKGROUND: Adrenal hemangiomas are extremely rare benign tumors that often need to be distinguished from malignancies. Adrenal tumors >4 cm in size are treated surgically because the possibility of malignancy cannot be ruled out. Traditionally, open surgery has been the mainstay of treatment; however, in recent years, robot-assisted surgery has been increasingly used for tumors of larger size and suspected malignancy. Here, we report a case of robot-assisted adrenalectomy for an 11 cm adrenal hemangioma.
CASE REPORT: A 62-year-old male with lateral abdominal pain was referred to our hospital for further examination and treatment. His medical history was significant for hypertension, diabetes, and dyslipidemia. Computed tomography revealed an 11 cm left adrenal tumor, and all endocrinological screening tests were negative. Because the possibility of malignancy could not be ruled out, a robot-assisted adrenalectomy was performed. The operation time was 129 min, and the estimated blood loss was 7 ml. Pathological findings revealed an adrenal hemangioma. The postoperative course was uneventful, and patient's condition subsequently improved postoperatively.
CONCLUSION: Robot-assisted adrenalectomy was performed for a giant adrenal hemangioma without any complications. Robotic surgery is useful for resecting adrenal hemangiomas even exceeding 11 cm in diameter.
PMID:39187351 | PMC:PMC11363784 | DOI:10.21873/invivo.13728
Retina. 2024 Jul 3. doi: 10.1097/IAE.0000000000004199. Online ahead of print.
NO ABSTRACT
PMID:39187262 | DOI:10.1097/IAE.0000000000004199
Circulation. 2024 Aug 27;150(9):738-741. doi: 10.1161/CIRCULATIONAHA.123.067438. Epub 2024 Aug 26.
NO ABSTRACT
PMID:39186529 | DOI:10.1161/CIRCULATIONAHA.123.067438
Abdom Radiol (NY). 2024 Aug 27. doi: 10.1007/s00261-024-04522-2. Online ahead of print.
ABSTRACT
The discontinuous peripheral enhancement is a pattern of enhancement usually attributed to typical cavernous hemangioma, that is the most common benign solid lesion of the liver. The discontinuous peripheral enhancement, however, may be encountered in many other benign and malignant focal liver lesions as an atypical presentation or evolution, and hemangiomas with discontinuous peripheral hyperenhancement on hepatic arterial phase may not always have the typical post-contrast pattern on portal venous and delayed phases. Therefore, abdominal radiologists may be challenged in their practice by lesions with discontinuous peripheral enhancement. This pictorial essay aims to review the spectrum of benign and malignant focal liver lesions that may show discontinuous peripheral enhancement. A particular point of interest is the diagnostic tree pathway that may guide the radiologists in the differential diagnosis.
PMID:39192088 | DOI:10.1007/s00261-024-04522-2
Discov Oncol. 2024 Aug 27;15(1):373. doi: 10.1007/s12672-024-01260-1.
ABSTRACT
BACKGROUND: Retroperitoneal infantile hemangioma (RIH), a type of primary retroperitoneal tumors, are exceptionally rare in clinical practice. Infantile hemangiomas typically manifest on the skin's surface. RIHs are exceptionally rare and typically small. In adults, these tumors often manifest without specific clinical symptoms or detectable signs for a definitive diagnosis. This case report details a patient diagnosed with RIH. We recommend complete excision of the tumor after a comprehensive evaluation, followed by postoperative pathology, to achieve a conclusive diagnosis. We believe that managing critical retroperitoneal structures and vessels intraoperatively presents a significant challenge for all procedures involving primary retroperitoneal tumors. A 47-year-old male was diagnosed with gallstones and underwent surgery 3 months ago at other institution for unexplained nausea and vomiting. Follow-up imaging 2 months after surgery revealed a retroperitoneal mass below the left renal pole. Upon presentation to our hospital, the patient continued to experience intermittent nausea and vomiting, with no other significant symptoms or signs. Considering the patient's 8-year history of hypertension, a paraganglioma was initially suspected. We performed the laparoscopic mass resection after a detailed assessment. However, postoperative pathology revealed it a capillary hemangioma (old term)/infantile hemangioma.
CONCLUSION: RIHs are exceedingly rare benign tumor. The possibility of malignancy should be ruled out, and surgical resection is recommended following a thorough evaluation, with the diagnosis confirmed through pathological examination.
PMID:39190162 | PMC:PMC11349721 | DOI:10.1007/s12672-024-01260-1
J Neurosurg Case Lessons. 2024 Aug 26;8(9):CASE24265. doi: 10.3171/CASE24265. Print 2024 Aug 26.
ABSTRACT
BACKGROUND: Anastomosing hemangiomas are benign vascular neoplasms occurring mainly in the genitourinary tract and paraspinal soft tissues. There have been no reported cases of anastomosing hemangiomas occurring in the spinal epidural space; therefore, their clinical and radiological presentations remain unclear.
OBSERVATIONS: A 55-year-old man presented with progressive back pain and motor and sensory disturbances in both lower extremities. Magnetic resonance imaging (MRI) revealed an extradural lesion at the T4-6 level that extended into the extraspinal region through the left T5-6 intervertebral foramen. Axial MRI revealed that the tumor encircled the spinal cord, with sharp horn-like ends in the intraspinal canal. The patient underwent complete tumor resection with T4-6 laminectomy and left T5-6 foraminotomy. The histopathological diagnosis was an anastomo-sing hemangioma exhibiting anastomosing proliferation of capillary vessels with hobnailed endothelial cells. His symptoms improved, and tumor control was achieved 4 months after surgery without adjuvant therapy.
LESSONS: Although rare, anastomosing hemangiomas can occur in the spinal epidural space. Radiologically, the tumors show horn-like projections encircling the spinal cord, which can be diagnostic cues. Anastomosing hemangiomas should be included in the differential diagnosis of spinal epidural tumors, especially when characteristic horn-like projections are present. [https:]
PMID:39186826 | DOI:10.3171/CASE24265
Ann Ital Chir. 2024;95(4):481-496. doi: 10.62713/aic.3316.
ABSTRACT
AIM: Congenital tumors of the tongue are rare in pediatric patients but encompass a diverse range of entities. Each tumor type exhibits distinct clinical behaviors, necessitating a precise approach to differentiating the tumor types and a tailored, tumor-specific treatment regimen. Advanced imaging techniques, such as diffusion-weighted imaging and perfusion studies, play a vital role in differentiating benign and malignant tongue tumors. This review summarizes current knowledge regarding the presentation, imaging features, and treatment of congenital tongue tumors.
METHODS: A literature review was conducted by searching studies on congenital tongue tumors in databases such as PubMed, Embase, Web of Science, and Scopus. Relevant data, such as clinical features, radiologic characteristics, treatment modalities, and outcomes for different tumor types, were extracted from the selected articles.
RESULTS: Our literature review reveals the various entities of congenital tongue tumors, which can be categorized in terms of hereditary pattern, phenotype, and rarity. Congenital tongue tumors include a range of vascular malformations, such as hemangiomas, lymphatic malformations, arteriovenous malformations, and venous malformations. Another entity is represented by cystic lesions, including dermoid cysts, epidermoid cysts, ranulas, and mucous retention cysts. Rare malignant neoplasms include teratomas and rhabdomyosarcomas. These tumor types vary in terms of swelling, respiratory distress, or impaired oral function, depending on size and location. The detection of these tumors can be carried out using imaging modalities, such as ultrasound, magnetic resonance imaging, and computed tomography, which are utilized to facilitate diagnosis and differentiation. At present, surgical excision remains the cornerstone of treatment, while other modalities may be adopted, depending on tumor type and extent. The prognosis of congenital tongue tumors can be affected by tumor's site, size, involvement of vital structures, and malignancy.
CONCLUSIONS: Given their diversity and complexity, congenital tongue tumors, albeit uncommon, require specialized clinical treatments tailored to each tumor type's characteristics. Understanding the variable presentations and imaging features enables accurate diagnosis, while customized treatment strategies are key to optimizing outcomes and minimizing morbidity in pediatric tongue tumors. This review summarizes current knowledge aimed at enhancing differential diagnosis and management of these diverse entities.
PMID:39186358 | DOI:10.62713/aic.3316
Eur Radiol. 2024 Aug 26. doi: 10.1007/s00330-024-11024-8. Online ahead of print.
ABSTRACT
OBJECTIVES: To create prediction models (PMs) for distinguishing between benign and malignant liver lesions using quantitative data from dual-energy CT (DECT) without contrast agents.
MATERIALS AND METHODS: This retrospective study included patients with liver lesions who underwent DECT, including non-contrast-enhanced scans. Benign lesions included hepatic hemangioma, whereas malignant lesions included hepatocellular carcinoma, metastatic liver cancer, and intrahepatic cholangiocellular carcinoma. Patients were divided into derivation and validation groups. In the derivation group, two radiologists calculated ten multiparametric data using univariate and multivariate logistic regression to generate PMs. In the validation group, two additional radiologists measured the parameters to assess the diagnostic performance of PMs.
RESULTS: The study included 121 consecutive patients (mean age 67.4 ± 13.8 years, 80 males), with 97 in the derivation group (25 benign and 72 malignant) and 24 in the validation group (7 benign and 17 malignant). Oversampling increased the benign lesion sample to 75, equalizing the malignant group for building PMs. All parameters were statistically significant in univariate analysis (all p < 0.05), leading to the creation of five PMs in multivariate analysis. The area under the curve for the five PMs of two observers was as follows: PM1 (slope K, blood) = 0.76, 0.74; PM2 (slope K, fat) = 0.55, 0.51; PM3 (effective-Z difference, blood) = 0.75, 0.72; PM4 (slope K, blood, fat) = 0.82, 0.78; and PM5 (slope K, effective-Z difference, blood) = 0.90, 0.87. PM5 yielded the best diagnostic performance.
CONCLUSION: Multiparametric non-contrast-enhanced DECT is a highly effective method for distinguishing between liver lesions.
CLINICAL RELEVANCE STATEMENT: The utilization of non-contrast-enhanced DECT is extremely useful for distinguishing between benign and malignant liver lesions. This approach enables physicians to plan better treatment strategies, alleviating concerns associated with contrast allergy, contrast-induced nephropathy, radiation exposure, and excessive medical expenses.
KEY POINTS: Distinguishing benign from malignant liver lesions with non-contrast-enhanced CT would be desirable. This model, incorporating slope K, effective Z, and blood quantification, distinguished benign from malignant liver lesions. Non-contrast-enhanced DECT has benefits, particularly in patients with an iodine allergy, renal failure, or asthma.
PMID:39186105 | DOI:10.1007/s00330-024-11024-8
Ophthalmol Retina. 2024 Aug 26:S2468-6530(24)00357-9. doi: 10.1016/j.oret.2024.07.022. Online ahead of print.
NO ABSTRACT
PMID:39186026 | DOI:10.1016/j.oret.2024.07.022
Ear Nose Throat J. 2024 Aug 26:1455613241272466. doi: 10.1177/01455613241272466. Online ahead of print.
ABSTRACT
Primary intraosseous hemangiomas of the maxillofacial region are rare lesions that comprise less than 1% of all osseous tumors. A review of the literature on primary intraosseous hemangiomas of the facial bones revealed a limited number of publications, much of which was largely limited to case reports. This case report summarizes the workup and surgical treatment of a 37-year-old female with a primary intraosseous hemangiomas of the left maxillary bone. The image, histology, treatment, and literature are reviewed.
PMID:39185817 | DOI:10.1177/01455613241272466
Curr Med Imaging. 2024;20:e15734056293540. doi: 10.2174/0115734056293540240325042225.
ABSTRACT
BACKGROUND: Ovarian hemangioma is an extremely rare tumor with atypical clinical manifestations, often discovered incidentally during autopsy or surgery. Approximately 60 cases have been reported in the past, but no more than 10 cases have been investigated by MRI and ultrasound (US).
CASE PRESENTATION: ln this paper, we reported a 51-year-old female patient with Ovarian Hemangioma who had no symptoms of abdominal pain, abnormal vaginal bleeding or discharge, or any other discomfort. Laboratory tests revealed an elevated serum carbohydrate antigen (CA125) of 48.99U/ml (reference range: 0-35U/ml). Multiparametric 3.0T magnetic resonance imaging (MRI) showed a cystic solid mass with a clear boundary and regular shape in the left ovarian area and minimal ascites in the abdominal cavity. The histological examination of the mass confirmed an ovarian hemangioma.
CONCLUSION: The MRI findings of ovarian hemangiomas are highly similar to those observed in hepatic hemangiomas, emphasizing the distinctive radiological characteristics specific to this condition in the ovary. This paper presents an overview of the typical MRI findings associated with ovarian hemangioma, which holds great importance for accurate diagnosis and effective treatment.
PMID:39185656 | DOI:10.2174/0115734056293540240325042225
Curr Med Imaging. 2024;20:e210524230162. doi: 10.2174/157340562001240521165946.
ABSTRACT
An error occurred in the Ethical Commiittie details of the manuscript titled "Imaging Features and Misdiagnosis of Giant Cerebral Cavernous Malformations", 2024; 20: e15734056273891 [1]. Original: ETHICS APPROVAL AND CONSENT TO PARTICIPATE: This study was approved by the institutional review committee of First Affiliated Hospital of Sun Yat-sen University (ZF2022-216). Corrected: ETHICS APPROVAL AND CONSENT TO PARTICIPATE: The ethics approval was provied by the Biomedical Ethics Committee of Guangdong Provincial Hospital of Taditional Chinese Medicine and the written informed consent was exempted. The original article can be found online at [https:]
PMID:39185654 | DOI:10.2174/157340562001240521165946
Ann Maxillofac Surg. 2024 Jan-Jun;14(1):102-105. doi: 10.4103/ams.ams_217_23. Epub 2024 Apr 9.
ABSTRACT
RATIONALE: Haemangiomas are benign vascular malformations. They are common neoplasms of infancy but seldom manifest at birth. Although common in the head & neck region, they are rare in the submandibular region. Despite being benign, surgical treatment is necessary if it causes significant functional or aesthetic compromise.
PATIENT CONCERNS: The patient's primary complaint was swelling in the submandibular region.
DIAGNOSIS: Initial examination and imaging studies were suggestive of sialolithiasis due to the presence of multiple calcifications. Ultrasonographic and magnetic resonance imaging were suggestive of haemangioma with multiple phleboliths.
TREATMENT: Excisional biopsy was done without compromising the submandibular gland. A histopathological examination was done post-operatively to confirm the diagnosis.
OUTCOMES: Post-operative recovery was uneventful and 6-month follow-up showed no recurrence.
TAKE-AWAY LESSON: This case highlights the importance of considering haemangioma in the differential diagnosis of submandibular region masses and emphasises the preservation of the submandibular gland when the extent of the lesion permits.
PMID:39184411 | PMC:PMC11340828 | DOI:10.4103/ams.ams_217_23
Asian J Surg. 2024 Aug 23:S1015-9584(24)01742-1. doi: 10.1016/j.asjsur.2024.08.004. Online ahead of print.
NO ABSTRACT
PMID:39181854 | DOI:10.1016/j.asjsur.2024.08.004
Eur J Obstet Gynecol Reprod Biol. 2024 Sep;300:351-354. doi: 10.1016/j.ejogrb.2024.08.009. Epub 2024 Aug 14.
ABSTRACT
INTRODUCTION: Placental chorioangioma is a benign placenta tumour. Majority of cases, the placental chorioangioma are small and no maternal and fetal complications. We highlight a case diagnosed with large placental chorioangioma with an intrapartum event associated with significant maternal and perinatal morbidity.
METHOD: A 38-year-old woman, Gravida 3 Para 1, with one previous miscarriage, presented with preterm labour at 33 weeks gestation. Antenatally, she was referred to a feto-maternal specialist for finding a placental tumour size 12 × 10 cm. Features are consistent with placental chorioangioma with polyhydramnios. The anomaly scan was normal. Antenatal fetal surveillance with Doppler studies were normal.
RESULTS: During this admission, corticosteroid was given together with a tocolytic agent and opioid analgesia. Unfortunately, the labour progressed, and the patient felt reduced in fetal movement. The cardiotograph showed suspicious tracing. We proceed with emergency caesarean delivery. The placenta was sent for histopathology assessment which confirmed a large placental chorioangioma. The baby was born with Apgar's score of 9 at 1 min, pH of 7.28 and lactate of 7.28 with anaemia and thrombocytopenia. The uterus developed intermittent uterine atony, and the uterotonic agent was given. She recovered well post-delivery. The baby was admitted to the neonatal intensive care unit (NICU) and received a blood product transfusion and discharged from NICU on day 15 of life.
DISCUSSION: Large placental chorioangioma is associated with polyhydramnios, preterm labour, postpartum haemorrhage, fetal anaemia, fetal distress, fetal hydrops and possible perinatal death. Multidisciplinary team involvement with feto-maternal specialists, anaesthetic and neonatologists would improve the outcome of both mother and fetus.
PMID:39152085 | DOI:10.1016/j.ejogrb.2024.08.009
ACG Case Rep J. 2024 Aug 22;11(8):e01471. doi: 10.14309/crj.0000000000001471. eCollection 2024 Aug.
ABSTRACT
Liver embolization is a common procedure for management of liver lesions. Embolization can be performed using only an embolic material or along with chemotherapy agents. Infrequent complications seen postliver embolization include pulmonary thromboembolism, hepatic infarct, liver abscess, liver failure, ischemic biliary strictures, and less frequently pancreatic damage (incidence of 1.7%). We describe a case of necrotizing pancreatitis after bland embolization of a large hepatic hemangioma. The exact mechanisms of acute pancreatitis after liver embolization are uncertain, although direct ischemic mechanisms, toxic effects of antineoplastic agents, and volume of embospheres used are believed to play a role.
PMID:39176214 | PMC:PMC11340917 | DOI:10.14309/crj.0000000000001471
Zh Vopr Neirokhir Im N N Burdenko. 2024;88(4):107-116. doi: 10.17116/neiro202488041107.
ABSTRACT
BACKGROUND: Stereotactic radiosurgery (SRS) for cerebral cavernous malformations has been used for more than 30 years. However, indications for this method and outcomes are still discussable.
OBJECTIVE: To analyze available literature data on SRS for cerebral cavernous malformations with assessment of indications for treatment, radiation parameters, radiological and clinical complications and outcomes.
RESULTS: The final analysis included 20 reports describing post-SRS outcomes in 1834 patients with cerebral cavernous malformations. The main radiation parameter was mean radiation dose to the edge of cavernous malformation (prescribed dose, 13.25±2.16 Gy). In natural course of malformation, mean incidence of hemorrhages from cavernous malformation when counted after the second hemorrhage was 25.9±14.6%, after the patient's birth - 2.59±0.44%. Mean follow-up period after SRS was 66.7±24.1 months. Incidence of hemorrhages from cavernous malformation after SRS for the first 2 years of follow-up was assessed in 14 studies (4.67±3.51%). Incidence of hemorrhages ≥2 years after SRS was analyzed in 12 studies (1.55±0.8%).
CONCLUSION: Despite significant global experience in SRS for cerebral cavernous malformations and many studies devoted to this problem, clear patient selection criteria have not yet been formulated. Modern selection principles have insufficient evidence base.
PMID:39169589 | DOI:10.17116/neiro202488041107
BMJ Case Rep. 2024 Aug 21;17(8):e260483. doi: 10.1136/bcr-2024-260483.
ABSTRACT
An unusual clinical presentation of any pathology prompts a diagnostic dilemma, which further brings challenges in treatment planning. Pyogenic granuloma (microscopically lobular capillary haemangioma) is a benign vascular tumour that commonly involves the skin and mucous membranes and usually manifests as a solitary papule with a friable pedunculated base. Rapid exophytic growth and an ulcerated surface with bleeding on provocation are characteristic, with a gingival predominance for the site. This case report aims to elaborate on an unusual clinical presentation of oral pyogenic granuloma in a middle-childhood female patient. The clinical and radiographic characteristics of the lesion were unremarkable to the diagnosis but rather mimicked other intraoral lesions. It is histopathology that could solve the puzzle with the microscopic and immunohistochemical findings that rendered the final diagnosis of lobular capillary haemangioma.
PMID:39174046 | DOI:10.1136/bcr-2024-260483
Neurology. 2024 Sep 24;103(6):e209799. doi: 10.1212/WNL.0000000000209799. Epub 2024 Aug 22.
NO ABSTRACT
PMID:39173099 | DOI:10.1212/WNL.0000000000209799
Vestn Otorinolaringol. 2024;89(4):54-59. doi: 10.17116/otorino20248904154.
ABSTRACT
Hemangiomas of the nasal cavity are extremely rare in the practice of an otorhinolaryngologist and can be presented in various histopathological variants. Scientific data on hemangiomas of the sinonasal region are analyzed and systematized. The article describes the principles of diagnosis and choice of the method of surgical treatment of hemangiomas. An analysis of the literature data shows that with hemangiomas of the nasal cavity, a comprehensive examination of the patient is required, including collection of complaints and anamnesis, endoscopy of the nasal cavity and computed tomography of the paranasal sinuses, and with significant hemangiomas spreading to neighboring anatomical areas, magnetic resonance imaging with intravenous contrast.
PMID:39171878 | DOI:10.17116/otorino20248904154
Case Reports Plast Surg Hand Surg. 2024 Aug 20;11(1):2393819. doi: 10.1080/23320885.2024.2393819. eCollection 2024.
ABSTRACT
Intramuscular hemangiomas (IMH) are extremely rare, accounting for 0.8% of all hemangiomas. IMH must be included in the differential diagnosis of soft tissue masses, and unexplained muscular pain. We herein describe the case of a patient who presented with an atypical localization of IMH in the infraspinatus muscle.
PMID:39170854 | PMC:PMC11338197 | DOI:10.1080/23320885.2024.2393819
Eur Spine J. 2024 Aug 21. doi: 10.1007/s00586-024-08382-8. Online ahead of print.
ABSTRACT
INTRODUCTION: SH is considered to be the most common benign tumor within the human spine. 1-2% of SH get symptomatic with back pain in most cases. Less often, ingrowth of vessels into the spinal canal is seen. In these cases, more invasive surgical treatment is required. Recurrence of SH following surgical treatment is a very rare condition.
METHODS: We present the results of a retrospective chart review, with description of the clinical course of 12 patients with spinal canal invading SH without recurrence and of 5 patients with a recurrent SH.
RESULTS: Recurrence was diagnosed 70 months (mean value) after the first procedure. All patients with a recurrent SH had received an incomplete tumor resection as a first treatment. The distribution of gender, age at the diagnosis, levels involved by the tumor, and initial symptoms were comparable in the group of patients with and without recurrence. The patients with recurrent SH were treated with preoperative embolization and en bloc resection or complete piecemeal resection of the affected vertebra. For stabilization, a combined, circumferential fixation was used consisting in a TSM cage filled by bone graft and posterior pedicle screw rod instrumentation in all patients. We followed these patients until 09/ 2023, mean FU 122 months (range 72-184 months). At this time, we found no evidence of tumor recurrence, good clinical conditions in all patients, no signs of implant failure or pseudarthrosis.
DISCUSSION: Thus, radical excision, sufficient stabilization and fusion is considered to be an adequate treatment concept for recurrent SH.
PMID:39168894 | DOI:10.1007/s00586-024-08382-8
Case Rep Orthop. 2024 Aug 13;2024:2307950. doi: 10.1155/2024/2307950. eCollection 2024.
ABSTRACT
Aggressive thoracic hemangiomas are rare, benign tumors that extend into the spinal canal and cause neurological symptoms. Delayed diagnosis and treatment, due to a paucity of literature on optimal treatment strategies, can increase morbidity. This case report describes a 19-year-old male patient with aggressive thoracic hemangioma who presented with upper back pain and progressive weakness of the lower extremities. The patient underwent preoperative embolization and sclerotherapy, followed by decompression, posterior instrumentation, and stabilization. The final diagnosis was confirmed by biopsy, and there was a significant improvement in neurology after the surgical intervention. The diagnosis of rare lesions, such as aggressive hemangiomas, requires a high level of clinical suspicion and the assistance of imaging modalities in patients with features of compressive myelopathy. A combination of endovascular and surgical approaches can lead to optimal outcomes.
PMID:39165484 | PMC:PMC11335424 | DOI:10.1155/2024/2307950