Infantile Subglottic Hemangioma: A Case ReportInfantile Subglottic Hemangioma: A Case Report.

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.

Persistent Thrombotic Hemangioma With Organizing/Anastomosing Features: A Case Report of a Guanine Nucleotide-Binding Protein Alpha Subunit (GNA)-Mutated Cutaneous Vascular Lesion.

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.

Synovial Hemangioma: An Overlooked Cause of Chronic Knee Pain in Children.

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.

PHACE syndrome: A review.

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.

A Rare Case of Intravascular Lobular Capillary Hemangioma Mimicking a Testicular Tumor: Diagnostic Challenges.

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.

Safety of Prior Propranolol Therapy for Infantile Hemangioma.

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.

Cryoprobe-Assisted Excision of Cavernous Hemangioma of the Orbit: A Clinical Case Report.

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.

Laparoscopic excision of an incidental vascular tumor.

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.

Cryocarboxy Surgery: A New Armamentarium in the Treatment of Infantile Hemangiomas of the Lips.

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.

A Rare Encounter: Hepatomegaly Unmasked as Multiple Giant Hepatic Hemangiomas.

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.

Long-Term Outcomes of Transarterial Chemoembolization of Giant Liver Hemangiomas with Lipiodol-Bleomycin Emulsion.

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.

Cardiac Hemangioma Mimicking Infective Endocarditis.

Cardiac hemangiomas are rare and often misdiagnosed due to their nonspecific clinical presentations. We report a case of a 70-year-old man presenting with chills and cold sweats, initially suspected of having infective endocarditis based on echocardiographic findings of a mobile mass on the mitral valve. Laboratory results showed leukocytosis and elevated C-reactive protein, but blood cultures were negative. Transesophageal echocardiography later revealed a well-defined mass with characteristics suggestive of a tumor. Surgical excision confirmed the diagnosis of hemangioma. Postoperative recovery was uneventful, with no mitral regurgitation. This case highlights the importance of considering cardiac tumors in the differential diagnosis of intracardiac masses.

Liver transplantation using an otherwise-wasted partial liver resection graft.

Liver transplantation represents a complex surgical procedure and serves as a curative treatment for patients presenting an acute or chronic end-stage liver disease, or carefully selected liver malignancy. A significant gap still exists between the number of available donor organs and potential recipients. The use of an otherwise-wasted resected liver lobe from patients with benign liver tumors is a new, albeit small, option to alleviate the allograft shortage. This review provides evidence that resected liver lobes may be used successfully in liver transplantation.

Cerebellar Venous Hemangioma: Two Case Reports and Literature Review.

Venous hemangiomas within the central nervous system (CNS) represent a rare pathological entity described by sporadic case reports so far. Comprehensive insights into their histological and imaging features, pathogenesis, natural course, and therapeutic modalities are lacking. This review article presents two patients with contrast-enhancing cerebellar lesions near the tentorium cerebelli lacking edema or diffusion restriction. Despite meticulous preoperative neuroradiological examination, diagnostic classification remained inconclusive. Confronted with both-progressive size and diagnostic uncertainty-surgical intervention was undertaken, resulting in uneventful and complete resection of the lesions. Histopathological analyses subsequently revealed a venous hemangioma in each case. In the literature, the term "hemangioma" is often misapplied and inaccurately used to describe a broad spectrum of vascular anomalies. Therefore, a precise identification is essential since the particular type of vascular anomaly affects its natural course and the treatment options available. We aim to contribute to the understanding of this diagnostically intricate entity by presenting the two cases and by providing a detailed overview of radiological and histopathological features of venous hemangiomas.

Immunohistochemical and ultrastructural identification of telocytes in the infantile hemangioma.

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.

Rare Case of an Intramuscular Hemangioma of the Foot: A Case Report With a Review of the Literature.

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.

Unusual Imaging Findings of Epithelioid Hemangioma: Case Report of Single Intramedullary Sclerotic Bone Lesion.

Epithelioid hemangioma (EH) is an uncommon, benign vascular tumor of mesenchymal origin. It mainly presents as a tumor with a lytic appearance and septations. However, no case reports have documented the predominantly sclerotic appearance of EH. Here, we present the imaging findings, including X-ray, CT, MRI, and histopathological findings of a 24-year-old female with EH.

Case report: A case of intercostal intramuscular hemangioma with sternal invasion.

Background: Intramuscular hemangioma is a vascular malformation occurring in muscle tissues. It is most common in skeletal muscles of limbs, especially lower limbs in childhood. The intercostal intramuscular hemangioma with sternal invasion is very rare. Case presentation: The 47-year-old female patient was hospitalized due to "suddenly chest pain for 4 days". Chest contrast-enhanced CT indicated bony changes in the sternum, accompanied by soft tissue shadows in the posterior sternum and diaphragmatic angle. After admission, physical examination showed: peristernal tenderness, percussion pain, other no obvious positive signs. PET-CT examination indicated that there was a high possibility of benign sternal changes. No cancer cells were found by sternal puncture. Surgical biopsy was performed and pathological findings were consistent with intramuscular hemangioma. Conclusion: Intramuscular hemangioma with bone invasion is very rare, imaging examination is difficult to indicate obvious evidence, preoperative diagnosis is difficult, complete surgical resection is very important, more personalized treatment should be provided according to the overall situation of the patient.

On Vascular Lesions of the Thyroid Gland with Emphasis on Intrathyroidal Hemangioma: Clinicopathologic Characterization of Two Cases and Review of the Literature.

Mesenchymal neoplasms of the thyroid gland are exceptionally rare accounting for less than 0.5% of all intrathyroidal tumors with hemangiomas comprising merely 6% of them. The clinicopathologic characteristics of two additional examples of thyroid hemangioma together with a thorough review of the pertinent literature are presented. A 62-year-old man and an 18-year-old woman presented with asymptomatic, soft-to-palpation, mobile nodules of the right thyroid lobe classified as TI-RADS 5 and TI-RADS 4, respectively, on ultrasound imaging. Microscopically, lesions featured a circumscribed, unencapsulated, lobular proliferation of variably-sized, congested, vascular channels lined by a single layer of flattened, cytologically bland endothelial cells, together with interspersed residual follicles. Vascular endothelial cells were strongly positive for CD31, CD34 and ERG, and negative for pancytokeratin AE1/AE3, TTF1, and PAX8. A diagnosis of cavernous hemangioma was rendered in the clinical setting of Hashimoto thyroiditis and follicular adenoma, respectively. Following inclusion of the current cases, a total of 53 intrathyroidal hemangiomas were identified in the literature with a patient mean age of 48.9 years (range = 0.17-84) and a slight female predilection (F:M = 1.4:1). A proclivity for the right thyroid lobe (59.6%) was noted with the striking majority of cases exhibiting features of cavernous hemangioma (95.2%). Prognosis is favorable and surgical resection is considered curative. The occasionally alarming clinical presentation in conjunction with absence of pathognomonic imaging features and limited diagnostic accuracy of FNA cytopathology for such lesions renders surgical intervention necessary for definitive diagnosis of intrathyroidal hemangiomas and exclusion of other epithelial and non-epithelial pathologic entities.