Adult diffuse hepatic hemangiomatosis lesion occupying the entire abdominal and pelvic cavities: a case report.

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.

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.

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.

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.

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.

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.

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.

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.

Ramsay Hunt Syndrome: A Rare Complication of Herpes Zoster Infection With an Incidental Finding of Submandibular Hemangioma.

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.

A Case Report: Giant Liver Hemangioma Treated with Transcatheter Embolization.

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.

Lobular Capillary Hemangioma of the Buccal Mucosa: A Rare Presentation.

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.

Anorectal hemangioma, a rare cause of lower gastrointestinal bleeding, treated with selective embolization: A case report.

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.

Thermal imaging for monitoring the treatment of hemangioma in pediatric patient: case report.

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.

Microsurgical treatment of spinal intradural capillary hemangioma: A consecutive case series of 18 patients and literature review.

PURPOSE: Intradural capillary hemangioma is a rare condition with unclear etiology. Although intradural capillary hemangiomas are benign, they exhibit significant proliferative activity, and their clinical significance should not be underestimated. METHODS: We report a series of spinal intradural capillary hemangiomas to illustrate the characteristics, surgical management, and outcomes. METHODS: A total of 18 consecutive patients who underwent microsurgical treatment were retrospectively reviewed. Patient characteristics were recorded in each case, including presenting symptoms, imaging findings, neurologic status, a surgical procedure performed and follow-up. RESULTS: There were 11(61.1 %) male and 7(38.9 %) female patients, with the ages ranging from 25 to 62 years. The thoracic spine was the most commonly affected site, accounting for 77.8 % (14/18) of the cases. 9 tumors were identified as intradural extramedullary, 3 tumors as intramedullary, and 2 tumors as both extramedullary and intramedullary. There were also 4 cases of tumors localized to the cauda equina. Clinical presentations included back pain, sensory deficits, weakness and gait ataxia with a duration of symptoms ranging from 1 to 12 months. The lesion was hypointense or isointense with the spinal cord on T1- weighted images and hyperintense on T2-weighted images and showed intense enhancement after contrast medium injection. All patients underwent surgical treatment, and no significant postoperative complications were observed. Postoperatively, patients were followed up for an average of 44 months. Follow-up showed that the majority of patients experienced significant improvement in neurological function, with no cases of recurrence. CONCLUSION: Surgical resection is typically the preferred method for treating spinal intradural capillary hemangiomas. Complete resection can relieve spinal cord compression and minimize the risk of recurrence.

A Novel Treatment for Skin Manifestations in Dermatomyositis: A Case Report.

We present a case of refractory cutaneous dermatomyositis (DM) in a 51-year-old Hispanic female which failed multiple treatments but found symptomatic relief with anifrolumab. Anifrolumab was the only treatment that was associated with significant improvement in the rash and pruritis of the patient and lowered her corticosteroid needs. To our knowledge, this is the only second case report that has shown success in treating refractory cutaneous symptoms of DM with anifrolumab after failing standard and multiple combinations of therapies. Anifrolumab is a new first-in-class human monoclonal antibody, which inhibits type 1 interferon receptor (IFN-1) and is used to treat systemic lupus erythematosus (SLE). It is FDA-approved for non-renal manifestations of SLE. This IFN pathway seems to be also active in patients with DM. The presence of IFN-1 and IFN-2 has been reported in muscle biopsies of patients with inflammatory myopathies. Moreover, the IFN activation signature is present in the muscle, blood, and skin of patients with DM. IFN-1 has been assumed to activate toll-like receptors which activate the dendritic cells leading to the secretion of cytokines and chemokines. This potential pathophysiological role of IFN in DM may explain the symptom improvement experienced by our patient after starting anifrolumab treatment. Anifrolumab has additionally been shown to have a good safety profile when used to treat patients with SLE with up to three years of treatment on background conventional disease-modifying antirheumatic drug (DMARD) therapies. In conclusion, SLE and DM share similarities in their pathophysiology and cutaneous disease involvement and can be differentiated clinically. Skin manifestations of DM can persist despite combinations of therapies even when weakness resolves. With this case report, we aim to highlight the possibility of utilizing anifrolumab for treating DM skin manifestations, especially in refractory cases. More research is needed to guide where anifrolumab stands in the therapeutic algorithm for DM. It is unknown whether it treats the myositis component, DM-related arthritis, or coexistent rheumatoid arthritis.

Cavernous hemangioma within the cavernous sinus: illustrative case.

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://thejns.org/doi/abs/10.3171/CASE24227.