Primary intraosseous cavernous hemangioma of the clivus: A case report and literature review.

Background: The radiographic presentation of the primary intraosseous cavernous hemangiomas (PICHs) is nonspecific. We report a case of clival PICH mimicking a chordoma with a literature review. Case Description: A 57-year-old woman presented with diplopia that started a few days before the presentation. She had transient diplopia at the right lateral gaze and upper gaze with normal eye movement. The symptoms disappeared spontaneously 1 week later. She had no other complaints or neurological deficits. Computed tomography revealed an intraosseous mass lesion and bone erosion of the middle and lower clivus, extending laterally to the right occipital condyle. Magnetic resonance imaging (MRI) showed hyperintense and hypointense components on T2- and T1-weighted images, respectively. The lesion was larger than on MRI performed 10 years earlier. Chordoma or chondroma was considered a possible preoperative diagnosis. An endoscopic transsphenoidal approach removed the tumor. In the operating view, the lesion appeared as "moth-eaten" bony interstices filled with vascular soft tissue. Histologically, an intraosseous cavernous hemangioma was diagnosed. Conclusion: Diagnosis before surgery is difficult without characteristic radiographic findings. When making a differential diagnosis of malignant skull lesions, PICH should be considered.

Assessing the Relationship between Surgical Timing and Postoperative Seizure Outcomes in Cavernoma-Related Epilepsy: A Single-Institution Retrospective Analysis of 63 Patients with a Review of the Literature.

Background: Patients with supratentorial cavernous malformations (SCMs) commonly present with seizures. First-line treatments for cavernoma-related epilepsy (CRE) include conservative management (antiepileptic drugs (AEDs)) and surgery. We compared seizure outcomes of CRE patients after early (≤6 months) vs. delayed (>6 months) surgery. Methods: We compared outcomes of CRE patients with SCMs surgically treated at our large-volume cerebrovascular center (1 January 2010-31 July 2020). Patients with 1 sporadic SCM and ≥1-year follow-up were included. Primary outcomes were International League Against Epilepsy (ILAE) class 1 seizure freedom and AED independence. Results: Of 63 CRE patients (26 women, 37 men; mean ± SD age, 36.1 ± 14.6 years), 48 (76%) vs. 15 (24%) underwent early (mean ± SD, 2.1 ± 1.7 months) vs. delayed (mean ± SD, 6.2 ± 7.1 years) surgery. Most (32 (67%)) with early surgery presented after 1 seizure; all with delayed surgery had ≥2 seizures. Seven (47%) with delayed surgery had drug-resistant epilepsy. At follow-up (mean ± SD, 5.4 ± 3.3 years), CRE patients with early surgery were more likely to have ILAE class 1 seizure freedom and AED independence than those with delayed surgery (92% (44/48) vs. 53% (8/15), p = 0.002; and 65% (31/48) vs. 33% (5/15), p = 0.03, respectively). Conclusions: Early CRE surgery demonstrated better seizure outcomes than delayed surgery. Multicenter prospective studies are needed to validate these findings.

Pathological Hip Dislocation in a Neurofibromatosis Patient Secondary to Capsular Hemangioma.

Neurofibromatosis is an autosomal-dominant multi-system disease affecting the nervous, integumentary, ocular, and musculoskeletal systems. In the small number of reported cases, the cause was either atraumatic or an intra-articular neurofibroma. Only a couple of articles in medical literature have reported synovial and capsular hemangioma originating within the hip joint. In this article, we present a rare case of pathological hip dislocation in a neurofibromatosis patient secondary to capsular hemangioma that would be reported for the first time in medical literature. We present the case of a 20-year-old female, resident of Karachi, who presented to the outpatient clinic with pain in the left hip join and inability to bear weight on left leg for 1 week. Diagnosed as a case of posterior hip dislocation after physical examination and imaging, her dislocation was reduced by the Allis method. This maneuver, however, was unsuccessful. Ultimately, the joint was reduced with open reduction via Steinmann pin because post-reduction hip joint was unstable to be contained into the acetabulum; therefore, Steinmann pin was used. The biopsy specimen taken from joint capsule and femoral neck during the surgery revealed cavernous hemangioma within the capsule. Hence, the etiology of posterior hip dislocation was attributed to the presence of capsular hemangioma within the hip joint. The surgery proved successful. The patient had remarkable recovery. The Steinmann pin was removed at 6 weeks, full weight-bearing started at 3 months, and range of motion extended from 0 to 90 degrees at 1 year with imaging studies showing a normally placed hip joint. The presented case reports an unusual etiology of a rare pathology occurring in association with a common genetic disease. It focuses on the importance of thorough examination and extensive relevant investigations in patients presenting with rare pathologies. These practices not only expedite the diagnosis and treatment of such patients, but can also reveal unusual etiologies responsible for uncommon pathologies. This case would help widen the differential diagnosis and treatment strategies of the physicians while dealing with neurofibromatosis patients with pathological hip dislocation. Level of Evidence This is a case report having Level of Evidence 4 in accordance with the levels developed by the Centre of Evidence Based Medicine (CEBM) for treatment.

Surgical management of lobar cerebral cavernous malformations in children: a single-center experience.

PURPOSE: We aimed to determine the surgical indications and postoperative outcomes among pediatric patients with lobar cerebral cavernous malformations (CCMs). METHODS: We retrospectively reviewed pediatric patients operated on for lobar CCM between March 2010 and August 2021. Indications for surgery included (1) intracranial hemorrhage, (2) symptomatic superficially located lesion, and (3) asymptomatic CCM in non-eloquent area in case of strong parental preferences. Patients presenting with seizures were assessed using Engel Epilepsy Surgery Outcome Scale. RESULTS: Twenty-one patients were included. The predominant symptoms were seizures (57.1%), headaches (33.3%), and focal neurological deficits (23.8%). Patients were qualified for surgery due to symptomatic intracranial hemorrhage (47.6%), drug-resistant epilepsy (28.6%), and focal neurological deficits (9.5%). Three patients (14.3%) were asymptomatic. A gross total resection of CCM with the surrounding hemosiderin rim was achieved in all patients. The mean follow-up was 52 months. No patient experienced surgery-related complications. In all individuals with a preoperative first episode of seizures or focal neurological deficits, the symptoms subsided. All six patients with drug-resistant epilepsy improved to Engel classes I (67%) and II (33%). CONCLUSION: Surgical removal of symptomatic lobar CCMs in properly selected candidates remains a safe option. Parental preferences may be considered a sole qualifying criterion for asymptomatic lobar CCM excision.

Endoscopic ultrasound-guided lauromacrogol injection for treatment of colorectal cavernous hemangioma: Two case reports.

BACKGROUND: Colorectal cavernous hemangioma is a rare vascular malformation resulting in recurrent lower gastrointestinal hemorrhage, and can be misinterpreted as colitis. Surgical resection is currently the mainstay of treatment, with an emphasis on sphincter preservation. CASE SUMMARY: We present details of two young patients with a history of persistent hematochezia diagnosed with colorectal cavernous hemangioma by endoscopic ultrasound (EUS). Cavernous hemangioma was relieved by several EUS-guided lauromacrogol injections and the patients achieved favorable clinical prognosis. CONCLUSION: Multiple sequential EUS-guided injections of lauromacrogol is a safe, effective, cost-efficient, and minimally invasive alternative for colorectal cavernous hemangioma.

Clinical course of patients with conservatively managed cerebral cavernous malformations.

INTRODUCTION: There is uncertainty whether patients with a cerebral cavernous malformation (CCM) should undergo conservative or surgical treatment, resulting in practice variation among hospitals. Our objective was to report clinical outcomes of patients with primarily conservatively managed CCMs. PATIENTS AND METHODS: This single-center cohort study included consecutive adult CCM patients, diagnosed in 2000-2023, who underwent conservative management as primary treatment strategy. Data were extracted from medical records, and we systematically conducted telephone and questionnaire follow-up. Functional status was assessed on the modified Rankin Scale (mRS). RESULTS: Of 345 patients, we included 265 patients with a CCM (median age 46 years; 45% male). At baseline, 131 (49%) patients presented with symptomatic hemorrhage (SH), and 134 (51%) with other symptoms or asymptomatically. During 58 months (IQR 35-94) median follow-up, 51 (19%) patients experienced a SH, 33 (12%) a seizure, and 13 (5%) focal neurological deficits. Fourteen (5%) patients underwent intervention (surgery n = 11, radiosurgery n = 4). Presentation with SH was associated with higher annual bleeding rates (6.0% vs 1.5%, p < 0.001), and higher cumulative 5-/10-year bleeding risks (31%/41% vs 7%, p < 0.001). Brainstem CCM was associated with higher cumulative 5-/10-year bleeding risks (27%/38% vs 17%/21%, p = 0.038). Nineteen (7%) patients died; two (0.8%) directly attributable to CCM. Of 246 surviving patients, 205 (83%) completed the questionnaire. At follow-up, 172/224 (77%) patients were functionally independent (mRS score ⩽2). DISCUSSION AND CONCLUSION: The majority of conservatively managed CCM patients remained free of a SH during follow-up. Few patients required intervention, and death attributable to the CCM was rare. These data may help patient counseling and treatment decisions.

Transumbilical single-site laparoscopic treatment of small intestinal cavernous hemangioma in child: a case report.

Background: While hemangiomas are the most commonly occurring benign vascular tumors, their occurrence in the gastrointestinal system is rare. This case report presents a unique instance of small intestinal hemangioma in a pediatric patient. Case description: A 21-month-old girl was admitted to the hospital with a history of "recurrent blood in the stool for one year and anemia for five months." Upon evaluation at our facility, abdominal color ultrasound and enhanced CT scans revealed a protruding mass in the wall of the small intestine, leading to a preliminary diagnosis of small intestinal hemangioma. Subsequent single-site umbilical laparoscopic exploration identified a tumor measuring approximately 6cm×2.5cm×1.2cm on the jejunum wall. Consequently, segmental resection of the intestine was performed, and the postoperative pathological diagnosis confirmed cavernous hemangioma. Conclusion: Small intestinal hemangiomas, particularly in pediatric patients, are exceptionally rare and challenging to diagnose as the cause of gastrointestinal bleeding prior to surgery. Hence, small intestinal hemangiomas should be considered in such cases. Laparoscopic surgical resection emerges as the optimal approach for addressing small intestinal hemangiomas.

Primary retroperitoneal cavernous hemangioma: A case report and review of the literature.

Retroperitoneal cavernous hemangioma, a rare vascular tumor, has only 30 PubMed cases. Preoperative diagnostic criteria are unclear and often present asymptomatically until complications such as rupture or compression arise. We present a 73-year-old with chronic abdominal pain and a giant retroperitoneal tumor. Computed tomography (CT) and magnetic resonance imaging (MRI) revealed an irregular space-occupying mass in the retroperitoneum, suggesting a retroperitoneal chronic expanding hematoma. Total surgical resection confirmed the diagnosis as retroperitoneal cavernous hemangioma.

Extensive Spinal Hemangioma Associated With Cutaneous Nevus in the Same Metamere: An Unusual Case of Paraplegia in the Peripartum Period.

Cavernous hemangiomas occur most commonly in the cerebral hemispheres but can involve any part of the neuroaxis, including the spine. Very rare cases of spinal angiomas are associated with a skin lesion in the same metameric segment. This condition, known as segmental neurovascular syndrome or Cobb syndrome, was first described in 1915. We report a rare case of segmental neurovascular syndrome with extensive cervical and thoracic lumbar involvement expressed as peripartum spinal cord compression syndrome. A 37-year-old female with a cutaneous nevus from the C7 dermatome to the L3 dermatome experienced pelvic limb paralysis 48 hours after giving birth to a healthy newborn by cesarean section. Magnetic resonance imaging (MRI) revealed an enhancing extensive epidural mass from C7 to T7 and subsequently from T10 to L3. Histopathology confirmed a spinal cavernous hemangioma. Although rare, segmental neurovascular syndrome must be considered in patients with cutaneous angioma and radiculopathy or myelopathy. Early diagnosis can lead to curative surgical treatment and more favorable outcomes.

Subarachnoid Hemorrhage Caused by Supratentorial Cerebral Cavernous Malformation: A Case Report.

Cavernous malformations (CMs) are acquired vascular abnormalities of the central nervous system that are typically asymptomatic. Clinically symptomatic lesions may present with seizures, intracerebral hemorrhage, or focal neurological deficits. Very rarely, CMs have been described as the cause of subarachnoid hemorrhage. We report a case of a previously healthy 58-year-old man who presented with acute onset of severe headache associated with vomiting. Head computed tomography (CT) scan showed subarachnoid hemorrhage with intraventricular extension. Subsequent CT angiography (CTA) and digital subtraction angiography (DSA) studies showed no evidence of vascular abnormalities. The patient was initially managed conservatively but later required neurosurgical and radiological interventions due to a complicated hospital course and worsening clinical condition. During surgery, an incidental mass was found in the temporal lobe, and subsequent histopathological examination confirmed the diagnosis of cavernoma, which was likely the underlying cause of the subarachnoid hemorrhage. This report highlights the importance of considering CMs in the differential diagnoses of subarachnoid hemorrhage, especially in the absence of informative results from CTA and DSA studies. Timely detection and management of CMs may positively impact the clinical outcome, leading to reduced morbidity and mortality rates.

Transoral surgery with Thunderbeat© for hemangioma of the tongue base: A novel procedure. Case report.

INTRODUCTION: Hemangiomas are the most common benign tumors of the head-neck region in children and mainly affect the face, oral mucosa, lips, and tongue. The base of tongue is an extremely rare site of involvement. The incidence is higher in women and occur more frequently in infants and childhood. PRESENTATION OF CASE: We present a rare case of cavernous hemangioma of the base of tongue in a 70-year-old male patient surgically removed by Transoral Ultrasonic Surgery (TOUSS). 1-year follow up didn't show sign of recurrence. DISCUSSION: Hemangiomas are benign proliferations of endothelial cells common in the head and neck. The etiology is uncertain: an imbalance in angiogenesis related to substances such as vascular endothelial growth factor (VEGF) and basic fibroblast growth factor (BFGF) with uncontrolled proliferation of vascular elements is proposed. It can be asymptomatic or, when affecting the tongue, lead to difficulty swallowing, pain, bleeding and dyspnea. CONCLUSION: This case report aims to stress that hemangioma should be considered in differential diagnosis in case of richly vascularized tongue base lesion, also in adult population. It would like to highlight the role of transoral ultrasonic surgery (TOUSS), which is able to achieve the same advantages as TORS with lower costs and shorter learning curve.

Ovarian hemangioma: Differential diagnosis of ovarian cancer.

INTRODUCTION AND IMPORTANCE: Ovarian cavernous hemangioma is a rare benign vascular tumor primarily found as either an isolated ovarian mass or as diffuse abdominopelvic hemangiomatosis. Its discovery is often incidental, but symptomatic presentations can occur, including ovarian torsion, can occur without any specification. CASE PRESENTATION: We present the case of a 55-year-old menopausal woman with chronic pelvic pain. Initial diagnostic imaging and tumor markers suggested ovarian malignancy. Subsequent investigations, including ultrasound and MRI, revealed a vascularized adnexal mass with characteristics mimicking malignancy. Surgical exploration through laparoscopy unveiled an 8 cm multicystic solid-cystic lesion with histopathological analysis confirming a 9 cm cavernous hemangioma. CLINICAL DISCUSSION: Histologically, the lesion displayed features consistent with a benign vascular malformation. Literature review indicates variability in presentations and diagnostic challenges in distinguishing these lesions from malignant ovarian tumors. Treatment involved successful cystectomy via laparoscopic surgery, ensuring ovarian preservation so as not to increase the cardiovascular risk due to bilateral annexectomy. CONCLUSION: Ovarian cavernous hemangiomas pose diagnostic challenges due to their rarity and potential mimicry of malignancy. Timely recognition through imaging modalities, especially MRI, and confirmation via histopathological examination remain pivotal. Ovarian-sparing surgical interventions are recommended, particularly for younger patients without suspicious tumor features. Despite their rarity, awareness of these lesions is crucial for accurate diagnosis and appropriate management.

Rare Co-occurrence of Spinal Cord Hemorrhage from Radiation-induced Cavernous Hemangioma and Classical Hodgkin Lymphoma Post-transplant Lymphoproliferative Disorder.

Post-transplant lymphoproliferative disorders (PTLDs) are lymphoproliferative diseases that occur after solid organ transplantation or hematopoietic stem cell transplantation (HSCT). The development of PTLD is often associated with reactivation of Epstein-Barr virus (EBV). A 26-year-old woman with a history of HSCT and total-body irradiation developed spinal cord hemorrhage from a radiation-induced cavernous hemangioma (RICH) shortly after the development of classical Hodgkin lymphoma PTLD with EBV reactivation. Although little is known about the factors leading to hemorrhagic events from spinal cord RICH, we suspect that EBV reactivation may have been a factor contributing to the hemorrhage in the present case.

A Huge Hemangioma of the Urinary Bladder: A Case Report and Literature Review.

Cavernous hemangioma of the bladder is a benign and very rare vascular tumor. It can be isolated or part of a syndrome. Neither clinical symptoms nor imaging modalities lead to a definitive diagnosis as there are no specific findings on imaging studies or specific symptoms. Painless gross hematuria is the most common chief complaint and presentation and should never be underestimated. Here, we report a case of a large hemangioma of the urinary bladder in a young man who presented with recurrent recent episodes of painless gross hematuria and, surprisingly, with a previous episode of painless hematuria 15 years ago, which was treated successfully with partial cystectomy. We discuss the clinical features, evaluation, diagnosis, biopsy, management, the challenges encountered in treatment, and assert the value of tissue diagnosis and follow-up pattern and period. Due to the rarity of the tumor and lack of trials and evidence-based guidelines for management, treatment modalities vary and the risk for recurrence is questionable and not known.

Perimesencephalic subarachnoid hemorrhage as a rare delayed complication of radiation therapy in a patient with parotid basaloid squamous cell carcinoma.

In this case report, we address a rare entity of parotid cancer: basaloid squamous cell carcinoma, which was surgically unresectable and had thus far only been treated with radiation therapy. Following twenty years of continuous remission, our patient presented with an acute perimesencephalic subarachnoid hemorrhage. The cause of the acute perimesencephalic subarachnoid hemorrhage was a delayed complication of radiation therapy.

Two cavernous hemangiomas masquerading as single space occupying tumors in the orbital apex were removed under nasal endoscope: A case report.

INTRODUCTION AND IMPORTANCE: This report details the unusual presentation of two hidden cavernous hemangiomas in the orbital apex, initially appearing as one tumor. The rarity and diagnostic complexity of this case underscore the need for meticulous surgical exploration and verification in orbital apex tumors. CASE PRESENTATION: A physical examination of an elderly male with a three-year history of headaches revealed a space-occupying lesion in the left orbital apex. Imaging confirmed a tumor in the extraconical space above the optic nerve. Initial nasal endoscopy removed an orbital apex tumor, pathologically confirmed as a cavernous hemangioma. CLINICAL DISCUSSION: Postoperative examination revealed incomplete tumor removal, prompting a second surgery for full excision. This case underscores the diagnostic and management challenges of orbital apex tumors, especially when imaging indicates a single mass. The endoscopic transsphenoidal approach for cavernous hemangiomas in the medial orbital apex, as illustrated in this case, appears promising. CONCLUSION: Clinicians must be aware of the potential for multiple tumors in orbital apex cases, even if imaging does not explicitly reveal them. This case highlights the importance of thorough surgical exploration and illustrates the effectiveness of endoscopic methods in intricate orbital apex surgeries.

Extra-Axial Cavernous Angioma: A Case Report and Review of the Literature.

Cavernous angiomas (CAs) are benign vascular malformations predominantly seen in the brain parenchyma and therefore referred to as intra-axial. Extra-axial dural-based cavernous angiomas, on the other hand, are rare vascular lesions found outside of the brain parenchyma. They occur in the middle fossa and may be easily misdiagnosed as meningiomas due to their extra-axial location. In addition, CAs that are located outside the middle fossa, such as in the convexity, have a better prognosis since they are more surgically accessible. Surgical resection is the main treatment of choice in CAs. However, other options, such as embolization and radiotherapy, may also be considered therapeutic choices or additive treatment options. The pathogenesis of CA and the involvement of other factors (genetics or environmental factors) are still unknown and require further investigation. We are presenting a young man who presented for evaluation of seizure-like events without any family history of neurologic conditions. The physical examination was unremarkable except for a slightly antalgic gait. Imaging studies showed an extra-axial left tentorial mass suggestive of a meningioma, hemangiopericytoma, or other extra-axial lesions. The lesion was resected where its vascular nature was mentioned initially, and the histology proved the diagnosis of cavernous angioma. Here we give an overview of the known pathogenesis, causes, clinical features, and diagnostic and therapeutic options in CA. Better knowledge about CA, its causes, clinical features, and treatment options would help clinicians in early diagnosis and patient management.

Hepatocellular carcinoma­cavernous hemangioma collision tumor: A case report.

Collision tumors consisting of hepatocellular carcinoma (HCC) and cavernous hemangioma (CH) are rare and the clinicopathological characteristics or cause of the tumors remain unclear. The present study reports the case of a 71-year-old male patient who was admitted to Sunshine Union Hospital (Weifang, China) due to a liver mass found during a routine physical examination. computed tomography scans showed a main lesion of ~4.0×4.2×3.5 cm in segment IV of the patient's liver and a nodule of ~2.4×2.2×1.3 cm in the lower-left part of the lesion, which was clearly demarcated from the main lesion. The capsule of the lesion was found to be intact during the operation performed to remove the tumor. The final patient diagnosis was of a HCC-CH collision tumor based on pathology. The patient underwent follow-up for 6 months after surgery and no recurrence was observed.

Imaging Features and Misdiagnosis of Giant Cerebral Cavernous Malformations.

BACKGROUND: While cerebral cavernous malformations (CCMs) have been extensively described, few reports have described the imaging appearance of giant CCMs (GCCMs). OBJECTIVE: To describe the imaging characteristics of GCCMs and study the reasons for preoperative misdiagnosis. METHODS: We retrospectively analyzed the data of 12 patients (5 men, 7 women; mean age, 35.23 ± 12.64 years) with histopathologically confirmed GCCMs. Two radiologists analyzed the CT (n = 12) and MRI (n = 10) features: location, number, size, shape, boundary, signal intensity, and enhancement. RESULTS: The sellar region, cerebral hemisphere, skull bone, and ventricle were involved in 5, 4, 2, and 1 patients, respectively. Three tumors were irregularly shaped, while nine were oval. Eleven lesions showed slightly high- and/or high-density on CT; 1 lesion appeared as a low-density cyst. Calcifications were found in 11 lesions. Four tumors showed uniform hypointensity on T1-weighted imaging (T1WI) and hyperintense signals on T2-weighted imaging (T2WI). Six tumors showed mixed low-, equal-, and high-intensity signals on T1WI and T2WI. Noticeable contrast enhancement and gradual strengthening were noted on T1WI. Ten lesions showed hemorrhage and hemosiderin deposition. The GCCMs were wrongly diagnosed as cartilage-derived tumors/ meningioma (3 patients); tumor and hematoma (2 patients each); and pituitary tumor/ meningioma, chondroma, chordoma, ependymoma, and macroadenoma (1 patient each). CONCLUSIONS: GCCMs present as an oval mass with slightly high- and/or high-density calcifications on CT and show hemorrhage and hemosiderin accumulation on MRI. Therefore, slightly high- and/or high-density calcification and hemosiderin accumulation are critical clinical characteristics of GCCMs.