Capillary Hemangioma of the Spinal Cord: Case Report and Systematic Review of Literature.

BACKGROUND: Capillary hemangiomas are rare vascular lesions that rarely affect the central nervous system. When they present within the spinal canal, they are typically confined intradurally, with intramedullary extension rare. We present a rare case of spinal intramedullary capillary hemangioma, with a systematic review of the literature. METHODS: Medical records and imaging data were retrospectively reviewed using the health record software EPIC (Verona, Wisconsin, USA) and the radiology management software system RIS/PACS (Radiology Information System/Picture Archiving and Communication System; QREADS). The report was written in accordance with the CARE (case reports) guidelines. We also performed a systematic review of the literature on all cases of intramedullary spinal capillary hemangiomas in accordance with PRISMA (preferred reporting items for systematic reviews and meta-analyses) guidelines. RESULTS: We report a case of a 54-year-old man who presented with progressive paraplegia and sensory deficits in the lower extremities. Spinal magnetic resonance imaging showed an intramedullary enhancing lesion centered at T11 with associated spinal cord compression. He underwent thoracic laminectomy and gross total resection of the lesion without complications and subsequent improvement on his neurological examination. Histological examination showed findings consistent with a capillary hemangioma. The literature review also documented 21 studies with a combined total of 38 cases of intramedullary spinal capillary hemangioma. CONCLUSIONS: Purely intramedullary capillary hemangiomas are unusual spinal lesions with only a few cases reported in the literature. These should be considered in the differential diagnosis of intramedullary tumors. Surgical management remains the first line of treatment for symptomatic patients.

Intramedullary spinal capillary hemangioma with secondary neurulation defect in children.

Intramedullary spinal capillary hemangioma is a rare occurrence in pediatric patients, and only limited cases have been reported. This study presents the first two cases of spinal capillary hemangioma co-present with retained medullary cord and one case of spinal capillary hemangioma with lumbosacral lipomatous malformation. Previous literature on ten patients with this pathology was reviewed. We speculated pathogenesis, imaging features, and histopathologic findings of the disease.

Documented growth of an intracranial capillary hemangioma: A case report.

Intracranial capillary hemangiomas in adults are rare, and diagnosis can be challenging. Hemangiomas, in general (and particularly in the skin), are more often noted in the pediatric population. Due to the lack of imaging undertaken in the presymptomatic phase, the literature provides few clues on the rate of growth of these unusual tumors. Therefore, we report a case of a 64-year-old man with a medical history of Lyme disease who presented with exhaustion and confusion. Imaging demonstrated an intra-axial lesion with vascularity in the posterior right temporal lobe, raising the possibility of a glioma. Imaging two years prior revealed a very small lesion in the same location. The patient underwent a craniectomy, total resection of the lesion was completed, and his symptoms of confusion resolved. Biopsy revealed a capillary hemangioma composed of small vascular channels lined by endothelial cells and pericytes without smooth muscle. Features of glioma, vascular neoplasms or neuroborreliosis (cerebral Lyme disease) were not identified. Our case documents the growth over two years of a rare intracranial capillary hemangioma in an older adult male.

Spontaneous hemorrhage of spinal epidural capillary hemangioma resulting in hyperacute neurologic deficit: A case report.

INTRODUCTION: Spinal epidural capillary hemangioma is a very rare variety of tumors, usually with a predilection for the thoracic spine. CASE PRESENTATION: A 16-year-old female complained of hyperacute neurologic deficit progressed within hour, which presented by acute paraplegia, and loss of all sensations from her lower limbs up to her breasts. Neurologic exam revealed paralysis of lower limbs (0/5 on both legs) with a flaccid tone, absence of reflexes, weakness of the trunk with sensory level T4, bilateral flexion of plantar reflexes, and loss of sphincters' controls. Emergent magnetic resonance imaging showed a dumbbell-shaped epidural mass in the posterior aspect of the spinal canal at the T1-T2 level, measuring approximately 1.1 × 4.5 × 1.5 cm in size. The lesion was isointense on T1-weighted, hyperintense on T2-weighted, and a little enhancement after gadolinium administration. The surgery was obtained nearly 16 hours after paralysis, which eradicated the lesion with good hemostasis. Histological examination showed a well-organized vascular tissue that haphazardly arranged and confirmed the diagnosis of capillary hemangioma. Neurological improvement was quickly observed within days after surgery and further complete recovery was achieved 2 months after discharge. CONCLUSION: We report an extremely rare case of spinal epidural capillary hemangioma, where acute spontaneous hemorrhage in the lesion resulted in the hyperacute neurologic deficit within an hour. Since these are benign lesions, the immediate surgical intervention results in a very favorable prognosis and is considered the treatment of choice. Also, this case highlighted and rose the question of a better neurologic improvement in younger age patients with spinal cord injury.

Capillary Haemangioma of Gingiva - A Rare Lesion.

Capillary Haemangioma is a benign vascular tumour characterized by proliferation of blood vessels with a very striking similarity to pyogenic granuloma with a predilection to occur on the gingiva. Though the head and neck are the most common region of occurrence, but is considered to be rare occurring on the gingiva, intra-orally. The lesion clinically mimicked pyogenic granuloma but was histopathologically diagnosed as capillary haemangioma. It was surgically excised and followed up for 6 months without any recurrence.

Perinatal Presentation of a Congenital Intramedullary Capillary Hemangioma in a Neonate Born with Hydrocephalus and Paraplegia.

Spinal intramedullary capillary hemangioma is a rare condition. Although most spinal/spinal cord hemangiomas are inborn in origin, perinatal presentation is uncommon. We present a neonate with an intramedullary hemangioma, born with communicating hydrocephalus and complete paraplegia. Spinal imagining showed an intradural mass with hemorrhagic foci, a low-lying conus medullaris, and scalloping of dorsal elements. Ventriculoperitoneal shunting was performed in an emergent setting, with cerebrospinal fluid showing a brownish appearance with high viscosity which implied a possible old hemorrhage. Tissue sample was taken from the spinal lesion in an elective setting. Histopathological examination revealed capillary lobules and extralobular large vessels resembling abnormal veins. The mentioned clues suggested that this congenital hemangioma had become symptomatic from the prenatal period. Though it is a are event, it should be kept in mind while evaluating a neonate with communicating hydrocephalus and weakness of extremities with or without cutaneous hemangiomatous stigmata.

Management of Capillary Hemangioma of the Sphenoid Sinus.

Background and objectives: Capillary hemangiomas are rare, benign vascular tumors that mainly affect the skin and soft tissue, with scarce appearance within the nasal cavities and paranasal sinuses. Materials and methods: We present a case report of capillary hemangioma of the sphenoid sinus and a review of the literature in the last ten years. Results: Clinical and endoscopic examination of the nose, radiologic assessment and particular histologic features contribute to the correct diagnosis of capillary hemangioma of the nose and paranasal sinuses. Conclusions: Transnasal endoscopic resection of capillary hemangioma located in the nose and paranasal sinuses is a valuable treatment method with good outcomes.

Characteristics, Natural Course and Treatment of Intramuscular Capillary-type Haemangioma: A Systematic Literature Review.

Intramuscular capillary-type haemangiomas (ICTH) are rare vascular anomalies that can easily be misdiagnosed as other entities. A systematic review was performed of all cases of ICTH in the literature since its first description in 1972. An adjudication committee reviewed cases to include only ICTHs. Among 1,143 reports screened, 43 were included, involving 75 patients. The most frequent differential diagnosis was intramuscular venous malformations. The mean age of patients at diagnosis was 21.2 years. ICTH was mainly described as a gradually increasing mass (81.8%), painless (73.9%), that could occur anywhere in the body but most frequently on the head and neck (44.0%). Magnetic resonance imaging (MRI) was mainly used for diagnosis (69.1%) and displayed specific features. The most frequent treatment was complete surgical removal (73.9%), which could be preceded by embolization, and led to complete remission without recurrence in all but 1 case.

Spinal epidural capillary hemangioma: case report and review of the literature.

BACKGROUND: Almost all of the epidural hemangiomas reported are cavernous hemangiomas. Purely extradural spinal capillary hemangiomas are very rare. Capillary hemangiomas are hamartomatous malformations that result from proliferations of vascular endothelial cells. Only ten cases have been reported in the English literature, treated with surgical excision. CASE DESCRIPTION: A case of a dorsal extradural spinal capillary hemangioma is described. A total surgical removal has been performed after spinal angiography and embolization. Complete surgical removal should always be the goal in these lesions. Embolization did not show to reduce bleeding during the surgical procedure in this case.

Multicenter retrospective review of pulsed dye laser in nonulcerated infantile hemangioma.

BACKGROUND/OBJECTIVES: We sought to describe the experience among members of the Hemangioma Investigator Group with pulsed dye laser (PDL) in the treatment of nonulcerated infantile hemangioma (IH) in pediatric patients in the pre- and post-beta-blocker era. METHODS: A multicenter retrospective cohort study was conducted in patients with nonulcerated IH treated with laser therapy. Patient demographics, IH characteristics, indications for/timing of laser therapy, as well as laser parameters were collected. Responses to laser therapy were evaluated using a visual analog scale (VAS). RESULTS: One hundred and seventeen patients with IH were treated with PDL. 18/117 (15.4%) had early intervention (defined as <12 months of life), and 99/117 (84.6%) had late intervention (≥12 months of life). In the late intervention group, 73.7% (73/99) had additional medical management of their IH. The mean age at PDL initiation for the late intervention group was 46.7 ± 35.3 months of life (range 12-172 months) with total number of treatments to maximal clearing of 4.2 ± 2.8 (range 1-17). Those who received propranolol prior to PDL received fewer sessions (1.1 fewer sessions, approaching significance [p = .056]).     On the VAS, there was a mean 85% overall improvement compared to baseline (range 18%-100%), with most improvement noted in erythema and/or telangiectasias. The incidence of adverse effects was 6/99 (6.1%). CONCLUSIONS: PDL is a useful tool in the treatment of IH, with notable improvement of telangiectasia and erythema and low risk of complications.   PDL is often introduced after the maximal proliferative phase.

Masseteric giant capillary haemangioma: a bulky mass treated with scarless and minimally invasive surgery.

In this article, the authors report their management with minimally invasive surgery and embolisation of a bulky intramuscular capillary haemangioma. Masseteric capillary haemangioma is a rare tumour in adults: in cases of large-sized tumours we suggest a multidisciplinary approach in choosing the best treatment and minimally invasive, scarless surgery.

Vitrectomy-Assisted Excision in the Treatment of Juxtapapillary Retinal Capillary Hemangioma.

Juxtapapillary retinal capillary hemangiomas are sight-threatening hamartomas located on or adjacent to the optic nerve. Nonsurgical approaches including laser photocoagulation and cryotherapy have been shown to be effective to reduce exudation in peripheral hemangiomas. However, in juxtapapillary hemangiomas, the functional outcomes are limited due to associated potential damage of the retinal nerve fiber layer. We present an 18-year-old female patient with von Hippel-Lindau (VHL) disease who presented with a juxtapapillary retinal capillary hemangioma associated with a tractional epiretinal membrane (ERM) and secondary macular hole. After vitrectomy-assisted excision of the lesion and inner limiting membrane (ILM) peeling around the macular hole, visual acuity and macular anatomy were recovered at 10 months of follow-up. [Ophthalmic Surg Lasers Imaging Retina 2022;53:570-573.].

Fetal cardiac capillary hemangioma resulting in tachyarrhythmia.

We present the case of a fetus with cardiac capillary hemangioma in the right atrial cavity. The tumor showed dramatic growth between the 28th and 32nd week of gestation and resulted in tachyarrhythmia. The patient was born at the 33 weeks of gestation weighing 2430 g via urgent cesarean section because the rapidly growing cardiac tumor caused incessant tachyarrhythmia, pericardial effusion, and fetal circulatory incompetence. Coronary angiography revealed that the right coronary artery drained into the tumor. Due to hemodynamic deterioration, the patient underwent subtotal resection of the tumor on the 2nd day after birth. Histopathological examination revealed an undifferentiated capillary hemangioma. The patient was discharged at the age of 86 days, as the tachyarrhythmia and hemodynamic incompetence had subsided; however, bradycardia and intermittent atrioventricular conduction disturbance gradually developed. Capillary hemangioma, a rare primary cardiac space-occupying tumor in children, can invade the conduction system.

Intradural extramedullary capillary hemangioma with intramedullary component: A case series.

RATIONALE: Capillary hemangioma in the spinal cord is an exceedingly rare entity that is liable to be misdiagnosed. To summarize the clinical presentation, radiological characteristics, and pathological features of capillary hemangioma in the spinal cord and to share our experience for the diagnosis and treatment of intradural extramedullary capillary hemangioma. PATIENT CONCERNS: Three patients underwent surgical treatment at our hospital between January 2020 and August 2020. All patients were male[median age at presentation: 57 years (range: 56-60)]. Two patients were experiencing pain and numbness in the lower back, and 1 patient was experiencing numbness and weakness in the left lower limb. The duration of symptoms ranged from 1 to 5 months. DIAGNOSIS: All patients were diagnosed with spinal cord capillary hemangioma after treatment. All lesions were in an intradural extramedullary location and involved spinal cord components. Two patients had lesions in thoracic segments (T8, Th9-10), and 1 patient had a lesion in lumbar segment (L1). INTERVENTIONS: All patients underwent microscopic laminectomy and complete resection of the extramedullary and intramedullary components of the spinal cord capillary hemangiomas. OUTCOMES: Postoperatively, all patients experienced transient numbness and pain in the lower limbs, which gradually decreased over time. None of the patients experienced bleeding, severe numbness or pain, or recurrence of symptoms at 3-month follow-up. CONCLUSION: Intradural extramedullary capillary hemangioma has unique morphological characteristics. Gross-total resection of the extramedullary and intramedullary components of spinal cord capillary hemangioma is recommended for patients with symptoms of spinal cord compression. Careful preoperative imaging and intraoperative decision-making may help avoid residual lesions or reoperation.

Resection of an Intradural Extramedullary Capillary Hemangioma in the Lumbar Spine.

BACKGROUND AND IMPORTANCE: Capillary hemangiomas are space-occupying lesions that rarely affect the central nervous system. When they present within the spinal canal, they can cause insidious symptoms and threaten neurological function. In this study, we present a case of an intradural extramedullary capillary hemangioma of the lumbar spine, discuss our management strategy, and review the current literature. For the first time for this diagnosis, we also provide an operative video. CLINICAL PRESENTATION: The patient is a previously healthy 40-year-old man who presented with complaints of progressive low back and leg pain, numbness, and intermittent subjective urinary incontinence. MRI revealed a discrete, homogenously enhancing intradural extramedullary lesion at L4. This lesion was resected by performing an L4 laminoplasty, which entails en bloc removal of the L4 lamina and then securing it back into place once the intradural resection and dural closure are completed. Histological analysis revealed a diagnosis of capillary hemangioma. The patient had full resolution of his symptoms postoperatively. DISCUSSION: Definitive management of spinal capillary hemangiomas involves gross total resection and can be accomplished with laminoplasty. Because these benign tumors can be adherent to adjacent structures, intraoperative neuromonitoring is helpful adjunct to preserve neurological function for a good outcome. CONCLUSION: Capillary hemangiomas rarely affect the spine but should be considered on the list of differential diagnoses of intradural lesions.

Spinal epidural capillary hemangioma: A systematic literature review and an illustrative case.

BACKGROUND: Spinal hemangiomas are rare vascular malformations resulting from proliferation of vascular endothelial cells. The cavernous form is the most common and represents 5-12% of spinal vascular malformations, while the capillary form is rare. CASE DESCRIPTION: A 56-year-old patient with no past medical history presented with progressive spinal cord compression symptoms localizing to the T10 level with MRC grade 4 proximal paraparesis. Preoperative MRI demonstrated a well-delineated, dumbbell-shaped, epidural lesion, without bony involvement, resulting in spinal cord compression at the T7 and T8 levels. The patient underwent gross total surgical resection of the lesion. At the one month follow up, the patient's strength improved to MRC grade 5, and sensation had fully returned. The histopathological diagnosis was a capillary hemangioma. Exclusively epidural capillary hemangiomas are extremely rare with only 26 cases reported in the literature. They are mainly located at the thoracic level (T4-T6). The MRI features include a well-circumscribed mass, hyperintense on T2-weighted sequence in 92% of cases, isointense on T1-weighted sequence in 88% of cases, and homogeneous contrast enhancement in 100% of cases. No tumor recurrence has been observed after gross total surgical removal. CONCLUSIONS: When evaluating progressive spinal cord compression by a purely epidural spinal lesion, the differential diagnosis should include capillary hemangioma, in addition to schwannoma, meningioma, and lymphoma. Early and complete surgical removal is the first line treatment.