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.

Isolated spinal artery aneurysm: etiology, clinical characteristics, and outcomes.

OBJECTIVE: Isolated spinal aneurysms (ISAs) are rare causes of subarachnoid hemorrhage (SAH), which encompass a highly heterogeneous group of clinical entities with multifarious pathogeneses, clinical characteristics, and treatment strategies. Therefore, knowledge about the ISAs remains inadequate. In this study, the authors present a comprehensive analysis of clinical data associated with ISAs at their institutions to enhance the understanding of this disease. METHODS: Patients with ISAs confirmed by spinal angiography or surgery at the authors' institutions between 2015 and 2022 were included. Data regarding clinical presentation, lesion location, aneurysm morphology, comorbidities, treatment results, and clinical outcomes were reviewed. RESULTS: Seven patients with ISAs were included in the study. Among them, 4 patients (57.1%) experienced severe headache, and 3 patients (42.9%) reported sudden-onset back pain. Additionally, lower-extremity weakness and urinary retention were observed in 2 of these patients (28.6%). Four of the aneurysms exhibited fusiform morphology, whereas the remaining were saccular. All saccular aneurysms in this series were attributed to hemodynamic factors. Conservative treatment was administered to 3 patients, 2 of whom underwent follow-up digital subtraction angiography, which showed spontaneous occlusion of both aneurysms. Four patients ultimately underwent invasive treatments, including 2 who underwent microsurgery and 2 who received endovascular embolization. One patient died of recurrent SAH, while the remaining 6 patients had a favorable prognosis at the latest follow-up assessment. CONCLUSIONS: The morphology of aneurysms may be associated with their etiology. Saccular ISAs are usually caused by pressure due to abnormally increased blood flow, whereas fusiform lesions may be more likely to be secondary to vessel wall damage. The authors found that a saccular spinal aneurysm in young patients with a significant dilated parent artery may be a vestige of spinal cord arteriovenous shunts. ISAs can be managed by surgical, endovascular, or conservative procedures, and the clinical outcome is generally favorable. However, the heterogeneous nature of the disease necessitates personalized treatment decision-making based on specific clinical features of each patient.

SARS-CoV-2 infection and spontaneous spinal hemorrhage: a systematic review.

The neurological manifestations of severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection, including spontaneous spinal hemorrhage (SSH), are diverse. SSH is a detrimental neurosurgical event requiring immediate medical attention. We aimed to investigate the association between SARS-CoV-2 and SSH and delineate a rational clinical approach. The authors searched PubMed, Scopus, Web of Science, and Google Scholar for studies published up to January 25, 2023, on SSH and SARS-CoV-2 infection. For each dataset, the authors performed pooled estimates examining three outcomes of interest: (1) early post-intervention neurological status, (2) mortality, and (3) post-intervention neurological rehabilitation outcomes. After reviewing 1341 results, seven datasets were identified for the final analysis. Fifty-seven percent of patients were females. Twenty-eight percent of the patients experienced severe systemic infection. The mean interval between the SARS-CoV-2 infection and neurological presentation was 18 days. Pain and sensorimotor deficits were the most common (57%). Spinal epidural hematoma (EDH) was the most common presentation (71.4%). Three patients were treated conservatively, while 4 received neurosurgical intervention. Pain and sensorimotor deficits had the best treatment response (100%), while the sphincter had the worst response (0%). Long-term follow-up showed that 71% of patients had good recovery. SARS-CoV-2-associated SSH is a rare complication of infection, with an often insidious presentation that requires high clinical suspicion. Patients with SARS-CoV-2 infection and new neurological symptoms or disproportionate neck or back pain require a neuroaxis evaluation. Neurosurgical intervention and conservative management are both viable options to treat SSH following COVID-19. Still, a homogenous approach to the treatment paradigm of SSH cannot be obtained, but lesions with space-occupying effects are suitable for neurosurgical evacuation-decompression while more indolent lesions could be treated conservatively. These options should be tailored individually until larger studies provide a consensus.

A Zebra Diagnosis: A Case of Spontaneous Hematomyelia Associated With COVID-19.

Spontaneous non-traumatic spinal hematomyelia, characterized by intramedullary spinal hematoma, is a rare neurological emergency. Bleeding arteriovenous malformation, coagulopathies, and neoplasms are reported causes of this rare diagnosis. The authors present a case of a previously healthy man who presented with acute paraplegia and was found to have a spontaneous hematomyelia in association with covid infection. He underwent laminectomy and hematoma evacuation but did not recover any neurological function.

A Spinal Cavernous Malformation in Multiple Cerebral Cavernous Venous Malformations Syndrome.

Teaching Point: In patients with familial multiple cavernous malformation syndrome with acute focal neurological deficit, a symptomatic spinal cavernous malformation must be included in the differential diagnosis.

Acute Spinal Hemorrhage from a Cauda Equina Arteriovenous Fistula Fed by the Proximal Radicular Artery.

Spinal cauda equina arteriovenous fistulas (CEAVFs) and spinal hemorrhage from spinal AVF are relatively unusual. To our knowledge, such a case of CEAVF presenting with hemorrhage has not been reported. Here, we describe such a rare case of CEAVF. Spinal magnetic resonance imaging and angiography revealed a CEAVF at the L2 level, fed by the proximal radicular artery and with 2 associated venous varices. Considering the feeder from the proximal radicular artery and the presence of acute hemorrhage, direct surgery was performed. Intraoperatively, spinal hematoma and the fistula were identified at the L2 level. The fistula was clipped and divided successfully. The postoperative course was uneventful. CEAVF presenting with spinal hemorrhage is extremely rare. This case demonstrated the angioarchitecture features of CEAVF. The clinicians should be aware of the possibility of this condition and the importance of treatment selection.

Spontaneous Convexity Subarachnoid Hemorrhage Caused by Internal Carotid Occlusion: Radiological Features

A 79-year-old patient was admitted to the emergency room with transitory monoparesis in the left hand and dysphasia. The brain computed tomography (CT) and magnetic resonance imaging (MRI) showed a spontaneous right convexity subarachnoid hemorrhage (cSAH). Digital subtraction angiography (DSA) confirmed an asymptomatic occlusion of the right internal carotid artery (ICA) . Cases related to stenosis have already been described, but there is no similar report of a case related to occlusion, even though the pathophysiology of both entities is similar. Atraumatic SAH has been associated with intracranial and extracranial artery stenosis.

[Primary angiitis of the central nervous system with cerebral infarction and spinal hemorrhage].

A 61-year-old woman presented with acute intense lower back pain and weakness in her left leg. She also presented with throbbing headache on the same day. On admission, muscle weakness in her left leg, lower left quadrantanopia and left lower extremity deep sensory disturbance were observed. Laboratory data showed no coagulopathy and autoimmune antibody was negative. Cerebrospinal fluid examination showed bloody and inflammatory findings. Brain MRI revealed cerebral infarction with multiple intracranial arterial stenosis and convexal subarachnoid hemorrhage. Spinal MRI revealed spinal hemorrhage in the cervical, thoracic, and part of the lumbar spine. Because these lesions occurred simultaneously, we made a diagnosis of vasculitis. After high dose corticosteroids therapy was undertaken, the multiple arterial stenosis improved. Primary angiitis of the central nervous system is sometimes difficult to distinguish from reversible cerebral vasoconstriction syndrome in its initial stage; although symptoms, examination findings and treatment differ in both.

Sudden spinal hemorrhage in a pediatric case with total body irradiation-induced cavernous hemangioma.

Compared to cerebral radiation-induced cavernous hemangiomas (RICHs), little is known about intraspinal RICHs. A 13-year-old male suddenly developed symptomatic spinal hemorrhage eight years after hematopoietic stem cell transplantation using a total body irradiation (TBI) based myeloablative regimen. A solitary small hemangioma was detected on follow-up T2 star weighted magnetic resonance imaging of the spine. His neurological symptoms gradually improved with supportive treatment and rehabilitation, although he experienced rebleeding 2 years later. Intraspinal RICH is very rare but should be recognized as a possible late adverse effect in pediatric patients who received TBI.

A pediatric patient of hemorrhagic acute transverse myelitis.

An 11-year-old boy presented with progressive leg hypesthesia but no history of trauma. Dysuria and constipation appeared subsequent to gait difficulty. He was admitted 8days after onset. Spinal magnetic resonance imaging (MRI) revealed longitudinal hyperintensity with cord swelling and hypointensity on T2-weighted images, suggesting severe inflammation and microbleeding change, respectively. Gadolinium contrast-enhanced MRI demonstrated mild enhancement in the lesions. Platelet count and coagulation findings were normal, and cerebrospinal fluid analysis showed no pleocytosis. He was diagnosed with idiopathic acute transverse myelitis (ATM), and intravenous methylprednisolone pulse therapy and plasmapheresis were initiated. On day 14, motor dysfunction aggravated suddenly, accompanied by expanding hemorrhagic lesions. Thereafter, administration of intravenous immunoglobulin, repeated intravenous methylprednisolone pulse therapy and prednisolone for one month resulted in complete recovery four months later. Both anti-aquaporin-4 and anti-myelin oligodendrocyte glycoprotein antibodies were negative. We presented the first pediatric case showing hemorrhagic spinal lesions in the clinical course of ATM. This severe complication should be recognized in the management of ATM.

Iatrogenic spinal hematoma as a complication of lumbar puncture: What is the risk and best management plan?

BACKGROUND: Lumbar puncture (LP) rarely results in complications such as spinal hematomas. However, it remains unclear if certain variables increase likelihood of these events, or if surgical intervention improves outcome. METHODS: In addition to two clinical vignettes, we evaluated the post-1974 literature for cases of spinal hematoma and subsequent intervention. Based on our compilation of data, we evaluated outcome relative to numerous distinct variables. RESULTS: Based on 35 LP-related spinal hematoma cases in the post-1974 literature and our encounters, we found 28.6% of patients presenting with preexisting coagulopathy had poor outcomes regardless of intervention, relative to 14.3% of patients without coagulopathy; a highly significant difference (P = 0.02). Once diagnosed, 21 patients were treated surgically and 14 nonsurgically. Of the 60% surgical patients, 57.1% had good outcomes, and 42.9% had poor outcomes within 12 months. Of 40% nonsurgical patients, 57.1% had good outcomes and 42.9% had poor outcomes. Results in these groups were not statistically different. CONCLUSIONS: We found a significant correlation between preexisting coagulopathy and poor neurological outcome irrespective of intervention. However, outcomes for these patients may be confounded by comorbidities including underlying conditions contributing to their coagulopathy. No significant correlation between type of surgical intervention and good outcome was found, possibly attributable to the paucity of details in existing case reports and the difficulty defining the degree of spinal cord compromise from a given lesion. Despite our findings, emergent neurosurgical intervention may be beneficial for the management of complications such as cauda equina syndrome secondary to intrathecal spinal hematoma.

Intraspinal hemorrhage in spontaneous intracranial hypotension: link to superficial siderosis? Report of 2 cases.

Spontaneous intracranial hypotension due to a spinal CSF leak has become a well-recognized cause of headaches, but such spinal CSF leaks also are found in approximately half of patients with superficial siderosis of the CNS. It has been hypothesized that friable vessels at the site of the spinal CSF leak are the likely source of chronic bleeding in these patients, but such an intraspinal hemorrhage has never been visualized. The authors report on 2 patients with spontaneous intracranial hypotension and intraspinal hemorrhage, offering support for this hypothesis. A 33-year-old man and a 62-year-old woman with spontaneous intracranial hypotension were found to have a hemorrhage within the ventral spinal CSF collection and within the thecal sac, respectively. Treatment consisted of microsurgical repair of a ventral dural tear in the first patient and epidural blood patching in the second patient. The authors suggest that spontaneous intracranial hypotension should be included in the differential diagnosis of spontaneous intraspinal hemorrhage, and that the intraspinal hemorrhage can account for the finding of superficial siderosis when the CSF leak remains untreated.

Hemorrhagic thoracic schwannoma presenting with intradural hematoma and acute paraplegia after spinal manipulation therapy.

Hemorrhagic conversion of spinal schwannomas represents a rare occurrence; also rare is the development of a spinal intradural hematoma after spinal manipulation therapy. We report a unique presentation of paraplegia in a patient who underwent spinal manipulation therapy and was found to have a hemorrhagic thoracic schwannoma at time of surgery in the setting of anti-platelet therapy use. In patients with spinal schwannomas, tumor hemorrhage is a rare occasion, which can be considered in the setting of additive effects of spinal manipulation therapy and antiplatelet therapy.

Concomitant Intraspinal and Retroperitoneal Hemorrhage Caused by an Aneurysm on the Celiac Artery: A Case Report.

Spontaneous spinal hemorrhage is a rare condition. We present a case in which the diagnosis was complicated by a concomitant intra-abdominal hemorrhage. The patient, taking coumarins, presented with acute back pain and abdominal pain and progressive paresis of the lower limbs. Computed tomography angiography of the abdomen showed an intra-abdominal hemorrhage and an aneurysm of the celiac trunk. MR (magnetic resonance) imaging of the spine revealed a combined subdural and epidural hemorrhage from C1 to L1. Both sites were treated conservatively. After 6 months the patient regained strength in both legs with some persistent loss of strength in the left leg. Follow-up MR imaging showed complete resolution of the spinal hemorrhage. The celiac artery aneurysm was treated conservatively. We suggest that the rupture of the celiac artery aneurysm caused increased intra-abdominal pressure leading to spinal hemorrhage. Emergency staff should be aware of the possibility of two rare but concomitant conditions.

Spontaneous resolution of nontraumatic acute spinal subdural hematoma.

Spinal subdural hematoma (SSDH) is an extremely uncommon condition. Causative factors include trauma, anticoagulant drug administration, hemostatic disorders, and vascular disorders such as arteriovenous malformations and lumbar punctures. Of SSDH cases, those that do not have any traumatic event can be considered cases of nontraumatic acute spinal subdural hematoma, which is known to have diverse clinical progress. Treatment typically consists of surgical decompression and cases in which the condition is relieved with conservative treatment are rarely reported. We report two nontraumatic acute spinal subdural hematoma patients who were successfully treated without surgery.

Spontaneous Resolution of Nontraumatic Acute Spinal Subdural Hematoma

Spinal subdural hematoma (SSDH) is an extremely uncommon condition. Causative factors include trauma, anticoagulant drug administration, hemostatic disorders, and vascular disorders such as arteriovenous malformations and lumbar punctures. Of SSDH cases, those that do not have any traumatic event can be considered cases of nontraumatic acute spinal subdural hematoma, which is known to have diverse clinical progress. Treatment typically consists of surgical decompression and cases in which the condition is relieved with conservative treatment are rarely reported. We report two nontraumatic acute spinal subdural hematoma patients who were successfully treated without surgery.