• Sufyan Ibrahim, Salomon Cohen-Cohen, Karim Rizwan Nathani, Amir Nazem, Aditya Raghunathan, Jay J Pillai, and Mohamad Bydon.
• Department of Neurologic Surgery, Neuro-Informatics Laboratory, Mayo Clinic, Rochester, Minnesota, USA; Department of Neurologic Surgery, Mayo Clinic, Rochester, Minnesota, USA.
• World Neurosurg. 2024 May 1; 185: e951e962e951-e962.

BackgroundCapillary 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.MethodsMedical 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.ResultsWe 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.ConclusionsPurely 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.Copyright © 2024 Elsevier Inc. All rights reserved.

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