Uncommon Manifestation of a Common Disease: Neurogenic Claudication Secondary to Epidural Spillage of Vertebral Hemangioma

Sir, A 64-year-old male presented with a history of neurogenic claudication. Magnetic resonance imaging (MRI) of the lumbar spine demonstrated a lumbar 4 (L4) vertebral hemangioma (VH) with extraosseous spread and significant epidural involvement, causing severe lumbar stenosis from lumbar 3 to 5 [Figure 1] with no evidence of vertebral body collapse. Computed tomography (CT) of the lumbar spine lesions typical of VH with a polka dot appearance [Figure 2]. Symptoms of neurogenic claudication paired with radiographic evidence of involvement of the posterior spinal elements, paravertebral and epidural extension, and progressive growth of the lesion led to diagnosis of atypical VH (AVH). The patient underwent spinal digital subtraction angiography with particle embolization followed by L3-5 laminectomy, facetectomy and epidural tumor resection with an L2 to S1 instrumented fusion. No attempt to remove the tumor involving the vertebral body was made as there was no evidence of any vertebral collapse or fracture. Given the absence of any fracture or vertebral collapse, vertebroplasty was not performed in this case. Intra-operatively, the intracanalicular tumor appeared reddish-brown, very friable, and bled extensively despite preoperative embolization. Histopathology confirmed the diagnosis of hemangioma. Postoperative MRI of the lumbar spine showed adequate decompression [Figure 3]. No adjuvant treatment was recommended secondary to the benign nature of the tumor. At his last follow up at 6 years, the patient demonstrated an absence of preoperative symptoms and no evidence of progression of disease.Figure 1: Sagittal T2W (left), T1W (Center) and axial T2W (Right) MRI showing a typical hemangioma involving the L4 vertebral body but with significant soft tissue extraosseous component hyper intense on T2W MRI, and hypo intense on T1W MRI and causing severe spinal stenosis and nerve root compressionFigure 2: Sagittal (left) and axial (right) CT of the lumbar spine shows L4 vertebral lesion with a polka dot appearance typical of vertebral hemangiomaFigure 3: Sagittal post contrast MRI done at 2 years after surgery showing well decompressed spinal canal with no evidence of any significant neural compressionAVH are a rare subtype of VH.[1-3] Factors that influence treatment include age, severity of pain, neurological status, predominantly bony or extraosseous extension, location of the lesion, presence or absence of fracture, degree of spinal deformity/instability, and degree of epidural extension. Ethanol embolization and vertebroplasty are potential treatment options in cases of mild pain, minimal radiculopathy, and no spinal instability.[4] Presence of spinal instability or neurological deficits strongly favor surgical intervention. Radiotherapy is usually considered as an adjuvant to surgery but may be used as first-line for patients without significant epidural extension or neurological deficits. The majority of AVHs occur in the thoracic spine and patients present with symptoms of spinal cord compression.[1,3] This case highlights an unusual clinical presentation of a lumbar AVH that progressed with epidural invasion of the tumor resulting in a significant epidural tumor spread and symptoms of neurogenic claudication. The rarity of such extensive spread of VH, especially in the lumbar spine, raised the suspicion for other neoplastic etiologies including lymphoma, enplaque meningioma, and epidural metastasis.[4] AVH presenting as neurogenic claudication secondary to spinal stenosis in the lumbar spine is exceptionally rare. The recognition of uncommon presentation of this common pathology is important and once diagnosed; they can be treated via various treatment options depending on physicians’ preference. Declaration of patient consent The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed. Financial support and sponsorship Nil. Conflicts of interest There are no conflicts of interest.