a S c i T e c h n o l j o u r n a lCase Report

Journal of Spine & Neurosurgery

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Nardone et al., J Spine Neurosurg 2013, S1http://dx.doi.org/10.4172/2325-9701.S1-005

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Purely Spinal Epidural Cavernous Hemangiomas: A Case ReportAntonio Nardone1, Delia Cannizzaro1, Federico Caporlingua1, Lorenzo Pescatori1, Alessandro Landi1* and Antonio Santoro1

AbstractCavernous hemangiomas affect mainly the brain and infrequently they are located in the intramedullary space. Spinal epidural cavernous haemangiomas are extremely rare and are mainly located to the cervico-thoracic spine. It is infrequently considered in the differential diagnosis of spinal epidural masses. We report a rare case of a cavernous haemangioma confined to the lumbar spinal epidural space. A 27 year-old man came to our attention complaining of a progressive weakness and impaired cutaneous sensibility on the left inferior limb. An MRI showed a pure extradural mass without invasion of the bone. He was submitted to an open surgery and the mass was removed. The histopathology revealed a typical cavernous haemangioma. The patient showed significant clinical and neurological improvement after surgery. Radiological presentation could be confusing in a purely epidural cavernous haemangioma. Awareness of the characteristics of the lesion will facilitate its diagnosis and treatment.

KeywordsCavernous hemangioma; CT; Disc protrusion; Epidural mass; MRI

*Corresponding author: Alessandro Landi, Department of Neurology and Psychiatry, Sapienza University of Rome, Rome, Italy, Tel: +39-6-49979105; Fax: +39-6-49979105; E-mail: dott.alessandro.landi@gmail.com

Received: December 05, 2013 Accepted: December 10, 2013 Published: December 16, 2013

progressive weakness and increasing pain to the left inferior limb. Previous clinical history was negative.

Examination

Neurological examination revealed a grade 3 motor deficit at the extension of the left foot and big toe, a diminished plantar and ankle reflexes and positivity to the straight leg raising test at 30°. No bowel or bladder dysfunction was noted.

Neuroradiological investigation

A CT scan revealed a mass in the left S1 neural foramen. Its density was similar to the L5/S1 intervertebral disc. It extended cranially and medially occupying the left lateral recess and L5-S1 foramen.

An MRI confirmed the presence of a L5-S1 extradural mass. The lesion was iso-intense to the muscle and spinal cord on T1-weighted images and hyper-intense, despite being slightly less intense than cerebrospinal fluid, on T2-weighted images (Figure 1).

Surgical treatment

An L5-S1 hemilaminectomy was performed and no bony erosion was noted. An extradural mass strictly adherent to the left S1 nerve root was exposed. The lesion presented as a highly vascularized reddish soft mass. It was coagulated and easily detached from the dura mater with microsurgical technique. A total excision of the mass was achieved (Figure 2).

Histopathology

The lesion consisted of a large number of thin walled vascular channels in collagenous connective tissue, lined by a single layer of endothelial cells. A diagnosis of cavernous haemangioma was made.

Postoperative course

Patient’s postoperative course was uneventful and he recovered his deficit in the first 2 days after the surgery. He was discharged 3 day after the surgery.

DiscussionCavernous haemangiomas are developmental vascular

Core Tip Cavernous spinal epidural hemangiomas are rare lesions that

are to be distinguished from more common findings at this level. A correct preoperative diagnosis is fundamental to choose the right treatment, thus avoiding complications.

Introduction Cavernous hemangiomas are developmental lesions that affect

the cerebral hemispheres, cerebellum and brainstem [1,2]. Rarely they are found in the epidural spinal space and, whether it occurs, the differential diagnosis includes foraminal disc protrusion, schwannoma or neurofibroma, synovial cyst, metastasis, cavernous haemangioma, lymphoma, prominent epidural veins and, rarely, other mesenchymal tumors such as angiolipoma [3,4]. We present a case of an extremely rare spinal epidural cavernous haemangioma of a sacral nerve.

Case ReportA 27 year-old man presented to our attention complaining a

Figure 1: Round shaped lesions (132,2x14,5 mm) iso-intense with the muscle and spinal cord on T1-weighted images (a) and hyperintense on T2-weighted images with a 5 mm nodular peripheral component showing an intermediate signal on T2-weighted images (b).

Citation: Nardone A, Cannizzaro D, Caporlingua F, Pescatori L, Landi A, et al. (2013) Purely Spinal Epidural Cavernous Hemangiomas: A Case Report. J Spine Neurosurg S1.

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The Future of Neurosurgical Oncology

malformations composed of thin-walled vascular spaces lined by a single endothelial layer. The walls of these vascular spaces do not have a significant smooth muscle or elastin component [5]. Although they are not classified as true neoplasms, cavernous haemangiomas may present signs of growth. Cavernous haemangiomas may develop anywhere in the neuraxis and its surrounding membranes [6,7].

Haemangiomas of all kinds comprise ~ 8% of benign soft-tissue tumors referred for surgery and pathology [8]. Although high, the real prevalence of haemangiomas is difficult to estimate because most of them are clinically asymptomatic.

Spinal cord cavernomas were thought to be rare, but with the advent of MRI are more and more frequently diagnosed, and their frequency may be greater than previously thought [9,10].

With regard to the spine, cavernous haemangiomas are most commonly observed within the vertebral body, with a reported prevalence at autopsy series between 7–12% [2]. They represent approximately 5% to 12% of the vascular lesions of the spine [6]. Occasionally, haemangiomas extend beyond the vertebral margins into the epidural space, and in this compartment, they may cause neural compressions. Intramedullary cavernous haemangiomas are rarer, but those in a purely epidural location are even rarer, comprising ~ 1–2% of spinal hemangiomas [11]. Most of these lesions are located intracranially, supratentorial sites being the most commonly affected. Spinal epidural cavernous haemangioma more common location is the lumbar and thoracic spine [12].

The most frequent clinical presentation is a progressive compressive myelopathy, in the form of progressive paraparesis [13]. Patients may also present with pain, radiculopathy or acute paraplegia [14]. When radiculopathy occurs, the duration of symptoms is relatively short; long-standing radiculopathy is unusual [15]. Sudden onset of symptoms may be due to hemorrhage or venous thrombosis with consequent enlargement of the haemangioma and therefore neural compression [16].

The MR imaging of spinal cavernous haemangiomas are not as characteristic as those of cranial cavernomas. In most of the cases the T1-weighted images of spinal cavernous haemangioma shows homogenous signal intensity similar to that of spinal cord and muscle, while on T2-weighted images the signal is consistently high, as observed in the current case. Frequently, lesions are characterized by their extension into the intervertebral foramen [17]. The rim

of hypo-intensity resulting from hemosiderin deposits, seen in intramedullary cavernous haemangioma is not seen in epidural cavernous haemangioma [18]. These radiological characteristics help distinguishing cavernous haemangiomas from other lesions of the spine. The differential diagnoses for these types of lesion include neurinoma, meningioma, lymphoma and metastasis [19]. The diagnosis is much easier when the lesion is associated with epidural hemorrhagic changes, but then other vascular information can have similar MRI appearance.

Surgery is the mainstay of treatment. A complete removal is possible in almost every case and good recovery is the most frequent outcome [3,20]. An en-bloc resection is advisable as the lesions are easily shrank by bipolar coagulation and because piece-meal resection causes profuse bleeding, prolonging operative time.

Radiotherapy has been documented in single case report with good results [21] and it may be considered for patients who underwent an incisional bioptic or incomplete surgical removal.

In conclusions, we report a rare case of a purely epidural spinal cavernous haemangioma, which was easily and completely resected with surgery. Preoperative diagnosis is helpful to guide surgical resection. Surgery is the mainstay of therapy and good result may be achieved with complete removal.

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Figure 2: Postoperative MRI T2-weighted (a) and T1-weighted (b) images showing the complete resection of the lesion and the decompression of the lumbar root.

Citation: Nardone A, Cannizzaro D, Caporlingua F, Pescatori L, Landi A, et al. (2013) Purely Spinal Epidural Cavernous Hemangiomas: A Case Report. J Spine Neurosurg S1.

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The Future of Neurosurgical Oncology

16. Isla A, Alvarez F, Morales C, García Blazquez M (1993) Spinal epidural hemangiomas. J Neurosurg Sci 37: 39-42.

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21. Sohn MJ, Lee DJ, Jeon SR, Khang SK (2009) Spinal radiosurgical treatment for thoracic epidural cavernous hemangioma presenting as radiculomyelopathy: technical case report. Neurosurgery 64: 1202-1203.

Author Affiliations Top1Department of Neurology and Psychiatry, Neurosurgery, Sapienza University of Rome, Rome, Italy

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Submit your next manuscript at ● www.scitechnol.com/submissionThis article is published in the special issue, The Future of Neurosurgical Oncology and has been edited by Dr. Jeff Lehman, Spine Midwest, Inc. Jefferson City, USA.