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LETTER TO EDITOR
Year : 2011  |  Volume : 59  |  Issue : 1  |  Page : 134-136

Thoracic epidural angiolipoma with bilateral multilevel extraspinal extensions: A rare entity


Department of Neurosurgery, L.T.M.G. Hospital, Mumbai, India

Date of Submission19-Aug-2010
Date of Decision19-Aug-2010
Date of Acceptance08-Oct-2010
Date of Web Publication18-Feb-2011

Correspondence Address:
Batuk Diyora
Department of Neurosurgery, L.T.M.G. Hospital, Mumbai
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.76875

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How to cite this article:
Diyora B, Nayak N, Kukreja S, Kamble H, Sharma A. Thoracic epidural angiolipoma with bilateral multilevel extraspinal extensions: A rare entity. Neurol India 2011;59:134-6

How to cite this URL:
Diyora B, Nayak N, Kukreja S, Kamble H, Sharma A. Thoracic epidural angiolipoma with bilateral multilevel extraspinal extensions: A rare entity. Neurol India [serial online] 2011 [cited 2019 Dec 15];59:134-6. Available from: http://www.neurologyindia.com/text.asp?2011/59/1/134/76875


Sir,

Spinal angiolipomas are rare benign tumors and account for 0.04-1.2% of all spinal tumors and 2-3% of spinal extradural tumors. [1],[2],[3],[4] We present the operative findings and MRI features of a noninfiltrative angiolipoma with bilateral multilevel extraspinal extensions.

A 20-year-old male presented with upper abdominal pain, radiating to back, for the past 6 months and bilateral lower limb weakness since 1 week. Neurological examination revealed paraparesis and impaired sensation below T4 level. Deep tendon reflexes were hyperactive and plantar response was bilateral extensor. Magnetic resonance imaging (MRI) of the spine [Figure 1]a and b showed a space occupying lesion from T5 to T8 vertebral levels located in the posterior epidural space. It was spindle shaped, measuring 6.5 cm in length and 12 mm at maximum width and extending bilaterally into the extraspinal space through neural foramina. It was isointense on T1-weighted images and hyperintense on T2-weighted images [Figure 2]a and b and showed enhancement with contrast.
Figure 1: (a) MRI dorsal spine (sagittal view)— T1-weighted image showing a hypointense dorsally located extradural mass causing thecal sac compression. (b) MRI dorsal spine (Sagittal view)— Lesion appears hyperintense on T2-weighted image with obliteration of CSF spaces

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Figure 2: (a and b) MRI dorsal spine (axial view)— showing a flattened, ventrally displaced spinal cord due to compression by the tumor

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The patient underwent T4-T9 laminectomy with total removal of epidural mass. Intraoperatively, the mass was reddish, soft to firm, fleshy with minimal vascularity. It had bilateral, multilevel, extraspinal extensions with widening of the neural foramina. There was a good plane of cleavage between the mass and dura. Extraspinal extensions of the mass were dissected through the widened foramina on either side [Figure 3]a and b. A satisfactory decompression of the spinal cord as well as nerve roots was achieved. Histopathology revealed angiolipoma [Figure 4]. Patient had a complete neurological recovery over the next 3 months.
Figure 3: (a and b) MRI dorsal spine (coronal view)— showing the bilateral multiple extraspinal extensions of the tumor. Picture of the excised tumor along with its multiple extraspinal extensions

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Figure 4: Histomicrograph of the surgical specimen shows typical angiolipoma composed of mature adipose tissue and blood vessels (H and E; 40)

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Angiolipoma is a subgroup of lipoma, also known as hemangiolipoma, angiomyolipoma, and fibromyolipoma. [5] The pathogenesis of spinal angiolipoma is unknown. Their probable origin is from progenitor tissue as lipoma and hemangioma. [4] Their preponderance in peri- or postmenopausal women supports a role of hormonal influence. Pathologically, the lesion is composed of mature fat cells and capillaries with some fibrin thrombi. Presence of fibrinous microthrombi in the lumen is a diagnostic feature. Degenerative changes (i.e., hyalinization, myxoid change, and fibrosis) may be present in longstanding cases.

Angiolipomas are of two types: noninfiltrating and infiltrating. [6] Noninfiltrating angiolipomas are more common. They are encapsulated lesions which present with painful, soft subcutaneous nodules. Infiltrating angiolipomas are rare, usually involve the extremities and may extensively infiltrate into the surrounding tissues.

Progressive paraparesis, back pain without radiculopathy, lower extremity sensory changes, and hyperreflexia are the usual clinical presentations. Unusually, patient can present with sudden-onset paraparesis. Exacerbation of symptoms may occur during pregnancy and in obese patients probably because of hormonal changes and increase in fatty component within the angiolipoma. [5] However, the association of spinal angiolipoma with increased body mass index has not been systematically studied. [7]

MRI reveals a solid mass that is usually hyperintense on T1 than T2-weighted signals, but can also be of heterogeneous or mixed intensity. It may be isointense with fat suppression sequences. Because of the presence of capillaries and venous channels, angiolipoma does not contain vascular flow void on MRI.

Complete surgical excision of the lesion is the treatment of choice. Complete excision is always possible in noninfiltrating angiolipoma and difficult in infiltrating angiolipomas, especially when it is located anteriorly or anterolaterally. [8] Because of the slow growing nature of the tumor, even partial excision would result in significant relief of pain and neurological symptoms. The role of external beam radiation in partially excised tumors is controversial. Owing to concerns of potential malignancy postoperative radiotherapy has been used in patients following partial excision of spinal epidural angiolipoma. [9],[10],[11] Current literature suggests that because of the benign nature of the tumor and very good prognosis, adjuvant radiation should not be used. [12]

 
  References Top

1.Fourney DR, Tong KA, Macaulay RJ, Griebel RW. Spinal angiolipoma. Can J Neurol Sci 2001;28:82-8.   Back to cited text no. 1
    
2.Garg A, Gupta V, Gaikwad S, Deol P, Mishra NK, Sharma MC, et al. Spinal angiolipoma: Report of three cases and review of MRI features. Australas Radiol 2002;46:84-90.   Back to cited text no. 2
    
3.Kujas M, Lopes M, Lalam TF, Fohanno D, Poirier J. Infiltrating extradural spinal angiolipoma. Clin Neuropathol 1999;18:93-8.  Back to cited text no. 3
    
4.Preul MC, Leblanc R, Tampieri D, Robitaille Y, Pokrupa R. Spinal angiolipomas. Report of three cases. J Neurosurg 1993;78:280-6.  Back to cited text no. 4
    
5.Provenzale JM, Mclendon RE. Spinal angiolipomas: MR features. AJNR Am J Neuroradiol 1996;17:713-9.  Back to cited text no. 5
    
6.Lin JL, Lin F. Two entities in angiolipoma. A study of 459 cases of lipoma with review of literature on infiltrating angiolipoma. Cancer 1974;34:720-7.  Back to cited text no. 6
    
7.Hungs M, Paré LS. Spinal angiolipoma: Case report and literature review. J Spinal Cord Med 2008;31:315-8.  Back to cited text no. 7
    
8.Rocchi G, Caroli E, Frati A, Cimatti M, Savlati M. Lumbar spinal angiolipomas: Report of two cases and review of the literature. Spinal Cord 2004;42:313-6.  Back to cited text no. 8
    
9.Anson JA, Cybulski GR, Reyes M. Spinal extradural angiolipoma: A report of two cases and review of the literature. Surg Neurol 1990;34:173-8.  Back to cited text no. 9
    
10.Bucy PC, Ritchey H. Klippel-Feil syndrome associated with compression of spinal cord by an extradural hemangiolipoma. J Neurosurg 1947;4:476-81.   Back to cited text no. 10
    
11.Gonzalez-Crussi F, Enneking WF, Arean VM. Infiltrating angiolipoma. J Bone Joint Surg Am 1966;486:1111-24.  Back to cited text no. 11
    
12.Gelabert-González M, García-Allut A. Spinal extradural angiolipoma: Report of two cases and review of the literature. Eur Spine J 2009;18:324-35.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]

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