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ORIGINAL ARTICLE
Year : 2014  |  Volume : 62  |  Issue : 4  |  Page : 367-370

Spinal angiolipoma: Experience of twelve patients and literature


Department of Neurosurgery,Beijing Tiantan Hospital, Capital Medical University; Department of Neurosurgery,China National Clinical Research Center for Neurological Diseases, Beijing, China

Date of Submission14-Apr-2014
Date of Decision25-May-2014
Date of Acceptance10-Aug-2014
Date of Web Publication19-Sep-2014

Correspondence Address:
Pinan Liu
Department of Neurosurgery, Beijing Tiantan Hospital, Capital Medical University, Beijing 100050
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.141232

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 » Abstract 

Purpose: To summarize our experience in treating patients with spinal angiolipomas. Materials and Methods: We conducted a retrospective review of the case records of patients with spinal angiolipoma who underwent surgery between March 1992 and March 2013. Results: Of the 12 patients seen during the study period, 10 patients had total removal of the lesion and 2 patients had sub-total removal. The postoperative course was uneventful, and the outcomes were good in patients who underwent total excision of the lesion. No patient received adjuvant radiation even in the patients with sub-total excision. Conclusion: In patients with spinal angiolipoma, total excision should be achieved and is associated with good outcomes.


Keywords: Angiolipoma, diagnosis, spinal epidural tumor, treatment


How to cite this article:
Wang B, Yang Z, Yang J, Wang G, Xu Y, Liu P. Spinal angiolipoma: Experience of twelve patients and literature. Neurol India 2014;62:367-70

How to cite this URL:
Wang B, Yang Z, Yang J, Wang G, Xu Y, Liu P. Spinal angiolipoma: Experience of twelve patients and literature. Neurol India [serial online] 2014 [cited 2018 Dec 10];62:367-70. Available from: http://www.neurologyindia.com/text.asp?2014/62/4/367/141232



 » Introduction Top


Angiolipomas are common benign lesions, consisting of both mature fatty tissue and proliferating abnormal blood vessels, [1] which are usually located subcutaneously on the trunk and limbs. [2] In particular, spinal angiolipoma (SAL) is an uncommon clinicopathological neoplasm and accounts for 0.14-1.2% of all spinal tumors and 2-3% of extradural spinal tumors. [3],[4] In this report, we summarize the clinical features of SAL and our surgical experience with patients with SAL.


 » Materials and Methods Top


This is a retrospective analysis of case records of 12 patients with SAL who underwent surgery at our hospital between March 1992 and March 2013. All patients had undergone preoperative magnetic resonance imaging (MRI). Additionally, axial computed tomography (CT) was performed in 2 patients. According to the McCormick classification, the clinical outcome was evaluated pre- and post-operatively. The following details were collected from the medical records: Demographic details, presenting symptoms, clinical signs, imaging features, and treatments.

Electrophysiological monitoring, including somatosensory-evoked potentials and motor-evoked potentials, was used in all operations. All patients underwent operations in a semi-prone position. The posterior midline approach was adopted; the laminae and spinous processes, one level above and below the tumor, were removed to provide adequate exposure of the tumor margins. To maintain spinal stability after tumor resection, the laminae were repositioned and secured with multiple small plates and screws. In addition, 1 patient underwent assisted thoracoscopic surgery at the same time because of the invasion of both spinal and thoracic cavity. Postoperatively, patients were followed up at 3, 6, and 12 months and thereafter, once every 12 months. MRI was performed at every follow-up visit for each patient, and the mean follow-up time was 63.5 months.


 » Results Top


Of the 12 patients with SAL, 7 were females. The mean age was 48 years and the mean duration of symptoms was 16.6 months. The main clinical symptoms included back pain, unilateral or bilateral lower extremity numbness or progressive weakness, and symptoms of sciatica. Total excision of tumors was done in 10 patients and sub-total excision in the remaining 2 patients. The postoperative course ranged from 3 months to 192 months. The mean duration of follow up was 63.5 months. No recurrence was observed in any patients. The postoperative course was uneventful with obvious neurological improvement. Gradual improvement was noted during follow up [Table 1].
Table 1: Summary of 12 cases of spinal angiolipoma

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Representative case report

Case 1

A 55-year-old obese woman was admitted with a 17-month history of gradually progressing numbness and pain in both legs. She also complained of a gait disturbance and difficulty in walking since 2 months before admission. Physical examination revealed hypesthesia below the T7 level, analgesia below the T9 level, decreased muscle strength, increased muscle tone, and hyperreflexia in the lower extremities. Plain radiographs of the thoracic and lumbar area did not reveal any bone abnormalities. MRI revealed an area of abnormally high-signal intensity in the posterior extradural space at the T6-8 levels. Gadolinium-enhanced T1-weighted imaging showed homogenous contrast enhancement [Figure 1]. All patients had thoracic laminectomy (T6-T8 levels). Intraoperatively, a reddish soft mass was exposed in the extradural space. The mass was easily dissected from the dura mater and completely removed in 10 patients [Figure 2]. Histopathological examination revealed tumor composed of mature, well-differentiated adipose tissue with numerous admixed vascular elements [Figure 3]. The laminae were finally repositioned and secured with multiple small plates and screws to maintain spinal stability after tumor resection. Follow-up neurological examination at 3 months postoperatively showed only hyperreflexia in both the lower extremities and no other abnormal findings. No tumor recurrence was evident on MRI [Figure 4].
Figure 1: (a) Non-contrast T1-weighted sagittal MRI showing a homogeneous hyperintense mass compressing the spinal cord; (b) Non-contrast T2-weighted sagittal MRI showed that mass was also hyperintense; (c) Post-contrast T1-weighted sagittal MRI showed obvious enhancement of the mass, typically "nib" signal

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Figure 2: After bilateral laminectomies were performed, a reddish soft mass exposed in the extradural space

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Figure 3: Pathological specimen: A mix of adipocytes and blood-filled vascular channels of varying size and shape (H and E, ×100)

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Figure 4: Postoperative T1-weighted sagittal MR image showing a lack of recurrence

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 » Discussion Top


Spinal angiolipomas are considered to be benign and rare lesions of the spinal axis. The first report of SAL was in a 16-year-old boy at autopsy in 1890 [5] SAL was initially considered to be a subgroup of lipomas and has been referred to by various terms: Vascular lipoma, hemangiolipoma, and fibromyolipoma. [6] In 1960, SAL was described as a distinct anatomopathologic entity. [7]

In 1974, Lin et al. categorized SALs into two subtypes: Noninfiltrating and infiltrating types. The noninfiltrating type is more common and is usually well encapsulated. The infiltrating types are rare, partially or entirely unencapsulated, and ill defined, and they commonly infiltrate the surrounding tissues, especially the bone. [8] The infiltrating type tumors are located in the anterior or anterolateral parts of the extradural space, while the noninfiltrating type tumors are located in the posterior parts. [9] In cases of recurrent infiltrating SAL, wider resection followed by radiotherapy has been recommended. [8] Recently, some authors have reported that most patients have a good prognosis even with subtotal removal, because the tumors are usually slow growing and do not undergo malignant transformation. The outcomes of the infiltrating and noninfiltrating types are not different. [10],[11] Of the 12 patients, 2 patients had infiltrating tumors. It was difficult to achieve total resection; however, sub-total resection had no recurrence after 40 months of follow up. Other authors proposed a new subtype in which overweight people are more likely to have angiolipomas with lipomatosis in the upper and/or lower segments. The results in this type are slightly worse than other subtypes. In our series, MRI scans of the entire spine were performed in all patients. [12] Of 12 patients with SAL, 8 patients were overweight. We did not identify any patients with angiolipomas and lipomatosis in the upper and/or lower segments. The value of a category and the use of adjuvant radiotherapy after incomplete removal of the tumor remain controversial.

Most angiolipomas occur mostly in adults aged between 40-60 years. A female predominance has been observed. [13],[14] The most common location of spinal angiolipoma is the thoracic epidural space. Thoracic predominance of the tumor could be the result of the regional variation in the blood supply in the spine, wherein the mid-thoracic spine is the least perfused. [15] In our patients, the tumor location was 5 in the thoracic region, 4 in the lumbar region, and 1 in the lumbosacral region. The clinical symptoms of angiolipomas were accordance with other benign, space-occupying spinal lesions. The symptoms usually evolve over a span of months to years. [16],[17] The data in our cases were in agreement with those of other published studies.

MRI is the standard imaging modality for the diagnosis of SAL. On T1-weighted images, SAL are hyperintense and often inhomogeneous [Figure 1]a and variable intensity; they are often hyperintense on T2-weighted images [Figure 1]b Gadolinium enhancement is due to the vascularity of these tumors. Significant heterogeneity in the imaging studies is attributed to the variable vascular and adipose elements of the tumor, typically the "nib" signal [1],[18] [Figure 1]c. Moreover, the images do not typically contain vascular flow voids on MRI because of the tumor's low vascular flow characteristics. [9],[19]

The differential diagnosis includes extradural lipomatosis, nerve sheath tumor, meningioma, metastasis, chronic extradural hematoma, and lymphoma. To evaluate the degree of destruction, CT scanning is essential when the tumor infiltrates the vertebral body. [17],[18],[19],[20] An easy misdiagnosis is extradural lipomatosis, which has the following characteristics: The lipoma is always accompanied by spina bifida, is usually seen in 20-year-old patients, and is more common in men and in the lumbosacral region. [18],[21] According to our experience, the degree of central hypointensity on T1-weighted images is predictive of the degree of vascularity that is likely to be encountered at surgery; the fat suppression sequence is particularly important for this tumor type, which is helpful for identifying small levels of vascular components.

Pathologically, the most common feature is a non-encapsulated lesion with two distinct components-mature adipocytes with delicate, proliferating abnormal blood vessels that are haphazardly wedged [Figure 3]. The ratio of fat to vessels is variable, ranging from 1:3 to 2:3. [14] Immunohistochemical assays were performed in a few cases, and a low proliferation rate was found with Ki-67. Samdami et al. observed a positive stain for CD31, Factor XIIIa, and Factor VIII and a negative stain for gliofibrillar acid protein. [2],[22]

Spinal angiolipomas are treated exclusively by surgical removal of the lesion. In cases of dorsally located, non-infiltrating lesions, gross total resection of the tumor and the associated capsule is usually achievable. In our patients, a high-speed drill revealed a reddish, soft mass extending into the extradural space of the spinal canal. The mass appeared as well-circumscribed lesions with no adhesions to the dura [Figure 2]. The dura was not opened, and the entire mass was removed; and when the tumor involved both the spinal and thoracic cavity [Figure 5], the help of a cardiothoracic surgeon was taken to excise the lesion. To maintain spinal stabilization during surgery, the involved vertebral body should be preserved. Nevertheless, hemostasis of the operative site should be achieved. Adjuvant radiation therapy is not need for this benign lesion, even in patients with sub-total excision. [23]
Figure 5: MRI showing the tumor invading into both the spinal and thoracic cavity

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Spinal angiolipoma is a rare but well-defined clinical entity with distinct clinicopathological and imaging features. The outcome after surgery is very good for total or sub-total resection. Early clinical recognition of this benign entity is essential for a good functional outcome.

 
 » References Top

1.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. 1
    
2.Samdani AF, Garonzik IM, Jallo G, Eberhart CG, Zahos P. Spinal angiolipoma: Case report and review of the literature. Acta Neurochir (Wein) 2004;146:299-302.  Back to cited text no. 2
    
3.Fourney DR, Tong KA, Macaulay RJ, Griebel RW. Spinal angiolipoma. Can J Neurol Sci 2001;28:82-8.  Back to cited text no. 3
    
4.Pagni CA, Canavero S. Spinal epidural angiolipoma: Rare or unreported? Neurosurgery 1992;31:758-64.  Back to cited text no. 4
    
5.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. 5
    
6.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. 6
    
7.Howard WR, Helwig EB. Angiolipoma. Arch Dermatol 1960;82:924-31.  Back to cited text no. 7
    
8.Lin JJ, 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. 8
    
9.Leu NH, Chen CY, Shy CG, Lu CY, Wu CS, Chen DC, et al. MR imaging of an infiltrating spinal epidural angiolipoma. AJNR Am J Neuroradiol 2003;24:1008-11.  Back to cited text no. 9
    
10.Trabulo A, Cerqueira L, Monteiro J, Roque P, Reis FC, Coelho MR. Spinal angiolipomas revisited: Two case reports. Acta Neurochir (Wien) 1996;138:1311-9.  Back to cited text no. 10
    
11.Guzey FK, Bas NS, Ozkan N, Karabulut C, Bas SC, Turgut H. Lumbar extradural infiltrating angiolipoma: A case report and review of 17 previously reported cases with infiltrating spinal angiolipomas. Spine J 2007;7:739-44.  Back to cited text no. 11
    
12.Si Y, Wang Z, Pan Y, Lin G, Yu T. Spinal angiolipoma: Etiology, imaging findings, classification, treatment, and prognosis. Eur Spine J 2014;23:417-25.  Back to cited text no. 12
    
13.Rubin G, Gornish M, Sandbank J, Shevach I, Rappaport ZH. Spinal extradural angiolipoma case report and review of the literature. Spine (Phila Pa 1976) 1992;17:719-24.  Back to cited text no. 13
    
14.Reyes D, Candocia FJ. Thoracolumbar spinal angiolipoma demonstrating high signal on STIR imaging: A case report and review of the literature. Spine J 2013;13:e1-5.  Back to cited text no. 14
    
15.Labram EK, el-Shunnar K, Hilton DA, Robertson NJ. Revisited: Spinal angiolipoma-three additional cases. Br J Neurosurg 1999;13:25-9.  Back to cited text no. 15
    
16.Andaluz N, Balko G, Bui H, Zuccarello M. Angiolipomas of the central nervous system. J Neurooncol 2000;49:219-30.  Back to cited text no. 16
    
17.Yamashita K, Fuji T, Nakai T, Hamada H, Kotoh K. Extradural spinal angiolipoma: Report of a case studied with MRI. Surg Neurol 1993;39:49-52.  Back to cited text no. 17
    
18.Hu S, Hu CH, Hu XY, Wang XM, Dai H, Fang XM, et al. MRI Features of spinal epidural angiolipomas. Korean J Radiol 2013;14:810-7.  Back to cited text no. 18
    
19.Turanzas F, Domínguez J, Eyerbe P, Dhimes P. Infiltrating spinal angiolipoma. Case report. Neurocirugia 1994;5:246-9.  Back to cited text no. 19
    
20.Provenzale JM, McLendon RE. Spinal angiolipomas: MR features. AJNR Am J Neuroradiol 1996;17:713-9.  Back to cited text no. 20
    
21.Petrella G, Tamburrini G, Lauriola L, Di Rocco C. Spinal epidural angiolipoma complicated by an intratumoral abscess: Case report. J Neurosurg 2005;103 (2 Suppl):166-9.  Back to cited text no. 21
    
22.Chotai S, Hur JS, Moon HJ, Kwon TH, Park YK, Kim JH. Spinal angiolipoma--case report. Neurol Med Chir (Tokyo) 2011;51:539-42.  Back to cited text no. 22
    
23.Meng J, Du Y, Yang HF, Hu FB, Huang YY, Li B, et al. Thoracic epidural angiolipoma: A case report and review of the literature. World J Radiol 2013;5:187-92.  Back to cited text no. 23
    


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