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Dinger TF, Eerikäinen MS, Michel A, Gembruch O, Darkwah Oppong M, Chihi M, Blau T, Uerschels AK, Pierscianek D, Deuschl C, Jabbarli R, Sure U, Wrede KH. A New Subform? Fast-Progressing, Severe Neurological Deterioration Caused by Spinal Epidural Lipomatosis. J Clin Med 2022; 11:jcm11020366. [PMID: 35054059 PMCID: PMC8781155 DOI: 10.3390/jcm11020366] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/12/2021] [Revised: 01/03/2022] [Accepted: 01/10/2022] [Indexed: 02/04/2023] Open
Abstract
Spinal epidural lipomatosis (SEL) is a rare condition caused by hypertrophic growth of epidural fat. The prevalence of SEL in the Western world is approximately 1 in 40 patients and is likely to increase due to current medical and socio-economic developments. Rarely, SEL can lead to rapid severe neurological deterioration. The pathophysiology, optimal treatment, and outcome of these patients remain unclear. This study aims to widen current knowledge about this “SEL subform” and to improve its clinical management. A systematic literature review according to the PRISMA guidelines using PubMed, Scopus, Web of Science, and Cochrane Library was used to identify publications before 7 November 2021 reporting on acute/rapidly progressing, severe SEL. The final analysis comprised 12 patients with acute, severe SEL. The majority of the patients were male (9/12) and multimorbid (10/12). SEL mainly affected the thoracic part of the spinal cord (11/12), extending a median number of 7 spinal levels (range: 4–19). Surgery was the only chosen therapy (11/12), except for one critically ill patient. Regarding the outcome, half of the patients regained independence (6/11; = modified McCormick Scale ≤ II). Acute, severe SEL is a rare condition, mainly affecting multimorbid patients. The prognosis is poor in nearly 50% of the patients, even with maximum therapy. Further research is needed to stratify patients for conservative or surgical treatment.
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Affiliation(s)
- Thiemo Florin Dinger
- Department of Neurosurgery and Spine Surgery, University Hospital of Essen, University of Duisburg-Essen, 47057 Duisburg, Germany; (A.M.); (O.G.); (M.D.O.); (M.C.); (A.-K.U.); (D.P.); (R.J.); (U.S.); (K.H.W.)
- Correspondence: ; Tel.: +49-201-723-1201
| | - Maija Susanna Eerikäinen
- Institute for Diagnostic and Interventional Radiology and Neuroradiology, University Hospital Essen, University of Duisburg-Essen, 47057 Duisburg, Germany; (M.S.E.); (C.D.)
| | - Anna Michel
- Department of Neurosurgery and Spine Surgery, University Hospital of Essen, University of Duisburg-Essen, 47057 Duisburg, Germany; (A.M.); (O.G.); (M.D.O.); (M.C.); (A.-K.U.); (D.P.); (R.J.); (U.S.); (K.H.W.)
| | - Oliver Gembruch
- Department of Neurosurgery and Spine Surgery, University Hospital of Essen, University of Duisburg-Essen, 47057 Duisburg, Germany; (A.M.); (O.G.); (M.D.O.); (M.C.); (A.-K.U.); (D.P.); (R.J.); (U.S.); (K.H.W.)
| | - Marvin Darkwah Oppong
- Department of Neurosurgery and Spine Surgery, University Hospital of Essen, University of Duisburg-Essen, 47057 Duisburg, Germany; (A.M.); (O.G.); (M.D.O.); (M.C.); (A.-K.U.); (D.P.); (R.J.); (U.S.); (K.H.W.)
| | - Mehdi Chihi
- Department of Neurosurgery and Spine Surgery, University Hospital of Essen, University of Duisburg-Essen, 47057 Duisburg, Germany; (A.M.); (O.G.); (M.D.O.); (M.C.); (A.-K.U.); (D.P.); (R.J.); (U.S.); (K.H.W.)
| | - Tobias Blau
- Institute of Neuropathology, University Hospital of Essen, University of Duisburg-Essen, 47057 Duisburg, Germany;
| | - Anne-Kathrin Uerschels
- Department of Neurosurgery and Spine Surgery, University Hospital of Essen, University of Duisburg-Essen, 47057 Duisburg, Germany; (A.M.); (O.G.); (M.D.O.); (M.C.); (A.-K.U.); (D.P.); (R.J.); (U.S.); (K.H.W.)
| | - Daniela Pierscianek
- Department of Neurosurgery and Spine Surgery, University Hospital of Essen, University of Duisburg-Essen, 47057 Duisburg, Germany; (A.M.); (O.G.); (M.D.O.); (M.C.); (A.-K.U.); (D.P.); (R.J.); (U.S.); (K.H.W.)
| | - Cornelius Deuschl
- Institute for Diagnostic and Interventional Radiology and Neuroradiology, University Hospital Essen, University of Duisburg-Essen, 47057 Duisburg, Germany; (M.S.E.); (C.D.)
| | - Ramazan Jabbarli
- Department of Neurosurgery and Spine Surgery, University Hospital of Essen, University of Duisburg-Essen, 47057 Duisburg, Germany; (A.M.); (O.G.); (M.D.O.); (M.C.); (A.-K.U.); (D.P.); (R.J.); (U.S.); (K.H.W.)
| | - Ulrich Sure
- Department of Neurosurgery and Spine Surgery, University Hospital of Essen, University of Duisburg-Essen, 47057 Duisburg, Germany; (A.M.); (O.G.); (M.D.O.); (M.C.); (A.-K.U.); (D.P.); (R.J.); (U.S.); (K.H.W.)
| | - Karsten Henning Wrede
- Department of Neurosurgery and Spine Surgery, University Hospital of Essen, University of Duisburg-Essen, 47057 Duisburg, Germany; (A.M.); (O.G.); (M.D.O.); (M.C.); (A.-K.U.); (D.P.); (R.J.); (U.S.); (K.H.W.)
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Iampreechakul P, Tangviriyapaiboon T, Liengudom A, Lertbutsayanukul P, Thammachantha S, Siriwimonmas S. Sacral Extradural Angiolipoma Associated with Tight Filum Terminale and Spina Bifida Coexisting with Spinal Arteriovenous Fistula. World Neurosurg 2020; 140:37-45. [PMID: 32407913 DOI: 10.1016/j.wneu.2020.04.239] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/18/2020] [Revised: 04/29/2020] [Accepted: 04/30/2020] [Indexed: 10/24/2022]
Abstract
BACKGROUND Spinal arteriovenous fistula (AVF) may rarely associate with spinal dysraphism, that is, tethered spinal cord and spinal intradural lipoma. Spinal extradural angiolipoma coexisting with spinal AVF has not been reported in the literature. We reported an extremely rare case of sacral angiolipoma associated with tight filum terminale and sacral spina bifida coexisting with spinal AVF within this tumor. CASE DESCRIPTION A 55-year-old women presented with progressive myelopathy for 10 months. She had a painless, slow-growing mass at her left buttock since birth. Magnetic resonance imaging of the lumbosacral spine showed an extradural mass at the level of S3-S4, extending from the spinal canal through the spina bifida to the subcutaneous fat of the left buttock. There was a low conus medullaris at S2. Magnetic resonance imaging of the thoracic spine disclosed venous congestion with tortuous intradural flow voids along both ventral and dorsal surfaces of the spinal cord. Magnetic resonance angiography and spinal angiography revealed a hypervascular mass at the sacral level and associated arteriovenous shunt with cranial drainage into an enlarged medullary vein. Due to an infected pressure sore on the mass, endovascular treatment was initially performed with minimal recovery. Six months after complete healing of her infected pressure ulcer, the patient underwent surgical removal of extradural mass containing the AVF, and subsequent release of the tight filum. Histologic findings were consistent with angiolipoma. CONCLUSIONS Sacral extradural angiolipoma in the present case may be congenital in origin with development of an acquired spinal AVF within the tumor.
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Affiliation(s)
| | | | - Anusak Liengudom
- Department of Neurosurgery, Prasat Neurological Institute, Bangkok, Thailand
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Abstract
Angiolipomas are uncommon spinal tumors which differ from their cutaneous counterparts in having larger caliber vascular stroma. Although slow growing, they can cause rapid spinal cord compression and sudden-onset sensorimotor symptoms due to vascular engorgement, hemorrhage, or thrombosis. The goal of surgery is spinal decompression, and favorable outcome is the rule. We report a patient with spinal angiolipoma, vertebral hemangioma along with subcutaneous lipomas and angiolipomas, exhibiting the entire histopathological spectrum of these related soft-tissue tumors. Analysis of his family tree revealed a hereditary predilection. Familial angiolipomatosis is an uncommon genetic condition which has not been reported to occur with spinal angiolipomas thus far.
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Affiliation(s)
- Shradha Maheshwari
- Department of Neurosurgery Surgery, Dr. RN Cooper Hospital and HBT Medical College, Mumbai, Maharashtra, India
| | - Eham Lalit Arora
- Department of General Surgery, Grant Government Medical College and Sir JJ Group of Hospitals, Mumbai, Maharashtra, India
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Onishi FJ, Salem FAS, de Melo Lins DL, Dauar RFB, Stavale JN. Spinal thoracic extradural angiolipoma manifesting as acute onset of paraparesis: Case report and review of literature. Surg Neurol Int 2017; 8:150. [PMID: 28791193 PMCID: PMC5525459 DOI: 10.4103/sni.sni_467_16] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/28/2016] [Accepted: 01/22/2017] [Indexed: 11/04/2022] Open
Abstract
BACKGROUND Angiolipomas are benign tumors most commonly found in the thoracic spine. They are composed of mature adipocytes and abnormal vascular elements that usually present with a slowly progressive course of neurological deterioration. CASE DESCRIPTION A 35-year-old female, with a prior history of back pain, acutely developed paraparesis. When the thoracic magnetic resonance imaging (MRI) revealed a dorsal epidural mass at the T3-T5 level, she underwent a laminectomy for gross total excision of the lesion that proved to be an angiolipoma. On the second postoperative day, the patient was again able to ambulate. CONCLUSION The angiolipomas of spine are rare causes of spinal cord compression, and those presenting with acute neurological deficits should be immediately treated.
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Benvenutti-Regato M, De la Garza-Ramos R, Caro-Osorio E. Thoracic epidural spinal angiolipoma with coexisting lumbar spinal stenosis: Case report and review of the literature. Int J Spine Surg 2015; 9:67. [PMID: 26767159 PMCID: PMC4710163 DOI: 10.14444/2067] [Citation(s) in RCA: 16] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/13/2022] Open
Abstract
BACKGROUND Spinal angiolipomas (SALs) are uncommon benign lesions that may present insidiously with back pain or acutely with weakness due to tumor bleeding/thrombosis. Given their rarity, these lesions are often overlooked in the differential diagnosis of epidural masses. The purpose of this article is to report the case of an epidural SAL and to conduct a literature review on the topic. METHODS A case report and review of the literature using the PubMed/Medline databases. All case reports and case series were reviewed up to June 2015. RESULTS A 65-year old female presented with neurogenic claudication and magnetic resonance imaging (MRI) revealed lumbar spinal stenosis. Following decompressive surgery, she experienced symptom resolution, but three months postoperatively she presented to the emergency department with acute paraparesis. A thoracic MRI revealed a lesion located between T8 and T10 causing severe spinal cord compression. Following emergent laminectomy and en bloc resection, the patient regained function and the lesion was diagnosed as SAL. Our literature review revealed 178 reported cases, with a female and thoracic predominance. The majority of patients underwent surgical treatment, achieving a gross total resection in most cases. Similarly, complete symptom resolution was the most common outcome. CONCLUSION Spinal angiolipomas are uncommon spinal tumors. However, they may be treated as any other space-occupying lesion, and surgical resection allows for complete symptom recovery in most patients.
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Affiliation(s)
- Mario Benvenutti-Regato
- Tecnológico de Monterrey, School of Medicine and Health Sciences, Monterrey, México
- Neurology and Neurosurgery Institute, Centro Médico Hospital Zambrano Hellion, TecSalud, Monterrey, México
| | - Rafael De la Garza-Ramos
- Tecnológico de Monterrey, School of Medicine and Health Sciences, Monterrey, México
- Neurology and Neurosurgery Institute, Centro Médico Hospital Zambrano Hellion, TecSalud, Monterrey, México
| | - Enrique Caro-Osorio
- Tecnológico de Monterrey, School of Medicine and Health Sciences, Monterrey, México
- Neurology and Neurosurgery Institute, Centro Médico Hospital Zambrano Hellion, TecSalud, Monterrey, México
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Eap C, Bannwarth M, Jazeron JF, Kleber JC, Theret É, Duntze J, Litre CF. Spontaneous epidural hematoma due to cervico-thoracic angiolipoma. Neurochirurgie 2015; 61:398-400. [DOI: 10.1016/j.neuchi.2015.09.005] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/03/2012] [Revised: 08/28/2015] [Accepted: 09/23/2015] [Indexed: 10/22/2022]
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Ramdasi RV, Avinasha KM, Mahore A, Kawale J. Spinal angiolipoma manifesting with apoplexy. BMJ Case Rep 2014; 2014:bcr-2014-204379. [PMID: 24842366 DOI: 10.1136/bcr-2014-204379] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022] Open
Abstract
We report a 58-year-old man presenting with acute paraplegia. MRI showed a haematoma within a well-defined epidural lesion at C7-D1. Intraoperatively, organised epidural haematoma surrounded by tumour tissue was found. The final histopathology report was angiolipoma. The patient had dramatic recovery. Angiolipomas should be considered in the differential diagnosis of acute paraplegia when imaging shows well-circumscribed haematoma.
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Affiliation(s)
| | - K M Avinasha
- Department of Neurosurgery, Seth G.S. Medical College & KEM Hospital, Mumbai, Maharashtra, India
| | - Amit Mahore
- Department of Neurosurgery, Seth G.S. Medical College & KEM Hospital, Mumbai, Maharashtra, India
| | - Juhi Kawale
- Department of Medicine, Seth G.S. Medical College & KEM Hospital, Mumbai, Maharashtra, India
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Thoracolumbar spinal angiolipoma demonstrating high signal on STIR imaging: a case report and review of the literature. Spine J 2013; 13:e1-5. [PMID: 24071036 DOI: 10.1016/j.spinee.2013.06.057] [Citation(s) in RCA: 10] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/03/2012] [Revised: 02/05/2013] [Accepted: 06/14/2013] [Indexed: 02/03/2023]
Abstract
BACKGROUND Angiolipomas are rare benign tumors, accounting for 0.14% to 1.2% of all tumors of the spinal axis with vascular and fatty histological features. Spinal angiolipoma (SAL) is most commonly found in the thoracic region and has high signal on contrast enhanced fat-saturated T1-weighted imaging. Although the international literature is extensive, there are few cases reported in United States. OBJECTIVE To present a case of SAL located in the thoracolumbar region without high signal on contrast enhanced fat-saturated T1-weighted imaging and to review previously reported cases. STUDY DESIGN/METHODS Case report and review the literature. RESULTS Magnetic resonance imaging obtained in a 68-year-old man with a long history of lumbago showed a heterogeneous mass (T10-L1) hyperintense on T1-weighted imaging but not enhancing on suppression fat sequences, suggesting epidural hematoma. Surgical excision of the lesion was performed, and SAL was diagnosed and confirmed by pathology. The patient became asymptomatic. CONCLUSIONS The predominance of either vascular or fatty components inside the tumor might alter the expected results on magnetic resonance imaging with suppression fat sequences.
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Meng J, Du Y, Yang HF, Hu FB, Huang YY, Li B, Zee CS. Thoracic epidural angiolipoma: A case report and review of the literature. World J Radiol 2013; 5:187-192. [PMID: 23671756 PMCID: PMC3647212 DOI: 10.4329/wjr.v5.i4.187] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/06/2012] [Revised: 12/18/2012] [Accepted: 01/21/2013] [Indexed: 02/06/2023] Open
Abstract
Angiolipoma of the spine is a benign neoplasm consisting of both mature fatty tissue and abnormal vascular elements, and usually presents with a slow progressive clinical course. Our patient presented with bilateral lower extremity weakness and chest-back numbness. Physical examination revealed adipose elements superficial hypesthesia below the T5 level and analgesia below the T6 level. Magnetic resonance imaging (MRI) scan showed an avidly and heterogeneously enhancing mass which was located in the posterior epidural space. Compression of the thoracic cord by the fusiform mass was seen between T3-T4. During the operation, a flesh pink vascular mass (4.7 cm × 1.0 cm × 1.0 cm) with obscure margin and strong but pliable texture was found in the posterior epidural space extending from T3 to T4. There was no infiltration of the dura or the adjacent bony spine. Histopathological study of the surgical specimen showed a typical angiolipoma. We review the previously documented cases of spinal extradural angiolipomas performed with MRI.
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Abstract
BACKGROUND Acute paraplegia is a true emergency. It is often the result of trauma but is rarely reported in association with cervical disk herniation in patients without antecedent injury. METHODS Case report. FINDINGS This 75-year-old man presented with acute paraplegia due to severe compression of the spinal cord by herniation of the C4-C5 cervical disk. He underwent emergency diskectomy and anterior fusion. Postoperatively, his neurologic functions improved gradually. CONCLUSIONS Cervical disk herniation should be considered in the differential diagnosis of nontraumatic acute paraplegia. Pre-existing narrowed canal is an important predisposing factor and excessive neck movements are believed to be triggering factors. Immediate early decompressive surgery is recommended to avoid irreversible progression of neurologic deficit.
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Affiliation(s)
- Chao Liu
- Department of Orthopaedics, Sir Run Run Shaw Hospital, Institute of Clinical Medicine of Zhejiang University, Hang Zhou, China
| | - Yue Huang
- Department of Orthopaedics, Sir Run Run Shaw Hospital, Institute of Clinical Medicine of Zhejiang University, Hang Zhou, China
| | - Hong-Xin Cai
- Department of Orthopaedics, Sir Run Run Shaw Hospital, Institute of Clinical Medicine of Zhejiang University, Hang Zhou, China
| | - Shun-Wu Fan
- Department of Orthopaedics, Sir Run Run Shaw Hospital, Institute of Clinical Medicine of Zhejiang University, Hang Zhou, China,Please address correspondence to Shun-Wu Fan, MD, Sir Run Run Shaw Institute of Clinical Medicine of Zhejiang University, #3 East Qingchun Rd, Hang Zhou 310016, China, +86(0)571 8600 6297, +86(0)571 8609 0073, e-mail:
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Spinal extradural angiolipoma: report of two cases and review of the literature. EUROPEAN SPINE JOURNAL : OFFICIAL PUBLICATION OF THE EUROPEAN SPINE SOCIETY, THE EUROPEAN SPINAL DEFORMITY SOCIETY, AND THE EUROPEAN SECTION OF THE CERVICAL SPINE RESEARCH SOCIETY 2009; 18:324-35. [PMID: 19127373 DOI: 10.1007/s00586-008-0858-8] [Citation(s) in RCA: 50] [Impact Index Per Article: 3.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Received: 09/06/2008] [Revised: 10/26/2008] [Accepted: 12/10/2008] [Indexed: 10/21/2022]
Abstract
Spinal angiolipomas are benign uncommon neoplasm composed of mature lipocytes admixed with abnormal blood vessels. They account for only 0.04-1.2% of all spinal tumors. We report two cases of lumbar extradural angiolipoma and review previously reported cases. We found 118 cases of spinal epidural angiolipoma (70 females and 48 males; age range 1.5-85 years, mean 44.03) spanning from 1890 to 2006. Prior to diagnosis 40.6% of the patients had weakness of the lower limbs. The interval between the initial symptoms and tumor diagnosis ranged from 1 day to 17 years (mean 20.2 months). Except for four cases diagnosed at autopsy, 109 patients underwent surgery and gross-total resection was performed in 79 cases (72.4%). Spinal angiolipomas are tumors containing angiomatous and lipomatous tissue, predominantly located in the mid-thoracic region. All angiolipomas show iso- or hyperintensity on T1-weighted images and hyperintensity on T2-weighted images and most lesions enhance with gadolinium administration. The treatment for spinal extradural angiolipomas is total surgical resection and no adjuvant therapy should be administered.
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Park JH, Jeon SR, Rhim SC, Roh SW. Lumbar spinal extradural angiolipoma: case report and review of the literature. J Korean Neurosurg Soc 2008; 44:265-7. [PMID: 19096690 DOI: 10.3340/jkns.2008.44.4.265] [Citation(s) in RCA: 16] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/22/2008] [Accepted: 09/17/2008] [Indexed: 02/08/2023] Open
Abstract
Angiolipomas in the lumbar spinal region are extremely rare. The present report describes the identification of such a tumor and its removal, and discusses the tumor characteristics and prognosis. A 74-year-old woman was presented with a 5-month history of lower back pain. Severe radiculopathy was experienced in the left leg for 5 days prior to the presentation, and there were no neurological deficits. Magnetic resonance (MR) images showed an approximately 3.5 cm heterogeneously enhanced and elongated mass at the left L5-S1 level. A portion of the mass appeared with high signal intensity on T2-weighted MR images, with low signal intensity on T1-weighted images, and with high signal intensity on T1 fat suppression enhancement images. Resection of the tumor was approached via an L5 and S1 laminectomy. A fibrous sticky yellowish hypervascular tumor was identified. Histological study revealed the tumor as an angiolipoma. Symptoms were relieved after tumor excision, and there were no neurological sequelae. Although extremely rare, lumbar epidural angiolipoma should be considered in the differential diagnosis of lumbar spinal epidural lesions. The prognosis after surgical management of this lesion is favorable.
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Affiliation(s)
- Jin Hoon Park
- Department of Neurological Surgery, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
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Yen HL, Tsai SC, Liu SM. Infiltrating spinal angiolipoma. J Clin Neurosci 2008; 15:1170-3. [PMID: 18701297 DOI: 10.1016/j.jocn.2007.07.084] [Citation(s) in RCA: 15] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/02/2007] [Revised: 07/18/2007] [Accepted: 07/20/2007] [Indexed: 12/12/2022]
Abstract
Infiltrating angiolipomas are rarely encountered in the spine. We present a case involving a 71-year-old man with a dorsal epidural angiolipoma at the T5-T7 level. The tumor involved the T5-T6 vertebral bodies and left pedicle. The patient presented with acute paraparesis and MRI showed a homogeneously hyphointense lesion on T1-weighted images. The epidural component of the tumor was removed via laminectomy to achieve adequate cord decompression. The patient was symptom-free at a 2-year follow-up. This report emphasizes the unusual clinical presentation and MRI features of an infiltrating spinal angiolipoma and discusses therapeutic management options.
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Affiliation(s)
- Han-Lin Yen
- Department of Neurosurgery, Tainan Municipal Hospital, No. 670, Chongde Rd., East District, Tainan City 701, Taiwan.
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Akhaddar A, Albouzidi A, Elmostarchid B, Gazzaz M, Boucetta M. Sudden onset of paraplegia caused by hemorrhagic spinal epidural angiolipoma. A case report. EUROPEAN SPINE JOURNAL : OFFICIAL PUBLICATION OF THE EUROPEAN SPINE SOCIETY, THE EUROPEAN SPINAL DEFORMITY SOCIETY, AND THE EUROPEAN SECTION OF THE CERVICAL SPINE RESEARCH SOCIETY 2008; 17 Suppl 2:S296-8. [PMID: 18228054 DOI: 10.1007/s00586-008-0591-3] [Citation(s) in RCA: 34] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Received: 06/10/2007] [Revised: 08/22/2007] [Accepted: 12/22/2007] [Indexed: 12/29/2022]
Abstract
Spinal epidural angiolipoma is a rare benign tumor containing vascular and mature adipose elements. A slow progressive clinical course was mostly presented and rarely a fluctuating course during pregnancy. The authors report the original case of spontaneous spinal epidural bleeding resulting from thoracic epidural angiolipoma who presented with hyperacute onset of paraplegia, simulating an extradural hematoma. The patient was admitted with sudden non-traumatic hyperacute paraplegia during a prolonged walk. Neurologic examination showed sensory loss below T6 and bladder disturbances. Spinal MRI revealed a non-enhanced heterogeneous thoracic epidural lesion, extending from T2 to T3. A bilateral T2-T4 laminectomy was performed to achieve resection of a lipomatous tumor containing area of spontaneous hemorrhage. The postoperative course was uneventful with complete neurologic recovery. Histologic examination revealed the tumor as an angiolipoma. Because the prognosis after rapid surgical management of this lesion is favorable, the diagnosis of spinal angiolipoma with bleeding should be considered in the differential diagnosis of hyperacute spinal cord compression.
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Affiliation(s)
- Ali Akhaddar
- Department of Neurosurgery, Mohammed V Military Teaching Hospital, University of King Mohammed V-Souissi, Rabat, Morocco.
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Abstract
BACKGROUND/OBJECTIVE Spinal angiolipoma (SAL) is an uncommon clinico-pathological entity. DESIGN Single case report. METHODS Retrospective data analysis. FINDINGS An obese woman with a 1-year history of progressive spastic paraparesis and acute deterioration underwent magnetic resonance imaging of the thoracic spine, the results of which suggested a tumor compressing the thoracic spinal cord. The histopathological examination of the completely resected tumor revealed an epidural angiolipoma. CONCLUSIONS This case report offers a reminder that SAL should be considered in the differential diagnosis of long-standing, slowly progressive paraparesis. It remains unclear whether an increased body mass index might be a contributing factor to the development of SAL.
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Affiliation(s)
- Marcel Hungs
- Departament of Neurology, University of California-Irvine, Orange, California 92868, USA.
| | - Laura S Paré
- 2Department of Neurological Surgery, University of California–Irvine, Orange, California
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Vilela P, Saraiva P, Goulão A. Intracranial angiolipoma as cause of subarachnoid haemorrhage. Case report and review of the literature. Neuroradiology 2005; 47:91-6. [PMID: 15662498 DOI: 10.1007/s00234-004-1290-3] [Citation(s) in RCA: 10] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/01/2004] [Accepted: 08/12/2004] [Indexed: 10/25/2022]
Abstract
A 33-year-old female with a longstanding history of seizures was admitted to our hospital with subarachnoid haemorrhage (SAH). Computed tomography (CT), magnetic resonance imaging (MRI) and digital subtraction angiography (DSA) depicted a vascular fat-containing lesion overlying a right frontal cortical polymicrogyria. The diagnosis of angiolipoma was established. Conservatory management was undertaken with full recovery. She has been followed for 5 years since, with neither re-bleeding nor morphologic change of the lesion. This is a rare intracranial lesion, with only 11 intracranial angiolipomas published in the literature, and is the first case reported which is associated with SAH caused by this lesion.
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Affiliation(s)
- P Vilela
- Serviço de Neurorradiologia, Hospital Garcia de Orta, Av. Prof. Torrado Silva. Pragal, 2801-591, Almada, Portugal.
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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. [PMID: 15123997 DOI: 10.1038/sj.sc.3101535] [Citation(s) in RCA: 22] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/28/2022]
Abstract
STUDY DESIGN Case report and review of the literature. OBJECTIVES To describe two patients with angiolipoma in the ventral aspect of the lumbar epidural space, to discuss the clinical, radiologic, and surgical features of these lesions, and to review previously reported cases. SETTING Rome, Italy. METHODS Two cases, a 60-year-old man and a 54-year-old woman presented with lumbar-sciatic pain but with no abnormal neurological signs. Investigation (CT and MRI) demonstrated lumbar tumours. RESULTS Laminectomy and excision of the tumors were performed, and symptoms improved immediately. CONCLUSIONS Magnetic resonance imaging with suppression fat sequences allows the recognition of these lesions. The prognosis after surgical removal of spinal angiolipoma is favorable.
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Affiliation(s)
- G Rocchi
- Department of Neurological Sciences, Neurosurgery, University of Rome La Sapienza, Italy
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do Souto AA, Domingues FS, Chimelli L, Lemos AM. Lumbosacral angiolipoma: case report. ARQUIVOS DE NEURO-PSIQUIATRIA 2003; 61:269-73. [PMID: 12806510 DOI: 10.1590/s0004-282x2003000200021] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/22/2022]
Abstract
We present a case of a 46-year old woman with a ventral epidural angiolipoma at the lumbosacral level with erosion of the sacrum. About ninety cases of spinal angiolipomas have been previously described in the literature, most of them situated on the thoracic region, dorsal to the dural sac. Angiolipomas can be radically excised with a good prognosis even in the presence of bone erosion. We did not find any other angiolipoma at the sacral level surgically explored in the review of the literature.
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Affiliation(s)
- Antonio Aversa do Souto
- Servi o de Neurocirurgia, Hospital Universit rio Clementino Fraga Filho, Universidade Federal do Rio de Janeiro, Rio de Janeiro, RJ, Brasil.
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Pinto-Rafael JI, Vázquez-Barquero A, Martín-Laez R, García-Valtuille R, Sanz-Alonso F, Figols-Guevara FJ, Pérez-Castro MC, Abascal F, Cerezal L. [Spinal angiolipoma: case report]. Neurocirugia (Astur) 2002; 13:321-5. [PMID: 12355656 DOI: 10.1016/s1130-1473(02)70609-x] [Citation(s) in RCA: 12] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/03/2023]
Abstract
Spinal extradural angiolipomas are rare benign tumours, accounting for only 0.14-1.2% of all spinal neoplasms. They are usually localized within the thoracic extradural space of the spinal canal and their common clinical presentation is myelopathy, mainly in the way of a slowly progressive paraparesis and sometimes in an acute form. We report the case of a 85 year old man who complained of bilateral lower limb weakness with sphincter disturbance of acute instauration. The MRI showed a posterior extradural lesion at L1-L2 level that compressed conus medularis and cauda equina. He underwent urgent surgical treatment and we achieved total resection of the lesion via a L1-L2 laminectomy. The pathological examination confirmed the tumour as an angiolipoma.
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Affiliation(s)
- J I Pinto-Rafael
- Servicio de Neurocirugía, Hospital Universitario Marqués de Valdecilla, Santander
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Abstract
BACKGROUND Spinal epidural angiolipoma is a rare cause of spinal cord compression. We present a case and review the clinical presentation, radiological appearance, pathological aspects and treatment of this distinct clinico-pathological entity. METHODS A case of a 46-year-old woman with a five-month history of progressive myelopathy affecting her lower extremities is presented. CT and MRI revealed a large epidural fat-containing mass compressing the spinal cord dorsally at the T7-T8 level. A laminectomy was performed with gross total resection of the lesion. RESULTS The patient's neurologic symptoms improved postoperatively. A two-year follow-up period has revealed no signs of tumor recurrence and no neurological deficit. CONCLUSION The diagnosis of spinal angiolipoma should be considered in the differential diagnosis of spinal cord compression. Magnetic resonance imaging is the investigation of choice. The surgical objective is complete excision but, for anterior lesions involving bone, an overly aggressive approach should be tempered by an awareness of the overall indolent natural history of so-called "infiltrating" spinal angiolipomas that are only partially excised.
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Affiliation(s)
- D R Fourney
- Division of Neurosurgery, Royal University Hospital, Saskatoon, Saskatchewan, Canada
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21
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Abstract
Angiolipomas are neoplasms composed of mature adipocytes admixed with abnormal vascular elements. They are most commonly found in the subcutaneous tissue of the trunk and extremities, but other sites have been reported. The craniospinal axis is an uncommon but significant site. An extensive review of the literature is conducted. We summarize 94 cases of angiolipomas in the central nervous system (CNS) in 92 patients, including five in our own series, to highlight the most prominent features of these tumors. The increasing number of cases of CNS angiolipoma in the era of magnetic resonance imaging raises the question of the rarity of these lesions.
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Affiliation(s)
- N Andaluz
- Department of Neurosurgery, The Neuroscience Institute, Cincinnati, OH, USA
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