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Dewa Putu Wisnu Wardhana1, Christopher Lauren2, Rohadi Muhammad Rosyidi3
  1. Department of Surgery, Neurosurgery Division, Universitas Udayana, Academic Hospital of Universitas Udayana, Bali, Indonesia
  2. Department of Surgery, Neurosurgery Division, Universitas Udayana, Prof. Dr. I.G.N.G. Ngoerah General Hospital, Denpasar, Bali, Indonesia
  3. Department of Neurosurgery, Medical Faculty of Mataram University, West Nusa Tenggara General Hospital, Mataram, Indonesia

Correspondence Address:
Dewa Putu Wisnu Wardhana, Department of Surgery, Neurosurgery Division, Universitas Udayana, Academic Hospital of Universitas Udayana, Denpasar, Bali, Indonesia.

DOI:10.25259/SNI_400_2024

Copyright: © 2024 Surgical Neurology International This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 License, which allows others to remix, transform, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.

How to cite this article: Dewa Putu Wisnu Wardhana1, Christopher Lauren2, Rohadi Muhammad Rosyidi3. Multilevel thoracic spinal epidural angiolipoma. 26-Jul-2024;15:260

How to cite this URL: Dewa Putu Wisnu Wardhana1, Christopher Lauren2, Rohadi Muhammad Rosyidi3. Multilevel thoracic spinal epidural angiolipoma. 26-Jul-2024;15:260. Available from: https://surgicalneurologyint.com/surgicalint-articles/13010/

Date of Submission
23-May-2024

Date of Acceptance
01-Jul-2024

Date of Web Publication
26-Jul-2024

Abstract

Background: Spinal epidural angiolipomas (SEAL) are rare benign tumors composed of mature adipose tissue and abnormal blood vessels. SEALs account for 0.04–1.2% of all spinal tumors and 2–3% of spinal extradural lesions. They are usually found in the mid-thoracic dorsal extradural region. Here, we present the case of a 52-year-old female with a rare SEAL involving multiple thoracic levels.

Case Description: A 52-year-old female presented with mid-thoracic pain and paraparesis (motor strength 3/5 proximal/distal). Magnetic resonance imaging (MRI) documented a well-demarcated, homogeneous dorsal epidural mass extending from T7 to T10. She underwent a laminectomy for tumor resection and decompression. The tumor, measuring 6 × 2 × 0.5 cm, consisted of mature adipose tissue and abnormal blood vessels. Postoperatively, the patient’s motor deficit fully resolved within 6 months.

Conclusion: SEAL is a rare extradural dorsal thoracic spine tumor that can be readily diagnosed with MRI. The gold standard of treatment is gross total resection through a laminectomy.

Keywords: Epidural angiolipoma, Intrathoracic, Neurosurgery, Oncology, Spine

INTRODUCTION

Spinal epidural angiolipoma (SEAL) is a rare benign tumor that typically occurs in the dorsal thoracic spinal region. SEALs are comprised of mature adipose tissue and abnormal blood vessels.[ 10 ] They are usually found in the subcutaneous areas of the trunk and extremities, representing between 0.04% and 1.2% of all spinal tumors and 2–3% of all spinal extradural lesions, most commonly in the dorsal mid-thoracic spine.[ 2 , 5 , 7 ] Here, a 52-year-old female presented with back pain and paraparesis attributed to magnetic resonance (MR)-documented T7–T10 dorsal thoracic lesions. Following a laminectomy for tumor resection, the patient was neurologically intact within 6 postoperative months.

CASE DESCRIPTION

Clinical history

A 52-year-old female presented with 3 months of increasing back pain (Visual Analog Scale 5/10 and Karnofsky performance status 80/100) and a progressive 1-month paraparesis (motor 3/5 proximal/distal) that worsened over the past week. Thoracic MR imaging (MRI) showed a lesion surrounding vertebrae T7–T10. On T1-weighted imaging, it appeared as a well-demarcated, homogeneous dorsal epidural compressive mass. On T2-weighted imaging, it was high-intensity and distinct from surrounding structures [ Figure 1a - c ]. Spinal MR myelography confirmed a complete spinal block at the T10 level [ Figure 1d ].


Figure 1:

Magnetic resonance imaging showing a focal mass around the dorsal extradural space from thoracic 7–10. (a and b), Coronal view (a) T1-weighted and (b) T2-weighted images showing the mass (white arrow). (c) Axial T2-weighted images showing the dorsally located mass (white arrow). (d) Magnetic resonance myelography showing the blockage site (white arrow).

 

Surgery, pathology, and outcome

Using a C-arm, the patient underwent a laminectomy from T7 to T9 [ Figure 2 ]. A dorsal extradural tumor (6 × 2 × 0.5 cm) was encountered, which had a reddish-yellow hue and clear boundaries from surrounding structures [ Figure 3a ]. Microscopic examination revealed a mixture of mature adipose tissue and blood vessels (anomalous vascular channels) consistent with a SEAL [ Figure 3b and 3c ]. The histopathological findings are summarized in Table 1 . The patient was discharged on the 4th postoperative day with 4/5 strength in the lower extremities; 6 months after surgery, strength was 5/5 bilaterally.


Figure 2:

Intraoperative view. (a) A C-arm was used to locate the incision site. (b) After the laminectomy, a yellowish mass was found to extend from multiple thoracal levels.

 

Figure 3:

Histopathological findings. (a) Macroscopic view of the mass, showing a reddish-yellow color. (b) Microscopic view shows a mixture of mature adipose cells and blood vessels (Hematoxylin and eosin (HE) stain; ×100 magnifications). (c) The vascular component comprises small, thin-walled vessels surrounded by mature adipose cells (HE stain; ×400 magnifications).

 

Table 1:

Histopathological findings.

 

DISCUSSION

Spinal epidural angiolipoma (SEAL) tumors are rare, predominantly occurring in the dorsal thoracic spine,[ 5 , 9 ] and more frequently diagnosed in women between 40 and 60 years old.[ 2 , 10 ] A summary of the various cases of SEAL that have been reported in the literature is shown in Table 2 . These tumors are classified into two types: noninfiltrating and infiltrating.[ 4 ] The noninfiltrating type, which is more common, typically originates in the posterior epidural space and is usually encapsulated. In contrast, the infiltrating type, which is less common, generally starts in the anterior epidural space and may extend into the vertebral bodies. Both types predominantly affect the thoracic region, generally spanning three to four vertebrae. Symptoms commonly include progressive paraparesis, back or flank pain, possible radiculopathy, sensory changes in the lower extremities, and hyperreflexia.[ 2 , 10 ]


Table 2:

Summary of the various cases of spinal epidural angiolipoma.

 

On MRI, SEALs often appear iso- to hyperintense on T1-weighted images and show hyperintensity on T2-weighted images.[ 3 ] They present as well-defined extradural masses and may appear as soft, reddish, and possibly encapsulated masses upon macroscopic examination, easily separable from the dura mater.[ 8 ] The gold standard treatment for SEALs is gross total resection, which can be achieved through anterior or posterior surgical approaches. The infiltrating type might require a more extensive resection[ 5 ] and sometimes preoperative embolization due to potential high vascularity.[ 6 ] Unlike other tumors, adjuvant therapies such as chemotherapy or radiotherapy are not recommended for SEALs due to their benign nature and low recurrence rate.[ 1 ]

In reported cases, patients generally recover fully following complete tumor resection.

CONCLUSION

SEAL is a rare dorsal extradural spinal tumor that should be treated with gross total resection.

Ethical approval

Institutional Review Board approval is not required.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

Use of artificial intelligence (AI)-assisted technology for manuscript preparation

The authors confirm that there was no use of artificial intelligence (AI)-assisted technology for assisting in the writing or editing of the manuscript and no images were manipulated using AI.

Disclaimer

The views and opinions expressed in this article are those of the authors and do not necessarily reflect the official policy or position of the Journal or its management. The information contained in this article should not be considered to be medical advice; patients should consult their own physicians for advice as to their specific medical needs.

References

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2. 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

3. Ghanta RK, Koti K, Dandamudi S. Spinal epidural angiolipoma: A rare cause of spinal cord compression. J Neurosci Rural Pract. 2012. 3: 341-3

4. 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

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6. Maduri R, Belouaer A, Brouland JP, Ecker T, Messerer M. Spinal angiolipomas in pregnancy: Natural history and surgical treatment. Clin Neurol Neurosurg. 2019. 178: 25-30

7. Nanassis K, Tsitsopoulos P, Marinopoulos D, Mintelis A, Tsitsopoulos P. Lumbar spinal epidural angiolipoma. J Clin Neurosci. 2008. 15: 460-3

8. Rkhami M, Kedous MA, Achoura S, Zehani A, Bahri K, Zammel I. Epidural angiolipoma: A rare cause of spinal cord compression. Int J Surg Case Rep. 2018. 45: 72-6

9. Somrani K, Ammar H, Gargouri F, Mansouri N, Yedeas MD, Chkili R. A rare case report of sacral epidural angiolipoma: Diagnosis and treatment. Surg Neurol Int. 2022. 13: 38

10. Wang FF, Wang S, Xue WH, Cheng JL. Epidural spinal angiolipoma: A case series. BMC Res Notes. 2017. 10: 128

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