Abstract

Background: Spinal epidural angiolipomas (SEALs) are rare, benign tumors composed of mature adipocytes and abnormal vasculature, representing only 0.04–1.2% of spinal tumors. Predominantly located in the thoracic epidural space, they often cause progressive neurological deficits due to spinal cord compression, though acute deterioration may occur from hemorrhage or thrombosis. Misdiagnosis is common, and evidence-based management remains limited.

Case Description: a 74-year-old woman presented with a 3-month history of progressive paraparesis, gait instability, and left-sided hyperreflexia. Spinal magnetic resonance ımaging revealed an 11 cm posterior epidural mass extending from T3 to T7, causing severe cord compression with myelopathic signal changes. The patient underwent T4–T6 laminectomy with gross total resection of the highly vascular mass. Histopathology confirmed the diagnosis of SEAL. Postoperatively, her symptoms improved, and she was discharged without complications.

Conclusion: This case highlights the importance of recognizing SEALs in differential diagnoses of spinal cord compression, emphasizing meticulous surgical excision for favorable outcomes. A literature review underscores the need for standardized management guidelines for these rare lesions.

Keywords: Angiolipoma, Epidural, Laminectomy, Spinal cord neoplasms

INTRODUCTION

Spinal epidural angiolipomas (SEALs) are exceedingly rare benign neoplasms composed of mature adipocytes and abnormally proliferating vascular elements.[ 4 , 2 ] They account for only 0.04–1.2% of all spinal axis tumors and 2–3% of spinal epidural tumors, with a predisposition to the posterior thoracic epidural space.[ 14 ] Patients with these tumors typically present with gradually progressive symptoms due to chronic spinal cord or nerve root compression; however, in some cases, rapid symptoms combined with neurological deterioration may be noticed secondary to intratumoral hemorrhage or venous thrombosis.[ 1 , 8 ]

The incomplete characterization of SEAL in the literature is associated with frequent misdiagnosis. At the same time, the lack of outcome-based studies also continues to limit the development of evidence-based surgical approaches.[ 16 ] In this case, we report our experience with a rare SEAL case detailing the clinical course, imaging characteristics, and histopathological findings in accordance with the [The CARE guidelines (for CAse REports)] CARE guideline criteria.[ 11 ]

CASE PRESENTATION

A 74-year-old woman presented with a 3-month history of progressively worsening bilateral lower extremity weakness, gait instability, and inability to walk independently. The patient has a history of hypertension and thyroid gland enlargement, has not undergone any surgeries, and is a housewife with unremarkable family history. On neurological examination, she was alert and oriented with a Glasgow coma scale score of 15/15. Motor examination revealed significant paraparesis, with a muscle strength of 3/5 in the left lower extremity and 4/5 in the right lower extremity. She also exhibited hyperreflexia (+3) on the left lower extremity and required assistance to ambulate.

Subsequent contrast-enhanced spinal magnetic resonance ımaging (MRI) has revealed an intraspinal-extradural space-occupying lesion extending from the level of the T3 vertebral body to the T7, located posteriorly and spanning approximately 11 cm in length. The lesion causes external compression on the thoracic spinal cord, resulting in significant cord deformation and myelopathic signal changes. Following contrast material administration, intense contrast enhancement is noted in the cranial portion of the lesion over a segment approximately 2 cm in length and no significant contrast enhancement is observed in the remaining parts of the lesion [ Figure 1 ].

Figure 1:

Preoperative spinal MRI scans demonstrating a thoracic epidural space-occupying lesion. (a and b) Contrast-enhanced WATER LAVA Flex sequence images in sagittal (orange arrow) (a) and axial (b) sections spanning the lesion. (c and d) T2-weighted images in sagittal (orange arrow) (c) and axial (d) sections across the lesion. (e) T1-weighted FSE sagittal image (orange arrow). (f) Sagittal STIR sequence image. An intraspinal-extradural space-occupying lesion is observed extending from the level of the T3 vertebral body to the T7, located posteriorly and spanning approximately 11 cm in length. The lesion causes external compression on the thoracic spinal cord, resulting in significant cord deformation and myelopathic signal changes. Following contrast material administration, intense contrast enhancement is noted in the cranial portion of the lesion over a segment approximately 2 cm in length (orange star). No significant contrast enhancement is observed in the remaining parts of the lesion. FSE: Fast spin echo, STIR: Short tau inversion recovery.

The patient then underwent a surgery to remove the lesion. A three-level laminectomy (T4–T6) was performed under intraoperative neuromonitoring to expose the affected region. Intraoperatively, a dark yellow-colored, moderately firm lesion surrounded by dense vascularization was identified [ Figure 2 ] and gross-total excision was achieved [ Figure 3 ]. No intraoperative complications were documented. Postoperatively, the general condition of the patient was good, and her neurological symptoms had improved. In the immediate postoperative examination, muscle strength in both the right and left lower extremities was assessed as 4/5, and the patient reported a reduction in symptoms. She was discharged in good condition on postoperative day 2. At the 10-day follow-up visit, the patient was walking normally, and bilateral lower extremity muscle strength was evaluated as 5/5. At the 2-month follow-up, she had no complaints. At follow-up after 10 days, she reported improvement in her symptoms. Histopathological analysis confirmed the diagnosis of SEAL, with negative immunohistochemistry for pancytokeratin, effectively excluding metastatic disease [ Figure 4 ].

Figure 2:

Intraoperative view of the (a) dark yellow-colored, moderately firm lesion (blue arrow). (b) The dense vascularization of the lesion and its surroundings should also be noted (blue arrow). The blue arrows in A and B indicates the visible margin of the tumor capsule, which was well-circumscribed and partially adherent to the dura.

Figure 3:

Postoperative spinal magnetic resonance ımaging demonstrating gross total resection of the lesion: (a) T2-weighted sagittal and (b) axial images, and (c) a sagittal STIR sequence image.

Figure 4:

Histopathological examination revealed an angiolipoma composed of mature adipose tissue (blue stars) and blood vessels ranging from capillary to cavernous structures (yellow arrows). Hematoxylin & Eosin Staining, under (a) 40x and (b) 100x magnification. Findings were found to be consistent with mixed capillary/cavernous angiolipoma.

DISCUSSION

This case underscores a rare manifestation of SEAL, a benign and infrequently encountered neoplasm that predominantly arises in the dorsal thoracic spine and is more commonly diagnosed in women between the ages of 40 and 60.[ 4 , 7 , 15 ] Our case contributes to the existing body of knowledge by documenting a particularly extensive thoracic lesion (T3–T7) in a 74-year-old female who presented with progressive paraparesis, ultimately managed with successful surgical excision.

Although the exact factors predisposing individuals to the development of angiolipomas remain unclear, some had proposed that these SEALs arise from the differentiation of pluripotential mesenchymal cells following certain stimuli, while others suggest a hormonal influence, as angiolipomas tend to occur more frequently around menopause and can vary in size with hormonal changes such as those seen during pregnancy.[ 3 ]

Although SEALs have been reported in the literature, the majority of documented cases, including those by Guzey et al., Nanassis et al., and Ghanta et al., occurred in patients in their 40s and 50s, with symptom durations ranging from 3 to 24 months [ Table 1 ].[ 5 , 6 , 10 ] In this context, our case stands out as both one of the oldest reported patients (74 years) and one with a relatively short symptom duration (3 months), suggesting a potentially more aggressive or rapidly symptomatic lesion. Similar to cases reported by Ghanta et al. and Hungs and Paré, our patient exhibited progressive motor deficits without sensory level or sphincter dysfunction as reported by other studies [ Table 1 ], underscoring the variability in SEAL clinical presentations even though when the lesion at the same level.[ 5 , 7 ]

Table 1:

Summary of reported cases of spinal thoracic epidural angiolipoma.

Radiologically, SEALs typically appear as T1-and T2-hyperintense lesions with variable contrast enhancement, though this can depend on the relative composition of adipose and vascular elements. Our patient’s MRI revealed a large, posteriorly located thoracic mass extending over multiple levels (T3–T7), with cord compression and signal changes indicative of myelopathy–radiologic findings that were consistent with those described in large thoracic lesions by Wang et al. and Rkhami et al.[ 12 , 15 ] The elongated morphology and posterior localization in our case is in line with previous findings, which report a predilection for the thoracic spine and posterior epidural space. In the differential diagnosis of such lesions, other epidural masses should be considered, including meningiomas, schwannomas, epidural lipomatosis, metastases, and lymphomas.

Histopathologically, our case confirmed the diagnosis of angiolipoma through identification of mature adipose tissue interspersed with abnormal blood vessels. This aligns with the consistent pathological descriptions across prior reports. Our negative pancytokeratin staining effectively excluded metastatic carcinoma, which is an important differential diagnosis when encountering epidural masses in older patients.[ 13 ]

Surgical resection remains the mainstay of treatment, with the goal of gross total excision and decompression of neural elements. As with nearly all prior cases, our patient experienced significant postoperative improvement following decompressive laminectomy and lesion removal. The complete excision, absence of complications, and early symptom relief within 10 days postoperatively support the literature’s consensus on favorable outcomes after surgery. This shows that the real challenge, as reflected in the literature and our case, lies not in treating the condition but in accurately diagnosing it preoperatively.

A key feature to highlight in this case is the diagnostic challenge that it presents. The condition can be difficult to recognize, often leading to a delayed diagnosis. Such delays are critical, as they may allow progressive myelopathy to develop, potentially resulting in irreversible neurological deficits. Therefore, when an epidural mass is detected, especially in the thoracic region, SEAL should be considered as a differential diagnosis to ensure timely intervention and prevent permanent neurological damage.

The primary strength of this case lies in its comprehensive characterization of SEAL through clinical, radiological, surgical, and pathological perspectives. It contributes valuable insight into the management and outcomes of a rarely encountered spinal tumor. The adherence to the CARE guidelines enhances the educational value and replicability of the findings for clinicians facing similar presentations.

However, the report is not without limitations. As a single case, generalizability remains limited, and the lack of long-term follow-up data restricts conclusions about sustained neurological recovery or recurrence risk. Furthermore, while the surgical approach proved effective in this case, optimal timing for surgery and possible extents of resection remain areas of uncertainty due to the scarcity of prospective or comparative studies and case series.

Data availability statement

The datasets generated during and/or analyzed during the present study are available from the corresponding author on reasonable request.

Statement of adherence to preprint policy

The authors declare that this paper has not been published as a preprint or in any other form elsewhere.

CONCLUSION

SEALs, though rare, should be considered in the differential diagnosis of myelopathy, particularly when imaging reveals a posteriorly located epidural mass. MRI is a valuable diagnostic tool, but definitive diagnosis requires histopathological confirmation. Early surgical intervention can result in neurological stabilization or improvement, reinforcing the importance of prompt recognition and treatment. Given the limited number of reported cases, further studies from the national databases such as the surveillance, epidemiology, and end results database are needed to better understand the natural history, optimal management strategies, and long-term outcomes associated with SEAL.

Ethical approval:

The 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 ınterest.

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

The authors confirm that there was no use of artificial ıntelligence (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.

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