Aneurysmal bone cyst of thoracic spine with neurological deficit and its recurrence treated with multimodal intervention – A case report
- Department of Orthopaedics, Chettinad Hospital and Research Institute, Kanchipuram, Tamil Nadu, India.
DOI:10.25259/SNI_466_2020Copyright: © 2020 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, tweak, 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: B. Yogesh Kumar, R. Thirumal, S. G. Chander. Aneurysmal bone cyst of thoracic spine with neurological deficit and its recurrence treated with multimodal intervention – A case report. 05-Sep-2020;11:274
How to cite this URL: B. Yogesh Kumar, R. Thirumal, S. G. Chander. Aneurysmal bone cyst of thoracic spine with neurological deficit and its recurrence treated with multimodal intervention – A case report. 05-Sep-2020;11:274. Available from: https://surgicalneurologyint.com/surgicalint-articles/10243/
Background: Aneurysmal bone cysts (ABCs) are rare, representing about 1% of primary bone tumors, and 15% of all primary spine/sacral tumors. Notably, when they are located in poorly accessible regions such as the spine and pelvis, their management may be challenging. Treatment options include selective arterial embolization (SAE), curettage, en bloc excision with reconstruction, and radiotherapy.
Case Description: A 16-year-old male presented with 2 months of mid back pain, left-sided thoracic radiculopathy, and left lower limb weakness (MRC – 3/5). MR imaging revealed an expansile, lytic lesion involving the T9 vertebral body, and the left-sided posterior elements resulting in cord compression. He underwent SAE followed by intralesional excision, bone grafting, and a cage – instrumented fusion. ABC was diagnosed from the biopsy sample. Postoperatively, the pain was reduced, and he was neurologically intact. Five months later, he presented with a new lesion that was treated with repeated SAE and three doses of zoledronic acid. At the end of 2 years, the subsequent, MRI and CT studies documented new bone formation in the lytic areas, with healing of lesion; additionally, he clinically demonstrated sustained pain relief.
Conclusion: Here, we emphasized the importance of surgery for patients with ABC who develop focal neurological deficits. Treatment options should include SAE with bisphosphonate therapy for lesions that recur without neurological involvement.
Keywords: Aneurysmal bone cyst, Bisphosphonate therapy, Recurrence, Selective arterial embolization, Surgical excision
Aneurysmal bone cysts (ABCs) are rare, locally aggressive lesions that occur most frequently in the first or second decades of life.[
A 16-year-old male presented with 2 months of mid back pain and left-sided radiculopathy with the acute onset of the left lower limb monoparesis (MRC – 3/5). Other accompanying neurological findings included a T10 sensory level with loss of pin prick, temperature, vibration/position appreciation with hyperreflexia, and a left-sided positive Babinski response. The computed tomography (CT) scan of the thoracic spine demonstrated an expansile lytic lesion with the classical “egg shell layer” occupying the left side of T9 vertebral body destroying the lamina and pedicle with epidural extension [
Computed tomography of the spine. (a) Sagittal section showing typical expansile, osteolytic bony destruction of T9 vertebra with posterior epidural extension and cord compression. (b) Axial section showing lytic lesion with egg shell layer (marked with arrow) of the T9 body involving the left pedicle, lamina, and spinous process.
MRI of the spine. (a and b) T1- and T2-weighted sagittal image revealed a heterogeneous bony cystic mass with internal septation and fluid-fluid levels at the T9 vertebra. (c and d) Axial T1- and T2-weighted images showing large, expansile spinal lesion with multiple fluid levels typical for an aneurysmal bone cyst and cord compression.
SAE and surgery
The patient had SAE; intercostal feeders were embolized using coils and gel foam [
Grossly, the T9 tumor was a gray-red 3–4 cm fleshy mass containing multiple blood-filled cysts. Histopathological examination showed cavernous spaces filled with blood surrounded by fibrous septa with marked cellular proliferation of band fibroblasts, few spindle cells, and scattered giant cells consistent with the diagnosis of an ABC [
The pain was reduced, and his deficits improved significantly. Within 3 months, he was walking independently and performing routine activities.
At the 5th postoperative months, he presented with a new right-sided thoracic radiculopathy without any focal neurological deficits. The repeat CT scan showed recurrence of the lesion; it now involved the right side of the T9 vertebral body [
Computed tomography of the spine at the 5th month. (a) Sagittal section showing recurrence of lytic lesion at T9 vertebra with spinal stabilization. (b) Axial section showing lytic lesion at the right side of T9 vertebral body (marked with arrow) and cage with bone growth at previous left side lesion.
Computed tomography of the spine at end of 2 years. (a) Sagittal section showing complete bone T9 vertebra with intact stabilization and no signs of bony lysis. (b) Axial section showing peripheral sclerotic bone rim formation right side (marked with arrow) and bone formation inside the aneurysmal bone cysts mass.
ABCs predominantly occur at the second decade and have a slight preponderance for women.[
Treatment options for primary and recurrent lesions
Tumor recurrences managed with “en bloc” resection, although optimal for lesion control, may not be technically feasible due to – high intraoperative and postoperative morbidity for such extensive resections. Radiation therapy, although very effective, does introduce the risk of radiation-induced sarcoma/myelopathy. SAE may be used preoperatively and/or to treat local recurrences.[
Adjunctive use of denosumab versus bisphosphonate therapy
Denosumab is a human monoclonal antibody that binds the cytokine receptor activator of nuclear factor- kappa B ligand,[
We emphasized the importance of surgery for patients with ABC who have focal neurological deficits. However, for those with recurrent lesions without specific neurological findings, SAE with bisphosphonate therapy is effective alternatives to repeated surgical intervention.
The authors certify that they have obtained all appropriate patient consent.
Publication of this article was made possible by the James I. and Carolyn R. Ausman Educational Foundation.
There are no conflicts of interest.
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