Background: Spinal epidural lipomatosis (SEL) is an excessive fat deposition within the epidural space of spinal canal leading to compression of nervous structures and neurological manifestations. Exogenous glucocorticoid administration is the major cause of symptomatic SEL. The reported patient had SEL related to endogenous Cushing syndrome which is among few reported cases in the literature. Case presentation: A 14 year girl with features of Cushing syndrome had severe back pain and magnetic resonance imaging of spine showed SEL. Subsequently, computed tomography of abdomen showed right adrenal adenoma.The patient underwent adrenalectomy which improved her signs of hypercortisolism and SEL. Conclusion: In view of severe neurological deficits and therapeutic consequences, SEL should be considered in all patients with Cushing syndrome who present with symptoms of spinal cord compression. Correction of underlying endocrinopathy is imperative in management of SEL.

Keywords: Spinal epidural lipomatosis, Cushing syndrome, adrenal adenoma, case report