We describe an exceptionally rare case of sustentaculoma of the left adrenal medulla in a 39-year-old Japanese male. Abdominal computed tomography and magnetic resonance imaging revealed a well-defined, smoothly bordered left adrenal tumor measuring 3.3× 2.3 cm in size. Histologically, the neoplasm comprised irregularly sized solid nests and collagenous fibrosis in the stroma. Immunohistochemical staining revealed positivity for vimentin, S-100 protein, CD56 (NCAM), and CD45 (LCA). No recurrence or metastasis has been observed for 16 years post-surgery. The CD45-positive finding in this case is unique as it is the first reported case in Japan.

Adrenal medulla

Sustentaculoma

Pheochromocytoma

Schwannoma, S-100 protein

CD45(LCA

Case report

© 2025 The Authors. Published by Elsevier Inc.