Recurrence of hypercortisolism in patients after bilateral adrenalectomy for Cushing’s disease is extremely rare.
We present a rare case of a 27-year-old man who previously underwent bilateral adrenalectomy for Cushing’s disease with complete clinical resolution. Cushingoid features recurred 12 years later, along with bilateral testicular enlargement. Hormonal tests confirmed ACTH-dependent Cushing’s. Surgical resection of the testicular tumors led to clinical and biochemical remission.
We report bilateral testicular tumors occurring in a patient with recurrent Cushing’s disease 12 years after bilateral adrenalectomy. Using mRNA expression analysis and steroid metabolome profiling, the tumors demonstrated both adrenocortical and gonadal steroidogenic properties, similar to testicular adrenal rest tumors found in patients with congenital adrenal hyperplasia. This suggests the presence of pluripotent cells even in patients without CAH.
- 1Department of Medicine, Division of Endocrinology, Radboud University Medical Centre, 6500 HB Nijmegen, The Netherlands.
- 2Department of Endocrinology, Changi General Hospital, Singapore 529889, Singapore.
- 3Department of Paediatrics, Division of Endocrinology, Radboud University Medical Centre, 6500 HB Nijmegen, The Netherlands.
- 4Department of Laboratory Medicine, Radboud University Medical Centre, 6500 HB Nijmegen, The Netherlands.
- 5Department of Pathology, Radboud University Medical Centre, 6500 HB Nijmegen, The Netherlands.
- 6Department of Radiology, Radboud University Medical Centre, 6500 HB Nijmegen, The Netherlands.
- 7Institute of Metabolism and Systems Research, University of Birmingham, Birmingham N15 2TT, United Kingdom.
- 8Centre for Endocrinology, Diabetes, and Metabolism, Birmingham Health Partners, Birmingham B15 2TH, United Kingdom.
– See more at: http://press.endocrine.org/doi/abs/10.1210/jc.2016-2702#sthash.F4lfWg9j.dpuf
Related
Filed under: adrenal, Cushing's, Rare Diseases, Treatments | Tagged: abstract, ACTH, Adrenocorticotropic hormone, bilateral laparoscopic adrenalectomy, BLA, recurrence, testicular tumor |
Leave a Reply