Osilodrostat-associated Adrenal Gland Shrinkage: A Case Series of Patients With ACTH-dependent Cushing's Syndrome
Authors' conclusion
We found that in selected patients with ACTH-dependent CS, osilodrostat can induce significant adrenal shrinkage, with or without AI. Further confirmation by larger studies of different CS types and monitoring for AI is required for all patients.
Study context
- Population
- Patients with ACTH-dependent Cushing's syndrome (CS) treated with osilodrostat >3 months, with computed tomography (CT) before and after osilodrostat; Oregon Health and Science University, 2000–2025
- Participants
- 10 patients
- Exposure
- Adrenal volume before vs after osilodrostat
- Osilodrostatn = 10
- Outcome
- Percent reduction in adrenal volume (AV) before vs after osilodrostat initiation
Methodological detailsnoninterventional, longitudinal, single-centre
- Noninterventional
- Longitudinal
- Single-centre
Main finding
As reported in the abstract
Adrenal volume · median of 19 months
- Adrenal volume (AV) decrease
- 46.7 ± 22.2%
- AV before osilodrostat
- 25.5 ± 9.9 mL
- AV after osilodrostat
- 12.7 ± 6.4 mL
- P for change
- P < .001
Observational data: associations, not effects.
Further findings
Correlation of AV reduction with maximum osilodrostat dose
- R
- 0.626
- P
- P = 0.027
Osilodrostat doses · over 23 months
- Mean starting dose
- 7.7 mg/day
- Mean maximum dose
- 13.8 mg/day
- Mean final dose
- 5.9 mg/day
- Block-and-replace regimen
- 4 patients
Safety
Adrenal insufficiency
- Adrenal insufficiency (AI) developed
- 5 patients
Original publication
Osilodrostat-associated Adrenal Gland Shrinkage: A Case Series of Patients With ACTH-dependent Cushing's Syndrome. J Clin Endocrinol Metab. Published 7 October 2025. doi:10.
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