Perforated duodenal ulcer may predict disease progression in subclinical Cushings syndrome
MO Sharfi, AO Adjene, A Sharma, AA Panahloo & LJ Seal
Subclinical Cushings syndrome (CS) has not been adequately characterised and the natural history is unknown. We report two cases of subclinical CS that presented as perforated duodenal ulcers (DU) and in both cases the disease rapidly progressed to overt CS.
A 60-year-old women presented with perforated DU requiring laparotomy and repair. Abdominal CT scanning demonstrated a right adrenal mass 4.2 cm. There were no clinical signs to suggest CS. Formal dexamethasone suppression testing was consistent with adrenal CS (LDDST 93 nmol/l HDDST103 nmol/l) with basal ACTH 9 nmol/l. A diagnosis of subclinical CS was made. Over the next 18 months the patient developed central obesity, thinning of the skin and facial plethora converting to overt CS. She has been commenced on ketoconazole and is currently awaiting adrenalectomy.
A 69-year-old women presented with perforated DU requiring open repair. CT scanning demonstrated a 3.8 cm right adrenal. There were no clinical signs of CS. Dexamethasone suppression testing suggested adrenal CS, (LDDST 175 nmol/l), with basal ACTH <5 nmol/l. A diagnosis of subclinical CS was made. Over the next 24 months, she developed central obesity, thinning of the skin, and proximal muscle weakness. She has also required antihypertensive medications to normalise her blood pressure. She is also awaiting adrenalectomy.
In subclinical CS the progression to overt CS is considered to be rare. We report two cases that progressed to clinical disease within 1824 months. Both cases presented with perforated DU, which could suggest that this is a marker of early CS or a predictor of disease progression in subclinical CS.
Perforated DU may be a prognostic indicator for disease in subclinical CS.