Session: OR02-Diagnosing and Treating Cortisol Excess and Deficiency
W190 (McCormick Place West Building)
Transsphenoidal surgery (TSS) is first-line treatment for most patients with Cushing’s disease (CD); however, post-surgical recurrence is an ongoing concern that may arise years after initial remission. While there are currently no standardized guidelines for post-TSS follow-up in patients with CD, conventional tools for assessing recurrence include urinary free cortisol (UFC) and 1 mg overnight dexamethasone suppression testing (DST). More recently, late-night salivary cortisol (LNSC) has also gained recognition as a diagnostic tool for both de novo and recurrent CD. However, timely recognition and treatment may be delayed when biochemical results are discordant.
To study this diagnostic difficulty more closely we retrospectively reviewed clinic records from 2/2006 to 7/2013 at our institution to identify patients with proven post-TSS CD recurrence and normal UFC.
We identified 10 female patients with recurrent CD who had normal UFC at time of recurrence. All patients underwent primary TSS; mean time to CD recurrence was 3.5 years (range 1–9) after initial clinical and biochemical remission. Five patients had evidence of tumor on MRI at time of recurrence. All patients had normal renal function and underwent testing with LNSC, DST, and UFC. All 10 patients had normal UFC; however, DST was abnormal in 8, and all 10 had ≥1 elevated LNSC measurement. All patients with available follow-up data (9/10) demonstrated significant improvement upon treatment. Two patients underwent repeat TSS; in both cases, pathology confirmed ACTH-staining pituitary adenoma and the patient achieved clinical and biochemical remission. Six patients received pharmacological therapy (5 with mifepristone, 1 with cabergoline) and all have shown clinical and/or biochemical improvement. One patient, who had normal UFC and DST with abnormal LNSC, underwent bilateral adrenalectomy and, among other clinical improvements, experienced a 55-kg weight loss and resolution of hypertension.
While UFC is generally considered an accurate diagnostic tool for CD, our case series indicates that UFC is not a reliable marker of recurrence and supports the hypothesis that LNSC may be more sensitive than UFC or DST for detection of recurrent CD. Prompt intervention when LNSC is elevated, despite normal UFC, may yield significant clinical benefit for many patients with CD.
Disclosure: TBC: Consultant, Corcept. JWF: Consultant, Corcept, Consultant, Novartis Pharmaceuticals. Nothing to Disclose: BRJ
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