ECE2023 Eposter Presentations Pituitary and Neuroendocrinology (234 abstracts)
Diagnostic and therapeutic challenges in cyclic Cushing’s syndrome: A systematic literature review
Elisabeth Nowak 1 , Frederick Vogel 1 , Adriana Albani 1 , Leah Braun 1 , German Rubinstein 1 , Stephanie Zopp 1 , Katrin Ritzel 1 , Felix Beuschlein 1,2 , Marily Theodoropoulou 1 & Martin Reincke 1
1Department of Internal Medicine IV, University Hospital, LMU Munich, Munich, Germany; 2Department of Endocrinology, Diabetology and Clinical Nutrition, University Hospital Zurich (UHZ), Zurich, Switzerland
Background: Cyclic Cushing’s syndrome (cCS) is a sub-entity of Cushing’s syndrome (CS) associated with diagnostic and therapeutic challenges. It describes a condition, in which phases of clear-cut biochemical hypercortisolism are followed by spontaneous troughs of normal or subnormal cortisol secretion. We conducted the first systematic literature review to identify common features of cCS.
Methods: We searched MEDLINE (via PubMed) for eligible single case reports and case series from inception to October 10th, 2022, using two independent search terms. We compared outcomes to patients with noncyclic CS derived from the literature and to a longitudinally enrolled cohort from LMU hospital Munich as a reference standard.
Findings: In total, 212 cases with cCS were included for further analysis. Pituitary tumor origin accounted for 143 (67%), ectopic for 36 (17%), and adrenal for 23 (11%) cases. 4 (2%) cases were of occult origin, and 6 (3%) cases were unclassified. Clinical signs and symptoms were comparable to non-cyclic CS. In ACTHdependent forms, inferior petrosal sinus sampling had a positive and negative predictive value of 100% when performed during hypercortisolism vs 76% and 29% when performed irrespective of cortisolemic status. Overall, twelve (6%) patients underwent unnecessary surgery due to misdiagnoses. Remission rates were significantly lower and time to remission significantly longer when compared to the population with non-cyclic CS (P< 0.001).
Interpretation: We identified a heterogeneous population of patients with cCS with various tumor etiologies. Unpredictable cycle duration and frequency may cause diagnostic challenges resulting in misdiagnoses and missed diagnoses.
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Endocrine Abstracts
ISSN 1470-3947 (print) | ISSN 1479-6848 (online)
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