Diagnosis of cyclic Cushing’s disease manifests as early morning hyperglycemia in a patient with previously well-controlled type 1 diabetes

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Journal Name: Journal of Pediatric Endocrinology and Metabolism
Issue: Ahead of print

Abstract
Background
Cyclic Cushing’s disease (CCD) is reported to occur in approximately 15% of patients with Cushing’s disease (CD). CCD is a rare phenomenon in children.

Case presentation
A Portuguese female with well-controlled type 1 diabetes (T1DM) on an insulin pump developed transient uncontrolled blood sugar every morning. Increased basal and bolus insulin dosing was ineffective in lowering blood sugar and she began to miss school because of nausea, vomiting, fatigue, but no ketoacidosis. Therefore, other causes of sporadic hyperglycemia were explored. Multiple 6-h urinary free cortisol (UFC) samples revealed a spike in cortisol coincident with severe hyperglycemia. Pituitary magnetic resonance imaging (MRI) revealed a 3.5 mm microadenoma and inferior petrosal sinus sampling of adrenocorticotropic hormone (ACTH) after corticotropin releasing hormone (CRH) stimulation confirmed ACTH-dependent CD. Endoscopic endonasal tumor resection led to resolution of early morning hyperglycemia and symptoms.

Discussion
Our case illustrates an atypical presentation of CCD. There are no previous case reports of a pediatric patient with T1DM and CCD. Unexplained hyperglycemia in a patient with previous well-controlled T1DM should prompt assessment of other causes. CCD can be easily be missed if timed 6-h UFC measurements are not obtained.

Keywords: Cushing’s disease; cyclic; type 1 diabetes

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Corresponding author: Lissette Cespedes, MD, Assistant Professor of Medicine, Division of Endocrinology, Diabetes and Metabolism, Rutgers New Jersey Medical School, 185 South Orange Avenue, MSB I-588, Newark, NJ 07103, USA, Phone: +973-972-6170, Fax: +973-972-5185, E-mail: bleichda@njms.rutgers.edu

Received: 2018-11-20

Accepted: 2019-03-24

Published Online: 2019-06-28

Author contributions: All the authors have accepted responsibility for the entire content of this submitted manuscript and approved submission.

Research funding: None declared.

Employment or leadership: None declared.

Honorarium: None declared.

Competing interests: The funding organization(s) played no role in the study design; in the collection, analysis, and interpretation of data; in the writing of the report; or in the decision to submit the report for publication.

Disclosures: The authors have no disclosures.

Citation Information: Journal of Pediatric Endocrinology and Metabolism, 20180506, ISSN (Online) 2191-0251, ISSN (Print) 0334-018X, DOI: https://doi.org/10.1515/jpem-2018-0506.

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