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Case Report
1 Clinical Associate Professor, Pediatric Endocrinology, University of Florida, Gainesville, Florida, Unites States of America
Address correspondence to:
Henry John Rohrs III
MD, University of Florida, Department of Pediatrics, Box 100296, Gainesville, Florida-32610,
United States of America
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Article ID: 100005Z19HR2018
Introduction: Our case discusses a pediatric patient that had an ACTH secreting pituitary adenoma that was not biochemically diagnosed using desmopressin as the stimulating agent for inferior petrosal sinus sampling (IPSS), but as the patient had clinical findings suggestive of Cushing disease, the test was repeated using the gold standard of oCRH as the stimulant, and a pituitary adenoma was identified and subsequently removed.
Case Report: An 8-year-old male presented with physical findings and biochemical evidence of Cushing disease. MRI of the brain revealed a heterogeneous filling defect within the left and right lateral aspects of the pituitary without microadenoma visualized. IPSS was performed using desmopressin as the stimulant for IPSS. The test was negative for ACTH secreting adenoma. IPSS was repeated using the gold standard of oCRH and the patient was found to have Cushing disease.
Conclusion: While desmopressin-stimulated IPSS has shown sensitivity and specificity of over 90% in multiple adult studies and case reports in pediatrics have suggested efficacy, formal pediatric studies have yet to be conducted. Additional studies of desmopressin as a stimulant for pediatric IPSS are warranted and clinicians should be aware of the potential lack of sensitivity of desmopressin stimulated IPSS in pediatric patients.
Keywords: Cushing disease, IPSS, oCRH, Pituitary adenoma, Vasopressin
Henry John Rohrs III - Substantial contributions to conception and design, Acquisition of data, Analysis of data, Interpretation of data, Drafting the article, Revising it critically for important intellectual content, Final approval of the version to be published
Guaranter of SubmissionThe corresponding author is the guarantor of submission.
Source of SupportNone
Consent StatementWritten informed consent was obtained from the patient for publication of this study.
Data AvailabilityAll relevant data are within the paper and its Supporting Information files.
Conflict of InterestAuthor declares no conflict of interest.
Copyright© 2018 Henry John Rohrs III. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information.