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      Long-Term Follow-Up of Cushing’s Disease: A Case Report

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          Abstract

          Cushing’s disease is a condition in which hypercortisolism develops due to excessive hypophyseal adrenocorticotropic hormone production. It is rare in childhood. In this paper, we report the case of a 10-year-old male patient with hypophyseal microadenoma-related Cushing’s disease who presented with obesity and was found to show poor height growth at follow-up. The diagnosis was confirmed with inferior petrosal sinus sampling, and the adenoma was successfully removed by transsphenoidal surgery. While adrenal axis suppression continued for approximately 1 year, clinical improvement was clearly observed after the third month following surgery. The findings in this patient demonstrate that decreased growth rate despite rapid weight gain in children can be early sign of Cushing’s disease and emphasize the importance of monitoring of growth in obese children.

          Conflict of interest:None declared.

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          Cushing syndrome in pediatrics.

          Constantine Stratakis (2012)
          Cushing syndrome is characterized by truncal obesity, growth deceleration, skin changes, muscle weakness, and hypertension. Cushing syndrome in childhood usually results from the exogenous administration of glucocorticoids. This article presents the causes and discusses the treatment of endogenous Cushing syndrome. It also discusses the clinical and molecular genetics of inherited forms of this syndrome. Cushing syndrome needs to be diagnosed and treated properly when first recognized; improper treatment can turn this otherwise completely curable disorder into a chronic ailment. Barriers to optimal care of a pediatric patient with Cushing syndrome are discussed. Published by Elsevier Inc.
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            Accuracy of diagnostic tests for Cushing's syndrome: a systematic review and metaanalyses.

            Sarah Nadeem, Katherine Harris, Rebecca Mullan … (2008)
            The diagnosis of Cushing's syndrome (CS) requires the use of tests of unregulated hypercortisolism that have unclear accuracy. Our objective was to summarize evidence on the accuracy of common tests for diagnosing CS. We searched electronic databases (MEDLINE, EMBASE, Web of Science, Scopus, and citation search for key articles) from 1975 through September 2007 and sought additional references from experts. Eligible studies reported on the accuracy of urinary free cortisol (UFC), dexamethasone suppression test (DST), and midnight cortisol assays vs. reference standard in patients suspected of CS. Reviewers working in duplicate and independently extracted study characteristics and quality and data to estimate the likelihood ratio (LR) and the 95% confidence interval (CI) for each result. We found 27 eligible studies, with a high prevalence [794 (9.2%) of 8631 patients had CS] and severity of CS. The tests had similar accuracy: UFC (n = 14 studies; LR+ 10.6, CI 5.5-20.5; LR- 0.16, CI 0.08-0.33), salivary midnight cortisol (n = 4; LR+ 8.8, CI 3.5-21.8; LR- 0.07, CI 0-1.2), and the 1-mg overnight DST (n = 14; LR+ 16.4, CI 9.3-28.8; LR- 0.06, CI 0.03-0.14). Combined testing strategies (e.g. a positive result in both UFC and 1-mg overnight DST) had similar diagnostic accuracy (n = 3; LR+ 15.4, CI 0.7-358; LR- 0.11, CI 0.007-1.57). Commonly used tests to diagnose CS appear highly accurate in referral practices with samples enriched with patients with CS. Their performance in usual clinical practice remains unclear.
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              Childhood Cushing Disease: A Challenge in Diagnosis and Management

              Nalini Shah, Nanni S. Shah, Anurag Ranjan Lila (2011)
              Background: Cushing syndrome (CS) in childhood is an extremely rare disorder that poses unique diagnostic difficulties. Different etiopathological states account for endogenous hypercortisolism in various age groups. Diagnosis is especially challenging in individuals with adrenocorticotropic hormone-dependent CS. The two most common etiologies are adrenal tumors and pituitary corticotrophin- secreting adenomas (Cushing disease). Case Report: We present the case of a 9-year-old boy that illustrates some of the challenges of diagnosing and managing patients with CS. Conclusions: Diagnosis is usually made by an experienced clinician using a combination of tests to establish a state of endogenous hypercortisolism followed by localization of its source.Management requires an individualized approach and multidisciplinary care. Even after curative therapy, these children require long-term follow-up into adulthood to assess growth and body composition and routine monitoring for metabolic anomalies and/or recurrence of the disease.
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                Author and article information

                Journal
                Journal ID (nlm-ta): J Clin Res Pediatr Endocrinol
                Journal ID (iso-abbrev): J Clin Res Pediatr Endocrinol
                Journal ID (publisher-id): JCRPE
                Title: Journal of Clinical Research in Pediatric Endocrinology
                Publisher: Galenos Publishing
                ISSN (Print): 1308-5727
                ISSN (Electronic): 1308-5735
                Publication date (Print): September 2013
                Publication date (Electronic): 18 September 2013
                Volume: 5
                Issue: 3
                Pages: 202-205
                Affiliations
                [1 ] Dr. Sami Ulus Women Health, Children’s Education and Research Hospital, Clinics of Pediatric Endocrinology, Ankara, Turkey
                [2 ] Social Security Institution Children’s Hospital, Clinics of Pediatric Neurosurgery, Ankara, Turkey
                [3 ] Yıldırım Beyazıt University, Clinics of Pediatric Endocrinology, Ankara, Turkey
                Author notes
                * Address for Correspondence: Dr. Sami Ulus Women Health, Children’s Education and Research Hospital, Clinics of Pediatric Endocrinology, Ankara, Turkey Phone: +90 312 305 65 08 E-mail: veyselbas@ 123456yahoo.com
                Article
                Publisher Item ID: 623
                DOI: 10.4274/Jcrpe.993
                PMC ID: 3814537
                PubMed ID: 24072091
                SO-VID: 68ef9dd7-131e-4674-8dcc-0eb97919c2fb
                Copyright © © Journal of Clinical Research in Pediatric Endocrinology, Published by Galenos Publishing.
                License:

                This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                Date received : 19 February 2013
                Date accepted : 14 May 2013
                Categories
                Subject: Case Report

                ScienceOpen disciplines: Pediatrics
                Keywords: obesity,growth rate,cushing’s disease
                Data availability:
                ScienceOpen disciplines: Pediatrics
                Keywords: obesity, growth rate, cushing’s disease

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