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01.12.2015 | Case report | Ausgabe 1/2015 Open Access

BMC Endocrine Disorders 1/2015

Paediatric cyclical Cushing’s disease due to corticotroph cell hyperplasia

Zeitschrift:
BMC Endocrine Disorders > Ausgabe 1/2015
Autoren:
E. Noctor, S. Gupta, T. Brown, M. Farrell, M. Javadpour, C. Costigan, A. Agha
Wichtige Hinweise

Competing interests

The authors declare that they have no competing interests.

Authors’ contributions

EN treated the patient, gathered data and drafted the manuscript. SG treated the patient, gathered data and critically reviewed the manuscript. TB treated the patient, gathered data, and critically reviewed the manuscript. MF prepared, analysed, and interpreted the histopathological samples, and critically reviewed the manuscript. MJ treated the patient, carried out the surgery, gathered data, and critically reviewed the manuscript. CC treated the patient, gathered data, and critically reviewed the manuscript. AA treated the patient, conceptualized the case report, gathered data, and critically reviewed the manuscript. All authors approved the final version of the manuscript.

Abstract

Background

Cushing’s disease is very rare in the paediatric population. Although uncommon, corticotroph hyperplasia causing Cushing’s syndrome has been described in the adult population, but appears to be extremely rare in children. Likewise, cyclical cortisol hypersecretion, while accounting for 15 % of adult cases of Cushing’s disease, has only rarely been described in the paediatric population. Here, we describe a very rare case of a 13-year old boy with cyclical cortisol hypersecretion secondary to corticotroph cell hyperplasia.

Case presentation

The case is that of a 13-year old boy, presenting with a long history of symptoms and signs suggestive of hypercortisolism, who was found to have cyclical ACTH-dependent hypercortisolism following dynamic pituitary testing and serial late-night salivary cortisol measurements. The patient underwent endoscopic transsphenoidal resection of the pituitary. Early surgical remission was confirmed by undetectable post-operative morning plasma cortisol levels. Histology and immunocytochemistry of the resected pituitary tissue showed extensive corticotroph cell hyperplasia.

Conclusion

This report describes a rare case of cyclical Cushing’s disease secondary to corticotroph hyperplasia in a paediatric patient. This highlights the challenging and varied nature of Cushing’s disease and its diagnosis, and the need to keep a differential diagnosis in mind during the diagnostic process.
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