Long-term outcome in patients after treatment for Cushing’s disease in childhood


Autoři: Katarzyna Pasternak-Pietrzak aff001;  Elżbieta Moszczyńska aff001;  Marcin Roszkowski aff002;  Karolina Kot aff001;  Elżbieta Marczak aff001;  Wiesława Grajkowska aff003;  Maciej Pronicki aff003;  Mieczysław Szalecki aff001
Působiště autorů: Department of Endocrinology and Diabetology, The Children’s Memorial Health Institute (CMHI), Warsaw, Poland aff001;  Department of Neurosurgery, The Children’s Memorial Health Institute (CMHI), Warsaw, Poland aff002;  Pathology Department, The Children’s Memorial Health Institute, Warsaw, Poland aff003;  Collegium Medicum, University of Jan Kochanowski, Kielce, Poland aff004
Vyšlo v časopise: PLoS ONE 14(12)
Kategorie: Research Article
doi: 10.1371/journal.pone.0226033

Souhrn

Introduction

Cushing’s disease (CD) is a rare cause of hypercortisolemia presenting a major diagnostic and therapeutic challenge. Data on pituitary function in long-term follow-up after CD treatment in childhood is limited.

Aim

Long-term assessment of patients of the Children’s Memorial Health Institute (CMHI) after CD treatment in childhood.

Materials and methods

Retrospective analysis of 29 CD patients, mean age at the time of diagnosis 13.46 yrs. The long-term follow-up (FU) was done by: 1) obtaining the data from a patient’s questionnaire (75% of adult patients); 2) using the data from the last clinic visit for patients who did not respond to the questionnaire and for current CMHI patients. The average long-term FU from transsphenoidal pituitary surgery (TSS) was 10.23 yrs.

Results

At the latest FU: 18 patients (62%) had long-term disease remission after TSS1, 2 patients (6.9%) after TSS2, 1 patient (3.4%) after the post-TSS radiotherapy (XRT) cycle and 3 patients (10.3%) after bilateral adrenalectomy (BA). One patient (3.4%) died after TSS2 due to postoperative complications, 1 patient (3.4%) had persistent disease at latest FU, in 1 patient (3.4%) the long-term FU was not possible to perform. CD recurrence occurred in 4 out of 28 patients (14%) at an average time 3.6 yrs. from definitive treatment. One patient (3.4%) after BA was operated because of Nelson's syndrome. Two patients (6.9%) were suspected of relapse at latest assessment. At the time of the last evaluation, 17 patients (63%) were on levothyroxine therapy since definitive treatment, 16 patients (59%) were on hydrocortisone treatment, 10 patients (37%) were taking sex hormones replacement, 4 patients (15%)—antidiuretic hormone.

Conclusions

Relatively large number of patients after CD treatment in childhood have hormonal pituitary deficits as well as mood and cognitive disorders. CD recurrence can occur even after a long time post effective treatment.

Klíčová slova:

Cortisol – Growth hormone – Magnetic resonance imaging – Pediatrics – Pituitary gland – Sex hormones – Surgical and invasive medical procedures – Adenomas


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