Cargando…

Central Diabetes Insipidus in Children and Adolescents: Twenty-Six Year Experience from a Single Centre

INTRODUCTION: Paediatric cohorts of central diabetes insipidus (CDI) have shown varying prevalence for different causes of CDI. The objective of this study was to determine the causes of CDI and long-term outcome in children and adolescents from a Tertiary Paediatric Endocrinology unit. METHODS: The...

Descripción completa

Detalles Bibliográficos
Autores principales: Korkmaz, Hüseyin Anil, Kapoor, Ritika R, Kalitsi, Jennifer, Aylwin, Simon JB, Buchanan, Charles R, Arya, Ved Bhushan
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Hindawi 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8924606/
https://www.ncbi.nlm.nih.gov/pubmed/35311034
http://dx.doi.org/10.1155/2022/9397130
_version_ 1784669895553712128
author Korkmaz, Hüseyin Anil
Kapoor, Ritika R
Kalitsi, Jennifer
Aylwin, Simon JB
Buchanan, Charles R
Arya, Ved Bhushan
author_facet Korkmaz, Hüseyin Anil
Kapoor, Ritika R
Kalitsi, Jennifer
Aylwin, Simon JB
Buchanan, Charles R
Arya, Ved Bhushan
author_sort Korkmaz, Hüseyin Anil
collection PubMed
description INTRODUCTION: Paediatric cohorts of central diabetes insipidus (CDI) have shown varying prevalence for different causes of CDI. The objective of this study was to determine the causes of CDI and long-term outcome in children and adolescents from a Tertiary Paediatric Endocrinology unit. METHODS: The clinic database was searched to identify patients with CDI managed between 1993 and 2019. Relevant clinical information was collected from patient records. RESULTS: A total of 138 CDI patients, median age 6 years (range <1–18) at presentation, were identified. Principal CDI aetiologies were craniopharyngioma (n = 44), acute central nervous system (CNS) insult (n = 33), germinoma (n = 15), postneurosurgery (indication other than craniopharyngioma and germinoma, n = 20), midline CNS malformation (n = 14), Langerhans cell histiocytosis (n = 5), and familial (n = 2). Idiopathic CDI in this cohort was infrequent (n = 5). Patients with CNS malformations/infections presented with CDI at a younger age compared to patients with CNS tumours (p < 0.0001). Five patients, initially presenting as idiopathic CDI, were subsequently diagnosed with germinoma after a median interval of 3.3 years. All patients with CDI related to craniopharyngioma and nearly all (87%) patients with CDI related to germinoma had concomitant GH, ACTH, and TSH deficiency. The majority of patients who manifested CDI due to acute CNS insult either deceased (30%) or had transient CDI (33.3%). CONCLUSION: Surgery for craniopharyngioma was the most common underlying aetiology of CDI with ubiquitous occurrence of panhypopituitarism in these patients. Manifestation of CDI in patients with acute CNS insult carries poor prognosis. We affirm that neuroimaging assessment in idiopathic CDI should be continued beyond 3 years from diagnosis as a significant number of patients exhibited progression of infundibular thickening 3 years post-CDI diagnosis.
format Online
Article
Text
id pubmed-8924606
institution National Center for Biotechnology Information
language English
publishDate 2022
publisher Hindawi
record_format MEDLINE/PubMed
spelling pubmed-89246062022-03-17 Central Diabetes Insipidus in Children and Adolescents: Twenty-Six Year Experience from a Single Centre Korkmaz, Hüseyin Anil Kapoor, Ritika R Kalitsi, Jennifer Aylwin, Simon JB Buchanan, Charles R Arya, Ved Bhushan Int J Endocrinol Research Article INTRODUCTION: Paediatric cohorts of central diabetes insipidus (CDI) have shown varying prevalence for different causes of CDI. The objective of this study was to determine the causes of CDI and long-term outcome in children and adolescents from a Tertiary Paediatric Endocrinology unit. METHODS: The clinic database was searched to identify patients with CDI managed between 1993 and 2019. Relevant clinical information was collected from patient records. RESULTS: A total of 138 CDI patients, median age 6 years (range <1–18) at presentation, were identified. Principal CDI aetiologies were craniopharyngioma (n = 44), acute central nervous system (CNS) insult (n = 33), germinoma (n = 15), postneurosurgery (indication other than craniopharyngioma and germinoma, n = 20), midline CNS malformation (n = 14), Langerhans cell histiocytosis (n = 5), and familial (n = 2). Idiopathic CDI in this cohort was infrequent (n = 5). Patients with CNS malformations/infections presented with CDI at a younger age compared to patients with CNS tumours (p < 0.0001). Five patients, initially presenting as idiopathic CDI, were subsequently diagnosed with germinoma after a median interval of 3.3 years. All patients with CDI related to craniopharyngioma and nearly all (87%) patients with CDI related to germinoma had concomitant GH, ACTH, and TSH deficiency. The majority of patients who manifested CDI due to acute CNS insult either deceased (30%) or had transient CDI (33.3%). CONCLUSION: Surgery for craniopharyngioma was the most common underlying aetiology of CDI with ubiquitous occurrence of panhypopituitarism in these patients. Manifestation of CDI in patients with acute CNS insult carries poor prognosis. We affirm that neuroimaging assessment in idiopathic CDI should be continued beyond 3 years from diagnosis as a significant number of patients exhibited progression of infundibular thickening 3 years post-CDI diagnosis. Hindawi 2022-03-08 /pmc/articles/PMC8924606/ /pubmed/35311034 http://dx.doi.org/10.1155/2022/9397130 Text en Copyright © 2022 Hüseyin Anil Korkmaz et al. https://creativecommons.org/licenses/by/4.0/This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Research Article
Korkmaz, Hüseyin Anil
Kapoor, Ritika R
Kalitsi, Jennifer
Aylwin, Simon JB
Buchanan, Charles R
Arya, Ved Bhushan
Central Diabetes Insipidus in Children and Adolescents: Twenty-Six Year Experience from a Single Centre
title Central Diabetes Insipidus in Children and Adolescents: Twenty-Six Year Experience from a Single Centre
title_full Central Diabetes Insipidus in Children and Adolescents: Twenty-Six Year Experience from a Single Centre
title_fullStr Central Diabetes Insipidus in Children and Adolescents: Twenty-Six Year Experience from a Single Centre
title_full_unstemmed Central Diabetes Insipidus in Children and Adolescents: Twenty-Six Year Experience from a Single Centre
title_short Central Diabetes Insipidus in Children and Adolescents: Twenty-Six Year Experience from a Single Centre
title_sort central diabetes insipidus in children and adolescents: twenty-six year experience from a single centre
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8924606/
https://www.ncbi.nlm.nih.gov/pubmed/35311034
http://dx.doi.org/10.1155/2022/9397130
work_keys_str_mv AT korkmazhuseyinanil centraldiabetesinsipidusinchildrenandadolescentstwentysixyearexperiencefromasinglecentre
AT kapoorritikar centraldiabetesinsipidusinchildrenandadolescentstwentysixyearexperiencefromasinglecentre
AT kalitsijennifer centraldiabetesinsipidusinchildrenandadolescentstwentysixyearexperiencefromasinglecentre
AT aylwinsimonjb centraldiabetesinsipidusinchildrenandadolescentstwentysixyearexperiencefromasinglecentre
AT buchanancharlesr centraldiabetesinsipidusinchildrenandadolescentstwentysixyearexperiencefromasinglecentre
AT aryavedbhushan centraldiabetesinsipidusinchildrenandadolescentstwentysixyearexperiencefromasinglecentre