Long-term follow-up of a child with Wolcott-Rallison syndrome.


Journal

BMJ case reports
ISSN: 1757-790X
Titre abrégé: BMJ Case Rep
Pays: England
ID NLM: 101526291

Informations de publication

Date de publication:
13 May 2021
Historique:
entrez: 14 5 2021
pubmed: 15 5 2021
medline: 18 5 2021
Statut: epublish

Résumé

Wolcott-Rallison syndrome (WRS), the most common cause of permanent neonatal diabetes mellitus (DM) in consanguineous families, has a poor prognosis, with a mean survival of 5.8 years. Majority of children with WRS succumb to the disease in the first decade of life. We present the long-term follow-up of an 8-year-2-month-old girl with genetically proven WRS who was born to a non-consanguineous parentage. She is on basal bolus regimen of insulin therapy for DM. In addition, she was noted to have features of skeletal dysplasia at 3 years and 3 months of age, which has led to her short stature. Surprisingly, she has had no episodes of hepatitis or liver dysfunction so far, which is frequently seen in children with WRS. To the best of our knowledge, she is the oldest surviving patient with WRS reported in India and South Asia.

Identifiants

pubmed: 33986015
pii: 14/5/e242376
doi: 10.1136/bcr-2021-242376
pmc: PMC8126271
pii:
doi:

Substances chimiques

eIF-2 Kinase EC 2.7.11.1

Types de publication

Journal Article Case Reports

Langues

eng

Sous-ensembles de citation

IM

Informations de copyright

© BMJ Publishing Group Limited 2021. No commercial re-use. See rights and permissions. Published by BMJ.

Déclaration de conflit d'intérêts

Competing interests: None declared.

Références

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Auteurs

Nikhil Shah (N)

Paediatric Endocrinology, Hirabai Cowasji Jehangir Medical Research Institute, Jehangir Hopsital, Pune, Maharashtra, India.
Paediatrics, Cloudnine Hopsital, Malad, Mumbai, Maharashtra, India.

Madhura Bharat Karguppikar (MB)

Paediatric Endocrinology, Hirabai Cowasji Jehangir Medical Research Institute, Jehangir Hopsital, Pune, Maharashtra, India.

Vaman Khadilkar (V)

Paediatric Endocrinology, Hirabai Cowasji Jehangir Medical Research Institute, Jehangir Hopsital, Pune, Maharashtra, India.
Interdisciplinary School of Health Sciences, Savitribai Phule Pune University, Pune, Maharashtra, India.

Anuradha Khadilkar (A)

Paediatric Endocrinology, Hirabai Cowasji Jehangir Medical Research Institute, Jehangir Hopsital, Pune, Maharashtra, India anuradhavkhadilkar@gmail.com.
Interdisciplinary School of Health Sciences, Savitribai Phule Pune University, Pune, Maharashtra, India.

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