Neurological Deterioration in Three Siblings: Exploring the Spectrum of Argininemia.
Arginase deficiency
Argininemia
Spastic diplegia
Urea cycle disorders
Journal
Indian journal of pediatrics
ISSN: 0973-7693
Titre abrégé: Indian J Pediatr
Pays: India
ID NLM: 0417442
Informations de publication
Date de publication:
03 2021
03 2021
Historique:
received:
25
02
2020
accepted:
13
07
2020
pubmed:
10
8
2020
medline:
26
5
2021
entrez:
10
8
2020
Statut:
ppublish
Résumé
Argininemia or hyperargininemia is a urea cycle disorder caused by deficiency of the enzyme arginase 1. It is inherited in an autosomal recessive fashion. It commonly leads to spastic diplegia in childhood, but other important features include cognitive deterioration and epilepsy. Unlike other disorders of the urea cycle, hyperammonemia is not prominent. The authors report three siblings with genetically proven argininemia who presented with diverse phenotypes but with spasticity being a common feature. Sibling 1 developed motor regression in early childhood, sibling 2 developed delayed motor milestones from early infancy, whereas sibling 3 had global developmental delay in late infancy after a period of normal development. All siblings had mild hyperammonemia only. Early recognition is imperative, not only to initiate ammonia scavenging therapy which may lead to definite clinical improvement, but also to provide genetic counselling.
Identifiants
pubmed: 32770317
doi: 10.1007/s12098-020-03466-x
pii: 10.1007/s12098-020-03466-x
doi:
Substances chimiques
Arginase
EC 3.5.3.1
Types de publication
Case Reports
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
266-268Références
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