Neurological Deterioration in Three Siblings: Exploring the Spectrum of Argininemia.


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
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

266-268

Références

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doi: 10.1016/j.ymgme.2004.11.004
Carvalho DR, Brum JM, Speck-Martins CE, et al. Clinical features and neurologic progression of hyperargininemia. Pediatr Neurol. 2012;46:369–74.
Scaglia F, Lee B. Clinical, biochemical, and molecular spectrum of hyperargininemia due to arginase I deficiency. Am J Med Genet C Semin Med Genet. 2006;142C:113–20.
doi: 10.1002/ajmg.c.30091
Prasad AN, Breen JC, Ampola MG, Rosman NP. Argininemia: a treatable genetic cause of progressive spastic diplegia simulating cerebral palsy: case reports and literature review. J Child Neurol. 1997;12:301–9.
doi: 10.1177/088307389701200502
Deignan JL, De Deyn PP, Cederbaum SD, et al. Guanidino compound levels in blood, cerebrospinal fluid, and post-mortem brain material of patients with argininemia. Mol Genet Metab. 2010;100:S31–6.
Bijarnia-Mahay S, Häberle J, Jalan AB, et al. Urea cycle disorders in India: clinical course, biochemical and genetic investigations, and prenatal testing. Orphanet J Rare Dis. 2018;13:174.
Chandra SR, Christopher R, Ramanujam CN, Harikrishna GV. Hyperargininemia experiences over last 7 years from a tertiary care center. J Pediatr Neurosci. 2019;14:2–6.
doi: 10.4103/JPN.JPN_1_19
Batshaw ML, Brusilow S, Waber L, et al. Treatment of inborn errors of urea synthesis: activation of alternative pathways of waste nitrogen synthesis and excretion. N Engl J Med. 1982;306:1387–92.

Auteurs

Divyani Garg (D)

Department of Neurology, Lady Hardinge Medical College and Associated Smt Sucheta Kriplani Hospital, New Delhi, India.

Sunita Bijarnia-Mahay (S)

Institute of Medical Genetics and Genomics, Sir Ganga Ram Hospital, New Delhi, India.

Aman Elwadhi (A)

Department of Pediatrics, Lady Hardinge Medical College and Kalawati Saran Children's Hospital, New Delhi, India.

Sandip Ray (S)

Department of Pediatrics, Lady Hardinge Medical College and Kalawati Saran Children's Hospital, New Delhi, India.

Johannes Häberle (J)

University Children's Hospital Zurich and Children's Research Center, Steinwiesstr. 75, CH-8032, Zurich, Switzerland.

Suvasini Sharma (S)

Department of Pediatrics, Lady Hardinge Medical College and Kalawati Saran Children's Hospital, New Delhi, India. sharma.suvasini@gmail.com.

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Classifications MeSH