First presentation of LPIN1 acute rhabdomyolysis in adolescence and adulthood.


Journal

Neuromuscular disorders : NMD
ISSN: 1873-2364
Titre abrégé: Neuromuscul Disord
Pays: England
ID NLM: 9111470

Informations de publication

Date de publication:
07 2020
Historique:
received: 03 04 2020
revised: 12 05 2020
accepted: 14 05 2020
pubmed: 12 6 2020
medline: 12 8 2021
entrez: 12 6 2020
Statut: ppublish

Résumé

LPIN1 mutations are a known common cause of autosomal recessive, recurrent and life-threatening acute rhabdomyolysis of childhood-onset. The first episode of rhabdomyolysis usually happens in nearly all cases before the age of 5 and death is observed in 1/3 of patients. Here we present two cases of acute rhabdomyolysis with a milder phenotype caused by LPIN1 mutation presenting in adolescence (11 years old) and adulthood (40 years old) after Parvovirus infection and metabolic stress, respectively. In our opinion, the mutation types, epigenetic factors, the environment exposition to triggers or the existence of proteins with a similar structure of LPIN1, may have a role in modulating the onset of rhabdomyolysis. LPIN1 should be included on a panel of genes analysed in the investigation of adult individuals with rhabdomyolysis. Metabolic and viral stressors should be included in the list of possible rhabdomyolysis precipitant.

Identifiants

pubmed: 32522502
pii: S0960-8966(20)30120-6
doi: 10.1016/j.nmd.2020.05.004
pii:
doi:

Substances chimiques

LPIN1 protein, human EC 3.1.3.4
Phosphatidate Phosphatase EC 3.1.3.4

Types de publication

Case Reports

Langues

eng

Sous-ensembles de citation

IM

Pagination

566-571

Informations de copyright

Crown Copyright © 2020. Published by Elsevier B.V. All rights reserved.

Auteurs

Chiara Pizzamiglio (C)

MRC Centre for Neuromuscular Diseases, UCL Institute of Neurology and National Hospital for Neurology and Neurosurgery, Queen Square, London, United Kingdom. Electronic address: c.pizzamiglio@ucl.ac.uk.

Nayana Lahiri (N)

Clinical Genetics Department, St George's University Hospitals NHS Foundation Trust, London, United Kingdom.

Niranjanan Nirmalananthan (N)

Departments of Neurology and Neuroradiology, Atkinson Morley Regional Neurosciences Centre, St George's Hospital, London, United Kingdom.

Bhrigu Sood (B)

South West Thames Renal and Transplantation Unit and South West Thames Institute for Renal Research, Saint Helier Hospital, Carshalton, Surrey, United Kingdom.

Subash Somalanka (S)

South West Thames Renal and Transplantation Unit and South West Thames Institute for Renal Research, Saint Helier Hospital, Carshalton, Surrey, United Kingdom.

Philip Ostrowski (P)

South West Thames Regional Genetics Service, St George's University NHS Foundation Trust, London, United Kingdom.

Rahul Phadke (R)

Division of Neuropathology, Dubowitz Neuromuscular Centre, UCL Great Ormond Street Hospital for Children, United Kingdom; Division of Neuropathology, National Hospital for Neurology and Neurosurgery, Queen Square, London, United Kingdom.

Dominic Gerard O'Donovan (DG)

Neuropathology, Department of Histopathology, Addenbrooke's Hospital, Cambridge University Hospitals NHS Foundation Trust, Cambridge, United Kingdom.

Francesco Muntoni (F)

Paediatric Neurology, Dubowitz Neuromuscular Centre, UCL Institute of Child Health and Great Ormond Street Hospital for Children, London, United Kingdom.

Rosaline Quinlivan (R)

MRC Centre for Neuromuscular Diseases, UCL Institute of Neurology and National Hospital for Neurology and Neurosurgery, Queen Square, London, United Kingdom.

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Classifications MeSH