Acute rhabdomyolysis following viral infection with coxsackie A4 in a 50-day-old infant with Fukuyama congenital muscular dystrophy.


Journal

Journal of infection and chemotherapy : official journal of the Japan Society of Chemotherapy
ISSN: 1437-7780
Titre abrégé: J Infect Chemother
Pays: Netherlands
ID NLM: 9608375

Informations de publication

Date de publication:
May 2020
Historique:
received: 01 09 2019
revised: 07 11 2019
accepted: 21 12 2019
pubmed: 28 1 2020
medline: 11 11 2020
entrez: 28 1 2020
Statut: ppublish

Résumé

Fukuyama congenital muscular dystrophy (FCMD), which is characterized by generalized muscle weakness, hypotonia, and motor delay during early infancy, gradually progresses with advanced age. Although acute rhabdomyolysis following infection in patients with FCMD has occasionally been reported, no studies have investigated rhabdomyolysis following viral infection in FCMD patients during early infancy. We report the case of a 50-day-old girl with no apparent symptoms of muscular dystrophy who developed severe acute rhabdomyolysis caused by viral infection, resulting in quadriplegia and respiratory failure therefore requiring mechanical ventilation. Brain magnetic resonance imaging incidentally showed the typical characteristics of FCMD, and FCMD was confirmed by genetic analysis, which revealed a 3-kb retrotransposon insertion in one allele of the fukutin gene and a deep intronic splicing variant in intron 5 in another allele. The virus etiology was confirmed to be Coxsackie A4. We report a severe case of acute rhabdomyolysis with the earliest onset of symptoms due to the Coxsackie A4 virus in a patient with FCMD. The present findings indicate that physicians should consider FCMD with viral infection a differential diagnosis if the patient presents with acute rhabdomyolysis following a fever.

Sections du résumé

BACKGROUND BACKGROUND
Fukuyama congenital muscular dystrophy (FCMD), which is characterized by generalized muscle weakness, hypotonia, and motor delay during early infancy, gradually progresses with advanced age. Although acute rhabdomyolysis following infection in patients with FCMD has occasionally been reported, no studies have investigated rhabdomyolysis following viral infection in FCMD patients during early infancy.
CASE REPORT METHODS
We report the case of a 50-day-old girl with no apparent symptoms of muscular dystrophy who developed severe acute rhabdomyolysis caused by viral infection, resulting in quadriplegia and respiratory failure therefore requiring mechanical ventilation. Brain magnetic resonance imaging incidentally showed the typical characteristics of FCMD, and FCMD was confirmed by genetic analysis, which revealed a 3-kb retrotransposon insertion in one allele of the fukutin gene and a deep intronic splicing variant in intron 5 in another allele. The virus etiology was confirmed to be Coxsackie A4.
CONCLUSION CONCLUSIONS
We report a severe case of acute rhabdomyolysis with the earliest onset of symptoms due to the Coxsackie A4 virus in a patient with FCMD. The present findings indicate that physicians should consider FCMD with viral infection a differential diagnosis if the patient presents with acute rhabdomyolysis following a fever.

Identifiants

pubmed: 31983616
pii: S1341-321X(19)30384-8
doi: 10.1016/j.jiac.2019.12.015
pii:
doi:

Substances chimiques

FKTN protein, human 0
Membrane Proteins 0
RNA, Viral 0

Types de publication

Case Reports

Langues

eng

Sous-ensembles de citation

IM

Pagination

516-519

Informations de copyright

Copyright © 2019 Japanese Society of Chemotherapy and The Japanese Association for Infectious Diseases. Published by Elsevier Ltd. All rights reserved.

Déclaration de conflit d'intérêts

Declaration of Competing Interest None.

Auteurs

Hiroshi Yamaguchi (H)

Department of Neurology, Hyogo Prefectural Kobe Children's Hospital, Hyogo, Japan; Department of Pediatrics, Kobe University Graduate School of Medicine, Hyogo, Japan. Electronic address: hiyamaguchi_kch@hp.pref.hyogo.jp.

Mariko Taniguchi-Ikeda (M)

Department of Clinical Genetics, Fujita Health University Hospital, Toyoake, Aichi, Japan.

Hiroaki Nagase (H)

Department of Pediatrics, Kobe University Graduate School of Medicine, Hyogo, Japan.

Yusuke Ito (Y)

Division of Infectious Disease, Department of Pediatrics, Hyogo Prefectural Kobe Children's Hospital, Hyogo, Japan.

Shoichi Tokumoto (S)

Department of Neurology, Hyogo Prefectural Kobe Children's Hospital, Hyogo, Japan; Department of Pediatrics, Kobe University Graduate School of Medicine, Hyogo, Japan.

Daisaku Toyoshima (D)

Department of Neurology, Hyogo Prefectural Kobe Children's Hospital, Hyogo, Japan.

Sarantuya Enkhjargal (S)

Department of Molecular Genetics, Institute for Comprehensive Medical Science, Fujita Health University, Toyoake, Aichi, Japan.

Masahiro Nishiyama (M)

Department of Pediatrics, Kobe University Graduate School of Medicine, Hyogo, Japan.

Hiroyuki Awano (H)

Department of Pediatrics, Kobe University Graduate School of Medicine, Hyogo, Japan.

Hiroshi Kurosawa (H)

Division of Pediatric Critical Care Medicine, Hyogo Prefectural Kobe Children's Hospital, Hyogo, Japan.

Masashi Kasai (M)

Division of Infectious Disease, Department of Pediatrics, Hyogo Prefectural Kobe Children's Hospital, Hyogo, Japan.

Azusa Maruyama (A)

Department of Neurology, Hyogo Prefectural Kobe Children's Hospital, Hyogo, Japan.

Kazumoto Iijima (K)

Department of Pediatrics, Kobe University Graduate School of Medicine, Hyogo, Japan.

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Classifications MeSH