TY - JOUR
T1 - Acute rhabdomyolysis following viral infection with coxsackie A4 in a 50-day-old infant with Fukuyama congenital muscular dystrophy
AU - Yamaguchi, Hiroshi
AU - Taniguchi-Ikeda, Mariko
AU - Nagase, Hiroaki
AU - Ito, Yusuke
AU - Tokumoto, Shoichi
AU - Toyoshima, Daisaku
AU - Enkhjargal, Sarantuya
AU - Nishiyama, Masahiro
AU - Awano, Hiroyuki
AU - Kurosawa, Hiroshi
AU - Kasai, Masashi
AU - Maruyama, Azusa
AU - Iijima, Kazumoto
N1 - Publisher Copyright:
© 2019 Japanese Society of Chemotherapy and The Japanese Association for Infectious Diseases
PY - 2020/5
Y1 - 2020/5
N2 - 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: 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: 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.
AB - 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: 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: 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.
KW - Creatine kinase
KW - Fukutin
KW - Mechanical ventilation
KW - Quadriplegia
KW - Respiratory failure
KW - Retrotransposon
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U2 - 10.1016/j.jiac.2019.12.015
DO - 10.1016/j.jiac.2019.12.015
M3 - Article
C2 - 31983616
AN - SCOPUS:85078149187
SN - 1341-321X
VL - 26
SP - 516
EP - 519
JO - Journal of Infection and Chemotherapy
JF - Journal of Infection and Chemotherapy
IS - 5
ER -