4.2 Article

Prediction of poor neurological development in patients with symptomatic congenital cytomegalovirus diseases after oral valganciclovir treatment

Journal

BRAIN & DEVELOPMENT
Volume 41, Issue 9, Pages 743-750

Publisher

ELSEVIER
DOI: 10.1016/j.braindev.2019.04.016

Keywords

Antiviral treatment; Developmental quotient; Microcephaly; Sequelae; Small for gestational age

Funding

  1. Ministry of Health, Labour and Welfare of Japan [H23-Jisedai-Ippan-001, H25-Jisedai-Shitei003, H30-Kansei-Shitei-002]
  2. Japan Agency for Medical Research and Development, AMED [15gk0110003h0103, 16gk0110021h0001, 18ek0109265h0002]
  3. Kawano Masanori Memorial Foundation for the Promotion of Pediatrics [2018]

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Objective: This study aimed to evaluate the neurodevelopmental outcomes of infants with symptomatic congenital cytomegalovirus (SCCMV) disease after antiviral treatment and investigate the symptoms at birth associated with a developmental quotient (DQ) < 70. Methods: In this prospective study conducted from 2009 to 2018, infants with SCCMV disease who received oral valganciclovir (VGCV; 32 mg/kg/day) for 6 weeks (November 2009 to June 2015) or 6 months (July 2015 to March 2018) were evaluated for their neurodevelopmental outcomes at around 18 months of corrected age. Sequelae were categorized as follows: no impairment with a DQ >= 80 and no hearing dysfunction; mild sequelae including unilateral hearing dysfunction or a DQ of 70-79; and severe sequelae with a DQ < 70, bilateral hearing dysfunction requiring hearing aids, blindness or epilepsy requiring anti-epileptic drugs. DQ was assessed using the Kyoto Scale of Psychological Development. Symptoms at birth associated with a DQ < 70 were determined using univariate and receiver operating characteristic curve analyses. Results: Of the 24 treated infants, 21 reached > 18 months of corrected age. Six (29%) were no impairment, 4 (19%) had mild sequelae, and 11 (52%) developed severe sequelae. The symptoms at birth associated with a DQ < 70 were microcephaly and/or small for gestational age. Conclusion: In our cohort of infants with SCCMV disease after VGCV treatment, the incidence of severe sequelae at 18 months of corrected age was around 50%. When microcephaly and/or small for gestational age are seen at birth, a low DQ may appear even after oral VGCV treatment. (C) 2019 The Japanese Society of Child Neurology. Published by Elsevier B.V. All rights reserved.

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