Association Between Arthrogryposis and Mortality in Infants With Congenital Zika Syndrome: A Systematic Review and Meta-analysis

Pediatr Neurol. 2020 Sep:110:20-24. doi: 10.1016/j.pediatrneurol.2020.05.007. Epub 2020 May 25.


Background: Intrauterine Zika virus infection is associated with neurological disorders and other problems, including such as impaired visual and hearing function and orthopedic abnormalities, including arthrogryposis. We systematically investigated the prevalence of arthrogryposis in infants with congenital Zika syndrome and the respective risk of mortality.

Methods: We conducted a systematic review and meta-analysis of reports published in PubMed, Web of Science, SCOPUS, and World Health Organization Global Index Medicus databases, using the keywords Zika virus and arthrogryposis and related terms.

Results: After screening titles and abstracts, a total of four studies were included. Arthrogryposis was not associated with increased risk for fetal demise (risk ratio, 3.33; 95% confidence interval, 0.73 to 15.26). However, arthrogryposis was associated with a 13-fold increased risk of mortality in neonates with congenital Zika syndrome (risk ratio, 13.11; 95% confidence interval, 3.74 to 45.92) than neonates with congenital Zika syndrome but without arthrogryposis.

Conclusions: Neonates with both congenital Zika syndrome and arthrogryposis had higher morbidity and mortality risks, making it necessary to implement protocols for the early identification of neuromuscular changes and appropriate management of patients.

Keywords: Congenital Zika syndrome; Outcome; Zika virus.

Publication types

  • Meta-Analysis
  • Systematic Review

MeSH terms

  • Arthrogryposis / epidemiology*
  • Arthrogryposis / etiology
  • Female
  • Humans
  • Infant, Newborn
  • Pregnancy
  • Pregnancy Complications, Infectious / epidemiology*
  • Zika Virus Infection / complications
  • Zika Virus Infection / congenital
  • Zika Virus Infection / mortality*