Cerebral Palsy in Children With Congenital Zika Syndrome: A 2-Year Neurodevelopmental Follow-up

Abstract

Objective: To describe the 2-year neurodevelopmental outcome in children with cerebral palsy associated with congenital Zika (CZ) and explore variables associated with a more severe presentation. Methods: Data on 69 children with cerebral palsy associated with CZ, followed in a neurorehabilitation hospital, who consecutively attended the neurodevelopmental assessment at 2 years of age, were collected. Bayley III Scales of Infant and Toddler Development, Hammersmith Infant Neurological Examination, and Gross Motor Function Classification System were used for the outcome evaluation. Descriptive and inferential statistical analysis were performed. Results: The median age at follow-up was of 24.0 (23-32) months. Only 3 (4.3%) children were not microcephalic. The majority presented with bilateral (94.2%), spastic (100.0%), Gross Motor Function Classification System grade IV or V (92.8%) cerebral palsy, epilepsy (73.1%), extremely low performances on cognitive (94.2%), language (95.7%), and motor (95.7%) Bayley-III Scales of Infant and Toddler Development Test scores. The median Hammersmith Infant Neurological Examination score was of 21.0 (range 9-75). There was a correlation between birth head circumference with the cognitive ( r = 0.3, P < .01), language ( r = 0.3, P < .01), and motor ( r = 0.3, P < .01) Bayley-III Scales of Infant and Toddler Development Test scores, as well as with the Hammersmith Infant Neurological Examination score ( r = 0.2, P < .03). An association was observed between an inferior median Hammersmith Infant Neurological Examination score with congenital microcephaly ( P = .04), arthrogryposis ( P = .02), and epilepsy in the first year ( P < .01). Conclusion: Cerebral palsy related to CZ presents with a severe global impairment at a 2-year follow-up. Birth head circumference, arthrogryposis, and early epilepsy are associated with a worse outcome and may be considered as prognostic markers. These findings are important for the neurorehabilitation planning, parents’ guiding, and future prognostic studies.

Epilepsy in children with Congenital Zika Syndrome: A systematic review and meta‐analysis

“…The overall quality of the selected studies is considered good, with high (12/15; 80%) [8][9][10][11][12][14][15][16][17][18][19]21 or moderate (3/15; 20%) 13,20,22 methodological quality according to the Newcastle-Ottawa scale (Table S1, Supplementary file). None of the studies described the intra-and inter-assay variations of the serological and molecular tests used in diagnosing ZIKV infection.…”

“…The remaining 15 studies [8][9][10][11][12][13][14][15][16][17][18][19][20][21][22] were critically analyzed and the information of interest was extracted. Among the selected studies, 13 [8][9][10][11][12][15][16][17][18][19][20][21][22] are articles published in specialized journals, and 2 are theses from Brazilian public universities (Universidade Federal de Pernambuco 13 and Universidade Federal Fluminense 14 ). One study 17 was not included in the meta-analysis because it used the same patients of the study 16 with different follow-up times.…”

“…Among the selected studies, 13 [8][9][10][11][12][15][16][17][18][19][20][21][22] are articles published in specialized journals, and 2 are theses from Brazilian public universities (Universidade Federal de Pernambuco 13 and Universidade Federal Fluminense 14 ). One study 17 was not included in the meta-analysis because it used the same patients of the study 16 with different follow-up times. Finally, 14 selected studies [8][9][10][11][12][13][14][15][16][18][19][20][21][22] were quantitatively analyzed and used to perform a meta-analysis of the frequency of epilepsy in children with CZS.…”

“…4 More recently, the hypothesis that CZS is associated with a high risk of developing early epilepsy (ie, in the first 24 months of life) has been confirmed in several studies. [8][9][10][11][12][13][14][15][16][17][18][19][20][21][22] Indeed, children diagnosed with CZS often present recurrent and spontaneous seizures, epileptogenic changes on electroencephalography (EEG), and high irritability. 9,18,21,22 Although several studies have evaluated the epidemiology of epilepsy in children with CZS in Brazil, they are decentralized, and no effort to estimate the overall frequency of this neurological complication among patients exposed to ZIKV during pregnancy has been reported.…”

Congenital Zika Syndrome—Assessing the Need for a Family Support Programme in Brazil

“…The evidence on CZS that was emerging at the time of this study highlighted the similarities of the physical presentation of CZS to those of CP. More recent literature has further described the overlapping between CZS and CP [35][36][37]. The review of the literature and findings of the social and economic impact study also suggested that there are similarities between CP and CZS, particularly in terms of needs and unmet needs of the caregiver.…”