Children Newly Diagnosed with Fetal and Neonatal Alloimmune Thrombocytopenia: Neurodevelopmental Outcome at School Age: Newly Diagnosed FNAIT: Follow-up Study

Thijs W de Vos, Maud van Zagten, Masja de Haas, Dick Oepkes, Ratna N G B Tan, C Ellen van der Schoot, Sylke J Steggerda, Linda S de Vries, Enrico Lopriore, Jeanine M M van Klink

Research output: Contribution to journalArticleAcademicpeer-review

Abstract

Objective: To evaluate the neurodevelopmental outcome at school age in children newly diagnosed with fetal and neonatal alloimmune thrombocytopenia (FNAIT). Study design: This observational cohort study included children diagnosed with FNAIT between 2002 and 2014. Children were invited for cognitive and neurological testing. Behavioral questionnaires and school performance results were obtained. A composite outcome of neurodevelopmental impairment (NDI) was used, defined, and subdivided into mild-to-moderate and severe NDI. Primary outcome was severe NDI, defined as IQ <70, cerebral palsy with Gross Motor Functioning Classification System level ≥ III, or severe visual/hearing impairment. Mild-to-moderate NDI was defined as IQ 70-85, minor neurological dysfunction or cerebral palsy with Gross Motor Functioning Classification System level ≤ II, or mild visual/hearing impairment. Results: In total, 44 children were included at a median age of 12 years (range: 6-17 years). Neuroimaging at diagnosis was available in 82% (36/44) of children. High-grade intracranial hemorrhage (ICH) was detected in 14% (5/36). Severe NDI was detected in 7% (3/44); two children had high-grade ICH, and one had low-grade ICH and perinatal asphyxia. Mild-to-moderate NDI was detected in 25% (11/44); one child had high-grade ICH, and eight children were without ICH, yet for two children, neuroimaging was not performed. Adverse outcome (perinatal death or NDI) was 39% (19/49). Four children (9%) attended special needs education, three of whom had severe NDI and one had mild-to-moderate NDI. Total behavioral problems within the clinical range were reported in 12%, which is comparable with 10% in the general Dutch population. Conclusion: Children who are newly diagnosed with FNAIT are at increased risk for long-term neurodevelopmental problems, even those without ICH. Trial registration: The study was registered at ClinicalTrials.gov (Identifier: NCT04529382).

Original languageEnglish
Article number113385
JournalThe Journal of Pediatrics
Volume258
Early online date16 Mar 2023
DOIs
Publication statusPublished - Jul 2023
Externally publishedYes

Keywords

  • cognitive functioning
  • fetal and neonatal alloimmune thrombocytopenia
  • intracranial hemorrhage
  • minor neurological dysfunction
  • neurodevelopmental impairment

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