TY - JOUR
T1 - Children Newly Diagnosed with Fetal and Neonatal Alloimmune Thrombocytopenia
T2 - Neurodevelopmental Outcome at School Age: Newly Diagnosed FNAIT: Follow-up Study
AU - de Vos, Thijs W.
AU - van Zagten, Maud
AU - de Haas, Masja
AU - Oepkes, Dick
AU - Tan, Ratna N. G. B.
AU - van der Schoot, C. Ellen
AU - Steggerda, Sylke J.
AU - de Vries, Linda S.
AU - Lopriore, Enrico
AU - van Klink, Jeanine M. M.
N1 - Funding Information: Funded by Process and Product Development Diagnostic Services, Sanquin (SQI/00034). Publisher Copyright: © 2023 The Author(s)
PY - 2023/7
Y1 - 2023/7
N2 - 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).
AB - 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).
KW - cognitive functioning
KW - fetal and neonatal alloimmune thrombocytopenia
KW - intracranial hemorrhage
KW - minor neurological dysfunction
KW - neurodevelopmental impairment
UR - http://www.scopus.com/inward/record.url?scp=85159128130&partnerID=8YFLogxK
U2 - https://doi.org/10.1016/j.jpeds.2023.02.031
DO - https://doi.org/10.1016/j.jpeds.2023.02.031
M3 - Article
C2 - 36933767
SN - 0022-3476
VL - 258
JO - Journal of pediatrics
JF - Journal of pediatrics
M1 - 113385
ER -