Long-term neurodevelopmental outcome after intrauterine transfusion for hemolytic disease of the fetus/newborn: the LOTUS study.
OBJECTIVE
To determine the incidence and risk factors for neurodevelopmental impairment (NDI) in children with hemolytic disease of the fetus/newborn treated with intrauterine transfusion (IUT).


METHODS
Neurodevelopmental outcome in children at least 2 years of age was assessed using standardized tests, including the Bayley Scales of Infant Development, the Wechsler Preschool and Primary Scale of Intelligence, and the Wechsler Intelligence Scale for Children, according to the children's age. Primary outcome was the incidence of neurodevelopmental impairment defined as at least one of the following: cerebral palsy, severe developmental delay, bilateral deafness, and/or blindness.


RESULTS
A total of 291 children were evaluated at a median age of 8.2 years (range, 2-17 years). Cerebral palsy was detected in 6 (2.1%) children, severe developmental delay in 9 (3.1%) children, and bilateral deafness in 3 (1.0%) children. The overall incidence of neurodevelopmental impairment was 4.8% (14/291). In a multivariate regression analysis including only preoperative risk factors, severe hydrops was independently associated with neurodevelopmental impairment (odds ratio, 11.2; 95% confidence interval, 1.7-92.7).


CONCLUSIONS
Incidence of neurodevelopmental impairment in children treated with intrauterine transfusion for fetal alloimmune anemia is low (4.8%). Prevention of fetal hydrops, the strongest preoperative predictor for impaired neurodevelopment, by timely detection, referral and treatment may improve long-term outcome.
Department of Obstetrics, Leiden University Medical Center, Leiden, The Netherlands.
Irene T
IT
Lindenburg
Vivianne E
VE
Smits-Wintjens
Jeanine M
JM
van Klink
Esther
E
Verduin
Inge L
IL
van Kamp
Frans J
FJ
Walther
Henk
H
Schonewille
Ilias I
II
Doxiadis
Humphrey H
HH
Kanhai
Jan M
JM
van Lith
Erik W
EW
van Zwet
Dick
D
Oepkes
Anneke
A
Brand
Enrico
E
Lopriore
D000293:Adolescent; D001766:Blindness / Q000453:epidemiology* / Q000209:etiology; D001805:Blood Transfusion, Intrauterine; D002547:Cerebral Palsy / Q000453:epidemiology* / Q000209:etiology; D002648:Child; D002675:Child, Preschool; D002658:Developmental Disabilities / Q000453:epidemiology* / Q000209:etiology; D004899:Erythroblastosis, Fetal / Q000628:therapy*; D005260:Female; D005500:Follow-Up Studies; D006312:Hearing Loss, Bilateral / Q000453:epidemiology* / Q000209:etiology; D006801:Humans; D015994:Incidence; D007231:Infant, Newborn; D008297:Male; D011247:Pregnancy; D012307:Risk Factors; D016896:Treatment Outcome