OBJECTIVES: To evaluate the perinatal and long-term neurodevelopmental outcome in a cohort of children with intracranial haemorrhage (ICH) due to fetal and neonatal alloimmune thrombocytopenia (FNAIT) and to clearly outline the burden of this disease. SUBJECTS AND METHODS: We performed an observational cohort study and included all consecutive cases of ICH caused by FNAIT from 1993 to 2015 at Leiden University Medical Centre. Neurological, motor, and cognitive development were assessed at a minimum age of 1 year. The primary outcome was adverse outcome, defined as perinatal death or severe neurodevelopmental impairment (NDI). Severe NDI was defined as any of the following: cerebral palsy (Gross Motor Function Classification System [GMFCS] level ≥II), bilateral deafness, blindness, or severe motor and/or cognitive developmental delay (<-2 SD). RESULTS: In total, 21 cases of ICH due to FNAIT were included in the study. The perinatal mortality rate was 10/21 (48%). Long-term outcome was assessed in 10 children (n = 1 lost to follow-up). Severe and moderate NDI were diagnosed in 6/10 (60%) and 1/10 (10%) of the surviving children. The overall adverse outcome, including perinatal mortality or severe NDI, was 16/20 (80%). CONCLUSIONS: The risk of perinatal death or severe NDI in children with ICH due to FNAIT is high. Only screening and effective preventive treatment can avoid this burden.
OBJECTIVES: To evaluate the perinatal and long-term neurodevelopmental outcome in a cohort of children with intracranial haemorrhage (ICH) due to fetal and neonatal alloimmune thrombocytopenia (FNAIT) and to clearly outline the burden of this disease. SUBJECTS AND METHODS: We performed an observational cohort study and included all consecutive cases of ICH caused by FNAIT from 1993 to 2015 at Leiden University Medical Centre. Neurological, motor, and cognitive development were assessed at a minimum age of 1 year. The primary outcome was adverse outcome, defined as perinatal death or severe neurodevelopmental impairment (NDI). Severe NDI was defined as any of the following: cerebral palsy (Gross Motor Function Classification System [GMFCS] level ≥II), bilateral deafness, blindness, or severe motor and/or cognitive developmental delay (<-2 SD). RESULTS: In total, 21 cases of ICH due to FNAIT were included in the study. The perinatal mortality rate was 10/21 (48%). Long-term outcome was assessed in 10 children (n = 1 lost to follow-up). Severe and moderate NDI were diagnosed in 6/10 (60%) and 1/10 (10%) of the surviving children. The overall adverse outcome, including perinatal mortality or severe NDI, was 16/20 (80%). CONCLUSIONS: The risk of perinatal death or severe NDI in children with ICH due to FNAIT is high. Only screening and effective preventive treatment can avoid this burden.
Authors: Thijs W de Vos; Leendert Porcelijn; Suzanne Hofstede-van Egmond; Eva Pajkrt; Dick Oepkes; Enrico Lopriore; C Ellen van der Schoot; Dian Winkelhorst; Masja de Haas Journal: Br J Haematol Date: 2021-08-16 Impact factor: 8.615
Authors: Giulia S Porcari; Lori C Jordan; Rebecca N Ichord; Daniel J Licht; Sabrina E Smith; Lauren A Beslow Journal: Pediatr Neurol Date: 2019-12-09 Impact factor: 3.372
Authors: C Ellen van der Schoot; Masja de Haas; Dian Winkelhorst; Thijs W de Vos; Marije M Kamphuis; Leendert Porcelijn; Enrico Lopriore; Dick Oepkes Journal: BMJ Open Date: 2020-07-20 Impact factor: 2.692
Authors: Thijs W de Vos; Dian Winkelhorst; Hans J Baelde; Kyra L Dijkstra; Rianne D M van Bergen; Lotte E van der Meeren; Peter G J Nikkels; Leendert Porcelijn; C Ellen van der Schoot; Gestur Vidarsson; Michael Eikmans; Rick Kapur; Carin van der Keur; Leendert A Trouw; Dick Oepkes; Enrico Lopriore; Marie-Louise P van der Hoorn; Manon Bos; Masja de Haas Journal: Int J Mol Sci Date: 2021-06-23 Impact factor: 5.923