Corine Koopman

COL4A1 mutation in two preterm siblings with antenatal onset of parenchymal hemorrhage.

To report the presence of intracerebral hemorrhage and porencephaly, both present at birth, in two preterm infants with a mutation in the collagen 4 A1 gene.

MR Imaging and Outcome of Term Neonates with Perinatal Asphyxia: Value of Diffusion-weighted MR Imaging and H MR Spectroscopy

PURPOSE To compare the association between neurodevelopmental outcome in neonates with hypoxic-ischemic encephalopathy following perinatal asphyxia and (a) apparent diffusion coefficients (ADCs) in the thalamus and basal ganglia at diffusion-weighted (DW) magnetic resonance (MR) imaging and (b) hydrogen 1 (¹H) MR spectroscopic measurements in the basal ganglia. MATERIALS AND METHODS This retrospective study was approved by the local ethics committee, and the requirement to obtain informed consent was waived. Eighty-one term neonates with perinatal asphyxia underwent conventional and DW cranial MR imaging (median age, 4 days; age range, 1-14 days); 51 neonates also underwent ¹H MR spectroscopy. Neurodevelopment was assessed from 18 to 46 months. Patients with favorable and adverse outcomes were compared. Receiver operating characteristics analysis was performed in all patients, and uni- and multivariate logistic regression analyses were performed in 44 patients examined within 7 days of birth by using MR imaging scores, ADCs in the basal ganglia and thalamus, and ¹H MR spectroscopic measurements in the basal ganglia. RESULTS An adverse outcome was seen in 28 of all 81 neonates (20 died, seven developed cerebral palsy, and one had severe mental retardation) and 22 of the 44 neonates examined within 7 days of birth with both ADC and ¹H MR spectroscopy. Poor outcome was associated with (a) lower ADCs in the basal ganglia (P < .001) and thalamus (P = .001) of neonates examined within 7 days of birth and (b) a higher lactate (Lac)-N-acetylaspartate (NAA) ratio in the basal ganglia (P < .001). Multivariate analysis showed that MR imaging score combined with Lac/NAA ratios or ADCs in the basal ganglia within the 1st week of life had a better association with outcome than did MR imaging alone (P = .006, area under the receiver operating characteristic curve [AUC] = 0.85 with Lac/NAA ratio; P < .0001, AUC = 0.93 with ADCs in basal ganglia). CONCLUSION The combination of MR imaging score with ADCs or Lac/NAA ratios in the basal ganglia has a better association with outcome of asphyxiated term neonates than does MR imaging alone.

Postnatal growth of preterm born children ≤ 750 g at birth

BACKGROUND Extremely low birth weight (ELBW) infants are at risk of impaired postnatal growth. Impaired postnatal growth has been reported to be associated with delayed cognitive and motor development. AIMS To describe postnatal growth patterns of appropriate and small for gestational age (AGA and SGA) ELBW children in relation to their cognitive and motor outcome at age 5.5. STUDY DESIGN Retrospective cohort study. SUBJECTS One hundred one children with a BW ≤ 750g, born between 1996 and 2005 in the University Hospital Utrecht, The Netherlands. OUTCOME MEASURES Height (Ht), weight (Wt), occipital-frontal circumference (OFC) at birth, 15 months and 2 years corrected age and 3.5 and 5.5 years. Cognitive and motor outcome at 5.5 years of age, classified as normal (Z-score ≥-1), mildly delayed (-2≤Z-score <-1) or severely delayed (Z-score <-2). AGA (Ht, Wt or OFC at birth ≥-2 SDS) infants were compared with SGA (Ht, Wt or OFC at birth <-2 SDS) infants. RESULTS Between birth and 5.5 years catch-up growth in Ht, weight for height (Wt/Ht), Wt and OFC was seen in 72.2%, 55.2%, 28.6% and 68.9% respectively of the SGA infants. For AGA infants we found substantial catch-down growth in Ht (15.4%) and Wt (33.8%). Cognitive and motor outcome was normal in 76.2% and 41.6% of the 101 children. A significantly higher percentage of normal cognitive outcome was found in AGA infants with Wt growth remaining at ≥-2 SDS compared to AGA infants with catch-down growth (83% vs 63%). Next, SGA infants who caught-up in OFC had a higher prevalence of normal cognitive outcome compared to SGA infants who did not catch-up in OFC. Furthermore, a higher percentage of severely delayed motor outcome was found in SGA infants without catch-up growth in Wt compared to SGA infants who caught-up in Wt (61.5% vs 32.2%). CONCLUSIONS Catch-up growth in Ht, Wt/Ht and OFC occurred in the majority of the SGA infants with a BW ≤ 750 g, but was less common in Wt. AGA children who remained their Wt at ≥-2 SDS have a better cognitive and motor developmental outcome at 5.5 years of age. Catch-up growth in OFC was associated with a better cognitive outcome at 5.5 years of age.

Cognitive and neurological outcome at the age of 5-8 years of preterm infants with post-hemorrhagic ventricular dilatation requiring neurosurgical intervention.

Background: Preterm infants with progressive post-hemorrhagic ventricular dilatation (PHVD) in the absence of associated parenchymal lesions may have a normal neurodevelopmental outcome. Objectives: To evaluate neurodevelopmental and cognitive outcomes among preterm infants with severe intraventricular hemorrhage (IVH) and PHVD requiring neurosurgical intervention. Methods: 32 preterm infants were admitted to a neonatal intensive care unit with PHVD requiring neurosurgical intervention, and were seen in the follow-up clinic for standardized cognitive, behavioral and neurological assessments between 5 and 8 years of age. Only preterm infants with a gestational age (GA) of <30 weeks, as well as preterm and full-term infants with PHVD and full-term infants with perinatal asphyxia are seen in our follow-up clinic at this age. There were 23 infants with a GA of <30 weeks in this study population. For these 23, matched controls were available and compared with the IVH group. Results: The majority (59.4%) had no impairments. None of the children with grade III and 8 of the 15 children (53%) with grade IV hemorrhage developed cerebral palsy. More subtle motor problems assessed with the Movement-ABC score were seen in 39% (n = 9); the mean IQ of all children was 93.4, and 29% of the children had an IQ of <85 (–1 SD). Timing of intervention did not have a beneficial effect on outcome. With respect to cognition, no significant differences were found between the IVH and the control group. Conclusion: The majority of the children in our population had no impairments. Cerebral palsy was not seen in any of the infants with a grade III hemorrhage.

Neurodevelopmental outcome over time of preterm born children ≤750 g at birth.

BACKGROUND Extremely low birth weight (ELBW) infants are at risk of cognitive impairment and follow-up is therefore of major importance. The age at which their neurodevelopmental outcome (NDO) can reliably be predicted differs in the literature. AIMS To describe NDO at 2, 3.5 and 5.5 years in an ELBW cohort. To examine the value of NDO at 2 years corrected age (CA) for prediction of NDO at 3.5 and 5.5 years. STUDY DESIGN A retrospective cross-sectional and longitudinal cohort study. SUBJECTS 101 children with a BW≤750 g, born between 1996 and 2005, who survived NICU admission and were included in a follow-up program. OUTCOME MEASURES NDO, measured with different tests for general development and intelligence, depending on age of assessment and classified as normal (Z-score≥-1), mildly delayed (-2≤Z-score<-1) or severely delayed (Z-score<-2). RESULTS At 2, 3.5 and 5.5 years 74.3, 82.2 and 76.2% had a normal NDO. A normal NDO at 2 years CA predicted a normal NDO at 3.5 and 5.5 years in 92% and 84% respectively. Of the children with a mildly or severely delayed NDO at 2 years CA the majority showed an improved NDO at 3.5 (69.2%) and 5.5 years (65.4%) respectively. CONCLUSIONS The majority of the children with a BW≤750 g had a normal NDO at all ages. A normal NDO at 2 years CA is a good predictor for normal outcome at 3.5 and 5.5 years, whereas a delayed NDO at 2 years CA is subject to change with the majority of the children showing a better NDO at 3.5 and 5.5 years.

Neuroimaging and neurodevelopmental outcome of preterm infants with a periventricular haemorrhagic infarction located in the temporal or frontal lobe

The aim of the study was to compare clinical and neuroimaging characteristics and neurodevelopmental outcome in preterm infants with a periventricular haemorrhagic infarction (PVHI) located in the temporal or frontal periventricular white matter.