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Aim: The aim of this study was to improve the understanding of brain function in children with autism spectrum disorder (ASD) in relation to minor neurological dysfunctions (MNDs). Method: We studied MNDs in 122 children (93 males, 29 females; mean age 8y 1mo, SD 2y 6mo) who, among a total cohort of 705 children (513 males, 192 females; mean age 9y, SD 2y 0.5mo) referred to a regional outpatient non-academic psychiatric centre in the Netherlands, were diagnosed with ASD after an extensive multidisciplinary psychiatric assessment. Children with clear neurological abnormalities (e.g. cerebral palsy or spina bifida) were excluded from the study. MNDs were assessed in all 705 children using the Touwen examination method. Special attention was paid to the severity and type of MND. Data of the children with ASD were compared with neurological morbidity data of children with other psychiatric disorders and with children in the general population, who were born at Groningen University Hospital between 1975 and 1978. Results: Seventy-four percent of the children with ASD showed complex MNDs compared with 52% of the children with other psychiatric disorders and 6% of the reference group (chi[superscript 2] = 18.0, p less than 0.001; chi[superscript 2] = 937.5, p less than 0.001 respectively). Specific dysfunctions frequently encountered in ASD were dysfunctional posture and muscle tone, fine manipulative disability, dyscoordination, and excessive associated movements. Conclusion: These findings suggest a contribution of dysfunctional supraspinal networks involving multiple parts of the brain in the pathogenesis of ASD. This is consistent with findings from neuroimaging studies, and highlights the importance of neurological examinations in paediatric psychiatric assessments.

This study of 570 children (age 9) in the Netherlands found that the presence and severity of minor neurological dysfunction was significantly related to poor performance on standardized reading, spelling, and arithmetic tests and to a lesser extent was related to ratings of distractible behavior. (Author/JDD)

Purpose: To compare family and functional outcome in infants at very high risk of cerebral palsy, after receiving the family centred programme "Coping with and Caring for infants with special needs (COPCA)" or typical infant physiotherapy. Materials and methods: Forty-three infants at very high risk were included before 9 months corrected age and randomly assigned to one year COPCA (n = 23) or typical infant physiotherapy (n = 20). Family and infant outcome were assessed before and during the intervention. Physiotherapy intervention sessions were analysed quantitatively for process analysis. Outcome was evaluated with non-parametric tests and linear mixed-effect models. Results: Between-group comparisons revealed no differences in family and infant outcomes. Within-group analysis showed that family's quality of life improved over time in the COPCA-group. Family empowerment was positively associated with intervention elements, including "caregiver coaching." Conclusions: One year of COPCA or typical infant physiotherapy resulted in similar family and functional outcomes. Yet, specific intervention elements, e.g., coaching, may increase empowerment of families of very high risk infants and may influence quality of life, which emphasizes the importance of family centred services. One year of the family centred programme "Coping with and a Caring for infants with special needs" compared with typical infant physiotherapy resulted in similar family outcome and similar functional outcome for the infants at very high risk for cerebral palsy. Specific contents of intervention, such as caregiver coaching, are associated with more family empowerment and increased quality of life. Emphasis on family needs is important in early intervention for infants at very high risk for cerebral palsy. [ABSTRACT FROM AUTHOR]

Study Question: Does Day-3 cleavage-stage PGS affect neurodevelopment of 9-year-old IVF offspring?Summary Answer: We did not find evidence of adverse consequences of Day-3 cleavage-stage PGS on neurodevelopment of 9-year-old IVF offspring, although children born after IVF with or without PGS often had a non-optimal neurological condition.What Is Known Already: Knowledge on long-term sequelae for development and health of children born following PGS is lacking. This is striking as evidence accumulates that IVF itself is associated with increased risk for impaired health and development in the offspring.Study Design Size, Duration: This prospective, assessor-blinded, multicentre, follow-up study evaluated development and health of 9-year-old IVF children born to women who were randomly assigned to IVF with PGS (PGS group) or without PGS (control group). The follow-up examination at 9 years took place between March 2014 and May 2016.Participants/materials, Setting, Methods: In total, 408 women were included and randomly assigned to IVF with or without Day-3 cleavage-stage PGS. This resulted in 52 ongoing pregnancies in the PGS group and 74 in the control group. In the PGS group, 59 children were born alive; in the control group, 85 children were born alive. At the age of 9 years, 43 children born after PGS and 56 control children participated in the study. Our primary outcome was the neurological optimality score, a sensitive measure of neurological condition assessed with a standardized, age-specific test (Touwen test). Secondary outcomes were adverse neurological condition (neurologically abnormal and the complex form of minor neurological dysfunction), cognitive development (intelligence quotient and specific domains), behaviour (parental and teacher's questionnaires), blood pressure and anthropometrics.Main Results and the Role Of Chance: Neurodevelopmental outcome of PGS children did not differ from that of controls; the neurological optimality scores (mean values [(95% CI]: PGS children 51.5 [49.3; 53.7], control children 53.1 [50.5; 55.7]) were not significantly different. The prevalences of adverse neurological outcome (in all but one child implying the presence of the complex form of minor neurological dysfunction) did not differ between the groups (PGS group 17/43 [40%], control group 19/56 [34%]), although the prevalence of complex minor neurological dysfunction in both groups was rather high. Also intelligence quotient scores of the two groups were not significantly different (PGS group 114 [108; 120]); control group 117 [109; 125]), and the behaviour, blood pressure and anthropometrics of both groups did not differ. Mean blood pressures of both groups were above the 60th percentile.Limitations Reasons For Caution: The power analysis of the study was not based on the number of children needed for the follow-up study, but on the number of women who were needed to detect an increase in ongoing pregnancy rates after PGS. In addition, our study evaluated embryo biopsy in the form of PGS at cleavage stage (Day-3 embryo biopsy), while currently PGS at blastocyst stage (Day-5 embryo biopsy) is recommended and increasingly being used.Wider Implications Of the Findings: Our findings indicate that PGS in cleavage stage embryos is not associated with adverse effects on neurological, cognitive and behavioural development, blood pressure and anthropometrics of offspring at 9 years. This is a reassuring finding as embryo biopsy in the forms of PGS and PGD is increasingly applied. However, both groups of IVF offspring showed high prevalences of the clinically relevant form of minor neurological dysfunction, which is a point of concern for the IVF community. In addition, our study confirms findings of others that IVF offspring may be at risk of an unfavourable cardiovascular outcome. These findings are alarming and highlight the importance of research on the underlying mechanisms of unfavourable neurodevelopmental and cardiovascular outcomes in IVF offspring.Study Funding/competing Interest(s): The randomized controlled trial was financially supported by the Organization for Health Research and Development (ZonMw), The Netherlands (Grant number 945-03-013). The follow-up was financially supported by the University Medical Center Groningen (Grant number: 754510), the Cornelia Foundation, and the graduate schools BCN and Share, Groningen, The Netherlands. The sponsors of the study had no role in study design, data collection, data analysis, data interpretation or writing of the report. There are no conflicts of interest.Trial Registration Number: ISRCTN76355836. [ABSTRACT FROM AUTHOR]

Background. A difficult birth at term (DBAT) may manifest as fetal acidosis and low Apgar scores and is often referred to as "perinatal asphyxia," especially when infants show signs of neonatal encephalopathy (NE). In contrast to DBAT resulting in moderate-to-severe NE, which is associated with neurodevelopmental disorders, little is known about the prognosis of less severe forms of DBAT, with or without NE. Objective. The purpose of this study was to evaluate the International Classification of Functioning, Disability and Health, Children & Youth Version activity "mobility" and other neurodevelopmental sequelae in infants with DBAT at age 6 years. Methods. The index cohort (n=62; 35 boys, 27 girls) consisted of consecutive term infants with DBAT based on clinical criteria in a Dutch nonacademic hospital from 1999 to 2005. Neonatal encephalopathy was assessed according to the Sarnat grading system and excluded infants with severe NE. The matched reference cohort (n=81; 49 boys, 32 girls) consisted of healthy term infants. The primary outcome at 6 years was limited mobility (Movement Assessment Battery for Children score ≤15th percentile). Secondary outcomes included learning and behavioral problems and the presence of minor neurological dysfunction. Results. Three children developed cerebral palsy and were excluded from analyses. Children with DBAT more often had limited mobility than children without DBAT (risk ratio [RR] = 2.44; 95% confidence interval [95% CI]= 1.16, 5.14). The risk of limited mobility rose with increasing severity of NE (mild NE: RR=3.38; 95% CI=1.40, 8.16; moderate NE: RR=4.00; 95% Cl=1.54, 10.40), and manual abilities especially were affected (RR=4.12; 95% CI=1.40, 12.14). Learning problems, need for physical therapy, and complex minor neurological dysfunction were more common in children with DBAT than in children without DBAT. Conclusions. Term infants who develop mild or moderate NE following DBAT are at increased risk for limited mobility at age 6 years. Routine monitoring of neuromotor development in these children is warranted. [ABSTRACT FROM AUTHOR]

Background. It is debatable whether adaptive riding (AR) in children with cerebral palsy (CP) improves postural control and gross motor development. Objective. The study aim was to explore the feasibility of an extensive assessment protocol for a randomized controlled trial of therapist-designed adaptive riding (TDAR) in children with CP, with the goals of assessing the effect on child outcomes and evaluating working mechanisms of sitting postural control. Design. A pretest-posttest group design with 2 baseline measurements was used. Methods. Six children (1 girl, 5 boys; age range=6-12 years, median age=8 years 9 months) with bilateral spastic CP (Gross Motor Function Classification System level III) participated. Outcomes were evaluated 3 times (TO, Tl, and T2) at 6-week intervals. TO and Tl were baseline measurements; between Tl and T2, a TDAR intervention including an integrated program of postural challenge exercises (2 times per week for 1 hour) was applied. The complex protocol included the 88-item Gross Motor Function Measure (GMFM-88) and electromyographic (EMG) recording of postural muscle activity during reaching while sitting (EMG recording at T1 and T2 only). Results. The protocol was feasible. Median GMFM-88 scores changed from 64.4 at TO to 66.7 at T1 and from 66.7 at T1 to 73.2 at T2. The change scores for all children exceeded the minimal clinically important difference of the GMFM-88. Five of 6 children showed a decrease in stereotyped top-down recruitment between T1 and T2. Limitations. Study limitations included the lack of a control group, small sample size, and potential assessor bias for all but the EMG parameters. Conclusions. The feasibility of the complex protocol was established. The data suggested that a 6-week TDAR intervention may improve gross motor function and may reduce stereotyped postural adjustments in children with CP. The limited results warrant replication in a well-powered randomized controlled trial. [ABSTRACT FROM AUTHOR]

Objective: To examine the effects of a neoprene thumb opponens splint on hand function during a self-selected activities of daily living task in children with unilateral spastic cerebral palsy with thumb-in-palm position of the affected hand.Design: Systematic evaluation of seven cases using a multiple baseline design across individuals.Setting: Outpatient clinic.Subjects: Seven children with unilateral cerebral palsy (2–7 years old), Manual Ability Classification System level 2–3 participated in the study.Interventions: Neoprene thumb opponens splints (McKie splint) were used. Children were followed for about four months. Baseline period ranged from 4 to 9 weeks, intervention period was two months and duration of follow-up one month.Main measures: Hand function was assessed using goal attainment scaling and visual analogue scales. Data was assessed visually.Results: In four children goal attainment scaling and/or visual analogue scale scores increased after introducing the splint. These effects remained when splints were not worn. Two children only benefited from the splint when it was worn. Thumb opponens splints were tolerated well by all children who participated in this study.Conclusions: Thumb opponens splints may have a positive effect on hand function in children with unilateral spastic cerebral palsy. [ABSTRACT FROM PUBLISHER]

Background. Systematic reviews have suggested that early intervention by means of specific motor training programs and general developmental programs in which parents learn how to promote infant development may be the most promising ways to promote infant motor and cognitive development of infants with or at high risk for developmental motor disorders. Objective. The purpose of this study was to investigate the effects of a recently developed pediatric physical therapy intervention program ("Coping With and Caring for Infants With Special Needs" [COPCA]) on the development of infants at high risk for developmental disorders using a combined approach of a 2-arm randomized trial and process evaluation. Setting. The study was conducted at the University Medical Center Groningen in the Netherlands. Participants and Intervention. Forty-six infants at high risk for developmental disorders were randomly assigned to receive COPCA (a family-centered program) (n=21) or traditional infant physical therapy (TIP) (n = 25) between 3 to 6 months corrected age (CA). Developmental outcome was assessed by blinded assessors at 3. 6, and 18 months CA with a neurological examination, the Alberta Infant Motor Scales, the Pediatric Evaluation of Disability Inventory, and the Mental Developmental Index (MDI) of the Bayley Scales of Infant Development. Contents of the intervention were analyzed by a quantitative video analysis of therapy sessions. Quantified physical therapy actions were correlated to evaluate associations between intervention and developmental outcome components. Results. The trial revealed that developmental outcome in both groups was largely identical. Process evaluation showed that typical COPCA actions--(1) family involvement and educational actions, (2) application of a wide variation in challenging the infant to produce motor behavior by himself or herself and allowing the infant to continue this activity, and (3) stimulation of motor behavior at the limit of the infant's capabilities--had positive correlations with developmental outcome at 18 months CA. The use of handling techniques was negatively associated with the Pediatric Evaluation of Disability Inventory outcome at 18 months CA. Limitations. Major limitations were the limited size of the groups studied and the differences between the groups in frequency and duration of physical therapy sessions. Conclusion. Extending the randomized trial with process evaluation was needed to obtain insight into associations between the components of intervention and developmental outcome. Specific therapist behaviors of parent coaching are associated with improved developmental outcome measures. Further studies are needed to examine whether these associations are caused by therapist behavior or whether therapist behavior is modified by children's motor skills. INSET: The Bottom Line. [ABSTRACT FROM AUTHOR]

Background. Evidence for effectiveness of pediatric physical therapy in infants at high risk for developmental motor disorders is limited. Therefore, "Coping With and Caring for Infants With Special Needs" (COPCA), a family-centered, early intervention program, was developed. The COPCA program is based on 2 components: (1) family involvement and educational parenting and (2) the neuromotor principles of the neuronal group selection theory. The COPCA coach uses principles of coaching to encourage the family's own capacities for solving problems of daily care and incorporating variation, along with trial and error in daily activities. Objective. The purpose of this study was to evaluate whether the content of sessions of the home-based, early intervention COPCA program differs from that of traditional infant physical therapy (TIP) sessions, which in the Netherlands are largely based on neurodevelopmental treatment. Setting. The study was conducted at the University Medical Center Groningen in the Netherlands. Design. A quantitative video analysis of therapy sessions was conducted with infants participating in a 2-arm randomized trial. Patients and Intervention. Forty-six infants at high risk for developmental motor disorders were randomly assigned to receive COPCA (n=21) or TIP (n=25) between 3 and 6 months corrected age. Intervention sessions were videotaped at 4 and 6 months corrected age and analyzed with a standardized observation protocol for the classification of physical therapy actions. Outcome parameters were relative amounts of time spent on specific physical therapy actions. Results. The content of COPCA and TIP differed substantially. For instance, in TIP sessions, more time was spent on facilitation techniques, including handling, than in COPCA sessions (29% versus 3%, respectively). During COPCA, more time was spent on family coaching and education than during TIP (16% versus 4%, respectively). Limitations. The major limitation of the study was its restriction to the Netherlands, implying that findings cannot be generalized automatically to other countries. Conclusion. The COPCA program differs broadly from TIP as applied in the Netherlands. Studies on the effectiveness of this family-centered program are needed. INSET: The Bottom Line. [ABSTRACT FROM AUTHOR]

The aim of this study was to improve the understanding of brain function in children with autism spectrum disorder (ASD) in relation to minor neurological dysfunctions (MNDs). We studied MNDs in 122 children (93 males, 29 females; mean age 8y 1mo, SD 2y 6mo) who, among a total cohort of 705 children (513 males, 192 females; mean age 9y, SD 2y 0.5mo) referred to a regional outpatient non-academic psychiatric centre in the Netherlands, were diagnosed with ASD after an extensive multidisciplinary psychiatric assessment. Children with clear neurological abnormalities (e.g. cerebral palsy or spina bifida) were excluded from the study. MNDs were assessed in all 705 children using the Touwen examination method. Special attention was paid to the severity and type of MND. Data of the children with ASD were compared with neurological morbidity data of children with other psychiatric disorders and with children in the general population, who were born at Groningen University Hospital between 1975 and 1978. Seventy-four percent of the children with ASD showed complex MNDs compared with 52% of the children with other psychiatric disorders and 6% of the reference group (χ=18.0, p<0.001; χ=937.5, p<0.001 respectively). Specific dysfunctions frequently encountered in ASD were dysfunctional posture and muscle tone, fine manipulative disability, dyscoordination, and excessive associated movements. These findings suggest a contribution of dysfunctional supraspinal networks involving multiple parts of the brain in the pathogenesis of ASD. This is consistent with findings from neuroimaging studies, and highlights the importance of neurological examinations in paediatric psychiatric assessments. [ABSTRACT FROM AUTHOR]

The aim of this study was to examine the effects of intervention in infants at risk of developmental disorders on motor outcome, as measured by the Infant Motor Profile (IMP) and using the combined approach of a randomized controlled trial and process evaluation. At a corrected age of 3 months, 46 infants (20 males, 26 females) recruited from the neonatal intensive care unit at the University Medical Centre Groningen (median birthweight 1210g, range 585-4750g; median gestational age 30wks, range 25-40wks) were included on the basis of definitely abnormal general movements. Exclusion criteria were severe congenital disorders and insufficient understanding of the Dutch language. The infants were assigned to either the family-centred COPing with and CAring for Infants with Special Needs (COPCA) intervention group ( n=21; 9 males, 12 females) or the traditional infant physiotherapy (TIP) intervention group ( n=25; 11 males, 14 females) for a period of 3 months. Three infants assigned to the TIP group (one male, two females) did not receive physiotherapy. IMP scores were measured by blinded assessors at 3, 4, 5, 6, and 18 months. At each age, the infants were neurologically examined. Physiotherapeutic sessions at 4 and 6 months were videotaped. Quantified physiotherapeutic actions were correlated with IMP scores at 6 and 18 months. The IMP scores of both the COPCA and TIP groups before, during, and after the intervention did not differ. Some physiotherapeutic actions were associated with IMP outcomes; the associations differed for infants who developed cerebral palsy ( n=10) and those who did not ( n=33). At randomized controlled trial level, the scores of both the TIP and COPCA groups did not differ in effect on motor outcome, as measured with the IMP. The analysis of physiotherapeutic actions revealed associations between these actions and IMP outcomes. However, the small sample size of this study precludes pertinent conclusions. [ABSTRACT FROM AUTHOR]

Objective: To evaluate the reliability of assessing infants' general movements (GMs) using a new classification and its validity in predicting complex minor neurological dysfunction (MND) at toddler and at school age. Design: Prospective study of two groups of infants, each consisting of a mix of low-risk and high-risk infants. Setting: University Hospital Groningen, the Netherlands. Subjects: Group A consisted of 16 Iow-risk and 21 high-risk infants; group B of 28 Iow-risk and 24 high-risk infants. Main outcome measures: Between term age and four months post term: multiple assessments of neurological condition by means of (a) assessment of GMs, and (b) a traditional neurological examination. GMs were classified into four classes using a standardized qualitative description: two classes of normal movements (normal-optimal and normal-suboptimal) and two classes of abnormal movements (mildly and definitely abnormal movements). Follow-up neurological examination with special attention to presence of MND was carried out in group A at 1‰ years, in group B at 4-9 years. Results: GMs could be assessed reliably. They were stable over age in about 60% of the infants. Both the condition of the GMs and the infant neurological condition were significantly related to neurological condition at follow-up. Best prediction of complex MND was achieved when both types of infant assessment at the age of 2-4 months post term were combined. Conclusions: The assessment of GMs is a valuable tool, in particular when combined with the traditional neurological examination, to predict at early age the development of complex MND. [ABSTRACT FROM AUTHOR]

Investigates whether minor neurological dysfunction was more closely related to learning problems than to behavioral difficulties by using the Groningen Perinatal Project (GPP) birth cohort in the Netherlands. Extrapolation of the GPP data; Description of the neurological condition in terms of the number of deviant clusters; Ratings of distractible behavior on parent and teacher questionnaires.

Objective: To evaluate whether children’s cognitive and psychomotor development and behavior at 2 years are affected by ovarian hyperstimulation and the IVF laboratory procedures or subfertility. Design: Prospective longitudinal cohort study. Setting: University Medical Center Groningen, the Netherlands. Patient(s): Singletons born after controlled ovarian hyperstimulation (COH)–IVF (n = 66) and modified natural cycle–IVF (n = 56), singletons born to subfertile couples who conceived naturally (subfertile–naturally conceived, n = 87), and a reference group of 101 2-year-old singletons born to fertile couples. Intervention(s): None. Main Outcome Measure(s): Bayley Scales of Infant Development and Achenbach Child Behavior Checklist. Result(s): Mental and psychomotor development and behavioral outcome in COH-IVF, modified natural cycle–IVF, and subfertile–natural cycle groups was not different. Developmental outcome and behavior of the subfertile groups were largely similar to those of the fertile reference group. Nevertheless, the subfertile groups scored higher on the scale of anxious-depressed behavior than the reference group. Conclusion(s): This present relatively small study found no differences in cognitive and psychomotor development and behavior at 2 years in children born after COH-IVF or modified natural cycle–IVF or naturally conceived children of subfertile parents. Replication of the study is needed before firm conclusions can be drawn. Furthermore, long-term follow-up is needed to confirm these findings in older children. [ABSTRACT FROM AUTHOR]

Background: The Alberta Infant Motor Scale (AIMS) has been developed in Canada in the 90ies. The AIMS and its Canadian norms are frequently used across the world to monitor infants' gross motor development. Currently, it is disputed whether the Canadian norms are valid for non-Canadian infants.Aims: To compare scores on the AIMS of Dutch infants with that of the Canadian norms, to compare the sequence of motor milestones in Dutch and Canadian infants, and to establish Dutch AIMS norms.Study Design: Cross-sectional study.Subjects: 1697 infants, aged 2-18 months, representative of the Dutch population (gestational age 39.7 weeks (27-42)).Outcome Measure: AIMS assessments, based on standardized video. Perinatal and social information was obtained by questionnaire and medical records. To create Dutch reference values quantile regression with polynomial splines was used.Results: 1236 Dutch infants (73%) scored below the 50th (P50) percentile of the Canadian norms, 653 (38%) below the P10 and 469 (28%) below the P5. In infants aged 6 to 12 months these values were: 567 infants (81%) < P50, 288 infants (41%) < P10, 201 infants (29%) < P5. The sequence of achievement of motor milestones of Dutch and Canadian infants was similar. Dutch norm-reference values of the AIMS were calculated.Conclusions and Implications: Gross motor development of Dutch infants is considerably slower than that of the Canadian AIMS norms sample. To prevent overdiagnosis of developmental delay and overreferral to paediatric physiotherapy Dutch AIMS norms are required. The paper introduces these norms, including percentile ranks. [ABSTRACT FROM AUTHOR]

Objective: To study birthweight in consecutively born sibling singletons conceived with and without in vitro fertilization (IVF) to disentangle the effects of maternal characteristics from those of the IVF treatment itself.Design: Population-based study.Setting: Not applicable.Patient(s): Firstborn and secondborn children from a 9-year birth cohort (1999-2007) comprising of 272,551 women who conceived two siblings.Intervention(s): No intervention; children were conceived naturally or through IVF.Main Outcome Measure(s): Birthweight.Result(s): The study included 545,102 children born by natural conception (NC) or IVF with the data set obtained from the population-based Netherlands Perinatal Registry (PRN) containing information on pregnancies, deliveries, and neonatal outcomes. We used two approaches: [1] the intersibling approach and [2] the sibling-ship approach. In the first approach we included children born to four groups of mothers who conceived in the following order (numbers indicate birth order): NC1-NC2 (reference, n = 254,721), IVF1-NC2 (n = 1342), NC1-IVF2 (n = 471), and IVF1-IVF2 (n = 687). Several comparisons were made to interpret the effects of IVF and maternal characteristics separately. In the second approach, perinatal outcomes of IVF children (n = 1,813) were compared with those of their NC siblings (n = 1,813). The intersibling analyses suggested an association between maternal characteristics and a lower birthweight, with estimates of the maternal effect ranging from -7 g (95% CI, -40; 26) to -101 g (95% CI, -170; -32). Neither the intersibling analyses nor the sibling-ship analyses indicated an additional adverse effect of IVF treatment itself.Conclusion(s): Maternal characteristics of subfertile women are associated with a lower birthweight. In vitro fertilization treatment itself does not additionally contribute to a lower birthweight in the offspring. [ABSTRACT FROM AUTHOR]

Objective To study whether specific congenital anomalies occur more often with a history of subfertility and/or the use of in vitro fertilization (IVF)/intracytoplasmic sperm injection (ICSI). Design Case-only analyses. Setting Not applicable. Patient(s) We included live births, stillbirths, and terminated pregnancies with congenital anomalies without a known cause that had a birth year between 1997 and 2010 (n = 4,525). A total of 4,185 malformed cases were born to fertile couples and 340 to subfertile couples, of whom 139 had conceived after IVF/ICSI and 201 had conceived naturally after >12 months. Intervention(s) None. Main Outcome Measure(s) The contribution, expressed in odds ratios (ORs), of a history of subfertility and IVF/ICSI to each specific type of congenital anomaly, imprinting disorder, and syndromal disorder. Result(s) We found subfertility to be associated with an increase in abdominal wall defects (adjusted OR [aOR] 2.43, 95% CI 1.05–5.62), penoscrotal hypospadia (aOR 9.83, 95% CI 3.58–27.04), right ventricular outflow tract obstruction (aOR 1.77, 95% CI 1.06–2.97), and methylation defects causing imprinting disorders (aOR 13.49, 95% CI 2.93–62.06). In vitro fertilization/ICSI was associated with an increased risk of polydactyly (OR 4.83, 95% CI 1.39–16.77) and more specifically polydactyly of the hands (OR 5.02, 95% CI 1.43–17.65). Conclusion(s) In our registry-based study, parental subfertility was associated with an increase in abdominal wall defects, penoscrotal hypospadia, right ventricular outflow tract obstruction, and methylation defects causing imprinting disorders. In vitro fertilization/ICSI was associated with an increase in polydactyly, mainly of the hands. [ABSTRACT FROM AUTHOR]

Background: Recent studies suggest that in vitro fertilization (IVF) and intracytoplasmic sperm injection (ICSI) are associated with suboptimal cardiometabolic outcome in offspring. It is unknown whether preimplantation genetic screening (PGS), which involves embryo biopsy, affects blood pressure (BP), anthropometrics, and the frequency of received medical care., Methods: In this prospective multicenter follow-up study, we assessed BP, anthropometrics, and received medical care of 4-y-old children born to women who were randomly assigned to IVF/ICSI with PGS (n = 49) or without PGS (controls; n = 64). We applied linear and generalized linear mixed-effects models to investigate possible effects of PGS., Results: BP in the PGS and control groups was similar: 102/64 and 100/64 mm Hg, respectively. Main anthropometric outcomes in the PGS vs. control group were: BMI: 16.1 vs. 15.8; triceps skinfold: 108 vs. 98 mm; and subscapular skinfold: 54 vs. 53 mm (all P values > 0.05). More PGS children than controls had received paramedical care (speech, physical, or occupational therapy: 14 (29%) vs. 9 (14%); P = 0.03 in multivariable analysis). The frequency of medicial treatment was comparable., Conclusion: PGS does not seem to affect BP or anthropometrics in 4-y-old children. The higher frequency of received paramedical care after PGS may suggest an effect of PGS on subtle developmental parameters.

Objective: To evaluate the effect of preimplantation genetic screening (PGS) on morphologic outcome in children., Design: Follow-up of a randomized controlled trial (RCT)., Setting: University hospital., Patient(s): Two-year-old children born to mothers who participated in an RCT on the efficacy of PGS: 50 children born after in vitro fertilization (IVF)/intracytoplasmic sperm injection (ICSI) with PGS (intervention group; PGS+) and 72 children born after IVF/ICSI only (control group; PGS-). Sixty-six age-matched children conceived without any form of assisted reproduction were recruited separately in a local public health service center (reference group)., Intervention(s): PGS., Main Outcome Measure(s): Body surface examination and anthropometry. The evaluation of morphologic abnormalities allowed assessment of children's phenotype in detail. Morphologic abnormalities were classified as major abnormalities (abnormal development in organogenesis, deformations, disruptions, or dysplasia) and minor anomalies (deviations in phenogenesis)., Result(s): The percentage of children with ≥ 1 major abnormality was 28% in the PGS+ and 35% in the PGS- group [difference -7%, 95% CI -23% to 10%]. The percentage of children with ≥ 1 minor anomaly was 64% in the PGS+ and 67% in the PGS- group [difference -3%, 95% CI -15% to 20%]. In the reference group 30% of the children had ≥ 1 major abnormality [95% CI 20% to 43%] and 74% had ≥ 1 minor anomaly [95% CI 62% to 84%]., Conclusion(s): No statistically significant differences were found in minor anomalies between children conceived after IVF/ICSI with or without PGS. There is < 2.5% chance of ≥ 10% more major abnormalities in children born after PGS., (Copyright © 2013 American Society for Reproductive Medicine. Published by Elsevier Inc. All rights reserved.)

Aim: the study investigated the relationships between handwriting, visuomotor integration, and neurological condition. We paid particular attention to the presence of minor neurological dysfunction (MND)., Method: participants were 200 children (131 males, 69 females; age range 8-13y) of whom 118 received mainstream education (mean age 10y 5mo, SD 1y 4mo) and 82 special education (mean age 10y 8mo, SD 1y 2mo). Each child had four assessments: a neurological examination, which paid attention to the type and severity of MND, a test to measure motor performance, a handwriting test, and the Developmental Test of Visual Motor Integration., Results: dysgraphic handwriting and slow writing speed were closely related to the severity of neurological dysfunction (both p<0.001); impaired visuomotor integration was related to the presence of MND (p<0.001) but somewhat less to its severity. Impaired handwriting and visuomotor integration were strongly related to two specific dysfunctions: fine manipulative disability and coordination problems (both p<0.001). Impaired visuomotor integration was weakly related to dysfunctional muscle tone regulation (p=0.009) and sensory dysfunction (p=0.042)., Interpretation: poor handwriting and impaired visuomotor integration are related to MND, but in a differential way. Poor handwriting is related to the severity of neurological dysfunction and to dysfunctions of complex supraspinal circuitries. Impaired visuomotor integration is associated with the presence of any of the most common types of MND.

Introduction: Child psychiatric diagnoses are generally based on a clinical examination and not on standardized questionnaires. The present study assessed whether symptom diagnostics based on clinical records facilitates the use of non-standardized clinical material for research., Method: Six hundred and eighty-five children, referred to a third level child psychiatric centre in the Netherlands, were, after extensive multidisciplinary examination, classified according to the multi-axial classification scheme for psychiatric disorders in childhood and adolescence (MAC-ICD-9). By two raters 44 behavioural symptoms were scored based on the clinical records of these children. Interrater agreement on symptoms in 50 records was performed. Principal components analysis on symptom scores of all children was performed; factor scores were related with MAC-ICD-9 classifications., Results: Interrater reliability for behavioural symptoms was excellent (kappa = 0.88). Many children with psychiatric problems suffer from a large number of behavioural symptoms. Factor scores of the symptoms revealed recognizable and well interpretable entities and indicated overlap in symptomatology and comorbidity., Conclusion: A symptom-based diagnostic approach based on extensive clinical patient files may provide a special dimension to improve the reliability of psychiatric classification.

Aim: To assess the knowledge from memory of caregivers about the most significant impairments contributing to additional care needs in children with developmental disabilities in therapeutic toddler groups., Methods: Children's needs for additional care due to impairments of physical health, motor, sensory, mental and voice and speech functions were separately classified using the capacity profile (CAP). Twenty-three therapists and teachers of toddler groups in two regional centres for paediatric rehabilitation assessed the CAP individually, unprepared and without consulting their notes or the clinical record. These CAP scores (150 CAPs of 44 children) were compared with those based on the clinical record using weighted kappa statistics., Results: Weighted kappa values for the two sets of CAP scores ranged from 0.22-0.74 (median 0.53), with the lowest scores for the sensory domain (median 0.32, range 0.22-0.52) and the highest scores for the motor domain (median 0.62, range 0.56-0.74)., Conclusion: Team members in general had only moderate remembered knowledge of the current impairments determining need of additional care. Remembered knowledge was the poorest for domains not easily observable, such as sensory functions. As this knowledge is essential for optimizing the child's daily environment, improvement of this type of knowledge should be facilitated.

Objective: To investigate the test-retest, inter-, and intraobserver reliability of the Quality of Upper Extremity Skills Test (QUEST) in young children with cerebral palsy (CP)., Design: For test-retest reliability, a test-retest design was used; for the intra- and interobserver reliability, the videotaped test was scored on 2 occasions by 1 observer and by various observers., Setting: Groups of preschool-age children in 2 general rehabilitation centers., Participants: Twenty-one children with CP (12 boys, 9 girls) aged 2 to 4.5 years (mean, 39 mo)., Interventions: Not applicable., Main Outcome Measure: Spearman correlation coefficient., Results: The data indicated that test-retest reliability was strong (rho range, .85-.94). Intraobserver agreement (rho range, .63-.95) and agreement between various observers (rho range, .72-.90) were moderate to strong., Conclusions: Test-retest and inter- and intraobserver reliability of the QUEST in preschool-age children with CP is good.