Developmental Medicine and Child Neurology最新文献

Aim: To explore longitudinal associations between ophthalmological outcome and development of visual perception, fine motor precision, and visual-motor integration (VMI) in children born very preterm (gestational age <32 weeks).

Method: This was a prospective longitudinal cohort study of 113 children (51 females, 62 males) born very preterm. Visual acuity, low-contrast visual acuity, stereopsis, and strabismus were investigated at 2 years 6 months (n = 98), 6 years 6 months (n = 84), and 12 years (n = 59). VMI was assessed at 6 years 6 months and 12 years, and visual perception and fine motor precision at 12 years, with the Beery-Buktenica Developmental Test of Visual-Motor Integration. Controls born at term were assessed at 6 years 6 months (n = 64) and 12 years (n = 46).

Results: For children born preterm, subnormal visual acuity at 2 years 6 months was related to both poorer VMI and fine motor precision at 12 years. Subnormal visual acuity, strabismus, and stereopsis at 6 years 6 months were also related to poorer VMI, visuo-perceptual, and fine motor measures at 12 years. These associations were not present in controls born at term.

Interpretation: Subnormal ophthalmological function at 2 years 6 months and 6 years 6 months in children born very preterm is related to poorer performance on VMI, visuo-perceptual, and fine motor precision tasks at 12 years, indicating that the quality of visual input interacts with developing visual-motor abilities. The results of this study provide new insights on the relationship between ophthalmological function and neurodevelopment after preterm birth.

Aim: To assess the effect of the augmented reality application called Minidocs on pain reduction during botulinum neurotoxin A (BoNT-A) injections in children with cerebral palsy (CP).

Method: Children with CP aged 3 to 8 years undergoing BoNT-A injection were randomized to usual pain management alone (n = 41) or combined with Minidocs (n = 39). Minidocs is an augmented reality application on a digital device offering several games including active distraction, hypnotic suggestion, and counter-aggression features. The primary outcome was the child's pain during BoNT-A injection, combining patient-reported (Faces Pain Scale [FPS]) and observer-reported (Face, Legs, Activity, Cry, Consolability scale [FLACC]) outcomes. Secondary outcomes included anxiety of children (Modified Yale Preoperative Anxiety Scale) and parents (State-Trait Anxiety Inventory Form Y-1), and satisfaction with Minidocs.

Results: In total, 14 out of 41 (34.1%) children in the control group and 7 out of 39 (17.9%) children in the experimental group experienced pain (i.e. FPS or FLACC scores ≥4). The difference between groups was not statistically significant (odds ratio 0.36; 95% confidence interval 0.11-1.16; p = 0.087). Changes in anxiety scores from before to after injection did not differ between groups. Satisfaction with the use of Minidocs was high.

Interpretation: This study did not demonstrate the benefit of augmented reality on pain reduction. The counter-aggression feature is an innovation of Minidocs worth exploring. Further studies are needed to identify profiles of children who respond to non-drug therapies.

Aim: To expand the scope of the Hand Assessment for Infants (HAI) by validating its internal scale structure and reliability in infants with a corrected age of 3 to 12 months who are at risk of bilateral or unilateral cerebral palsy (CP).

Method: This test-validation study included 274 HAI assessments collected from 221 infants (mean age = 7 months, SD = 2.4 months, 132 males) at risk of unilateral (n = 123) or bilateral (n = 98) CP. Each assessment was scored on 17 HAI items (12 unimanual and five bimanual) and analysed using the Rasch measurement model.

Results: Strong internal scale validity and person and item reliability were found for the HAI items when analysed separately for infants at risk of unilateral and bilateral CP. Logit measures for both scales were linked to a common reference frame, allowing comparable overall bimanual performance measures (HAI unit) while maintaining distinct item difficulty hierarchies.

Interpretation: The HAI provides valid and reliable measures of hand use in infants at risk of bilateral and unilateral CP regardless of the severity of manual impairments. By linking the scales, the HAI unit provides a comparable measure of bimanual performance across CP subtypes. Thus, the subtype of CP does not need to be determined before data collection.

Aim: To investigate the content and construct validity of the Both Hands Assessment (BoHA) for children and adolescents with bilateral cerebral palsy (CP), classified in Manual Ability Classification System (MACS) levels I to III, aged 18 months to 18 years.

Method: In this cross-sectional study, we included 61 adolescents with bilateral CP (37 males, 24 females, mean age 15 years 6 months, SD = 2 years 3 months) who were assessed with the BoHA to investigate content validity. Their BoHA results were combined with data from 210 children (121 males, 89 females, mean age 6 years 4 months, SD = 3 years 1 month), resulting in 271 BoHA assessments for the Rasch measurement model analyses investigating construct validity.

Results: After revising the bimanual item 'orients objects', the BoHA items were suitable for scoring bimanual performance in adolescents. Strong internal scale validity and reliable item and person measures were demonstrated for the 16 BoHA items, analysed separately for individuals with asymmetric (n = 94) and symmetric (n = 177) hand use. The BoHA logit measures were linked to the same reference frame, enabling comparable overall bimanual performance measures while maintaining separate item difficulty hierarchies.

Interpretation: The BoHA scale can measure bimanual performance in individuals with bilateral CP, classified in MACS levels I to III, aged 18 months to 18 years. This allows for monitoring development and evaluating the effectiveness of intervention from early childhood to adulthood.

Hypotonia in childhood may arise because of dysfunction across the neuroaxis. Hypotonia is termed central when it arises because of dysfunction of the central nervous system, in contrast to peripheral hypotonia which is due to neuromuscular disorders. Central hypotonia predominantly affects the axial region of the body and is the most common form of hypotonia. For many children and young people a mixed picture is seen, with some elements of inappropriately high or low tone coinciding. This review explores the pathophysiological mechanisms of central hypotonia, focusing particularly on the role of descending motor pathways and the cerebellum as potential avenues for intervention. A case is made that hypotonia is an underappreciated and underexplored component of the central motor disorder.

Aim: To explore the integrated cardiopulmonary, metabolic, and muscular response to incremental exercise in individuals with cerebral palsy (CP) compared with typically developing participants.

Method: This was a prospective cross-sectional study. Sixteen (seven male) individuals with CP (classified in Gross Motor Function Classification System levels II-V) and 30 (15 male) typically developing participants performed a treadmill-based incremental submaximal test and an exercise test to task failure. Participants used running frames (CP) or performed traditional running (typically developing participants). Metabolic and cardiopulmonary parameters were measured during both tests. Electromyography of the vastus lateralis and gastrocnemius medialis was recorded during the test to task failure.

Results: Compared with typically developing participants, individuals with CP showed decreased minute ventilation (p < 0.05), increased respiratory frequency at a comparable exercise intensity (p < 0.05), and an altered metabolic response, on the basis of the partial pressure of carbon dioxide (p < 0.05) and lactate levels (p < 0.001), during both tests. In addition, participants with CP exhibited a lower ventilatory efficiency during the test to task failure (p < 0.01). Electromyography analysis suggested peripheral skeletal muscle fatigue in the lower limbs (p < 0.05) in individuals with CP compared with typically developing participants.

Interpretation: Individuals with CP have a disproportional ventilatory response to incremental exercise, not driven by metabolic perturbations. The increased breathing frequency resulted in high rate of perceived exertion and signs of peripheral muscle fatigue compared with typically developing participants. Our findings stress the importance of interventions focused on ventilatory function in individuals with CP.