Developmental Medicine and Child Neurology最新文献

Aim: To identify developmental trajectories of impaired hand function in infants aged 3 to 15 months with unilateral cerebral palsy (CP).

Method: Sixty-three infants (37 male; median gestational age 37 weeks [interquartile range 30-39.1 weeks]) recruited as part of a randomized trial with a confirmed diagnosis of unilateral CP were included. All infants received early upper limb therapy. The Hand Assessment for Infants (HAI) was completed at baseline and until 12 to 15 months corrected age. Group-based trajectory modelling identified groups with similar trajectories of development of impaired hand function. Multinomial logistic regression determined associations between demographic variables and trajectory membership.

Results: The three-group trajectory model comprised 'low' 29%, 'moderate' 35%, and 'high' 36% functioning groups. The relative risk ratio of being in the low or moderate relative to high group increased by 16% (95% confidence interval [CI] 1.02-1.32) and 14% (95% CI 1.01-1.29) respectively for each 1 week increase in gestational age. Males were more likely to be in the low relative to high group (relative risk ratio 7.22; 95% CI 1.6-32.5).

Interpretation: Three distinct trajectories of development of the impaired hand were identified. Males and infants born closer to term age were at higher risk of being in a low group with little improvement over time, despite receiving early intervention.

Aim: To systematically review the prevalence and incidence of osteoporosis, osteopenia, low bone mass, and fragility fracture in adults with cerebral palsy (CP), and identify the risk factors for osteoporosis and fracture.

Method: A systematic literature search was performed in the MEDLINE, PubMed, CINAHL, AMED, Cochrane Reviews, EMBASE, and EBM database reviews from inception until May 2024. Search terms covered a combination of keywords for CP, fracture, osteoporosis, incidence and prevalence, and risk factors. Participants were adults with CP aged 18 years and older. JBI critical appraisal instruments were used to assess quality and risk of bias.

Results: Seventeen of 303 studies met the eligibility criteria to assess the prevalence and incidence of osteoporosis and fracture, and 16 of 663 studies to assess risk factors. Osteoporosis prevalence was 5% in a general adult population with CP, increasing to 43% in those attending outpatient clinics. Osteoporosis incidence reported in one study was 2.85 per 1000 person years. Prevalence of fragility fracture was 5.5% overall but up to 38% in outpatient settings. Risk factors for osteoporosis and fracture included mobility status, nutritional status, and anticonvulsant use.

Interpretation: Low bone density and fracture is common in adults with CP with reduced mobility. The main risk factors for poor bone health are related to reduced mobility, nutrition, and anticonvulsant use.

Aim: To quantify optic nerve hypoplasia (ONH) and septo-optic-pituitary dysplasia (SOD) morbidities and comorbidities.

Method: A retrospective population-based study with a case-control design was undertaken using administrative health data from Manitoba, Canada. Cases were 124 patients with ONH or SOD (70 males, 54 females; age range 6 months-36 years 8 months [mean 13 years, SD 7 years 2 months]) diagnosed from 1990 to 2019, matched to 620 unrelated population-based controls (350 males, 270 females; age range 0-36 years 8 months [mean 12 years 5 months, SD 7 years 2 months]) on birth year, sex, and area of residence. Additionally, 76 cases with ONH or SOD (46 males, 30 females; age range 2 years 5 months-36 years 8 months [mean 13 years 11 months, SD 7 years 3 months]) were matched one-to-one with sibling controls (40 males, 36 females; age range 7 months-33 years 1 month [mean 11 years 8 months, SD 7 years 3 months]). We used χ² or Fisher's exact tests to test for differences in prevalence in morbidities and comorbidities between cases and controls; odds ratios (ORs) with 95% confidence intervals (CIs) were estimated. Cox proportional hazards models were used to test for differences in subgroups of cases; hazard ratios and 95% CIs were estimated.

Results: Visual impairment and visual impairment with hypopituitarism were core morbidities associated with ONH and SOD cases respectively compared to unrelated controls (OR = 58.6, 95% CI = 22.5-152.5; OR = 243.4, 95% CI = 32.9-1799.0 respectively). Developmental delay or intellectual disability (OR = 6.9, 95% CI = 3.3-14.4), autism spectrum disorder (OR = 4.0, 95% CI = 2.0-8.3), epilepsy (OR = 14.9, 95% CI = 6.1-36.5), cerebral palsy (OR = 40.9, 95% CI = 14.0-119.6), and mood or anxiety disorders (OR = 1.7, 95% CI = 1.0-2.8) were the comorbidities more common among cases with ONH and SOD. Cases matched to siblings showed similar results except for mood and anxiety disorders.

Interpretation: Visual impairment and visual impairment with hypopituitarism are the main morbidities in patients with ONH and SOD respectively, while developmental delay or intellectual disability, autism spectrum disorder, epilepsy, cerebral palsy, and mood or anxiety disorders are important comorbidities.