The role of fidgety movements and early motor repertoire in predicting mobility outcomes in infants with myelomeningocele.

OBJECTIVE: To describe fidgety movements and co-occurring movements and postures in infants with myelomeningocele (MMC) and their association with mobility at preschool ages. METHODS: A retrospective cohort with early assessment via general movement assessment, followed by mobility assessment between 36 and 70 months of age. RESULTS: Twelve infants were included; 12 of 12 had fidgety movements in the upper limbs, with seven exhibiting them also in the hips and three in both the hips and ankles. The presence of fidgety movements in the lower limbs, kicking, a non-flat posture, a non-monotonous movement character, and a non-absent age-adequate movement repertoire were independently associated with mobility using the Hoffer modified classification and functional mobility scale (FMS) at 5 and 50 m. An optimality score was calculated based on leg movements and postures, ranging from 0 to 10 points. Infants who scored at least 4 points achieved household ambulation and FMS (5 m) of at least level 4. Community ambulation and an FMS (50 m) of level 5 were achieved with a score of at least 7.5. CONCLUSIONS: Assessing fidgety movements with other leg movements and postures in infants with MMC provided relevant information that could potentially predict mobility at preschool age and thus could be used for early intervention planning.

The General Movement Optimality Score-Revised (GMOS-R) with Socioeconomically Stratified Percentile Ranks.

Background: The general movement optimality score (GMOS) quantifies the details of general movements (GMs). We recently conducted psychometric analyses of the GMOS and developed a revised scoresheet. Consequently, the GMOS-Revised (GMOS-R) instrument necessitated validation using new percentile ranks. This study aimed to provide these percentile ranks for the GMOS-R and to investigate whether sex, preterm birth, or the infant's country of birth and residence affected the GMOS-R distribution. Methods: We applied the GMOS-R to an international sample of 1983 infants (32% female, 44% male, and 24% not disclosed), assessed in the extremely and very preterm period (10%), moderate (12%) and late (22%) preterm periods, at term (25%), and post-term age (31%). Data were grouped according to the World Bank's classification into lower- and upper-middle-income countries (LMICs and UMICs; 26%) or high-income countries (HICs; 74%), respectively. Results: We found that sex and preterm or term birth did not affect either GM classification or the GMOS-R, but the country of residence did. A lower median GMOS-R for infants with normal or poor-repertoire GMs from LMICs and UMICs compared with HICs suggests the use of specific percentile ranks for LMICs and UMICs vs. HICs. Conclusion: For clinical and scientific use, we provide a freely available GMOS-R scoring sheet, with percentile ranks reflecting socioeconomic stratification.

Health-related quality of life in children with cerebral palsy associated with congenital Zika infection.

OBJECTIVE: To describe the health-related quality of life (QOL) in children with cerebral palsy (CP) associated with congenital Zika infection. METHODS: Cross-sectional study of a consecutive series of children, followed in a referral multicentric rehabilitation network in Brazil. We invited the caregivers to respond to the Brazilian version of the Caregiver Priorities & Child Health Index of Life with Disabilities (CPCHILDTM) questionnaire. Statistical analysis was performed with the Statistical Package for the Social Sciences (SPSS) 26.0™. We used absolute and relative frequencies for categorical variables and mean and standard deviation for continuous variables. RESULTS: The sample consisted of 193 children, at mean age of 50.3±7.6 months. We observed a predominance of children with cerebral palsy (CP) with Gross Motor Function Classification System (GMFCS) level V (93.7%). Epilepsy (88.4%) was the most common comorbidity. CPCHILDTM mean scores were activities of daily living (ADL)/personal care 43.2±12.6; positioning, transferring and mobility 33.7±16.5; comfort and emotions 84.4±15.2; communication and social interaction (CoSI) 48.2±24.3; health 70.9±17.1; and overall quality of life (OQOL) 72.1±17.1. Total score was 54.8±11.3. CONCLUSIONS: Among children with cerebral palsy (CP) related to congenital Zika syndrome, the quality of life (QOL) scores were very similar to other populations with cerebral palsy (CP). The activities of positioning, transferring and mobility had the greatest impact on health-related quality of life (QOL). Rehabilitation strategies and public policies should prioritize aspects related to mobility for this population.

Health-related quality of life in children with cerebral palsy associated with congenital Zika infection / Qualidade de vida relacionada à saúde em crianças com paralisia cerebral associada a síndrome da Zika congênita

Abstract Objective: To describe the health-related quality of life (QOL) in children with cerebral palsy (CP) associated with congenital Zika infection. Methods: Cross-sectional study of a consecutive series of children, followed in a referral multicentric rehabilitation network in Brazil. We invited the caregivers to respond to the Brazilian version of the Caregiver Priorities & Child Health Index of Life with Disabilities (CPCHILDTM) questionnaire. Statistical analysis was performed with the Statistical Package for the Social Sciences (SPSS) 26.0™. We used absolute and relative frequencies for categorical variables and mean and standard deviation for continuous variables. Results: The sample consisted of 193 children, at mean age of 50.3±7.6 months. We observed a predominance of children with cerebral palsy (CP) with Gross Motor Function Classification System (GMFCS) level V (93.7%). Epilepsy (88.4%) was the most common comorbidity. CPCHILDTM mean scores were activities of daily living (ADL)/personal care 43.2±12.6; positioning, transferring and mobility 33.7±16.5; comfort and emotions 84.4±15.2; communication and social interaction (CoSI) 48.2±24.3; health 70.9±17.1; and overall quality of life (OQOL) 72.1±17.1. Total score was 54.8±11.3. Conclusions: Among children with cerebral palsy (CP) related to congenital Zika syndrome, the quality of life (QOL) scores were very similar to other populations with cerebral palsy (CP). The activities of positioning, transferring and mobility had the greatest impact on health-related quality of life (QOL). Rehabilitation strategies and public policies should prioritize aspects related to mobility for this population.

RESUMO Objetivo: Descrever a qualidade de vida relacionada à saúde em crianças com paralisia cerebral associada à infecção congênita pelo zika. Métodos: Estudo transversal de série consecutiva de casos de crianças acompanhadas em uma rede multicêntrica de reabilitação no Brasil. Convidamos o(s) cuidador(es) a responder à versão brasileira do Caregiver Priorities & Child Health Index of Life with Disabilities (CPCHILDTM). A análise estatística foi realizada com o pacote estatístico Statistical Package for the Social Sciences (SPSS) 26.0™. Para variáveis categóricas, foram utilizadas frequências absolutas e relativas e, para variáveis contínuas, média e desvio padrão. Resultados: Cento e noventa e três crianças, com idade média de 50,3±7,6 meses, foram incluídas. Observou-se predomínio de crianças com paralisia cerebral Gross Motor Function Classification System (GMFCS) nível V (93,7%). Epilepsia (88,4%) foi a comorbidade mais comum. As pontuações médias do CPCHILDTM foram: atividades de vida diária/cuidados pessoais 43,2±12,6; posicionamento, transferência e mobilidade 33,7±16,5; conforto e emoções 84,4±15,2; comunicação e interação social 48,2±24,3; saúde 70,9±17,1; e qualidade de vida geral 72,1±17,1. A pontuação total foi 54,8±11,3. Conclusões: Entre as crianças com paralisia cerebral relacionada à síndrome do zika congênita, as atividades de posicionamento, transferência e mobilidade tiveram maior impacto na qualidade de vida relacionada à saúde, semelhantemente a outras etiologias de paralisia cerebral. As estratégias de reabilitação e as políticas públicas devem priorizar os aspectos relacionados à mobilidade dessa população.

Cerebral Palsy: Early Markers of Clinical Phenotype and Functional Outcome.

The Prechtl General Movement Assessment (GMA) has become a cornerstone assessment in early identification of cerebral palsy (CP), particularly during the fidgety movement period at 3-5 months of age. Additionally, assessment of motor repertoire, such as antigravity movements and postural patterns, which form the Motor Optimality Score (MOS), may provide insight into an infant's later motor function. This study aimed to identify early specific markers for ambulation, gross motor function (using the Gross Motor Function Classification System, GMFCS), topography (unilateral, bilateral), and type (spastic, dyskinetic, ataxic, and hypotonic) of CP in a large worldwide cohort of 468 infants. We found that 95% of children with CP did not have fidgety movements, with 100% having non-optimal MOS. GMFCS level was strongly correlated to MOS. An MOS > 14 was most likely associated with GMFCS outcomes I or II, whereas GMFCS outcomes IV or V were hardly ever associated with an MOS > 8. A number of different movement patterns were associated with more severe functional impairment (GMFCS III-V), including atypical arching and persistent cramped-synchronized movements. Asymmetrical segmental movements were strongly associated with unilateral CP. Circular arm movements were associated with dyskinetic CP. This study demonstrated that use of the MOS contributes to understanding later CP prognosis, including early markers for type and severity.

Association of Infants Exposed to Prenatal Zika Virus Infection With Their Clinical, Neurologic, and Developmental Status Evaluated via the General Movement Assessment Tool.

Importance: There is an urgent need to assess neurodevelopment in Zika virus (ZIKV)-exposed infants. Objectives: To perform general movement assessment (GMA) at 9 to 20 weeks' postterm age and to evaluate whether the findings are associated with neurodevelopmental outcomes at age 12 months in infants prenatally exposed to acute maternal illness with rash in Brazil during the ZIKV outbreak and in age-matched controls. Design, Setting, and Participants: In this cohort study, infants prenatally exposed to acute maternal illness with rash were recruited at medical institutions in Rio de Janeiro and Belo Horizonte, Brazil, from February 1, 2016, to April 30, 2017, while infants without any exposure to maternal illness originated from the Graz University Audiovisual Research Database for the Interdisciplinary Analysis of Neurodevelopment. Participants were 444 infants, including 76 infants without congenital microcephaly, 35 infants with microcephaly, and 333 neurotypical children matched for sex, gestational age at birth, and age at GMA. Main Outcomes and Measures: General movement assessment performed at 9 to 20 weeks' postterm age, with negative predictive value, positive predictive value, sensitivity, and specificity generated, as well as clinical, neurologic, and developmental status (Bayley Scales of Infant and Toddler Development, Third Edition [Bayley-III] scores) at age 12 months. Motor Optimality Scores were generated based on the overall quality of the motor repertoire. Adverse outcomes were defined as a Bayley-III score less than 2 SD in at least 1 domain, a score less than 1 SD in at least 2 domains, and/or atypical neurologic findings. Results: A total of 444 infants were enrolled, including 111 children prenatally exposed to a maternal illness with rash and 333 children without any prenatal exposure to maternal illness (57.7% male and mean [SD] age, 14 [2] weeks for both groups); 82.1% (46 of 56) of ZIKV-exposed infants without congenital microcephaly were healthy at age 12 months. Forty-four of 46 infants were correctly identified by GMA at 3 months, with a negative predictive value of 94% (95% CI, 85%-97%). Seven of 10 ZIKV-exposed children without microcephaly with adverse neurodevelopmental outcomes were identified by GMA. The GMA positive predictive value was 78% (95% CI, 46%-94%), sensitivity was 70% (95% CI, 35%-93%), specificity was 96% (95% CI, 85%-99%), and accuracy was 91% (95% CI, 80%-97%). Children with microcephaly had bilateral spastic cerebral palsy; none had normal movements. The Motor Optimality Score differentiated outcomes: the median Motor Optimality Score was 23 (interquartile range [IQR], 21-26) in children with normal development, 12 (IQR, 8-19) in children with adverse outcomes, and 5 (IQR, 5-6) in children with microcephaly, a significant difference (P = .001). Conclusions and Relevance: This study suggests that although a large proportion of ZIKV-exposed infants without microcephaly develop normally, many do not. The GMA should be incorporated into routine infant assessments to enable early entry into targeted treatment programs.

Dimensionamento de amostras: determinaçäo de sua utilizaçäo e aplicaçäo metodológica em trabalhos finais de pós-graduaçäo (Mestrado e Doutorado) aprovados na Faculdade de Medicina da UFMG, 1995-1998 / Sample size calculations: a study of methodology utilization in graduate final works from School of Medicine UFMG, 1995-1998

Estudaraqm-se 134 trabalhos finais de pós-graduaçäo (entre dissertaçöes e teses com resultados numéricos), defendidos e aprovados na Faculdade de Medicina da Universidade Federal de Minas Gerais (UFMG), no período de 1995 a 1998, com o objetivo de verificar a frequência da utilizaçäo de metodologia visando à determinaçäo do tamanho da amostra a ser estudada. Vinte e sete (20,15 por cento) apresentavam-no. Relataram o cálculo 6,67 por cento (2/30) dos trabalhos experimentais e 24,04 por cento (25/104) observacionais. Nenhum estudo com animais o indicou. Verificou-se maior proporçäo de dimensionamento de amostras nos trabalhos com orientaçöes estatísticas (p=0,024; IC 95 por cento para razäo de chances = [1,13; 7,97]). Houve fraca relaçäo da presença desse cálculo com a titulaçäo, Mestrado/Doutorado, (p=0,087). Apenas 13 trabalhos (48,15 por cento) apresentaram descriçäo completa do procedimento. Em conclusäo, percebe-se valorizaçäo incipiente em relaçäo à determinaçäo do tamanho de amostra, sugerindo desconhecimento ou negligência da metodologia e das repercussöes de sua inadequaçäo. Torna-se necessário investimento em formaçäo de pesquisadores e desenvolvimento de modelos de avaliaçäo dos aspectos estatísticos dos trabalhos desenvolvidos.