Perfil neuropsicológico de crianças brasileiras submetidas à cirurgia fetal da mielomeningocele
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Data
2024-03-22
Autores
Amoêdo, Sheila da Costa [UNIFESP]
Orientadores
Mello, Claudia Berlim de [UNIFESP]
Tipo
Tese de doutorado
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A cirurgia fetal marca um avanço histórico no tratamento da mielomeningocele, uma complexa malformação do sistema nervoso central, que impacta diversos aspectos do desenvolvimento humano. A hidrocefalia, as malformações encefálicas e fatores ambientais contribuem para a formação de um perfil cognitivo característico desse diagnóstico. Os resultados alcançados com a cirurgia fetal, como a redução tanto na frequência quanto na gravidade da hidrocefalia, a diminuição da necessidade de tratamento com derivação liquórica e o aumento significativo de nascimentos prematuros motivaram a investigação do funcionamento neuropsicológico de crianças brasileiras que passaram por esse procedimento. Objetivo: Analisar o funcionamento neuropsicológico de crianças brasileiras submetidas à cirurgia fetal da mielomeningocele e o efeito de variáveis sociodemográficas, clínicas, gestacionais e neurológicas. Método: Participaram do estudo 31 crianças submetidas à cirurgia fetal da mielomeningocele (MMC) e 17 crianças prematuras (PR), da mesma faixa etária e de ambos os sexos. Para a avaliação do funcionamento neuropsicológico foram aplicadas a Escala Wechsler Abreviada de Inteligência (WASI), a bateria neuropsicológica NEPSY II, a Escala de Comportamento Adaptativo - Vineland 3, o Inventário Behavior Rating Inventory of Executive Function (BRIEF), o Child Behavior Checklist (CBCL 6/18) e o questionário MTA-SNAP-IV. Foram selecionadas variáveis de exposição de relevância clínica e da literatura. Resultados: A amostra apresentou a mesma média de idade ⦏MMC (M=7, DP=1,06); PR (M=7, DP=1,18)⦐ teve o predomínio do sexo masculino e os pais tinham escolaridade entre o ensino médio e a pós graduação. O grupo MMC apresentou frequência mais baixa de herniação de Chiari (24%), de histórico de hidrocefalia (35%) e de derivação liquórica (19%) comparada às crianças que realizam o fechamento pós-natal. Em termos de desempenho intelectual, as crianças com mielomeningocele apresentaram resultados dentro da média populacional, mas com diferença estatisticamente significativa e média de escores mais baixa no subteste Raciocínio Matricial ⦏MMC (M=42, DP= 10,3); PR (M=56, DP=6,7); p< ,001⦐, Vocabulário ⦏MMC (M=51, DP= 13,8); PR (M=64, DP=9,9); p=0,002⦐, do Quociente de Inteligência Total ⦏MMC (M=91,2, DP= 17,2); PR (M=109, DP=16,1); p=0,002⦐ e de Execução ⦏MMC (M=83,5, DP= 14,0); PR (M=99,8, DP=13,1); p﹤,001⦐, em comparação às crianças prematuras. O comportamento
adaptativo do grupo mielomeningocele destacou-se positivamente no domínio de socialização, porém em comparação ao grupo de prematuros, apresentou desafios nas atividades de vida diárias, principalmente pelas especificidades relacionadas ao diagnóstico. O grupo mielomeningocele apresentou escores inferiores ao grupo prematuro em todos os domínios neuropsicológicos avaliados, especialmente em algumas tarefas que envolveram controle inibitório e flexibilidade cognitiva, memória verbal e visual de curto e longo prazo, teoria da mente em contextos verbais e percepção visoespacial. Por outro lado, o mesmo grupo obteve altos escores em tarefas de velocidade de nomeação e de repetição de pseudopalavras. Em relação ao funcionamento executivo no ambiente de casa e na escola, os grupos demonstraram diferença estatística no índice Flexibilidade ⦏MMC (M= 55, DP= 11,8); PR (M= 49, DP= 6,5); p= 0,03⦐, quando o grupo MMC demonstrou mais desafios nesta habilidade. No que diz respeito aos problemas comportamentais, o grupo MMC apresentou índice mais alto de problemas de ansiedade ⦏MMC (M= 61,5, DP= 7,7); PR (M= 56, DP= 4,8); p= 0,04⦐, enquanto o grupo PR em problemas de conduta ⦏MMC (M=52, DP= 3,7); PR (M= 53,6, DP= 5,3); p= 0,05⦐. A escolaridade dos pais, a composição familiar, índices de prematuridade, hidrocefalia e uso de válvula de derivação liquórica foram alguns dos preditores do funcionamento neuropsicológico. Conclusões: O funcionamento neuropsicológico de crianças brasileiras submetidas à cirurgia fetal para mielomeningocele se caracteriza por um desempenho intelectual dentro da média populacional, porém com médias de escores significativamente mais baixas de Quociente Intelectual de Execução e Total. Elas enfrentam desafios principalmente em tarefas que envolvem atenção, controle inibitório, flexibilidade, memória e percepção visuoespacial, além de apresentarem um índice mais alto de problemas de ansiedade. Fatores familiares, neurológicos e relacionados à prematuridade são preditores do funcionamento neuropsicológico dessas crianças.
Introduction: Fetal surgery marks a historic advance in the treatment of myelomeningocele, a complex malformation of the central nervous system, which impacts several aspects of human development. Hydrocephalus, brain malformations and environmental factors contribute to the formation of a cognitive profile characteristic of this diagnosis. The results achieved with fetal surgery, such as the reduction in the frequency and severity of hydrocephalus, the reduction in the need for treatment with CSF diversion and the significant increase in premature births, motivated the investigation of the neuropsychological functioning of Brazilian children who underwent this procedure. Objective: To analyze the neuropsychological functioning of Brazilian children undergoing fetal myelomeningocele surgery and the effect of sociodemographic, clinical, gestational and neurological variables. Method: The study included 31 children who underwent fetal myelomeningocele (MMC) surgery and 17 premature children (PR), within the same age group and of both genders. To assess the neuropsychological functioning, the Wechsler Abbreviated Scale of Intelligence (WASI), the neuropsychological battery (NEPSY II), the Adaptive Behavior Scale (Vineland 3), the Behavior Rating Inventory of ExecutiveFunction (BRIEF), the Child Behavior Checklist (CBCL 6/18) and the MTA-SNAP-IV questionnaire were applied. Exposure variables of clinical and literature relevance were selected. Results: The sample had the same average age ⦏MMC (M=7, SD=1.06); PR (M=7, SD=1.18)⦐, there was a predominance of males and the parents had education between highschool and post graduation. The MMC group had a lower frequency of Chiari herniation (24%), history of hydrocephalus (35%) and shunt (19%), compared to children who underwent postnatal closure. Concerning the intellectual performance, children with myelomeningocele showed results within the population average, but with a statistically significant difference and lower mean scores in the Vocabulary subtest ⦏MMC (M=51, SD= 13.8); PR (M=64, SD=9.9); p=0.002⦐, of the Total Intelligence Quotient ⦏MMC (M=91.2, SD= 17.2); PR (M=109, SD=16.1); p=0.002⦐ and the Execution Quotient ⦏MMC (M=83.5, SD= 14.0); PR (M=99.8, SD=13.1); p﹤.001⦐, compared to premature children. The adaptive behavior of the myelomeningocele group had a positive highlight in the socialization domain, however, compared to the premature group, it presented challenges in activities of daily living, mainly due to the specificities related to the diagnosis. However, the myelomeningocele group presented lower scores than the premature group in all neuropsychological domains assessed, especially in some tasks involving inhibitory control and cognitive flexibility, short and long-term verbal and visual memory, theory of mind in verbal contexts and visuospatial perception. On the other hand, the same group obtained high scores in naming speed and pseudoword repetition tasks. Concerning the executive functioning in different environment, such as home and school, the groups demonstrated a statistical difference in the Flexibility index ⦏MMC (M= 55, SD= 11.8); PR (M= 49, SD= 6.5); p= 0.03⦐, with the MMC group showing more challenges in this skill. Regarding to behavioral problems, the MMC group had a higher rate of anxiety problems ⦏MMC (M= 61.5, SD= 7.7); PR (M= 56, SD= 4.8); p= 0.04⦐, while the PR group showed greater vulnerability in conduct problems ⦏MMC (M=52, SD= 3.7); PR (M= 53.6, SD= 5.3); p= 0.05⦐. Parental education, family composition, rates of prematurity, hydrocephalus and use of a shunt were some of the predictors of neuropsychological functioning. Conclusions: The neuropsychological functioning of Brazilian children that underwent fetal surgery for myelomeningocele is characterized by an intellectual performance within the population average, but with significantly lower average scores in the Execution Quotient and Total Quotient. They face challenges mainly in tasks involving attention, inhibitory control, flexibility, memory and visuospatial perception, besides a higher rate of anxiety problems. Neurological factors, in addition to those related to prematurity and the family, are predictors of the neuropsychological functioning of these children.
Introduction: Fetal surgery marks a historic advance in the treatment of myelomeningocele, a complex malformation of the central nervous system, which impacts several aspects of human development. Hydrocephalus, brain malformations and environmental factors contribute to the formation of a cognitive profile characteristic of this diagnosis. The results achieved with fetal surgery, such as the reduction in the frequency and severity of hydrocephalus, the reduction in the need for treatment with CSF diversion and the significant increase in premature births, motivated the investigation of the neuropsychological functioning of Brazilian children who underwent this procedure. Objective: To analyze the neuropsychological functioning of Brazilian children undergoing fetal myelomeningocele surgery and the effect of sociodemographic, clinical, gestational and neurological variables. Method: The study included 31 children who underwent fetal myelomeningocele (MMC) surgery and 17 premature children (PR), within the same age group and of both genders. To assess the neuropsychological functioning, the Wechsler Abbreviated Scale of Intelligence (WASI), the neuropsychological battery (NEPSY II), the Adaptive Behavior Scale (Vineland 3), the Behavior Rating Inventory of ExecutiveFunction (BRIEF), the Child Behavior Checklist (CBCL 6/18) and the MTA-SNAP-IV questionnaire were applied. Exposure variables of clinical and literature relevance were selected. Results: The sample had the same average age ⦏MMC (M=7, SD=1.06); PR (M=7, SD=1.18)⦐, there was a predominance of males and the parents had education between highschool and post graduation. The MMC group had a lower frequency of Chiari herniation (24%), history of hydrocephalus (35%) and shunt (19%), compared to children who underwent postnatal closure. Concerning the intellectual performance, children with myelomeningocele showed results within the population average, but with a statistically significant difference and lower mean scores in the Vocabulary subtest ⦏MMC (M=51, SD= 13.8); PR (M=64, SD=9.9); p=0.002⦐, of the Total Intelligence Quotient ⦏MMC (M=91.2, SD= 17.2); PR (M=109, SD=16.1); p=0.002⦐ and the Execution Quotient ⦏MMC (M=83.5, SD= 14.0); PR (M=99.8, SD=13.1); p﹤.001⦐, compared to premature children. The adaptive behavior of the myelomeningocele group had a positive highlight in the socialization domain, however, compared to the premature group, it presented challenges in activities of daily living, mainly due to the specificities related to the diagnosis. However, the myelomeningocele group presented lower scores than the premature group in all neuropsychological domains assessed, especially in some tasks involving inhibitory control and cognitive flexibility, short and long-term verbal and visual memory, theory of mind in verbal contexts and visuospatial perception. On the other hand, the same group obtained high scores in naming speed and pseudoword repetition tasks. Concerning the executive functioning in different environment, such as home and school, the groups demonstrated a statistical difference in the Flexibility index ⦏MMC (M= 55, SD= 11.8); PR (M= 49, SD= 6.5); p= 0.03⦐, with the MMC group showing more challenges in this skill. Regarding to behavioral problems, the MMC group had a higher rate of anxiety problems ⦏MMC (M= 61.5, SD= 7.7); PR (M= 56, SD= 4.8); p= 0.04⦐, while the PR group showed greater vulnerability in conduct problems ⦏MMC (M=52, SD= 3.7); PR (M= 53.6, SD= 5.3); p= 0.05⦐. Parental education, family composition, rates of prematurity, hydrocephalus and use of a shunt were some of the predictors of neuropsychological functioning. Conclusions: The neuropsychological functioning of Brazilian children that underwent fetal surgery for myelomeningocele is characterized by an intellectual performance within the population average, but with significantly lower average scores in the Execution Quotient and Total Quotient. They face challenges mainly in tasks involving attention, inhibitory control, flexibility, memory and visuospatial perception, besides a higher rate of anxiety problems. Neurological factors, in addition to those related to prematurity and the family, are predictors of the neuropsychological functioning of these children.
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Citação
AMOÊDO, Sheila da Costa. Perfil neuropsicológico de crianças brasileiras submetidas à cirurgia fetal da mielomeningocele. 2024. 204 f. Tese (Doutorado em Psicobiologia) - Escola Paulista de Medicina, Universidade Federal de São Paulo (UNIFESP). São Paulo, 2024.