Akihito Takeuchi

Predicting executive function task scores with the Rey-Osterrieth Complex Figure.

The Boston Qualitative Scoring System (BQSS) is one of the scoring methods of the Rey-Osterrieth Complex Figure Test (ROCF). With BQSS, 17 qualitative ratings are generated, and subsequently 6 summary scores are calculated by combining several qualitative scores. Previously we showed that 5 of 6 BQSS summary scores were correlated with the scores of the several executive function tests in children. The objective of this study was to develop a new summary score which correlates with the executive function test scores more strongly than the existing summary scores. For this purpose, we conducted multiple regression analysis to predict PEN, CA, DSM of the Wisconsin Card Sorting Test Keio Version, and the Mazes scores from WISC-III, by the BQSS qualitative scores derived from ROCF drawings in copy condition. The subjects were 78 children with various neuropsychological disorders (5 years 5 months-14 years 11 months; mean: 9 years 2 months; F 22, M 56). Significant predictive models were generated for PEN, CA, and Mazes scores. Among them the models for the Mazes scores were the most accurate. The second model for the Mazes scores was most suitable for a new summary score. Its degree-of-freedom-adjusted coefficient of multiple determination and multiple correlation coefficient reached 0.467 and 0.695, respectively. A new summary score should be applied in future studies to evaluate its clinical usefulness.

Neurodevelopment in full-term small for gestational age infants: A nationwide Japanese population-based study

OBJECTIVE To investigate neurological development in small for gestational age (SGA) infants, with a focus on full-term SGA infants. METHODS We analyzed data from a large, Japanese, nationwide, population-based longitudinal survey started in 2001. We restricted the study to participants born before 42weeks of gestation (n=46,563). Parents were asked questions about motor and language development when the children were 2.5years old, and about behavioral development at 5.5years. We analyzed the relationships between SGA status and development by logistic regression. Odds ratios (ORs) and 95% confidence intervals (95% CI) were calculated for each outcome for full-term and preterm births, adjusting for potential infant- and parent-related confounding factors. We also calculated the population-attributable fractions to estimate the public impact of SGA births. RESULTS SGA full-term children were more likely to demonstrate developmental delays at 2.5years, e.g., being unable to walk alone (OR 3.0, 95% CI: 1.7, 5.3), compose a two-phrase sentence (OR 1.5, 95% CI: 1.2, 1.8), or use a spoon to eat (OR 1.5, 95% CI: 1.1, 1.9). SGA status also had some degree of negative impacts on behavioral problems at 5.5years among term children, e.g., being unable to listen without fidgeting (OR 1.2, 95% CI: 1.1, 1.3), or remain patient (OR 1.1, 95% CI: 1.0, 1.2). The public health impacts were comparable between full-term and preterm SGA children at 2.5years. CONCLUSION SGA is a risk factor for developmental delay, even in full-term infants, with non-negligible public health impacts.

Reading skills of Japanese second-graders.

Background:  A few studies have explored the prevalence of dyslexia among children who speak Japanese as their native language by evaluating them individually by means of reading‐based tasks. The present study was designed to clarify the frequency of suspected dyslexia among second‐graders attending ordinary classes.

Predicting the reading skill of Japanese children

OBJECTIVE To clarify cognitive processes underlining the development of reading in children speaking Japanese as their first language, we examined relationships between performances of cognitive tasks in the preschool period and later reading abilities. METHODS Ninety-one normally developing preschoolers (41 girls and 50 boys; 5years 4months to 6years 4months, mean 5years 10months) participated as subjects. We conducted seven cognitive tasks including phonological awareness tasks, naming tasks, and working memory tasks in the preschool period. In terms of reading tasks, the hiragana naming task was administered in the preschool period; the reading times, which is a composite score of the monomoraic syllable reading task, the word and the non-word reading tasks, and the single sentence reading task, was evaluated in first and second grade; and the kanji reading task (naming task) was tested in second grade. Ravens colored progressive matrices and picture vocabulary test revised were also conducted in first grade. Correlation analyses between task scores and stepwise multiple regression analyses were implemented. RESULTS Tasks tapping phonological awareness, lexical access, and verbal working memory showed significant correlations with reading tasks. In the multiple regression analyses the performances in the verbal working memory task played a key role in predicting character naming task scores (the hiragana naming task and the kanji reading task) while the digit naming task was an important predictor of reading times. Unexpectedly, the role of phonological (mora) awareness was modest among children speaking Japanese. CONCLUSION Cognitive functions including phonological awareness, digit naming, and verbal working memory (especially the latter two) were involved in the development of reading skills of children speaking Japanese.

Reading difficulty in school-aged very low birth weight infants in Japan

OBJECTIVE To investigate the prevalence of and the perinatal risk factors related to reading difficulty in school-aged very low birth weight infants (VLBWI) with normal intelligence. METHODS Subjects were 79 Japanese children in the second to fourth grade of elementary school who had been born at very low birth weight and who regularly visited a follow-up clinic at one of four hospitals. All members had a full-scale IQ score of 80 or higher. Perinatal information was obtained retrospectively from medical records. Each subject underwent four reading tasks, testing monomoratic syllable reading, word reading, non-word reading and short sentence reading. Subjects with an SD reading time score greater than 2.0 in two or more tasks were considered to have reading difficulty (RD). Furthermore we investigated the relations between RD and perinatal factors using logistic regression analysis adjusted for potential confounding factors. RESULTS Twenty-five (31.6%) out of 79 subjects had RD. We discovered that treated retinopathy of prematurity (tRoP) was a significant risk factor (adjusted OR=5.80, 95% confidence interval=1.51-22.33). CONCLUSION The rate of RD in school-aged VLBWI was higher than the estimated prevalence of dyslexia in Japan. Even in children with normal intelligence, long-term developmental follow-up including support for reading skills is necessary for VLBWI. Further investigation is desired to elucidate the relations between visual problems and RD in school-aged children.

Behavioral outcomes of school-aged full-term small-for-gestational-age infants: A nationwide Japanese population-based study

BACKGROUND Small for gestational age (SGA) birth is linked with neurological deficits among children at pre-school age, but the evidence is still limited on whether such deficits are still observable at school age. We investigated the association between SGA birth and behavioral development at school age among full-term infants. METHODS We analyzed data from a large, Japanese, nationwide, population-based longitudinal survey that started in 2001. We restricted the study participants to children born at 37-41weeks of gestation with information on birth weight and behavioral outcomes at 8years of age (n=33,795). Behavioral outcomes including three attentional problems and four aggressive behaviors queried at 8years of age by survey questions were used as outcome indicators. We then used logistic regression to estimate odds ratios (OR) and 95% confidence intervals (95% CI) for the associations between SGA birth and each outcome, adjusting for potential infant- and parent-related confounding factors. RESULTS Among full-term children, SGA children were more likely to interrupt people (OR 1.10, 95% CI 1.01, 1.20), unable to wait his/her turn (OR 1.17, 95% CI 1.00, 1.38), and destroy toys and/or books (OR 1.15, 95% CI 1.00, 1.31). CONCLUSIONS This is the largest study ever conducted on this issue. SGA birth is negatively associated with some attentional problems and aggressive behavior at school age among full-term children. Appropriate long-term developmental follow-up and support may be needed for full-term SGA infants.

Amelioration of disabling myoclonus in a case of DRPLA by levetiracetam

We report on an 18-year-old male patient with dentatorubral-pallidoluysian atrophy (DRPLA) (number of CAG repeats: 68) with progressive myoclonus epilepsy (PME), who showed a dramatic response to levetiracetam in terms of the intensity of myoclonus. He began to have convulsive seizures and myoclonus at 7 and 10 years of age, respectively, and his intelligence deteriorated from 12 years of age. EEG showed multifocal and diffuse spike-and-wave complexes. His convulsive seizures were suppressed from 13 years of age. At 17 years of age, the patient showed gradual intensification of erratic segmental positive myoclonus as well as frequent atonic falls that were probably attributable to negative myoclonus. Back averaging of EEG data revealed cortical discharges associated with positive myoclonus. Photosensitive myoclonic seizures were also observed. The administration of levetiracetam alleviated positive myoclonus and suppressed atonic falls, resulting in a remarkable improvement in the patients quality of daily life. Reports on the efficacy of levetiracetam for myoclonus in DRPLA are still rare, though its effect on PME is known in the context of other neurological disorders. Thus levetiracetam should be subjected to clinical trials as a means of disabling myoclonus in DRPLA.

Catch-Up Growth and Neurobehavioral Development among Full-Term, Small-for-Gestational-Age Children: A Nationwide Japanese Population-Based Study

Objective To examine the relationship between catch‐up growth of full‐term, small for gestational age (SGA) children and their neurobehavioral development. Study design Data were obtained from a population‐based nationwide Japanese longitudinal survey that started in 2001. Study participants were full‐term children with information on height at 2 years of age (n = 32 533). Catch‐up growth for SGA infants was defined as achieving a height at 2 years of age of more than ‐2.0 standard deviations for chronological age. Logistic regression analyses were used to estimate ORs and 95% CIs for the associations of SGA and catch‐up growth status with neurobehavioral development at 2.5 and 8 years of age, adjusting for potential infant‐ and parent‐related confounding factors. Results Fifteen percent of term SGA infants failed to catch up in height. At 2.5 years of age, SGA children without catch‐up growth were more likely to be unable to climb stairs (OR, 10.42; 95% CI, 5.55‐19.56) and unable to compose a 2‐word sentence (OR, 3.58; 95% CI, 1.81‐7.08) compared with children with normal growth at birth. Furthermore, SGA children without catch‐up growth were at increased risk for aggressive behaviors (OR, 3.85; 95% CI, 1.19‐12.47) at 8 years of age. Conclusions Continuous follow‐up for full‐term SGA infants with failure of catch‐up growth or poor postnatal growth may be beneficial for early detection and intervention for behavioral problems.

Intelligence test at preschool-age predicts reading difficulty among school-aged very low birth weight infants in Japan

OBJECTIVE To elucidate whether the results of an intelligence test at preschool age are predictive of reading difficulty (RD) at school age among very low birth weight infants (VLBWI). METHODS Subjects were 48 Japanese children whose birth weight was <1500 g and who regularly visited a follow-up clinic. All subjects completed the Wechsler Intelligence Scale for Children-III (WISC-III) during the last grade of kindergarten, and four reading tasks during the second to fourth grade of elementary school. All participants had a full-scale intelligence quotient score of 85 or higher. Subjects with a standard deviation reading time score greater than 2.0 in two or more tasks were considered to have RD. We evaluated the associations between each WISC-III score and RD using logistic regression analyses. Furthermore, we performed receiver operating characteristic (ROC) analysis to determine a cutoff WISC-III score predictive of RD. RESULTS In the mutually-adjusted model, the adjusted odds ratio per 1 score increase of freedom from distractibility (FD) was 0.832 (95% confidence interval: 0.720-0.962). In the ROC analysis, an FD score of <95.5 was chosen as the cutoff value for predicting RD (sensitivity, 0.77; specificity, 0.74). CONCLUSION The present study indicated that a lower FD score at preschool age, which was associated with deficits in verbal working memory and attention, is a risk factor for RD at school age among Japanese VLBWI. Further investigation is desired to clarify the cognitive deficits underlying RD in Japanese-speaking preterm children, and to establish appropriate interventions for these children.

Fetal ultrasonographic findings including cerebral hyperechogenicity in a patient with non-lethal form of Raine syndrome

Raine syndrome is a rare osteosclerotic bone dysplasia characterized by craniofacial anomalies and intracranial calcification. Most patients with Raine syndrome are of Arab ancestry and die during the neonatal period. We herein report a Japanese patient with non‐lethal Raine syndrome who presented with characteristic cerebral hyperechogenicity and a hypoplastic nose by fetal ultrasonography. She was admitted to the NICU due to pyriform aperture stenosis. Craniofacial abnormalities, intracranial calcification, osteosclerosis, chondrodysplasia punctata, and a mutation of FAM20C was identified. She was subsequently discharged without surgical intervention and is now 2 years old with mild neurodevelopmental delays. Images of cerebral hyperechogenicity by fetal ultrasonography in a non‐lethal case were described herein for the first time. This patient represents a rare occurrence of a child with Raine syndrome born to Japanese parents and confirms that this syndrome is not always lethal. Even if Raine syndrome is suspected in a fetus due to cerebral hyperechogenicity and a hypoplastic nose, cerebral hyperechogenicity without pulmonary hypoplasia does not always predict lethality or severe neurodevelopmental delays. The information provided herein will be useful for prenatal counseling.