The ESSENCE-Q: Can specialist supervision improve scoring agreement across specialist and public health nurses/nursery teachers?

AIM: This study aimed to explore whether the supervision of community public health nurses (PHNs) and nursery school teachers (NSTs) by a specialist, familiar with Early Symptomatic Syndromes Eliciting Neurodevelopmental Clinical Examinations (ESSENCE), improved the agreement of ESSENCE-Questionnaire (ESSENCE-Q) scoring, across raters. METHODS: A PHN, two NSTs, and a speech-language pathologist (SLP), familiar with ESSENCE, independently assessed 32 children. The ESSENCE-Q results were divided into the first (child 1-18) and second groups (the 19th child and the following children). Changes in score discrepancies were analysed for ESSENCE-Q cutoff scores and total ESSENCE-Q scores across raters. The SLP scores were used as a reference to evaluate sensitivity and specificity. RESULTS: The total ESSENCE-Q scores of the PHN and NSTs showed higher concordance in the second group (p < 0.05). Comparisons of the differences between the PHN/NSTs and SLP in total ESSENCE-Q scores showed a significantly smaller difference in the NSTs' scores in the second group (p < 0.05). CONCLUSION: The findings suggest that specialist supervision may lead to a better agreement between PHN and NSTs regarding ESSENCE-Q scores.

How Accurately Does the Information on Motor Development Collected During Health Checkups for Infants Predict the Diagnosis of Neurodevelopmental Disorders? - A Bayesian Network Model-Based Study.

Purpose: We investigated to what extent early motor development problems predict a future diagnosis of neurodevelopmental disorders (NDDs)/Early Symptomatic Syndromes Eliciting Neurodevelopmental Examinations (ESSENCE) by using a Bayesian network model (BN). Subjects and methods: Subjects were the children who had participated in the 18- and 36-month checkups in two cities in Japan between April 2014 and March 2015. Their motor development data at the 4-, 10- and 18-month-checkups were collected with ethical consideration. The diagnosis was confirmed at the age of six, after regular assessment in all developmental areas at a neurodevelopmental clinic. The accuracy of prediction of NDD based on posterior probabilities determined using the BN was evaluated using the area under the receiver operating characteristic (ROC) curve (AUC). The posterior probability (the optimal cut-off value) yielding the maximum Youden Index (sensitivity + specificity - 1) is determined with the ROC curve, and the sensitivity, specificity, positive predictive value (PPV), negative predictive value (NPV), and utility index (UI) were computed. Results: BN models showed associations between early motor items and developmental coordination disorders, borderline intelligence/intellectual disability, and speech and language disorder. The ROC curve for any NDD had an AUC of 0.735. The posterior probability with the maximal Youden Index was 0.138; at the optimal cut-off value, the sensitivity, specificity, PPV, NPV, UI+, and UI- were 0.619, 0.761, 0.250, 0.940, 0.155 and 0.715, respectively. Conclusion: We utilized a novel approach in detailing the associations between certain early motor problems and specific NDDs. We showed that the presence of motor development problems early in development increases the probability of a future diagnosis of any NDD. Still, the sensitivity of early motor development problems as a screening tool was not high enough to be the sole instrument for detecting NDDs. The need for a broad, holistic ESSENCE perspective when looking at the course of motor development problems was stressed.

Early neurodevelopmental problems and risk for avoidant/restrictive food intake disorder (ARFID) in 4-7-year-old children: A Japanese birth cohort study.

Background: An overrepresentation of neurodevelopmental problems (NDPs) has been observed in individuals with avoidant/restrictive food intake disorder (ARFID). Previous studies on the association between ARFID and NDPs have been limited by cross-sectional data from clinical samples of small size. This study aimed to extend previous research by using prospectively collected data in a non-clinical child cohort. We examined the occurrence of early NDPs in 4-7-year-old children with suspected ARFID and how predictive early NDPs are of ARFID. Methods: Data were collected via parent-report a sub-sample of the Japan Environment and Children's Study (JECS) including 3728 children born 2011-2014 in Kochi prefecture. NDPs were assessed biannually between 0.5 and 3 years of age with the Ages and Stages Questionnaire-3, at age 2.5 years with the ESSENCE-Q, and at age 1 and 3 years via parent-reported clinical diagnoses. ARFID was identified cross-sectionally (at age 4-7 years) using a newly developed screening tool. Logistic regressions were used to test association of (1) a composite early NDP risk score, (2) specific early NDPs, and (3) neurodevelopmental trajectories over time with ARFID. Results: Children in the highest risk percentiles of the NDP risk score had roughly three times higher odds of having suspected ARFID; the absolute risk of later ARFID for children above the 90th percentile was 3.1%. Early NDPs (excluding early feeding problems) were more predictive of later ARFID than were early feeding problems. Specific NDPs predictive of ARFID were problems with general development, communication/language, attention/concentration, social interaction, and sleep. Neurodevelopmental trajectories of children with and without suspected ARFID started to divert after age 1 year. Conclusions: The results mirror the previously observed overrepresentation of NDPs in ARFID populations. In this non-clinical child cohort, early feeding problems were common and rarely developed into ARFID; however, our findings imply that they should be monitored closely in children with high NDP risk to prevent ARFID.

Development of a parent-reported screening tool for avoidant/restrictive food intake disorder (ARFID): Initial validation and prevalence in 4-7-year-old Japanese children.

The prevalence of avoidant/restrictive food intake disorder (ARFID) in the general child population is still largely unknown and validated screening instruments are lacking. The aims of this study were (1) to investigate the prevalence of children screening positive for ARFID in a Japanese birth cohort using a newly developed parent-reported screening tool, (2) to estimate the prevalence of children with ARFID experiencing physical versus psychosocial consequences of their eating pattern, and (3) to provide preliminary evidence for the validity of the new screening tool. Data were collected from 3728 4-7-year-old children born between 2011 and 2014 in Kochi prefecture, Japan (response rate was 56.5%); a sub-sample of the Japan Environment and Children's Study (JECS). Parents completed a questionnaire including the ARFID screener and several other measures to assess convergent validity. The point prevalence of children screening positive for ARFID was 1.3%; half of them met criteria for ARFID based on psychosocial impairment alone, while the other half met diagnostic criteria relating to physical impairment (and additional psychosocial impairment in many cases). Sensory sensitivity to food characteristics (63%) and/or lack of interest in eating (51%) were the most prevalent drivers of food avoidance. Children screening positive for ARFID were lighter in weight and shorter in height, they showed more problem behaviors related to mealtimes and nutritional intake, and they were more often selective eaters and more responsive to satiety, which together provides preliminary support for the validity of the new screening tool. This is the largest screening study to date of ARFID in children up to 7 years. Future studies should examine the diagnostic validity of the new ARFID screener using clinically ascertained cases. Further research on ARFID prevalence in the general population is needed.

ESSENCE-Q obtained in routine Japanese public child health check-ups may be a valuable tool in neurodevelopmental screening.

AIM: Our aim was to extend the validity of a questionnaire developed for screening and identifying early symptomatic syndromes eliciting neurodevelopmental clinical examinations-questionnaire (ESSENCE-Q) in young children. METHODS: Early symptomatic syndromes eliciting neurodevelopmental clinical examinations-questionnaire data for 207 children, living in Aki City, Japan, in 2014-2015, were obtained from mothers, public health nurses and psychologists at 20- and 40-month routine check-ups at child healthcare centres. These were checked against subsequent ESSENCE diagnoses made by physicians. Receiver operating characteristic curves were constructed, and the area under the curves was compared. Sensitivity, specificity, positive predictive values (PPVs) and negative predictive values were calculated at optimal cut-off values. The clinical utility index was also calculated. RESULTS: When the ESSENCE-Q was used by public health nurses, it demonstrated good validity, in terms of high sensitivity and high NPVs, at the 20-month check-up, but not at 40 months. Psychologists demonstrated good validity at both ages, but mothers did not. Good negative utility indexes, indicating screening accuracy, were obtained from the psychologists at both check-ups and from nurses at 20 months. CONCLUSION: The ESSENCE-Q results used by nurses and psychologists showed good validity. Future studies should confirm the effectiveness of this tool to identify children in need of clinical detailed neurodevelopmental assessment.

Development of a new screening tool for neuromotor development in children aged two - the neuromotor 5 min exam 2-year-old version (N5E2).

OBJECTIVE: As a new screening tool for neuromotor development in children aged two, we developed the Neuromotor 5 min Exam 2-year-old version (N5E2), which can be easily administered by pediatricians or primary care physicians. In this study, as an initial attempt to examine the utility of the N5E2, the inter-rater reliability on scoring for the individual items in this scale was assessed. METHODS: The participants of the study were 29 children (aged 1-5 years, mean age = 2.79) diagnosed with a variety of neuromotor/developmental disorders/high-risk conditions. Inter-rater reliability was examined on the following 11 items in the N5E2: (1) Retrieving a rolling ball, (2) Gait, (3) Toe-walking, (4) Asymmetries of posture and/or movement, (5) Age at unsupported walking, (6) Speaking in two-word understandable sentences, (7) Hypotonus, (8) Hypertonus, (9) Eye movement, (10) Vision problem, (11) Hearing problem. The items were administered to children by two pediatricians with different expertise and clinical experience, separately. RESULTS: The results showed that among the eleven items in the N5E2 examined, a high level of agreement (κ ≥ 0.60) was found on 4 items, and a moderate level of agreement (0.40 ≤ κ < 0.60) was found on 5 items. The level of agreement somewhat improved after the dichotomization of the score; using this format, a high level of rater agreement (κ ≥ 0.60) was found on 6 out of 11 items. The analyses also revealed high inter-rater reliability on the sum score of the 11 items (r = 0.84). CONCLUSIONS: The results suggest the possibility that this brief screening tool could be feasible in settings where clinicians' experience varies, based on its inter-rater reliability on individual items between the clinicians with different expertise and amount of clinical experiences.

ESSENCE-Q - used as a screening tool for neurodevelopmental problems in public health checkups for young children in south Japan.

BACKGROUND: Screening for developmental disorders is an important task for Child Health Care. The concept of ESSENCE (early symptomatic syndromes eliciting neurodevelopmental clinical examinations) was created to cover all types of early developmental disorders and the ESSENCE-Questionnaire (ESSENCE-Q containing 12 questions with possible total scores ranging from 0 to 22) was developed as a tool for early detection of these disorders. The aim of this study was to perform a validation study in a public health situation in Japan. METHODS: The psychometric properties of the ESSENCE-Q, completed by mothers, public health nurses (PHNs), and psychologists at 18-month (n=143 children) and 36-month (n=149 children) checkups were evaluated in a small city of Japan. Results were validated against clinical ESSENCE diagnoses. Receiver operating characteristic curves were generated and compared by using the area under the curve (AUC). Optimal cutoff values were explored. RESULTS: At the 18-month checkup, AUC by mothers was 0.72, by PHNs 0.86, and by psychologists 0.82. An optimal cutoff was 3 with a high negative predictive value (NPV). At the 36-month checkup, AUC by mothers was 0.57, by PHNs 0.82, and by psychologists 0.87. Optimal cutoff was 2 with high NPV. CONCLUSION: The ESSENCE-Q completed by PHNs and psychologists had good diagnostic validity. The results suggested that almost all children scoring under cutoff would not have any ESSENCE problems/diagnoses.

Whether the Autism Spectrum Quotient consists of two different subgroups? Cluster analysis of the Autism Spectrum Quotient in general population.

The purpose of this study was to investigate whether the individuals in the general population with high scores on the Autism Spectrum Quotient constituted a single homogeneous group or not. A cohort of university students (n = 4901) was investigated by cluster analysis based on the original five subscales of the Autism Spectrum Quotient. Based on the results of the analysis, the students could be divided into six clusters: the first with low scores on all the five subscales, the second with high scores on only the 'attention to detail' subscale, the third and fourth with intermediate scores on all the subscales, the fifth with high scores on four of the five subscales but low scores on the 'attention to detail' subscale and the sixth with high scores on all the five subscales. The students with high total Autism Spectrum Quotient scores (n = 166) were divided into two groups: one with high scores on four subscales but low scores on the 'attention to detail' subscale and the other with high scores on all the five subscales. The results of this study suggested that individuals from the general population with high Autism Spectrum Quotient scores may consist of two qualitatively different groups.

ESSENCE-Q - a first clinical validation study of a new screening questionnaire for young children with suspected neurodevelopmental problems in south Japan.

BACKGROUND: Early identification of autism spectrum disorder, intellectual developmental disorder, attention-deficit/hyperactivity disorder, and other neurodevelopmental disorders/problems is crucial, yet diagnosis is often delayed for years under the often misguided "wait-and-see" paradigm. The early symptomatic syndromes eliciting neurodevelopmental clinical examinations-questionnaire (ESSENCE-Q) is a brief (12-item) screening questionnaire developed specifically for the purpose of speeding up the identification process of a wide variety of neurodevelopmental problems. The aims were to 1) estimate the reliability of the ESSENCE-Q, 2) evaluate the clinical cutoff levels suggested by the author of the ESSENCE-Q, and 3) propose optimal cutoff levels based on receiver operating characteristic analysis. METHODS: The ESSENCE-Q was used for 1 year by a psychiatrist in Kochi, Japan, assessing children under the age of 6 years referred for developmental problems. The children were also clinically assessed with regard to whether or not they met criteria for a developmental disorder (diagnosis positive and diagnosis negative groups). We contrasted the results of the ESSENCE-Q and those of clinical diagnostic assessments in 130 cases. RESULTS: Cronbach's alpha was 0.82, sensitivity was 0.94 (95% confidence interval [CI]: [0.88, 0.98]), and specificity 0.53 (95% CI: [0.28, 0.77]), which are reasonable psychometrics for a first-step screening tool. Based on receiver operating characteristic analysis, we recommended an optimal cutoff level of yes ≥2 or maybe/a little ≥3 on the ESSENCE-Q (0.87 (95% CI: [0.79, 0.92]) sensitivity and 0.77 (95% CI: [0.50, 0.93]) specificity). CONCLUSION AND IMPLICATION: The ESSENCE-Q can be a good instrument for use as a screening tool for aiding in the process of early identification of neurodevelopmental disorders in clinical settings. To establish the broader validity and reliability of the ESSENCE-Q, case-control studies and general population studies of children in different age groups are needed.

Infant Motor Delay and Early Symptomatic Syndromes Eliciting Neurodevelopmental Clinical Examinations in Japan.

BACKGROUND: Abnormalities of early motor development have been reported in autism spectrum disorder, attention-deficit/hyperactivity disorder, intellectual developmental disorder, developmental coordination disorder, and other Early Symptomatic Syndromes Eliciting Neurodevelopmental Clinical Examinations (ESSENCE). However, few studies have been conducted with a view to following up a clinically representative cohort of children coming for assessment of motor delay before age two years. We performed a prospective clinical cohort study to examine whether or not early motor delay is often an indication of ESSENCE. METHODS: The sample comprised a one-year cohort of all children who came to a Japanese neurodevelopmental center before their second birthday because of delayed or abnormal gross motor development. The children were followed up from the ESSENCE viewpoint. RESULTS: Of the 30 children, 28 (18 boys and 10 girls) (93%) were given diagnoses subsumed under the ESSENCE umbrella. Of the 15 children with an identified or strongly suspected etiology, 13 (8 boys and 5 girls) (87%) had ESSENCE disorders or symptoms. Of the 15 children without a known etiology, all had ESSENCE disorders or symptoms. CONCLUSION: This study indicated that the vast majority of children with motor delay or abnormality in the first two years of life meet criteria for a disorder within the group of ESSENCE at follow-up; this means that young children, presenting with motor problems always need a broad clinical assessment, not just related to motor function, and systematic follow-up.