Validity of the ESSENCE-Q neurodevelopmental screening tool in Japan

Kahoko Yasumitsu-Lovell; Lucy Thompson; Elisabeth Fernell; Masamitsu Eitoku; Narufumi Suganuma; Christopher Gillberg; Japan Environment and Children’s Study Group

doi:10.1111/dmcn.15956

Validity of the ESSENCE-Q neurodevelopmental screening tool in Japan

Kahoko Yasumitsu-Lovell^* (Corresponding Author)
, Lucy Thompson
, Elisabeth Fernell
, Masamitsu Eitoku
, Narufumi Suganuma
, Christopher Gillberg
, Japan Environment and Children’s Study Group

^*Corresponding author for this work

Research output: Contribution to journal › Article › peer-review

5 Citations (Scopus)

Abstract

Aim: To assess the validity of the Early Symptomatic Syndromes Eliciting Neurodevelopmental Clinical Examinations–Questionnaire (ESSENCE-Q), a simple screening tool for neurodevelopmental problems, in Japan. Method: Parents/caregivers completed the 11-item ESSENCE-Q for 77 612 children aged 2 years 6 months included in a national birth cohort study. Information about neurodevelopmental disorders (NDDs: autism spectrum disorder; intellectual disability and/or developmental language disorder; motor delay/motor disorder) was collected at age 3 years. Each ESSENCE-Q item was scored on a binary (0,1) scale, with a total score range of 0 to 11. Total scores and individual items were compared across children with and without NDDs. Results: NDDs were recorded in 854 children (1.1%). With a total ESSENCE-Q score cut-off of ≥3, receiver operating characteristic curve analysis showed an area under the curve of 0.91, with sensitivity 84.9%, specificity 84.8%, positive predictive value 5.9%, and negative predictive value 99.8%. The proportion of parental concerns at 2 years 6 months differed significantly by NDD status for communication (89.5% vs 14.2%) and general development (80.2% vs 7.4%). ESSENCE-Q total scores were moderately negatively correlated (−0.36, p < 0.001) with Japanese Ages and Stages Questionnaire scores. Interpretation: The parent/caregiver-completed ESSENCE-Q is useful as a tool for screening out children with neurotypical development at this early age. Further research into longer-term predictive validity will be possible as more NDD diagnoses are given as the children grow up.

Original language	English
Pages (from-to)	1611-1621
Number of pages	11
Journal	Developmental Medicine and Child Neurology
Volume	66
Issue number	12
Early online date	17 May 2024
DOIs	https://doi.org/10.1111/dmcn.15956
Publication status	Published - 1 Dec 2024

Bibliographical note

ACKNOWLEDGEMENTS
Members of the Japan Environment and Children's Study Group are as follows: Michihiro Kamijima (principal investigator, Nagoya City University, Nagoya, Japan), Shin Yamazaki (National Institute for Environmental Studies, Tsukuba, Japan), Yukihiro Ohya (National Center for Child Health and Development, Tokyo, Japan), Reiko Kishi (Hokkaido University, Sapporo, Japan), Nobuo Yaegashi (Tohoku University, Sendai, Japan), Koichi Hashimoto (Fukushima Medical University, Fukushima, Japan), Chisato Mori (Chiba University, Chiba, Japan), Shuichi Ito (Yokohama City University, Yokohama, Japan), Zentaro Yamagata (University of Yamanashi, Chuo, Japan), Hidekuni Inadera (University of Toyama, Toyama, Japan), Takeo Nakayama (Kyoto University, Kyoto, Japan), Tomotaka Sobue (Osaka University, Suita, Japan), Masayuki Shima (Hyogo Medical University, Nishinomiya, Japan), Seiji Kageyama (Tottori University, Yonago, Japan), Narufumi Suganuma (Kochi University, Nankoku, Japan), Shoichi Ohga (Kyushu University, Fukuoka, Japan), and Takahiko Katoh (Kumamoto University, Kumamoto, Japan).

We thank the JECS participants, as well as Nagamasa Maeda and Mikiya Fujieda of the Kochi Regional Centre of the JECS.

Data Availability Statement

Data are unsuitable for public deposition due to ethical restrictions and legal framework of Japan. It is prohibited by the Act on the Protection of Personal Information (Act No. 57 of 30 May 2003, amendment on 9 September 2015) to publicly deposit the data containing personal information. Ethical Guidelines for Medical and Health Research Involving Human Subjects enforced by the Japan Ministry of Education, Culture, Sports, Science and Technology and the Ministry of Health, Labour and Welfare also restricts the open sharing of the epidemiological data. All inquiries about access to data should be sent to: [email protected]. The person responsible for handling enquiries sent to this e-mail address is Dr Shoji F. Nakayama, JECS Programme Office, National Institute for Environmental Studies.(https://www.env.go.jp/chemi/ceh/en/).

Funding

The Japan Environment and Children's Study was funded by the Ministry of Environment, Japan. CG was funded by the Swedish state under the ALFGBG agreement, Ann Mari and Per Ahlqvist Foundation, and the Swedish Child Neuropsychiatry Foundation.

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Cite this

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title = "Validity of the ESSENCE-Q neurodevelopmental screening tool in Japan",

abstract = "Aim: To assess the validity of the Early Symptomatic Syndromes Eliciting Neurodevelopmental Clinical Examinations–Questionnaire (ESSENCE-Q), a simple screening tool for neurodevelopmental problems, in Japan. Method: Parents/caregivers completed the 11-item ESSENCE-Q for 77 612 children aged 2 years 6 months included in a national birth cohort study. Information about neurodevelopmental disorders (NDDs: autism spectrum disorder; intellectual disability and/or developmental language disorder; motor delay/motor disorder) was collected at age 3 years. Each ESSENCE-Q item was scored on a binary (0,1) scale, with a total score range of 0 to 11. Total scores and individual items were compared across children with and without NDDs. Results: NDDs were recorded in 854 children (1.1\%). With a total ESSENCE-Q score cut-off of ≥3, receiver operating characteristic curve analysis showed an area under the curve of 0.91, with sensitivity 84.9\%, specificity 84.8\%, positive predictive value 5.9\%, and negative predictive value 99.8\%. The proportion of parental concerns at 2 years 6 months differed significantly by NDD status for communication (89.5\% vs 14.2\%) and general development (80.2\% vs 7.4\%). ESSENCE-Q total scores were moderately negatively correlated (−0.36, p < 0.001) with Japanese Ages and Stages Questionnaire scores. Interpretation: The parent/caregiver-completed ESSENCE-Q is useful as a tool for screening out children with neurotypical development at this early age. Further research into longer-term predictive validity will be possible as more NDD diagnoses are given as the children grow up.",

author = "Kahoko Yasumitsu-Lovell and Lucy Thompson and Elisabeth Fernell and Masamitsu Eitoku and Narufumi Suganuma and Christopher Gillberg and \{Japan Environment and Children{\textquoteright}s Study Group\}",

note = "ACKNOWLEDGEMENTS Members of the Japan Environment and Children's Study Group are as follows: Michihiro Kamijima (principal investigator, Nagoya City University, Nagoya, Japan), Shin Yamazaki (National Institute for Environmental Studies, Tsukuba, Japan), Yukihiro Ohya (National Center for Child Health and Development, Tokyo, Japan), Reiko Kishi (Hokkaido University, Sapporo, Japan), Nobuo Yaegashi (Tohoku University, Sendai, Japan), Koichi Hashimoto (Fukushima Medical University, Fukushima, Japan), Chisato Mori (Chiba University, Chiba, Japan), Shuichi Ito (Yokohama City University, Yokohama, Japan), Zentaro Yamagata (University of Yamanashi, Chuo, Japan), Hidekuni Inadera (University of Toyama, Toyama, Japan), Takeo Nakayama (Kyoto University, Kyoto, Japan), Tomotaka Sobue (Osaka University, Suita, Japan), Masayuki Shima (Hyogo Medical University, Nishinomiya, Japan), Seiji Kageyama (Tottori University, Yonago, Japan), Narufumi Suganuma (Kochi University, Nankoku, Japan), Shoichi Ohga (Kyushu University, Fukuoka, Japan), and Takahiko Katoh (Kumamoto University, Kumamoto, Japan). We thank the JECS participants, as well as Nagamasa Maeda and Mikiya Fujieda of the Kochi Regional Centre of the JECS.",

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doi = "10.1111/dmcn.15956",

language = "English",

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AU - Yasumitsu-Lovell, Kahoko

AU - Thompson, Lucy

AU - Fernell, Elisabeth

AU - Eitoku, Masamitsu

AU - Suganuma, Narufumi

AU - Gillberg, Christopher

AU - Japan Environment and Children’s Study Group

N1 - ACKNOWLEDGEMENTS Members of the Japan Environment and Children's Study Group are as follows: Michihiro Kamijima (principal investigator, Nagoya City University, Nagoya, Japan), Shin Yamazaki (National Institute for Environmental Studies, Tsukuba, Japan), Yukihiro Ohya (National Center for Child Health and Development, Tokyo, Japan), Reiko Kishi (Hokkaido University, Sapporo, Japan), Nobuo Yaegashi (Tohoku University, Sendai, Japan), Koichi Hashimoto (Fukushima Medical University, Fukushima, Japan), Chisato Mori (Chiba University, Chiba, Japan), Shuichi Ito (Yokohama City University, Yokohama, Japan), Zentaro Yamagata (University of Yamanashi, Chuo, Japan), Hidekuni Inadera (University of Toyama, Toyama, Japan), Takeo Nakayama (Kyoto University, Kyoto, Japan), Tomotaka Sobue (Osaka University, Suita, Japan), Masayuki Shima (Hyogo Medical University, Nishinomiya, Japan), Seiji Kageyama (Tottori University, Yonago, Japan), Narufumi Suganuma (Kochi University, Nankoku, Japan), Shoichi Ohga (Kyushu University, Fukuoka, Japan), and Takahiko Katoh (Kumamoto University, Kumamoto, Japan). We thank the JECS participants, as well as Nagamasa Maeda and Mikiya Fujieda of the Kochi Regional Centre of the JECS.

PY - 2024/12/1

Y1 - 2024/12/1

N2 - Aim: To assess the validity of the Early Symptomatic Syndromes Eliciting Neurodevelopmental Clinical Examinations–Questionnaire (ESSENCE-Q), a simple screening tool for neurodevelopmental problems, in Japan. Method: Parents/caregivers completed the 11-item ESSENCE-Q for 77 612 children aged 2 years 6 months included in a national birth cohort study. Information about neurodevelopmental disorders (NDDs: autism spectrum disorder; intellectual disability and/or developmental language disorder; motor delay/motor disorder) was collected at age 3 years. Each ESSENCE-Q item was scored on a binary (0,1) scale, with a total score range of 0 to 11. Total scores and individual items were compared across children with and without NDDs. Results: NDDs were recorded in 854 children (1.1%). With a total ESSENCE-Q score cut-off of ≥3, receiver operating characteristic curve analysis showed an area under the curve of 0.91, with sensitivity 84.9%, specificity 84.8%, positive predictive value 5.9%, and negative predictive value 99.8%. The proportion of parental concerns at 2 years 6 months differed significantly by NDD status for communication (89.5% vs 14.2%) and general development (80.2% vs 7.4%). ESSENCE-Q total scores were moderately negatively correlated (−0.36, p < 0.001) with Japanese Ages and Stages Questionnaire scores. Interpretation: The parent/caregiver-completed ESSENCE-Q is useful as a tool for screening out children with neurotypical development at this early age. Further research into longer-term predictive validity will be possible as more NDD diagnoses are given as the children grow up.

AB - Aim: To assess the validity of the Early Symptomatic Syndromes Eliciting Neurodevelopmental Clinical Examinations–Questionnaire (ESSENCE-Q), a simple screening tool for neurodevelopmental problems, in Japan. Method: Parents/caregivers completed the 11-item ESSENCE-Q for 77 612 children aged 2 years 6 months included in a national birth cohort study. Information about neurodevelopmental disorders (NDDs: autism spectrum disorder; intellectual disability and/or developmental language disorder; motor delay/motor disorder) was collected at age 3 years. Each ESSENCE-Q item was scored on a binary (0,1) scale, with a total score range of 0 to 11. Total scores and individual items were compared across children with and without NDDs. Results: NDDs were recorded in 854 children (1.1%). With a total ESSENCE-Q score cut-off of ≥3, receiver operating characteristic curve analysis showed an area under the curve of 0.91, with sensitivity 84.9%, specificity 84.8%, positive predictive value 5.9%, and negative predictive value 99.8%. The proportion of parental concerns at 2 years 6 months differed significantly by NDD status for communication (89.5% vs 14.2%) and general development (80.2% vs 7.4%). ESSENCE-Q total scores were moderately negatively correlated (−0.36, p < 0.001) with Japanese Ages and Stages Questionnaire scores. Interpretation: The parent/caregiver-completed ESSENCE-Q is useful as a tool for screening out children with neurotypical development at this early age. Further research into longer-term predictive validity will be possible as more NDD diagnoses are given as the children grow up.

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DO - 10.1111/dmcn.15956

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JO - Developmental Medicine and Child Neurology

JF - Developmental Medicine and Child Neurology

IS - 12

ER -

Validity of the ESSENCE-Q neurodevelopmental screening tool in Japan

Abstract

Bibliographical note

Data Availability Statement

Funding

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