The DOI matches its Crossref record; 13 of the 13 references it cites were found in a registry or an index.
Autism and other psychiatric comorbidity in neurofibromatosis type 1: evidence from a population‐based study
Open accessAbstract
Aim To investigate psychopathology in children with neurofibromatosis type 1 (NF1), particularly the prevalence of autism spectrum disorder (ASD) and attention‐deficit–hyperactivity disorder (ADHD) symptomatology, using a population‐based sampling approach. Method Standard questionnaire screen reports were analysed for ASD (Social Responsiveness Scale, SRS), ADHD (Conners’ Parent Rating Scale‐ Revised, CPRS‐R), and other psychiatric morbidity (Strengths and Difficulties Questionnaire, SDQ) from parents and teachers of children aged from 4 to 16 years (112 females, 95 males) on the UK North West Regional Genetic Service register for NF1. Results Parental response rate was 52.7% (109/207 children; 59 females, 50 males, mean age 9y 11mo, SD 3y 3mo). The SRS showed that in 29.4% (32/109) of children, autism was in the severe, clinical range (T‐score>75) and in 26.6% (29/109) in the mild to moderate range (T‐score 60–75). CPRS‐R scores showed that in 53.8% (57/106) of children autism was in the clinical ADHD range (ADHD index T‐score>65). Based on their scores on the SDQ total difficulties scale, 41.5% (44/106) of children were in the abnormal range and 14.2% (15/106) were in the borderline range. Twenty‐five per cent (26/104) of children met criteria for both clinical autism and ADHD. Interpretation This representative population‐based sample of children with NF1 indicates a high prevalence of ASD symptoms associated with NF1 as well as substantial co‐occurrence with ADHD symptoms. The findings clarify the psychopathology of NF1 and show the disorder as a potentially important single‐gene cause for autism symptoms.
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Cites
The Strengths and Difficulties Questionnaire: A research note
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“The questionnaires used are standard and well validated as screening proxies for diagnosis in population studies in their respective diagnostic fields.(43,44) In validation studies, SRS clinical range T-scores greater than 75 show sensitivity of 0.”
MethodComorbidity in Child Psychopathology: Concepts, Issues and Research Strategies
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Ratner, N., & Miller, S. J. (2015) · Nature Reviews Cancer · doi:10.1038/nrc3911 · Cited by 493
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Johnson, M. H., Gliga, T., Jones, E., & Charman, T. (2015) · Journal of Child Psychology and Psychiatry · doi:10.1111/jcpp.12328 · Cited by 257
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Adviento, B., Corbin, I. L., Widjaja, F., Desachy, G., Enrique, N., Rosser, T., Risi, S., Marco, E. J., Hendren, R. L., Bearden, C. E., et al. (2014) · Journal of Medical Genetics · doi:10.1136/jmedgenet-2013-101951 · Cited by 174
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Antshel, K. M., Zhang-James, Y., & Faraone, S. V. (2013) · Expert Review of Neurotherapeutics · doi:10.1586/14737175.2013.840417 · Cited by 147
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Metadata
- Authors
- GARG, S., LEHTONEN, A., HUSON, S. M., EMSLEY, R., TRUMP, D., EVANS, D. G., & GREEN, J.
- Venue
- Developmental Medicine & Child Neurology
- Publisher
- Wiley
- Published
- 2012-11-16
- Volume
- 55
- Issue
- 2
- Pages
- 139-145
- ISSN
- 0012-1622 (ISSN-L)
- 1469-8749
- OpenAlex
- W1572843489
- Cited by
- 182
- References
- 13
- FWCI
- 7.48
- Type
- journal article
- Language
- en
- Open access
- bronze
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