Maladaptive behaviors and risk factors among the genetic subtypes of Prader-Willi syndrome
Open accessAbstract
Maladaptive behaviors among 65 people with Prader-Willi syndrome were assessed using the Reiss Screen for maladaptive behaviors. Young adults in their twenties were more likely to display aggressive behavior than adolescents and older adults. Differences in maladaptive behaviors between the typical deletion and uniparental disomy (UPD) subtypes were evaluated. The typical deletion subtype had higher self-injury and stealing scores than the UPD subtype. Subject characteristics were differentially related to maladaptive behavior among the typical deletion and UPD subtypes. Differences in maladaptive behavior were also examined between the typical deletion type I and type II subtypes. The type I deletion subtype had greater physical depression scores than the type II deletion subtype. The Reiss Screen cut-off scores were used to determine whether differences occurred between the subtypes at a clinically significant level. These findings offer insight into the health care needs of people with PWS.
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Individuals with the larger typical 15q11-q13 type I deletion in Prader-Willi and Angelman syndromes have more severe neurodevelopmental symptoms than those with the smaller typical type II deletion.
“Several studies have shown that individuals with the larger typical 15q11–q13 type I deletion which is found in both Prader-Willi and Angelman syndromes are reported to have more severe neurodevelopmental symptoms as compared to those individuals with the smaller typical type II deletion [5,6,7,8,9,10].”
From The 15q11.2 BP1–BP2 Microdeletion Syndrome: A Review (2015) · cites this paper as [7]
Individuals with PWS having type I deletions had significantly higher Reiss maladaptive behavior scores for depression than individuals with type II deletions.
“Hartley et al. [7] also found that individuals with PWS having type I deletions had significantly higher Reiss maladaptive behavior scores for depression (physical signs) than individuals with type II deletions.”
From The 15q11.2 BP1–BP2 Microdeletion Syndrome: A Review (2015) · cites this paper as [7]
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References 23 · Cited by 112
- Authors
- Hartley, S. L., MacLean, W. E., Butler, M. G., Zarcone, J., & Thompson, T.
- Affiliations
- University of Wyoming (US)
- Children's Mercy Hospital (US)
- University of Kansas Medical Center (US)
- Corresponding author
- Hartley, Sigan L.
- Venue
- American Journal of Medical Genetics Part A
- Publisher
- Wiley
- Published
- 2005-06-06
- Volume
- 136A
- Issue
- 2
- Pages
- 140-145
- ISSN
- 1552-4825 (ISSN-L)
- 1552-4833
- PMC
- PMC1896317
- OpenAlex
- W2066563455
- Cited by
- 112
- References
- 23
- Citations by year
- 2025: 4 · 2024: 4 · 2023: 5 · 2022: 7 · 2021: 7 · 2020: 8 · 2019: 5 · 2018: 5 · 2017: 5 · 2016: 2 · 2015: 5 · 2014: 11 · 2013: 4 · 2012: 12
- FWCI
- 2.16
- Type
- journal article
- Language
- en
- Open access
- green
- www.ncbi.nlm.nih.gov
- Locations
- American Journal of Medical Genetics Part A (publishedVersion)
- PubMed (publishedVersion)
- PubMed Central (acceptedVersion)
- Funders
- Eunice Kennedy Shriver National Institute of Child Health and Human Development (R01HD041672)
- Topics
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetic Syndromes and Imprinting
- Life Sciences › Neuroscience › Developmental Neuroscience › Williams Syndrome Research
- Social Sciences › Psychology › Clinical Psychology › Child and Adolescent Psychosocial and Emotional Development
- Keywords
- Prader-Willi syndrome, maladaptive behavior, aggression, self-harm, theft, uniparental disomy, genetic subtypes, age differences, depression, risk factors
- MeSH terms
- Adaptation, Psychological, Adolescent, Adult, Age Factors, Mental Disorders, Child, Chromosome Deletion, Chromosomes, Human, Pair 15, Female, Humans, Male, Middle Aged, Prader-Willi Syndrome, Risk Factors, Uniparental Disomy
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