Genetics of Prader-Willi syndrome

Prader–Willi syndrome (PWS) is a complex genomic imprinting disorder associated with a spectrum of medical, cognitive, behavioural, and psychiatric problems and is also the most common cause of life-threatening obesity that can be effectively treated with hormone therapy and restricted diet, if dete...

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Language:English
Published: MDPI - Multidisciplinary Digital Publishing Institute 2022
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Online Access:ONIX_20221117_9783036550268_9
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description Prader–Willi syndrome (PWS) is a complex genomic imprinting disorder associated with a spectrum of medical, cognitive, behavioural, and psychiatric problems and is also the most common cause of life-threatening obesity that can be effectively treated with hormone therapy and restricted diet, if detected early. PWS is usually caused by the loss of the paternally inherited 15q11.2-q13 region and abnormal expression of genes within that region and beyond. While some genotype–phenotype correlations with delineation of clinical characteristics and natural history have emerged when comparing the three main molecular classes of PWS (maternal uniparental disomy (UPD) 15, imprinting centre defect, and deletion of paternal 15q11-q13), better awareness and informative biomarkers are still needed. These could facilitate early diagnosis, counseling, prognostic testing, as well as patient stratification for clinical trials, to improve outcomes for the affected children and their families. This Special Issue will comprise reviews and original research articles focused on the recent advances of genetics/genomics, testing, and epigenetic processes along with clinical description, co-morbidities, and natural history of PWS. Current and future directions with focus on improved screening, diagnosis, and treatment will be addressed in this rare neurodevelopmental genetic imprinting disorder influenced by the PWS genetic subtypes.
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spelling doab-20.500.12854ir-937522024-03-30T23:22:31Z Genetics of Prader-Willi syndrome Butler, Merlin G. Godler, David E. Prader-Willi syndrome appetite treatment Caralluma fimbriata extract single-case Prader–Willi syndrome thromboembolism risk factors vasculitis blood clots registry natural history age diagnosis obesity deletion uniparental disomy genetic syndrome mental illness psychosis major depressive illness obsessive-compulsive disorder autism eating disorder skin picking insurance health claims thrombosis pulmonary embolism deep venous thrombosis individuals with exogenous obesity confirmatory ICD-9 diagnostic codes Prader–Willi 15q11.2 SNORD116 atypical microdeletion food-related behavior childhood scoliosis kyphosis spinal deformities junctional kyphosis treatment options surgery bracing KATP channel activation hyperphagic obesity animal models weight BMI pediatric linear mixed models gut microbiota bacteria fungi diet hyperphagia cross-sectional games parents home exercise bone health Prader-Willi syndrome (PWS) PWS molecular classes PWS genetic subtype–phenotype correlations psychiatric behavioral phenotype growth hormone treatment pharmacogenetic testing cytochrome P450 enzymes drug interactions medication management n/a thema EDItEUR::M Medicine and Nursing Prader–Willi syndrome (PWS) is a complex genomic imprinting disorder associated with a spectrum of medical, cognitive, behavioural, and psychiatric problems and is also the most common cause of life-threatening obesity that can be effectively treated with hormone therapy and restricted diet, if detected early. PWS is usually caused by the loss of the paternally inherited 15q11.2-q13 region and abnormal expression of genes within that region and beyond. While some genotype–phenotype correlations with delineation of clinical characteristics and natural history have emerged when comparing the three main molecular classes of PWS (maternal uniparental disomy (UPD) 15, imprinting centre defect, and deletion of paternal 15q11-q13), better awareness and informative biomarkers are still needed. These could facilitate early diagnosis, counseling, prognostic testing, as well as patient stratification for clinical trials, to improve outcomes for the affected children and their families. This Special Issue will comprise reviews and original research articles focused on the recent advances of genetics/genomics, testing, and epigenetic processes along with clinical description, co-morbidities, and natural history of PWS. Current and future directions with focus on improved screening, diagnosis, and treatment will be addressed in this rare neurodevelopmental genetic imprinting disorder influenced by the PWS genetic subtypes. 2022-11-17T16:22:33Z 2022-11-17T16:22:33Z 2022 book ONIX_20221117_9783036550268_9 9783036550268 9783036550251 https://directory.doabooks.org/handle/20.500.12854/93752 eng image/jpeg Attribution 4.0 International https://mdpi.com/books/pdfview/book/6180 https://mdpi.com/books/pdfview/book/6180 MDPI - Multidisciplinary Digital Publishing Institute 10.3390/books978-3-0365-5026-8 10.3390/books978-3-0365-5026-8 46cabcaa-dd94-4bfe-87b4-55023c1b36d0 9783036550268 9783036550251 224 Basel open access
spellingShingle Prader-Willi syndrome
appetite treatment
Caralluma fimbriata extract
single-case
Prader–Willi syndrome
thromboembolism
risk factors
vasculitis
blood clots
registry
natural history
age diagnosis
obesity
deletion
uniparental disomy
genetic syndrome
mental illness
psychosis
major depressive illness
obsessive-compulsive disorder
autism
eating disorder
skin picking
insurance health claims
thrombosis
pulmonary embolism
deep venous thrombosis
individuals with exogenous obesity
confirmatory ICD-9 diagnostic codes
Prader–Willi
15q11.2
SNORD116
atypical microdeletion
food-related behavior
childhood
scoliosis
kyphosis
spinal deformities
junctional kyphosis
treatment options
surgery
bracing
KATP channel activation
hyperphagic obesity
animal models
weight
BMI
pediatric
linear mixed models
gut microbiota
bacteria
fungi
diet
hyperphagia
cross-sectional
games
parents
home
exercise
bone health
Prader-Willi syndrome (PWS)
PWS molecular classes
PWS genetic subtype–phenotype correlations
psychiatric behavioral phenotype
growth hormone treatment
pharmacogenetic testing
cytochrome P450 enzymes
drug interactions
medication management
n/a
thema EDItEUR::M Medicine and Nursing
Genetics of Prader-Willi syndrome
title Genetics of Prader-Willi syndrome
title_full Genetics of Prader-Willi syndrome
title_fullStr Genetics of Prader-Willi syndrome
title_full_unstemmed Genetics of Prader-Willi syndrome
title_short Genetics of Prader-Willi syndrome
title_sort genetics of prader willi syndrome
topic Prader-Willi syndrome
appetite treatment
Caralluma fimbriata extract
single-case
Prader–Willi syndrome
thromboembolism
risk factors
vasculitis
blood clots
registry
natural history
age diagnosis
obesity
deletion
uniparental disomy
genetic syndrome
mental illness
psychosis
major depressive illness
obsessive-compulsive disorder
autism
eating disorder
skin picking
insurance health claims
thrombosis
pulmonary embolism
deep venous thrombosis
individuals with exogenous obesity
confirmatory ICD-9 diagnostic codes
Prader–Willi
15q11.2
SNORD116
atypical microdeletion
food-related behavior
childhood
scoliosis
kyphosis
spinal deformities
junctional kyphosis
treatment options
surgery
bracing
KATP channel activation
hyperphagic obesity
animal models
weight
BMI
pediatric
linear mixed models
gut microbiota
bacteria
fungi
diet
hyperphagia
cross-sectional
games
parents
home
exercise
bone health
Prader-Willi syndrome (PWS)
PWS molecular classes
PWS genetic subtype–phenotype correlations
psychiatric behavioral phenotype
growth hormone treatment
pharmacogenetic testing
cytochrome P450 enzymes
drug interactions
medication management
n/a
thema EDItEUR::M Medicine and Nursing
topic_facet Prader-Willi syndrome
appetite treatment
Caralluma fimbriata extract
single-case
Prader–Willi syndrome
thromboembolism
risk factors
vasculitis
blood clots
registry
natural history
age diagnosis
obesity
deletion
uniparental disomy
genetic syndrome
mental illness
psychosis
major depressive illness
obsessive-compulsive disorder
autism
eating disorder
skin picking
insurance health claims
thrombosis
pulmonary embolism
deep venous thrombosis
individuals with exogenous obesity
confirmatory ICD-9 diagnostic codes
Prader–Willi
15q11.2
SNORD116
atypical microdeletion
food-related behavior
childhood
scoliosis
kyphosis
spinal deformities
junctional kyphosis
treatment options
surgery
bracing
KATP channel activation
hyperphagic obesity
animal models
weight
BMI
pediatric
linear mixed models
gut microbiota
bacteria
fungi
diet
hyperphagia
cross-sectional
games
parents
home
exercise
bone health
Prader-Willi syndrome (PWS)
PWS molecular classes
PWS genetic subtype–phenotype correlations
psychiatric behavioral phenotype
growth hormone treatment
pharmacogenetic testing
cytochrome P450 enzymes
drug interactions
medication management
n/a
thema EDItEUR::M Medicine and Nursing
url ONIX_20221117_9783036550268_9