Psychiatric Diagnoses and Medications in Wolfram Syndrome.
Anxiety
ER stress
Obsessive Compulsive Disorder
Psychiatry
Wolfram Syndrome
medication
Journal
Scandinavian journal of child and adolescent psychiatry and psychology
ISSN: 2245-8875
Titre abrégé: Scand J Child Adolesc Psychiatr Psychol
Pays: Poland
ID NLM: 101608905
Informations de publication
Date de publication:
Jan 2022
Jan 2022
Historique:
entrez:
23
1
2023
pubmed:
24
1
2023
medline:
24
1
2023
Statut:
epublish
Résumé
Wolfram Syndrome is a rare genetic disorder usually resulting from pathogenic variation in the To examine lifetime psychiatric diagnoses and medication history in Wolfram Syndrome. Child, adolescent, and young adult Wolfram Syndrome participants (n=39) were assessed by a child & adolescent psychiatrist to determine best estimate DSM-5 lifetime psychiatric diagnoses as well as psychoactive medication history. In addition, the Child & Adolescent Symptom Inventory-5 (CASI-5) Parent Checklist was used to determine likely psychiatric diagnoses based on symptom counts in Wolfram Syndrome patients (n=33), type 1 diabetes (n=15), and healthy comparison (n=18) groups. Study participants with Wolfram Syndrome had high lifetime rates of anxiety disorders (77%). Also, 31% had an obsessive-compulsive spectrum disorder, 33% had a mood disorder, 31% had a neurodevelopmental or disruptive behavior disorder, and 31% had a sleep-wake disorder. More than half of Wolfram Syndrome participants had taken at least one psychoactive medication, and one third had taken at least one selective serotonin reuptake inhibitor (SSRI). Some individuals reported poor response to sertraline but better response after switching to another SSRI (fluoxetine or citalopram). In general, people with Wolfram Syndrome often reported benefit from psychotherapy and/or commonly used psychoactive medications appropriate for their psychiatric diagnoses. Wolfram Syndrome may be associated with elevated risk for anxiety and obsessive-compulsive spectrum disorders, which seem generally responsive to usual treatments for these disorders.
Sections du résumé
Background
UNASSIGNED
Wolfram Syndrome is a rare genetic disorder usually resulting from pathogenic variation in the
Objective
UNASSIGNED
To examine lifetime psychiatric diagnoses and medication history in Wolfram Syndrome.
Method
UNASSIGNED
Child, adolescent, and young adult Wolfram Syndrome participants (n=39) were assessed by a child & adolescent psychiatrist to determine best estimate DSM-5 lifetime psychiatric diagnoses as well as psychoactive medication history. In addition, the Child & Adolescent Symptom Inventory-5 (CASI-5) Parent Checklist was used to determine likely psychiatric diagnoses based on symptom counts in Wolfram Syndrome patients (n=33), type 1 diabetes (n=15), and healthy comparison (n=18) groups.
Results
UNASSIGNED
Study participants with Wolfram Syndrome had high lifetime rates of anxiety disorders (77%). Also, 31% had an obsessive-compulsive spectrum disorder, 33% had a mood disorder, 31% had a neurodevelopmental or disruptive behavior disorder, and 31% had a sleep-wake disorder. More than half of Wolfram Syndrome participants had taken at least one psychoactive medication, and one third had taken at least one selective serotonin reuptake inhibitor (SSRI). Some individuals reported poor response to sertraline but better response after switching to another SSRI (fluoxetine or citalopram). In general, people with Wolfram Syndrome often reported benefit from psychotherapy and/or commonly used psychoactive medications appropriate for their psychiatric diagnoses.
Conclusions
UNASSIGNED
Wolfram Syndrome may be associated with elevated risk for anxiety and obsessive-compulsive spectrum disorders, which seem generally responsive to usual treatments for these disorders.
Identifiants
pubmed: 36687263
doi: 10.2478/sjcapp-2022-0017
pii: sjcapp-2022-0017
pmc: PMC9828213
doi:
Types de publication
Journal Article
Langues
eng
Pagination
163-174Informations de copyright
© 2022 Angela M. Reiersen, Jacob S. Noel, Tasha Doty, Richa A. Sinkre, Anagha Narayanan, Tamara Hershey, published by Sciendo.
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