Understanding activity participation among individuals with Wolfram syndrome / Emily Bumpus in The British Journal of Occupational Therapy, Vol.81 Issue 6 (Juin 2018)

Public ISBD [article]  inThe British Journal of Occupational Therapy > Vol.81 Issue 6 (Juin 2018) . - p. 348-357 Titre : Understanding activity participation among individuals with Wolfram syndrome Type de document : texte imprimé Auteurs : Emily Bumpus ; Tamara Hershey ; Tasha Doty ; Samantha Ranck ; Meredith Gronski ; Fumihko Urano ; Erin R. Foster Année de publication : 2018 Article en page(s) : p. 348-357 Langues : Anglais (eng) Mots-clés : Wolfram syndrome  participation  occupational therapy  neurodegeneration  function Résumé : Introduction Wolfram syndrome is a rare genetic disease associated with a variety of progressive metabolic and neurologic impairments. Previous research has focused on Wolfram syndrome-related impairments and biomarkers for disease progression; however, information about how Wolfram syndrome impacts participation in daily activities is lacking. Method Wolfram syndrome (n = 45; 20 children, 25 adults) participants completed an online questionnaire about activity participation. Thirty-six non-Wolfram syndrome comparison participants (11 children; 25 adults) completed a portion of the questionnaire. Symptom data from a subset of Wolfram syndrome participants (n = 20) were also examined in relation to participation data. Results Wolfram syndrome children and adults had lower participation than non-Wolfram syndrome children and adults in almost all activity domains, and social and exercise-related activities were the most problematic. In the subset of Wolfram syndrome adults with symptom data, poorer vision, balance, gait, hearing, and overall symptom severity were related to lower participation. Conclusion Wolfram syndrome appears to negatively impact participation in a variety of activities, and this effect may increase as people age and/or Wolfram syndrome progresses. The most functionally pertinent Wolfram syndrome symptoms are those associated with neurodegeneration, especially vision loss and walking and balance problems. This study revealed symptoms and activity domains that are most relevant for people with Wolfram syndrome and, thus, can inform current practice and treatment development research. Permalink : ./index.php?lvl=notice_display&id=80154 [article] Understanding activity participation among individuals with Wolfram syndrome [texte imprimé] / Emily Bumpus ; Tamara Hershey ; Tasha Doty ; Samantha Ranck ; Meredith Gronski ; Fumihko Urano ; Erin R. Foster . - 2018 . - p. 348-357. Langues : Anglais (eng) in The British Journal of Occupational Therapy > Vol.81 Issue 6 (Juin 2018) . - p. 348-357 Mots-clés : Wolfram syndrome  participation  occupational therapy  neurodegeneration  function Résumé : Introduction Wolfram syndrome is a rare genetic disease associated with a variety of progressive metabolic and neurologic impairments. Previous research has focused on Wolfram syndrome-related impairments and biomarkers for disease progression; however, information about how Wolfram syndrome impacts participation in daily activities is lacking. Method Wolfram syndrome (n = 45; 20 children, 25 adults) participants completed an online questionnaire about activity participation. Thirty-six non-Wolfram syndrome comparison participants (11 children; 25 adults) completed a portion of the questionnaire. Symptom data from a subset of Wolfram syndrome participants (n = 20) were also examined in relation to participation data. Results Wolfram syndrome children and adults had lower participation than non-Wolfram syndrome children and adults in almost all activity domains, and social and exercise-related activities were the most problematic. In the subset of Wolfram syndrome adults with symptom data, poorer vision, balance, gait, hearing, and overall symptom severity were related to lower participation. Conclusion Wolfram syndrome appears to negatively impact participation in a variety of activities, and this effect may increase as people age and/or Wolfram syndrome progresses. The most functionally pertinent Wolfram syndrome symptoms are those associated with neurodegeneration, especially vision loss and walking and balance problems. This study revealed symptoms and activity domains that are most relevant for people with Wolfram syndrome and, thus, can inform current practice and treatment development research. Permalink : ./index.php?lvl=notice_display&id=80154

Exemplaires (1)

Cote	Support	Localisation	Section	Disponibilité
Revue	Revue	Centre de Documentation HELHa Campus Montignies	Armoires à volets	Document exclu du prêt - à consulter sur place Exclu du prêt

---

1 (1 - 1 / 1)