Burden of adult neurofibromatosis 1 questionnaire: translation and psychometric properties of the Persian version

Orphanet Journal of Rare Diseases

Research

Reza Jahanshahi¹  Shohreh Ghasemi²  Fatemeh Mirzaei³  Zahra Yasaghi⁴  Samira Foji⁵  Akram Sanagoo⁶  Leila Jouybari⁶ 

[1] BSN, Student Research Committee, Golestan University of Medical Sciences, Gorgan, Iran;DDS, MSc of Oral Surgery, Adjunct Clinical, Department of Augusta University, Augusta, GA, USA;DDS, Student Research Committee, Golestan University of Medical Sciences, Gorgan, Iran;Master of critical care nursing, school of nursing and midwifery, Golestan University of Medical Sciences, Gorgan, Iran;School of Nursing and Midwifery, Sabzevar university of medical scinces, Sabzevar, Iran;School of nursing and midwifery, Golestan University of Medical Sciences, Gorgan, Iran;

关键词: Cost of illness;    Iran;    Psychometrics;    Rare diseases;    Von Recklinghausen’s disease;   

DOI  :  10.1186/s13023-023-02681-x

 received in 2022-08-01, accepted in 2023-04-02,  发布年份 2023

来源: Springer

【 摘 要 】

BackgroundThe notion of “burden” has taken a key place in the evaluation of care, particularly in the case of rare diseases. The aim of this study was to evaluate the psychometric properties of the burden of neurofibromatosis 1 questionnaire (BoN) and to determine the perceived disease burden.ResultsThe 15-item BoN was translated into Persian, and no items were removed based on content validity. The adequacy of the sample was acceptable (KMO = 0.902), and Bartlett’s test of sphericity revealed statistically significant results (P < 0.001). Exploratory factor analysis revealed three factors. The reliability of the scale was good (Cronbach’s alpha: 0.90), and the intraclass coefficient was 0.85. The severity of the burden of neurofibromatosis was moderate, and the total mean burden score was 33.12 ± 16.12.ConclusionsThe Persian version of the BoN is an acceptable tool in terms of structure and content, and it specifically assesses the practical aspects of daily activities for patients with neurofibromatosis.

【 授权许可】

CC BY   
© The Author(s) 2023