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Longitudinal evaluation of quality of life in 288 patients with neurofibromatosis 2

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Longitudinal evaluation of quality of life in 288 patients with neurofibromatosis 2. / Ferner, Rosalie E.; Shaw, Adam; Evans, D. Gareth; McAleer, Dympna; Halliday, Dorothy; Parry, Allyson; Raymond, F. Lucy; Durie-Gair, Juliette; Hanemann, C. Oliver; Hornigold, Rachel; Axon, Patrick; Golding, John F.

In: Journal of Neurology, Vol. 261, No. 5, 05.2014, p. 963-969.

Research output: Contribution to journalArticle

Harvard

Ferner, RE, Shaw, A, Evans, DG, McAleer, D, Halliday, D, Parry, A, Raymond, FL, Durie-Gair, J, Hanemann, CO, Hornigold, R, Axon, P & Golding, JF 2014, 'Longitudinal evaluation of quality of life in 288 patients with neurofibromatosis 2', Journal of Neurology, vol. 261, no. 5, pp. 963-969. https://doi.org/10.1007/s00415-014-7303-1

APA

Ferner, R. E., Shaw, A., Evans, D. G., McAleer, D., Halliday, D., Parry, A., Raymond, F. L., Durie-Gair, J., Hanemann, C. O., Hornigold, R., Axon, P., & Golding, J. F. (2014). Longitudinal evaluation of quality of life in 288 patients with neurofibromatosis 2. Journal of Neurology, 261(5), 963-969. https://doi.org/10.1007/s00415-014-7303-1

Vancouver

Ferner RE, Shaw A, Evans DG, McAleer D, Halliday D, Parry A et al. Longitudinal evaluation of quality of life in 288 patients with neurofibromatosis 2. Journal of Neurology. 2014 May;261(5):963-969. https://doi.org/10.1007/s00415-014-7303-1

Author

Ferner, Rosalie E. ; Shaw, Adam ; Evans, D. Gareth ; McAleer, Dympna ; Halliday, Dorothy ; Parry, Allyson ; Raymond, F. Lucy ; Durie-Gair, Juliette ; Hanemann, C. Oliver ; Hornigold, Rachel ; Axon, Patrick ; Golding, John F. / Longitudinal evaluation of quality of life in 288 patients with neurofibromatosis 2. In: Journal of Neurology. 2014 ; Vol. 261, No. 5. pp. 963-969.

Bibtex Download

@article{8abf597c7dc2433facfc99227d54caf7,
title = "Longitudinal evaluation of quality of life in 288 patients with neurofibromatosis 2",
abstract = "Advances in molecular biology have resulted in novel therapy for neurofibromatosis 2-related (NF2) tumours, highlighting the need for robust outcome measures. The disease-focused NF2 impact on quality of life (NFTI-QOL) patient questionnaire was assessed as an outcome measure for treatment in a multi-centre study. NFTI-QOL was related to clinician-rated severity (ClinSev) and genetic severity (GenSev) over repeated visits. Data were evaluated for 288 NF2 patients (n = 464 visits) attending the English national NF2 clinics from 2010 to 2012. The male-to-female ratio was equal and the mean age was 42.2 (SD 17.8) years. The analysis included NFTI-QOL eight-item score, ClinSev graded as mild, moderate, or severe, and GenSev as a rank order of the number of NF2 mutations (graded as mild, moderate, severe). The mean (SD) 8.7 (5.4) score for NFTI-QOL for either a first visit or all visits 9.2 (5.4) was similar to the published norm of 9.4 (5.5), with no significant relationships with age or gender. NFTI-QOL internal reliability was good, with a Cronbach's alpha score of 0.85 and test re-test reliability r = 0.84. NFTI related to ClinSev (r = 0.41, p <0.001; r = 0.46 for all visits), but weakly to GenSev (r = 0.16, p <0.05; r = 0.15 for all visits). ClinSev related to GenSev (r = 0.41, p <0.001; r = 0.42 for all visits). NFTI-QOL showed a good reliability and ability to detect significant longitudinal changes in the QOL of individuals. The moderate relationships of NFTI-QOL with clinician- and genetic-rated severity suggest that NFTI-QOL taps into NF2 patient experiences that are not encompassed by ClinSev rating or genotype.",
keywords = "Neurofibromatosis 2, NF2, NFTI-QOL, Vestibular schwannoma, TYPE-2, DISEASE, SEVERITY, BEVACIZUMAB, MUTATIONS",
author = "Ferner, {Rosalie E.} and Adam Shaw and Evans, {D. Gareth} and Dympna McAleer and Dorothy Halliday and Allyson Parry and Raymond, {F. Lucy} and Juliette Durie-Gair and Hanemann, {C. Oliver} and Rachel Hornigold and Patrick Axon and Golding, {John F.}",
year = "2014",
month = may,
doi = "10.1007/s00415-014-7303-1",
language = "English",
volume = "261",
pages = "963--969",
journal = "Journal of Neurology",
issn = "0340-5354",
publisher = "D. Steinkopff-Verlag",
number = "5",

}

RIS (suitable for import to EndNote) Download

TY - JOUR

T1 - Longitudinal evaluation of quality of life in 288 patients with neurofibromatosis 2

AU - Ferner, Rosalie E.

AU - Shaw, Adam

AU - Evans, D. Gareth

AU - McAleer, Dympna

AU - Halliday, Dorothy

AU - Parry, Allyson

AU - Raymond, F. Lucy

AU - Durie-Gair, Juliette

AU - Hanemann, C. Oliver

AU - Hornigold, Rachel

AU - Axon, Patrick

AU - Golding, John F.

PY - 2014/5

Y1 - 2014/5

N2 - Advances in molecular biology have resulted in novel therapy for neurofibromatosis 2-related (NF2) tumours, highlighting the need for robust outcome measures. The disease-focused NF2 impact on quality of life (NFTI-QOL) patient questionnaire was assessed as an outcome measure for treatment in a multi-centre study. NFTI-QOL was related to clinician-rated severity (ClinSev) and genetic severity (GenSev) over repeated visits. Data were evaluated for 288 NF2 patients (n = 464 visits) attending the English national NF2 clinics from 2010 to 2012. The male-to-female ratio was equal and the mean age was 42.2 (SD 17.8) years. The analysis included NFTI-QOL eight-item score, ClinSev graded as mild, moderate, or severe, and GenSev as a rank order of the number of NF2 mutations (graded as mild, moderate, severe). The mean (SD) 8.7 (5.4) score for NFTI-QOL for either a first visit or all visits 9.2 (5.4) was similar to the published norm of 9.4 (5.5), with no significant relationships with age or gender. NFTI-QOL internal reliability was good, with a Cronbach's alpha score of 0.85 and test re-test reliability r = 0.84. NFTI related to ClinSev (r = 0.41, p <0.001; r = 0.46 for all visits), but weakly to GenSev (r = 0.16, p <0.05; r = 0.15 for all visits). ClinSev related to GenSev (r = 0.41, p <0.001; r = 0.42 for all visits). NFTI-QOL showed a good reliability and ability to detect significant longitudinal changes in the QOL of individuals. The moderate relationships of NFTI-QOL with clinician- and genetic-rated severity suggest that NFTI-QOL taps into NF2 patient experiences that are not encompassed by ClinSev rating or genotype.

AB - Advances in molecular biology have resulted in novel therapy for neurofibromatosis 2-related (NF2) tumours, highlighting the need for robust outcome measures. The disease-focused NF2 impact on quality of life (NFTI-QOL) patient questionnaire was assessed as an outcome measure for treatment in a multi-centre study. NFTI-QOL was related to clinician-rated severity (ClinSev) and genetic severity (GenSev) over repeated visits. Data were evaluated for 288 NF2 patients (n = 464 visits) attending the English national NF2 clinics from 2010 to 2012. The male-to-female ratio was equal and the mean age was 42.2 (SD 17.8) years. The analysis included NFTI-QOL eight-item score, ClinSev graded as mild, moderate, or severe, and GenSev as a rank order of the number of NF2 mutations (graded as mild, moderate, severe). The mean (SD) 8.7 (5.4) score for NFTI-QOL for either a first visit or all visits 9.2 (5.4) was similar to the published norm of 9.4 (5.5), with no significant relationships with age or gender. NFTI-QOL internal reliability was good, with a Cronbach's alpha score of 0.85 and test re-test reliability r = 0.84. NFTI related to ClinSev (r = 0.41, p <0.001; r = 0.46 for all visits), but weakly to GenSev (r = 0.16, p <0.05; r = 0.15 for all visits). ClinSev related to GenSev (r = 0.41, p <0.001; r = 0.42 for all visits). NFTI-QOL showed a good reliability and ability to detect significant longitudinal changes in the QOL of individuals. The moderate relationships of NFTI-QOL with clinician- and genetic-rated severity suggest that NFTI-QOL taps into NF2 patient experiences that are not encompassed by ClinSev rating or genotype.

KW - Neurofibromatosis 2

KW - NF2

KW - NFTI-QOL

KW - Vestibular schwannoma

KW - TYPE-2

KW - DISEASE

KW - SEVERITY

KW - BEVACIZUMAB

KW - MUTATIONS

U2 - 10.1007/s00415-014-7303-1

DO - 10.1007/s00415-014-7303-1

M3 - Article

VL - 261

SP - 963

EP - 969

JO - Journal of Neurology

JF - Journal of Neurology

SN - 0340-5354

IS - 5

ER -

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