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Voice-related quality of life in adults with neurofibromatosis type 1

Marjan Cosyns UGent, Geert Mortier UGent, Sandra Janssens UGent and John Van Borsel UGent (2012) JOURNAL OF VOICE. 26(2). p.e57-e62
abstract
Objective. Neurofibromatosis type 1 (NF1) is an autosomal dominant neurocutaneous disorder, which occurs in about one in 3000-4000 people. Its hallmark features include multiple cafe-au-lait spots and neurofibromas. Voice characteristics of NF1 patients have been documented using both subjective and objective evaluations. However, the relative impact of these voice characteristics on daily activities has, as far as we know, not been examined yet. Methods. Thirty adults with NF1 were asked to complete the Flemish Dutch version of the Voice Handicap Index (VHI). Additionally, the level of severity of NF1 was rated and Dysphonia Severity Indices (DSIs) were collected. The results of the patient group were compared with those of a healthy control group frequency matched for age, gender, smoking behavior, and vocal usage. Results. NF1 patients obtained significantly higher VHI scores compared with healthy controls. Differences between the patient and control group were especially marked for daily functioning and affective responses. Men and women with NF1 had similar VHI scores, but total VHI score did increase with age. An apparent association between VHI, DSI, and severity of NF1 could not be demonstrated. Conclusion. It is likely that the elevated VHI scores observed in the patient group were not merely caused by the voice characteristics associated with NF1. A flow over from other psychophysical issues relating to the disease might have played a role.
Please use this url to cite or link to this publication:
author
organization
year
type
journalArticle (original)
publication status
published
subject
keyword
Voice Handicap Index (VHI), Voice disorders, Neurofibromatosis type 1, DISORDERS, IMPACT, PARKINSONS-DISEASE, HANDICAP INDEX, GENE, DYSPHONIA, MUTATIONS, IMPAIRMENT
journal title
JOURNAL OF VOICE
J. Voice
volume
26
issue
2
pages
e57 - e62
Web of Science type
Article
Web of Science id
000307782100004
JCR category
OTORHINOLARYNGOLOGY
JCR impact factor
1.55 (2012)
JCR rank
17/43 (2012)
JCR quartile
2 (2012)
ISSN
0892-1997
DOI
10.1016/j.jvoice.2010.11.001
language
English
UGent publication?
yes
classification
A1
copyright statement
I have transferred the copyright for this publication to the publisher
id
1230595
handle
http://hdl.handle.net/1854/LU-1230595
date created
2011-05-23 14:37:59
date last changed
2013-07-08 15:45:08
@article{1230595,
  abstract     = {Objective. Neurofibromatosis type 1 (NF1) is an autosomal dominant neurocutaneous disorder, which occurs in about one in 3000-4000 people. Its hallmark features include multiple cafe-au-lait spots and neurofibromas. Voice characteristics of NF1 patients have been documented using both subjective and objective evaluations. However, the relative impact of these voice characteristics on daily activities has, as far as we know, not been examined yet. 
Methods. Thirty adults with NF1 were asked to complete the Flemish Dutch version of the Voice Handicap Index (VHI). Additionally, the level of severity of NF1 was rated and Dysphonia Severity Indices (DSIs) were collected. The results of the patient group were compared with those of a healthy control group frequency matched for age, gender, smoking behavior, and vocal usage. 
Results. NF1 patients obtained significantly higher VHI scores compared with healthy controls. Differences between the patient and control group were especially marked for daily functioning and affective responses. Men and women with NF1 had similar VHI scores, but total VHI score did increase with age. An apparent association between VHI, DSI, and severity of NF1 could not be demonstrated. 
Conclusion. It is likely that the elevated VHI scores observed in the patient group were not merely caused by the voice characteristics associated with NF1. A flow over from other psychophysical issues relating to the disease might have played a role.},
  author       = {Cosyns, Marjan and Mortier, Geert and Janssens, Sandra and Van Borsel, John},
  issn         = {0892-1997},
  journal      = {JOURNAL OF VOICE},
  keyword      = {Voice Handicap Index (VHI),Voice disorders,Neurofibromatosis type 1,DISORDERS,IMPACT,PARKINSONS-DISEASE,HANDICAP INDEX,GENE,DYSPHONIA,MUTATIONS,IMPAIRMENT},
  language     = {eng},
  number       = {2},
  pages        = {e57--e62},
  title        = {Voice-related quality of life in adults with neurofibromatosis type 1},
  url          = {http://dx.doi.org/10.1016/j.jvoice.2010.11.001},
  volume       = {26},
  year         = {2012},
}

Chicago
Cosyns, Marjan, Geert Mortier, Sandra Janssens, and John Van Borsel. 2012. “Voice-related Quality of Life in Adults with Neurofibromatosis Type 1.” Journal of Voice 26 (2): e57–e62.
APA
Cosyns, Marjan, Mortier, G., Janssens, S., & Van Borsel, J. (2012). Voice-related quality of life in adults with neurofibromatosis type 1. JOURNAL OF VOICE, 26(2), e57–e62.
Vancouver
1.
Cosyns M, Mortier G, Janssens S, Van Borsel J. Voice-related quality of life in adults with neurofibromatosis type 1. JOURNAL OF VOICE. 2012;26(2):e57–e62.
MLA
Cosyns, Marjan, Geert Mortier, Sandra Janssens, et al. “Voice-related Quality of Life in Adults with Neurofibromatosis Type 1.” JOURNAL OF VOICE 26.2 (2012): e57–e62. Print.