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PMID: 27666129 Published · ppublish English Journal Article

Using a qualitative approach to conceptualize concerns of patients with neurofibromatosis type 1 associated plexiform neurofibromas (pNF) across the lifespan.

American journal of medical genetics. Part A ·Vol. 173 ·No. 1 ·2017-01-00 ·页码 79-87

Lai JS, Jensen SE, Patel ZS, Listernick R, Charrow J

Abstract

Neurofibromatosis Type 1 (NF1) plexiform neurofibromas (pNFs) are associated with a variety of symptoms and concerns that affect patients' quality of life (QOL), highlighting the value of incorporating the patients' perspective when evaluating treatment outcomes. To better conceptualize the experience of patients with pNFs, this qualitative study sought to identify the most important outcomes to assess from the perspective of patients, families, and clinicians. Clinicians, patients age 5 years old and above, and parents of patients aged 5-17 years participated in semi-structured interviews to elicit the pNF symptoms/concerns considered most important to assess. The data were analyzed using an iterative coding procedure and the frequency with which symptoms/concerns emerged was tabulated. Eight clinicians, 31 patients, and 17 parents of patients participated in semi-structured interviews. The most frequently reported concerns raised by patients across all age groups included pain, appearance/disfigurement, social activity/role participation, stigma, and anxiety. For parents, physical functioning was the primary concern, followed by pain, social activity/role participation, appearance/disfigurement, and social relationships. The resulting conceptual framework included five domains to represent the most important identified symptoms/concerns: pain, social functioning, physical function impact, stigma, and emotional distress. This conceptual framework describing the symptoms/concerns of patients with pNF can help investigators create a measurement system to improve assessment of aspects of QOL only patients can report on. It may also provide the ability to identify symptoms/concerns that might warrant referrals to various clinical disciplines. © 2016 Wiley Periodicals, Inc.

Keywords
lifespan neurofibromatosis type 1 patient-centered outcomes plexiform neurofibromas
MeSH 主题词
Adolescent Adult Aged Child Child, Preschool Female Humans Male Middle Aged Neurofibroma, Plexiform/complications,diagnosis,epidemiology Neurofibromatosis 1/complications,diagnosis,epidemiology Patient Outcome Assessment Phenotype Qualitative Research Quality of Life Self Report Socioeconomic Factors Surveys and Questionnaires Young Adult
作者与单位
共 5 位作者,点击展开单位 / ORCID
Lai Jin-Shei
Department of Medical Social Sciences, Northwestern University Feinberg School of Medicine, Chicago, Illinios. | Department of Pediatrics, Northwestern University Feinberg School of Medicine, Chicago, Illinios. | Ann & Robert H. Lurie, Children's Hospital of Chicago, Chicago, Illinios.
Jensen Sally E
Department of Medical Social Sciences, Northwestern University Feinberg School of Medicine, Chicago, Illinios. | Department of Surgery (Division of Organ Transplantation), Northwestern University Feinberg School of Medicine, Chicago, Illinios.
Patel Zabin S
Department of Medical Social Sciences, Northwestern University Feinberg School of Medicine, Chicago, Illinios.
Listernick Robert
Department of Pediatrics, Northwestern University Feinberg School of Medicine, Chicago, Illinios. | Ann & Robert H. Lurie, Children's Hospital of Chicago, Chicago, Illinios.
Charrow Joel
Department of Pediatrics, Northwestern University Feinberg School of Medicine, Chicago, Illinios. | Ann & Robert H. Lurie, Children's Hospital of Chicago, Chicago, Illinios.
Article Info
Journal
American journal of medical genetics. Part A
Abbr.
Am J Med Genet A
ISSN
1552-4833
Published
2017-01-00
电子出版
2016-00-26
页码
79-87
Language
English
Country/Region
United States
NLM ID
101235741
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