Highlights
- •Current outcomes do not capture the full range of small-fiber polyneuropathy (SFPN) symptoms.
- •We report the development and initial validation of a new survey for SFPN symptoms.
- •This survey is suitable for patients with ill-defined causes of SFPN.
- •It can support diagnosis and monitoring symptoms, for clinical and research use.
Abstract
Small-fiber polyneuropathy (SFPN) affects unmyelinated and thinly myelinated peripheral
axons. Several questionnaires have been developed to assess polyneuropathy from diabetes
or chemotherapy, but none for SFPN from other or unknown causes. A comprehensive survey
could help clinicians diagnose and assess treatment responses, define prevalence natural
history and cures, and identify research subjects. Thus, we developed the 1-page Small-Fiber
Symptom Survey, using input from patients and 21 medical/scientific experts. Participants
comprised consenting consecutive patients evaluated for SFPN at the Massachusetts
General Hospital plus normal control subjects. Participants SFPN status was stratified
on the basis of the results of their objective diagnostic tests (distal leg skin biopsy
and autonomic function testing). We measured internal consistency, test retest reliability,
convergent validity, and performed a receiver operating curve analysis. The 179 participants
averaged 46.6 ± 15.6 years old; they were 73.2% female and 92.2% Caucasian. Eighty-five
had confirmed SFPN, mostly idiopathic. Principal component analysis revealed 5 symptom
clusters. The questionnaire had good internal consistency (Cronbach α = .893), excellent
test retest reliability (r = .927, P < .001) and good to fair convergent validity. Participants with confirmed SFPN had
more severe symptoms than others (P = .009). The Small-Fiber Symptom Survey has satisfactory psychometric properties,
indicating potential future utility for surveying patient-reported symptoms of SFPN
regardless of its cause.
Perspective
This article reports the initial development and early psychometric validation of
a new patient-reported outcome measure intended to capture the wide range of multisystem
symptoms of SFPN. When further developed, it could potentially help clinicians diagnose
and monitor patients, and help advance research.
Key words
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Article info
Publication history
Published online: January 04, 2017
Accepted:
December 21,
2016
Received in revised form:
December 11,
2016
Received:
August 2,
2016
Footnotes
R.T. and M.L. contributed equally to this work.
This work was supported by the National Institutes of Health (R01-NS093653 and UL1 TR001102), the Lundbeck Foundation Scholarship in Neurology, and the U.S. Department of Defense (GW140169).
The authors have no conflicts of interest to declare.
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© 2017 by the American Pain Society
