RT Journal Article
SR Electronic
T1 Epidermal Neurite Density in Skin Biopsies from Patients with Juvenile Fibromyalgia
JF The Journal of Rheumatology
JO J Rheumatol
FD The Journal of Rheumatology
SP jrheum.200378
DO 10.3899/jrheum.200378
A1 Alexis Boneparth
A1 Shan Chen
A1 Daniel B.  Horton
A1 L. Nandini  Moorthy
A1 Ian Farquhar
A1 Heather M.  Downs
A1 Hang Lee
A1 Anne Louise  Oaklander
YR 2020
UL http://www.jrheum.org/content/early/2020/08/10/jrheum.200378.abstract
AB Objective Fibromyalgia is defined by idiopathic, chronic, widespread musculoskeletal pain. In adults with fibromyalgia, meta-analysis of lower-leg skin biopsy demonstrated 45% pooled prevalence of abnormally low epidermal neurite density (END). END <5th centile of the normal distribution is the consensus diagnostic threshold for small-fiber neuropathy. However, the clinical significance of END findings in fibromyalgia is unknown. The prevalence of small fiber pathology has not yet been studied in juvenile fibromyalgia. Methods We screened 21 patients aged 13-20y with fibromyalgia diagnosed by pediatric rheumatologists. Fifteen meeting the American College of Rheumatology criteria (modified for juvenile fibromyalgia) underwent lower-leg measurements of END and completed validated questionnaires assessing pain, functional disability, and dysautonomia symptoms. The primary outcome was proportion of fibromyalgia patients with END <5th centile of age/gender/race-based laboratory norms. Cases were systematically matched by ethnicity, race, sex, and age to a group of previously biopsied healthy adolescents with selection blinded to biopsy results. All 23 controls matching demographic criteria were included. Results Among biopsied JFM patients, 53% (8/15) had END <5th centile versus 4% (1/23) of healthy controls (p<0.001). Mean patient END was 273/mm2 skin surface (95% confidence interval: 198-389) versus 413 (95% CI: 359-467; p<0.001). As expected, fibromyalgia patients reported more functional disability, dysautonomia, and pain than healthy controls. Conclusion Abnormal END reduction is common in adolescents with fibromyalgia, with similar prevalence to adults with fibromyalgia. More studies are needed to fully characterize the significance of low END in fibromyalgia and to elucidate the clinical implications of these findings.