Epidermal Neurite Density in Skin Biopsies From Patients With Juvenile Fibromyalgia.
Epidermal Neurite Density in Skin Biopsies From Patients With Juvenile Fibromyalgia.
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DOI：
10.3899/jrheum.200378
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发表时间：
2021-04
期刊：
影响因子：
--
通讯作者：
Oaklander AL
中科院分区：
文献类型：
--
作者：
Boneparth A;Chen S;Horton DB;Moorthy LN;Farquhar I;Downs HM;Lee H;Oaklander AL
纤维肌痛是指特发性、慢性、广泛的肌肉骨骼疼痛。在患有纤维肌痛的成人中，小腿皮肤活检的荟萃分析表明，表皮神经突密度 (END) 异常低的汇总患病率为 45%。 END <正态分布的第 5 个百分位是小纤维神经病的共识诊断阈值。然而，END 发现在纤维肌痛中的临床意义尚不清楚。尚未研究青少年纤维肌痛中小纤维病理学的患病率。我们筛查了 21 名 13-20 岁、由儿科风湿病专家诊断患有纤维肌痛的患者。 15 名符合美国风湿病学会标准（针对青少年纤维肌痛进行修改）的患者接受了小腿 END 测量，并完成了评估疼痛、功能障碍和自主神经功能障碍症状的有效问卷。主要结局是纤维肌痛患者 END 低于年龄/性别/种族实验室标准第 5 个百分位的比例。根据种族、种族、性别和年龄，将病例与一组先前接受过活检的健康青少年进行系统匹配，并在对活检结果不知情的情况下进行选择。包括所有 23 个符合人口统计标准的对照。在进行活检的 JFM 患者中，53% (8/15) 的 END < 5th 百分位数相比，健康对照的这一比例为 4% (1/23) (p<0.001)。患者平均 END 为 273/mm2 皮肤表面（95% 置信区间：198-389），而患者 END 为 413（95% CI：359-467；p<0.001）。正如预期的那样，纤维肌痛患者比健康对照者报告了更多的功能障碍、自主神经功能障碍和疼痛。 END 异常减少在患有纤维肌痛的青少年中很常见，其患病率与患有纤维肌痛的成人相似。需要更多的研究来充分描述低 END 在纤维肌痛中的重要性，并阐明这些发现的临床意义。
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. 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. 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. 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.
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