ArticlePediatrics2013
Evidence of small-fiber polyneuropathy in unexplained, juvenile-onset, widespread pain syndromes.
Article in Pediatrics, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 33 papers, 1 of them a synthesis that pooled it.
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Who cites it
33 citing papers in PubMed, 1 synthesis or guideline pooled it, 98 citations in OpenAlex.
- Epidermal Neurite Density in Skin Biopsies From Patients With Juvenile Fibromyalgia.The Journal of rheumatology · 2021Pooled it
- Article
- Defining the Chronic Complexities of hEDS and HSD: A Global Survey of Diagnostic Challenges, Life-Long Comorbidities, and Unmet Needs.Journal of clinical medicine · 2025Article
- Article
- Relapsing-Remitting Immunotherapy Responsive Small-Fiber Neuropathy: Longitudinal Tracking Through 10 Years Including Pregnancies.Neurology(R) neuroimmunology & neuroinflammation · 2024Article
- Correlation Between Orofacial Pain and Sensory and Autonomic Neuropathies.Journal of pain research · 2024Article
- Advances in diagnosis and management of distal sensory polyneuropathies.Journal of neurology, neurosurgery, and psychiatry · 2023Review
- A Review of the Therapeutic Targeting of SCN9A and Nav1.7 for Pain Relief in Current Human Clinical Trials.Journal of pain research · 2023Review
- Small fiber polyneuropathy: A new therapeutic target in patients with interstitial cystitis/bladder pain syndrome?Frontiers in urology · 2023Review
- What is the Next Step after an Electrodiagnostic Study in Children with Polyneuropathies? Rationale for Laboratory and Other Diagnostic Tests.Iranian journal of child neurology · 2023Review
- The puzzle of fibromyalgia between central sensitization syndrome and small fiber neuropathy: a narrative review on neurophysiological and morphological evidence.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2022Review
- Implications of Nerve Fiber Density on the Diagnosis and Treatment of Juvenile Fibromyalgia.Journal of pain research · 2022Review
- Cutaneous manifestations of orthostatic intolerance syndromes.International journal of women's dermatology · 2021Article
- Article
- Antiplexin D1 Antibodies Relate to Small Fiber Neuropathy and Induce Neuropathic Pain in Animals.Neurology(R) neuroimmunology & neuroinflammation · 2021Article
- Long-term efficacy of immunoglobulins in small fiber neuropathy related to Sjögren's syndrome.Journal of neurology · 2020Article
- Acute monophasic erythromelalgia pain in five children diagnosed as small-fiber neuropathy.European journal of paediatric neurology : EJPN : official journal of the European Paediatric Neurology Society · 2020Article
- Association of small-fiber polyneuropathy with three previously unassociated rare missense SCN9A variants.Canadian journal of pain = Revue canadienne de la douleur · 2020Article
- Current Diagnosis and Treatment of Painful Small Fiber Neuropathy.Current neurology and neuroscience reports · 2019Review
- Scientific Advances in and Clinical Approaches to Small-Fiber Polyneuropathy: A Review.JAMA neurology · 2019Article
Corrections and comments
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Authors and funding
2 authors at 2 institutions in 1 country.
Funding
Abstract
objectiveWe tested the hypothesis that acquired small-fiber polyneuropathy (SFPN), previously uncharacterized in children, contributes to unexplained pediatric widespread pain syndromes.
methodsForty-one consecutive patients evaluated for unexplained widespread pain beginning before age 21 had medical records comprehensively analyzed regarding objective diagnostic testing for SFPN (neurodiagnostic skin biopsy, nerve biopsy, and autonomic function testing), plus histories, symptoms, signs, other tests, and treatments. Healthy, demographically matched volunteers provided normal controls for SFPN tests.
resultsAge at illness onset averaged 12.3 ± 5.7 years; 73% among this poly-ethnic sample were female (P = .001). Sixty-eight percent were chronically disabled, and 68% had hospitalizations. Objective testing diagnosed definite SFPN in 59%, probable SFPN in 17%, and possible SFPN in 22%. Only 1 of 41 had entirely normal SFPN test results. Ninety-eight percent of patients had other somatic complaints consistent with SFPN dysautonomia (90% cardiovascular, 82% gastrointestinal, and 34% urologic), 83% reported chronic fatigue, and 63% had chronic headache. Neurologic examinations identified reduced sensation in 68% and vasomotor abnormalities in 55%, including 23% with erythromelalgia. Exhaustive investigations for SFPN causality identified only history of autoimmune illnesses in 33% and serologic markers of disordered immunity in 89%. Treatment with corticosteroids and/or intravenous immune globulin objectively and subjectively benefited 80% of patients (12/15).
conclusionsMore than half among a large series of patients with childhood-onset, unexplained chronic widespread pain met rigorous, multitest, diagnostic criteria for SFPN, which extends the age range of acquired SFPN into early childhood. Some cases appeared immune-mediated and improved with immunomodulatory therapies.
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Registered trials
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