Reduction of Intraepidermal Nerve Fiber Density (IENFD) in the skin biopsies of patients with fibromyalgia: A controlled study

Kosmidis, Michalis L.; Κουτσογεωργοπούλου, Λουκία; Alexopoulos, Harry; Mamali, Ioanna; Vlachoyiannopoulos, P.; Voulgarelis, Michalis; Moutsopoulos, Haralampos Μ.; Tzioufas, Athanasios G.; Dalakas, Marinos C.

doi:10.1016/j.jns.2014.09.035

Cited by 80 publications

(62 citation statements)

References 20 publications

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“…Some studies have found that symmetric axonal sensory or sensorimotor polyneuropathy is the most common form of neuropathy (34.0 %), followed by cranial neuropathies (19.5 %), multiple mononeuropathies (8.5 %), ganglionopathies (<5 %), and CIDP (1.3 %) [20,82,90]. In our large series of patients, as well as in other studies, small fiber sensory neuropathy was the most common neuropathy, while demyelinating neuropathies were the rarest [20,41,90,91].…”

Section: Sjögren's Syndromesupporting

confidence: 54%

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Immunotherapies for Neurological Manifestations in the Context of Systemic Autoimmunity

et al. 2016

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Neurological involvement is relatively common in the majority of systemic autoimmune diseases and may lead to severe morbidity and mortality, if not promptly treated. Treatment options vary greatly, depending on the underlying systemic pathophysiology and the associated neurological symptoms. Selecting the appropriate therapeutic scheme is further complicated by the lack of definite therapeutic guidelines, the necessity to differentiate primary neurological syndromes from those related to the underlying systemic disease, and to sort out adverse neurological manifestations caused by immunosuppressants or the biological agents used to treat the primary disease. Immunotherapy is a sine qua non for treating most, if not all, neurological conditions presenting in the context of systemic autoimmunity. Specific agents include classical immune modulators such as corticosteroids, cyclophosphamide, intravenous immunoglobulin, and plasma exchange, as well as numerous biological therapies, for example antitumor necrosis factor agents and monoclonal antibodies that target various immune pathways such as B cells, cytokines, and co-stimulatory molecules. However, experience regarding the use of these agents in neurological complications of systemic diseases is mainly empirical or based on small uncontrolled studies and case series. The aim of this review is to present the state-of-the-art therapies applied in various neurological manifestations encountered in the context of systemic autoimmune diseases; evaluate all treatment options on the basis of existing guidelines; and compliment these data with our personal experience derived from a large number of patients.

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Section: Sjögren's Syndromesupporting

confidence: 54%

“…In such cases, a skin biopsy assessing epidermal small fiber density could be diagnostic [22,41]. Whether in some of these patients the small fiber neuropathy has an autoimmune etiology remains to be determined.…”

Section: Peripheral Neuropathiesmentioning

confidence: 99%

Immunotherapies for Neurological Manifestations in the Context of Systemic Autoimmunity

et al. 2016

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“…Fibromyalgia has neuropathic pain features: it is a stimulus-independent pain state, accompanied by allodynia and paresthesias (2). The notion of fibromyalgia as a neuropathic pain syndrome has been recently reinforced by several controlled studies describing small fiber neuropathy in patients with fibromyalgia (3)(4)(5)(6)(7)(8). Small fiber neuropathy is a disorder of the peripheral nerves that mainly affects small sensory fibers and sympathetic fibers resulting in pain, paresthesias and autonomic dysfunction.…”

Section: Discussionmentioning

confidence: 99%

Small fiber neuropathy in women with fibromyalgia. An in vivo assessment using corneal confocal bio-microscopy

Ramı́rez

Martínez-Martínez

Hernández-Quíntela

et al. 2015

Seminars in Arthritis and Rheumatism

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“…Initial pathology skin biopsy studies identifying loss of cutaneous innervation were followed by electrophysiologic evidence that C-fibers fire spontaneously and excessively in patients with fibromyalgia as in positive-control patients with SFPN but not normal controls [74,[92][93][94]. Subsequent studies have so far all been supportive (e.g., [95]), so biological overlap between fibromyalgia and High-dose corticosteroids resulted in immediate improvement in all signs and symptoms, including pain. One month later during prednisone taper, the redness and edema were resolved, the ulcers were healing, and the left first dorsal interosseous muscle had neurogenic atrophy, consistent with bystander damage to myelinated motor fibers.…”

Section: Small-fiber Polyneuropathymentioning

confidence: 99%

“…A proportion of patients with fibromyalgia have evidence for inflammation/dysimmune mechanisms by virtue of also having systemic autoimmune diseases, dysimmune profiles in blood or skin, and/or improvement in fibromyalgia symptoms after immunotherapy [93]. Comparing patients with fibromyalgia with those without systemic autoimmune rheumatologic disorders revealed that 44 % of those with systemic autoimmunity versus 27 % of those without it had skin biopsies consistent with SFPN [95]. Thus, it may be reasonable to more seriously consider immunotherapy for the subset of patients with fibromyalgia with confirmed SFPN, evidence of immune mediation, exclusion of other causes, and inadequate response to symptom-based treatments.…”

Section: Small-fiber Polyneuropathymentioning

confidence: 99%

Immunotherapy Prospects for Painful Small-fiber Sensory Neuropathies and Ganglionopathies

Oaklander

2016

Neurotherapeutics

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The best-known peripheral neuropathies are those affecting the large, myelinated motor and sensory fibers. These have well-established immunological causes and therapies. Far less is known about the somatic and autonomic "small fibers"; the unmyelinated C-fibers, thinly myelinated A-deltas, and postganglionic sympathetics. The small fibers sense pain and itch, innervate internal organs and tissues, and modulate the inflammatory and immune responses. Symptoms of small-fiber neuropathy include chronic pain and itch, sensory impairment, edema, and skin color, temperature, and sweating changes. Small-fiber polyneuropathy (SFPN) also causes cardiovascular, gastrointestinal, and urological symptoms, the neurologic origin of which often remains unrecognized. Routine electrodiagnostic study does not detect SFPN, so skin biopsies immunolabeled to reveal axons are recommended for diagnostic confirmation. Preliminary evidence suggests that dysimmunity causes some cases of small-fiber neuropathy. Several autoimmune diseases, including Sjögren and celiac, are associated with painful small-fiber ganglionopathy and distal axonopathy, and some patients with "idiopathic" SFPN have evidence of organ-specific dysimmunity, including serological markers. Dysimmune SFPN first came into focus in children and teenagers as they lack other risk factors, for example diabetes or toxic exposures. In them, the rudimentary evidence suggests humoral rather than cellular mechanisms and complement consumption. Preliminary evidence supports efficacy of corticosteroids and immunoglobulins in carefully selected children and adult patients. This paper reviews the evidence of immune causality and the limited data regarding immunotherapy for small-fiberpredominant ganglionitis, regional neuropathy (complex regional pain syndrome), and distal SFPN. These demonstrate the need to develop case definitions and outcome metrics to improve diagnosis, enable prospective trials, and dissect the mechanisms of small-fiber neuropathy.

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Reduction of Intraepidermal Nerve Fiber Density (IENFD) in the skin biopsies of patients with fibromyalgia: A controlled study

Cited by 80 publications

References 20 publications

Immunotherapies for Neurological Manifestations in the Context of Systemic Autoimmunity

Immunotherapies for Neurological Manifestations in the Context of Systemic Autoimmunity

Small fiber neuropathy in women with fibromyalgia. An in vivo assessment using corneal confocal bio-microscopy

Immunotherapy Prospects for Painful Small-fiber Sensory Neuropathies and Ganglionopathies

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