Long-term efficacy of immunoglobulins in small fiber neuropathy related to Sjögren’s syndrome

Sala, Taylor Pindi; Villedieu, Mona; Damian, Laura; Crave, Jean-Charles; Pautot, Vivien; Stojanovich, Ljudmila; Tervaert, Jan Willem Cohen; Chérìn, P.; Bélizna, C.

doi:10.1007/s00415-020-10033-z

Cited by 21 publications

(26 citation statements)

References 46 publications

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“…Genetic SFN is because of mutations in a number of genes, such as GLA (Fabry disease), TTR (transthyretin-related amyloidosis), or SNCA (alpha-synucleinopathy) and many others. Secondary causes of SFN prevail and include, for example, diabetes, renal failure, thyroid dysfunction, hypovitaminoses, acute infections (SARS-CoV-2,[ 8 ] borreliosis[ 9 ]), vaccinations (rabies, varicella, human papillomavirus, lyme, SARS-CoV-2),[ 8 10 ] auto-immune disease,[ 11 12 ] pure autonomic failure because of alpha-synuclein deposition,[ 13 ] sarcoidosis,[ 14 ] Sjögren syndrome,[ 15 ] Parkinson’s disease,[ 16 ] and many others. [ 17 ] SFN may go along with or without affection of large motor or sensory fibers.…”

Section: Discussionmentioning

confidence: 99%

Small fiber neuropathy as a complication of SARS-CoV-2 vaccinations

Finsterer

2022

Journal of Family Medicine and Primary Care

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Generally, severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) vaccinations are not free of side effects. A rarely reported adverse reaction to SARS-CoV-2 vaccinations is small fiber neuropathy (SFN). Here, we present three patients with SFN after the second dose of messenger ribonucleic acid-based SARS-CoV-2 vaccines. Data for this study were collected via the self-made platform “Pubbly” for reporting side effects of SARS-CoV-2 vaccinations. Three patients with post-SARS-CoV-2 vaccination SFN were identified: a 40 yo Caucasian female (patient 1), a 52 yo Caucasian female (patient 2), and a 32 yo Caucasian female (patient 3). Patient 1 complained about fatigue, dizziness, flushing, palpitations, diarrhea, muscle weakness, and gait disturbance 10 days after the second Pfizer jab. Patient 2 reported dizziness, balance problems, brain fog, palpitations, dysphagia, and sleep problems. Patient 3 complained about profound fatigue, brain fog, vertigo, pre-syncopal sensations, hair loss, chest pain, dyspnea, palpitations, paresthesias, irregular menstrual cycles, muscle weakness, and hives 1 day after the second Moderna dose. All three patients underwent skin biopsy upon which SFN was diagnosed. Patient 1 profited from immunoglobulins, but patient 2 did not require any treatment. Symptoms in patient 3 resolved upon symptomatic treatment. Despite treatment, patient 1 did not completely recover. SFN can be a rare side effect of SARS-CoV-2 vaccinations. Post-SARS-CoV-2 vaccination SFN can be mild or severe and may or may not require treatment. Post-SARS-CoV-2 vaccination SFN is most likely immune-mediated as it responds to intravenous immunoglobulins.

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Section: Discussionmentioning

confidence: 99%

Small fiber neuropathy as a complication of SARS-CoV-2 vaccinations

Finsterer

2022

Journal of Family Medicine and Primary Care

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“…Successful trials with cibinetide (ARA 290) were reported in sarcoidosis patients with SFN of distal or unspecified phenotype 271,272 . The efficacy of IVIg was confirmed in larger studies of patients with SFN associated with Sjögren syndrome, 273 and with various autoimmune conditions including several patients with apparently small‐fiber‐restricted autoimmunity 274 . However, these studies did not differentiate between distal SFN and NLD‐SFN, a potentially important point, as it could be expected that therapeutic response differs according to the site of damage.…”

Section: Treatmentmentioning

confidence: 97%

Non‐length‐dependent small fiber neuropathy: Not a matter of stockings and gloves

et al. 2021

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The clinical spectrum of small fiber neuropathy (SFN) encompasses manifestations related to the involvement of thinly myelinated A‐delta and unmyelinated C fibers, including not only the classical distal phenotype, but also a non–length‐dependent (NLD) presentation that can be patchy, asymmetrical, upper limb‐predominant, or diffuse. This narrative review is focused on NLD‐SFN. The diagnosis of NLD‐SFN can be problematic, due to its varied and often atypical presentation, and diagnostic criteria developed for distal SFN are not suitable for NLD‐SFN. The topographic pattern of NLD‐SFN is likely related to ganglionopathy restricted to the small neurons of dorsal root ganglia. It is often associated with systemic diseases, but about half the time is idiopathic. In comparison with distal SFN, immune‐mediated diseases are more common than dysmetabolic conditions. Treatment is usually based on the management of neuropathic pain. Disease‐modifying therapy, including immunotherapy, may be effective in patients with identified causes. Future research on NLD‐SFN is expected to further clarify the interconnected aspects of phenotypic characterization, diagnostic criteria, and pathophysiology.

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“…The study selection process is illustrated in the PRISMA chart (Figure 1). All of the 49 studies that met our inclusion criteria [19–67] were of an observational nature. The majority of the papers were published by research teams that included both rheumatologists/internists and neurologists ( n = 31) followed by research teams that included either only rheumatologists/internists ( n = 12) or only neurologists ( n = 6).…”

Section: Resultsmentioning

confidence: 99%