Primary Sjögren Syndrome (NeuroSenSS)

Primary Sjögren syndrome (pSS) is an inflammatory, autoimmune, multiorgan disease often involving the central and peripheral nervous systems. Fifteen to twenty percent of patients with the primary Sjögren's syndrome have neurological complications involving the peripheral nervous system. Although some patients have large fiber neuropathy, around forty percent of patients with Sjögren's syndrome experience neuropathic pain with normal electrodiagnostic studies. Although these patients may be diagnosed with fibromyalgia or depressive symptoms, some have been shown to have small fiber neuropathy (SFN). A recent study proved that more than 90% of pSS patients with such neuropathic pain have SFN {Fauchais, 2010 #188}.

The aim of this study will be to investigate the occurrence of small fiber neuropathy in patients with pSS and neuropathic pain with normal electromyographic studies and to determine the existence of a conjoint local inflammatory process mediated by cellular, cytokinic or auto-antibody response. Quantification of epidermal nerve fiber density after skin biopsy is a valuable tool to diagnose small fiber neuropathy and the method has been widely validated. A skin biopsy will be performed in patients and control and will allow quantification of small fiber density in skin sample along with measurement of sweat gland innervation. Moreover, pathophysiological studies will be carried on in order to evaluate the causal relationship between cellular and humoral inflammation and small fiberneuropathy.

Recent studies have pointed out the inconstant efficacy of both corticosteroid and immunosuppressive drugs in pSS-related SFN. Dissecting the molecular mechanisms of small fiber neuropathy in these patients may help designing new therapeutic strategies.

Study Overview

• Status: Completed
• Conditions: Primary Sjögren's Syndrome
• Intervention / Treatment: Other: skin biopsy

• Study Type: Observational
• Enrollment (Actual): 44

Contacts and Locations

Study Locations

• France
  • Albi, France, 81000
    • CH d'Albi
  • Bordeaux, France, 33000
    • CHU Bordeaux
  • Brive la Gaillarde, France, 19312
    • CH de Brive
  • Lille, France, 59037
    • CHU de LILLE
  • Limoges, France, 87 042
    • University Hospital, Limoges
  • Rodez, France, 12000
    • CH de Rodez
  • Toulouse, France, 31059
    • Chu Toulouse

Participation Criteria

Eligibility Criteria

• Ages Eligible for Study: 18 years and older (ADULT, OLDER_ADULT)
• Accepts Healthy Volunteers: Yes
• Genders Eligible for Study: All

• Sampling Method: Non-Probability Sample

Study Population

Sjögren's syndrome

Description

Inclusion Criteria:

• Patients with pSS according to revised European criteria and exhibiting neuropathic pain without any electromyographic abnormality will be included (Group I).
• Age of entry into the study ≥ 18 yrs (both groups).
• Affiliated or profit patient of a social security system (both groups).
• Informed consent signed up (both groups).

Exclusion Criteria:

• Chronic alcoholism
• Type I or type II diabetes
• Peripheral neuropathy unrelated to pSS complications
• Coagulation disease
• Previous allergy to xylocaine, lidocaine, prilocaine or ricin oil
• Porphyria, methemoglobinemia
• Patients under measure of maintenance of justice.
• Patients unable to understand or to participate to the study.
• Child and major patients making the object of a measure of lawful protection.
• Patients deprived of freedom.
• Pregnant, nursing women.
• Participation in biomedical research within one month prior to inclusion.

Study Plan

How is the study designed?

Number of groups / cohorts

3

Cohorts and Interventions

Group / Cohort	Intervention / Treatment
patients with Sjögren's syndrome; A skin biopsy will be performed in patients	Other: skin biopsy
control group; A skin biopsy will be performed in control group	Other: skin biopsy
non auto-immune small fiber neuropathies

What is the study measuring?

Primary Outcome Measures

Outcome Measure	Time Frame
To evaluate the cutaneous lymphocytic infiltration	1 year

Secondary Outcome Measures

Outcome Measure	Time Frame
Cutaneous infiltration T, B and Th17	1 year

Collaborators and Investigators

• Sponsor: University Hospital, Limoges
• Investigators: Principal Investigator: Anne-Laure FAUCHAIS, MD, University Hospital, Limoges

Study record dates

Study Major Dates

• Study Start (ACTUAL): September 1, 2012
• Primary Completion (ACTUAL): September 1, 2016
• Study Completion (ACTUAL): June 1, 2017

Study Registration Dates

• First Submitted: November 6, 2013
• First Submitted That Met QC Criteria: November 15, 2013
• First Posted (ESTIMATE): November 21, 2013

Study Record Updates

• Last Update Posted (ACTUAL): January 11, 2019
• Last Update Submitted That Met QC Criteria: January 10, 2019
• Last Verified: June 1, 2016

More Information

Terms related to this study

• Additional Relevant MeSH Terms: Pathologic Processes; Immune System Diseases; Autoimmune Diseases; Eye Diseases; Disease; Joint Diseases; Musculoskeletal Diseases; Rheumatic Diseases; Connective Tissue Diseases; Arthritis; Stomatognathic Diseases; Mouth Diseases; Lacrimal Apparatus Diseases; Arthritis, Rheumatoid; Xerostomia; Salivary Gland Diseases; Dry Eye Syndromes; Syndrome; Sjogren's Syndrome
• Other Study ID Numbers: I10 023