Case of primary Sjogren's syndrome preceded by dystonia.

There are only six cases in literature that describe development of dystonia with Sjogren's syndrome (SS). We describe a case of a 43-year-old woman who presented with symptoms including movement disorder, sensory neurogenic bladder, sensory loss and neuropathic pain, migraine like headaches, musculoskeletal pain, Raynaud's phenomenon and dysautonomia. Symptoms started in 2000, with weakness that progressed to dystonia in 2003. Diagnostic work-up was inconclusive with negative inflammatory serologies, cerebrospinal fluid and MRI for many years. After patient developed sicca syndrome with dry eyes and mouth in 2009, her rheumatoid factor titre was elevated (550 IU/mL), erythrocyte sedimentation rate, anti-Sjogrens syndrome-related antigen A (anti-Ro/SSA) and anti-SSB/La: anti-Sjogrens syndrome-related antigen B (anti-La/SSB) became positive. Lip biopsy confirmed diagnosis of SS. She was diagnosed with primary SS with neurological involvement. Her symptoms responded well to intravenous methylprednisolone. Symptoms stabilised with trials of immune-suppressive therapy. This is a case that demonstrates the delay of diagnosing SS with preceding unique neurological association.

Antikeratin antibodies in serum and synovial fluid show specificity for rheumatoid arthritis in a study of connective tissue diseases.

Tests for antikeratin antibodies (AKA) were performed on 2152 disease-associated and control sera by indirect immunofluorescence (IF) on rat oesophagus substrate. The incidence of AKA was significantly raised in rheumatoid arthritis (37%) in comparison with systemic sclerosis (8%), psoriasis (7%), ankylosing spondylitis (6%), systemic lupus erythematosus (3%), and normal controls (2%). AKA were detected in synovial fluid obtained from patients with rheumatoid arthritis (RA) (48%) but not from patients with other conditions. Further experiments on AKA-positive sera showed reactivity with stratum corneum of rabbit prepuce and lips. A specific rabbit antihuman keratin antiserum was shown, by IF and inhibition studies, to have a different specificity from that of spontaneous human AKA. AKA were associated with the presence of subcutaneous nodules in RA (p = 0.05), but not with Raynaud's phenomenon, Sjögren's syndrome, or HLA-DR4 positivity. Rheumatoid factor (RF) was not associated with AKA either in RA or in RF-positive disease controls.

Detection of immune complexes in systemic sclerosis and Raynaud's phenomenon.

Phagocytosed immune complexes in polymorphonuclear leukocytes of nineteen patients with Raynaud's phenomenon were detected by scoring the cells for IgG and C3 inclusions. Parallel estimations of circulating immune complexes were made by liquid phase C1q binding and polyethylene glycol (PEG) precipitation methods. The results were compared with the degree of cold stress-induced vasospasm measured by quantified infra-red thermography. Thirteen of nineteen patients with Raynaud's phenomenon compared with only one of seventeen normal controls showed significantly elevated levels of phagocytosed IgG. The correlation of IgG and C3 scores for individual patients was high and the IgG and C3 scores correlated well with the thermographic assessment of the severity of the Raynaud's phenomenon. Two of the patients had elevated levels of circulating immune complexes detectable by raised levels of C1q binding, four patients had raised IgG and three patients raised IgM complexes detectable by PEG precipitation. None of the controls had elevated levels of circulating immune complexes detectable by either method. There was no correlation between the levels of circulating and phagocytosed immune complexes.

[Liver enzyme function and humoral immunity in workers exposed to vinyl chloride]. / Izuchavane enzimnata funktsiia na cherniia drob i khumoralniia imunitet na rabotnitsi, eksponirani na vinilkhlorid.

The work aims at studying the enzyme function of the liver, as well as the humoral immunity of workers engaged in the production of vinyl chloride and polyvinylchloride with proved hepatotropic and Raynaud syndromes. About 191 workers /experimental group/ from the synthesis and polymerization of vinyl chloride and 2 control groups--34 persons from Varna and 54 from G. Traikov were examined. The following biochemical and immunological studies were performed: GOT, GPT, GGTP, alkaline phosphatase, IGG, circulating immune complexes, titer of the complement, cryoglobulins in the serum and identification of the sediments by immunoelectrophoresis. It was established that the immune system was included in the early phases of toxic effect. Disturbances in the enzyme function and humoral immunity were registered both in workers with hepatotropic and Raynaud syndromes.