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Anti-salivary gland protein 1 antibodies in two patients with Sjogren's syndrome: two case reports.

Abstract Current diagnostic criteria for Sjogren's syndrome developed by the American College of Rheumatology include the presence of antinuclear antibodies, rheumatoid factor, anti-Ro or anti-La autoantibodies. The purpose of this report is to describe two patients with biopsy-proven Sjogren's syndrome lacking these autoantibodies but identified by antibodies to salivary gland protein 1. Diagnosis was delayed until salivary gland tumors developed in these patients because of the lack of the classic autoantibodies. This report emphasizes the existence of patients with primary Sjogren's syndrome who lack autoantibodies anti-Ro or anti-La and may therefore be misdiagnosed. Antibodies to salivary gland protein 1 identify some of these patients.
PMID
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Authors

Mayor MeshTerms
Keywords
Journal Title journal of medical case reports
Publication Year Start




PMID- 24885364
OWN - NLM
STAT- MEDLINE
DA  - 20140604
DCOM- 20150117
LR  - 20150805
IS  - 1752-1947 (Electronic)
IS  - 1752-1947 (Linking)
VI  - 8
DP  - 2014 May 11
TI  - Anti-salivary gland protein 1 antibodies in two patients with Sjogren's syndrome:
      two case reports.
PG  - 145
LID - 10.1186/1752-1947-8-145 [doi]
AB  - INTRODUCTION: Current diagnostic criteria for Sjogren's syndrome developed by the
      American College of Rheumatology include the presence of antinuclear antibodies, 
      rheumatoid factor, anti-Ro or anti-La autoantibodies. The purpose of this report 
      is to describe two patients with biopsy-proven Sjogren's syndrome lacking these
      autoantibodies but identified by antibodies to salivary gland protein 1.
      Diagnosis was delayed until salivary gland tumors developed in these patients
      because of the lack of the classic autoantibodies. This report emphasizes the
      existence of patients with primary Sjogren's syndrome who lack autoantibodies
      anti-Ro or anti-La and may therefore be misdiagnosed. Antibodies to salivary
      gland protein 1 identify some of these patients. CASE PRESENTATION: Two patients 
      are described and were seen in the autoimmune disease clinics of the State
      University of New York (SUNY) at the Buffalo School of Medicine. In both
      patients, chronic dry mouth and dry eye had been dismissed as idiopathic because 
      test results for autoantibodies anti-Ro and anti-La were negative. Both patients 
      had swelling of major salivary glands that prompted biopsies. Biopsies of major
      salivary glands from both cases demonstrated salivary gland tumors and existence 
      of inflammation consistent with Sjogren's syndrome. Serologic testing revealed
      antibodies to salivary gland protein 1. CONCLUSIONS: Patients presenting with
      classic clinical symptoms of dry mouth and eyes do not always show the current
      serologic markers of Sjogren's syndrome, anti-Ro and anti-La. In these cases,
      investigation for antibodies to salivary gland protein 1 is of importance to make
      the diagnosis of Sjogren's syndrome. Early diagnosis of Sjogren's syndrome is
      necessary for improved management as well as for vigilance regarding potential
      complications, such as salivary gland tumors as were seen in the described cases.
FAU - Vishwanath, Sahana
AU  - Vishwanath S
FAU - Shen, Long
AU  - Shen L
FAU - Suresh, Lakshmanan
AU  - Suresh L
FAU - Ambrus, Julian L Jr
AU  - Ambrus JL Jr
AD  - Department of Medicine, SUNY at Buffalo School of Medicine, 100 High Street,
      Buffalo, NY 14203, USA. [email protected]
LA  - eng
PT  - Case Reports
PT  - Journal Article
PT  - Research Support, Non-U.S. Gov't
DEP - 20140511
PL  - England
TA  - J Med Case Rep
JT  - Journal of medical case reports
JID - 101293382
RN  - 0 (Autoantibodies)
RN  - 0 (Salivary Proteins and Peptides)
SB  - IM
MH  - Adenoma, Pleomorphic/complications/*pathology
MH  - Autoantibodies/*immunology
MH  - Female
MH  - Humans
MH  - Lymphoma, B-Cell, Marginal Zone/complications/*pathology
MH  - Middle Aged
MH  - Parotid Neoplasms/complications/*pathology
MH  - Salivary Proteins and Peptides/*immunology
MH  - Sjogren's Syndrome/complications/*immunology
PMC - PMC4031912
OID - NLM: PMC4031912
EDAT- 2014/06/03 06:00
MHDA- 2015/01/18 06:00
CRDT- 2014/06/03 06:00
PHST- 2013/11/22 [received]
PHST- 2014/02/18 [accepted]
AID - 1752-1947-8-145 [pii]
AID - 10.1186/1752-1947-8-145 [doi]
PST - epublish
SO  - J Med Case Rep. 2014 May 11;8:145. doi: 10.1186/1752-1947-8-145.

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