title: Primary Sjögren's syndrome accompanied by pleural effusion: a case report and literature review.
abstract: Sjögren's syndrome (SS) is a systemic autoimmune disease characterized by the infiltration of lymphocytes in exocrine glands, specifically the salivary and lacrimal glands, resulting in the typical symptoms of xerophthalmia and xerostomia. SS may be accompanied by pleural effusion when the lung is involved, but this occurrence has been reported in only 10 cases in the literature. We report the case of a 42 year-old woman with severe bilateral pleural effusion for eight years. Primary Sjögren's Syndrome was finally diagnosed based on the presence of xerophthalmia and xerostomia, biopsy of the minor salivary glands, and positive anti-SS-A antibody in the serum and pleural effusion. Biopsy of the parietal pleura through video-assisted thoracoscopy revealed infiltration of lymphocytes. The patient had a long history of pleural effusion without clear etiology. Malignant disease was first suspected because of abnormal density lesion on the left lung and malignant cells found on cytology, but PET-CT revealed no malignant lesion. Examinations did not support infection, malignant tumor, pulmonary sarcoidosis, or other connective tissue diseases. This data could be useful for the future study of pleural effusion in SS.
authors.affiliation: Department of Respiratology, Qilu Hospital Shandong University Jinan 250012, China.
authors.forename: Dedong
authors.initials: D
authors.lastname: Ma
authors.affiliation: Department of Anaesthesiology, Affiliated Hospital of Shandong University of Traditional Chinese Medicine Jinan 250011, China.
authors.forename: Hongxiu
authors.initials: H
authors.lastname: Lu
authors.affiliation: Department of Respiratology, Qilu Hospital Shandong University Jinan 250012, China.
authors.forename: Yiqing
authors.initials: Y
authors.lastname: Qu
authors.affiliation: Department of Respiratology, Qilu Hospital Shandong University Jinan 250012, China.
authors.forename: Shanshan
authors.initials: S
authors.lastname: Wang
authors.affiliation: Department of ICU, Ningbo Peoples Hospital Ningbo 315800, China.
authors.forename: Yangyang
authors.initials: Y
authors.lastname: Ying
authors.affiliation: Department of Respiratology, Qilu Hospital Shandong University Jinan 250012, China.
authors.forename: Wei
authors.initials: W
authors.lastname: Xiao
mesh_terms: D000328:Adult; D000974:Antibodies, Antinuclear / Q000097:blood; D015415:Biomarkers / Q000097:blood; D001706:Biopsy; D057210:Delayed Diagnosis; D003937:Diagnosis, Differential; D005260:Female; D006801:Humans; D010996:Pleural Effusion / Q000175:diagnosis / Q000188:drug therapy / Q000209:etiology* / Q000276:immunology; D011237:Predictive Value of Tests; D012859:Sjogren's Syndrome / Q000150:complications* / Q000175:diagnosis / Q000188:drug therapy / Q000276:immunology; D013997:Time Factors; D014057:Tomography, X-Ray Computed; D014985:Xerophthalmia / Q000209:etiology; D014987:Xerostomia / Q000209:etiology
chemical_list: D000974:Antibodies, Antinuclear; D015415:Biomarkers; C035356:SS-A antibodies
keywords: Primary Sjögren’s syndrome; anti-SSA antibody; pleural effusion
references.citation: BMC Med. 2013;11:93
references.pmid: 23556533
references.citation: Mod Rheumatol. 2015 Jul 20;:1-4
references.pmid: 26052802
references.citation: Am Fam Physician. 2009 Mar 15;79(6):465-70
references.pmid: 19323359
references.citation: Respirology. 2008 Jan;13(1):155-8
references.pmid: 18197929
references.citation: Ann Rheum Dis. 2002 Jun;61(6):554-8
references.pmid: 12006334
references.citation: Curr Allergy Asthma Rep. 2013 Aug;13(4):354-60
references.pmid: 23797265
references.citation: Case Rep Rheumatol. 2012;2012:640353
references.pmid: 23198246
references.citation: Nihon Kokyuki Gakkai Zasshi. 2000 Aug;38(8):628-31
references.pmid: 11061090
references.citation: Nihon Kyobu Shikkan Gakkai Zasshi. 1995 Nov;33(11):1325-9
references.pmid: 8583730
references.citation: Intern Med. 1995 Aug;34(8):811-4
references.pmid: 8563128
references.citation: Respiration. 2008;75(4):361-71
references.pmid: 18477860
references.citation: Intern Med. 2008;47(23):2049-52
references.pmid: 19043259
references.citation: Chest. 1989 Dec;96(6):1440-1
references.pmid: 2582863
references.citation: Clin Exp Rheumatol. 1997 Mar-Apr;15(2):193-6
references.pmid: 9196874
references.citation: BMJ. 2012;344:e3821
references.pmid: 22700787
references.citation: Br J Rheumatol. 1996 Jan;35(1):72-5
references.pmid: 8624627
references.citation: Intern Med. 2000 Nov;39(11):979-84
references.pmid: 11065256
references.citation: Chin Med J (Engl). 2010 Nov;123(22):3252-7
references.pmid: 21163125
references.citation: Nat Rev Rheumatol. 2012 Jul;8(7):399-411
references.pmid: 22549247