| Primary Sjögren syndrome in a 2-year-old patient: role of the dentist in diagnosis and dental management with a 6-year follow-up. | |
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MedLine Citation:
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PMID: 21623979 Owner: NLM Status: Publisher |
Abstract/OtherAbstract:
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International Journal of Paediatric Dentistry 2011 Background. Primary Sjögren syndrome is a rare autoimmune disease, especially in children, mainly affecting girls (77%), and usually diagnosed around 10 years of age. Diagnosis during childhood is difficult, especially because of the diversity of the clinical presentation and difficulty obtaining reliable history data, accounting for a higher frequency of underdiagnosed cases. Differential conditions should be considered, especially the ones that promote xerostomia, such as diabetes, ectodermal dysplasia, rheumatoid arthritis, scleroderma, systemic lupus erythematosus, sarcoidosis, lymphoma, HIV and HTLV infection. Conditions associated with parotid enlargement should also be excluded, including juvenile recurrent parotitis (JRP), sialadenosis, sarcoidosis, lymphoma, infectious parotitis caused by streptococcal and staphylococcal infections, viral infections (paramyxovirus, Epstein-Barr virus, cytomegalovirus, and parvovirus), and diffuse infiltrative lymphocytosis syndrome (associated with HIV infection), and rare congenital conditions, such as polycystic parotid disease. Case report. A paediatric female patient was referred to our clinic for dental treatment complaining about dry mouth, oral discomfort, and dysphagia. The patient presented five of the required criteria to establish the diagnosis of pSS, including ocular symptoms, oral symptoms, evidence of keratoconjunctivitis sicca, focal sialadenitis confirmed by minor salivary gland biopsy, and evidence of major salivary gland involvement. Our patient did not have positive SS-A and SS-B autoantibodies. According to the literature, about 29% of individuals with pSS can present seronegativity for SS-A (anti-Ro) antibodies and about 33% can present seronegativity for SS-B (anti-La) antibodies. Conclusion. To the best of our knowledge, this is the youngest patient reported in the scientific English literature with pSS. Primary Sjögren syndrome has a wide clinical and immunologic spectrum and may progress with increased morbidity. Clinicians must be aware of the development of pSS in such an early age and exclude all possible differential findings to provide early diagnosis and treatment. |
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Authors:
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Marcio Augusto De Oliveira; Nathalie Pepe Medeiros De Rezende; Célia Márcia Fernandes Maia; Marina Gallottini |
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Publication Detail:
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Type: JOURNAL ARTICLE Date: 2011-5-31 |
Journal Detail:
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Title: International journal of paediatric dentistry / the British Paedodontic Society [and] the International Association of Dentistry for Children Volume: - ISSN: 1365-263X ISO Abbreviation: - Publication Date: 2011 May |
Date Detail:
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Created Date: 2011-5-31 Completed Date: - Revised Date: - |
Medline Journal Info:
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Nlm Unique ID: 9107511 Medline TA: Int J Paediatr Dent Country: - |
Other Details:
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Languages: ENG Pagination: - Citation Subset: - |
Copyright Information:
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© 2011 The Authors. International Journal of Paediatric Dentistry © 2011 BSPD, IAPD and Blackwell Publishing Ltd. |
Affiliation:
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Special Care Dentistry Center, Department of Oral Pathology, School of Dentistry, University of São Paulo, São Paulo, SP, Brazil Private Practice Dentist, Montes Claros, MG, Brazil. |
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From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine
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