Juvenile Sjogren's Syndrome: Clinical Characteristics With Focus on Salivary Gland Ultrasonography

Carregando...
Imagem de Miniatura
Citações na Scopus
33
Tipo de produção
article
Data de publicação
2020
Título da Revista
ISSN da Revista
Título do Volume
Editora
WILEY
Autores
HAMMENFORS, Daniel S.
VALIM, Valeria
BICA, Blanca E. R.
LILLEBY, Vibke
NIETO-GONZALEZ, Juan Carlos
MOSSEL, Esther
COELHO, Aline
Citação
ARTHRITIS CARE & RESEARCH, v.72, n.1, p.78-87, 2020
Projetos de Pesquisa
Unidades Organizacionais
Fascículo
Resumo
Objective Juvenile Sjogren's syndrome (SS) is a rare, poorly defined, and possibly underdiagnosed condition affecting children and adolescents. The aim of this study was to characterize symptoms and clinical findings of juvenile SS and to explore the clinical application of major salivary gland ultrasonography (SGUS) in patients with juvenile SS. Methods A cross-sectional multicenter study recruited patients with disease onset until age 18 years (n = 67). Disease characteristics were recorded, and unstimulated whole sialometry and SGUS examination of the parotid and submandibular salivary glands were performed. Results The female:male ratio was 58:9. The mean age at first symptom was 10.2 years and 12.1 years at diagnosis. Ocular and oral symptoms were noted in 42 of 67 patients (63%) and 53 of 66 patients (80%), respectively. The American-European Consensus Group or American College of Rheumatology/European League Against Rheumatism classification criteria for primary SS were fulfilled by 42 of 67 patients (63%). Pathologic SGUS findings were observed in 41 of 67 patients (61%); 26 of 41 SGUS+ patients (63%) fulfilled primary SS criteria. Salivary gland enlargements/parotitis were noted in 37 of 58 patients and were nonsignificantly associated with SGUS+ status (P = 0.066). The mean levels of saliva were 5.6 ml/15 minutes in SGUS- patients compared to 3.3 ml/15 minutes in the SGUS+ patients (P = 0.049). A total of 36 of 41 SGUS+ patients (88%) were anti-Ro/La+ compared to 14 of 26 SGUS- patients (54%) (P = 0.001). In addition, 24 of 39 SGUS+ patients (62%) were positive for rheumatoid factor (RF), whereas only 5 of 25 SGUS- patients (20%) were RF+ (P = 0.001). Conclusion Juvenile SS is characterized by a large spectrum of clinical symptoms and findings. Several glandular and extraglandular parameters such as hyposalivation, swollen salivary glands, and autoantibodies are associated with pathologic SGUS findings.
Palavras-chave
Referências
  1. ANAYA JM, 1995, J RHEUMATOL, V22, P1152
  2. Bogdanovic R, 2013, MOD RHEUMATOL, V23, P182, DOI 10.1007/s10165-012-0633-x
  3. Bossini N, 2001, NEPHROL DIAL TRANSPL, V16, P2328, DOI 10.1093/ndt/16.12.2328
  4. Both T, 2017, INT J MED SCI, V14, P191, DOI 10.7150/ijms.17718
  5. Carotti M, 2014, CLIN EXP RHEUMATOL, V32, pS61
  6. Cimaz R, 2003, EUR J PEDIATR, V162, P661, DOI 10.1007/s00431-003-1277-9
  7. Civilibal M, 2007, CLIN PEDIATR, V46, P738, DOI 10.1177/0009922807301945
  8. Cornec D, 2014, RHEUMATOLOGY, V53, P1604, DOI 10.1093/rheumatology/keu037
  9. DEVITA S, 1992, CLIN EXP RHEUMATOL, V10, P351
  10. Fang QG, 2013, J CRANIOFAC SURG, V24, pE413, DOI 10.1097/SCS.0b013e3182942ce3
  11. Goransson LG, 2011, SCAND J RHEUMATOL, V40, P221, DOI 10.3109/03009742.2010.536164
  12. Hammenfors DS, 2015, CLIN EXP RHEUMATOL, V33, P56
  13. Haugen AJ, 2008, SCAND J RHEUMATOL, V37, P30, DOI 10.1080/03009740701678712
  14. Houghton K, 2005, J RHEUMATOL, V32, P2225
  15. Johnsen SJ, 2013, ARTHRIT CARE RES, V65, P816, DOI 10.1002/acr.21887
  16. Jonsson R, 2007, ANN NY ACAD SCI, V1108, P433, DOI 10.1196/annals.1422.046
  17. Lee J, 2016, CLIN EXP RHEUMATOL, V34, P663
  18. Lieberman SM, 2013, CURR OPIN RHEUMATOL, V25, P651, DOI 10.1097/BOR.0b013e328363ed23
  19. Milic VD, 2009, J RHEUMATOL, V36, P1495, DOI 10.3899/jrheum.081267
  20. Nieto-Gonzalez JC, 2014, CLIN EXP RHEUMATOL, V32, P578
  21. Nikitakis NG, 2003, ORAL SURG ORAL MED O, V96, P42, DOI 10.1016/S1079-2104(03)00159-8
  22. Obinata K, 2010, ORAL SURG ORAL MED O, V109, P129, DOI 10.1016/j.tripleo.2009.08.033
  23. Pertovaara M, 2001, CLIN NEPHROL, V56, P10
  24. Salaffi F, 2000, J RHEUMATOL, V27, P1229
  25. Schuetz C, 2010, CLIN EXP RHEUMATOL, V28, P434
  26. Sepulveda JIR, 2017, BIOL SEX DIFFER, V8, DOI 10.1186/s13293-017-0146-6
  27. Seror R, 2015, RMD OPEN, V1, DOI 10.1136/rmdopen-2014-000022
  28. Shiboski CH, 2017, ARTHRITIS RHEUMATOL, V69, P35, DOI 10.1002/art.39859
  29. Virdee S, 2017, CLIN RHEUMATOL, V36, P2225, DOI 10.1007/s10067-017-3745-z
  30. VITALI C, 1993, ARTHRITIS RHEUM, V36, P340, DOI 10.1002/art.1780360309
  31. Vitali C, 2002, ANN RHEUM DIS, V61, P554, DOI 10.1136/ard.61.6.554
  32. Yokogawa N, 2016, CLIN EXP RHEUMATOL, V34, P343
  33. Yokogawa N, 2014, J RHEUMATOL, V41, P1178, DOI 10.3899/jrheum.131511