Associations between salivary gland histopathologic diagnoses and phenotypic features of Sjögren's syndrome among 1,726 registry participants

Troy E Daniels; Darren Cox; Caroline H Shiboski; Morten Schiødt; Ava Wu; Hector Lanfranchi; Hisanori Umehara; Yan Zhao; Stephen Challacombe; Mi Y Lam; Yvonne De Souza; Julie Schiødt; Helena Holm; Patricia A M Bisio; Mariana S Gandolfo; Toshioki Sawaki; Mengtao Li; Wen Zhang; Beni Varghese-Jacob; Per Ibsen; Alicia Keszler; Nozomu Kurose; Takayuki Nojima; Edward Odell; Lindsey A Criswell; Richard Jordan; John S Greenspan; Sjögren's International Collaborative Clinical Alliance Research Groups

doi:10.1002/art.30381

Associations between salivary gland histopathologic diagnoses and phenotypic features of Sjögren's syndrome among 1,726 registry participants

Troy E Daniels, Darren Cox, Caroline H Shiboski, Morten Schiødt, Ava Wu, Hector Lanfranchi, Hisanori Umehara, Yan Zhao, Stephen Challacombe, Mi Y Lam, Yvonne De Souza, Julie Schiødt, Helena Holm, Patricia A M Bisio, Mariana S Gandolfo, Toshioki Sawaki, Mengtao Li, Wen Zhang, Beni Varghese-Jacob, Per IbsenAlicia Keszler, Nozomu Kurose, Takayuki Nojima, Edward Odell, Lindsey A Criswell, Richard Jordan, John S Greenspan, Sjögren's International Collaborative Clinical Alliance Research Groups

164 Citations (Scopus)

Abstract

Objective To examine associations between labial salivary gland (LSG) histopathology and other phenotypic features of Sjögren's syndrome (SS). Methods The database of the Sjögren's International Collaborative Clinical Alliance (SICCA), a registry of patients with symptoms of possible SS as well as those with obvious disease, was used for the present study. LSG biopsy specimens from SICCA participants were subjected to protocol-directed histopathologic assessments. Among the 1,726 LSG specimens exhibiting any pattern of sialadenitis, we compared biopsy diagnoses against concurrent salivary, ocular, and serologic features. Results LSG specimens included 61% with focal lymphocytic sialadenitis (FLS; 69% of which had focus scores of âyen;1 per 4 mm²) and 37% with nonspecific or sclerosing chronic sialadenitis (NS/SCS). Focus scores of âyen;1 were strongly associated with serum anti-SSA/SSB positivity, rheumatoid factor, and the ocular component of SS, but not with symptoms of dry mouth or dry eyes. Those with positive anti-SSA/SSB were 9 times (95% confidence interval [95% CI] 7.4-11.9) more likely to have a focus score of âyen;1 than were those without anti-SSA/SSB, and those with an unstimulated whole salivary flow rate of <0.1 ml/minute were 2 times (95% CI 1.7-2.8) more likely to have a focus score of âyen;1 than were those with a higher flow rate, after controlling for other phenotypic features of SS. Conclusion Distinguishing FLS from NS/SCS is essential in assessing LSG biopsies, before determining focus score. A diagnosis of FLS with a focus score of âyen;1 per 4 mm², as compared to FLS with a focus score of <1 or NS/SCS, is strongly associated with the ocular and serologic components of SS and reflects SS autoimmunity.

Original language	English
Journal	Arthritis & Rheumatism
Volume	63
Issue number	7
Pages (from-to)	2021-30
Number of pages	10
ISSN	0004-3591
DOIs	https://doi.org/10.1002/art.30381
Publication status	Published - Jul 2011

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10.1002/art.30381

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Daniels, T. E., Cox, D., Shiboski, C. H., Schiødt, M., Wu, A., Lanfranchi, H., Umehara, H., Zhao, Y., Challacombe, S., Lam, M. Y., De Souza, Y., Schiødt, J., Holm, H., Bisio, P. A. M., Gandolfo, M. S., Sawaki, T., Li, M., Zhang, W., Varghese-Jacob, B., ... Sjögren's International Collaborative Clinical Alliance Research Groups (2011). Associations between salivary gland histopathologic diagnoses and phenotypic features of Sjögren's syndrome among 1,726 registry participants. Arthritis & Rheumatism, 63(7), 2021-30. https://doi.org/10.1002/art.30381

Daniels, TE, Cox, D, Shiboski, CH, Schiødt, M, Wu, A, Lanfranchi, H, Umehara, H, Zhao, Y, Challacombe, S, Lam, MY, De Souza, Y, Schiødt, J, Holm, H, Bisio, PAM, Gandolfo, MS, Sawaki, T, Li, M, Zhang, W, Varghese-Jacob, B, Ibsen, P, Keszler, A, Kurose, N, Nojima, T, Odell, E, Criswell, LA, Jordan, R, Greenspan, JS & Sjögren's International Collaborative Clinical Alliance Research Groups 2011, 'Associations between salivary gland histopathologic diagnoses and phenotypic features of Sjögren's syndrome among 1,726 registry participants', Arthritis & Rheumatism, vol. 63, no. 7, pp. 2021-30. https://doi.org/10.1002/art.30381

@article{f7bd9fb8e60f49a7a8bcd210313c6978,

title = "Associations between salivary gland histopathologic diagnoses and phenotypic features of Sj{\"o}gren's syndrome among 1,726 registry participants",

abstract = "Objective To examine associations between labial salivary gland (LSG) histopathology and other phenotypic features of Sj{\"o}gren's syndrome (SS). Methods The database of the Sj{\"o}gren's International Collaborative Clinical Alliance (SICCA), a registry of patients with symptoms of possible SS as well as those with obvious disease, was used for the present study. LSG biopsy specimens from SICCA participants were subjected to protocol-directed histopathologic assessments. Among the 1,726 LSG specimens exhibiting any pattern of sialadenitis, we compared biopsy diagnoses against concurrent salivary, ocular, and serologic features. Results LSG specimens included 61% with focal lymphocytic sialadenitis (FLS; 69% of which had focus scores of {\^a}yen;1 per 4 mm2) and 37% with nonspecific or sclerosing chronic sialadenitis (NS/SCS). Focus scores of {\^a}yen;1 were strongly associated with serum anti-SSA/SSB positivity, rheumatoid factor, and the ocular component of SS, but not with symptoms of dry mouth or dry eyes. Those with positive anti-SSA/SSB were 9 times (95% confidence interval [95% CI] 7.4-11.9) more likely to have a focus score of {\^a}yen;1 than were those without anti-SSA/SSB, and those with an unstimulated whole salivary flow rate of <0.1 ml/minute were 2 times (95% CI 1.7-2.8) more likely to have a focus score of {\^a}yen;1 than were those with a higher flow rate, after controlling for other phenotypic features of SS. Conclusion Distinguishing FLS from NS/SCS is essential in assessing LSG biopsies, before determining focus score. A diagnosis of FLS with a focus score of {\^a}yen;1 per 4 mm2, as compared to FLS with a focus score of <1 or NS/SCS, is strongly associated with the ocular and serologic components of SS and reflects SS autoimmunity.",

author = "Daniels, {Troy E} and Darren Cox and Shiboski, {Caroline H} and Morten Schi{\o}dt and Ava Wu and Hector Lanfranchi and Hisanori Umehara and Yan Zhao and Stephen Challacombe and Lam, {Mi Y} and {De Souza}, Yvonne and Julie Schi{\o}dt and Helena Holm and Bisio, {Patricia A M} and Gandolfo, {Mariana S} and Toshioki Sawaki and Mengtao Li and Wen Zhang and Beni Varghese-Jacob and Per Ibsen and Alicia Keszler and Nozomu Kurose and Takayuki Nojima and Edward Odell and Criswell, {Lindsey A} and Richard Jordan and Greenspan, {John S} and Ibsen, {Per Holger}",

note = "Copyright {\textcopyright} 2011 by the American College of Rheumatology.",

year = "2011",

month = jul,

doi = "10.1002/art.30381",

language = "English",

volume = "63",

pages = "2021--30",

journal = "Arthritis & Rheumatism",

issn = "0004-3591",

publisher = "Wiley",

number = "7",

}

TY - JOUR

T1 - Associations between salivary gland histopathologic diagnoses and phenotypic features of Sjögren's syndrome among 1,726 registry participants

AU - Daniels, Troy E

AU - Cox, Darren

AU - Shiboski, Caroline H

AU - Schiødt, Morten

AU - Wu, Ava

AU - Lanfranchi, Hector

AU - Umehara, Hisanori

AU - Zhao, Yan

AU - Challacombe, Stephen

AU - Lam, Mi Y

AU - De Souza, Yvonne

AU - Schiødt, Julie

AU - Holm, Helena

AU - Bisio, Patricia A M

AU - Gandolfo, Mariana S

AU - Sawaki, Toshioki

AU - Li, Mengtao

AU - Zhang, Wen

AU - Varghese-Jacob, Beni

AU - Ibsen, Per

AU - Keszler, Alicia

AU - Kurose, Nozomu

AU - Nojima, Takayuki

AU - Odell, Edward

AU - Criswell, Lindsey A

AU - Jordan, Richard

AU - Greenspan, John S

AU - Sjögren's International Collaborative Clinical Alliance Research Groups

PY - 2011/7

Y1 - 2011/7

N2 - Objective To examine associations between labial salivary gland (LSG) histopathology and other phenotypic features of Sjögren's syndrome (SS). Methods The database of the Sjögren's International Collaborative Clinical Alliance (SICCA), a registry of patients with symptoms of possible SS as well as those with obvious disease, was used for the present study. LSG biopsy specimens from SICCA participants were subjected to protocol-directed histopathologic assessments. Among the 1,726 LSG specimens exhibiting any pattern of sialadenitis, we compared biopsy diagnoses against concurrent salivary, ocular, and serologic features. Results LSG specimens included 61% with focal lymphocytic sialadenitis (FLS; 69% of which had focus scores of âyen;1 per 4 mm2) and 37% with nonspecific or sclerosing chronic sialadenitis (NS/SCS). Focus scores of âyen;1 were strongly associated with serum anti-SSA/SSB positivity, rheumatoid factor, and the ocular component of SS, but not with symptoms of dry mouth or dry eyes. Those with positive anti-SSA/SSB were 9 times (95% confidence interval [95% CI] 7.4-11.9) more likely to have a focus score of âyen;1 than were those without anti-SSA/SSB, and those with an unstimulated whole salivary flow rate of <0.1 ml/minute were 2 times (95% CI 1.7-2.8) more likely to have a focus score of âyen;1 than were those with a higher flow rate, after controlling for other phenotypic features of SS. Conclusion Distinguishing FLS from NS/SCS is essential in assessing LSG biopsies, before determining focus score. A diagnosis of FLS with a focus score of âyen;1 per 4 mm2, as compared to FLS with a focus score of <1 or NS/SCS, is strongly associated with the ocular and serologic components of SS and reflects SS autoimmunity.

AB - Objective To examine associations between labial salivary gland (LSG) histopathology and other phenotypic features of Sjögren's syndrome (SS). Methods The database of the Sjögren's International Collaborative Clinical Alliance (SICCA), a registry of patients with symptoms of possible SS as well as those with obvious disease, was used for the present study. LSG biopsy specimens from SICCA participants were subjected to protocol-directed histopathologic assessments. Among the 1,726 LSG specimens exhibiting any pattern of sialadenitis, we compared biopsy diagnoses against concurrent salivary, ocular, and serologic features. Results LSG specimens included 61% with focal lymphocytic sialadenitis (FLS; 69% of which had focus scores of âyen;1 per 4 mm2) and 37% with nonspecific or sclerosing chronic sialadenitis (NS/SCS). Focus scores of âyen;1 were strongly associated with serum anti-SSA/SSB positivity, rheumatoid factor, and the ocular component of SS, but not with symptoms of dry mouth or dry eyes. Those with positive anti-SSA/SSB were 9 times (95% confidence interval [95% CI] 7.4-11.9) more likely to have a focus score of âyen;1 than were those without anti-SSA/SSB, and those with an unstimulated whole salivary flow rate of <0.1 ml/minute were 2 times (95% CI 1.7-2.8) more likely to have a focus score of âyen;1 than were those with a higher flow rate, after controlling for other phenotypic features of SS. Conclusion Distinguishing FLS from NS/SCS is essential in assessing LSG biopsies, before determining focus score. A diagnosis of FLS with a focus score of âyen;1 per 4 mm2, as compared to FLS with a focus score of <1 or NS/SCS, is strongly associated with the ocular and serologic components of SS and reflects SS autoimmunity.

U2 - 10.1002/art.30381

DO - 10.1002/art.30381

M3 - Journal article

SN - 0004-3591

VL - 63

SP - 2021

EP - 2030

JO - Arthritis & Rheumatism

JF - Arthritis & Rheumatism

IS - 7

ER -

Associations between salivary gland histopathologic diagnoses and phenotypic features of Sjögren's syndrome among 1,726 registry participants

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