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Lee and Song: A Meta-analysis of the Diagnostic Value of Minor Salivary Gland Biopsy for Primary Sjogren's Syndrome
Original Article

J Rheum Dis 2014;21(6):303-310.

Published online: 31 October 2014

DOI: https://doi.org/10.4078/jrd.2014.21.6.303

A Meta-analysis of the Diagnostic Value of Minor Salivary Gland Biopsy for Primary Sjogren's Syndrome

Young Ho Lee, Gwan Gyu Song

Division of Rheumatology, Korea University Medical Center, Division of Rheumatology, Department of Internal Medicine, Korea University College of Medicine, Seoul, Korea

Corresponding to: Young Ho Lee, Division of Rheumatology, Korea University Medical Center, Korea University College of Medicine, 216-1, Anam-dong 5-ga, Seongbuk-gu, Seoul 136-705, Korea. E-mail: lyhcgh@korea.ac.kr

Received 3 September 2014       Revised 26 October 2014       Accepted 27 October 2014

Copyright © 2014 The Korean College of Rheumatology

This is a Free Access article, which permits unrestricted non-commerical use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract

Objective

The purpose of this study is to evaluate the diagnostic performance of minor salivary gland biopsy (MSGB) for patients with primary Sjogren's syndrome (pSS).

Methods

We have conducted a search from Medline, Embase, and Cochrane Library databases, and performed a meta-analysis on the diagnostic accuracy of MSGB in pSS patients.

Results

A total of eight studies, including 583 pSS and 627 non-pSS patients, were available for the meta-analysis. The pooled sensitivity and specificity of MSGB were 75.7% (95% confidence interval [CI], 72.0∼79.1) and 90.7% (88.1∼92.9), respectively. The positive likelihood ratio, negative likelihood ratio, and diagnostic odds ratio were 9.475 (4.051∼22.16), 0.266 (0.208∼0.340), and 38.92 (19.12∼72.21), respectively. The area under the curve was 0.901 and the Q* index was 0.902, indicating a high diagnostic accuracy. Some between-study heterogeneity was found in the meta-analyses; however, there was no evidence of a threshold effect (Spearman correlation co-efficient=0.419; p=0.301). Meta-regression showed that the study quality, sample size, study design, and diagnostic criteria were not sources of heterogeneity, and subgroup meta-analyses did not change the overall diagnostic accuracy.

Conclusion

Our meta-analysis of published studies demonstrates that MSGB has a high diagnostic accuracy and may play an important role in the diagnosis of pSS.

Keywords: Primary Sjogren's syndrome, Minor salivary gland biopsy, Diagnostic accuracy, Meta-analysis

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Figure 1.
Flow diagram of study selection.
jrd-21-303f1.tif
Figure 2.
Sensitivity (A) and specificity (B) estimates for minor salivary gland biopsy for the diagnosis of primary Sjogren's syndrome. Circles and lines represent point estimates and 95% confidence intervals, respectively. Circled areas represent relative study sizes.
jrd-21-303f2.tif
Figure 3.
Positive (A) and negative (B) likelihood ratios, and diagnostic odds ratio (C) estimates for minor salivary gland biopsy for the diagnosis of primary Sjogren's syndrome. Circles and lines represent point estimates and 95% confidence intervals, respectively. Circled areas represent the relative study size.
jrd-21-303f3.tif
Figure 4.
Summary receiver-operating characteristic curves for minor salivary gland biopsy for the diagnosis of primary Sjogren's syndrome. Solid circles represent individual studies included in this meta-analysis. The curve shown is a regression line that summarizes the overall diagnostic accuracy. SE (AUC): standard error of the area under the curve, Q*: an index defined by the point on the SROC curve where the sensitivity and specificity are equal, SE(Q*): Q* index standard error.
jrd-21-303f4.tif
Table 1.
Characteristics of individual studies included in the meta-analysis
Author Country Diagnostic criteria pSS Non-pSS Study design Cut-off TP TN FN FP Anti-Ro (%) Anti-La (%) Study quality*
Cornec, 2013 (5) France Expert Opinion 78 80 Prospective FS ≥1 63 67 15 13 56.4† Na 10
Obinata, 2010 (6) Japan Revised Japanese criteria 36 37 Retrospective FS ≥1 23 34 13 3 Na Na 12
Nakamura, 2010 (7) Japan AECG 63 49 Retrospective FS ≥1 58 30 5 19 58.7 17.5 11
Milic, 2009 (8) Serbia AECG 107 28 Prospective FS ≥1 70 28 37 0 Na Na 10
Yazisiz, 2009 (9) Turkey AECG 99 54 Retrospective FS ≥1 78 54 21 0 17.7 8.3 10
Caporali, 2008 (10) Italy AECG 124 254 Retrospective Cumulative FS ≥1 87 248 37 6 80.6 29.6 11
Teppo, 2007 (11) Finland AECG 60 100 Retrospective FS ≥1 49 83 11 17 Na Na 11
Kessel, 2006 (12) Israel AECG 16 25 Prospective FS ≥1 15 25 1 0 62.5 37.5 9

AECG: American-European Consensus Group criteria, pSS: primary Sjogren's syndrome, FS: focal score, TP: true positive, FP: false positive, FN: false negative, TN: true negative, Na: not available.

* Quality Assessment of Diagnostic Accuracy Studies (QUADAS) criteria.

† Anti-Ro or La positivity.

Table 2.
Summary results of the meta-analysis
Subgroup Population Study no. N
 Sensitivity Specificity PLR NLR DOR
pSS Non-pSS
All combined Overall 8 583 627 0.757 0.907 9.475 0.266 38.92
          (0.720∼0.791) (0.881∼0.929) (4.051∼22.16) (0.208∼0.340) (19.12∼72.21)
Study quality QUADAS＞11 4 283 440 0.767 0.898 6.888 0.274 31.01
          (0.712∼0.815) (0.866∼0.924) (2.288∼20.73) (0.193∼0.389) (13.33∼72.14)
  QUADAS≤10 4 300 187 0.748 0.929 23.59 0.246 101.27
          (0.695∼0.796) (0.882∼0.962) (2.600∼214.0) (0.154∼0.395) (14.71∼696.9)
Study design Prospective 3 201 133 0.729 0.900 14.23 0.247 62.03
          (0.662∼0.789) (0.835∼0.946) (2.033∼99.74) (0.121∼0.502) (9.287∼414.4)
  Retrospective 5 382 494 0.772 0.909 9.020 0.261 37.60
          (0.727∼0.813) (0.880∼0.933) (2.803∼29.02) (0.196∼0.347) (15.21∼92.92)

pSS: primary Sjogren's syndrome, PLR: positive likelihood ratio, NLR: negative likelihood ratio, DOR: diagnostic OR, QUADAS: Quality Assessment of Diagnostic Accuracy Studies criteria.

Table 3.
Estimates of summary receiver operating characteristic curve parameters
Subgroup Population Study no. Numbers
 AUC SE(AUC) Q* SE(Q*)
pSS Non-pSS
All combined Overall 8 583 627 0.902 0.014 0.833 0.016
Study quality QUADAS＞11 4 283 440 0.903 0.019 0.835 0.020
  QUADAS≤10 4 300 187 0.886 0.032 0.816 0.033
Study design Prospective 3 201 133 0.881 0.032 0.818 0.033
  Retrospective 5 382 494 0.905 0.017 0.836 0.018

AUC: area under the curve, SE: standard error, QUADAS: Quality Assessment of Diagnostic Accuracy Studies criteria, NA: not available.

Table 4.
Meta-regression analysis of potential sources of heterogeneity
Covariates Coefficient SE RDOR (95% CI) p-value
QUADAS −1.477 1.517 0.23 (0.00∼156.4) 0.433
Sample size −0.598 0.925 0.55 (0.01∼29.47) 0.584
Study design −0.407 0.670 0.67 (0.00∼879.7) 0.830
Diagnostic criteria 1.464 0.824 4.32 (0.12∼150.1) 0.217

SE: standard error, RDOR: relative diagnostic odds ratio, QUADAS: Quality Assessment of Diagnostic Accuracy Studies criteria.

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