Journal of Shanghai Jiao Tong University (Medical Science) >

2025 , Vol. 45 >Issue 3: 373 - 380

DOI: https://doi.org/10.3969/j.issn.1674-8115.2025.03.015

Clinical research

A nomogram based on ultrasound scoring parameters and clinical indicators for differentiating primary Sjὅgren′s syndrome from IgG4-related sialadenitis

  • LIU Chuxuan ,
  • ZUO Jiaxin ,
  • XIONG Ping
Expand
  • Department of Ultrasound, Shanghai Ninth People's Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai 200011, China
XIONG Ping, E-mail: xiong_ping_xp@163.com.

Received date: 2024-09-02

  Accepted date: 2024-12-13

  Online published: 2025-03-28

Supported by

Cross-Disciplinary Research Fund Project of Shanghai Ninth People′s Hospital, Shanghai Jiao Tong University School of Medicine(JYJC202132);Discipline Cluster Construction Project of Shanghai Ninth People′s Hospital, Shanghai Jiao Tong University School of Medicine

Fold

Abstract

Objective ·To construct and validate a nomogram for distinguishing primary Sjὅgren′s syndrome (PSS) from immunoglobulin G4-related sialadenitis (IgG4-RS) based on ultrasound scoring parameters and clinical indicators. Methods ·A total of 141 patients with PSS and 31 patients with IgG4-RS were retrospectively recruited from Shanghai Ninth People′s Hospital, Shanghai Jiao Tong University School of Medicine from January 2018 to December 2023. The ultrasound scoring parameters, including parotid gland ultrasound (PGUS) score, submandibular gland ultrasound (SMGUS) score, and salivary gland ultrasound (SGUS) score, along with clinical indicators such as gender, age, anti-SSB/La antibody, anti-SSA/Ro60 antibody, anti-SSA/Ro52 antibody, IgG, and rheumatoid factor (RF), were collected. The optimal US scoring parameters and clinical indicators were screened by least absolute shrinkage and selection operator (LASSO) regression, and a nomogram model was constructed to distinguish PSS from IgG4-RS. The internal validation of the model was carried out through bootstrap method. Receiver operator characteristic (ROC) curve, calibration curve and decision curve analysis (DCA) were used to estimate the discrimination, calibration, and clinical utility of the nomogram model, respectively. Results ·LASSO regression identified six major variables: gender, age, anti-SSA/Ro60 antibody, anti-SSA/Ro52 antibody, PGUS score, and SMGUS score. These variables were used to construct the nomogram. ROC curve of the nomogram showed that the area under the curve (AUC) was 0.976, indicating the nomogram had strong discrimination ability. The bootstrap method was used for internal validation with 1 000 resampling iterations, and the average absolute error was 0.018. Calibration curve demonstrated good agreement between predicted and observed values. DCA indicated that the nomogram had certain clinical utility. Conclusion ·The nomogram based on ultrasound scoring parameters and clinical indicators demonstrates excellent discrimination and calibration in differentiating PSS from IgG4-RS. It has the potential to assist in clinical diagnosis and decision-making.

Key words： ultrasound scoring system; primary sj?gren′s syndrome; IgG4-related sialadenitis; differential diagnosis; nomogram

Cite this article

LIU Chuxuan , ZUO Jiaxin , XIONG Ping . A nomogram based on ultrasound scoring parameters and clinical indicators for differentiating primary Sjὅgren′s syndrome from IgG4-related sialadenitis[J]. Journal of Shanghai Jiao Tong University (Medical Science), 2025 , 45(3) : 373 -380 . DOI: 10.3969/j.issn.1674-8115.2025.03.015

References

1 俞光岩. 多发性唾液腺肿大的鉴别诊断及处理[J]. 北京大学学报(医学版), 2021, 53(1): 1-4.
  YU G Y. Differential diagnosis and management of multiple salivary gland enlargements[J]. Journal of Peking University (Health Sciences), 2021, 53(1): 1-4.
2 NARVáEZ J, SáNCHEZ-FERNáNDEZ S á, SEOANE-MATO D, et al. Prevalence of Sj?gren′s syndrome in the general adult population in Spain: estimating the proportion of undiagnosed cases[J]. Sci Rep, 2020, 10(1): 10627.
3 BRITO-ZERóN P, RAMOS-CASALS M, BOSCH X, et al. The clinical spectrum of IgG4-related disease[J]. Autoimmun Rev, 2014, 13(12): 1203-1210.
4 L?HR J M, VUJASINOVIC M, ROSENDAHL J, et al. IgG4-related diseases of the digestive tract[J]. Nat Rev Gastroenterol Hepatol, 2022, 19(3): 185-197.
5 FRAGOULIS G E, ZAMPELI E, MOUTSOPOULOS H M. IgG4-related sialadenitis and sj?gren′s syndrome[J]. Oral Dis, 2017, 23(2): 152-156.
6 SEROR R, NOCTURNE G, MARIETTE X. Current and future therapies for primary Sj?gren syndrome[J]. Nat Rev Rheumatol, 2021, 17(8): 475-486.
7 HONG X, ZHANG Y Y, LI W, et al. Treatment of immunoglobulin G4-related sialadenitis: outcomes of glucocorticoid therapy combined with steroid-sparing agents[J]. Arthritis Res Ther, 2018, 20(1): 12.
8 杜晶晶, 董兴红, 高洁, 等. 原发性干燥综合征患者抗SSB与其他实验室参数相关性研究[J]. 检验医学与临床, 2023, 20(16): 2395-2399.
  DU J J, DONG X H, GAO J, et al. Correlations between anti-SSB and other laboratory parameters in patients with primary Sj?gren′s syndrome[J]. Laboratory Medicine and Clinic,2023, 20(16): 2395-2399.
9 罗盈, 文章雪, 王子霞, 等. IgG4水平及IgG4/IgG比值对IgG4相关性疾病的临床价值[J]. 国际检验医学杂志, 2024, 45(7): 804-808.
  LUO Y, WEN Z X, WANG Z X, et al. Clinical value of IgG4 level and IgG4/IgG ratioin in IgG4-related diseases[J]. International Journal of Laboratory Medicine, 2024, 45(7): 804-808.
10 蒋佳纯, 许世豪, 王晓冰, 等. 涎腺超声评分联合血清学指标诊断原发性干燥综合征的临床价值[J]. 中国现代医生, 2021, 59(31): 131-134, 193.
  JIANG J C, XU S H, WANG X B, et al. Clinical value of salivary gland ultrasound scoring combined with serological indicators in diagnosis of primary sj?gren′s syndrome[J]. China Modern Doctor, 2021, 59(31): 131-134, 193.
11 CULVER L, SADLER R, SIMPSON D, et al. Elevated serum IgG4 levels in diagnosis, treatment response, organ involvement, and relapse in a prospective IgG4-related disease UK cohort[J]. Am J Gastroenterol, 2016, 111(5): 733-743.
12 吴海兰, 王婧玲, 陈莉. 唾液腺超声评分系统在干燥综合征诊断中的应用进展[J]. 中国医学影像学杂志, 2022, 30(11): 1188-1191, 1196.
  WU H L, WANG J L, CHEN L. Application progress of salivary gland ultrasound scoring system in Sj?gren′s syndrome[J].Chinese Journal of Medical Imaging, 2022, 30(11): 1188-1191, 1196.
13 刘杨, 程昉, 王艳玲, 等. 涎腺超声对原发性干燥综合征的诊断和评估价值[J]. 中华临床免疫和变态反应杂志, 2021, 15(5): 528-533.
  LIU Y, CHEN F, WANG Y L, et al. Diagnostic and evaluation value of salivary gland ultrasonography in primary Sj?gren′s syndrome[J]. Chinese Journal of Allergy & Clinical Immunology, 2021, 15(5): 528-533.
14 宁晓然, 王子乔, 张珊珊, 等. 超声评分系统在IgG4相关涎腺炎评估中的应用[J]. 北京大学学报(医学版), 2019, 51(6): 1032-1035.
  NING X R, WANG Z Q, ZHANG S S, et al. Application of ultrasonography scoring system in the assessment of IgG4-related sialadenitis[J]. Journal of Peking University (Health Sciences), 2019, 51(6): 1032-1035.
15 NEGRINI S, EMMI G, GRECO M, et al. Sj?gren′s syndrome: a systemic autoimmune disease[J]. Clin Exp Med, 2022, 22(1): 9-25.
16 UMEHARA H, OKAZAKI K, MASAKI Y, et al. Comprehensive diagnostic criteria for IgG4-related disease (IgG4-RD), 2011[J]. Mod Rheumatol, 2012, 22(1): 21-30.
17 HOC?EVAR A, AMBROZ?IC? A, ROZMAN B, et al. Ultrasonographic changes of major salivary glands in primary Sjo?gren′s syndrome. Diagnostic value of a novel scoring system[J]. Rheumatology (Oxford), 2005, 44(6): 768-772.
18 KOO T K, LI M Y. A guideline of selecting and reporting intraclass correlation coefficients for reliability research[J]. J Chiropr Med, 2016, 15(2): 155-163.
19 ALENZI F, ALQAHTANI B, ALHAMAD E H, et al. Fatigue in Saudi patients with primary sj?gren′s syndrome and its correlation with disease characteristics and outcome measures: a cross-sectional study[J]. Open Access Rheumatol, 2020, 12: 303-308..
20 ZEHRFELD N, ABELMANN M, BENZ S, et al. Primary Sj?gren′s syndrome independently promotes premature subclinical atherosclerosis[J]. RMD Open, 2024, 10(2): e003559.
21 MULLER R, EBBO M, HABERT P, et al. Thoracic manifestations of IgG4-related disease[J]. Respirology, 2023, 28(2): 120-131.
22 HONG X, LI W, XIE X Y, et al. Differential diagnosis of IgG4-related sialadenitis, primary Sj?gren syndrome, and chronic obstructive submandibular sialadenitis[J]. Br J Oral Maxillofac Surg, 2017, 55(2): 179-184.
23 CONTICINI E, D′ALESSANDRO R, BARDELLI M, et al. Routine IgG4 staining in minor salivary gland biopsy in a cohort of Italian Caucasian patients suffering from xerostomia[J]. Reumatologia, 2022, 60(1): 12-15.
24 范文强, 吴洁, 马玲, 等. 抗SSA抗体、抗SSB抗体、抗α-胞衬蛋白抗体联合检测在干燥综合征诊断中价值探讨[J]. 中华生物医学工程杂志, 2017, 23(3): 239-243.
  FAN W Q, WU J, MA L, et al. Value of combined detection of SSA antibody, anti-SSB antibody, and anti-α-fodrin antibody in diagnosis of Sj?gren′s syndrome[J]. Chinese Journal of Biomedical Engineering, 2017, 23(3): 239-243.
25 喻晓雯, 王琴, 冯婧, 等. 原发性干燥综合征的自身抗体研究进展[J]. 中国免疫学杂志, 2018, 34(2): 301-305.
  YU X W, WANG Q, FENG J, et al. Advances in research on autoantibodies of primary sj?gren′s syndrome[J]. Chinese Journal of Immunology, 2018, 34(2): 301-305.
26 ABDEL RAZEK A A K, MUKHERJI S. Imaging of sialadenitis[J]. Neuroradiol J, 2017, 30(3): 205-215.
27 MOSSEL E, VAN GINKEL M S, HAACKE E A, et al. Histopathology, salivary flow and ultrasonography of the parotid gland: three complementary measurements in primary Sj?gren′s syndrome[J]. Rheumatology (Oxford), 2022, 61(6): 2472-2482.
28 ZHOU M Z, LIU Y Y, ZHANG S S, et al. Diagnostic value of a novel salivary gland ultrasound scoring system in IgG4-related sialadenitis[J]. Rheumatology (Oxford), 2025, 64(2): 747-755.
Options
Outlines

/

〈 〉