Journal of Peking University(Health Sciences) ›› 2019, Vol. 51 ›› Issue (1): 1-3. doi: 10.19723/j.issn.1671-167X.2019.01.001

    Next Articles

Clinicopathological characteristics and diagnosis of IgG4-related sialadenitis

Guang-ya YU1,2,(),Xia HONG1,2,Wei LI1,Yan-yan ZHANG1,Yan GAO3,Yan CHEN3,Zu-yan ZHANG4,Xiao-yan XIE4,Zhan-guo LI5,Yan-ying LIU5,Jia-zeng SU1,Wen-xuan ZHU1,Zhi-peng SUN4   

  1. 1. Department of Oral & Maxillofacial Surgery, Peking University School and Hospital of Stomatology & National Engineering Laboratory for Digital and Material Technology of Stomatology & Beijing Key Laboratory of Digital Stomatology, Beijing 100081, China
    2. Center of Stomatology, Peking University Shenzhen Hospital, Shenzhen 518036, Guangdong, China
    3. Department of Oral Pathology, Peking University School and Hospital of Stoma-tology, Beijing 100081, China
    4. Department of Oral Radiology, Peking University School and Hospital of Stoma-tology, Beijing 100081, China
    5. Department of Rheumatology and Immunology, Peking University People’s Hospital, Beijing, 100044, China
  • Received:2018-11-26 Online:2019-02-18 Published:2019-02-26
  • Contact: Guang-ya YU E-mail:gyyu@263.net
  • Supported by:
    Supported by the National Natural Science Foundation of China(81470756);Supported by the National Natural Science Foundation of China(81671005);Supported by the National Natural Science Foundation of China(81611540351)

Abstract:

SUMMARY Immunoglobulin G4-related sialadenitis (IgG4-RS) is a newly recognized immune-mediated disease and one of immunoglobulin G4-related diseases (IgG4-RD). Our multidisciplinary research group investigated the clinicopathological characteristics and diagnosis of IgG4-RS during the past 10 years. Clinically, it showed multiple bilateral enlargement of major salivary glands (including sublingual and accessory parotid glands) and lacrimal glands. The comorbid diseases of head and neck region including rhinosinusitis, allergic rhinitis, and lymphadenopathy were commonly seen, which could occur more early than enlargement of major salivary glands. Internal organ involvements, such as autoimmune pancreatitis, sclerosing cholangitis, and interstitial pneumonia could also be seen. Thirty-five (38.5%)patients had the symptom of xerostomia. Saliva flow at rest was lower than normal. Secretory function was reduced more severely in the submandibular glands than in the parotid glands. Serum levels of IgG4 were elevated in almost all the cases and the majority of the patients had increased IgE levels. CT, ultrasonography, and sialography showed their imaging characteristics. Histologically it showed marked lymphoplasmacytic inflammation, large irregular lymphoid follicles with expanded germinal centers, prominent cellular interlobular fibrosis, eosinophil infiltration, and obliterative phlebitis. Their immunohistological examination showed marked IgG-positive and IgG4-positive plasma cell infiltration and high IgG4/IgG ratio. The disease could be divided into three stages according to severity of glandular fibrosis. The serum IgG4 level was higher and the saliva secretion lower as glandular fibrosis increased. IgG4-RS should be differentiated from other diseases with enlargement of major salivary gland and lacrimal gland, such as primary Sjögren syndrome, chronic obstructive submandibular sialadenitis, and eosinophilic hyperplastic lymphogranuloma.

Key words: IgG4-related sialadentitis, IgG4-related disease, Salivary gland, Submandibular gland, Parotid gland, Lacrimal gland

CLC Number: 

  • R780.2
[1] Geyer JT, Deshpande V . IgG4-associated sialadenitis[J]. Curr Opin Rheumatol, 2011,23(1):95-101.
doi: 10.1097/BOR.0b013e3283413011
[2] Brito-Zeron P, Ramos-Casals M, Bosch X , et al. The clinical spectrum of IgG4-related disease[J]. Autoimmun Rev, 2014,13(12):1203-1210.
doi: 10.1016/j.autrev.2014.08.013 pmid: 25151972
[3] Li W, Chen Y, Sun ZP , et al. Clinicopathological characteristics of immunoglobulin G4-related sialadenitis[J]. Arthritis Res Ther, 2015,17(1):186-195.
doi: 10.1186/s13075-015-0698-y pmid: 4508811
[4] Hong X, Sun ZP, Li W , et al. Comorbid diseases of IgG4-related sialadenitis in the head and neck region[J]. Laryngoscope, 2015,125(9):2113-2118.
doi: 10.1002/lary.25387 pmid: 25994602
[5] Hong X, Li W, Su JZ , et al. Internal organ involvement in IgG4-related sialadenitis: a systemic review[J]. Chin J Dent Res, 2015,18(2):85-94.
pmid: 26167546
[6] 李巍, 孙志鹏, 刘筱菁 , 等. 腮腺和颌下腺CT体积的测量[J]. 北京大学学报(医学版), 2014,46(2):288-293.
doi: 10.3969/j.issn.1671-167X.2014.02.022
[7] Li W, Xie XY, Su JZ , et al. Ultrasonographic features of immunoglobulin G4-related sialadenitis[J]. Ultrasound Med Biol, 2016,42(1):167-175.
doi: 10.1016/j.ultrasmedbio.2015.09.014 pmid: 26518180
[8] Hong X, Li W, Xie XY , et al. Differential diagnosis of IgG4-related sialadenitis, primary Sjögren syndrome, and chronic obstructive submandibular sialadenitis[J]. Brit J Oral Maxillofac Surg, 2017,55(1):179-184.
doi: 10.1016/j.bjoms.2016.10.021 pmid: 27866757
[9] 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.
doi: 10.3109/s10165-011-0571-z
[10] Seifert G . Sialadenitis[M] //Seifert G, Haubrich J. Diseases of thesalivary gland. New York: Thieme Inc., 1986: 140-146.
[11] Khosroshahi A, Wallace ZS, Crowe JL , et al. International consensus guidance statement on the management and treatment of IgG4-related disease. Arthr Rheumatol, 2015,67(7):1688-1699.
doi: 10.1002/art.39132 pmid: 25809420
[12] Yamamoto M, Yajima H, Takahashi H , et al. Everyday clinical practice in IgG4-related dacryoadenitis and/or sialadenitis: results from the SMART database[J]. Mod Rheumatol, 2015,25(2):199-204.
doi: 10.3109/14397595.2014.950036 pmid: 25159154
[13] Hong X, Zhang YY, 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):12-21.
doi: 10.1186/s13075-017-1507-6 pmid: 29382364
[1] YU Guang-yan,LIU Deng-gao,LI Wei,HONG Xia,ZHANG Yan-yan,ZHU Wen-xuan,ZHANG Ke-fu,LI Xiao,LI Zhan-guo,LIU Yan-ying,CHEN Yan,GAO Yan,SU Jia-zeng. Studies on newly recognized chronic sialadenitis [J]. Journal of Peking University (Health Sciences), 2022, 54(1): 13-17.
[2] CHEN Chao-lun,SU Jia-zeng,YU Guang-yan. Effects of acid stimulation on saliva flow rate and compositions of human parotid and submandibular glands [J]. Journal of Peking University (Health Sciences), 2022, 54(1): 89-94.
[3] ZHU Yi-ying,MIN Sai-nan,YU Guang-yan. Effect of topical injection of cyclosporine A on saliva secretion and inflammation in the submandibular gland of non-obese diabetic mice [J]. Journal of Peking University (Health Sciences), 2021, 53(4): 750-757.
[4] WANG Yi-ping,CAI Zhi-gang,PENG Xin,ZHANG Jie,SUN Zhi-peng,LI Wei,ZHANG Lei,YU Guang-yan. Measurement of the weight and volume of submandibular gland in vitro [J]. Journal of Peking University (Health Sciences), 2021, 53(1): 126-132.
[5] Huan-bin YU,Wen-jie WU,Xiao-ming LV,Yan SHI,Lei ZHENG,Jian-guo ZHANG. 125I seed brachytherapy for recurrent salivary gland carcinoma after external radiotherapy [J]. Journal of Peking University (Health Sciences), 2020, 52(5): 919-923.
[6] Ye ZHANG,Ni ZHANG,Xiao-xiao LIU,Chuan-xiang ZHOU. Cervical lymph node metastasis in adenoid cystic carcinoma of the salivary glands: A clinicopathologic study [J]. Journal of Peking University(Health Sciences), 2020, 52(1): 30-34.
[7] Zi-qiao WANG,Yan-ying LIU,Xia ZHANG,Tian LIU,Li-min REN,Dan-hua SHEN,Yi WANG,Zhan-guo LI. Analysis of the clinical features and misdiagnosis reasons of 17 patients misdiagnosed with IgG4-related disease [J]. Journal of Peking University(Health Sciences), 2019, 51(6): 1025-1031.
[8] Xin CONG,Sai-nan MIN,Li-ling WU,Zhi-gang CAI,Guang-yan YU. Role and mechanism of muscarinic acetylcholine receptor in the regulation of submandibular gland secretion [J]. Journal of Peking University(Health Sciences), 2019, 51(3): 390-396.
[9] Qian SU,Xin PENG,Chuan-xiang ZHOU,Guang-yan YU. Clinicopathological features and possible prognostic factors in parotid lymphomas [J]. Journal of Peking University(Health Sciences), 2019, 51(1): 35-42.
[10] ZHANG Ya-qiong, YE Xin, LIU Deng-gao, ZHAO Ya-ning, XIE Xiao-yan, YU Guang-yan. Endoscopy-assisted sialodochoplasty for the treatment of severe sialoduct stenosis [J]. Journal of Peking University(Health Sciences), 2018, 50(1): 160-164.
[11] YU Guang-yan, WU Li-ling, CAI Zhi-gang, LV Lan, CONG Xin. A 20-year study on microvascular autologous transplantation of submandibular gland for treatment of severe dry eye [J]. Journal of Peking University(Health Sciences), 2018, 50(1): 1-4.
[12] WANG Wei, ZHENG Lei, LIU Shu-ming, HUANG Ming-wei, SHI Yan, LV Xiao-ming, ZHANG Jie, ZHANG Jian-guo. Distant metastases of malignant salivary gland carcinoma after treated by 125Ⅰinternal brachy therapy alone [J]. Journal of Peking University(Health Sciences), 2017, 49(3): 547-550.
[13] LI Qiong, CHANG Zhi-fang, YANG Guo-an, PANG Chun-yan, WANG Yong-fu. Effect of type 1 sphingosine-1-phosphate receptor siRNA on human salivary gland cells [J]. Journal of Peking University(Health Sciences), 2016, 48(6): 987-993.
[14] WANG Wei, ZHENG Lei, LIU Shu-Ming, HUANG Ming-Wei, SHI Yan, LV Xiao-Ming , ZHANG Jie, ZHANG Jian-Guo. Clinical investigation on distant metastases of salivary gland carcinoma after being treated by surgery combined with 125I internal brachytherapy [J]. Journal of Peking University(Health Sciences), 2015, 47(3): 504-508.
[15] CONG Xin, ZHANG Yan, YU Guang-Yan, WU Li-Ling.  Activation of transient receptor potential vanilloid subtype 1 serves as a novel pathway to modulate secretion in submandibular gland [J]. Journal of Peking University(Health Sciences), 2015, 47(1): 8-12.
Viewed
Full text


Abstract

Cited

  Shared   
  Discussed   
[1] . [J]. Journal of Peking University(Health Sciences), 2009, 41(4): 456 -458 .
[2] . [J]. Journal of Peking University(Health Sciences), 2009, 41(2): 125 -128 .
[3] . [J]. Journal of Peking University(Health Sciences), 2009, 41(2): 135 -140 .
[4] . [J]. Journal of Peking University(Health Sciences), 2009, 41(2): 158 -161 .
[5] . [J]. Journal of Peking University(Health Sciences), 2009, 41(2): 217 -220 .
[6] . [J]. Journal of Peking University(Health Sciences), 2009, 41(1): 52 -55 .
[7] . [J]. Journal of Peking University(Health Sciences), 2009, 41(1): 109 -111 .
[8] . [J]. Journal of Peking University(Health Sciences), 2009, 41(3): 297 -301 .
[9] . [J]. Journal of Peking University(Health Sciences), 2009, 41(5): 599 -601 .
[10] . [J]. Journal of Peking University(Health Sciences), 2009, 41(5): 516 -520 .