EOSINOPHILIC SIALODOCHITIS: DESCRIPTION OF TWO CASES AND LITERATURE REVIEW
https://doi.org/10.25587/2587-5590-2024-2-17-29
Abstract
Eosinophilic sialodochitis (or eosinophilic sialoadenitis, synonyms sialodochitis fibrinosa, Kussmaul’s disease) is a rare disease and is clinically manifested by recurrent swelling of the parotid and/or submandibular salivary glands, often accompanied by pain or itching over the affected glands, and the secretion of mucous plugs from the gland ducts due to obstruction of the excretory ducts. The pathogenesis of the disease is based on a local and systemic atopic reaction associated with activation of eosinophils, their primarily infiltration of the large salivary gland ducts, which leads to sialodochitis, accompanied by eosinophilia, increased synthesis of IgE and often related with concomitant allergic diseases, such as bronchial asthma, vasomotor rhinitis. We have not found any descriptions of this rare disorder in the Russian literature. In this article, we present two cases of eosinophilic sialodochitis, also focusштп on clinical and laboratory manifestations, pathogenesis, and differential diagnosis of diseases with similar manifestations, analyzing the literature and approaches to the treatment of this allergic condition. On the example of one patient with a long and severe course of eosinophilic sialodochitis in combination with a poorly controlled bronchial asthma with inefficacy of the standard treatment, prescription of a biological agent from the group of IL-5 inhibitors – benralizumab, we succeeded in achievement of a significant improvement in the both diseases courses.
About the Authors
V. I. Vasiliev
LLC "Revmotsentr"
Russian Federation
Russian Federation
VASILIEV V.I. – Doctor of Medical Sciences, Professor, rheumatologist,
Moscow.
Phone: +7-926-246-50-15.
I. V. Gaiduk
Moscow State Medical University named after A.I.Evdokimov
Russian Federation
Russian Federation
GAIDUK I.V. – Candidate of Medical Sciences, Associate Professor of the Department of Oral Mucosa,
Moscow.
Phone: +7-916-118-74-76.
S. G. Palshina
LLC "Revmotsentr"
Russian Federation
Russian Federation
PALSHINA S.G. – Ph.D., rheumatologist,
Moscow.
Phone: +7-919-776-59-61.
N. S. Shornikova
Revmotsentr LLC
Russian Federation
Russian Federation
SHORNIKOVA N.S. – Ph.D., rheumatologist, head,
Moscow.
Phone: +7-910- 446-56-69.
References
1. Lee L.I., Pawar R.R., Whitley S., Makdissi J. Incidence of different causes of benign obstruction of the salivary glands: retrospective analysis of 493 cases using fluoroscopy and subtraction sialography. Br.J.Oral. Maxillofac Surg. 2015, 53: 54 – 57. https://doi.org/10.1016/j.bjoms.2014.09.017.
2. Afanas’ev V.V. Sljunnye zhelezy. Bolezni i travmy. Rukovodstvo dlja vrachej. «GJeOTAR-Media», Moskva. 2012: 1 – 295.
3. Panin A.M. Hirurgicheskaja stomatologija. Vospalitel’nye i distroficheskie zabolevanija sljunnyh zheljoz. Uchebnoe posobie. (pod red. A. M. Panina). «Litterra». Moskva. 2020: 1 – 236. ISBN 978-5-4235-0354-3.
4. Zhu W.X., Chen Y., Liu D.G., Yu G.Y. Eosinophilic Sialodochitis: A Type of Chronic Obstructive Sialoadenitis Related to Allergy. Laryngoscope. 2021; 131 (3): 800 – 806. doi: 10.1002/lary.28772.
5. Dinca O.M., Didilesku A.C., Vladan G.C., Bucur M.B., Padurariu L.C., Bucur A. Eosinophilic sialodochitis: a case report of a rare disease. Rom.J.Morphol.Embryol. 2021; 62 (2): 621 – 624. doi:10.47162/RJME.62.2.32.
6. Kussmaul I. Reccurent salivary gland tumor due to chronic fibrotic inflammation of Stensen’s ducts [Anfallsweise auftertende Speichelgeschwulst in Folge von chronischer eitrig-fibrinoeser Entzundung des Stenonschen Ganges. Berlin Klin.Wschr. 1879; 15: 209 – 211.
7. Waldbott G., Shea J. Allergic parotitis. J.Allergy. 1947; 18 (1): 51 –54. doi: 10.1016/0021-8707(47)90067-1.
8. Ogawa T., Yoshitaki R., Lida S., Takigami K., Mori M., Yamaguchi Y., Tsuda Y. Two cases of sialodochitis fibrinosa (Kussmaul’s disease). Japanese Journal of Oral and Maxillofacial Surgery.1995; 41 (6): 549 – 551.
9. Darling M.R., Phillips V.M., Erasmus J.H. Bilateral submandibular salivary gland swelling-A report of chronic sialodochitis with eosinophilia. SADJ; 57 (3): 104 – 106.
10. Frati F., Boccardo R., Scurati S., Gelardi M., Incorvaia C. Idiopatic eosinophilic parotitis in an eight yearold boy. A case report. Journal of Medical Case Report. 2011; 5: 385. doi: 10.1186/1752-1947-5-385.
11. Baer A.N., Okuhama A., Eisele D.W., Tversky J.R., Gniadek T.J. Eosinophilic sialodochitis: redefinition of “allergic parotitis” and “sialodochitis fibrinosa”. Oral Dis. 2017; 23 (7): 840 – 848. doi: 10.1111/odi.12595.
12. Robles B.J.F., Avarez B.B., Sanchinel A.A.S., Andrus R.F., Malpartida M.E., Giraldes C.R., Verdejo F.L., Argumanez C.M., Pimiento J.A.P., Menendes C.B., Alcazar L.F.V., Sanchez J.L.A., Palop M.J., Tundidor H.G., Esteban J.C., Sanz J.S., Mateos C.B., Zaragoza C.M.I., Mendoza J.B.M. Sialodochitis fibrinosa (kussmaul disease) report of 3 cases and literature review. Medicine 2016; 95 (42): 1 – 5. doi: 10.1097/MD.0000000000005132.
13. Carey B., O’Neill N., Drown J., Escudier M., Yullah E., Beneng K., Odel E., Thomas B., Haque R. Eosinophilic sialodochitis: an emerging atopic condition. Oral Dis. 2022: 28 (3): 648 – 656. doi: 10.1111/odi.13821.
14. Sano T., Miyta J., Sano A., Ono Y., Tanaka Y., Matsucuma S., Ueki S., Kawana A. Eosinophilic sialoadenitis in patient with severe asthma: a case report. Asia Pac.Allergy. 2021: 11 (3): 1 – 7. doi:10.5415/apallergy.2021.11.e29.
15. Gonzales O., Picado C., Arismandi E., Alobid I., Ramirez J., Valero A., Bobolea I. Eosinophilic Sialodochitis: A Rare Comorbidity of Severe Asthma. J Investig Allergol Clin Immunol. 2023; 33 (2): 139 – 140. doi:10.18176/jiaci.0817 .
16. Okuda M., Ogami Y., Unno T. Sialodochitis fibrinosa (Kussmaul). Jibi to Rinsho Otologia Fucuoka. 1975; 21: 635 – 639. Article in Japanese.
17. Fucuchi M., Miybe Y., Furutani C., Saga T., Moritoki Y., Yamada T., Weller P.F., Ueki S. How to detect eosinophil ETosis (EETosis) and extracellular traps. Allergol.Int. 2021; 70: 19 – 29. doi: 10.1016/j.alit.2020.10.002.
18. Ueki S., Konno Y., Takeda M., Morioki Y., Hirokawa M., Matsuwaki Y., Honda K., Ohta N., Yamamoto S., Takagi Y., Wada A., Weller P.F. Eosinophil extracellular trap cell death-derived DNA traps: their presence in secretions and functional attributes. J. Allergy Clin. Immunol. 2016; 137: 258 – 267. doi: 10.1016/j.jaci.2015.04.041.
19. Ueki S., Melo R.C., Chiran I., Spencer L.A., Dvorak A.M., Weller P.F. Eosinophil extracellular DNA trap cell death mediates lytic release of free secretion-competent eosinophil granules in humans. Blood. 2013; 121: 2074 –2083. doi: 10.1182/blood-2012-05-432088.
20. Ueki S., Tokunaga T., Melo RCN., Saito H., Honda K., Fucushi M., Konno Y., Takeda M., Yamamoto Y., Hirokawa M., Fujieda S., Spencer LA., Weller PF. Charcot-Leyden crystal formation is closely associated with eosinophil extracellular trap cell death. Blood. 2018; 132: 2183 – 2187. doi: 10.1182/blood-2018-04-842260.
21. Melo RCN., Wang H., Silva TR., Imoto Y., Fujieda S., Fukuchi M., Miybe Y., Hirokawa M., Ueki S., Weller PF. Galectin-10, the protein that forms Charcot-Leyden crystals, is not stored in granules but resides in the peripheral cytoplasm of human eosinophils. J.Leukoc.Biol. 2020; 108: 139 – 149. doi: 10.1002/JLB.3AB0220-311R.
22. Kawamura Y., Ikeda R., Hori T., Sasaki T., Miyabe Y., Fukuchi M., Sakamoto K., Ohta N., Kawase T., Katori Y., Ueki S. Sialodochitis fibrinosa: salivary duct obstruction by eosinophil extracellular traps? Oral. Dis. 2020; 26: 1459 – 1463. doi: 10.1111/odi.13434.
23. Romachjova I.F., Judin L.A., Afanas’ev V.V., Morozov A.N. Zabolevanija i povrezhdenija sljunnyh zheljoz. Moskva. «Medicina». 1987: 1 –237.
24. Marx R.E., Stern D. Oral and Maxillofacial Pathology: A Rationale for Diagnosis and Treatment. Second Edition, Volume I, II. Quintessence Publishing Co, Inc 2012: 1 – 980. ISBN 978-0-86715-512-9.
25. Myers E.N., Ferris R.L. Editors. Salyvary Gland Disoders. 2007: 1 –517. Springer. ISBN 978-3-540-47070-0.Springer Berlin Heidelberg New York.
26. Safonova T.N., Vasil’ev V.I., Lihvanceva V.G. Sindrom Shjogrena. Rukovodstvo dlja vrachej. Izdatel’stvo Moskovskogo Universiteta. 2013: 1 – 597. ISBN: 978-5-19-010836-1.
27. Vasil’ev V.I., Sokol E.V., Rodionova E.B., Pal’shina S.G., Aleksandrova E.N., RadenskaLopovok S.G., Probatova N.A., Kokosadze N.V., Kovrigina A.M., Safonova T.N., Borovskaja A.B., Gajduk I.V. Svjazannye s IgG-4 porazhenija sljunnyh zheljoz. Ter.arh. 2015; 87 (8): 92 – 102. https://doi.org/10.17116/terarkh201587892-102
28. Vasil’ev V.I., Logvinenko O.A., Simonova M.V., Safonova T.N. Razvitie suhogo sindroma pri sarkoidoze s porazheniem sljunnyh i sljoznyh zheljoz. Terapevticheskij arhiv. 2005; 1: 62 – 67.
29. Vasil’ev V.I., Gajduk I.V., Pal’shina S.G., Gorodeckij V.R., Sokol E.V., Rodionova E.B., Burceva M.V., Shornikova N.S., Probatova N.N., Kokosadze N.V., Pavlovskaja A.I., Kupryshina N.A., Safonova T.N. Pervichnye onkogematologicheskie zabolevanija, debjutirujushhie s porazhenija bol’shih sljunnyh zheljoz v revmatologicheskoj praktike. Sovremennaja revmatologija. 2019; 13 (1): 44 – 51. https://cyberleninka.ru/article/n/pervichnye-onkogematologicheskie-zabolevaniya-debyutiruyuschie-s-porazheniya-bolshih-slyunnyhzhelez-v-revmatologicheskoy-praktike
30. Сhicamatsu K., Shino M., Fukudo Y., Sakakura K., Furuya N. Reccuring bilateral parotid gland swelling two cases of sialodochitis fibrinosa. J.Laryngol.Otol. 2006; 120: 330 – 333. doi: 10.1017/S0022215106000296.
31. Hayashi K., Onda T., Ochara H. et al. Case of suspected sialodochitis fibrinosa. Bull. Tokyo Dent Coll. 2016; 57: 91 – 96. doi: 10.2209/tdcpublication.2015-0028.
Review
For citations:
Vasiliev V.I., Gaiduk I.V., Palshina S.G., Shornikova N.S. EOSINOPHILIC SIALODOCHITIS: DESCRIPTION OF TWO CASES AND LITERATURE REVIEW. Vestnik of North-Eastern Federal University. Medical Sciences. 2024;(2):17-29. (In Russ.) https://doi.org/10.25587/2587-5590-2024-2-17-29
Views:
163
Email this article 