Background: IgG4-related disease is a rare autoimmune condition that presents with lymphoplasmacytic infiltrate and fibrosis in the organ affected. Isolated testicle involvement is uncommon and there are only a few cases reported in the literature. CasePresentation: We report a case of isolated chronic orchitis due to IgG4 hypersecretion in a 61-year-old patient that evolved with asymptomatic tumor-like mass growth and was treated with left orchiectomy. Histopathological study revealed orchitis related to IgG4 hypersecretion disease. Conclusion: IgG4-related disease can be manifested as a multi or single-organ disorder. Most diagnoses are made after surgery with histopathological analysis. Most of the cases in literature stand out the difficulty in diagnosis and necessity of high suspicion due to this condition’s similarity with neoplasm presentation.
References
[1]
Kamisawa, T., Zen, Y., Pillai, S. and Stone, J. (2015) IgG4-Related Disease. Lancet, 385, 1460-1471. https://doi.org/10.1016/S0140-6736(14)60720-0
[2]
Mahajan, V., Mattoo, H., Deshpande, V., Pillai, S. and Stone, J. (2014) IgG4-Related Disease. Annual Review of Pathology, 9, 315-347. https://doi.org/10.1146/annurev-pathol-012513-104708
[3]
Stone, J., Zen, Y. and Deshpande, V. (2012) IgG4-Related Disease. The New England Journal of Medicine, 366, 539-551. https://doi.org/10.1056/NEJMra1104650
[4]
Lal, J., Bhat, S., Doddamani, S. and Devi, L. (2016) Isolated Testicular Immunoglobulin G4-Related Disease: A Mimicker of Malignancy. Indian Journal of Urology, 32, 326-328. https://doi.org/10.4103/0970-1591.191273
[5]
Shams, A., DAS, A., Sinha, M., et al. (2021) IgG4-Related Disease with Selective Testicular Involvement—A Rare Entity: Case Report with Review of Literature. Turkish Journal of Pathology, 37, 78-83. https://doi.org/10.5146/tjpath.2020.01493
[6]
de Buy Wenniger, L., Scheltema, J., Verheij, J. and Beuers, U. (2013) Testicular Inflammation as a New Manifestation of IgG4-Associated Disease. Urology, 82, e15-e16. https://doi.org/10.1016/j.urology.2013.04.046
[7]
Zen, Y. and Nakanuma, Y. (2010) IgG4-Related Disease: A Cross-Sectional Study of 114 Cases. The American Journal of Surgical Pathology, 34, 1812-1819. https://doi.org/10.1097/PAS.0b013e3181f7266b
[8]
Opriţă, R., Opriţă, B., Berceanu, D. and Diaconescu, I. (2017) Overview of IgG4-Related Disease. Journal of Medicine and Life, 10, 203-207.
[9]
Lang, D., Zwerina, J. and Pieringer, H. (2016) IgG4-Related Disease: Current Challenges and Future Prospects. Therapeutics and Clinical Risk Management, 12, 189-199. https://doi.org/10.2147/TCRM.S99985
[10]
Karram, S., Kao, C., Osunkoya, A., Ulbright, T. and Epstein, J. (2014) Idiopathic Granulomatous Orchitis: Morphology and Evaluation of Its Relationship to IgG4 Related Disease. Human Pathology, 45, 844-850. https://doi.org/10.1016/j.humpath.2013.12.003
[11]
Deshpande, V., Zen, Y., Chan, J., et al. (2012) Consensus Statement on the Pathology of IgG4-Related Disease. Modern Pathology, 25, 1181-1192.
[12]
Migita, K., Miyashita, T., Mizuno, A., et al. (2014) IgG4-Related Epididymo-Orchitis Associated with Bladder Cancer: Possible Involvement of BAFF/BAFF-R Interaction in IgG4-Related Urogenital Disease. Modern Rheumatology, 24, 188-194. https://doi.org/10.3109/14397595.2013.852841
[13]
Khosroshahi, A., Wallace, Z., Crowe, J., et al. (2015) Second International Symposium on IgG4-Related Disease. International Consensus Guidance Statement on the Management and Treatment of IgG4-Related Disease. Arthritis & Rheumatology, 67, 1688-1699. https://doi.org/10.1002/art.39132
[14]
Roy, S., Hooda, S. and Parwani, A. (2011) Idiopathic Granulomatous Orchitis. Pathology—Research and Practice, 207, 275-278. https://doi.org/10.1016/j.prp.2011.02.005