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Testicular vasculitis in eosinophilic granulomatosis with polyangiitis: a case-based review

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Abstract

Testicular vasculitis (TV) develops when an organ is involved in systemic vasculitis. A 47-year-old man with eosinophilic granulomatosis with polyangiitis (EGPA) developed TV as the first clinical episode. The patient had bronchial asthma for 8 years and developed left testicular pain before developing arthralgia, abdominal involvement, and sensory polyneuropathy, which led to the diagnosis of EGPA. The induration of the affected testicle persisted even after initiating immunosuppressive therapy with corticosteroids and cyclophosphamide, raising concern for testicular neoplasm, while testicular pain and other symptoms resolved. The patient underwent inguinal orchiectomy, and a histology examination of the resected testicle revealed fibrinoid necrotizing vasculitis. Only three cases of biopsy-proven TV in patients with EGPA have been reported in our review of published English-language articles. Two of the three patients in the reviewed cases developed TV before being diagnosed with EGPA. Moreover, all patients underwent extirpation of the affected testicle, leading to a pathological diagnosis of TV. This report suggests that TV may develop and be the presenting condition in EGPA, although urogenital involvement is rare.

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References

  1. Jennette JC, Falk RJ, Bacon PA, Basu N, Cid MC, Ferrario F et al (2013) 2012 Revised International Chapel Hill Consensus Conference Nomenclature of Vasculitides. Arthritis Rheum 65:1–11. https://doi.org/10.1002/art.37715

    Article  Google Scholar 

  2. Furuta S, Iwamoto T, Nakajima H (2019) Update on eosinophilic granulomatosis with polyangiitis. Allergol Int 68:430–436. https://doi.org/10.1016/j.alit.2019.06.004

    Article  Google Scholar 

  3. Grayson PC, Ponte C, Suppiah R, Robson JC, Craven A, Judge A et al (2022) 2022 American College of Rheumatology/European Alliance of Associations for Rheumatology classification criteria for eosinophilic granulomatosis with polyangiitis. Ann Rheum Dis 81:309–314. https://doi.org/10.1136/annrheumdis-2021-221794

    Article  Google Scholar 

  4. Masi AT, Hunder GG, Lie JT, Michel BA, Bloch DA, Arend WP et al (1990) The American College of Rheumatology 1990 criteria for the classification of Churg-Strauss syndrome (allergic granulomatosis and angiitis). Arthritis Rheum 33:1094–1100. https://doi.org/10.1002/art.1780330806

    Article  Google Scholar 

  5. DaJusta DG, Mueller TJ, Ioffreda R, Deen M, Barone JA (2009) Churg-Strauss syndrome mimicking a testicular mass. Curr Urol 3:210–212. https://doi.org/10.1159/000253392

    Article  Google Scholar 

  6. Fukui S, Iwamoto N, Mochizuki Y, Nakajima H, Abe K, Kawakami A (2017) Eosinophilic granulomatosis with polyangiitis along with funiculitis and myelitis. Int J Rheum Dis 20:2203–2205. https://doi.org/10.1111/1756-185x.12876

    Article  Google Scholar 

  7. Li J, Yan M, Qin J, Ren L, Wen R (2020) Testicular infarction and pulmonary embolism secondary to nonasthmatic eosinophilic granulomatosis with polyangiitis: a case report. J Investig Allergol Clin Immunol 30:380–381. https://doi.org/10.18176/jiaci.0566

    Article  Google Scholar 

  8. Benzi TC, Logsdon NT, Sampaio FJB, Favorito LA (2022) Testicular arteries anatomy applied to Fowler-Sthephens surgery in high undescended testis - a narrative review. Int Braz J Urol 48:8–17. https://doi.org/10.1590/s1677-5538.Ibju.2021.99.11

    Article  Google Scholar 

  9. Hernández-Rodríguez J, Molloy ES, Hoffman GS (2008) Single-organ vasculitis. Curr Opin Rheumatol 20:40–46. https://doi.org/10.1097/BOR.0b013e3282f176a0

    Article  Google Scholar 

  10. Hernández-Rodríguez J, Tan CD, Koening CL, Khasnis A, Rodríguez ER, Hoffman GS (2012) Testicular vasculitis: findings differentiating isolated disease from systemic disease in 72 patients. Medicine (Baltimore) 91:75–85. https://doi.org/10.1097/MD.0b013e31824156a7

    Article  Google Scholar 

  11. Silva CA, Cocuzza M, Borba EF, Bonfá E (2012) Cutting-edge issues in autoimmune orchitis. Clin Rev Allergy Immunol 42:256–263. https://doi.org/10.1007/s12016-011-8281-z

    Article  Google Scholar 

  12. Atis G, Memis OF, Güngör HS, Arikan O, Saglican Y, Caskurlu T (2010) Testicular polyarteritis nodosa mimicking testicular neoplasm. Sci World J 10:1915–1918. https://doi.org/10.1100/tsw.2010.194

    Article  Google Scholar 

  13. Bhatia S, Herrera Hernandez LP, Kamboj AK, Rieck KM (2018) Isolated polyarteritis nodosa presenting as bilateral testicular swelling. Am J Med 131:e55–e56. https://doi.org/10.1016/j.amjmed.2017.08.040

    Article  Google Scholar 

  14. Meeuwissen J, Maertens J, Verbeken E, Blockmans D (2008) Case reports: testicular pain as a manifestation of polyarteritis nodosa. Clin Rheumatol 27:1463–1466. https://doi.org/10.1007/s10067-008-0970-5

    Article  Google Scholar 

  15. Dotan ZA, Laufer M, Heldenberg E, Langevitz P, Fridman E, Duvdevani M et al (2003) Isolated testicular polyarteritis nodosa mimicking testicular neoplasm-long-term follow-up. Urology 62:352. https://doi.org/10.1016/s0090-4295(03)00388-1

    Article  Google Scholar 

  16. Pannek J, Haupt G (1997) Orchitis due to vasculitis in autoimmune diseases. Scand J Rheumatol 26:151–154. https://doi.org/10.3109/03009749709065674

    Article  Google Scholar 

  17. Davenport A, Downey SE, Goel S, Maciver AG (1996) Wegener’s granulomatosis involving the urogenital tract. Br J Urol 78:354–357. https://doi.org/10.1046/j.1464-410x.1996.00166.x

    Article  Google Scholar 

  18. Agraharkar M, Gokhale S, Gupta R (2002) Wegener’s granulomatosis diagnosed by testicular biopsy. Int Urol Nephrol 34:559–564. https://doi.org/10.1023/a:1025642217482

    Article  Google Scholar 

  19. Kechida M, Ktari K, Jellazi M, Mesfar R, Khochtali I, Saad H et al (2022) Simultaneous testicle and epididymis vasculitis revealing granulomatosis with polyangiitis. Clin Case Rep 10:e6231. https://doi.org/10.1002/ccr3.6231

    Article  Google Scholar 

  20. Lee SS, Tang SH, Sun GH, Yu CP, Jin JS, Chang SY (2006) Limited Wegener’s granulomatosis of the epididymis and testis. Asian J Androl 8:737–739. https://doi.org/10.1111/j.1745-7262.2006.00207.x

    Article  Google Scholar 

  21. Alba MA, Moreno-Palacios J, Beça S, Cid MC (2015) Urologic and male genital manifestations of granulomatosis with polyangiitis. Autoimmun Rev 14:897–902. https://doi.org/10.1016/j.autrev.2015.05.012

    Article  Google Scholar 

  22. Suo L, Perez LC, Finch CJ (2019) Testicular granulomatous vasculitis mimicking testicular torsion in an anti-neutrophil cytoplasmic antibody-associated vasculitis patient. SAGE Open Med Case Rep 7:2050313x18823451. https://doi.org/10.1177/2050313x18823451

    Article  Google Scholar 

  23. Sundaram S, Smith DH (2001) Giant cell arteritis mimicking a testicular tumour. Rheumatol Int 20:215–216. https://doi.org/10.1007/s002960100113

    Article  Google Scholar 

  24. Ha TS, Lee JS (2007) Scrotal involvement in childhood Henoch-Schönlein purpura. Acta Paediatr 96:552–555. https://doi.org/10.1111/j.1651-2227.2006.00173.x

    Article  Google Scholar 

  25. Güneş M, Kaya C, Koca O, Keles MO, Karaman MI (2012) Acute scrotum in Henoch-Schönlein purpura: fact or fiction? Turk J Pediatr 54:194–197

    Google Scholar 

  26. Lenert P, Icardi M, Dahmoush L (2013) ANA (+) ANCA (+) systemic vasculitis associated with the use of minocycline: case-based review. Clin Rheumatol 32:1099–1106. https://doi.org/10.1007/s10067-013-2245-z

    Article  Google Scholar 

  27. Louthrenoo W, Norasetthada A, Khunamornpong S, Sreshthaputra A, Sukitawut W (1999) Childhood Churg-Strauss syndrome. J Rheumatol 26:1387–1393

    Google Scholar 

  28. George GH, Axford JS (1999) Asthma and epididymitis: the calm before the storm. Ann Rheum Dis 58:731–736. https://doi.org/10.1136/ard.58.12.731

    Article  Google Scholar 

  29. Kanzawa Y, Imai Y, Mizuno Y, Nishioka H (2017) Testicular lymphocytic vasculitis treated with prednisolone and azathioprine. Mod Rheumatol 27:705–707. https://doi.org/10.3109/14397595.2015.1026020

    Article  Google Scholar 

  30. Marth D, Scheidegger J, Studer UE (1990) Ultrasonography of testicular tumors. Urol Int 45:237–240. https://doi.org/10.1159/000281715

    Article  Google Scholar 

  31. Schultz-Lampel D, Bogaert G, Thüroff JW, Schlegel E, Cramer B (1991) MRI for evaluation of scrotal pathology. Urol Res 19:289–292. https://doi.org/10.1007/bf00299060

    Article  Google Scholar 

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Acknowledgements

We would like to thank Editage (www.editage.com) for English-language editing.

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Authors and Affiliations

  1. Department of Medicine (Neurology and Rheumatology), Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto, 390-8621, Japan

    Takanori Ichikawa, Yasuhiro Shimojima, Shun Nomura, Dai Kishida, Jun Tanimura & Yoshiki Sekijima

  2. Department of Urology, Shinshu University School of Medicine, 3-1-1 Asahi, Matsumoto, 390-8621, Japan

    Masashi Shiozaki

  3. Department of Neurology, Aizawa Hospital, 2-5-1 Honjo, Matsumoto, 390-8510, Japan

    Jun Tanimura

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  1. Takanori Ichikawa
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Contributions

All the authors participated in the treatment of this patient and developed an argument for this case. T-I and Y-Shi reviewed the literature and prepared the draft of this manuscript. Y-Shi contributed to revision of the manuscript. All authors have revised and approved the final manuscript.

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Correspondence to Yasuhiro Shimojima.

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Ichikawa, T., Shimojima, Y., Nomura, S. et al. Testicular vasculitis in eosinophilic granulomatosis with polyangiitis: a case-based review. Clin Rheumatol 42, 293–299 (2023). https://doi.org/10.1007/s10067-022-06453-z

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