ACUTE CHOLECYSTITIS CAUSED BY GRANULOMATOSIS WITH POLYANGIITIS - A RARE CASE

Vanja Petković; Sonja Golubović; Tatjana Ilić; Sandra Jelčić; Zorana Ostojić; Violeta Knežević

doi:10.5937/mckg59-55630

Vanja Petković Klinika za nefrologiju i kliničku imunologiju, Univerzitetski klinički centar Vojvodine, Novi Sad, Srbija https://orcid.org/0009-0001-2173-8640
Sonja Golubović Klinika za nefrologiju i kliničku imunologiju, Univerzitetski klinički centar Vojvodine, Novi Sad, Srbija https://orcid.org/0000-0003-3716-698X
Tatjana Ilić Klinika za nefrologiju i kliničku imunologiju, Univerzitetski klinički centar Vojvodine, Novi Sad, Srbija
Sandra Jelčić Klinika za medicinsku rehabilitaciju, Univerzitetski klinički centar Vojvodine, Novi Sad, Srbija; https://orcid.org/0009-0006-1766-2213
Zorana Ostojić Institut za javno zdravlje Vojvodine, Novi Sad, Srbija. https://orcid.org/0009-0002-0282-9499
Violeta Knežević Klinika za nefrologiju i kliničku imunologiju, Univerzitetski klinički centar Vojvodine, Novi Sad, Srbija

DOI: https://doi.org/10.5937/mckg59-55630

Keywords: ANCA vasculitis, žučna kesa, PR3

Abstract

The most commonly affected organs in Granulomatosis with polyangiitis (GPA) include kidneys and lungs, while presentation in other organ systems is rare. [1, 2, 3]. We hereby present a case of vasculitic changes in the gallbladder, as the initial manifestation of GPA. A 65-year-old woman presented with fever and generalized weakness and was admitted to hospital. Upon admission, physical examination revealed signs of dehydration, mild hypotension and tachycardia. There was no palpable tenderness in the abdomen, and Murphy’s sign was negative. Initial work-up revealed normocytic anemia, elevated markers of inflammation. Urinalysis revealed protein and red blood cells. Blood and urine cultures were negative, and no signs of inflammation were identified on radiographic imaging. Due to persisting fever and unresolved inflammatory process despite the usage of parenteral antibiotic treatment, a computed tomography (CT) was also performed and revealed calculous cholecystitis necessitating laparoscopic cholecystectomy. Histopathology of the resected gallbladder showed small-vessel vasculitis. Laboratory tests revealed positive proteinase-3-anti-neutrophil cytoplasmic antibodies (PR3-ANCA) with an onset of renal dysfunction. These results supported a diagnosis of granulomatosis with polyangiitis (GPA) and the patient was started with glucocorticoids and intravenous cyclophosphamide. After this treatment, the patient’s general condition stabilized, creatinine and C-reactive protein levels decreased, and anemia resolved as well. This case highlights the importance of considering vasculitis in patients with unexplained persistent fever and systemic symptoms, especially when common causes have been excluded.

References

References
1. Puéchal X. Granulomatosis with polyangiitis (Wegener's). Joint Bone Spine 2020;87(5):359-366.
2. Klapa S, Arnold S, Lamprecht P. Granulomatosis with polyangiitis and microscopic polyangiitis. LARYNGO RHINO OTOL 2024;103(07):490-499.
3. Greco A, Marinelli C, Fusconi M, Macri GF, Gallo A, De Virgilio A. et al. Clinic manifestations in granulomatosis with polyangiitis. Int J Immunopathol Pharmacol 2016;29(2):151-9.
4. Ross C, Makhzoum JP, Pagnoux C. Updates in ANCA-associated vasculitis. Eur J Rheumatol 2022;9(3):153-166.
5. Banerjee P, Jain A, Kumar U, Sabyasachi Senapati. Epidemiology and genetics of granulomatosis with polyangiitis. Rheumatol Int 2021;41:2069–2089.
6. Anderson E, Gakhar N, Stull C, Caplan L. Gastrointestinal and Hepatic Disease in Vasculitis. Rheum Dis Clin North Am 2018;44(1):1-14.
7. Bagai S, Sharma A, Gupta R, Kumar V, Rathi M, Kohli HS. et al. Ramachandran R. Gastrointestinal Involvement in Granulomatosis with Polyangiitis: Case Report and Review. Indian J Nephrol. 2019;29(6):415-418.
8. Pagnoux C, Mahr A, Cohen P, Guillevin L. Presentation and outcome of gastrointestinal involvement in systemic necrotizing vasculitides: analysis of 62 patients with polyarteritis nodosa, microscopic polyangiitis, Wegener granulomatosis, Churg-Strauss syndrome, or rheumatoid arthritis-associated vasculitis. Medicine (Baltimore). 2005;84(2):115-128.
9. Pérez-Macías JP, Rodarte-Shade M, Garza-García CA, Tueme-De la Peña, D. Rodríguez-Guerra ML. Granulomatosis with polyangiitis (Wegener’s granulomatosis) with gastrointestinal involvement: A case report. Rev Gastroenterol Mex. 2022;87(3):392-394.
10. Kitaguchi D, Kurata M, Shimomura O, Oda T, Ohkohchi N. Gallbladder bleeding associated with microscopic polyangiitis: a case report. J Surg Case Rep 2017;(8):159.
11. Hernández-Rodríguez J, Tan CD, Rodríguez ER, Hoffman GS, Hoffman GS. Single-organ gallbladder vasculitis: characterization and distinction from systemic vasculitis involving the gallbladder. An analysis of 61 patients. Medicine (Baltimore) 2014;93(24):405-413.
12. Juliano J, Wilson KD, Gertner E. Vasculitis of the gallbladder: case report and spectrum of disease. J Clin Rheumatol 2009;15(2):75-7.
13. Mohammad AJ, Jacobsson LT, Westman KW, Sturfelt G, Segelmark M, Incidence and survival rates in Wegener's granulomatosis, microscopic polyangiitis, Churg–Strauss syndrome and polyarteritis nodosa. Rheumatology 2009;48(12):1560-1565.
14. Ohta R, Ikeda H, Kubota S, Sano C. Acute Cholecystitis in an Elderly Patient With Antineutrophil Cytoplasmic Antibody-Associated Vasculitis: A Case Report. Cureus. 2022;14(2):21877.
15. Seth N, Tran PP. Cholecystitis: A Rare Presentation for Diffuse Eosinophilic Granulomatosis With Polyangiitis. ACG Case Rep J. 2023;10(4):01023.
16. Ohnuki Y, Moriya Y, Yutani S, Mizuma A, Nakayama T, Ohnuki Y. et al. Eosinophilic Granulomatosis with Polyangiitis (Churg-Strauss Syndrome) Complicated by Perforation of the Small Intestine and Cholecystitis. Intern Med 2018;57(5):737-740.
17. Ito H, Mishima Y, Cho T, Ogiwara N, Shinma Y, Yokota M. et al. Eosinophilic Cholecystitis Associated with Eosinophilic Granulomatosis with Polyangiitis. Case Rep Gastroenterol 2020;14(3):668-674.
18. Ichinose K, Iwanaga N, Okada A, Tamai M, Yamasaki S, Nakamura H. et al. A case of microscopic polyangiitis in an elderly patient presenting predominantly with cholecystitis successfully treated with mizoribine. Mod Rheumatol 2014;24(6):1011-4.
19. Ye L, Lu X, Xue J. Eosinophilic granulomatosis with polyangiitis complicated by cholecystitis: a case report and review of the literature. Clin Rheumatol. 2016;35(1):259-63.
20. Gallaher JR, Charles A. Acute Cholecystitis: A Review. JAMA 2022;327(10):965-975.
21. Kitching AR, Anders HJ, Basu N, Brouwer E, Gordon J, Jayne DR. et al. ANCA-associated vasculitis. Nat Rev Dis Primers 2020;6(1):71.
22. Bielejewska A, Bociek A, Bociek M, Jaroszyński A. A unique case of ovarian manifestation of systemic vasculitis that mimics ovarian cancer. Sarcoidosis Vasc Diffuse Lung Dis 2020;37(2):179-183.
23. Yaseen K, Mandell BF. ANCA associated vasculitis (AAV): a review for internists. Postgrad Med 2023;135(1):3-13.
24. Francescutti V, Ellis AK, Bourgeois JM, Ward C. Acute acalculous cholecystitis: an unusual presenting feature of Churg-Strauss vasculitis. Can J Surg 2008;51(6):E129-30.
25. Gorgun E, Ozmen V. Acalculous gangrenous cholecystitis in a young adult: a gastrointestinal manifestation of polyarteritis nodosa. Surg Laparosc Endosc Percutan Tech 2002;12(5):359-61.