Abstract
An 82-year-old woman was previously diagnosed with cholecystitis and treated with antibiotics at another hospital. Because her fever and inflammation persisted, therapeutic cholecystectomy was performed. Histopathology of the gallbladder revealed periarterial vasculitis. After transfer to our hospital, an elevated titer of myeloperoxidase–antineutrophil cytoplasmic antibody (MPO–ANCA) was observed (47 U/mL). The patient’s renal dysfunction had previously been thought to be sequelae of her cholecystectomy. We diagnosed microscopic polyangiitis (MPA) and began treatment with 40 mg orally of prednisolone daily. The titer of MPO–ANCA decreased with the treatment, but fever recurred with prednisolone taper. We, therefore, added 50 mg orally of mizoribine (MZR) daily as an immunosuppressant and increased the MZR to 100 mg daily while monitoring its blood peak concentration. The peak level of MZR was 1.58 μg/mL at 6 h after administration. After adding MZR, we successfully tapered the orally dosed prednisolone without recurrent fever or complications. We describe this case of MPA in an elderly patient manifesting predominantly with cholecystitis and successfully treated with orally dosed prednisolone and MZR.
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References
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):115–28. doi:00005792-200503000-00006.
Ichinose K, Origuchi T, Kawashiri SY, Iwamoto N, Fujikawa K, Aramaki T, Kamachi M, Arima K, Tamai M, Nakamura H, Ida H, Kawakami A, Tsukada T, Ueki Y, Eguchi K. Efficacy and safety of mizoribine by one single dose administration for patients with rheumatoid arthritis. Intern Med. 2010;49(20):2211–8. doi:JST.JSTAGE/internalmedicine/49.3810.
Sonda K, Takahashi K, Tanabe K, Funchinoue S, Hayasaka Y, Kawaguchi H, Teraoka S, Toma H, Ota K. Clinical pharmacokinetic study of mizoribine in renal transplantation patients. Transpl Proc. 1996;28(6):3643–8.
Nishioka Y, Horita Y, Tadokoro M, Taura K, Suyama N, Miyazaki M, Harada T, Kohno S. Mizoribine induces remission of relapsed ANCA-associated renal vasculitis. Nephrol Dial Transplant. 2006;21(4):1087–8. doi:gfi29510.1093/ndt/gfi295.
LiVolsi VA, Perzin KH, Porter M. Polyarteritis nodosa of the gallbladder, presenting as acute cholecystitis. Gastroenterology. 1973;65(1):115–23.
Chen KT. Gallbladder vasculitis. J Clin Gastroenterol. 1989;11(5):537–40.
Newbold KM, Allum WH, Downing R, Symmons DP, Oates GD. Vasculitis of the gall bladder in rheumatoid arthritis and systemic lupus erythematosus. Clin Rheumatol. 1987;6(2):287–9.
Hashimoto A, Matsushita R, Iizuka N, Kimura M, Matsui T, Tanaka S, Ishikawa A, Endo H, Hirohata S. Henoch–Schonlein purpura complicated by perforation of the gallbladder. Rheumatol Int. 2009;29(4):441–3. doi:10.1007/s00296-008-0727-0.
Papaioannou CC, Hunder GG, Lie JT. Vasculitis of the gallbladder in a 70-year-old man with giant cell (temporal) arteritis. J Rheumatol. 1979;6(1):71–6.
Manoharan S, Muir J. Gallbladder vasculitis associated with cutaneous leucocytoclastic vasculitis. Australas J Dermatol. 2004;45(4):216–9. doi:AJD10010.1111/j.1440-0960.2004.00100.x.
Rajvanshi P, Atac BS, Seno R, Gupta S. Gallbladder vasculitis associated with type-1 cryoglobulinemia. Dig Dis Sci. 2001;46(2):296–300.
Tatsukawa H, Nagano S, Umeno Y, Oribe M. Churg–strauss syndrome with cholecystitis and renal involvement. Intern Med. 2003;42(9):893–6.
Goritsas CP, Repanti M, Papadaki E, Lazarou N, Andonopoulos AP. Intrahepatic bile duct injury and nodular regenerative hyperplasia of the liver in a patient with polyarteritis nodosa. J Hepatol. 1997;26(3):727–30. doi:S0168-8278(97)80441-2.
Juliano J, Wilson KD, Gertner E. Vasculitis of the gallbladder: case report and spectrum of disease. J Clin Rheumatol. 2009;15(2):75–7. doi:10.1097/RHU.0b013e318194b29900124743-200903000-00008.
Ribi C, Cohen P, Pagnoux C, Mahr A, Arene JP, Puechal X, Carli P, Kyndt X, Le Hello C, Letellier P, Cordier JF, Guillevin L. Treatment of polyarteritis nodosa and microscopic polyangiitis without poor-prognosis factors: a prospective randomized study of one hundred twenty-four patients. Arthritis Rheum. 2010;62(4):1186–97. doi:10.1002/art.27340.
Aihara Y, Miyamae T, Ito S, Kobayashi S, Imagawa T, Mori M, Ibe M, Mitsuda T, Yokota S. Mizoribine as an effective combined maintenance therapy with prednisolone in child-onset systemic lupus erythematosus. Pediatr Int. 2002;44(2):199–204.
Tanaka H, Suzuki K, Nakahata T, Tsugawa K, Ito E, Waga S. Mizoribine oral pulse therapy for patients with disease flare of lupus nephritis. Clin Nephrol. 2003;60(6):390–4.
Canada R, Chaudry S, Gaber L, Waters B, Martinez A, Wall B. Polyarteritis nodosa and cryoglobulinemic glomerulonephritis related to chronic hepatitis C. Am J Med Sci. 2006;331(6):329–33. doi:00000441-200606000-00008.
Hegarty J, Chiu DY, Middleton RJ, Haeney MM, Newstead CG, Bruce IN, Kalra PA, O’Donoghue DJ. Systemic lupus erythematosus, eosinophilic vasculitis and acalculous cholecystitis. Nephrol Dial Transpl. 2006;21(3):792–5. doi:gfi29910.1093/ndt/gfi299.
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This work was supported by a Grant-in-Aid for Scientific Research (to K.I.; No. 24790999).
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K. Ichinose and N. Iwanaga contributed equally to this work.
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Ichinose, K., Iwanaga, N., Okada, A. et al. A case of microscopic polyangiitis in an elderly patient presenting predominantly with cholecystitis successfully treated with mizoribine. Mod Rheumatol (2013). https://doi.org/10.1007/s10165-013-0841-z
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DOI: https://doi.org/10.1007/s10165-013-0841-z