Abstract
A 47-year-old man presented with severe hypokalemic paralysis and respiratory failure. A large amount of potassium was administered along with providing intensive care, and his condition improved. Hypokalemia was attributed to increased urinary potassium excretion. A kidney biopsy was performed to make a definitive histological diagnosis. It revealed acute tubulointerstitial nephritis (TIN). After the diagnosis, prednisolone was administered, and the TIN gradually improved. From the clinical course and laboratory findings, the TIN was presumed to be an autoimmune disorder. Further specific autoantibody tests were positive for anti-mitochondrial antibody (AMA), which has been gaining increasing attention in regard to TIN. In addition, all previous cases of TIN associated with AMA have affected females. The detailed pathogenetic mechanisms are as yet unclear and require further investigation.
This is a preview of subscription content, log in via an institution to check access.
Similar content being viewed by others
References
Schwarz A, Krause PH, Kunzendorf U, Keller F, Distler A. The outcome of acute interstitial nephritis: risk factors for the transition from acute to chronic interstitial nephritis. Clin Nephrol. 2000;54:179–90.
Takahashi N, Saeki T, Komatsuda A, Munemura C, Fukui T, Imai N, Homma N, Hatta T, Samejima KI, Fujimoto T, Omori H, Ito Y, Nishikawa Y, Kobayashi M, Morikawa Y, Fukushima S, Yokoi S, Mikami D, Kasuno K, Kimura H, Nemoto T, Nakamoto Y, Sada K, Sugai M, Naiki H, Yoshida H, Narita I, Saito Y, Iwano M. Tubulointerstitial nephritis with IgM-positive plasma cells. J Am Soc Nephrol. 2017;28:3688–98.
Saeki T, Nishi S, Imai N, Ito T, Yamazaki H, Kawano M, Yamamoto M, Takahashi H, Matsui S, Nakada S, Origuchi T, Hirabayashi A, Homma N, Tsubata Y, Takata T, Wada Y, Saito A, Fukase S, Ishioka K, Miyazaki K, Masaki Y, Umehara H, Sugai S, Narita I. Clinicopathological characteristics of patients with IgG4-related tubulointerstitial nephritis. Kidney Int. 2010;78:1016–23.
Mandeville JT, Levinson RD, Holland GN. The tubulointerstitial nephritis and uveitis syndrome. Surv Ophthalmol. 2001;46:195–208.
Ye X, Maras JE, Bakun PJ, Tucker KL. Dietary intake of vitamin B-6, plasma pyridoxal 5′-phosphate, and homocysteine in Puerto Rican adults. J Am Diet Assoc. 2010;110:1660–8.
Kunisawa J, Sugiura Y, Wake T, Nagatake T, Suzuki H, Nagasawa R, Shikata S, Honda K, Hashimoto E, Suzuki Y, Setou M, Suematsu M, Kiyono H. Mode of bioenergetic metabolism during B cell differentiation in the intestine determines the distinct requirement for vitamin B1. Cell Rep. 2015;13:122–31.
Saeki T, Kawano M. IgG4-related kidney disease. Kidney Int. 2014;85:251–7.
Takahashi N, Kimura H, Kawajiri Y, Mikami D, Yamamoto C, Kasuno K, Imai N, Kuroda T, Nishi S, Yamamoto M, Yoshida H. Tubulointerstitial nephritis with IgM-positive plasmacytoid large lymphocyte infiltration in a patient with primary biliary cirrhosis and sjögren’s syndrome. Clin Nephrol. 2010;74:74–80.
Lino M, Binaut R, Noël LH, Patey N, Rustin P, Daniel L, Serpaggi J, Varaut A, Vanhille P, Knebelmann B, Grünfeld JP, Fakhouri F. Tubulointerstitial nephritis and Fanconi syndrome in primary biliary cirrhosis. Am J Kidney Dis. 2005;46:e41-6.
Kodama T, Imai H, Wakui H, Ohtani H, Komatsuda A, Miura AB. Tubulointerstitial nephritis with renal tubular acidosis and asymptomatic primary biliary cirrhosis accompanied by antibody to a 52-kDa mitochondrial protein alone. Clin Nephrol. 1996;45:401–5.
Bando H, Hashimoto N, Hirota Y, Sakaguchi K, Hisa I, Inoue Y, Imanishi Y, Seino S, Kaji H. Severe hypophosphatemic osteomalacia with Fanconi syndrome, renal tubular acidosis, vitamin D deficiency and primary biliary cirrhosis. Intern Med. 2009;48:353–8.
Hara M, Miyazawa R, Takagi A, Kado H, Maki K, Sawada K, You K, Hatta T. Case of fanconi syndrome positive for anti-M2 antibodies revealed by severe hypokalemia and multiple bone fracture. Nihon Jinzo Gakkai Shi. 2011;53:719–25.
Saeki T, Nakajima A, Ito T, Takata T, Imai N, Yoshita K, Kabasawa H, Yamazaki H, Narita I. Tubulointerstitial nephritis and Fanconi syndrome in a patient with primary sjögren’s syndrome accompanied by antimitochondrial antibodies: a case report and review of the literature. Mod Rheumatol. 2016;4:1–4.
Macdougall IC, Isles CG, Whitworth JA, More IA, MacSween RN. Interstitial nephritis and primary biliary cirrhosis: a new association? Clin Nephrol. 1987;27:36–40.
Kamouchi M, Tsuji H, Hirakata H, Okamura K, Ishitsuka T, Murai K, Onoyama K, Fujishima M. Tubulointerstitial disorders in the kidney associated with primary biliary cirrhosis (PBC). Clin Nephrol. 1991;35:134–5.
Terrier B, Fakhouri F, Berezne A, Bouldouyre MA, Guilpain P, Sogni P, Terris B, Noël LH, Guillevin L, Mouthon L. Osteomalacia revealing celiac disease and primary biliary cirrhosis-related fanconi syndrome in a patient with systemic sclerosis. Clin Exp Rheumatol. 2008;26:467–70.
Komatsuda A, Wakui H, Ohtani H, Masai R, Okuyama S, Nimura T, Suzuki N, Sawada K. Tubulointerstitial nephritis and renal tubular acidosis of different types are rare but important complications of primary biliary cirrhosis. Nephrol Dial Transpl. 2010;25:3575–9.
Bansal T, Takou A, Khwaja A. Progressive chronic kidney disease secondary to tubulointerstitial nephritis in primary biliary cirrhosis. Clin Kidney J. 2012;5:442–4.
Iwakura T, Fujigaki Y, Matsuyama T, Fujikura T, Ohashi N, Yasuda H, Kato A, Baba S. Tubulointerstitial nephritis and primary biliary cirrhosis with a T cell-dominant profile of infiltrating cells and granulomas in both organs. Intern Med. 2013;52:467–71.
Rasolzadegan M-H, Bakhshayesh H, Amid N. Tubulointerstitial nephritis associated with primary biliary cirrhosis. J Nephropharmacol. 2014;3:29–31.
Yamaguchi S, Maruyama T, Wakino S, Tokuyama H, Hashiguchi A, Tada S, Homma K, Monkawa T, Thomas J, Miyashita K, Kurihara I, Yoshida T, Konishi K, Hayashi K, Hayashi M, Itoh H. A case of severe osteomalacia caused by Tubulointerstitial nephritis with Fanconi syndrome in asymptomotic primary biliary cirrhosis. BMC Nephrol. 2015;16:187.
Malmborg AC, Shultz DB, Luton F, Mostov KE, Richly E, Leung PS, Benson GD, Ansari AA, Coppel RL, Gershwin ME, Van de Water J. Penetration and co-localization in MDCK cell mitochondria of IgA derived from patients with primary biliary cirrhosis. J Autoimmun. 1998;11:573–80.
Acknowledgements
We are deeply grateful for the direction provided by Atsushi Takahashi M.D., Ph.D.; Tomoko Namba-Hamano M.D., Ph.D.; and Yoshitsugu Takabatake M.D., Ph.D. (Osaka University Hospital, Osaka, Japan).
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Conflict of interest
The authors have declared that no conflict of interest exists.
Ethical approval
All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional research committee at which the studies were conducted (IRB approval number 29–94) and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.
Informed consent
Informed consent was obtained from all individual participants included in the study.
Additional information
Publisher’s Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
This article has previously been submitted to another academic medical journal written in Japanese (Rinshotaieki, 2015; 42: 37–41.). However, the main message of this article has changed, and the current figures and tables have not been replicated in the initial Japanese version, except for the table of laboratory examination results.
About this article
Cite this article
Morita, M., Yamaguchi, Y., Masuyama, S. et al. Anti-mitochondria antibody-related tubulointerstitial nephritis accompanied by severe hypokalemic paralysis. CEN Case Rep 8, 119–124 (2019). https://doi.org/10.1007/s13730-019-00376-6
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s13730-019-00376-6