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Published in: BMC Nephrology 1/2023

Open Access 01-12-2023 | Kidney Transplantation | Case Report

Karyomegalic interstitial nephritis as a rare cause of kidney graft dysfunction: case report and review of literature

Authors: Fatma El-Husseiny Moustafa, Eman Nagy, Salwa Mahmoud Elwasif, Mohamed Sobh

Published in: BMC Nephrology | Issue 1/2023

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Abstract

Karyomegalic interstitial nephritis (KIN) is a rare cause of chronic interstitial nephritis characterized by enlarged renal tubular epithelial nuclei. The first case of KIN reported in a kidney graft was in 2019. Here, we report the first case of KIN in 2 brothers receiving kidneys from 2 different unrelated living donors. A male kidney transplant recipient with focal segmental glomerulosclerosis as the original kidney disease presented with graft impairment and proteinuria, and graft biopsy revealed KIN. This patient had a brother who was also a kidney transplant recipient and had one episode of graft impairment and was diagnosed with KIN as well.
Literature
1.
go back to reference Bennani Guebessi N, Karkouri M. Karyomegalic interstitial nephritis. CEN case reports. 2016;5(1):23–5.CrossRefPubMed Bennani Guebessi N, Karkouri M. Karyomegalic interstitial nephritis. CEN case reports. 2016;5(1):23–5.CrossRefPubMed
2.
go back to reference Bhandari S, Kalowski S, Collett P, Cooke BE, Kerr P, Newland R, et al. Karyomegalic nephropathy: an uncommon cause of progressive renal failure. Nephrol Dialysis Transplantation. 2002;17(11):1914–20.CrossRef Bhandari S, Kalowski S, Collett P, Cooke BE, Kerr P, Newland R, et al. Karyomegalic nephropathy: an uncommon cause of progressive renal failure. Nephrol Dialysis Transplantation. 2002;17(11):1914–20.CrossRef
3.
go back to reference Monga G, Banfi G, Salvadore M, Amatruda O, Bozzola C, Mazzucco G. Karyomegalic interstitial nephritis: report of 3 new cases and review of the literature. Clin Nephrol. 2006;65(5). Monga G, Banfi G, Salvadore M, Amatruda O, Bozzola C, Mazzucco G. Karyomegalic interstitial nephritis: report of 3 new cases and review of the literature. Clin Nephrol. 2006;65(5).
4.
go back to reference Burry A. Extreme dysplasla in renal epithelium of a young woman dying from hepatocarcinoma. J Pathol. 1974;113(3):147–50.CrossRefPubMed Burry A. Extreme dysplasla in renal epithelium of a young woman dying from hepatocarcinoma. J Pathol. 1974;113(3):147–50.CrossRefPubMed
5.
go back to reference Mihatsch M, Gudat F, Zollinger H, Heierli C, Thölen H, Reutter F. Systemic karyomegaly associated with chronic interstitial nephritis. A new disease entity? Clin Nephrol. 1979;12(2):54–62.PubMed Mihatsch M, Gudat F, Zollinger H, Heierli C, Thölen H, Reutter F. Systemic karyomegaly associated with chronic interstitial nephritis. A new disease entity? Clin Nephrol. 1979;12(2):54–62.PubMed
6.
go back to reference Ravindran A, Cortese C, Larsen CP, Wadei HM, Gandhi MJ, Cosio FG, et al. Karyomegalic interstitial nephritis in a renal allograft. Am J Transplant. 2019;19(1):285–90.CrossRefPubMed Ravindran A, Cortese C, Larsen CP, Wadei HM, Gandhi MJ, Cosio FG, et al. Karyomegalic interstitial nephritis in a renal allograft. Am J Transplant. 2019;19(1):285–90.CrossRefPubMed
7.
go back to reference Matsuura T, Wakino S, Yoshifuji A, Nakamura T, Tokuyama H, Hashiguchi A, et al. Improvement in karyomegalic interstitial nephritis three years after ifosfamide and cisplatin therapy by corticosteroid. CEN case reports. 2014;3(2):226–31.CrossRefPubMedPubMedCentral Matsuura T, Wakino S, Yoshifuji A, Nakamura T, Tokuyama H, Hashiguchi A, et al. Improvement in karyomegalic interstitial nephritis three years after ifosfamide and cisplatin therapy by corticosteroid. CEN case reports. 2014;3(2):226–31.CrossRefPubMedPubMedCentral
8.
go back to reference Isnard P, Rabant M, Labaye J, Antignac C, Knebelmann B, Zaidan M. Karyomegalic interstitial nephritis: a case report and review of the literature. Medicine. 2016;95(20). Isnard P, Rabant M, Labaye J, Antignac C, Knebelmann B, Zaidan M. Karyomegalic interstitial nephritis: a case report and review of the literature. Medicine. 2016;95(20).
9.
go back to reference Hassen W, Abid-Essafi S, Achour A, Guezzah N, Zakhama A, Ellouz F, et al. Karyomegaly of tubular kidney cells in human chronic interstitial nephropathy in Tunisia: respective role of Ochratoxin A and possible genetic predisposition. Hum Exp Toxicol. 2004;23(7):339–46.CrossRefPubMed Hassen W, Abid-Essafi S, Achour A, Guezzah N, Zakhama A, Ellouz F, et al. Karyomegaly of tubular kidney cells in human chronic interstitial nephropathy in Tunisia: respective role of Ochratoxin A and possible genetic predisposition. Hum Exp Toxicol. 2004;23(7):339–46.CrossRefPubMed
10.
go back to reference McCulloch T, Prayle A, Lunn A, Watson AR. Karyomegalic-like nephropathy, Ewing’s sarcoma and ifosfamide therapy. Pediatr Nephrol. 2011;26(7):1163–6.CrossRefPubMed McCulloch T, Prayle A, Lunn A, Watson AR. Karyomegalic-like nephropathy, Ewing’s sarcoma and ifosfamide therapy. Pediatr Nephrol. 2011;26(7):1163–6.CrossRefPubMed
11.
go back to reference Spoendlin M, Moch H, Brunner F, Brunner W, Burger H-R, Kiss D, et al. Karyomegalic interstitial nephritis: further support for a distinct entity and evidence for a genetic defect. Am J Kidney Dis. 1995;25(2):242–52.CrossRefPubMed Spoendlin M, Moch H, Brunner F, Brunner W, Burger H-R, Kiss D, et al. Karyomegalic interstitial nephritis: further support for a distinct entity and evidence for a genetic defect. Am J Kidney Dis. 1995;25(2):242–52.CrossRefPubMed
12.
go back to reference Zhou W, Otto EA, Cluckey A, Airik R, Hurd TW, Chaki M, et al. FAN1 mutations cause karyomegalic interstitial nephritis, linking chronic kidney failure to defective DNA damage repair. Nat Genet. 2012;44(8):910–5.CrossRefPubMedPubMedCentral Zhou W, Otto EA, Cluckey A, Airik R, Hurd TW, Chaki M, et al. FAN1 mutations cause karyomegalic interstitial nephritis, linking chronic kidney failure to defective DNA damage repair. Nat Genet. 2012;44(8):910–5.CrossRefPubMedPubMedCentral
13.
go back to reference Rejeb I, Jerbi M, Jilani H, Gaied H, Elaribi Y, Hizem S, et al. New familial cases of karyomegalic interstitial nephritis with mutations in the FAN1 gene. BMC Med Genom. 2021;14(1):1–8.CrossRef Rejeb I, Jerbi M, Jilani H, Gaied H, Elaribi Y, Hizem S, et al. New familial cases of karyomegalic interstitial nephritis with mutations in the FAN1 gene. BMC Med Genom. 2021;14(1):1–8.CrossRef
14.
go back to reference Murray SL, Connaughton DM, Fennelly NK, Kay EW, Dorman A, Doyle B, et al. Karyomegalic interstitial nephritis: cancer risk following transplantation. Nephron. 2020;144(1):49–54.CrossRefPubMed Murray SL, Connaughton DM, Fennelly NK, Kay EW, Dorman A, Doyle B, et al. Karyomegalic interstitial nephritis: cancer risk following transplantation. Nephron. 2020;144(1):49–54.CrossRefPubMed
15.
go back to reference De Chiara L, Conte C, Semeraro R, Diaz-Bulnes P, Angelotti ML, Mazzinghi B, et al. Tubular cell polyploidy protects from lethal acute kidney injury but promotes consequent chronic kidney disease. Nat Commun. 2022;13(1):5805.CrossRefPubMedPubMedCentral De Chiara L, Conte C, Semeraro R, Diaz-Bulnes P, Angelotti ML, Mazzinghi B, et al. Tubular cell polyploidy protects from lethal acute kidney injury but promotes consequent chronic kidney disease. Nat Commun. 2022;13(1):5805.CrossRefPubMedPubMedCentral
16.
go back to reference Airik M, Phua YL, Huynh AB, McCourt BT, Rush BM, Tan RJ, et al. Persistent DNA damage underlies tubular cell polyploidization and progression to chronic kidney disease in kidneys deficient in the DNA repair protein FAN1. Kidney Int. 2022;102(5):1042–56.CrossRefPubMed Airik M, Phua YL, Huynh AB, McCourt BT, Rush BM, Tan RJ, et al. Persistent DNA damage underlies tubular cell polyploidization and progression to chronic kidney disease in kidneys deficient in the DNA repair protein FAN1. Kidney Int. 2022;102(5):1042–56.CrossRefPubMed
17.
go back to reference De Chiara L, Romagnani P. Polyploid tubular cells and chronic kidney disease. Kidney Int. 2022;102(5):959–61.CrossRefPubMed De Chiara L, Romagnani P. Polyploid tubular cells and chronic kidney disease. Kidney Int. 2022;102(5):959–61.CrossRefPubMed
18.
go back to reference Radha S, Tameem A, Rao B. Karyomegalic interstitial nephritis with focal segmental glomerulosclerosis: a rare association. Indian J Nephrol. 2014;24(2):117.CrossRefPubMedPubMedCentral Radha S, Tameem A, Rao B. Karyomegalic interstitial nephritis with focal segmental glomerulosclerosis: a rare association. Indian J Nephrol. 2014;24(2):117.CrossRefPubMedPubMedCentral
19.
go back to reference Tagliente DJ, Voss JS, Peters SG, Aubry MC, Cornell LD, Maleszewski JJ. Systemic karyomegaly with primary pulmonary presentation. Hum Pathol. 2014;45(1):180–4.CrossRefPubMed Tagliente DJ, Voss JS, Peters SG, Aubry MC, Cornell LD, Maleszewski JJ. Systemic karyomegaly with primary pulmonary presentation. Hum Pathol. 2014;45(1):180–4.CrossRefPubMed
20.
go back to reference Akyürek LM, Hussein A, Nicholson AG, Mauritz N-J, Mölne J. Pulmonary manifestations of systemic karyomegaly. Respiratory Med Case Rep. 2020;29:101032. Akyürek LM, Hussein A, Nicholson AG, Mauritz N-J, Mölne J. Pulmonary manifestations of systemic karyomegaly. Respiratory Med Case Rep. 2020;29:101032.
21.
go back to reference Wang Z, Ni X, Zhu S, Yue S. IgA Nephropathy Concomitant with Karyomegalic interstitial nephritis. Am J Med Sci. 2020;360(3):287–92.CrossRefPubMed Wang Z, Ni X, Zhu S, Yue S. IgA Nephropathy Concomitant with Karyomegalic interstitial nephritis. Am J Med Sci. 2020;360(3):287–92.CrossRefPubMed
22.
go back to reference Chand MT, Zaka A, Qu H. Association of karyomegalic interstitial nephritis with focal segmental glomerulosclerosis. Autopsy and Case Reports. 2021;11. Chand MT, Zaka A, Qu H. Association of karyomegalic interstitial nephritis with focal segmental glomerulosclerosis. Autopsy and Case Reports. 2021;11.
23.
go back to reference Gast C, Pengelly RJ, Lyon M, Bunyan DJ, Seaby EG, Graham N, et al. Collagen (COL4A) mutations are the most frequent mutations underlying adult focal segmental glomerulosclerosis. Nephrol Dialysis Transplantation. 2016;31(6):961–70.CrossRef Gast C, Pengelly RJ, Lyon M, Bunyan DJ, Seaby EG, Graham N, et al. Collagen (COL4A) mutations are the most frequent mutations underlying adult focal segmental glomerulosclerosis. Nephrol Dialysis Transplantation. 2016;31(6):961–70.CrossRef
24.
go back to reference Abrahamson DR, Hudson BG, Stroganova L, Borza D-B, John PLS. Cellular origins of type IV collagen networks in developing glomeruli. J Am Soc Nephrol. 2009;20(7):1471–9.CrossRefPubMedPubMedCentral Abrahamson DR, Hudson BG, Stroganova L, Borza D-B, John PLS. Cellular origins of type IV collagen networks in developing glomeruli. J Am Soc Nephrol. 2009;20(7):1471–9.CrossRefPubMedPubMedCentral
25.
go back to reference Becker J, Hoerning A, Schmid K, Hoyer P. Immigrating progenitor cells contribute to human podocyte turnover. Kidney Int. 2007;72(12):1468–73.CrossRefPubMed Becker J, Hoerning A, Schmid K, Hoyer P. Immigrating progenitor cells contribute to human podocyte turnover. Kidney Int. 2007;72(12):1468–73.CrossRefPubMed
26.
go back to reference Katsuma A, Nakada Y, Yamamoto I, Horita S, Furusawa M, Unagami K, et al. Long-term survival in japanese renal transplant recipients with Alport syndrome: a retrospective study. BMC Nephrol. 2018;19(1):1–9.CrossRef Katsuma A, Nakada Y, Yamamoto I, Horita S, Furusawa M, Unagami K, et al. Long-term survival in japanese renal transplant recipients with Alport syndrome: a retrospective study. BMC Nephrol. 2018;19(1):1–9.CrossRef
27.
go back to reference Sugimoto H, Mundel TM, Sund M, Xie L, Cosgrove D, Kalluri R. Bone-marrow-derived stem cells repair basement membrane collagen defects and reverse genetic kidney disease. Proceedings of the National Academy of Sciences. 2006;103(19):7321-6. Sugimoto H, Mundel TM, Sund M, Xie L, Cosgrove D, Kalluri R. Bone-marrow-derived stem cells repair basement membrane collagen defects and reverse genetic kidney disease. Proceedings of the National Academy of Sciences. 2006;103(19):7321-6.
28.
go back to reference Prodromidi EI, Poulsom R, Jeffery R, Roufosse CA, Pollard PJ, Pusey CD, et al. Bone marrow-derived cells contribute to podocyte regeneration and amelioration of renal disease in a mouse model of Alport syndrome. Stem Cells. 2006;24(11):2448–55.CrossRefPubMed Prodromidi EI, Poulsom R, Jeffery R, Roufosse CA, Pollard PJ, Pusey CD, et al. Bone marrow-derived cells contribute to podocyte regeneration and amelioration of renal disease in a mouse model of Alport syndrome. Stem Cells. 2006;24(11):2448–55.CrossRefPubMed
Metadata
Title
Karyomegalic interstitial nephritis as a rare cause of kidney graft dysfunction: case report and review of literature
Authors
Fatma El-Husseiny Moustafa
Eman Nagy
Salwa Mahmoud Elwasif
Mohamed Sobh
Publication date
01-12-2023

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