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Primary hyperoxaluria type 2 successfully treated with combined liver-kidney transplantation after failure of isolated kidney transplantation

2020; Elsevier BV; Volume: 20; Issue: 6 Linguagem: Inglês

10.1111/ajt.15829

1600-6143

Arnaud Del Bello, Olivier Cointault, Audrey Delas, Nassim Kamar,

Biomedical Research and Pathophysiology

American Journal of TransplantationVolume 20, Issue 6 p. 1752-1753 LETTER TO THE EDITORFree Access Primary hyperoxaluria type 2 successfully treated with combined liver-kidney transplantation after failure of isolated kidney transplantation Arnaud Del Bello, Corresponding Author Arnaud Del Bello delbello.a@chu-toulouse.fr orcid.org/0000-0003-3115-868X Department of Nephrology and Organ Transplantation, CHU Rangueil, Toulouse, France Correspondence Arnaud Del Bello Email: delbello.a@chu-toulouse.frSearch for more papers by this authorOlivier Cointault, Olivier Cointault Department of Nephrology and Organ Transplantation, CHU Rangueil, Toulouse, FranceSearch for more papers by this authorAudrey Delas, Audrey Delas Department of Pathology, Institut Universitaire du Cancer, CHU, Toulouse, FranceSearch for more papers by this authorNassim Kamar, Nassim Kamar Department of Nephrology and Organ Transplantation, CHU Rangueil, Toulouse, France Université Paul Sabatier, Toulouse, France INSERM U1043, IFR–BMT, CHU Purpan, Toulouse, FranceSearch for more papers by this author Arnaud Del Bello, Corresponding Author Arnaud Del Bello delbello.a@chu-toulouse.fr orcid.org/0000-0003-3115-868X Department of Nephrology and Organ Transplantation, CHU Rangueil, Toulouse, France Correspondence Arnaud Del Bello Email: delbello.a@chu-toulouse.frSearch for more papers by this authorOlivier Cointault, Olivier Cointault Department of Nephrology and Organ Transplantation, CHU Rangueil, Toulouse, FranceSearch for more papers by this authorAudrey Delas, Audrey Delas Department of Pathology, Institut Universitaire du Cancer, CHU, Toulouse, FranceSearch for more papers by this authorNassim Kamar, Nassim Kamar Department of Nephrology and Organ Transplantation, CHU Rangueil, Toulouse, France Université Paul Sabatier, Toulouse, France INSERM U1043, IFR–BMT, CHU Purpan, Toulouse, FranceSearch for more papers by this author First published: 24 February 2020 https://doi.org/10.1111/ajt.15829Citations: 5AboutSectionsPDF ToolsRequest permissionExport citationAdd to favoritesTrack citation ShareShare Give accessShare full text accessShare full-text accessPlease review our Terms and Conditions of Use and check box below to share full-text version of article.I have read and accept the Wiley Online Library Terms and Conditions of UseShareable LinkUse the link below to share a full-text version of this article with your friends and colleagues. Learn more.Copy URL Share a linkShare onFacebookTwitterLinked InRedditWechat To the Editor: Primary hyperoxaluria type 2 (PH2) is a very rare disease that usually has a less severe course than primary hyperoxaluria type 1. In patients who have PH2 with end-stage renal disease (ESRD), isolated kidney transplant is recommended.1 However, there are very few reported cases of kidney transplant in this setting, and the expert recommendations are based on low-level evidence.2 Similar to previous case reports,3, 4 we had recently reported a case of reoccurrence of oxalate nephropathy after isolated kidney transplant leading to early graft loss in a patient with PH2.5 A 41-year-old man had undergone a kidney transplant from a deceased donor for a PH2-related ESRD. He required daily dialysis for 3 days for hyperkaliemia and uremia. Biopsy-proven recurrence of kidney oxalate nephropathy was observed early after transplant, leading to graft loss at 6 months of posttransplant. The report of the success of combined liver-kidney transplant (CLKT) to prevent oxalate deposit by Dhondup et al3 prompted us to consider CLKT in our patient. Hence, 2 years after kidney allograft loss, the patient was offered a CLKT from a deceased donor. At transplant, preformed donor-specific anti–HLA class II antibodies were detected, and the complement-dependent cytoxicity crossmatch was positive. He was given antilymphocyte T globulins (lymphoglobulins 9 mg/kg at day 0), rituximab (375 mg/m2 on days 0 and 7), tacrolimus (target trough level at 7-10 ng/mL), mycophenolate mofetil (1 g/d), and low-dose steroids (5 mg/d). After transplant, regarding the presence of a delayed graft function, he required hemodialysis for 3 weeks. At transplant, plasma oxalate level was at 27 µmol/L, which was below the saturation level established in this setting: <30 µmol/L.2 At 3 and 12 months of posttransplant, plasma oxalate level was at 11 and 12 µmol/L. Urine oxalate/creatinine ratio decreased after transplant and reached the normal range at 9 months of posttransplant (N < 40 µmol/mmol2) and remained unchanged thereafter (Figure 1A). Biliary tract complications were the main complications observed during follow-up. Kidney biopsies were performed at 1, 6, and 12 months of posttransplant: we observed some oxalate crystals at months 1 and 6 but not at month 12 post–combined liver-kidney transplant (Figure 1B-D). At 15 months of posttransplant, kidney function was stable; that is, estimated glomerular filtration rate was at 49 mL/min/1.73 m2 (Figure 1A). Figure 1Open in figure viewerPowerPoint A, Course of posttransplant kidney graft function (represented by CKD-EPI eGFR (mL/min/1.73 m2) and urine oxalate/creatinine ratio. The recipient received continuous hemodiafiltration for the 2 first weeks of posttransplant, and intermittent dialysis for 1 additional week. Initial maintenance immunosuppressive therapy relied on tacrolimus, mycophenolate mofetil, and low-dose steroids. Because of delayed graft function, belatacept was added at month 1 and tacrolimus trough levels targets were decreased from 7-10 to 3-5 ng/mL. At 4 months of posttransplant, tacrolimus was stopped, leading to an improvement in kidney function. Abbreviation: CLKT, Combined liver-kidney transplantation. B, Kidney biopsy 1 month post combined liver-kidney transplantation were some crystal oxalate (arrows) on Masson's trichrome stain (magnification × 200) can be seen. C, Kidney biopsy 6 months post combined liver-kidney transplantation were some crystal oxalate (arrows) on H&E stain (magnification × 100) (left) and Masson's trichrome stain (magnification × 200) (right) can be seen. D, Kidney biopsy 12 months showing the absence of crystal oxalate on H&E stain (left) (magnification × 100) and Masson's trichrome stain (magnification × 200) (right) [Color figure can be viewed at wileyonlinelibrary.com] Although the glyoxylate-reductase/hydroxypyruvate-reductase has a wide tissue distribution,6 similar to the report by Dhondup et al,3 we have also observed that combined liver-kidney transplant prevents early reoccurrence of the disease on kidney transplants. The most striking finding is that our patient had a rapid and severe relapse of oxalate nephropathy after isolated kidney transplant whereas he did not after CLKT. Nevertheless, we cannot exclude that, in addition to liver transplant, posttransplant dialysis for 3 weeks may have had a beneficial effect in preventing oxalate deposits. Collecting data from a large patient sample with PH2 who had undergone isolated kidney transplant or CLKT is required to establish robust guidelines. REFERENCES 1Cochat P, Rumsby G. Primary hyperoxaluria. N Engl J Med. 2013; 369(7): 649- 658. CrossrefCASPubMedWeb of Science®Google Scholar 2Hoppe B. An update on primary hyperoxaluria. Nat Rev Nephrol. 2012; 8(8): 467- 475. CrossrefCASPubMedWeb of Science®Google Scholar 3Dhondup T, Lorenz EC, Milliner DS, Lieske JC. Combined liver-kidney transplantation for primary hyperoxaluria Type 2: a case report. Am J Transplant. 2018; 18(1): 253- 257. Wiley Online LibraryCASPubMedWeb of Science®Google Scholar 4Liu SI, Gao B, Wang G, et al. Recurrent primary hyperoxaluria type 2 leads to early post-transplant renal function loss: a case report. Exp Ther Med. 2018; 15(4): 3169- 3172. CASPubMedWeb of Science®Google Scholar 5Del Bello A, Cointault O, Delas A, Kamar N. Recurrence of oxalate nephropathy after isolated kidney transplantation for primary hyperoxaluria type 2. Am J Transplant. 2018; 18(2): 525- 526. Wiley Online LibraryPubMedWeb of Science®Google Scholar 6Cregeen DP, Williams EL, Hulton S, Rumsby G. Molecular analysis of the glyoxylate reductase (GRHPR) gene and description of mutations underlying primary hyperoxaluria type 2. Hum Mutat. 2003; 22(6): 497- 497. Wiley Online LibraryPubMedWeb of Science®Google Scholar Citing Literature Volume20, Issue6June 2020Pages 1752-1753 AST and ASTS members - please log in via your Society website for full journal access.AST Members >> ASTS Members >> FiguresReferencesRelatedInformation