Sustained pyridoxine response in primary hyperoxaluria type 1 recipients of kidney alone transplant

E. C. Lorenz, John C Lieske, B. M. Seide, A. M. Meek, J. B. Olson, E. J. Bergstralh, D. S. Milliner

Research output: Contribution to journalArticle

15 Citations (Scopus)

Abstract

Combined liver kidney transplant is the preferred transplant option for most patients with primary hyperoxaluria type 1 (PH1) given that it removes the hepatic source of oxalate production and improves renal allograft survival. However, PH1 patients homozygous for the G170R mutation can develop normal urine oxalate levels with pyridoxine therapy and may be candidates for kidney alone transplant (KTx). We examined the efficacy of pyridoxine therapy following KTx in five patients homozygous for G170R transplanted between September 1999 and July 2013. All patients were maintained on pyridoxine posttransplant. Median age at transplant was 39 years (range 33-67 years). Median follow-up posttransplant was 8.5 years (range 0.2-13.9 years). At the end of follow-up, four grafts were functioning. One graft failed 13.9 years posttransplant due to recurrent oxalate nephropathy following an acute medical illness. After tissue oxalate stores had cleared, posttransplant urine oxalate levels were <0.5 mmol/24 h the majority of times checked. Calcium oxalate crystals were noted in only 3/13 allograft biopsies. This series suggests that a subgroup of PH1 patients demonstrate sustained response to pyridoxine therapy following KTx. Therefore, pyridoxine combined with KTx should be considered for PH1 patients with a homozygous G170R mutation. The authors demonstrate sustained response to pyridoxine therapy following kidney alone transplant in a series of patients with primary hyperoxaluria type 1.

Original languageEnglish (US)
Pages (from-to)1433-1438
Number of pages6
JournalAmerican Journal of Transplantation
Volume14
Issue number6
DOIs
StatePublished - 2014

Fingerprint

Pyridoxine
Oxalates
Transplants
Kidney
Allografts
Urine
Calcium Oxalate
Mutation
Liver
Therapeutics
Primary hyperoxaluria type 1
Biopsy

Keywords

  • Kidney disease
  • kidney transplantation
  • living donor
  • pathology
  • recurrent disease

ASJC Scopus subject areas

  • Transplantation
  • Immunology and Allergy
  • Pharmacology (medical)
  • Medicine(all)

Cite this

Lorenz, E. C., Lieske, J. C., Seide, B. M., Meek, A. M., Olson, J. B., Bergstralh, E. J., & Milliner, D. S. (2014). Sustained pyridoxine response in primary hyperoxaluria type 1 recipients of kidney alone transplant. American Journal of Transplantation, 14(6), 1433-1438. https://doi.org/10.1111/ajt.12706

Sustained pyridoxine response in primary hyperoxaluria type 1 recipients of kidney alone transplant. / Lorenz, E. C.; Lieske, John C; Seide, B. M.; Meek, A. M.; Olson, J. B.; Bergstralh, E. J.; Milliner, D. S.

In: American Journal of Transplantation, Vol. 14, No. 6, 2014, p. 1433-1438.

Research output: Contribution to journalArticle

Lorenz, E. C. ; Lieske, John C ; Seide, B. M. ; Meek, A. M. ; Olson, J. B. ; Bergstralh, E. J. ; Milliner, D. S. / Sustained pyridoxine response in primary hyperoxaluria type 1 recipients of kidney alone transplant. In: American Journal of Transplantation. 2014 ; Vol. 14, No. 6. pp. 1433-1438.
@article{29052061917a429086b761c3b62ffda0,
title = "Sustained pyridoxine response in primary hyperoxaluria type 1 recipients of kidney alone transplant",
abstract = "Combined liver kidney transplant is the preferred transplant option for most patients with primary hyperoxaluria type 1 (PH1) given that it removes the hepatic source of oxalate production and improves renal allograft survival. However, PH1 patients homozygous for the G170R mutation can develop normal urine oxalate levels with pyridoxine therapy and may be candidates for kidney alone transplant (KTx). We examined the efficacy of pyridoxine therapy following KTx in five patients homozygous for G170R transplanted between September 1999 and July 2013. All patients were maintained on pyridoxine posttransplant. Median age at transplant was 39 years (range 33-67 years). Median follow-up posttransplant was 8.5 years (range 0.2-13.9 years). At the end of follow-up, four grafts were functioning. One graft failed 13.9 years posttransplant due to recurrent oxalate nephropathy following an acute medical illness. After tissue oxalate stores had cleared, posttransplant urine oxalate levels were <0.5 mmol/24 h the majority of times checked. Calcium oxalate crystals were noted in only 3/13 allograft biopsies. This series suggests that a subgroup of PH1 patients demonstrate sustained response to pyridoxine therapy following KTx. Therefore, pyridoxine combined with KTx should be considered for PH1 patients with a homozygous G170R mutation. The authors demonstrate sustained response to pyridoxine therapy following kidney alone transplant in a series of patients with primary hyperoxaluria type 1.",
keywords = "Kidney disease, kidney transplantation, living donor, pathology, recurrent disease",
author = "Lorenz, {E. C.} and Lieske, {John C} and Seide, {B. M.} and Meek, {A. M.} and Olson, {J. B.} and Bergstralh, {E. J.} and Milliner, {D. S.}",
year = "2014",
doi = "10.1111/ajt.12706",
language = "English (US)",
volume = "14",
pages = "1433--1438",
journal = "American Journal of Transplantation",
issn = "1600-6135",
publisher = "Wiley-Blackwell",
number = "6",

}

TY - JOUR

T1 - Sustained pyridoxine response in primary hyperoxaluria type 1 recipients of kidney alone transplant

AU - Lorenz, E. C.

AU - Lieske, John C

AU - Seide, B. M.

AU - Meek, A. M.

AU - Olson, J. B.

AU - Bergstralh, E. J.

AU - Milliner, D. S.

PY - 2014

Y1 - 2014

N2 - Combined liver kidney transplant is the preferred transplant option for most patients with primary hyperoxaluria type 1 (PH1) given that it removes the hepatic source of oxalate production and improves renal allograft survival. However, PH1 patients homozygous for the G170R mutation can develop normal urine oxalate levels with pyridoxine therapy and may be candidates for kidney alone transplant (KTx). We examined the efficacy of pyridoxine therapy following KTx in five patients homozygous for G170R transplanted between September 1999 and July 2013. All patients were maintained on pyridoxine posttransplant. Median age at transplant was 39 years (range 33-67 years). Median follow-up posttransplant was 8.5 years (range 0.2-13.9 years). At the end of follow-up, four grafts were functioning. One graft failed 13.9 years posttransplant due to recurrent oxalate nephropathy following an acute medical illness. After tissue oxalate stores had cleared, posttransplant urine oxalate levels were <0.5 mmol/24 h the majority of times checked. Calcium oxalate crystals were noted in only 3/13 allograft biopsies. This series suggests that a subgroup of PH1 patients demonstrate sustained response to pyridoxine therapy following KTx. Therefore, pyridoxine combined with KTx should be considered for PH1 patients with a homozygous G170R mutation. The authors demonstrate sustained response to pyridoxine therapy following kidney alone transplant in a series of patients with primary hyperoxaluria type 1.

AB - Combined liver kidney transplant is the preferred transplant option for most patients with primary hyperoxaluria type 1 (PH1) given that it removes the hepatic source of oxalate production and improves renal allograft survival. However, PH1 patients homozygous for the G170R mutation can develop normal urine oxalate levels with pyridoxine therapy and may be candidates for kidney alone transplant (KTx). We examined the efficacy of pyridoxine therapy following KTx in five patients homozygous for G170R transplanted between September 1999 and July 2013. All patients were maintained on pyridoxine posttransplant. Median age at transplant was 39 years (range 33-67 years). Median follow-up posttransplant was 8.5 years (range 0.2-13.9 years). At the end of follow-up, four grafts were functioning. One graft failed 13.9 years posttransplant due to recurrent oxalate nephropathy following an acute medical illness. After tissue oxalate stores had cleared, posttransplant urine oxalate levels were <0.5 mmol/24 h the majority of times checked. Calcium oxalate crystals were noted in only 3/13 allograft biopsies. This series suggests that a subgroup of PH1 patients demonstrate sustained response to pyridoxine therapy following KTx. Therefore, pyridoxine combined with KTx should be considered for PH1 patients with a homozygous G170R mutation. The authors demonstrate sustained response to pyridoxine therapy following kidney alone transplant in a series of patients with primary hyperoxaluria type 1.

KW - Kidney disease

KW - kidney transplantation

KW - living donor

KW - pathology

KW - recurrent disease

UR - http://www.scopus.com/inward/record.url?scp=84901276046&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=84901276046&partnerID=8YFLogxK

U2 - 10.1111/ajt.12706

DO - 10.1111/ajt.12706

M3 - Article

C2 - 24797341

AN - SCOPUS:84901276046

VL - 14

SP - 1433

EP - 1438

JO - American Journal of Transplantation

JF - American Journal of Transplantation

SN - 1600-6135

IS - 6

ER -