Simultaneous kidney and pancreas transplantation in a patient with nail-patella syndrome and insulin-dependent diabetes

Pediatr Nephrol. 2023 Jun;38(6):1985-1989. doi: 10.1007/s00467-022-05817-6. Epub 2022 Nov 24.

Abstract

Background: Nail-patella syndrome (NPS) is a rare autosomal dominant disorder caused by a mutation in LIM-homeodomain transcription factor 1-beta (LMX1B) and characterized by nail dystrophy, skeletal changes, glaucoma, and kidney disease with up to 30% of patients progressing to kidney failure. Autoimmune diseases, including thyroid disease, have been reported previously in patients with NPS.

Case-diagnosis/treatment: We report the case of a pediatric patient with NPS with kidney failure, hypothyroidism, and type 1 diabetes mellitus. The patient's pedigree and identification of a kidney specific mutation in LMX1B was a result of whole exome sequencing. Clinical data was obtained from retrospective chart review and included the 1-year post-transplant follow-up period. At 15 years of age, our patient received a simultaneous kidney-pancreas transplantation, from a 3 HLA antigen mismatched deceased donor. The donor was CMV + , EBV - and our patient was CMV - , EBV - at time of transplant. Our patient maintained normal kidney function and euglycemia without insulin therapy at 1 year post-transplant.

Conclusions: The patient's hypothyroidism, diabetes mellitus, and kidney failure may all be related to LMX1B mutation. Further study is needed to clarify the genetic link between these processes. Simultaneous kidney-pancreas transplantation can be used to successfully treat diabetes mellitus and kidney failure in a pediatric patient.

Publication types

  • Case Reports

MeSH terms

  • Child
  • Cytomegalovirus Infections*
  • Diabetes Mellitus, Type 1* / complications
  • Diabetes Mellitus, Type 1* / genetics
  • Diabetes Mellitus, Type 1* / surgery
  • Homeodomain Proteins / genetics
  • Humans
  • Insulins*
  • Kidney
  • Kidney Transplantation*
  • Nail-Patella Syndrome* / complications
  • Nail-Patella Syndrome* / genetics
  • Nail-Patella Syndrome* / surgery
  • Pancreas Transplantation*
  • Renal Insufficiency*
  • Retrospective Studies

Substances

  • Insulins
  • Homeodomain Proteins