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Bone Deformities and Kidney Failure: Coincidence of PHEX-Related Hypophosphatemic Rickets and m.3243A>G Mitochondrial Disease.
Nielsen, Simone Rask; Hansen, Stinus Gadegaard; Bistrup, Claus; Brusgaard, Klaus; Frederiksen, Anja Lisbeth.
Afiliação
  • Nielsen SR; Department of Clinical Genetics, Aalborg University Hospital, Ladegaardsgade 5, 5. Floor, 9000, Aalborg, Denmark. s.rask@rn.dk.
  • Hansen SG; Department of Clinical Research, Aalborg University, Aalborg, Denmark. s.rask@rn.dk.
  • Bistrup C; Department of Endocrinology, Hospital South West Jutland, Esbjerg, Denmark.
  • Brusgaard K; Department of Nephrology, Odense University Hospital, Odense, Denmark.
  • Frederiksen AL; Department of Clinical Research, University of Southern Denmark, Odense, Denmark.
Calcif Tissue Int ; 111(6): 641-645, 2022 12.
Article em En | MEDLINE | ID: mdl-35916905
X-linked hypophosphatemic rickets (XLH) and m.3243A>G mitochondrial disease share several clinical findings, including short stature, hearing impairment (HI), nephropathy, and hypertension. Here, we report on a case with the rare coincidence of these two genetic conditions. In early childhood, the patient presented with hypophosphatemia and bone deformities and was clinically diagnosed with XLH. This was genetically verified in adulthood with the identification of a de novo pathogenic deletion in phosphate-regulating endopeptidase homolog X-linked (PHEX). In addition, the patient developed HI and hypertension and when his mother was diagnosed with m.3243A>G, subsequent genetic testing confirmed the patient to carry the same variant. Over the next two decades, the patient developed progressive renal impairment however without nephrocalcinosis known to associate with XLH which could indicate an m.3243A>G-related kidney disease. Parallel with the progression of renal impairment, the patient developed hyperphosphatemia and secondary hyperparathyroidism. In conclusion, this case represents a complex clinical phenotype with the reversal of hypo- to hyperphosphatemia in XLH potentially mediated by the development of an m.3243A>G-associated nephropathy.
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Texto completo: 1 Base de dados: MEDLINE Assunto principal: Doenças Mitocondriais / Doenças Genéticas Ligadas ao Cromossomo X / Insuficiência Renal / Raquitismo Hipofosfatêmico Familiar / Hiperfosfatemia / Raquitismo Hipofosfatêmico / Hipertensão Tipo de estudo: Prognostic_studies Limite: Child, preschool / Humans Idioma: En Ano de publicação: 2022 Tipo de documento: Article

Texto completo: 1 Base de dados: MEDLINE Assunto principal: Doenças Mitocondriais / Doenças Genéticas Ligadas ao Cromossomo X / Insuficiência Renal / Raquitismo Hipofosfatêmico Familiar / Hiperfosfatemia / Raquitismo Hipofosfatêmico / Hipertensão Tipo de estudo: Prognostic_studies Limite: Child, preschool / Humans Idioma: En Ano de publicação: 2022 Tipo de documento: Article