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Domino liver transplant from a donor with maple syrup urine disease into a recipient with phenylketonuria

Classical phenylketonuria (PKU) presents a unique challenge for women of child-bearing age. In the context of pregnancy, poorly controlled hyperphenylalaninemia can result in a devastating constellation of outcomes for the baby referred to as the maternal PKU Syndrome. We present the case of a woman...

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Autores principales: Raghu, Vikram K., Dobrowolski, Steven F., Sindhi, Rakesh, Strauss, Kevin A., Mazariegos, George V., Vockley, Jerry, Soltys, Kyle
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Elsevier 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9248231/
https://www.ncbi.nlm.nih.gov/pubmed/35782613
http://dx.doi.org/10.1016/j.ymgmr.2022.100866
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author Raghu, Vikram K.
Dobrowolski, Steven F.
Sindhi, Rakesh
Strauss, Kevin A.
Mazariegos, George V.
Vockley, Jerry
Soltys, Kyle
author_facet Raghu, Vikram K.
Dobrowolski, Steven F.
Sindhi, Rakesh
Strauss, Kevin A.
Mazariegos, George V.
Vockley, Jerry
Soltys, Kyle
author_sort Raghu, Vikram K.
collection PubMed
description Classical phenylketonuria (PKU) presents a unique challenge for women of child-bearing age. In the context of pregnancy, poorly controlled hyperphenylalaninemia can result in a devastating constellation of outcomes for the baby referred to as the maternal PKU Syndrome. We present the case of a woman with classical PKU unable to maintain a restricted diet and refractory to pharmacological therapies. She elected to undergo a domino liver transplant, receiving an organ from a donor with classical branched chain ketoacid dehydrogenase deficiency (maple syrup urine disease). Plasma phenylalanine concentrations normalized within a few days after transplant and remained so on an unrestricted diet during the first year of follow-up. The patient reports subjective improvements in mood, energy level, and overall quality of life. In the appropriate clinical setting, liver transplant should be considered to provide metabolic stability for PKU patients, particularly women of childbearing age.
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spelling pubmed-92482312022-07-02 Domino liver transplant from a donor with maple syrup urine disease into a recipient with phenylketonuria Raghu, Vikram K. Dobrowolski, Steven F. Sindhi, Rakesh Strauss, Kevin A. Mazariegos, George V. Vockley, Jerry Soltys, Kyle Mol Genet Metab Rep Case Report Classical phenylketonuria (PKU) presents a unique challenge for women of child-bearing age. In the context of pregnancy, poorly controlled hyperphenylalaninemia can result in a devastating constellation of outcomes for the baby referred to as the maternal PKU Syndrome. We present the case of a woman with classical PKU unable to maintain a restricted diet and refractory to pharmacological therapies. She elected to undergo a domino liver transplant, receiving an organ from a donor with classical branched chain ketoacid dehydrogenase deficiency (maple syrup urine disease). Plasma phenylalanine concentrations normalized within a few days after transplant and remained so on an unrestricted diet during the first year of follow-up. The patient reports subjective improvements in mood, energy level, and overall quality of life. In the appropriate clinical setting, liver transplant should be considered to provide metabolic stability for PKU patients, particularly women of childbearing age. Elsevier 2022-04-21 /pmc/articles/PMC9248231/ /pubmed/35782613 http://dx.doi.org/10.1016/j.ymgmr.2022.100866 Text en © 2022 Published by Elsevier Inc. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
spellingShingle Case Report
Raghu, Vikram K.
Dobrowolski, Steven F.
Sindhi, Rakesh
Strauss, Kevin A.
Mazariegos, George V.
Vockley, Jerry
Soltys, Kyle
Domino liver transplant from a donor with maple syrup urine disease into a recipient with phenylketonuria
title Domino liver transplant from a donor with maple syrup urine disease into a recipient with phenylketonuria
title_full Domino liver transplant from a donor with maple syrup urine disease into a recipient with phenylketonuria
title_fullStr Domino liver transplant from a donor with maple syrup urine disease into a recipient with phenylketonuria
title_full_unstemmed Domino liver transplant from a donor with maple syrup urine disease into a recipient with phenylketonuria
title_short Domino liver transplant from a donor with maple syrup urine disease into a recipient with phenylketonuria
title_sort domino liver transplant from a donor with maple syrup urine disease into a recipient with phenylketonuria
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9248231/
https://www.ncbi.nlm.nih.gov/pubmed/35782613
http://dx.doi.org/10.1016/j.ymgmr.2022.100866
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