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Fainting Fanconi syndrome clarified by proxy: a case report
BACKGROUND: Rare diseases may elude diagnosis due to unfamiliarity of the treating physicians with the specific disorder. Yet, advances in genetics have tremendously enhanced our ability to establish specific and sometimes surprising diagnoses. CASE PRESENTATION: We report a case of renal Fanconi sy...
Autores principales: | , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
BioMed Central
2017
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5504823/ https://www.ncbi.nlm.nih.gov/pubmed/28693455 http://dx.doi.org/10.1186/s12882-017-0649-8 |
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author | Walsh, Stephen Benedict Unwin, Robert Kleta, Robert van’t Hoff, William Bass, Paul Hussain, Khalid Ellard, Sian Bockenhauer, Detlef |
author_facet | Walsh, Stephen Benedict Unwin, Robert Kleta, Robert van’t Hoff, William Bass, Paul Hussain, Khalid Ellard, Sian Bockenhauer, Detlef |
author_sort | Walsh, Stephen Benedict |
collection | PubMed |
description | BACKGROUND: Rare diseases may elude diagnosis due to unfamiliarity of the treating physicians with the specific disorder. Yet, advances in genetics have tremendously enhanced our ability to establish specific and sometimes surprising diagnoses. CASE PRESENTATION: We report a case of renal Fanconi syndrome associated with intermittent hypoglycemic episodes, the specific cause for which remained elusive for over 30 years, despite numerous investigations, including three kidney and one liver biopsy. The most recent kidney biopsy showed dysmorphic mitochondria, suggesting a mitochondrial disorder. When her son presented with hypoglycemia in the neonatal period, he underwent routine genetic testing for hyperinsulinemic hypoglycemia, which revealed a specific mutation in HNF4A. Subsequent testing of the mother confirmed the diagnosis also in her. CONCLUSION: Modern sequencing technologies that test multiple genes simultaneously enable specific diagnoses, even if the underlying disorder was not clinically suspected. The finding of mitochondrial dysmorphology provides a potential clue for the mechanism, by which the identified mutation causes renal Fanconi syndrome. |
format | Online Article Text |
id | pubmed-5504823 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2017 |
publisher | BioMed Central |
record_format | MEDLINE/PubMed |
spelling | pubmed-55048232017-07-12 Fainting Fanconi syndrome clarified by proxy: a case report Walsh, Stephen Benedict Unwin, Robert Kleta, Robert van’t Hoff, William Bass, Paul Hussain, Khalid Ellard, Sian Bockenhauer, Detlef BMC Nephrol Case Report BACKGROUND: Rare diseases may elude diagnosis due to unfamiliarity of the treating physicians with the specific disorder. Yet, advances in genetics have tremendously enhanced our ability to establish specific and sometimes surprising diagnoses. CASE PRESENTATION: We report a case of renal Fanconi syndrome associated with intermittent hypoglycemic episodes, the specific cause for which remained elusive for over 30 years, despite numerous investigations, including three kidney and one liver biopsy. The most recent kidney biopsy showed dysmorphic mitochondria, suggesting a mitochondrial disorder. When her son presented with hypoglycemia in the neonatal period, he underwent routine genetic testing for hyperinsulinemic hypoglycemia, which revealed a specific mutation in HNF4A. Subsequent testing of the mother confirmed the diagnosis also in her. CONCLUSION: Modern sequencing technologies that test multiple genes simultaneously enable specific diagnoses, even if the underlying disorder was not clinically suspected. The finding of mitochondrial dysmorphology provides a potential clue for the mechanism, by which the identified mutation causes renal Fanconi syndrome. BioMed Central 2017-07-11 /pmc/articles/PMC5504823/ /pubmed/28693455 http://dx.doi.org/10.1186/s12882-017-0649-8 Text en © The Author(s). 2017 Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. |
spellingShingle | Case Report Walsh, Stephen Benedict Unwin, Robert Kleta, Robert van’t Hoff, William Bass, Paul Hussain, Khalid Ellard, Sian Bockenhauer, Detlef Fainting Fanconi syndrome clarified by proxy: a case report |
title | Fainting Fanconi syndrome clarified by proxy: a case report |
title_full | Fainting Fanconi syndrome clarified by proxy: a case report |
title_fullStr | Fainting Fanconi syndrome clarified by proxy: a case report |
title_full_unstemmed | Fainting Fanconi syndrome clarified by proxy: a case report |
title_short | Fainting Fanconi syndrome clarified by proxy: a case report |
title_sort | fainting fanconi syndrome clarified by proxy: a case report |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5504823/ https://www.ncbi.nlm.nih.gov/pubmed/28693455 http://dx.doi.org/10.1186/s12882-017-0649-8 |
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