Cargando…

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...

Descripción completa

Detalles Bibliográficos
Autores principales: Walsh, Stephen Benedict, Unwin, Robert, Kleta, Robert, van’t Hoff, William, Bass, Paul, Hussain, Khalid, Ellard, Sian, Bockenhauer, Detlef
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2017
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
_version_ 1783249356091031552
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
work_keys_str_mv AT walshstephenbenedict faintingfanconisyndromeclarifiedbyproxyacasereport
AT unwinrobert faintingfanconisyndromeclarifiedbyproxyacasereport
AT kletarobert faintingfanconisyndromeclarifiedbyproxyacasereport
AT vanthoffwilliam faintingfanconisyndromeclarifiedbyproxyacasereport
AT basspaul faintingfanconisyndromeclarifiedbyproxyacasereport
AT hussainkhalid faintingfanconisyndromeclarifiedbyproxyacasereport
AT ellardsian faintingfanconisyndromeclarifiedbyproxyacasereport
AT bockenhauerdetlef faintingfanconisyndromeclarifiedbyproxyacasereport