Hydroxychloroquine-induced renal phospholipidosis resembling Fabry disease in undifferentiated connective tissue disease: A case report
Identifieur interne : 000766 ( Main/Exploration ); précédent : 000765; suivant : 000767Hydroxychloroquine-induced renal phospholipidosis resembling Fabry disease in undifferentiated connective tissue disease: A case report
Auteurs : Song-Zhao Wu ; Xiang Liang ; Jian Geng ; Meng-Bi Zhang ; Na Xie ; Xiao-Yan SuSource :
- World Journal of Clinical Cases [ 2307-8960 ] ; 2019.
Abstract
Fabry disease is a kind of lysosomal storage disease resulting from deficient activity of the lysosomal hydrolase alpha-galactosidase A (GLA). A mutation in the
We report the case of a 41-year-old female patient who was diagnosed with undifferentiated connective tissue disease in 2008. Hydroxychloroquine treatment started 2 years ago, and proteinuria and hematuria increased. Renal biopsy demonstrated renal phospholipidosis. Zebra bodies and myelin figures were found by renal electron microscopy and were initially thought to be indicators of Fabry disease. A genetic analysis of the patient and her family members did not reveal mutations in the
This report reveals one of the adverse effects of hydroxychloroquine. We should pay more attention to hydroxychloroquine-induced renal phospholipidosis.
Url:
DOI: 10.12998/wjcc.v7.i24.4377
PubMed: 31911921
PubMed Central: 6940343
Affiliations:
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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Hydroxychloroquine-induced renal phospholipidosis resembling Fabry disease in undifferentiated connective tissue disease: A case report</title><author><name sortKey="Wu, Song Zhao" sort="Wu, Song Zhao" uniqKey="Wu S" first="Song-Zhao" last="Wu">Song-Zhao Wu</name></author><author><name sortKey="Liang, Xiang" sort="Liang, Xiang" uniqKey="Liang X" first="Xiang" last="Liang">Xiang Liang</name></author><author><name sortKey="Geng, Jian" sort="Geng, Jian" uniqKey="Geng J" first="Jian" last="Geng">Jian Geng</name></author><author><name sortKey="Zhang, Meng Bi" sort="Zhang, Meng Bi" uniqKey="Zhang M" first="Meng-Bi" last="Zhang">Meng-Bi Zhang</name></author><author><name sortKey="Xie, Na" sort="Xie, Na" uniqKey="Xie N" first="Na" last="Xie">Na Xie</name></author><author><name sortKey="Su, Xiao Yan" sort="Su, Xiao Yan" uniqKey="Su X" first="Xiao-Yan" last="Su">Xiao-Yan Su</name></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">31911921</idno><idno type="pmc">6940343</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6940343</idno><idno type="RBID">PMC:6940343</idno><idno type="doi">10.12998/wjcc.v7.i24.4377</idno><date when="2019">2019</date><idno type="wicri:Area/Pmc/Corpus">000326</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">000326</idno><idno type="wicri:Area/Pmc/Curation">000326</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Curation">000326</idno><idno type="wicri:Area/Pmc/Checkpoint">000709</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Checkpoint">000709</idno><idno type="wicri:Area/Ncbi/Merge">000E14</idno><idno type="wicri:Area/Ncbi/Curation">000E14</idno><idno type="wicri:Area/Ncbi/Checkpoint">000E14</idno><idno type="wicri:Area/Main/Merge">000766</idno><idno type="wicri:Area/Main/Curation">000766</idno><idno type="wicri:Area/Main/Exploration">000766</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Hydroxychloroquine-induced renal phospholipidosis resembling Fabry disease in undifferentiated connective tissue disease: A case report</title><author><name sortKey="Wu, Song Zhao" sort="Wu, Song Zhao" uniqKey="Wu S" first="Song-Zhao" last="Wu">Song-Zhao Wu</name></author><author><name sortKey="Liang, Xiang" sort="Liang, Xiang" uniqKey="Liang X" first="Xiang" last="Liang">Xiang Liang</name></author><author><name sortKey="Geng, Jian" sort="Geng, Jian" uniqKey="Geng J" first="Jian" last="Geng">Jian Geng</name></author><author><name sortKey="Zhang, Meng Bi" sort="Zhang, Meng Bi" uniqKey="Zhang M" first="Meng-Bi" last="Zhang">Meng-Bi Zhang</name></author><author><name sortKey="Xie, Na" sort="Xie, Na" uniqKey="Xie N" first="Na" last="Xie">Na Xie</name></author><author><name sortKey="Su, Xiao Yan" sort="Su, Xiao Yan" uniqKey="Su X" first="Xiao-Yan" last="Su">Xiao-Yan Su</name></author></analytic><series><title level="j">World Journal of Clinical Cases</title><idno type="eISSN">2307-8960</idno><imprint><date when="2019">2019</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><sec><title>BACKGROUND</title><p>Fabry disease is a kind of lysosomal storage disease resulting from deficient activity of the lysosomal hydrolase alpha-galactosidase A (GLA). A mutation in the <italic>GLA</italic> gene leads to a loss of activity of alpha-galactosidase A. Some drugs, such as hydroxychloroquine, can cause pathological changes similar to those usually seen in Fabry disease.</p></sec><sec><title>CASE SUMMARY</title><p>We report the case of a 41-year-old female patient who was diagnosed with undifferentiated connective tissue disease in 2008. Hydroxychloroquine treatment started 2 years ago, and proteinuria and hematuria increased. Renal biopsy demonstrated renal phospholipidosis. Zebra bodies and myelin figures were found by renal electron microscopy and were initially thought to be indicators of Fabry disease. A genetic analysis of the patient and her family members did not reveal mutations in the <italic>GLA</italic> gene, supporting a diagnosis of hydroxychloroquine-induced renal phospholipidosis.</p></sec><sec><title>CONCLUSION</title><p>This report reveals one of the adverse effects of hydroxychloroquine. 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sort="Zhang, Meng Bi" uniqKey="Zhang M" first="Meng-Bi" last="Zhang">Meng-Bi Zhang</name></noCountry></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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