{"id":"https://openalex.org/W2810745812","doi":"https://doi.org/10.1136/bcr-2018-225059","title":"Regressive pyridoxine-induced sensory neuronopathy in a patient with homocystinuria","display_name":"Regressive pyridoxine-induced sensory neuronopathy in a patient with homocystinuria","publication_year":2018,"publication_date":"2018-06-28","ids":{"openalex":"https://openalex.org/W2810745812","doi":"https://doi.org/10.1136/bcr-2018-225059","mag":"2810745812","pmid":"https://pubmed.ncbi.nlm.nih.gov/29954767","pmcid":"https://www.ncbi.nlm.nih.gov/pmc/articles/6040505"},"language":"en","primary_location":{"id":"doi:10.1136/bcr-2018-225059","doi":"https://doi.org/10.1136/bcr-2018-225059","is_oa":false,"landing_page_url":"https://doi.org/10.1136/bcr-2018-225059","pdf_url":null,"source":{"id":"https://openalex.org/S141953298","display_name":"BMJ Case Reports","issn_l":"1757-790X","issn":["1757-790X"],"is_oa":false,"is_in_doaj":false,"is_core":true,"listed_in":["anvur-scientific","cwts-core","dongbi-d","jufo-1","ki-jl-1","medline","norway-1","poland-20","russia-white-list-2","vabb-shw"],"host_organization":"https://openalex.org/P4310319945","host_organization_name":"BMJ","host_organization_lineage":["https://openalex.org/P4310319945"],"host_organization_lineage_names":["BMJ"],"type":"journal"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"BMJ Case Reports","raw_type":"journal-article"},"volume":null,"type":"article","indexed_in":["crossref","pubmed"],"open_access":{"is_oa":true,"oa_status":"green","oa_url":"https://www.ncbi.nlm.nih.gov/pmc/articles/6040505","any_repository_has_fulltext":true},"authorships":[{"author_position":"first","author":{"id":"https://openalex.org/A5041504666","display_name":"Andoni Echaniz\u2010Laguna","orcid":"https://orcid.org/0000-0003-1012-9783","observed_orcids":["https://orcid.org/0000-0003-1012-9783"]},"institutions":[{"id":"https://openalex.org/I4210145324","display_name":"H\u00f4pitaux Universitaires de Strasbourg","ror":"https://ror.org/04bckew43","country_code":"FR","type":"healthcare","lineage":["https://openalex.org/I4210145324"]}],"countries":["FR"],"is_corresponding":false,"raw_author_name":"Andoni Echaniz-Laguna","raw_affiliation_strings":["Neurologie, Hopitaux universitaires de Strasbourg, Strasbourg, France"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Neurologie, Hopitaux universitaires de Strasbourg, Strasbourg, France","institution_ids":["https://openalex.org/I4210145324"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5113818286","display_name":"Rachel Mourot-Cottet","orcid":null,"observed_orcids":[]},"institutions":[{"id":"https://openalex.org/I4210145324","display_name":"H\u00f4pitaux Universitaires de Strasbourg","ror":"https://ror.org/04bckew43","country_code":"FR","type":"healthcare","lineage":["https://openalex.org/I4210145324"]}],"countries":["FR"],"is_corresponding":false,"raw_author_name":"Rachel Mourot-Cottet","raw_affiliation_strings":["Medecine Interne, Hopitaux universitaires de Strasbourg, Strasbourg, France"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Medecine Interne, Hopitaux universitaires de Strasbourg, Strasbourg, France","institution_ids":["https://openalex.org/I4210145324"]}]},{"author_position":"middle","author":{"id":"https://openalex.org/A5114014005","display_name":"Esther No\u00ebl","orcid":null,"observed_orcids":[]},"institutions":[{"id":"https://openalex.org/I4210145324","display_name":"H\u00f4pitaux Universitaires de Strasbourg","ror":"https://ror.org/04bckew43","country_code":"FR","type":"healthcare","lineage":["https://openalex.org/I4210145324"]}],"countries":["FR"],"is_corresponding":false,"raw_author_name":"Esther Noel","raw_affiliation_strings":["Medecine Interne, Hopitaux universitaires de Strasbourg, Strasbourg, France"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Medecine Interne, Hopitaux universitaires de Strasbourg, Strasbourg, France","institution_ids":["https://openalex.org/I4210145324"]}]},{"author_position":"last","author":{"id":"https://openalex.org/A5010661354","display_name":"Jean\u2010Baptiste Chanson","orcid":"https://orcid.org/0000-0003-4221-3840","observed_orcids":["https://orcid.org/0000-0003-4221-3840"]},"institutions":[{"id":"https://openalex.org/I4210145324","display_name":"H\u00f4pitaux Universitaires de Strasbourg","ror":"https://ror.org/04bckew43","country_code":"FR","type":"healthcare","lineage":["https://openalex.org/I4210145324"]}],"countries":["FR"],"is_corresponding":true,"raw_author_name":"Jean-Baptiste Chanson","raw_affiliation_strings":["Neurologie, Hopitaux universitaires de Strasbourg, Strasbourg, France","jean-baptiste.chanson{at}chru-strasbourg.fr"],"raw_orcid":null,"affiliations":[{"raw_affiliation_string":"Neurologie, Hopitaux universitaires de Strasbourg, Strasbourg, France","institution_ids":["https://openalex.org/I4210145324"]},{"raw_affiliation_string":"jean-baptiste.chanson{at}chru-strasbourg.fr","institution_ids":[]}]}],"institutions":[],"countries_distinct_count":1,"institutions_distinct_count":1,"corresponding_author_ids":["https://openalex.org/A5010661354"],"corresponding_institution_ids":["https://openalex.org/I4210145324"],"apc_list":null,"apc_paid":null,"fwci":2.7658,"has_fulltext":false,"cited_by_count":23,"citation_normalized_percentile":{"value":0.89548502,"is_in_top_1_percent":false,"is_in_top_10_percent":false},"cited_by_percentile_year":{"min":89,"max":98},"biblio":{"volume":"2018","issue":null,"first_page":"bcr","last_page":"2018"},"is_retracted":false,"is_paratext":false,"is_xpac":false,"primary_topic":{"id":"https://openalex.org/T10499","display_name":"Folate and B Vitamins Research","score":0.9106000065803528,"subfield":{"id":"https://openalex.org/subfields/2745","display_name":"Rheumatology"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},"topics":[{"id":"https://openalex.org/T10499","display_name":"Folate and B Vitamins Research","score":0.9106000065803528,"subfield":{"id":"https://openalex.org/subfields/2745","display_name":"Rheumatology"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},{"id":"https://openalex.org/T11735","display_name":"Peripheral Neuropathies and Disorders","score":0.04470000043511391,"subfield":{"id":"https://openalex.org/subfields/2728","display_name":"Neurology"},"field":{"id":"https://openalex.org/fields/27","display_name":"Medicine"},"domain":{"id":"https://openalex.org/domains/4","display_name":"Health Sciences"}},{"id":"https://openalex.org/T12331","display_name":"Hereditary Neurological Disorders","score":0.02239999920129776,"subfield":{"id":"https://openalex.org/subfields/2804","display_name":"Cellular and Molecular Neuroscience"},"field":{"id":"https://openalex.org/fields/28","display_name":"Neuroscience"},"domain":{"id":"https://openalex.org/domains/1","display_name":"Life Sciences"}}],"keywords":[{"id":"https://openalex.org/keywords/pyridoxine","display_name":"pyridoxine","score":0.9110000133514404},{"id":"https://openalex.org/keywords/vitamin-b6","display_name":"vitamin B6","score":0.7379999756813049},{"id":"https://openalex.org/keywords/homocystinuria","display_name":"homocystinuria","score":0.8939999938011169},{"id":"https://openalex.org/keywords/sensory-neuronopathy","display_name":"sensory neuronopathy","score":0.9010000228881836},{"id":"https://openalex.org/keywords/sensory-ganglionopathy","display_name":"sensory ganglionopathy","score":0.9890000224113464},{"id":"https://openalex.org/keywords/neurotoxicity","display_name":"neurotoxicity","score":0.8970000147819519},{"id":"https://openalex.org/keywords/ataxia","display_name":"ataxia","score":0.8349999785423279},{"id":"https://openalex.org/keywords/cystathionine-beta-synthase-deficiency","display_name":"cystathionine beta-synthase deficiency","score":0.8960000276565552},{"id":"https://openalex.org/keywords/dose-reduction","display_name":"dose reduction","score":0.5849999785423279}],"concepts":[{"id":"https://openalex.org/C2779375456","wikidata":"https://www.wikidata.org/wiki/Q994859","display_name":"Homocystinuria","level":4,"score":0.9415326118469238},{"id":"https://openalex.org/C71924100","wikidata":"https://www.wikidata.org/wiki/Q11190","display_name":"Medicine","level":0,"score":0.8854886293411255},{"id":"https://openalex.org/C2776514930","wikidata":"https://www.wikidata.org/wiki/Q423746","display_name":"Pyridoxine","level":2,"score":0.7959960699081421},{"id":"https://openalex.org/C16568411","wikidata":"https://www.wikidata.org/wiki/Q83042","display_name":"Neurology","level":2,"score":0.4126986563205719},{"id":"https://openalex.org/C126322002","wikidata":"https://www.wikidata.org/wiki/Q11180","display_name":"Internal medicine","level":1,"score":0.4065743386745453},{"id":"https://openalex.org/C60644358","wikidata":"https://www.wikidata.org/wiki/Q128570","display_name":"Bioinformatics","level":1,"score":0.34135153889656067},{"id":"https://openalex.org/C177713679","wikidata":"https://www.wikidata.org/wiki/Q679690","display_name":"Intensive care medicine","level":1,"score":0.3244314193725586},{"id":"https://openalex.org/C118552586","wikidata":"https://www.wikidata.org/wiki/Q7867","display_name":"Psychiatry","level":1,"score":0.11948215961456299},{"id":"https://openalex.org/C54355233","wikidata":"https://www.wikidata.org/wiki/Q7162","display_name":"Genetics","level":1,"score":0.10567298531532288},{"id":"https://openalex.org/C2780912031","wikidata":"https://www.wikidata.org/wiki/Q180341","display_name":"Methionine","level":3,"score":0.0},{"id":"https://openalex.org/C86803240","wikidata":"https://www.wikidata.org/wiki/Q420","display_name":"Biology","level":0,"score":0.0},{"id":"https://openalex.org/C515207424","wikidata":"https://www.wikidata.org/wiki/Q8066","display_name":"Amino acid","level":2,"score":0.0}],"mesh":[{"descriptor_ui":"D000328","descriptor_name":"Adult","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D001259","descriptor_name":"Ataxia","qualifier_ui":"Q000139","qualifier_name":"chemically induced","is_major_topic":true},{"descriptor_ui":"D001259","descriptor_name":"Ataxia","qualifier_ui":"Q000503","qualifier_name":"physiopathology","is_major_topic":false},{"descriptor_ui":"D004305","descriptor_name":"Dose-Response Relationship, Drug","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D004827","descriptor_name":"Epilepsy","qualifier_ui":"Q000188","qualifier_name":"drug therapy","is_major_topic":true},{"descriptor_ui":"D005260","descriptor_name":"Female","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D006712","descriptor_name":"Homocystinuria","qualifier_ui":"Q000188","qualifier_name":"drug therapy","is_major_topic":true},{"descriptor_ui":"D006801","descriptor_name":"Humans","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D011115","descriptor_name":"Polyneuropathies","qualifier_ui":"Q000139","qualifier_name":"chemically induced","is_major_topic":true},{"descriptor_ui":"D011115","descriptor_name":"Polyneuropathies","qualifier_ui":"Q000503","qualifier_name":"physiopathology","is_major_topic":false},{"descriptor_ui":"D011736","descriptor_name":"Pyridoxine","qualifier_ui":"Q000008","qualifier_name":"administration & dosage","is_major_topic":false},{"descriptor_ui":"D011736","descriptor_name":"Pyridoxine","qualifier_ui":"Q000009","qualifier_name":"adverse effects","is_major_topic":true},{"descriptor_ui":"D014815","descriptor_name":"Vitamins","qualifier_ui":"Q000008","qualifier_name":"administration & dosage","is_major_topic":false},{"descriptor_ui":"D014815","descriptor_name":"Vitamins","qualifier_ui":"Q000009","qualifier_name":"adverse effects","is_major_topic":true},{"descriptor_ui":"D016896","descriptor_name":"Treatment Outcome","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D019966","descriptor_name":"Substance-Related Disorders","qualifier_ui":null,"qualifier_name":null,"is_major_topic":false},{"descriptor_ui":"D020127","descriptor_name":"Recovery of Function","qualifier_ui":"Q000502","qualifier_name":"physiology","is_major_topic":true}],"locations_count":4,"locations":[{"id":"doi:10.1136/bcr-2018-225059","doi":"https://doi.org/10.1136/bcr-2018-225059","is_oa":false,"landing_page_url":"https://doi.org/10.1136/bcr-2018-225059","pdf_url":null,"source":{"id":"https://openalex.org/S141953298","display_name":"BMJ Case Reports","issn_l":"1757-790X","issn":["1757-790X"],"is_oa":false,"is_in_doaj":false,"is_core":true,"listed_in":["anvur-scientific","cwts-core","dongbi-d","jufo-1","ki-jl-1","medline","norway-1","poland-20","russia-white-list-2","vabb-shw"],"host_organization":"https://openalex.org/P4310319945","host_organization_name":"BMJ","host_organization_lineage":["https://openalex.org/P4310319945"],"host_organization_lineage_names":["BMJ"],"type":"journal"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"BMJ Case Reports","raw_type":"journal-article"},{"id":"pmh:oai:pubmedcentral.nih.gov:6040505","doi":null,"is_oa":true,"landing_page_url":"https://www.ncbi.nlm.nih.gov/pmc/articles/6040505","pdf_url":null,"source":{"id":"https://openalex.org/S2764455111","display_name":"PubMed Central","issn_l":null,"issn":null,"is_oa":true,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I1299303238","host_organization_name":"National Institutes of Health","host_organization_lineage":["https://openalex.org/I1299303238"],"host_organization_lineage_names":[],"type":"repository"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"BMJ Case Rep","raw_type":"Text"},{"id":"pmid:29954767","doi":null,"is_oa":false,"landing_page_url":"https://pubmed.ncbi.nlm.nih.gov/29954767","pdf_url":null,"source":{"id":"https://openalex.org/S4306525036","display_name":"PubMed","issn_l":null,"issn":null,"is_oa":false,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I1299303238","host_organization_name":"National Institutes of Health","host_organization_lineage":["https://openalex.org/I1299303238"],"host_organization_lineage_names":[],"type":"repository"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"BMJ case reports","raw_type":"Case Reports"},{"id":"pmh:oai:open-archive.highwire.org:casereports:2018/jun28_1/bcr-2018-225059","doi":null,"is_oa":false,"landing_page_url":"http://casereports.bmj.com/cgi/content/short/2018/jun28_1/bcr-2018-225059","pdf_url":null,"source":{"id":"https://openalex.org/S4406923041","display_name":"HighWire Press Open Archive","issn_l":null,"issn":null,"is_oa":false,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":null,"host_organization_name":null,"host_organization_lineage":[],"host_organization_lineage_names":[],"type":"repository"},"license":null,"license_id":null,"version":"submittedVersion","is_accepted":false,"is_published":false,"raw_source_name":"","raw_type":"TEXT"}],"best_oa_location":{"id":"pmh:oai:pubmedcentral.nih.gov:6040505","doi":null,"is_oa":true,"landing_page_url":"https://www.ncbi.nlm.nih.gov/pmc/articles/6040505","pdf_url":null,"source":{"id":"https://openalex.org/S2764455111","display_name":"PubMed Central","issn_l":null,"issn":null,"is_oa":true,"is_in_doaj":false,"is_core":false,"listed_in":[],"host_organization":"https://openalex.org/I1299303238","host_organization_name":"National Institutes of Health","host_organization_lineage":["https://openalex.org/I1299303238"],"host_organization_lineage_names":[],"type":"repository"},"license":null,"license_id":null,"version":"publishedVersion","is_accepted":true,"is_published":true,"raw_source_name":"BMJ Case Rep","raw_type":"Text"},"sustainable_development_goals":[{"display_name":"Good health and well-being","score":1.0,"id":"https://metadata.un.org/sdg/3"}],"study_designs":[{"display_name":"Case Report","id":"https://openalex.org/study-designs/case-report"}],"awards":[],"funders":[],"has_content":{"pdf":false,"grobid_xml":false},"content_urls":null,"referenced_works_count":13,"referenced_works":["https://openalex.org/W1994698400","https://openalex.org/W2008006501","https://openalex.org/W2017861431","https://openalex.org/W2036623679","https://openalex.org/W2052097887","https://openalex.org/W2077199654","https://openalex.org/W2090224716","https://openalex.org/W2095038666","https://openalex.org/W2149679351","https://openalex.org/W2537029225","https://openalex.org/W2614590942","https://openalex.org/W2736086209","https://openalex.org/W2802831923"],"related_works":["https://openalex.org/W1970842543","https://openalex.org/W1994698400","https://openalex.org/W2017861431","https://openalex.org/W2471243515","https://openalex.org/W2807944728","https://openalex.org/W2040934932","https://openalex.org/W2546417782","https://openalex.org/W2055876879","https://openalex.org/W2393509161","https://openalex.org/W2523267837"],"abstract_inverted_index":{"Pyridoxine":[0,15,101],"(vitamin":[1],"B6)":[2],"is":[3,16,35,50],"an":[4],"essential":[5],"vitamin":[6,32,142],"playing":[7],"a":[8,40,59],"crucial":[9],"role":[10],"in":[11,84,109],"amino":[12],"acid":[13],"metabolism.":[14],"used":[17],"for":[18,71,144],"isoniazid":[19],"side-effects":[20],"prevention,":[21],"pyridoxine-dependent":[22],"epilepsy":[23],"treatment":[24],"and":[25,37,74,81,96,115,117],"cystathionine":[26],"beta-synthase":[27],"deficiency":[28],"(homocystinuria)":[29],"treatment.":[30],"However,":[31],"B6":[33,143],"hypervitaminosis":[34],"neurotoxic":[36],"may":[38,131],"provoke":[39],"progressive":[41,76],"sensory":[42,77,93,97,113,121,129],"neuronopathy":[43],"(sensory":[44],"ganglionopathy),":[45],"usually":[46],"when":[47],"daily":[48],"uptake":[49],"above":[51],"50":[52],"mg.":[53],"We":[54],"describe":[55],"the":[56,85,110,118],"case":[57,125],"of":[58,92,112,120,138,141],"30-year-old":[60],"patient":[61],"with":[62,67,79],"homocystinuria":[63],"who":[64],"was":[65,99,103],"treated":[66],"pyridoxine":[68],"1250-1750":[69],"mg/day":[70],"20":[72,147],"years":[73],"developed":[75],"neuropathy":[78],"ataxia":[80],"impaired":[82],"sensation":[83],"extremities.":[86],"Electrodiagnostic":[87],"testing":[88],"demonstrated":[89],"non-length-dependent":[90],"abnormalities":[91],"nerve":[94,122],"potentials,":[95],"ganglionopathy":[98,130],"diagnosed.":[100],"dosage":[102],"reduced":[104],"to":[105],"500":[106],"mg/day,":[107],"resulting":[108],"disappearance":[111],"symptoms":[114],"ataxia,":[116],"normalisation":[119],"potentials.":[123],"Our":[124],"indicates":[126],"that":[127],"pyridoxine-induced":[128],"be":[132],"reversible,":[133],"even":[134],"after":[135],"prolonged":[136],"ingestion":[137],"high":[139],"doses":[140],"more":[145],"than":[146],"years.":[148]},"counts_by_year":[{"year":2026,"cited_by_count":1},{"year":2025,"cited_by_count":3},{"year":2024,"cited_by_count":1},{"year":2023,"cited_by_count":2},{"year":2022,"cited_by_count":3},{"year":2021,"cited_by_count":4},{"year":2020,"cited_by_count":5},{"year":2019,"cited_by_count":2},{"year":2018,"cited_by_count":2}],"updated_date":"2026-10-31T12:09:11.096373","created_date":"2018-07-10T00:00:00"}