A prospective, observational study of patients with uncommon distal symmetric painful small-fiber neuropathy
To investigate the clinical characteristics of patients with uncommon distal symmetric painful small-fiber neuropathy (DSPSFN). From September 2012 to September 2014, participants between 18-70 years of age that had DSPSFN defined by clinical signs/symptoms and ID pain > 2 or DN4 > 4 on questi...
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| Veröffentlicht in: | PloS one 2017-09, Vol.12 (9), p.e0183948-e0183948 |
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| creator | Hsu, Jung-Lung Liao, Ming-Feng Hsu, Hui-Ching Weng, Yi-Ching Lo, Ai-Lun Chang, Kuo-Hsuan Chang, Hong-Shiu Kuo, Hung-Chou Huang, Chin-Chang Ro, Long-Sun |
| description | To investigate the clinical characteristics of patients with uncommon distal symmetric painful small-fiber neuropathy (DSPSFN).
From September 2012 to September 2014, participants between 18-70 years of age that had DSPSFN defined by clinical signs/symptoms and ID pain > 2 or DN4 > 4 on questionnaires for more than 1 month were included. Participants who had previous historical or laboratory evidence of common etiologies of DSPSFN were excluded. Enzyme activity and genetic studies for Fabry diseaseand familial amyloid polyneuropathy were performed after participants fulfilled the inclusion and exclusion criteria. The cryoglobulin test, autoantibodies studies and electrophysiological studies were performed in these participants.
In total, 100 cases were enrolled in the current study. Three cases of subclinical diabetes mellitus and two cases of fibromyalgia were found. Fabry disease (1%) and familial amyloid polyneuropathy (3%) with Ala97Ser transthyretin (TTR) mutations were also detected. The cryoglobulin test was positive in 30% of participants, and these participants had higher DN4 scores than the negative group. In the autoantibodies studies, 59% of the participants had abnormal anti-Ro/SSA and/or anti-La/SSB antibodies.
Cryoglobulinemia is not a rare etiology of uncommon DSPSFN. The long-term prognosis is quite good in these participants. From our structuralized protocol, Fabry disease and familial amyloid polyneuropathy could be easily detected in these cases of uncommon DSPSFN. |
| doi_str_mv | 10.1371/journal.pone.0183948 |
| format | Article |
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From September 2012 to September 2014, participants between 18-70 years of age that had DSPSFN defined by clinical signs/symptoms and ID pain > 2 or DN4 > 4 on questionnaires for more than 1 month were included. Participants who had previous historical or laboratory evidence of common etiologies of DSPSFN were excluded. Enzyme activity and genetic studies for Fabry diseaseand familial amyloid polyneuropathy were performed after participants fulfilled the inclusion and exclusion criteria. The cryoglobulin test, autoantibodies studies and electrophysiological studies were performed in these participants.
In total, 100 cases were enrolled in the current study. Three cases of subclinical diabetes mellitus and two cases of fibromyalgia were found. Fabry disease (1%) and familial amyloid polyneuropathy (3%) with Ala97Ser transthyretin (TTR) mutations were also detected. The cryoglobulin test was positive in 30% of participants, and these participants had higher DN4 scores than the negative group. In the autoantibodies studies, 59% of the participants had abnormal anti-Ro/SSA and/or anti-La/SSB antibodies.
Cryoglobulinemia is not a rare etiology of uncommon DSPSFN. The long-term prognosis is quite good in these participants. From our structuralized protocol, Fabry disease and familial amyloid polyneuropathy could be easily detected in these cases of uncommon DSPSFN.</description><identifier>ISSN: 1932-6203</identifier><identifier>EISSN: 1932-6203</identifier><identifier>DOI: 10.1371/journal.pone.0183948</identifier><identifier>PMID: 28957343</identifier><language>eng</language><publisher>United States: Public Library of Science</publisher><subject>Antibodies ; Autoantibodies ; Autoantibodies - immunology ; Biochemistry ; Biology and Life Sciences ; Biopsy ; Care and treatment ; Chinese medicine ; Complications and side effects ; Cryoglobulinemia ; Cryoglobulinemia - complications ; Demographic aspects ; Demography ; Development and progression ; Diabetes ; Diabetes mellitus ; Diabetic neuropathy ; Diagnosis ; Electrophysiological Phenomena ; Enzymatic activity ; Enzyme activity ; Etiology ; Fabry's disease ; Female ; Fibromyalgia ; Hepatitis ; HIV ; Hospitals ; Human immunodeficiency virus ; Humans ; Infections ; Kidney diseases ; Laboratories ; Male ; Medicine and Health Sciences ; Metabolism ; Middle Aged ; Mutation ; Neurology ; Neuropathy ; Observational studies ; Pain ; Patients ; Peripheral neuropathy ; Physicians ; Polyneuropathy ; Prospective Studies ; Research and Analysis Methods ; Risk factors ; Ro(SSA) antigen ; Small Fiber Neuropathy - complications ; Small Fiber Neuropathy - diagnosis ; Small Fiber Neuropathy - immunology ; Small Fiber Neuropathy - physiopathology ; Transthyretin ; β-Amyloid</subject><ispartof>PloS one, 2017-09, Vol.12 (9), p.e0183948-e0183948</ispartof><rights>COPYRIGHT 2017 Public Library of Science</rights><rights>2017 Hsu et al. This is an open access article distributed under the terms of the Creative Commons Attribution License: http://creativecommons.org/licenses/by/4.0/ (the “License”), which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.</rights><rights>2017 Hsu et al 2017 Hsu et al</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c692t-edfa1db6a53f1bb775a4de379ca32de7b05675f301aa7ebd0d0267b24424c0253</citedby><cites>FETCH-LOGICAL-c692t-edfa1db6a53f1bb775a4de379ca32de7b05675f301aa7ebd0d0267b24424c0253</cites><orcidid>0000-0001-6422-8870</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC5619719/pdf/$$EPDF$$P50$$Gpubmedcentral$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC5619719/$$EHTML$$P50$$Gpubmedcentral$$Hfree_for_read</linktohtml><link.rule.ids>230,314,727,780,784,864,885,2102,2928,23866,27924,27925,53791,53793,79600,79601</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/28957343$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><contributor>Malik, Rayaz Ahmed</contributor><creatorcontrib>Hsu, Jung-Lung</creatorcontrib><creatorcontrib>Liao, Ming-Feng</creatorcontrib><creatorcontrib>Hsu, Hui-Ching</creatorcontrib><creatorcontrib>Weng, Yi-Ching</creatorcontrib><creatorcontrib>Lo, Ai-Lun</creatorcontrib><creatorcontrib>Chang, Kuo-Hsuan</creatorcontrib><creatorcontrib>Chang, Hong-Shiu</creatorcontrib><creatorcontrib>Kuo, Hung-Chou</creatorcontrib><creatorcontrib>Huang, Chin-Chang</creatorcontrib><creatorcontrib>Ro, Long-Sun</creatorcontrib><title>A prospective, observational study of patients with uncommon distal symmetric painful small-fiber neuropathy</title><title>PloS one</title><addtitle>PLoS One</addtitle><description>To investigate the clinical characteristics of patients with uncommon distal symmetric painful small-fiber neuropathy (DSPSFN).
From September 2012 to September 2014, participants between 18-70 years of age that had DSPSFN defined by clinical signs/symptoms and ID pain > 2 or DN4 > 4 on questionnaires for more than 1 month were included. Participants who had previous historical or laboratory evidence of common etiologies of DSPSFN were excluded. Enzyme activity and genetic studies for Fabry diseaseand familial amyloid polyneuropathy were performed after participants fulfilled the inclusion and exclusion criteria. The cryoglobulin test, autoantibodies studies and electrophysiological studies were performed in these participants.
In total, 100 cases were enrolled in the current study. Three cases of subclinical diabetes mellitus and two cases of fibromyalgia were found. Fabry disease (1%) and familial amyloid polyneuropathy (3%) with Ala97Ser transthyretin (TTR) mutations were also detected. The cryoglobulin test was positive in 30% of participants, and these participants had higher DN4 scores than the negative group. In the autoantibodies studies, 59% of the participants had abnormal anti-Ro/SSA and/or anti-La/SSB antibodies.
Cryoglobulinemia is not a rare etiology of uncommon DSPSFN. The long-term prognosis is quite good in these participants. From our structuralized protocol, Fabry disease and familial amyloid polyneuropathy could be easily detected in these cases of uncommon DSPSFN.</description><subject>Antibodies</subject><subject>Autoantibodies</subject><subject>Autoantibodies - immunology</subject><subject>Biochemistry</subject><subject>Biology and Life Sciences</subject><subject>Biopsy</subject><subject>Care and treatment</subject><subject>Chinese medicine</subject><subject>Complications and side effects</subject><subject>Cryoglobulinemia</subject><subject>Cryoglobulinemia - complications</subject><subject>Demographic aspects</subject><subject>Demography</subject><subject>Development and progression</subject><subject>Diabetes</subject><subject>Diabetes mellitus</subject><subject>Diabetic neuropathy</subject><subject>Diagnosis</subject><subject>Electrophysiological Phenomena</subject><subject>Enzymatic activity</subject><subject>Enzyme activity</subject><subject>Etiology</subject><subject>Fabry's disease</subject><subject>Female</subject><subject>Fibromyalgia</subject><subject>Hepatitis</subject><subject>HIV</subject><subject>Hospitals</subject><subject>Human immunodeficiency virus</subject><subject>Humans</subject><subject>Infections</subject><subject>Kidney diseases</subject><subject>Laboratories</subject><subject>Male</subject><subject>Medicine and Health Sciences</subject><subject>Metabolism</subject><subject>Middle Aged</subject><subject>Mutation</subject><subject>Neurology</subject><subject>Neuropathy</subject><subject>Observational studies</subject><subject>Pain</subject><subject>Patients</subject><subject>Peripheral neuropathy</subject><subject>Physicians</subject><subject>Polyneuropathy</subject><subject>Prospective Studies</subject><subject>Research and Analysis Methods</subject><subject>Risk factors</subject><subject>Ro(SSA) antigen</subject><subject>Small Fiber Neuropathy - complications</subject><subject>Small Fiber Neuropathy - diagnosis</subject><subject>Small Fiber Neuropathy - immunology</subject><subject>Small Fiber Neuropathy - physiopathology</subject><subject>Transthyretin</subject><subject>β-Amyloid</subject><issn>1932-6203</issn><issn>1932-6203</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2017</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>AZQEC</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><sourceid>DWQXO</sourceid><sourceid>GNUQQ</sourceid><sourceid>DOA</sourceid><recordid>eNqNk12L1DAUhoso7jr6D0QLgig4Yz6aprkRhsWPgYUFv25DmqQzWdKmJuno_HvTne4ylb2QXrScPuc9yXvOybLnEKwgpvD9tRt8J-yqd51eAVhhVlQPsnPIMFqWCOCHJ99n2ZMQrgEguCrLx9kZqhihuMDnmV3nvXeh1zKavX6XuzpovxfRuKSdhzioQ-6avE8R3cWQ_zZxlw-ddG3rulyZEEfs0LY6eiMTZ7pmSJFWWLtsTK193unBuySwOzzNHjXCBv1sei-yH58-fr_4sry8-ry5WF8uZclQXGrVCKjqUhDcwLqmlIhCaUyZFBgpTWtASkoaDKAQVNcKKIBKWqOiQIUEiOBF9vKo21sX-GRU4JAlgpKCwURsjoRy4pr33rTCH7gTht8EnN9y4aORVnOMBVZUEQmkKhoJRQ1oSVihKlaKBoKk9WGqNtStVjL55IWdic7_dGbHt27PSQkZTT1aZG8mAe9-DTpE3pogtbWi0264OTdBCGE81nr1D3r_7SZqK9IFUkdcqitHUb4mgEKAWDW6tLqHSo_SrZFpqhqT4rOEt7OExET9J27FEALffPv6_-zVzzn7-oTdaWHjLjg7jEMY5mBxBGUa2eB1c2cyBHxcils3-LgUfFqKlPbitEF3SbdbgP8CblUJbg</recordid><startdate>20170928</startdate><enddate>20170928</enddate><creator>Hsu, Jung-Lung</creator><creator>Liao, Ming-Feng</creator><creator>Hsu, Hui-Ching</creator><creator>Weng, Yi-Ching</creator><creator>Lo, Ai-Lun</creator><creator>Chang, Kuo-Hsuan</creator><creator>Chang, Hong-Shiu</creator><creator>Kuo, Hung-Chou</creator><creator>Huang, Chin-Chang</creator><creator>Ro, Long-Sun</creator><general>Public Library of Science</general><general>Public Library of Science (PLoS)</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>IOV</scope><scope>ISR</scope><scope>3V.</scope><scope>7QG</scope><scope>7QL</scope><scope>7QO</scope><scope>7RV</scope><scope>7SN</scope><scope>7SS</scope><scope>7T5</scope><scope>7TG</scope><scope>7TM</scope><scope>7U9</scope><scope>7X2</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8AO</scope><scope>8C1</scope><scope>8FD</scope><scope>8FE</scope><scope>8FG</scope><scope>8FH</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABJCF</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>ARAPS</scope><scope>ATCPS</scope><scope>AZQEC</scope><scope>BBNVY</scope><scope>BENPR</scope><scope>BGLVJ</scope><scope>BHPHI</scope><scope>C1K</scope><scope>CCPQU</scope><scope>D1I</scope><scope>DWQXO</scope><scope>FR3</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>GNUQQ</scope><scope>H94</scope><scope>HCIFZ</scope><scope>K9.</scope><scope>KB.</scope><scope>KB0</scope><scope>KL.</scope><scope>L6V</scope><scope>LK8</scope><scope>M0K</scope><scope>M0S</scope><scope>M1P</scope><scope>M7N</scope><scope>M7P</scope><scope>M7S</scope><scope>NAPCQ</scope><scope>P5Z</scope><scope>P62</scope><scope>P64</scope><scope>PATMY</scope><scope>PDBOC</scope><scope>PIMPY</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PTHSS</scope><scope>PYCSY</scope><scope>RC3</scope><scope>7X8</scope><scope>5PM</scope><scope>DOA</scope><orcidid>https://orcid.org/0000-0001-6422-8870</orcidid></search><sort><creationdate>20170928</creationdate><title>A prospective, observational study of patients with uncommon distal symmetric painful small-fiber neuropathy</title><author>Hsu, Jung-Lung ; Liao, Ming-Feng ; Hsu, Hui-Ching ; Weng, Yi-Ching ; Lo, Ai-Lun ; Chang, Kuo-Hsuan ; Chang, Hong-Shiu ; Kuo, Hung-Chou ; Huang, Chin-Chang ; Ro, Long-Sun</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c692t-edfa1db6a53f1bb775a4de379ca32de7b05675f301aa7ebd0d0267b24424c0253</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2017</creationdate><topic>Antibodies</topic><topic>Autoantibodies</topic><topic>Autoantibodies - immunology</topic><topic>Biochemistry</topic><topic>Biology and Life Sciences</topic><topic>Biopsy</topic><topic>Care and treatment</topic><topic>Chinese medicine</topic><topic>Complications and side effects</topic><topic>Cryoglobulinemia</topic><topic>Cryoglobulinemia - complications</topic><topic>Demographic aspects</topic><topic>Demography</topic><topic>Development and progression</topic><topic>Diabetes</topic><topic>Diabetes mellitus</topic><topic>Diabetic neuropathy</topic><topic>Diagnosis</topic><topic>Electrophysiological Phenomena</topic><topic>Enzymatic activity</topic><topic>Enzyme activity</topic><topic>Etiology</topic><topic>Fabry's disease</topic><topic>Female</topic><topic>Fibromyalgia</topic><topic>Hepatitis</topic><topic>HIV</topic><topic>Hospitals</topic><topic>Human immunodeficiency virus</topic><topic>Humans</topic><topic>Infections</topic><topic>Kidney diseases</topic><topic>Laboratories</topic><topic>Male</topic><topic>Medicine and Health Sciences</topic><topic>Metabolism</topic><topic>Middle Aged</topic><topic>Mutation</topic><topic>Neurology</topic><topic>Neuropathy</topic><topic>Observational studies</topic><topic>Pain</topic><topic>Patients</topic><topic>Peripheral neuropathy</topic><topic>Physicians</topic><topic>Polyneuropathy</topic><topic>Prospective Studies</topic><topic>Research and Analysis Methods</topic><topic>Risk factors</topic><topic>Ro(SSA) antigen</topic><topic>Small Fiber Neuropathy - 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Academic</collection><collection>PubMed Central (Full Participant titles)</collection><collection>DOAJ Directory of Open Access Journals</collection><jtitle>PloS one</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Hsu, Jung-Lung</au><au>Liao, Ming-Feng</au><au>Hsu, Hui-Ching</au><au>Weng, Yi-Ching</au><au>Lo, Ai-Lun</au><au>Chang, Kuo-Hsuan</au><au>Chang, Hong-Shiu</au><au>Kuo, Hung-Chou</au><au>Huang, Chin-Chang</au><au>Ro, Long-Sun</au><au>Malik, Rayaz Ahmed</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>A prospective, observational study of patients with uncommon distal symmetric painful small-fiber neuropathy</atitle><jtitle>PloS one</jtitle><addtitle>PLoS One</addtitle><date>2017-09-28</date><risdate>2017</risdate><volume>12</volume><issue>9</issue><spage>e0183948</spage><epage>e0183948</epage><pages>e0183948-e0183948</pages><issn>1932-6203</issn><eissn>1932-6203</eissn><abstract>To investigate the clinical characteristics of patients with uncommon distal symmetric painful small-fiber neuropathy (DSPSFN).
From September 2012 to September 2014, participants between 18-70 years of age that had DSPSFN defined by clinical signs/symptoms and ID pain > 2 or DN4 > 4 on questionnaires for more than 1 month were included. Participants who had previous historical or laboratory evidence of common etiologies of DSPSFN were excluded. Enzyme activity and genetic studies for Fabry diseaseand familial amyloid polyneuropathy were performed after participants fulfilled the inclusion and exclusion criteria. The cryoglobulin test, autoantibodies studies and electrophysiological studies were performed in these participants.
In total, 100 cases were enrolled in the current study. Three cases of subclinical diabetes mellitus and two cases of fibromyalgia were found. Fabry disease (1%) and familial amyloid polyneuropathy (3%) with Ala97Ser transthyretin (TTR) mutations were also detected. The cryoglobulin test was positive in 30% of participants, and these participants had higher DN4 scores than the negative group. In the autoantibodies studies, 59% of the participants had abnormal anti-Ro/SSA and/or anti-La/SSB antibodies.
Cryoglobulinemia is not a rare etiology of uncommon DSPSFN. The long-term prognosis is quite good in these participants. From our structuralized protocol, Fabry disease and familial amyloid polyneuropathy could be easily detected in these cases of uncommon DSPSFN.</abstract><cop>United States</cop><pub>Public Library of Science</pub><pmid>28957343</pmid><doi>10.1371/journal.pone.0183948</doi><tpages>e0183948</tpages><orcidid>https://orcid.org/0000-0001-6422-8870</orcidid><oa>free_for_read</oa></addata></record> |
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| recordid | cdi_plos_journals_1944275491 |
| source | MEDLINE; DOAJ Directory of Open Access Journals; Elektronische Zeitschriftenbibliothek - Frei zugängliche E-Journals; Public Library of Science (PLoS); PubMed Central; Free Full-Text Journals in Chemistry |
| subjects | Antibodies Autoantibodies Autoantibodies - immunology Biochemistry Biology and Life Sciences Biopsy Care and treatment Chinese medicine Complications and side effects Cryoglobulinemia Cryoglobulinemia - complications Demographic aspects Demography Development and progression Diabetes Diabetes mellitus Diabetic neuropathy Diagnosis Electrophysiological Phenomena Enzymatic activity Enzyme activity Etiology Fabry's disease Female Fibromyalgia Hepatitis HIV Hospitals Human immunodeficiency virus Humans Infections Kidney diseases Laboratories Male Medicine and Health Sciences Metabolism Middle Aged Mutation Neurology Neuropathy Observational studies Pain Patients Peripheral neuropathy Physicians Polyneuropathy Prospective Studies Research and Analysis Methods Risk factors Ro(SSA) antigen Small Fiber Neuropathy - complications Small Fiber Neuropathy - diagnosis Small Fiber Neuropathy - immunology Small Fiber Neuropathy - physiopathology Transthyretin β-Amyloid |
| title | A prospective, observational study of patients with uncommon distal symmetric painful small-fiber neuropathy |
| url | https://sfx.bib-bvb.de/sfx_tum?ctx_ver=Z39.88-2004&ctx_enc=info:ofi/enc:UTF-8&ctx_tim=2025-01-04T16%3A21%3A48IST&url_ver=Z39.88-2004&url_ctx_fmt=infofi/fmt:kev:mtx:ctx&rfr_id=info:sid/primo.exlibrisgroup.com:primo3-Article-gale_plos_&rft_val_fmt=info:ofi/fmt:kev:mtx:journal&rft.genre=article&rft.atitle=A%20prospective,%20observational%20study%20of%20patients%20with%20uncommon%20distal%20symmetric%20painful%20small-fiber%20neuropathy&rft.jtitle=PloS%20one&rft.au=Hsu,%20Jung-Lung&rft.date=2017-09-28&rft.volume=12&rft.issue=9&rft.spage=e0183948&rft.epage=e0183948&rft.pages=e0183948-e0183948&rft.issn=1932-6203&rft.eissn=1932-6203&rft_id=info:doi/10.1371/journal.pone.0183948&rft_dat=%3Cgale_plos_%3EA507102985%3C/gale_plos_%3E%3Curl%3E%3C/url%3E&disable_directlink=true&sfx.directlink=off&sfx.report_link=0&rft_id=info:oai/&rft_pqid=1944275491&rft_id=info:pmid/28957343&rft_galeid=A507102985&rft_doaj_id=oai_doaj_org_article_33a3d7d5c0cd4fc1ab076594d896af10&rfr_iscdi=true |