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Diagnosis and Clinical Development of Sporadic Inclusion Body Myositis and Polymyositis With Mitochondrial Pathology: A Single-Center Retrospective Analysis
Abstract To review our diagnostic and treatment approaches concerning sporadic inclusion body myositis (sIBM) and polymyositis with mitochondrial pathology (PM-Mito), we conducted a retrospective analysis of clinical and histological data of 32 patients diagnosed as sIBM and 7 patients diagnosed as...
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| Published in: | Journal of neuropathology and experimental neurology 2021-11, Vol.80 (11), p.1060-1067 |
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| container_issue | 11 |
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| container_title | Journal of neuropathology and experimental neurology |
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| creator | Winkler, Maren von Landenberg, Christina Kappes-Horn, Karin Neudecker, Stephan Kornblum, Cornelia Reimann, Jens |
| description | Abstract
To review our diagnostic and treatment approaches concerning sporadic inclusion body myositis (sIBM) and polymyositis with mitochondrial pathology (PM-Mito), we conducted a retrospective analysis of clinical and histological data of 32 patients diagnosed as sIBM and 7 patients diagnosed as PM-Mito by muscle biopsy. Of 32 patients identified histologically as sIBM, 19 fulfilled the 2011 European Neuromuscular Center (ENMC) diagnostic criteria for “clinico-pathologically defined sIBM” at the time of biopsy. Among these, 2 patients developed sIBM after years of immunosuppressive treatment for organ transplantation. Of 11 patients fulfilling the histological but not the clinical criteria, including 3 cases with duration |
| doi_str_mv | 10.1093/jnen/nlab101 |
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To review our diagnostic and treatment approaches concerning sporadic inclusion body myositis (sIBM) and polymyositis with mitochondrial pathology (PM-Mito), we conducted a retrospective analysis of clinical and histological data of 32 patients diagnosed as sIBM and 7 patients diagnosed as PM-Mito by muscle biopsy. Of 32 patients identified histologically as sIBM, 19 fulfilled the 2011 European Neuromuscular Center (ENMC) diagnostic criteria for “clinico-pathologically defined sIBM” at the time of biopsy. Among these, 2 patients developed sIBM after years of immunosuppressive treatment for organ transplantation. Of 11 patients fulfilling the histological but not the clinical criteria, including 3 cases with duration <12 months, 8 later fulfilled the criteria for clinico-pathologically defined sIBM. Of 7 PM-Mito patients, 4 received immunosuppression with clinical improvement in 3. One of these later developed clinico-pathologically defined sIBM; 1 untreated patient progressed to clinically defined sIBM. Thus, muscle histology remains important for this differential diagnosis to identify sIBM patients not matching the ENMC criteria and the PM-Mito group. In the latter, we report at least 50% positive, if occasionally transient, response to immunosuppressive treatments and progression to sIBM in a minority. The mitochondrial abnormalities defining PM-Mito do not seem to define the threshold to immunosuppression unresponsiveness.</description><identifier>ISSN: 0022-3069</identifier><identifier>EISSN: 1554-6578</identifier><identifier>DOI: 10.1093/jnen/nlab101</identifier><identifier>PMID: 34643702</identifier><language>eng</language><publisher>England: Oxford University Press</publisher><subject>Adenosine triphosphatase ; Aged ; Biopsy ; Care and treatment ; Cytotoxicity ; Development and progression ; Diagnosis ; Diagnosis, Differential ; Disease Progression ; Female ; Histology ; Humans ; Immunosuppressive Agents - therapeutic use ; Immunotherapy ; Inclusion body myositis ; Inflammatory diseases ; Laboratories ; Male ; Middle Aged ; Mitochondria - pathology ; Mitochondrial diseases ; Mitochondrial DNA ; Muscle, Skeletal - pathology ; Musculoskeletal diseases ; Myositis ; Myositis, Inclusion Body - diagnosis ; Myositis, Inclusion Body - drug therapy ; Myositis, Inclusion Body - pathology ; Neurology ; Neuromuscular diseases ; Organ Transplantation ; Pathology ; Polymyositis ; Polymyositis - drug therapy ; Polymyositis - pathology ; Proteins ; Retrospective Studies</subject><ispartof>Journal of neuropathology and experimental neurology, 2021-11, Vol.80 (11), p.1060-1067</ispartof><rights>2021 American Association of Neuropathologists, Inc. 2021</rights><rights>2021 American Association of Neuropathologists, Inc.</rights><rights>COPYRIGHT 2021 Oxford University Press</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c456t-e892e9ba9e20ca81c91dbbe91e4576b3a58aad1ceab83399499e2c8879e94ed93</citedby><cites>FETCH-LOGICAL-c456t-e892e9ba9e20ca81c91dbbe91e4576b3a58aad1ceab83399499e2c8879e94ed93</cites><orcidid>0000-0003-3349-6877 ; 0000-0002-0111-7281</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,780,784,27924,27925</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/34643702$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Winkler, Maren</creatorcontrib><creatorcontrib>von Landenberg, Christina</creatorcontrib><creatorcontrib>Kappes-Horn, Karin</creatorcontrib><creatorcontrib>Neudecker, Stephan</creatorcontrib><creatorcontrib>Kornblum, Cornelia</creatorcontrib><creatorcontrib>Reimann, Jens</creatorcontrib><title>Diagnosis and Clinical Development of Sporadic Inclusion Body Myositis and Polymyositis With Mitochondrial Pathology: A Single-Center Retrospective Analysis</title><title>Journal of neuropathology and experimental neurology</title><addtitle>J Neuropathol Exp Neurol</addtitle><description>Abstract
To review our diagnostic and treatment approaches concerning sporadic inclusion body myositis (sIBM) and polymyositis with mitochondrial pathology (PM-Mito), we conducted a retrospective analysis of clinical and histological data of 32 patients diagnosed as sIBM and 7 patients diagnosed as PM-Mito by muscle biopsy. Of 32 patients identified histologically as sIBM, 19 fulfilled the 2011 European Neuromuscular Center (ENMC) diagnostic criteria for “clinico-pathologically defined sIBM” at the time of biopsy. Among these, 2 patients developed sIBM after years of immunosuppressive treatment for organ transplantation. Of 11 patients fulfilling the histological but not the clinical criteria, including 3 cases with duration <12 months, 8 later fulfilled the criteria for clinico-pathologically defined sIBM. Of 7 PM-Mito patients, 4 received immunosuppression with clinical improvement in 3. One of these later developed clinico-pathologically defined sIBM; 1 untreated patient progressed to clinically defined sIBM. Thus, muscle histology remains important for this differential diagnosis to identify sIBM patients not matching the ENMC criteria and the PM-Mito group. In the latter, we report at least 50% positive, if occasionally transient, response to immunosuppressive treatments and progression to sIBM in a minority. The mitochondrial abnormalities defining PM-Mito do not seem to define the threshold to immunosuppression unresponsiveness.</description><subject>Adenosine triphosphatase</subject><subject>Aged</subject><subject>Biopsy</subject><subject>Care and treatment</subject><subject>Cytotoxicity</subject><subject>Development and progression</subject><subject>Diagnosis</subject><subject>Diagnosis, Differential</subject><subject>Disease Progression</subject><subject>Female</subject><subject>Histology</subject><subject>Humans</subject><subject>Immunosuppressive Agents - therapeutic use</subject><subject>Immunotherapy</subject><subject>Inclusion body myositis</subject><subject>Inflammatory diseases</subject><subject>Laboratories</subject><subject>Male</subject><subject>Middle Aged</subject><subject>Mitochondria - pathology</subject><subject>Mitochondrial diseases</subject><subject>Mitochondrial DNA</subject><subject>Muscle, Skeletal - pathology</subject><subject>Musculoskeletal diseases</subject><subject>Myositis</subject><subject>Myositis, Inclusion Body - diagnosis</subject><subject>Myositis, Inclusion Body - drug therapy</subject><subject>Myositis, Inclusion Body - pathology</subject><subject>Neurology</subject><subject>Neuromuscular diseases</subject><subject>Organ Transplantation</subject><subject>Pathology</subject><subject>Polymyositis</subject><subject>Polymyositis - drug therapy</subject><subject>Polymyositis - pathology</subject><subject>Proteins</subject><subject>Retrospective Studies</subject><issn>0022-3069</issn><issn>1554-6578</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2021</creationdate><recordtype>article</recordtype><sourceid>TOX</sourceid><recordid>eNp9kk9v1DAQxS0EotvCjTOyxAEOpLUTJ7F7W7b8qdSKioI4Ro4z2fXKsYPtVMp36YfFq92CQAj5YGn0e2-eZgahF5ScUiKKs60Fe2aNbCmhj9CCliXLqrLmj9GCkDzPClKJI3QcwpYQIohgT9FRwSpW1CRfoPsLLdfWBR2wtB1eGW21kgZfwB0YNw5gI3Y9vh2dl51W-NIqMwXtLH7nuhlfz0kaD-IbZ-bhofBdxw2-1tGpjbOd18nzRsaNM249n-MlvtV2bSBbpQbg8ReI3oURVNR3gJdWmjlFeoae9NIEeH74T9C3D--_rj5lV58_Xq6WV5liZRUz4CIH0UoBOVGSUyVo17YgKLCyrtpCllzKjiqQLS8KIZhIpOK8FiAYdKI4QW_2vqN3PyYIsRl0UGCMtOCm0OQlpzxnnNGEvvoL3brJp7yJqhhJ1mnev6m1NNBo27vopdqZNsu64ixngu7anv6DSq-DQStnodep_ofg7V6g0rCCh74ZvR6knxtKmt0xNLtjaA7HkPCXh6xTO0D3C37YfgJe7wE3jf-3-gmC8MBM</recordid><startdate>20211101</startdate><enddate>20211101</enddate><creator>Winkler, Maren</creator><creator>von Landenberg, Christina</creator><creator>Kappes-Horn, Karin</creator><creator>Neudecker, Stephan</creator><creator>Kornblum, Cornelia</creator><creator>Reimann, Jens</creator><general>Oxford University Press</general><scope>TOX</scope><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>3V.</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>88G</scope><scope>88I</scope><scope>8AF</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>AZQEC</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>DWQXO</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>GNUQQ</scope><scope>HCIFZ</scope><scope>K9.</scope><scope>M0S</scope><scope>M1P</scope><scope>M2P</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>Q9U</scope><scope>S0X</scope><scope>7X8</scope><orcidid>https://orcid.org/0000-0003-3349-6877</orcidid><orcidid>https://orcid.org/0000-0002-0111-7281</orcidid></search><sort><creationdate>20211101</creationdate><title>Diagnosis and Clinical Development of Sporadic Inclusion Body Myositis and Polymyositis With Mitochondrial Pathology: A Single-Center Retrospective Analysis</title><author>Winkler, Maren ; von Landenberg, Christina ; Kappes-Horn, Karin ; Neudecker, Stephan ; Kornblum, Cornelia ; Reimann, Jens</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c456t-e892e9ba9e20ca81c91dbbe91e4576b3a58aad1ceab83399499e2c8879e94ed93</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2021</creationdate><topic>Adenosine triphosphatase</topic><topic>Aged</topic><topic>Biopsy</topic><topic>Care and treatment</topic><topic>Cytotoxicity</topic><topic>Development and progression</topic><topic>Diagnosis</topic><topic>Diagnosis, Differential</topic><topic>Disease Progression</topic><topic>Female</topic><topic>Histology</topic><topic>Humans</topic><topic>Immunosuppressive Agents - therapeutic use</topic><topic>Immunotherapy</topic><topic>Inclusion body myositis</topic><topic>Inflammatory diseases</topic><topic>Laboratories</topic><topic>Male</topic><topic>Middle Aged</topic><topic>Mitochondria - pathology</topic><topic>Mitochondrial diseases</topic><topic>Mitochondrial DNA</topic><topic>Muscle, Skeletal - pathology</topic><topic>Musculoskeletal diseases</topic><topic>Myositis</topic><topic>Myositis, Inclusion Body - diagnosis</topic><topic>Myositis, Inclusion Body - drug therapy</topic><topic>Myositis, Inclusion Body - pathology</topic><topic>Neurology</topic><topic>Neuromuscular diseases</topic><topic>Organ Transplantation</topic><topic>Pathology</topic><topic>Polymyositis</topic><topic>Polymyositis - drug therapy</topic><topic>Polymyositis - pathology</topic><topic>Proteins</topic><topic>Retrospective Studies</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Winkler, Maren</creatorcontrib><creatorcontrib>von Landenberg, Christina</creatorcontrib><creatorcontrib>Kappes-Horn, Karin</creatorcontrib><creatorcontrib>Neudecker, Stephan</creatorcontrib><creatorcontrib>Kornblum, Cornelia</creatorcontrib><creatorcontrib>Reimann, Jens</creatorcontrib><collection>Oxford Academic Journals (Open Access)</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>Psychology Database (Alumni)</collection><collection>Science Database (Alumni Edition)</collection><collection>STEM Database</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni)</collection><collection>ProQuest Central</collection><collection>ProQuest Central Essentials</collection><collection>AUTh Library subscriptions: ProQuest Central</collection><collection>ProQuest One Community College</collection><collection>ProQuest Central</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Central Student</collection><collection>SciTech Premium Collection</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>Science Database</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>ProQuest Central Basic</collection><collection>SIRS Editorial</collection><collection>MEDLINE - Academic</collection><jtitle>Journal of neuropathology and experimental neurology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Winkler, Maren</au><au>von Landenberg, Christina</au><au>Kappes-Horn, Karin</au><au>Neudecker, Stephan</au><au>Kornblum, Cornelia</au><au>Reimann, Jens</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Diagnosis and Clinical Development of Sporadic Inclusion Body Myositis and Polymyositis With Mitochondrial Pathology: A Single-Center Retrospective Analysis</atitle><jtitle>Journal of neuropathology and experimental neurology</jtitle><addtitle>J Neuropathol Exp Neurol</addtitle><date>2021-11-01</date><risdate>2021</risdate><volume>80</volume><issue>11</issue><spage>1060</spage><epage>1067</epage><pages>1060-1067</pages><issn>0022-3069</issn><eissn>1554-6578</eissn><abstract>Abstract
To review our diagnostic and treatment approaches concerning sporadic inclusion body myositis (sIBM) and polymyositis with mitochondrial pathology (PM-Mito), we conducted a retrospective analysis of clinical and histological data of 32 patients diagnosed as sIBM and 7 patients diagnosed as PM-Mito by muscle biopsy. Of 32 patients identified histologically as sIBM, 19 fulfilled the 2011 European Neuromuscular Center (ENMC) diagnostic criteria for “clinico-pathologically defined sIBM” at the time of biopsy. Among these, 2 patients developed sIBM after years of immunosuppressive treatment for organ transplantation. Of 11 patients fulfilling the histological but not the clinical criteria, including 3 cases with duration <12 months, 8 later fulfilled the criteria for clinico-pathologically defined sIBM. Of 7 PM-Mito patients, 4 received immunosuppression with clinical improvement in 3. One of these later developed clinico-pathologically defined sIBM; 1 untreated patient progressed to clinically defined sIBM. Thus, muscle histology remains important for this differential diagnosis to identify sIBM patients not matching the ENMC criteria and the PM-Mito group. In the latter, we report at least 50% positive, if occasionally transient, response to immunosuppressive treatments and progression to sIBM in a minority. The mitochondrial abnormalities defining PM-Mito do not seem to define the threshold to immunosuppression unresponsiveness.</abstract><cop>England</cop><pub>Oxford University Press</pub><pmid>34643702</pmid><doi>10.1093/jnen/nlab101</doi><tpages>8</tpages><orcidid>https://orcid.org/0000-0003-3349-6877</orcidid><orcidid>https://orcid.org/0000-0002-0111-7281</orcidid><oa>free_for_read</oa></addata></record> |
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| subjects | Adenosine triphosphatase Aged Biopsy Care and treatment Cytotoxicity Development and progression Diagnosis Diagnosis, Differential Disease Progression Female Histology Humans Immunosuppressive Agents - therapeutic use Immunotherapy Inclusion body myositis Inflammatory diseases Laboratories Male Middle Aged Mitochondria - pathology Mitochondrial diseases Mitochondrial DNA Muscle, Skeletal - pathology Musculoskeletal diseases Myositis Myositis, Inclusion Body - diagnosis Myositis, Inclusion Body - drug therapy Myositis, Inclusion Body - pathology Neurology Neuromuscular diseases Organ Transplantation Pathology Polymyositis Polymyositis - drug therapy Polymyositis - pathology Proteins Retrospective Studies |
| title | Diagnosis and Clinical Development of Sporadic Inclusion Body Myositis and Polymyositis With Mitochondrial Pathology: A Single-Center Retrospective Analysis |
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