Ovoid palatal patch: a clue to anti-TIF1γ dermatomyositis
An 80-year-old woman presented with a several-year history of progressive hair loss and scalp pruritus. No other rashes or muscle weakness were noted on examination. Scalp biopsy showed interface dermatitis, dense perivascular and periadnexal lymphocytic infiltrate, mucin and scarring alopecia. Labo...
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| creator | Franciosi, Ellen Blankenship, Kaitlin Houk, Laura Rashighi, Mehdi |
| description | An 80-year-old woman presented with a several-year history of progressive hair loss and scalp pruritus. No other rashes or muscle weakness were noted on examination. Scalp biopsy showed interface dermatitis, dense perivascular and periadnexal lymphocytic infiltrate, mucin and scarring alopecia. Laboratory analysis did not show evidence of myositis. The patient was started on hydroxychloroquine for possible cutaneous lupus erythematosus. On follow-up, she presented with a new violaceous rash on the superior eyelids and a well-defined oval patch on the mid-hard palate suspicious for dermatomyositis. Myositis-specific autoantibodies revealed presence of anti-transcriptional intermediary factor-1γ (anti-TIF1γ) in the serum. Anti-TIF1γ autoantibody-positive dermatomyositis is a newly recognised subtype of dermatomyositis that is highly associated with amyopathic disease and has an increased risk of malignancy, making prompt diagnosis crucial. This case highlights the utility of a thorough oral exam in patients suspected to have connective tissue disease as the distinctive ovoid palatal patch is nearly pathognomonic for anti-TIF1γ dermatomyositis. |
| doi_str_mv | 10.1136/bcr-2019-234111 |
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No other rashes or muscle weakness were noted on examination. Scalp biopsy showed interface dermatitis, dense perivascular and periadnexal lymphocytic infiltrate, mucin and scarring alopecia. Laboratory analysis did not show evidence of myositis. The patient was started on hydroxychloroquine for possible cutaneous lupus erythematosus. On follow-up, she presented with a new violaceous rash on the superior eyelids and a well-defined oval patch on the mid-hard palate suspicious for dermatomyositis. Myositis-specific autoantibodies revealed presence of anti-transcriptional intermediary factor-1γ (anti-TIF1γ) in the serum. Anti-TIF1γ autoantibody-positive dermatomyositis is a newly recognised subtype of dermatomyositis that is highly associated with amyopathic disease and has an increased risk of malignancy, making prompt diagnosis crucial. This case highlights the utility of a thorough oral exam in patients suspected to have connective tissue disease as the distinctive ovoid palatal patch is nearly pathognomonic for anti-TIF1γ dermatomyositis.</description><identifier>ISSN: 1757-790X</identifier><identifier>EISSN: 1757-790X</identifier><identifier>DOI: 10.1136/bcr-2019-234111</identifier><identifier>PMID: 32332045</identifier><language>eng</language><publisher>England: BMJ Publishing Group LTD</publisher><subject>Aged, 80 and over ; Alopecia ; Autoantibodies - blood ; Baldness ; Cancer ; Case reports ; Dermatologic Agents - therapeutic use ; Dermatomyositis - diagnosis ; Dermatomyositis - drug therapy ; Diagnosis, Differential ; Erythema - pathology ; Exanthema - pathology ; Female ; Hair loss ; Humans ; Immunoglobulins ; Lupus ; Medical screening ; Methotrexate - therapeutic use ; Palate, Hard - pathology ; Patients ; Pruritus ; Rare Disease ; Rheumatology ; Transcription Factors - immunology ; Ultrasonic imaging</subject><ispartof>BMJ case reports, 2020-04, Vol.13 (4), p.e234111</ispartof><rights>BMJ Publishing Group Limited 2020. No commercial re-use. See rights and permissions. Published by BMJ.</rights><rights>2020 BMJ Publishing Group Limited 2020. No commercial re-use. See rights and permissions. Published by BMJ.</rights><rights>BMJ Publishing Group Limited 2020. No commercial re-use. See rights and permissions. Published by BMJ. 2020</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-b423t-2263aa33b808d369457dbe832ac23f087499ebaed850fd1f23c764012499b8de3</citedby><cites>FETCH-LOGICAL-b423t-2263aa33b808d369457dbe832ac23f087499ebaed850fd1f23c764012499b8de3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC7202738/pdf/$$EPDF$$P50$$Gpubmedcentral$$H</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC7202738/$$EHTML$$P50$$Gpubmedcentral$$H</linktohtml><link.rule.ids>230,314,727,780,784,885,27924,27925,53791,53793</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/32332045$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Franciosi, Ellen</creatorcontrib><creatorcontrib>Blankenship, Kaitlin</creatorcontrib><creatorcontrib>Houk, Laura</creatorcontrib><creatorcontrib>Rashighi, Mehdi</creatorcontrib><title>Ovoid palatal patch: a clue to anti-TIF1γ dermatomyositis</title><title>BMJ case reports</title><addtitle>BMJ Case Rep</addtitle><description>An 80-year-old woman presented with a several-year history of progressive hair loss and scalp pruritus. No other rashes or muscle weakness were noted on examination. Scalp biopsy showed interface dermatitis, dense perivascular and periadnexal lymphocytic infiltrate, mucin and scarring alopecia. Laboratory analysis did not show evidence of myositis. The patient was started on hydroxychloroquine for possible cutaneous lupus erythematosus. On follow-up, she presented with a new violaceous rash on the superior eyelids and a well-defined oval patch on the mid-hard palate suspicious for dermatomyositis. Myositis-specific autoantibodies revealed presence of anti-transcriptional intermediary factor-1γ (anti-TIF1γ) in the serum. Anti-TIF1γ autoantibody-positive dermatomyositis is a newly recognised subtype of dermatomyositis that is highly associated with amyopathic disease and has an increased risk of malignancy, making prompt diagnosis crucial. This case highlights the utility of a thorough oral exam in patients suspected to have connective tissue disease as the distinctive ovoid palatal patch is nearly pathognomonic for anti-TIF1γ dermatomyositis.</description><subject>Aged, 80 and over</subject><subject>Alopecia</subject><subject>Autoantibodies - blood</subject><subject>Baldness</subject><subject>Cancer</subject><subject>Case reports</subject><subject>Dermatologic Agents - therapeutic use</subject><subject>Dermatomyositis - diagnosis</subject><subject>Dermatomyositis - drug therapy</subject><subject>Diagnosis, Differential</subject><subject>Erythema - pathology</subject><subject>Exanthema - pathology</subject><subject>Female</subject><subject>Hair loss</subject><subject>Humans</subject><subject>Immunoglobulins</subject><subject>Lupus</subject><subject>Medical screening</subject><subject>Methotrexate - therapeutic use</subject><subject>Palate, Hard - pathology</subject><subject>Patients</subject><subject>Pruritus</subject><subject>Rare Disease</subject><subject>Rheumatology</subject><subject>Transcription Factors - immunology</subject><subject>Ultrasonic imaging</subject><issn>1757-790X</issn><issn>1757-790X</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2020</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><recordid>eNqFkM9KAzEQh4MottSevcmCN2Ftktk_2R4EKVYLhV4qeAvJJmu37G5qNi30uXwPn8mUrVJP5jJh8uU3w4fQNcH3hEAykrkNKSZZSCEihJyhPknjNEwz_HZ-cu-hYduusT9AIhbBJeoBBaA4ivtovNiZUgUbUQknKl9dvhoHIsirrQ6cCUTjynA5m5Kvz0BpWwtn6r1pS1e2V-iiEFWrh8c6QK_Tp-XkJZwvnmeTx3koIwoupDQBIQAkw0xBkkVxqqRmQEVOocAsjbJMS6EVi3GhSEEhT5MIE-r7kikNA_TQ5W62stYq142zouIbW9bC7rkRJf_70pQr_m52PKWYpsB8wO0xwJqPrW4dX5utbfzOnEIGmZ_GwFOjjsqtaVuri98JBPODb-5984Nv3vn2P25OF_vlf-x64K4DZL3-N-0bOSyIPw</recordid><startdate>20200423</startdate><enddate>20200423</enddate><creator>Franciosi, Ellen</creator><creator>Blankenship, Kaitlin</creator><creator>Houk, Laura</creator><creator>Rashighi, Mehdi</creator><general>BMJ Publishing Group LTD</general><general>BMJ Publishing Group</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>3V.</scope><scope>7RV</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>BENPR</scope><scope>BTHHO</scope><scope>CCPQU</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>K9.</scope><scope>KB0</scope><scope>M0S</scope><scope>M1P</scope><scope>NAPCQ</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>5PM</scope></search><sort><creationdate>20200423</creationdate><title>Ovoid palatal patch: a clue to anti-TIF1γ dermatomyositis</title><author>Franciosi, Ellen ; Blankenship, Kaitlin ; Houk, Laura ; Rashighi, Mehdi</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-b423t-2263aa33b808d369457dbe832ac23f087499ebaed850fd1f23c764012499b8de3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2020</creationdate><topic>Aged, 80 and over</topic><topic>Alopecia</topic><topic>Autoantibodies - blood</topic><topic>Baldness</topic><topic>Cancer</topic><topic>Case reports</topic><topic>Dermatologic Agents - therapeutic use</topic><topic>Dermatomyositis - diagnosis</topic><topic>Dermatomyositis - drug therapy</topic><topic>Diagnosis, Differential</topic><topic>Erythema - pathology</topic><topic>Exanthema - pathology</topic><topic>Female</topic><topic>Hair loss</topic><topic>Humans</topic><topic>Immunoglobulins</topic><topic>Lupus</topic><topic>Medical screening</topic><topic>Methotrexate - therapeutic use</topic><topic>Palate, Hard - pathology</topic><topic>Patients</topic><topic>Pruritus</topic><topic>Rare Disease</topic><topic>Rheumatology</topic><topic>Transcription Factors - immunology</topic><topic>Ultrasonic imaging</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Franciosi, Ellen</creatorcontrib><creatorcontrib>Blankenship, Kaitlin</creatorcontrib><creatorcontrib>Houk, Laura</creatorcontrib><creatorcontrib>Rashighi, Mehdi</creatorcontrib><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>Nursing & Allied Health Database</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</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 Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central</collection><collection>BMJ Journals</collection><collection>ProQuest One Community College</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Nursing & Allied Health Database (Alumni Edition)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>Nursing & Allied Health Premium</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>PubMed Central (Full Participant titles)</collection><jtitle>BMJ case reports</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Franciosi, Ellen</au><au>Blankenship, Kaitlin</au><au>Houk, Laura</au><au>Rashighi, Mehdi</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Ovoid palatal patch: a clue to anti-TIF1γ dermatomyositis</atitle><jtitle>BMJ case reports</jtitle><addtitle>BMJ Case Rep</addtitle><date>2020-04-23</date><risdate>2020</risdate><volume>13</volume><issue>4</issue><spage>e234111</spage><pages>e234111-</pages><issn>1757-790X</issn><eissn>1757-790X</eissn><abstract>An 80-year-old woman presented with a several-year history of progressive hair loss and scalp pruritus. No other rashes or muscle weakness were noted on examination. Scalp biopsy showed interface dermatitis, dense perivascular and periadnexal lymphocytic infiltrate, mucin and scarring alopecia. Laboratory analysis did not show evidence of myositis. The patient was started on hydroxychloroquine for possible cutaneous lupus erythematosus. On follow-up, she presented with a new violaceous rash on the superior eyelids and a well-defined oval patch on the mid-hard palate suspicious for dermatomyositis. Myositis-specific autoantibodies revealed presence of anti-transcriptional intermediary factor-1γ (anti-TIF1γ) in the serum. Anti-TIF1γ autoantibody-positive dermatomyositis is a newly recognised subtype of dermatomyositis that is highly associated with amyopathic disease and has an increased risk of malignancy, making prompt diagnosis crucial. This case highlights the utility of a thorough oral exam in patients suspected to have connective tissue disease as the distinctive ovoid palatal patch is nearly pathognomonic for anti-TIF1γ dermatomyositis.</abstract><cop>England</cop><pub>BMJ Publishing Group LTD</pub><pmid>32332045</pmid><doi>10.1136/bcr-2019-234111</doi><oa>free_for_read</oa></addata></record> |
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| subjects | Aged, 80 and over Alopecia Autoantibodies - blood Baldness Cancer Case reports Dermatologic Agents - therapeutic use Dermatomyositis - diagnosis Dermatomyositis - drug therapy Diagnosis, Differential Erythema - pathology Exanthema - pathology Female Hair loss Humans Immunoglobulins Lupus Medical screening Methotrexate - therapeutic use Palate, Hard - pathology Patients Pruritus Rare Disease Rheumatology Transcription Factors - immunology Ultrasonic imaging |
| title | Ovoid palatal patch: a clue to anti-TIF1γ dermatomyositis |
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