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Outcomes of 847 childhood-onset systemic lupus erythematosus patients in three age groups
Objective The objective of this study was to assess outcomes of childhood systemic lupus erythematosus (cSLE) in three different age groups evaluated at last visit: group A early-onset disease (
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| Published in: | Lupus 2017-08, Vol.26 (9), p.996-1001 |
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| creator | Lopes, S R M Gormezano, N W S Gomes, R C Aikawa, N E Pereira, R M R Terreri, M T Magalhães, C S Ferreira, J C Okuda, E M Sakamoto, A P Sallum, A M E Appenzeller, S Ferriani, V P L Barbosa, C M Lotufo, S Jesus, A A Andrade, L E C Campos, L M A Bonfá, E Silva, C A |
| description | Objective
The objective of this study was to assess outcomes of childhood systemic lupus erythematosus (cSLE) in three different age groups evaluated at last visit: group A early-onset disease ( |
| doi_str_mv | 10.1177/0961203317690616 |
| format | article |
| fullrecord | <record><control><sourceid>proquest_cross</sourceid><recordid>TN_cdi_proquest_miscellaneous_1862936900</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sage_id>10.1177_0961203317690616</sage_id><sourcerecordid>1862936900</sourcerecordid><originalsourceid>FETCH-LOGICAL-c379t-ec6b273013130af14e34104a2303aff29b80499d32839150ee4d189c4a66a1a83</originalsourceid><addsrcrecordid>eNp1kL1PwzAQxS0EouVjZ0IeWQJ3sXHiEVV8SZVYYGCK3ORCg5I4-Jyh_z2pWhiQmE5373dPek-IC4RrxCy7AWswBaUwMxYMmgMxR51lyXRPD8V8KydbfSZOmD8BQKE1x2KW5qg0qHwu3l_GWPqOWPpa5jqT5bppq7X3VeJ7pih5w5G6ppTtOIwsKWzimjoXPU_b4GJDfWTZ9DKuA5F0HyQ_gh8HPhNHtWuZzvfzVLw93L8unpLly-Pz4m6ZlCqzMaHSrNJMASpU4GrUpDSCdqkC5eo6tasctLWVSnNl8RaIdIW5LbUzxqHL1am42vkOwX-NxLHoGi6pbV1PfuQCc5NaNfUDEwo7tAyeOVBdDKHpXNgUCMW20OJvodPL5d59XHVU_T78NDgByQ7gKXrx6cfQT2n_N_wGkNd8gw</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>1862936900</pqid></control><display><type>article</type><title>Outcomes of 847 childhood-onset systemic lupus erythematosus patients in three age groups</title><source>SAGE Journals</source><creator>Lopes, S R M ; Gormezano, N W S ; Gomes, R C ; Aikawa, N E ; Pereira, R M R ; Terreri, M T ; Magalhães, C S ; Ferreira, J C ; Okuda, E M ; Sakamoto, A P ; Sallum, A M E ; Appenzeller, S ; Ferriani, V P L ; Barbosa, C M ; Lotufo, S ; Jesus, A A ; Andrade, L E C ; Campos, L M A ; Bonfá, E ; Silva, C A</creator><creatorcontrib>Lopes, S R M ; Gormezano, N W S ; Gomes, R C ; Aikawa, N E ; Pereira, R M R ; Terreri, M T ; Magalhães, C S ; Ferreira, J C ; Okuda, E M ; Sakamoto, A P ; Sallum, A M E ; Appenzeller, S ; Ferriani, V P L ; Barbosa, C M ; Lotufo, S ; Jesus, A A ; Andrade, L E C ; Campos, L M A ; Bonfá, E ; Silva, C A ; Brazilian Childhood-onset Systemic Lupus Erythematosus Group ; Brazilian Childhood-onset Systemic Lupus Erythematosus Group</creatorcontrib><description>Objective
The objective of this study was to assess outcomes of childhood systemic lupus erythematosus (cSLE) in three different age groups evaluated at last visit: group A early-onset disease (<6 years), group B school age (≥6 and <12 years) and group C adolescent (≥12 and <18 years).
Methods
An observational cohort study was performed in ten pediatric rheumatology centers, including 847 cSLE patients.
Results
Group A had 39 (4%), B 395 (47%) and C 413 (49%). Median disease duration was significantly higher in group A compared to groups B and C (8.3 (0.1–23.4) vs 6.2 (0–17) vs 3.3 (0–14.6) years, p < 0.0001). The median Systemic Lupus International Collaborating Clinics/American College of Rheumatology Damage Index (SLICC/ACR-DI) (0 (0–9) vs 0 (0–6) vs 0 (0–7), p = 0.065) was comparable in the three groups. Further analysis of organ/system damage revealed that frequencies of neuropsychiatric (21% vs 10% vs 7%, p = 0.007), skin (10% vs 1% vs 3%, p = 0.002) and peripheral vascular involvements (5% vs 3% vs 0.3%, p = 0.008) were more often observed in group A compared to groups B and C. Frequencies of severe cumulative lupus manifestations such as nephritis, thrombocytopenia, and autoimmune hemolytic anemia were similar in all groups (p > 0.05). Mortality rate was significantly higher in group A compared to groups B and C (15% vs 10% vs 6%, p = 0.028). Out of 69 deaths, 33/69 (48%) occurred within the first two years after diagnosis. Infections accounted for 54/69 (78%) of the deaths and 38/54 (70%) had concomitant disease activity.
Conclusions
This large multicenter study provided evidence that early-onset cSLE group had distinct outcomes. This group was characterized by higher mortality rate and neuropsychiatric/vascular/skin organ damage in spite of comparable frequencies of severe cumulative lupus manifestations. We also identified that overall death in cSLE patients was an early event mainly attributed to infection associated with disease activity.</description><identifier>ISSN: 0961-2033</identifier><identifier>EISSN: 1477-0962</identifier><identifier>DOI: 10.1177/0961203317690616</identifier><identifier>PMID: 28134038</identifier><language>eng</language><publisher>London, England: SAGE Publications</publisher><subject>Adolescent ; Age of Onset ; Anemia, Hemolytic, Autoimmune - complications ; Anemia, Hemolytic, Autoimmune - diagnosis ; Anemia, Hemolytic, Autoimmune - pathology ; Brazil - epidemiology ; Child ; Child, Preschool ; Cohort Studies ; Female ; Humans ; Immunosuppressive Agents - therapeutic use ; Infant ; Lupus Erythematosus, Systemic - complications ; Lupus Erythematosus, Systemic - drug therapy ; Lupus Erythematosus, Systemic - epidemiology ; Lupus Erythematosus, Systemic - mortality ; Mortality ; Nephritis - complications ; Nephritis - diagnosis ; Nephritis - epidemiology ; Nephritis - mortality ; Pregnancy ; Retrospective Studies ; Severity of Illness Index ; Thrombocytopenia - complications ; Thrombocytopenia - diagnosis ; Thrombocytopenia - pathology ; Treatment Outcome</subject><ispartof>Lupus, 2017-08, Vol.26 (9), p.996-1001</ispartof><rights>The Author(s) 2017</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c379t-ec6b273013130af14e34104a2303aff29b80499d32839150ee4d189c4a66a1a83</citedby><cites>FETCH-LOGICAL-c379t-ec6b273013130af14e34104a2303aff29b80499d32839150ee4d189c4a66a1a83</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,780,784,27924,27925,79364</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/28134038$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Lopes, S R M</creatorcontrib><creatorcontrib>Gormezano, N W S</creatorcontrib><creatorcontrib>Gomes, R C</creatorcontrib><creatorcontrib>Aikawa, N E</creatorcontrib><creatorcontrib>Pereira, R M R</creatorcontrib><creatorcontrib>Terreri, M T</creatorcontrib><creatorcontrib>Magalhães, C S</creatorcontrib><creatorcontrib>Ferreira, J C</creatorcontrib><creatorcontrib>Okuda, E M</creatorcontrib><creatorcontrib>Sakamoto, A P</creatorcontrib><creatorcontrib>Sallum, A M E</creatorcontrib><creatorcontrib>Appenzeller, S</creatorcontrib><creatorcontrib>Ferriani, V P L</creatorcontrib><creatorcontrib>Barbosa, C M</creatorcontrib><creatorcontrib>Lotufo, S</creatorcontrib><creatorcontrib>Jesus, A A</creatorcontrib><creatorcontrib>Andrade, L E C</creatorcontrib><creatorcontrib>Campos, L M A</creatorcontrib><creatorcontrib>Bonfá, E</creatorcontrib><creatorcontrib>Silva, C A</creatorcontrib><creatorcontrib>Brazilian Childhood-onset Systemic Lupus Erythematosus Group</creatorcontrib><creatorcontrib>Brazilian Childhood-onset Systemic Lupus Erythematosus Group</creatorcontrib><title>Outcomes of 847 childhood-onset systemic lupus erythematosus patients in three age groups</title><title>Lupus</title><addtitle>Lupus</addtitle><description>Objective
The objective of this study was to assess outcomes of childhood systemic lupus erythematosus (cSLE) in three different age groups evaluated at last visit: group A early-onset disease (<6 years), group B school age (≥6 and <12 years) and group C adolescent (≥12 and <18 years).
Methods
An observational cohort study was performed in ten pediatric rheumatology centers, including 847 cSLE patients.
Results
Group A had 39 (4%), B 395 (47%) and C 413 (49%). Median disease duration was significantly higher in group A compared to groups B and C (8.3 (0.1–23.4) vs 6.2 (0–17) vs 3.3 (0–14.6) years, p < 0.0001). The median Systemic Lupus International Collaborating Clinics/American College of Rheumatology Damage Index (SLICC/ACR-DI) (0 (0–9) vs 0 (0–6) vs 0 (0–7), p = 0.065) was comparable in the three groups. Further analysis of organ/system damage revealed that frequencies of neuropsychiatric (21% vs 10% vs 7%, p = 0.007), skin (10% vs 1% vs 3%, p = 0.002) and peripheral vascular involvements (5% vs 3% vs 0.3%, p = 0.008) were more often observed in group A compared to groups B and C. Frequencies of severe cumulative lupus manifestations such as nephritis, thrombocytopenia, and autoimmune hemolytic anemia were similar in all groups (p > 0.05). Mortality rate was significantly higher in group A compared to groups B and C (15% vs 10% vs 6%, p = 0.028). Out of 69 deaths, 33/69 (48%) occurred within the first two years after diagnosis. Infections accounted for 54/69 (78%) of the deaths and 38/54 (70%) had concomitant disease activity.
Conclusions
This large multicenter study provided evidence that early-onset cSLE group had distinct outcomes. This group was characterized by higher mortality rate and neuropsychiatric/vascular/skin organ damage in spite of comparable frequencies of severe cumulative lupus manifestations. We also identified that overall death in cSLE patients was an early event mainly attributed to infection associated with disease activity.</description><subject>Adolescent</subject><subject>Age of Onset</subject><subject>Anemia, Hemolytic, Autoimmune - complications</subject><subject>Anemia, Hemolytic, Autoimmune - diagnosis</subject><subject>Anemia, Hemolytic, Autoimmune - pathology</subject><subject>Brazil - epidemiology</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Cohort Studies</subject><subject>Female</subject><subject>Humans</subject><subject>Immunosuppressive Agents - therapeutic use</subject><subject>Infant</subject><subject>Lupus Erythematosus, Systemic - complications</subject><subject>Lupus Erythematosus, Systemic - drug therapy</subject><subject>Lupus Erythematosus, Systemic - epidemiology</subject><subject>Lupus Erythematosus, Systemic - mortality</subject><subject>Mortality</subject><subject>Nephritis - complications</subject><subject>Nephritis - diagnosis</subject><subject>Nephritis - epidemiology</subject><subject>Nephritis - mortality</subject><subject>Pregnancy</subject><subject>Retrospective Studies</subject><subject>Severity of Illness Index</subject><subject>Thrombocytopenia - complications</subject><subject>Thrombocytopenia - diagnosis</subject><subject>Thrombocytopenia - pathology</subject><subject>Treatment Outcome</subject><issn>0961-2033</issn><issn>1477-0962</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2017</creationdate><recordtype>article</recordtype><recordid>eNp1kL1PwzAQxS0EouVjZ0IeWQJ3sXHiEVV8SZVYYGCK3ORCg5I4-Jyh_z2pWhiQmE5373dPek-IC4RrxCy7AWswBaUwMxYMmgMxR51lyXRPD8V8KydbfSZOmD8BQKE1x2KW5qg0qHwu3l_GWPqOWPpa5jqT5bppq7X3VeJ7pih5w5G6ppTtOIwsKWzimjoXPU_b4GJDfWTZ9DKuA5F0HyQ_gh8HPhNHtWuZzvfzVLw93L8unpLly-Pz4m6ZlCqzMaHSrNJMASpU4GrUpDSCdqkC5eo6tasctLWVSnNl8RaIdIW5LbUzxqHL1am42vkOwX-NxLHoGi6pbV1PfuQCc5NaNfUDEwo7tAyeOVBdDKHpXNgUCMW20OJvodPL5d59XHVU_T78NDgByQ7gKXrx6cfQT2n_N_wGkNd8gw</recordid><startdate>201708</startdate><enddate>201708</enddate><creator>Lopes, S R M</creator><creator>Gormezano, N W S</creator><creator>Gomes, R C</creator><creator>Aikawa, N E</creator><creator>Pereira, R M R</creator><creator>Terreri, M T</creator><creator>Magalhães, C S</creator><creator>Ferreira, J C</creator><creator>Okuda, E M</creator><creator>Sakamoto, A P</creator><creator>Sallum, A M E</creator><creator>Appenzeller, S</creator><creator>Ferriani, V P L</creator><creator>Barbosa, C M</creator><creator>Lotufo, S</creator><creator>Jesus, A A</creator><creator>Andrade, L E C</creator><creator>Campos, L M A</creator><creator>Bonfá, E</creator><creator>Silva, C A</creator><general>SAGE Publications</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>7X8</scope></search><sort><creationdate>201708</creationdate><title>Outcomes of 847 childhood-onset systemic lupus erythematosus patients in three age groups</title><author>Lopes, S R M ; Gormezano, N W S ; Gomes, R C ; Aikawa, N E ; Pereira, R M R ; Terreri, M T ; Magalhães, C S ; Ferreira, J C ; Okuda, E M ; Sakamoto, A P ; Sallum, A M E ; Appenzeller, S ; Ferriani, V P L ; Barbosa, C M ; Lotufo, S ; Jesus, A A ; Andrade, L E C ; Campos, L M A ; Bonfá, E ; Silva, C A</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c379t-ec6b273013130af14e34104a2303aff29b80499d32839150ee4d189c4a66a1a83</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2017</creationdate><topic>Adolescent</topic><topic>Age of Onset</topic><topic>Anemia, Hemolytic, Autoimmune - complications</topic><topic>Anemia, Hemolytic, Autoimmune - diagnosis</topic><topic>Anemia, Hemolytic, Autoimmune - pathology</topic><topic>Brazil - epidemiology</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Cohort Studies</topic><topic>Female</topic><topic>Humans</topic><topic>Immunosuppressive Agents - therapeutic use</topic><topic>Infant</topic><topic>Lupus Erythematosus, Systemic - complications</topic><topic>Lupus Erythematosus, Systemic - drug therapy</topic><topic>Lupus Erythematosus, Systemic - epidemiology</topic><topic>Lupus Erythematosus, Systemic - mortality</topic><topic>Mortality</topic><topic>Nephritis - complications</topic><topic>Nephritis - diagnosis</topic><topic>Nephritis - epidemiology</topic><topic>Nephritis - mortality</topic><topic>Pregnancy</topic><topic>Retrospective Studies</topic><topic>Severity of Illness Index</topic><topic>Thrombocytopenia - complications</topic><topic>Thrombocytopenia - diagnosis</topic><topic>Thrombocytopenia - pathology</topic><topic>Treatment Outcome</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Lopes, S R M</creatorcontrib><creatorcontrib>Gormezano, N W S</creatorcontrib><creatorcontrib>Gomes, R C</creatorcontrib><creatorcontrib>Aikawa, N E</creatorcontrib><creatorcontrib>Pereira, R M R</creatorcontrib><creatorcontrib>Terreri, M T</creatorcontrib><creatorcontrib>Magalhães, C S</creatorcontrib><creatorcontrib>Ferreira, J C</creatorcontrib><creatorcontrib>Okuda, E M</creatorcontrib><creatorcontrib>Sakamoto, A P</creatorcontrib><creatorcontrib>Sallum, A M E</creatorcontrib><creatorcontrib>Appenzeller, S</creatorcontrib><creatorcontrib>Ferriani, V P L</creatorcontrib><creatorcontrib>Barbosa, C M</creatorcontrib><creatorcontrib>Lotufo, S</creatorcontrib><creatorcontrib>Jesus, A A</creatorcontrib><creatorcontrib>Andrade, L E C</creatorcontrib><creatorcontrib>Campos, L M A</creatorcontrib><creatorcontrib>Bonfá, E</creatorcontrib><creatorcontrib>Silva, C A</creatorcontrib><creatorcontrib>Brazilian Childhood-onset Systemic Lupus Erythematosus Group</creatorcontrib><creatorcontrib>Brazilian Childhood-onset Systemic Lupus Erythematosus Group</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><jtitle>Lupus</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Lopes, S R M</au><au>Gormezano, N W S</au><au>Gomes, R C</au><au>Aikawa, N E</au><au>Pereira, R M R</au><au>Terreri, M T</au><au>Magalhães, C S</au><au>Ferreira, J C</au><au>Okuda, E M</au><au>Sakamoto, A P</au><au>Sallum, A M E</au><au>Appenzeller, S</au><au>Ferriani, V P L</au><au>Barbosa, C M</au><au>Lotufo, S</au><au>Jesus, A A</au><au>Andrade, L E C</au><au>Campos, L M A</au><au>Bonfá, E</au><au>Silva, C A</au><aucorp>Brazilian Childhood-onset Systemic Lupus Erythematosus Group</aucorp><aucorp>Brazilian Childhood-onset Systemic Lupus Erythematosus Group</aucorp><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Outcomes of 847 childhood-onset systemic lupus erythematosus patients in three age groups</atitle><jtitle>Lupus</jtitle><addtitle>Lupus</addtitle><date>2017-08</date><risdate>2017</risdate><volume>26</volume><issue>9</issue><spage>996</spage><epage>1001</epage><pages>996-1001</pages><issn>0961-2033</issn><eissn>1477-0962</eissn><abstract>Objective
The objective of this study was to assess outcomes of childhood systemic lupus erythematosus (cSLE) in three different age groups evaluated at last visit: group A early-onset disease (<6 years), group B school age (≥6 and <12 years) and group C adolescent (≥12 and <18 years).
Methods
An observational cohort study was performed in ten pediatric rheumatology centers, including 847 cSLE patients.
Results
Group A had 39 (4%), B 395 (47%) and C 413 (49%). Median disease duration was significantly higher in group A compared to groups B and C (8.3 (0.1–23.4) vs 6.2 (0–17) vs 3.3 (0–14.6) years, p < 0.0001). The median Systemic Lupus International Collaborating Clinics/American College of Rheumatology Damage Index (SLICC/ACR-DI) (0 (0–9) vs 0 (0–6) vs 0 (0–7), p = 0.065) was comparable in the three groups. Further analysis of organ/system damage revealed that frequencies of neuropsychiatric (21% vs 10% vs 7%, p = 0.007), skin (10% vs 1% vs 3%, p = 0.002) and peripheral vascular involvements (5% vs 3% vs 0.3%, p = 0.008) were more often observed in group A compared to groups B and C. Frequencies of severe cumulative lupus manifestations such as nephritis, thrombocytopenia, and autoimmune hemolytic anemia were similar in all groups (p > 0.05). Mortality rate was significantly higher in group A compared to groups B and C (15% vs 10% vs 6%, p = 0.028). Out of 69 deaths, 33/69 (48%) occurred within the first two years after diagnosis. Infections accounted for 54/69 (78%) of the deaths and 38/54 (70%) had concomitant disease activity.
Conclusions
This large multicenter study provided evidence that early-onset cSLE group had distinct outcomes. This group was characterized by higher mortality rate and neuropsychiatric/vascular/skin organ damage in spite of comparable frequencies of severe cumulative lupus manifestations. We also identified that overall death in cSLE patients was an early event mainly attributed to infection associated with disease activity.</abstract><cop>London, England</cop><pub>SAGE Publications</pub><pmid>28134038</pmid><doi>10.1177/0961203317690616</doi><tpages>6</tpages><oa>free_for_read</oa></addata></record> |
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| identifier | ISSN: 0961-2033 |
| ispartof | Lupus, 2017-08, Vol.26 (9), p.996-1001 |
| issn | 0961-2033 1477-0962 |
| language | eng |
| recordid | cdi_proquest_miscellaneous_1862936900 |
| source | SAGE Journals |
| subjects | Adolescent Age of Onset Anemia, Hemolytic, Autoimmune - complications Anemia, Hemolytic, Autoimmune - diagnosis Anemia, Hemolytic, Autoimmune - pathology Brazil - epidemiology Child Child, Preschool Cohort Studies Female Humans Immunosuppressive Agents - therapeutic use Infant Lupus Erythematosus, Systemic - complications Lupus Erythematosus, Systemic - drug therapy Lupus Erythematosus, Systemic - epidemiology Lupus Erythematosus, Systemic - mortality Mortality Nephritis - complications Nephritis - diagnosis Nephritis - epidemiology Nephritis - mortality Pregnancy Retrospective Studies Severity of Illness Index Thrombocytopenia - complications Thrombocytopenia - diagnosis Thrombocytopenia - pathology Treatment Outcome |
| title | Outcomes of 847 childhood-onset systemic lupus erythematosus patients in three age groups |
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