{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T22:37:35Z","timestamp":1790807855815,"version":"4.1.0"},"reference-count":45,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"11","funder":[{"name":"St. Peter's Trust for Kidney, Bladder and Prostate Research"},{"DOI":"10.13039\/501100004185","name":"Northern Counties Kidney Research Fund","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100004185","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000265","name":"Medical Research Council","doi-asserted-by":"publisher","award":["MR\/K023519\/1"],"award-info":[{"award-number":["MR\/K023519\/1"]}],"id":[{"id":"10.13039\/501100000265","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100004440","name":"Wellcome Trust","doi-asserted-by":"publisher","award":["212252\/Z\/18\/Z"],"award-info":[{"award-number":["212252\/Z\/18\/Z"]}],"id":[{"id":"10.13039\/100004440","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000405","name":"Kids Kidney Research","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000405","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":["CJASN"],"published-print":{"date-parts":[[2021,11]]},"abstract":"<jats:sec>\n                    <jats:title>Background and objectives<\/jats:title>\n                    <jats:p>Membranoproliferative GN and C3 glomerulopathy are rare and overlapping disorders associated with dysregulation of the alternative complement pathway. Specific etiologic data for pediatric membranoproliferative GN\/C3 glomerulopathy are lacking, and outcome data are based on retrospective studies without etiologic data.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Design, setting, participants, &amp; measurements<\/jats:title>\n                    <jats:p>A total of 80 prevalent pediatric patients with membranoproliferative GN\/C3 glomerulopathy underwent detailed phenotyping and long-term follow-up within the National Registry of Rare Kidney Diseases (RaDaR). Risk factors for kidney survival were determined using a Cox proportional hazards model. Kidney and transplant graft survival was determined using the Kaplan\u2013Meier method.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Central histology review determined 39 patients with C3 glomerulopathy, 31 with immune-complex membranoproliferative GN, and ten with immune-complex GN. Patients were aged 2\u201315 (median, 9; interquartile range, 7\u201311) years. Median complement C3 and C4 levels were 0.31 g\/L and 0.14 g\/L, respectively; acquired (anticomplement autoantibodies) or genetic alternative pathway abnormalities were detected in 46% and 9% of patients, respectively, across all groups, including those with immune-complex GN. Median follow-up was 5.18 (interquartile range, 2.13\u20138.08) years. Eleven patients (14%) progressed to kidney failure, with nine transplants performed in eight patients, two of which failed due to recurrent disease. Presence of &gt;50% crescents on the initial biopsy specimen was the sole variable associated with kidney failure in multivariable analysis (hazard ratio, 6.2; 95% confidence interval, 1.05 to 36.6;\n                      <jats:italic toggle=\"yes\">P&lt;<\/jats:italic>\n                      0.05). Three distinct C3 glomerulopathy prognostic groups were identified according to presenting eGFR and &gt;50% crescents on the initial biopsy specimen.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Crescentic disease was a key risk factor associated with kidney failure in a national cohort of pediatric patients with membranoproliferative GN\/C3 glomerulopathy and immune-complex GN. Presenting eGFR and crescentic disease help define prognostic groups in pediatric C3 glomerulopathy. Acquired abnormalities of the alternative pathway were commonly identified but not a risk factor for kidney failure.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.2215\/cjn.00320121","type":"journal-article","created":{"date-parts":[[2021,9,22]],"date-time":"2021-09-22T14:45:14Z","timestamp":1632321914000},"page":"1639-1651","update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":45,"title":["C3 Glomerulopathy and Related Disorders in Children"],"prefix":"10.2215","volume":"16","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-4867-6117","authenticated-orcid":false,"given":"Edwin K.S.","family":"Wong","sequence":"first","affiliation":[{"name":"National Renal Complement Therapeutics Centre, Royal Victoria Infirmary, Newcastle upon Tyne, United Kingdom"},{"name":"Complement Therapeutics Research Group, Translational and Clinical Research Institute, Newcastle University, Newcastle upon Tyne, United Kingdom"},{"name":"Department of Renal Medicine, Freeman Hospital, Newcastle upon Tyne, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1312-5411","authenticated-orcid":false,"given":"Kevin J.","family":"Marchbank","sequence":"additional","affiliation":[{"name":"National Renal Complement Therapeutics Centre, Royal Victoria Infirmary, Newcastle upon Tyne, United Kingdom"},{"name":"Complement Therapeutics Research Group, Translational and Clinical Research Institute, Newcastle University, Newcastle upon Tyne, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hannah","family":"Lomax-Browne","sequence":"additional","affiliation":[{"name":"Department of Immunology and Inflammation, Imperial College, London, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Isabel Y.","family":"Pappworth","sequence":"additional","affiliation":[{"name":"Complement Therapeutics Research Group, Translational and Clinical Research Institute, Newcastle University, Newcastle upon Tyne, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Harriet","family":"Denton","sequence":"additional","affiliation":[{"name":"Complement Therapeutics Research Group, Translational and Clinical Research Institute, Newcastle University, Newcastle upon Tyne, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Katie","family":"Cooke","sequence":"additional","affiliation":[{"name":"Complement Therapeutics Research Group, Translational and Clinical Research Institute, Newcastle University, Newcastle upon Tyne, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sophie","family":"Ward","sequence":"additional","affiliation":[{"name":"Complement Therapeutics Research Group, Translational and Clinical Research Institute, Newcastle University, Newcastle upon Tyne, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amy-Claire","family":"McLoughlin","sequence":"additional","affiliation":[{"name":"Complement Therapeutics Research Group, Translational and Clinical Research Institute, Newcastle University, Newcastle upon Tyne, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Grant","family":"Richardson","sequence":"additional","affiliation":[{"name":"Complement Therapeutics Research Group, Translational and Clinical Research Institute, Newcastle University, Newcastle upon Tyne, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Valerie","family":"Wilson","sequence":"additional","affiliation":[{"name":"National Renal Complement Therapeutics Centre, Royal Victoria Infirmary, Newcastle upon Tyne, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0845-1730","authenticated-orcid":false,"given":"Claire L.","family":"Harris","sequence":"additional","affiliation":[{"name":"National Renal Complement Therapeutics Centre, Royal Victoria Infirmary, Newcastle upon Tyne, United Kingdom"},{"name":"Complement Therapeutics Research Group, Translational and Clinical Research Institute, Newcastle University, Newcastle upon Tyne, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"B. Paul","family":"Morgan","sequence":"additional","affiliation":[{"name":"Systems Immunity Research Institute, School of Medicine, Cardiff University, Cardiff, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Svetlana","family":"Hakobyan","sequence":"additional","affiliation":[{"name":"Systems Immunity Research Institute, School of Medicine, Cardiff University, Cardiff, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paul","family":"McAlinden","sequence":"additional","affiliation":[{"name":"Research and Development Department, Newcastle upon Tyne Hospitals National Health Service (NHS) Foundation Trust, Newcastle upon Tyne, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9170-1579","authenticated-orcid":false,"given":"Daniel P.","family":"Gale","sequence":"additional","affiliation":[{"name":"Department of Renal Medicine, University College London, London, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Heather","family":"Maxwell","sequence":"additional","affiliation":[{"name":"The Royal Hospital for Children, Glasgow, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Martin","family":"Christian","sequence":"additional","affiliation":[{"name":"Nottingham Children\u2019s Hospital, Queens Medical Centre, Nottingham, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Roger","family":"Malcomson","sequence":"additional","affiliation":[{"name":"Histopathology Department, Leicester Royal Infirmary, Leicester, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Timothy H.J.","family":"Goodship","sequence":"additional","affiliation":[{"name":"National Renal Complement Therapeutics Centre, Royal Victoria Infirmary, Newcastle upon Tyne, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9850-8352","authenticated-orcid":false,"given":"Stephen D.","family":"Marks","sequence":"additional","affiliation":[{"name":"Department of Paediatric Nephrology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, United Kingdom"},{"name":"National Institute for Health Research Great Ormond Street Hospital Biomedical Research Centre, University College London Great Ormond Street Institute of Child Health, London, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1153-0192","authenticated-orcid":false,"given":"Matthew C.","family":"Pickering","sequence":"additional","affiliation":[{"name":"Department of Immunology and Inflammation, Imperial College, London, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4718-0072","authenticated-orcid":false,"given":"David","family":"Kavanagh","sequence":"additional","affiliation":[{"name":"National Renal Complement Therapeutics Centre, Royal Victoria Infirmary, Newcastle upon Tyne, United Kingdom"},{"name":"Complement Therapeutics Research Group, Translational and Clinical Research Institute, Newcastle University, Newcastle upon Tyne, United Kingdom"},{"name":"Department of Renal Medicine, Freeman Hospital, Newcastle upon Tyne, United Kingdom"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"H. 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