{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,11,2]],"date-time":"2026-11-02T17:55:54Z","timestamp":1793642154746,"version":"4.5.1"},"reference-count":17,"publisher":"The Endocrine Society","issue":"11","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2011,11,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Context:<\/jats:title>\n                  <jats:p>Type 1 hyperlipoproteinemia (T1HLP) in childhood is most often due to genetic deficiency of lipoprotein lipase (LPL) or other related proteins.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Objective:<\/jats:title>\n                  <jats:p>The aim was to report a case of marked hypertriglyceridemia and recurrent acute pancreatitis due to the presence of LPL autoantibody in a young girl who was subsequently diagnosed with Sj\u00f6gren's syndrome.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Subject and Methods:<\/jats:title>\n                  <jats:p>A 9-yr-old African-American girl presented with acute pancreatitis and serum triglycerides of 4784 mg\/dl. Strict restriction of dietary fat reduced serum triglycerides, but she continued to experience recurrent pancreatitis. Approximately 18 months thereafter, she developed transient pauciarticular arthritis with elevated serum antinuclear antibody (&amp;gt;1:1280). Minor salivary gland biopsy revealed chronic sialadenitis with a dense periductal lymphocytic aggregate suggestive of Sj\u00f6gren's syndrome. Genomic DNA was analyzed for LPL, GPIHBP1, APOA5, APOC2, and LMF1. Immunoblotting was performed to detect serum LPL autoantibody.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results:<\/jats:title>\n                  <jats:p>The patient had no disease-causing variants in LPL, GPIHBP1, APOA5, APOC2, or LMF1. Immunoblotting revealed serum LPL antibody. The patient responded to immunosuppressive therapy for Sj\u00f6gren's syndrome with resolution of hypertriglyceridemia.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusions:<\/jats:title>\n                  <jats:p>Unexplained T1HLP in childhood could be secondary to LPL deficiency induced by autoantibodies. Therefore, diagnosis of autoimmune T1HLP should be entertained if clinical features are suggestive of an autoimmune process.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1210\/jc.2011-1113","type":"journal-article","created":{"date-parts":[[2011,9,1]],"date-time":"2011-09-01T11:19:20Z","timestamp":1314875960000},"page":"3302-3307","source":"Crossref","is-referenced-by-count":31,"title":["Type 1 Hyperlipoproteinemia and Recurrent Acute Pancreatitis due to Lipoprotein Lipase Antibody in a Young Girl with Sj\u00f6gren's Syndrome"],"prefix":"10.1210","volume":"96","author":[{"given":"Ambika P.","family":"Ashraf","sequence":"first","affiliation":[{"name":"Division of Pediatric Endocrinology (A.P.A.), University of Alabama at Birmingham, Birmingham, Alabama 35294"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Timothy","family":"Beukelman","sequence":"additional","affiliation":[{"name":"Children's Hospital, and Division of Rheumatology (T.B.), University of Alabama at Birmingham, Birmingham, Alabama 35294"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Valerie","family":"Pruneta-Deloche","sequence":"additional","affiliation":[{"name":"Unit\u00e9 Mixte de Recherch\u00e9 (V.P.-D.) Lyon University, Institut National des Sciences Appliqu\u00e9es de Lyon, CarMeN Laboratory, Institut National de la Sant\u00e9 et de la Recherche M\u00e9dicale Unit\u00e9 1060, Universit\u00e9 Lyon-1, F-69621 Villeurbanne, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David R.","family":"Kelly","sequence":"additional","affiliation":[{"name":"Department of Pathology (D.R.K.), University of Alabama at Birmingham, Birmingham, Alabama 35294"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Abhimanyu","family":"Garg","sequence":"additional","affiliation":[{"name":"Division of Nutrition and Metabolic Diseases (A.G.), Department of Internal Medicine and the Center for Human Nutrition, University of Texas Southwestern Medical Center, Dallas, Texas 75390"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"80","reference":[{"key":"2020071615372582300_B1","first-page":"1","article-title":"Familial lipoprotein lipase deficiency, apo C-II deficiency, and hepatic lipase deficiency","volume-title":"Online Metabolic and Molecular Bases of Inherited Disease","author":"Brunzell","year":"2006"},{"key":"2020071615372582300_B2","doi-asserted-by":"crossref","first-page":"151","DOI":"10.1139\/Y09-005","article-title":"Novel LPL mutations associated with lipoprotein lipase deficiency: two case reports and a literature review.","volume":"87","author":"Rahalkar","year":"2009","journal-title":"Can J Physiol Pharmacol"},{"key":"2020071615372582300_B3","doi-asserted-by":"crossref","first-page":"2862","DOI":"10.1172\/JCI24471","article-title":"Apoa5 Q139X truncation predisposes to late-onset hyperchylomicronemia due to lipoprotein lipase impairment.","volume":"115","author":"Mar\u00e7ais","year":"2005","journal-title":"J Clin Invest"},{"key":"2020071615372582300_B4","doi-asserted-by":"crossref","first-page":"1483","DOI":"10.1038\/ng.2007.24","article-title":"Mutations in LMF1 cause combined lipase deficiency and severe hypertriglyceridemia.","volume":"39","author":"P\u00e9terfy","year":"2007","journal-title":"Nat Genet"},{"key":"2020071615372582300_B5","doi-asserted-by":"crossref","first-page":"956","DOI":"10.1161\/ATVBAHA.109.186577","article-title":"Chylomicronemia with a mutant GPIHBP1 (Q115P) that cannot bind lipoprotein lipase.","volume":"29","author":"Beigneux","year":"2009","journal-title":"Arterioscler Thromb Vasc Biol"},{"key":"2020071615372582300_B6","doi-asserted-by":"crossref","first-page":"1581","DOI":"10.1210\/jc.2007-0275","article-title":"Update on dyslipidemia.","volume":"92","author":"Garg","year":"2007","journal-title":"J Clin Endocrinol Metab"},{"key":"2020071615372582300_B7","doi-asserted-by":"crossref","first-page":"1113","DOI":"10.1503\/cmaj.060963","article-title":"Hypertriglyceridemia: its etiology, effects and treatment.","volume":"176","author":"Yuan","year":"2007","journal-title":"CMAJ"},{"key":"2020071615372582300_B8","doi-asserted-by":"crossref","first-page":"15","DOI":"10.1111\/j.1399-0004.1994.tb04197.x","article-title":"Gene environment interaction and plasma triglyceride levels: the crucial role of lipoprotein lipase.","volume":"46","author":"Hayden","year":"1994","journal-title":"Clin Genet"},{"key":"2020071615372582300_B9","doi-asserted-by":"crossref","first-page":"1255","DOI":"10.1056\/NEJM198905113201906","article-title":"Autoimmune hyperchylomicronemia.","volume":"320","author":"Kihara","year":"1989","journal-title":"N Engl J Med"},{"key":"2020071615372582300_B10","doi-asserted-by":"crossref","first-page":"49","DOI":"10.1016\/S0301-2115(97)00154-1","article-title":"Rare case of autoimmune hyperchylomicronemia during pregnancy.","volume":"76","author":"Yoshimura","year":"1998","journal-title":"Eur J Obstet Gynecol Reprod Biol"},{"key":"2020071615372582300_B11","first-page":"791","article-title":"Characterization of a new case of autoimmune type I hyperlipidemia: long-term remission under immunosuppressive therapy.","volume":"82","author":"Pruneta","year":"1997","journal-title":"J Clin Endocrinol Metab"},{"key":"2020071615372582300_B12","doi-asserted-by":"crossref","first-page":"3995","DOI":"10.1210\/jc.2005-0205","article-title":"Combination of circulating antilipoprotein lipase (anti-LPL) antibody and heterozygous S172 fsX179 mutation of LPL gene leading to chronic hyperchylomicronemia.","volume":"90","author":"Pruneta-Deloche","year":"2005","journal-title":"J Clin Endocrinol Metab"},{"key":"2020071615372582300_B13","doi-asserted-by":"crossref","first-page":"502","DOI":"10.1530\/eje.0.1410502","article-title":"Acquired lipoprotein lipase deficiency associated with chronic urticaria. 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