{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,17]],"date-time":"2026-03-17T20:03:21Z","timestamp":1773777801179,"version":"3.50.1"},"reference-count":16,"publisher":"S. Karger AG","issue":"1","license":[{"start":{"date-parts":[[2026,1,27]],"date-time":"2026-01-27T00:00:00Z","timestamp":1769472000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"},{"start":{"date-parts":[[2026,1,27]],"date-time":"2026-01-27T00:00:00Z","timestamp":1769472000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Case Rep Gastroenterol"],"abstract":"<jats:p>Introduction: Macroamylasemia is a rare biochemical disorder in which the amylase molecule binds to other macromolecules, most commonly immunoglobulin A or G. The increased molecular weight of this complex reduces renal excretion of the enzyme, resulting in chronically elevated serum amylase levels. It can be associated with various non-pancreatic conditions, including celiac disease. In some reported cases, macroamylasemia resolved with a gluten-free diet. We present a pediatric case in which macroamylasemia was the initial manifestation of celiac disease. Case Presentation: A two-year-old boy presented with a 3-month history of abdominal pain, anemia, and constipation. Physical examination revealed paraumbilical tenderness. Laboratory tests showed hypochromic anemia; elevated serum amylase (171 U\/L); low serum iron (2.5 \u00b5mol\/L); vitamin D3 deficiency (15.9 ng\/mL), and low urine amylase with an amylase\/creatinine clearance ratio below 1%, confirming macroamylasemia. Imaging studies were normal. HLA typing revealed the HLA-DQ2 haplotype. Celiac serology was positive for tissue transglutaminase-IgA (87.55 IE\/mL) and endomysial antibody-IgA (1:&gt;100). Duodenal biopsy confirmed Marsh type IIIb histology. A gluten-free diet was initiated alongside iron and vitamin D3 supplementation. Symptoms resolved within 2 months, accompanied by normalization of serum amylase levels and a weight gain of over 1 kg. Vitamin D3 therapy was continued until serum levels normalized. Conclusion: Celiac disease should be considered in patients with unexplained macroamylasemia, even in the absence of classical gastrointestinal symptoms. The primary treatment in such cases is a strict gluten-free diet, which can lead to the normalization of serum amylase levels when macroamylasemia is associated with celiac disease.<\/jats:p>","DOI":"10.1159\/000550670","type":"journal-article","created":{"date-parts":[[2026,1,27]],"date-time":"2026-01-27T17:01:49Z","timestamp":1769533309000},"page":"40-45","source":"Crossref","is-referenced-by-count":0,"title":["Macroamylasemia in a Two-Year-Old Boy as the First Manifestation of Celiac Disease: A Case Report"],"prefix":"10.1159","volume":"20","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8641-6979","authenticated-orcid":false,"given":"Marija","family":"Dimitrovska-Ivanova","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Elena","family":"Simeonovska Joveva","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Marija","family":"Karakolevska Ilova","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Aneta","family":"Demerdzieva","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Biljana","family":"Coneska Jovanova","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Snezana","family":"Palcevska Kocevska","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"127","published-online":{"date-parts":[[2026,1,27]]},"reference":[{"key":"ref1","doi-asserted-by":"publisher","DOI":"10.1002\/j.1536-4801.1992.tb10500.x"},{"key":"ref2","doi-asserted-by":"publisher","DOI":"10.1016\/j.arcped.2004.12.022"},{"key":"ref3","doi-asserted-by":"publisher","DOI":"10.1157\/13127999"},{"key":"ref4","doi-asserted-by":"publisher","DOI":"10.1159\/000132771"},{"key":"ref5","doi-asserted-by":"publisher","DOI":"10.1097\/MPG.0000000000001715"},{"key":"ref6","doi-asserted-by":"publisher","DOI":"10.2169\/internalmedicine.39.1115"},{"key":"ref7","doi-asserted-by":"publisher","DOI":"10.1016\/j.cgh.2017.06.037"},{"key":"ref8","doi-asserted-by":"publisher","DOI":"10.19080\/ARGH.2020.15.555898"},{"key":"ref9","doi-asserted-by":"publisher","DOI":"10.1111\/j.1572-0241.2001.03746.x"},{"key":"ref10","doi-asserted-by":"publisher","DOI":"10.1097\/MPG.0000000000002497"},{"key":"ref11","doi-asserted-by":"publisher","DOI":"10.1016\/s1542-3565(05)00022-4"},{"key":"ref12","doi-asserted-by":"publisher","DOI":"10.1111\/j.1572-0241.2004.30203.x"},{"key":"ref13","doi-asserted-by":"publisher","DOI":"10.1159\/000077568"},{"key":"ref14","doi-asserted-by":"publisher","DOI":"10.4082\/kjfm.23.0195"},{"key":"ref15","doi-asserted-by":"publisher","DOI":"10.1542\/peds.107.6.e93"},{"key":"ref16","doi-asserted-by":"publisher","DOI":"10.1016\/j.cgh.2005.12.027"}],"container-title":["Case Reports in Gastroenterology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/karger.com\/article-pdf\/doi\/10.1159\/000550670","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,3,17]],"date-time":"2026-03-17T17:00:54Z","timestamp":1773766854000},"score":1,"resource":{"primary":{"URL":"https:\/\/karger.com\/article\/doi\/10.1159\/000550670"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,1,27]]},"references-count":16,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2026,1,1]]}},"URL":"https:\/\/doi.org\/10.1159\/000550670","archive":["Portico"],"relation":{"is-supplemented-by":[{"id-type":"doi","id":"10.6084\/m9.figshare.31155175","asserted-by":"subject"}]},"ISSN":["1662-0631"],"issn-type":[{"value":"1662-0631","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,1,27]]}}}