{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,23]],"date-time":"2026-09-23T13:07:18Z","timestamp":1790168838812,"version":"4.1.0"},"reference-count":37,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2026,9,23]],"date-time":"2026-09-23T00:00:00Z","timestamp":1790121600000},"content-version":"vor","delay-in-days":265,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,1,1]],"date-time":"2026-01-01T00:00:00Z","timestamp":1767225600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Case Reports in Endocrinology"],"published-print":{"date-parts":[[2026,1]]},"abstract":"<jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>\n                      Congenital hyperinsulinism (CHI) is characterized by inappropriate insulin secretion resulting in persistent neonatal hypoglycemia. CHI is often linked to mutations in the\n                      <jats:italic>ABCC8<\/jats:italic>\n                      and\n                      <jats:italic>KCNJ11<\/jats:italic>\n                      genes and can lead to persistent hypoglycemia, seizures, and neurological injury, making early diagnosis and management essential.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case Report<\/jats:title>\n                    <jats:p>\n                      We describe a late\u2010preterm Nepalese infant born at 34 weeks and 5 days of gestation, weighing 5.26\u2009kg, to a 32\u2010year\u2010old gravida four nondiabetic mother. The infant developed significant hypoglycemia just 2\u2009h after birth. Despite a glucose infusion rate (GIR) of 12\u2009mg\/kg\/min, the infant had persistent hypoglycemia. A critical sample confirmed hyperinsulinism, prompting treatment with octreotide, intravenous (IV) GIR of 17\u2009mg\/kg\/min and later diazoxide. Due to limited IV access, a continuous enteral GIR of 9\u2009mg\/kg\/min was administered through an orogastric (OG) tube. Genetic testing revealed a homozygous pathogenic p.Arg1214Gln mutation in the\n                      <jats:italic>ABCC8<\/jats:italic>\n                      gene, and\n                      <jats:sup>18<\/jats:sup>\n                      F\u2010DOPA PET\u2010CT (2\u2010deoxy\u20102\u2010[fluorine\u201018]fluoro\u2010D\u2010glucose positron emission computed tomography) showed diffuse pancreatic uptake. At 5 months, interruption of diazoxide therapy led to a hypoglycemic seizure (blood glucose level: 29\u2009mg\/dL), which resolved after the treatment resumed. Despite the typical association of homozygous\n                      <jats:italic>ABCC8<\/jats:italic>\n                      mutations with diazoxide nonresponsiveness, this infant achieved sustained glycemic control on diazoxide. On subsequent follow\u2010ups, the child remains stable and continues to achieve normal developmental milestones.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>\n                      Early recognition and timely management are crucial in effective management of CHI. This case shows rare diazoxide responsiveness in homozygous\n                      <jats:italic>ABCC8<\/jats:italic>\n                      mutation. This case suggests that a carefully monitored therapeutic trial of diazoxide may still be reasonable in selected patients with homozygous\n                      <jats:italic>ABCC8<\/jats:italic>\n                      mutations before concluding complete medical unresponsiveness.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1155\/crie\/7968566","type":"journal-article","created":{"date-parts":[[2026,9,23]],"date-time":"2026-09-23T12:40:00Z","timestamp":1790167200000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Diazoxide\u2010Responsive Congenital Hyperinsulinism in a Preterm Infant With Homozygous\n                    <i>ABCC8<\/i>\n                    Mutation: A Case Report"],"prefix":"10.1155","volume":"2026","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-9708-544X","authenticated-orcid":false,"given":"Prerana","family":"Kansakar","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0005-5693-2127","authenticated-orcid":false,"given":"Grisha","family":"Gurung","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-0732-6844","authenticated-orcid":false,"given":"Sadikshya","family":"Bhandari","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kriti","family":"Shrestha","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0005-5096-4785","authenticated-orcid":false,"given":"Bhumika","family":"G. C.","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shreya","family":"Thapa","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0009-8054-4393","authenticated-orcid":false,"given":"Rasik","family":"Acharya","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,9,23]]},"reference":[{"key":"e_1_2_14_1_2","doi-asserted-by":"publisher","DOI":"10.1530\/EDM-18-0120"},{"key":"e_1_2_14_2_2","doi-asserted-by":"publisher","DOI":"10.3389\/fendo.2022.873254"},{"key":"e_1_2_14_3_2","doi-asserted-by":"publisher","DOI":"10.4067\/S0370-41062017000300010"},{"key":"e_1_2_14_4_2","doi-asserted-by":"publisher","DOI":"10.11604\/pamj.2020.35.53.16604"},{"key":"e_1_2_14_5_2","volume-title":"UpToDate","author":"Rozance P. 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