{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,19]],"date-time":"2026-09-19T00:02:32Z","timestamp":1789776152749,"version":"4.0.1"},"reference-count":11,"publisher":"SAGE Publications","issue":"4","license":[{"start":{"date-parts":[[2026,5,11]],"date-time":"2026-05-11T00:00:00Z","timestamp":1778457600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Trop Doct"],"published-print":{"date-parts":[[2026,10]]},"abstract":"<jats:p>Thiamine deficiency is a reversible cause of severe pulmonary arterial hypertension (PAH) in early infancy, particularly among exclusively breastfed infants of mothers consuming polished rice-based diets. This retrospective case series (July 2023\u2013July 2025) included infants aged-12 months with echocardiographically confirmed severe PAH and no structural heart disease. Eight exclusively breastfed infants (median age 2.5 months) presented with respiratory distress. All mothers consumed polished rice-based diets. Echocardiography revealed severe PAH with right-sided cardiac dilation. Thiamine therapy resulted in rapid clinical improvement within 48 h and complete resolution within two weeks, with no deaths. Thiamine deficiency should be suspected in infants with acute severe PAH, as prompt treatment leads to excellent outcomes.<\/jats:p>","DOI":"10.1177\/00494755261451113","type":"journal-article","created":{"date-parts":[[2026,5,12]],"date-time":"2026-05-12T04:21:37Z","timestamp":1778559697000},"page":"720-722","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":0,"title":["Pulmonary arterial hypertension in infancy: A case series"],"prefix":"10.1177","volume":"56","author":[{"ORCID":"https:\/\/orcid.org\/0009-0007-9086-3801","authenticated-orcid":false,"given":"NV","family":"Shashikala","sequence":"first","affiliation":[{"name":"Resident, Department of Paediatrics, Bapuji Child Health Institute and Research Centre, Davanagere, Karnataka, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"KM","family":"Arun","sequence":"additional","affiliation":[{"name":"2Pediatric Cardiologist, Department of pediatrics, Bapuji Child Health Institute and Research Centre, Davanagere, Karnataka, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2342-0759","authenticated-orcid":false,"given":"RC","family":"Ashwini","sequence":"additional","affiliation":[{"name":"Professor, Neonatologist Department of Pediatrics, Bapuji Child Health Institute and Research Centre, Davanagere, Karnataka, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Muganagowda","family":"Patil","sequence":"additional","affiliation":[{"name":"Professor, HOD Department of Pediatrics, Bapuji Child Health Institute and Research Centre, Davanagere, Karnataka, India"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2026,5,11]]},"reference":[{"key":"e_1_3_2_2_2","first-page":"99","article-title":"Thiamine-responsive acute pulmonary hypertension of early infancy","volume":"7","author":"Panigrahy N","year":"2020","unstructured":"Panigrahy N, Mohanty PK, Sahoo SK. Thiamine-responsive acute pulmonary hypertension of early infancy. Children (Basel) 2020; 7: 99.","journal-title":"Children (Basel)"},{"key":"e_1_3_2_3_2","first-page":"104","article-title":"Infantile beriberi presenting as pulmonary arterial hypertension","volume":"12","author":"Kumar A","year":"2023","unstructured":"Kumar A, Mehra S, Patel V. Infantile beriberi presenting as pulmonary arterial hypertension. J Clin Neonatol 2023; 12: 104\u2013108.","journal-title":"J Clin Neonatol"},{"key":"e_1_3_2_4_2","first-page":"812","article-title":"Pulmonary arterial hypertension in infants related to micronutrient deficiency","volume":"59","author":"Choudhury T","year":"2023","unstructured":"Choudhury T, Bora R, Saikia D. Pulmonary arterial hypertension in infants related to micronutrient deficiency. J Paediatr Child Health 2023; 59: 812\u2013818.","journal-title":"J Paediatr Child Health"},{"key":"e_1_3_2_5_2","first-page":"843215","article-title":"Metabolic and myocardial effects of thiamine deficiency in infancy","volume":"10","author":"Ahmed Z","year":"2022","unstructured":"Ahmed Z, Khan M, Ali R. Metabolic and myocardial effects of thiamine deficiency in infancy. Front Pediatr 2022; 10: 843215.","journal-title":"Front Pediatr"},{"key":"e_1_3_2_6_2","first-page":"1245","article-title":"Cardiac dysfunction in thiamine deficiency: an echocardiographic study","volume":"32","author":"Sharma P","year":"2022","unstructured":"Sharma P, Malhotra A, Jain S. Cardiac dysfunction in thiamine deficiency: an echocardiographic study. Cardiol Young 2022; 32: 1245\u20131251.","journal-title":"Cardiol Young"},{"key":"e_1_3_2_7_2","article-title":"Reversible pulmonary hypertension due to thiamine deficiency in infancy","volume":"68","author":"Singh S","year":"2022","unstructured":"Singh S, Verma A, Gupta R. Reversible pulmonary hypertension due to thiamine deficiency in infancy. J Trop Pediatr 2022; 68: fmac021.","journal-title":"J Trop Pediatr"},{"key":"e_1_3_2_8_2","article-title":"Severe infantile thiamine deficiency presenting with pulmonary hypertension","volume":"15","author":"Narang M","year":"2023","unstructured":"Narang M, Kaur G, Bansal A. Severe infantile thiamine deficiency presenting with pulmonary hypertension. Cureus 2023; 15: e41862.","journal-title":"Cureus"},{"key":"e_1_3_2_9_2","first-page":"12","article-title":"Maternal thiamine deficiency and infantile cardiomyopathy: a prospective observational study","volume":"16","author":"Bhatia P","year":"2024","unstructured":"Bhatia P, Shah D, Iyer S. Maternal thiamine deficiency and infantile cardiomyopathy: a prospective observational study. Nutrients 2024; 16: 12.","journal-title":"Nutrients"},{"key":"e_1_3_2_10_2","first-page":"305","article-title":"Exclusive breastfeeding and risk of thiamine deficiency in early infancy","volume":"12","author":"Rao S","year":"2021","unstructured":"Rao S, Reddy V, Anil Kumar M. Exclusive breastfeeding and risk of thiamine deficiency in early infancy. Pediatr Health Med Ther 2021; 12: 305\u2013312.","journal-title":"Pediatr Health Med Ther"},{"key":"e_1_3_2_11_2","first-page":"210","article-title":"Maternal dietary patterns and risk of infantile thiamine deficiency","volume":"52","author":"Menon S","year":"2023","unstructured":"Menon S, Nair R, Joseph L. Maternal dietary patterns and risk of infantile thiamine deficiency. Clin Nutr ESPEN 2023; 52: 210\u2013216.","journal-title":"Clin Nutr ESPEN"},{"key":"e_1_3_2_12_2","first-page":"56","article-title":"Clinical manifestations of infantile thiamine deficiency in low-income settings","volume":"24","author":"Devi R","year":"2024","unstructured":"Devi R, Prasad S, Mishra S. Clinical manifestations of infantile thiamine deficiency in low-income settings. BMC Pediatr 2024; 24: 56.","journal-title":"BMC Pediatr"}],"container-title":["Tropical Doctor"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/00494755261451113","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1177\/00494755261451113","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/00494755261451113","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,9,18]],"date-time":"2026-09-18T23:20:14Z","timestamp":1789773614000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1177\/00494755261451113"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,5,11]]},"references-count":11,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2026,10]]}},"alternative-id":["10.1177\/00494755261451113"],"URL":"https:\/\/doi.org\/10.1177\/00494755261451113","relation":{},"ISSN":["0049-4755","1758-1133"],"issn-type":[{"value":"0049-4755","type":"print"},{"value":"1758-1133","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,5,11]]}}}