{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,27]],"date-time":"2026-08-27T14:21:43Z","timestamp":1787840503555,"version":"build-2784847793"},"reference-count":9,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"2","license":[{"start":{"date-parts":[[2025,2,1]],"date-time":"2025-02-01T00:00:00Z","timestamp":1738368000000},"content-version":"unspecified","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-sa\/4.0"}],"content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2025,2]]},"abstract":"<jats:title>\n                    A\n                    <jats:sc>BSTRACT<\/jats:sc>\n                  <\/jats:title>\n                  <jats:p>\n                    Lung agenesis (LA) is a rare congenital anomaly that refers to an absence of lung tissue and bronchi.\n                    <jats:italic toggle=\"yes\">Burkholderia cepacia<\/jats:italic>\n                    complex (BCC) causes a fatal acute necrotising pneumonia called Cepacia syndrome. The unique concomitant association of both these rare diseases has not been highlighted in the literature to the best of our knowledge. Here, we present a case of association of right-sided lung agenesis along with Cepacia syndrome. In our case, the\n                    <jats:italic toggle=\"yes\">Burkholderia cepacia<\/jats:italic>\n                    complex was found to be a multi-drug-resistant organism. Debilitating deterioration in the oxygen saturation was observed and initiation of treatment after culture sensitivity report yielded in remission of the disease. However, due to underlying congenital agenesis, therapeutic intervention with steroids and bronchodilators led to stabilising the patient by developing oxygenation. Underlying pulmonary congenital defects predispose individuals to infections. The implementation of antimicrobial therapy must be aided with culture and sensitivity reports for better outcomes.\n                  <\/jats:p>","DOI":"10.4103\/jfmpc.jfmpc_1424_24","type":"journal-article","created":{"date-parts":[[2025,2,20]],"date-time":"2025-02-20T07:03:18Z","timestamp":1740034998000},"page":"804-806","update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":0,"title":["Right lung agenesis with multi-drug-resistant Cepacia syndrome: A case report"],"prefix":"10.4103","volume":"14","author":[{"given":"Mohit","family":"Kondisetti","sequence":"first","affiliation":[{"name":"Department of Pulmonary Medicine, DY Patil Deemed to be University School of Medicine, Navi Mumbai, Maharashtra, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Priyavardhan","family":"Mishra","sequence":"additional","affiliation":[{"name":"DY Patil Deemed to be University School of Medicine, Navi Mumbai, Maharashtra, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anant","family":"Patil","sequence":"additional","affiliation":[{"name":"Department of Pharmacology, DY Patil Deemed to be University School of Medicine, Navi Mumbai, Maharashtra, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nikhil","family":"Sarangdhar","sequence":"additional","affiliation":[{"name":"Department of Pulmonary Medicine, DY Patil Deemed to be University School of Medicine, Navi Mumbai, Maharashtra, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sankeerth","family":"Chanamolu","sequence":"additional","affiliation":[{"name":"Department of Pharmacology, DY Patil Deemed to be University School of Medicine, Navi Mumbai, Maharashtra, India"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"276","published-online":{"date-parts":[[2025,2,21]]},"reference":[{"key":"R1-20260109","doi-asserted-by":"crossref","first-page":"e40","DOI":"10.1055\/a-2107-0651","article-title":"Unilateral lung agenesis:A case series and review of literature","volume":"13","author":"Weber","year":"2023","journal-title":"AJP Rep"},{"key":"R2-20260109","first-page":"4397641","article-title":"Right lung agenesis with tracheal stenosis due to complete tracheal rings and postpneumonectomy like syndrome treated with tissue expander placement","volume":"2016","author":"Agrawal","year":"2016","journal-title":"Case Rep Pulmonol"},{"key":"R3-20260109","first-page":"OD15","article-title":"Left pulmonary agenesis with right lung bronchiectasis in an adult","volume":"10","author":"Kumar","year":"2016","journal-title":"J Clin Diagn Res"},{"key":"R4-20260109","first-page":"100994","article-title":"Right unilateral lung agenesis with dextroposition;the first case in Saudi Arabia","volume":"29","author":"Ahmed","year":"2019","journal-title":"Respir Med Case Rep"},{"key":"R5-20260109","doi-asserted-by":"crossref","first-page":"e00029","DOI":"10.1128\/mSystems.00029-16","article-title":"Long-term evolution of Burkholderia multivorans during a chronic cystic fibrosis infection reveals shifting forces of selection","volume":"1","author":"Silva","year":"2016","journal-title":"mSystems"},{"key":"R6-20260109","doi-asserted-by":"crossref","first-page":"e00139","DOI":"10.1128\/CMR.00139-19","article-title":"Burkholderia cepacia complex bacteria:A feared contamination risk in water-based pharmaceutical products","volume":"33","author":"Tavares","year":"2020","journal-title":"Clin Microbiol Rev"},{"key":"R7-20260109","doi-asserted-by":"crossref","first-page":"86","DOI":"10.4269\/ajtmh.21-1338","article-title":"Case report:Community-acquired Burkholderia cepacia pneumonia of a patient with pulmonary tuberculosis","volume":"107","author":"Li","year":"2022","journal-title":"Am J Trop Med Hyg"},{"key":"R8-20260109","doi-asserted-by":"crossref","first-page":"447","DOI":"10.1007\/s00430-017-0521-2","article-title":"Burkholderia cepacia complex in cystic fibrosis in a Brazilian reference centre","volume":"206","author":"Dentini","year":"2017","journal-title":"Med Microbiol Immunol"},{"key":"R9-20260109","doi-asserted-by":"crossref","first-page":"550","DOI":"10.1016\/j.ijid.2022.07.004","article-title":"Management of Cepacia syndrome in an immunocompetent non-cystic fibrosis adult patient","volume":"122","author":"Ottu Para","year":"2022","journal-title":"Int J Infect Dis"}],"container-title":["Journal of Family Medicine and Primary Care"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.ovid.com\/10.4103\/jfmpc.jfmpc_1424_24","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,27]],"date-time":"2026-08-27T13:24:01Z","timestamp":1787837041000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.ovid.com\/10.4103\/jfmpc.jfmpc_1424_24"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,2]]},"references-count":9,"journal-issue":{"issue":"2","published-print":{"date-parts":[[2025]]}},"URL":"https:\/\/doi.org\/10.4103\/jfmpc.jfmpc_1424_24","relation":{},"ISSN":["2249-4863","2278-7135"],"issn-type":[{"value":"2249-4863","type":"print"},{"value":"2278-7135","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,2]]},"assertion":[{"value":"2024-08-19","name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-10-14","name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}}]}}