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We present a case of a 12-month-old normally developing baby girl who was admitted for hypothermia, acute hypoxic respiratory failure, altered mental status, and metabolic acidosis. A comprehensive workup confirmed the presence of fentanyl in her urine. A magnetic resonance image (MRI) of the brain revealed multiple areas of signal abnormalities in the subcortical and periventricular white matter. The infant recovered and was discharged with an age-appropriate neurologic examination. A repeat MRI at 18 months showed almost complete resolution of the previous findings along with a normal neurologic exam and development for age. This case emphasizes the importance of timely diagnosis and appropriate management in preventing long-term neurologic sequelae associated with fentanyl exposure.<\/jats:p>","DOI":"10.1177\/08830738251348044","type":"journal-article","created":{"date-parts":[[2025,6,23]],"date-time":"2025-06-23T00:55:08Z","timestamp":1750640108000},"page":"100-104","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":3,"title":["Novel Cause of Reversible Leukoencephalopathy in an Infant Associated With Illicit Fentanyl Exposure"],"prefix":"10.1177","volume":"41","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-1174-3785","authenticated-orcid":false,"given":"Emin","family":"Fidan","sequence":"first","affiliation":[{"name":"SUNY Downstate Health Sciences University and Kings County Hospital, Brooklyn, New York, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Apurva","family":"Devaraj","sequence":"additional","affiliation":[{"name":"SUNY Downstate Health Sciences University and Kings County Hospital, Brooklyn, New York, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Radha","family":"Giridharan","sequence":"additional","affiliation":[{"name":"SUNY Downstate Health Sciences University and Kings County Hospital, Brooklyn, New York, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Geetha","family":"Chari","sequence":"additional","affiliation":[{"name":"SUNY Downstate Health Sciences University and Kings County Hospital, Brooklyn, New York, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2025,6,22]]},"reference":[{"key":"e_1_3_2_2_2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMc2313270"},{"key":"e_1_3_2_3_2","unstructured":"House TW. 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