Objective: To report a case of isolated ankyloglossia superior in a one-month-old boy. Methods: Design: Case Report Setting: Tertiary Government Training Hospital Patient: One Results: A one-month-old boy with failure to thrive, feeding difficulties and a palatoglossal band that limited mouth opening and anterior posturing of the tongue was diagnosed to have non-syndromic ankyloglossia superior and underwent surgical transection of the fibrous ankylosis under intravenous sedation due to difficulty of insinuating an endotracheal tube orally. Direct latch breastfeeding was successfully adapted from the second to tenth postoperative day before they were lost to follow up. Conclusion: Despite a complicated pediatric airway, coordinated surgical and anesthesia management successfully restored tongue function and mouth opening to allow effective feeding.