A full-term male neonate delivered by midwife at home following an uneventful pregnancy to consanguineous union in Sudan was brought to emergency department with difficulty breathing, poor feeding, and a noticeable anterolateral neck swelling at six hours of life. The delivery was uncomplicated. On examination, the newborn was tachypneic, hypoxic and had tachycardia with weight 3.3 kg. A soft, non-tender neck mass measuring approximately 4×6 cm was noted in the anterior > right lateral region, displacing the head into extended position. The overlying skin was normal, and trachea was difficult to palpate (Image 1a). Non-invasive oxygen support initiated, and congenital neck masses like cystic hygroma or branchial cysts were considered as etiology for mass causing respiratory distress.Lateral neck radiography demonstrated tracheal deviation and narrowing. Neck ultrasound showed an enlarged, lobulated thyroid gland. Computed tomography confirmed a homogeneously enlarged thyroid with retrosternal extension compressing the upper airway. Thyroid function testing revealed markedly elevated thyroid-stimulating hormone (TSH reference: 152-292 nmol/L), consistent with congenital primary hypothyroidism. Maternal thyroid function was normal and hence, goiter is likely due to dyshormonogenesis. A case of twins with congenital goiter has been reported to require intubation and ventilation due to goiter.(1) Oral levothyroxine (25 μg/day) was initiated. The infant’s respiratory distress improved and by day 5, oxygen was discontinued, and oral feeds resumed. Reports of successful antenatal thyroxine treatment have been reported.(2,3) At the 3-month follow-up, the neck swelling had fully regressed, and the infant demonstrated normal growth, development, and neurologic function (Image 1b). Thyroid function normalized at 3 month follow up.Key Clinical Message:Our case highlights that neonatal goiter causing airway compression is a rare but significant presentation of congenital hypothyroidism. Early recognition through imaging and thyroid function testing, followed by timely initiation of levothyroxine, can rapidly reverse respiratory distress and support normal neurodevelopment.ReferencesReynolds BC, Simpson JH, Macara L, Watt AJ, Kubba H, Donaldson MD, Pohlen J. Goitrous congenital hypothyroidism in a twin pregnancy causing respiratory obstruction at birth: implications for management. Acta Paediatr. 2006 Nov;95(11):1345-8. doi: 10.1080/08035250600711074. PMID: 17062458.Hanono A, Shah B, David R, Buterman I, Roshan D, Shah S, Lam L, Timor-Tritsch I. Antenatal treatment of fetal goiter: a therapeutic challenge. J Matern Fetal Neonatal Med. 2009 Jan;22(1):76-80. doi: 10.1080/14767050802448299. PMID: 19085636.Abuhamad AZ, Fisher DA, Warsof SL, Slotnick RN, Pyle PG, Wu SY, Evans AT. Antenatal diagnosis and treatment of fetal goitrous hypothyroidism: case report and review of the literature. Ultrasound Obstet Gynecol. 1995 Nov;6(5):368-71. doi: 10.1046/j.1469-0705.1995.06050368.x. PMID: 8590211.Figure legendsImage 1a: Visible anterior neck swelling shortly after birth.. Image 1b: Resolution of goiter after 3 months of therapy.Written informed consent was taken from the parents of the index case for publication purposes.Author contribution:Khalid Makki was the primary physician managing the case, contributed to the clinical diagnosis, coordinated imaging and treatment, and drafted the initial manuscript.Fatima Abdallah contributed to the clinical management, collected relevant patient data, reviewed the literature, and assisted in manuscript preparation.Poorvi Agrawal supervised the case report development, contributed to the interpretation of findings, critically revised the manuscript for intellectual content, and ensured compliance with reporting standards.All authors approved the final manuscript and agree to be accountable for all aspects of the work.