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Zeynep Kamil Kadın ve Çocuk Hastalıkları Eğitim ve Araştırma Hastanesi ¹Pediatri Uzmanı, ²Pediatri Doçenti, ³Pediatri Araştırma Görevlisi, ⁴Genetik Uzmanı, ⁵Pediatri Profesörü Tetraamelia is seen very rarely as a result of interruption of embryonal development between 24-36 days of fertilization. it is a natal disorder characterized by the absence of four extremities and variable etiology it can be isolated or can be associated with other anomalies. Thalidomide is the most important teratogen for which a causal relationship has been determined. A male infant with gestational age of 37 weeks and birth weight of 1950 g was born to a non-consanguineous couple. He was diagnosed to have tetraamelia in the 12th-16th week of gestation, but his parents did not accept termination. Prenatal history revealed meclizine utilization in the first trimester. He was diagnosed to have isolated tetraamelia as he did not have any dysmorphology apart from hemangioma on the nose and philtrum, and there was no family history We aimed to emphasize association of this very rare disorder with meclizine utilization, which may be coincidental. Anahtar Kelimeler : ameli, meklizin.