The authors describe an 11-year-old girl with duplication of a pituitary gland and hypertelorism. Of the 14 cases described to date, only 2 others have lacked associated major facial or oral anomalies. Two separate glands, respective stalks and hypothalamic enlargement were incidentally found on MR imaging performed to evaluate the patient’s complaint of anosmia. A tortuous fusiform dilatation of one pericallosal artery was also discovered. A review of hypophyseal embryogenesis is presented. It is now evident that duplication of the pituitary can occur without other major dysmorphias. This may infer that notochord/prochordal plate deficits are not a prerequisite for hypophyseal duplication.

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