We report 2 cases of unilateral, multilocular, cystic adrenal masses in neonates. Both patients demonstrated somatic hemihypertrophy, which is considered to have the same implications as the Beckwith-Wiedemann syndrome in terms of tumor risk. Pathological examination showed multiple, adrenal cysts without evidence of tumor or routine adrenal hemorrhage. This finding appears to represent a previously unreported association between the spectrum of hemihypertrophy/Beckwith-Wiedemann syndrome and benign cystic adrenal disease.

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