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Byline: Yi-Ju Chen (1), Chih-Sung Hsieh (2), Hock-Liew Eng (1), Chao-Cheng Huang (1) Keywords: Ovarian fibroma; Ovarian tumour; Infancy; Gorlin's syndrome Abstract: Ovarian fibroma rarely occurs in prepubertal girls. We report an ovarian fibroma in a 7-month-old female infant presenting as a right abdominal mass. The tumour was composed of oval-to-spindle cells forming interlacing fascicles and intermingling with collagen fibres. Immunohistochemically, the tumour cells were weakly positive for estrogen receptor and progesterone receptor but negative for inhibin. To the best of our knowledge, this is the youngest case of ovarian fibroma up-to-date in the English literature. Because Gorlin's syndrome was not present in the patient's family, other factors such as germline or somatic mutations may predispose the occurrence of this ovarian fibroma in infancy. Author Affiliation: (1) Department of Pathology, Chang Gung Memorial Hospital, 123 Ta-Pei Road, Niao-Sung Hsiang, Kaohsiung , County 833, Taiwan (2) Department of Pediatric Surgery, Chang Gung Memorial Hospital, Kaohsiung, Taiwan Article History: Registration Date: 13/08/2004 Accepted Date: 25/07/2003 Online Date: 30/09/2004