Background: Lobular endocervical glandular hyperplasia (LEGH) is a rare entity of a pseudoneoplastic benign condition of the uterine cervix, and its histogenesis and pathological significance including a connection with carcinogenesis of the endocervical gland has not yet been fully recognized. Case: We describe a rare case of localized LEGH, which developed adjacent to a cesarean section scar. A 53-year-old premenopausal woman presented with a recent onset of abdominal distention and menorrhagia. Magnetic resonance imaging revealed multiple uterine myomas including submucosal myoma and localized small cystic lesions in the proximal area of the anterior wall of the cervix. Total hysterectomy was performed. The cystic lesions were diagnosed as LEGH. Thread-like foreign bodies and inflammatory reaction were demonstrated around several hyperplastic lesions. Focal immunoreactivity for MIB-1 was detected only in the LEGH cells adjacent to the fibrosis and foreign body reaction. Discussion: The histological findings, in relation to the previous cesarean section suggest that the ectopic pyloric hyperplasia in the present case could represent a heteroplastic or metaplastic process due to a multidirectional differentiation of cervical glands during chronic inflammation by foreign bodies.