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Innovating the conservative management of isolated cervical aplasia. Isolated cervical aplasia (ESHRE/ESGE U0C4V0) is a rare condition with an incidence of approximately 1:100,000 births.This congenital malformation of the female genital tract represents an impairment of the outflow tract and is an inevitable cause of infertility. Patients usually present with pelvic pain or haematometra and surgical treatment is needed. Conservative management is the first line of approach, allowing for future fertility. However, complications are not negligible. Choosing the best surgical technique remains controversial as few follow-up studies have been published.We describe a case report of isolated cervical aplasia diagnosed in a 16-year-old patient, managed by a canalisation procedure using a Foley catheter. Following failure of this approach, a levonorgestrel intrauterine system was inserted, which remained efficient after 4 years.This case adds to the few reports of success in the management of this challenging clinical entity and might guide clinicians to avoid non-conservative approaches in young patients.