Treatment of juvenile cystic adenomyoma through combined insertion of LNG-IUS and GyneFix®: a case report and literature review

Abstract Background Juvenile cystic adenomyoma (JCA) is an uncommon form of adenomyosis. No consensus exists regarding its optimal treatment. Despite the potential long-term complications, lesion resection is the radical therapy for JCA. Given the dual goals of symptom control and fertility preservation in such young adolescents, treatment selection is often challenging, highlighting the need for alternative therapeutic choices. Case presentation and literature review We report the case of a 14-year-old nulliparous female with severe dysmenorrhoea refractory to medical treatment who was successfully managed with a levonorgestrel-releasing intrauterine system (LNG-IUS) fixed to a GyneFix ® frameless intrauterine device (IUD) to prevent re-displacement. A review of 14 published articles (covering 19 reported cases) on JCA revealed a lack of consensus on standardized treatment and highlighted the frequent use of more invasive surgical procedures, such as laparoscopy or laparotomy, for lesion resection. These surgical approaches, while often effective, are associated with potential risks, including uterine wall injury and potential future fertility-related complications. In contrast, the combined IUD approach achieved long-term symptom relief for > 16 months. Conclusion For adolescents with JCA, the combined LNG-IUS and GyneFix ® technique represents a promising, minimally invasive, and fertility-preserving treatment strategy, particularly for patients who previously achieved satisfactory symptom control with Mirena ® therapy but experienced device expulsion or displacement. By effectively reducing the risk of device expulsion, this combined approach offers a conservative alternative to more invasive surgical intervention while aiming to preserve fertility.