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

In: BMC Women's Health · 2026 · doi:10.1186/s12905-026-05051-4 · W7224855843
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This case report and literature review describe a combined LNG-IUS and GyneFix technique that provides long-term symptom relief for juvenile cystic adenomyosis while preserving fertility and preventing device expulsion in adolescents.

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This paper presents a case report and literature review regarding juvenile cystic adenomyoma, an uncommon variant of adenomyosis affecting adolescents. The authors describe a 14-year-old patient successfully treated with a combined levonorgestrel-releasing intrauterine system and GyneFix device to prevent expulsion, achieving long-term symptom relief without invasive surgery. A review of existing literature highlights the lack of consensus on optimal treatment and notes that surgical resection carries risks to fertility, whereas this conservative approach offers a promising alternative for preserving reproductive potential in young patients. This paper is centrally about adenomyosis — specifically the management of juvenile cystic adenomyoma using hormonal intrauterine devices as a fertility-preserving intervention.

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Abstract

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.
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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. Similar content being viewed by others Abbreviations - JCA: - Juvenile cystic adenomyoma - LNG-IUS: - Levonorgestrel-releasing intrauterine system - IUD: - Intrauterine device - CPP: - Chronic pelvic pain - VAS: - Visual analog scale - CA-125: - Cancer antigen 125 - MRI: - Magnetic resonance imaging - TRUS: - Transrectal ultrasonography - OCs: - Oral contraceptives - GnRHa: - Gonadotropin-releasing hormone agonist

Acknowledgements

Not applicable. Funding The authors declare that no funds, grants, or other support were received during the preparation of this manuscript. Author information Authors and Affiliations Corresponding author Ethics declarations Ethics approval and consent to participate According to the regulations of our institution, ethical approval was not required for this single case report. The report was conducted in accordance with the Declaration of Helsinki. Consent for publication Written informed consent was obtained from the patient and her legal guardian for the publication of this case report and any accompanying medical information. Competing interests The authors declare no competing interests. Additional information Publisher’s note Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations. Rights and permissions Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. About this article Cite this article Han, C., Nie, L., Zhang, P. et al. Treatment of juvenile cystic adenomyoma through combined insertion of LNG-IUS and GyneFix®: a case report and literature review. BMC Women's Health (2026). https://doi.org/10.1186/s12905-026-05051-4 Received: Accepted: Published: DOI: https://doi.org/10.1186/s12905-026-05051-4

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