Aromatase inhibitors
Aromatase inhibitors (e.g., letrozole) may be considered by a specialist for use in the treatment of adenomyosis. The use of aromatase inhibitors is recommended by the European Society of Human Reproduction and Embryology (ESHRE) in conjunction with oral contraceptives, progestins, or gonadotropin-releasing hormone (GnRH) agonists for patients with pain associated with drug-resistant and surgery-resistant rectovaginal endometriosis.[154]Becker CM, Bokor A, Heikinheimo O, et al; ESHRE Endometriosis Guideline Group. ESHRE guideline: endometriosis. Hum Reprod Open. 2022 Feb 26;2022(2):hoac009.
https://academic.oup.com/hropen/article/2022/2/hoac009/6537540
http://www.ncbi.nlm.nih.gov/pubmed/35350465?tool=bestpractice.com
The rationale for using aromatase inhibitors in the treatment of adenomyosis and endometriosis is based on the abnormal expression of aromatase cytochrome P450 in both diseases.[20]Kitawaki J, Noguchi T, Amatsu T, et al. Expression of aromatase cytochrome P450 protein and messenger ribonucleic acid in human endometriotic and adenomyotic tissues but not in normal endometrium. Biol Reprod. 1997 Sep;57(3):514-9.
http://www.ncbi.nlm.nih.gov/pubmed/9282984?tool=bestpractice.com
[155]Yamamoto T, Noguchi T, Tamura T, et al. Evidence for estrogen synthesis in adenomyotic tissues. Am J Obstet Gynecol. 1993 Sep;169(3):734-8.
http://www.ncbi.nlm.nih.gov/pubmed/8372890?tool=bestpractice.com
However, there is no robust evidence regarding the use of aromatase inhibitors for adenomyosis management.[156]Stratopoulou CA, Donnez J, Dolmans MM. Conservative management of uterine adenomyosis: medical vs. surgical approach. J Clin Med. 2021 Oct 22;10(21):4878.
https://www.mdpi.com/2077-0383/10/21/4878
http://www.ncbi.nlm.nih.gov/pubmed/34768397?tool=bestpractice.com
One small randomized controlled trial involving 32 patients with adenomyosis compared the effectiveness of letrozole to the GnRH agonist goserelin over a period of 12 weeks; both groups showed a reduction in total adenomyoma volume at 12 weeks (40.9% for letrozole vs. 49.1% for goserelin) with no significant difference found between the two groups.[114]Badawy AM, Elnashar AM, Mosbah AA. Aromatase inhibitors or gonadotropin-releasing hormone agonists for the management of uterine adenomyosis: a randomized controlled trial. Acta Obstet Gynecol Scand. 2012 Apr;91(4):489-95.
https://obgyn.onlinelibrary.wiley.com/doi/10.1111/j.1600-0412.2012.01350.x
http://www.ncbi.nlm.nih.gov/pubmed/22229256?tool=bestpractice.com
GnRH antagonists
GnRH antagonists induce a dose-dependent suppression of the pituitary-gonadal axis and have an established role in the context of endometriosis and uterine leiomyomas. The data to support the use of GnRH antagonists in adenomyosis are limited, but case reports and pilot studies of GnRH antagonists for adenomyosis have shown promising results, with reduction of lesion size and improved patient quality of life. However, a rapid return to baseline uterine volume and bleeding patterns was observed within 12 weeks of discontinuation.[109]Etrusco A, Barra F, Chiantera V, et al. Current medical therapy for adenomyosis: from bench to bedside. Drugs. 2023 Nov;83(17):1595-611.
https://link.springer.com/article/10.1007/s40265-023-01957-7
http://www.ncbi.nlm.nih.gov/pubmed/37837497?tool=bestpractice.com
[157]Donnez O, Donnez J. Gonadotropin-releasing hormone antagonist (linzagolix): a new therapy for uterine adenomyosis. Fertil Steril. 2020 Sep;114(3):640-5.
https://www.fertstert.org/article/S0015-0282(20)30345-9/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/32507315?tool=bestpractice.com
[158]Donnez J, Donnez O, Dolmans MM. Evolution of uterine adenomyosis volume during and after GnRH antagonist (linzagolix) treatment: lessons for further clinical trials. Fertil Steril. 2023 Nov;120(5):1071-3.
https://www.fertstert.org/article/S0015-0282(23)00699-4/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/37495010?tool=bestpractice.com
[159]Donnez J, Donnez O, Brethous M, et al. Treatment of symptomatic uterine adenomyosis with linzagolix, an oral gonadotrophin-releasing hormone antagonist: a pilot study. Reprod Biomed Online. 2022 Jan;44(1):200-3.
http://www.ncbi.nlm.nih.gov/pubmed/34799277?tool=bestpractice.com
Large-scale studies are required.
Selective progesterone receptor modulators
Selective progesterone receptor modulators (e.g., ulipristal, mifepristone) have been shown to reduce heavy menstrual bleeding and improve quality of life in women with symptomatic fibroids.[157]Donnez O, Donnez J. Gonadotropin-releasing hormone antagonist (linzagolix): a new therapy for uterine adenomyosis. Fertil Steril. 2020 Sep;114(3):640-5.
https://www.fertstert.org/article/S0015-0282(20)30345-9/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/32507315?tool=bestpractice.com
[160]Murji A, Whitaker L, Chow TL, et al. Selective progesterone receptor modulators (SPRMs) for uterine fibroids. Cochrane Database Syst Rev. 2017 Apr 26;(4):CD010770.
https://www.cochranelibrary.com/cdsr/doi/10.1002/14651858.CD010770.pub2/full
http://www.ncbi.nlm.nih.gov/pubmed/28444736?tool=bestpractice.com
Available data suggest that selective progesterone receptor modulators may have a beneficial role in treating patients with abnormal uterine bleeding due to adenomyosis, while the evidence regarding pelvic pain and dysmenorrhea is conflicting.[161]Gonçalves-Henriques M, de Pinho A, Freixo M, et al. Ulipristal acetate in adenomyosis. Gynecol Minim Invasive Ther. 2022 Oct-Dec;11(4):198-202.
https://journals.lww.com/gmit/fulltext/2022/11040/ulipristal_acetate_in_adenomyosis.2.aspx
http://www.ncbi.nlm.nih.gov/pubmed/36660320?tool=bestpractice.com
In one retrospective cohort study, the efficacy of ulipristal was compared between patients with both leiomyomas and adenomyosis (cases) versus patients with leiomyomas alone (controls). The results showed that 90.2% of cases achieved optimal bleeding control compared with 73.8% in the control group (P = 0.028). Improvement in reported visual analog scale scores was also noted in patients treated with ulipristal.[162]Gracia M, Alcalà M, Ferreri J, et al. Ulipristal acetate improves clinical symptoms in women with adenomyosis and uterine myomas. J Minim Invasive Gynecol. 2018 Nov-Dec;25(7):1274-80.
http://www.ncbi.nlm.nih.gov/pubmed/29626678?tool=bestpractice.com
Similar results have been reported using mifepristone in patients with adenomyosis, with improvement in pain as reported by visual analog scale scores.[163]Che X, Wang J, He J, et al. The new application of mifepristone in the relief of adenomyosis-caused dysmenorrhea. Int J Med Sci. 2020 Jan 14;17(2):224-33.
https://www.medsci.org/v17p0224.htm
http://www.ncbi.nlm.nih.gov/pubmed/32038106?tool=bestpractice.com
In one randomized controlled trial investigating the use of ulipristal in patients with adenomyosis, significant improvements in bleeding, amenorrhea, and pain scores were reported. However, no difference was observed after 3 months of stopping the treatment, and there was no improvement in quality of life or anemia after 6 months.[164]Capmas P, Brun JL, Legendre G, et al. Ulipristal acetate use in adenomyosis: a randomized controlled trial. J Gynecol Obstet Hum Reprod. 2021 Jan;50(1):101978.
http://www.ncbi.nlm.nih.gov/pubmed/33186772?tool=bestpractice.com
Contradictory results were reported in a few case series and reports, where worsening of pelvic pain and radiologic findings were observed after 3 months of ulipristal treatment, not only in the extent of the disease but also in the size and number of lesions.[157]Donnez O, Donnez J. Gonadotropin-releasing hormone antagonist (linzagolix): a new therapy for uterine adenomyosis. Fertil Steril. 2020 Sep;114(3):640-5.
https://www.fertstert.org/article/S0015-0282(20)30345-9/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/32507315?tool=bestpractice.com
[165]Conway F, Morosetti G, Camilli S, et al. Ulipristal acetate therapy increases ultrasound features of adenomyosis: a good treatment given in an erroneous diagnosis of uterine fibroids. Gynecol Endocrinol. 2019 Mar;35(3):207-10.
http://www.ncbi.nlm.nih.gov/pubmed/30382803?tool=bestpractice.com
Note that ulipristal has significant adverse effects, including weight gain, fatigue, and abdominal discomfort.[166]Hong YH, Han SJ, Lee D, et al. Adverse symptoms during short-term use of ulipristal acetate in women with uterine myomas and/or adenomyosis. J Obstet Gynaecol Res. 2019 Apr;45(4):865-70.
http://www.ncbi.nlm.nih.gov/pubmed/30675965?tool=bestpractice.com
Safety concerns, including reports of liver injury requiring transplantation, have led to the withdrawal of ulipristal for the treatment of fibroids in some countries.[167]Middelkoop MA, Bet PM, Drenth JPH, et al. Risk-efficacy balance of ulipristal acetate compared to surgical alternatives. Br J Clin Pharmacol. 2021 Jul;87(7):2685-97.
https://bpspubs.onlinelibrary.wiley.com/doi/10.1111/bcp.14708
http://www.ncbi.nlm.nih.gov/pubmed/33341097?tool=bestpractice.com
High-intensity focused ultrasound (HIFU)
HIFU is a minimally invasive thermal ablation procedure that has shown promise for management of adenomyosis.[2]Dason ES, Maxim M, Sanders A, et al; Society of Obstetricians and Gynaecologists of Canada (SOGC). Guideline no. 437: diagnosis and management of adenomyosis. J Obstet Gynaecol Can. 2023 Jun;45(6):417-29.e1.
https://www.jogc.com/article/S1701-2163(23)00307-9/abstract
http://www.ncbi.nlm.nih.gov/pubmed/37244746?tool=bestpractice.com
This is a conservative alternative to surgery that employs focused ultrasound waves to heat targeted tissue, resulting in coagulative necrosis and cell death without damage to surrounding tissues.[168]Cheung VYT. High-intensity focused ultrasound therapy. Best Pract Res Clin Obstet Gynaecol. 2018 Jan;46:74-83.
http://www.ncbi.nlm.nih.gov/pubmed/29074037?tool=bestpractice.com
The procedure is typically monitored with magnetic resonance imaging (MRI) or ultrasound, and does not require incisions.[168]Cheung VYT. High-intensity focused ultrasound therapy. Best Pract Res Clin Obstet Gynaecol. 2018 Jan;46:74-83.
http://www.ncbi.nlm.nih.gov/pubmed/29074037?tool=bestpractice.com
Severe adverse effects have been found to be rare.[169]Fukunishi H, Funaki K, Sawada K, et al. Early results of magnetic resonance-guided focused ultrasound surgery of adenomyosis: analysis of 20 cases. J Minim Invasive Gynecol. 2008 Sep-Oct;15(5):571-9.
http://www.ncbi.nlm.nih.gov/pubmed/18657480?tool=bestpractice.com
[170]Zhou M, Chen JY, Tang LD, et al. Ultrasound-guided high-intensity focused ultrasound ablation for adenomyosis: the clinical experience of a single center. Fertil Steril. 2011 Mar 1;95(3):900-5.
https://www.fertstert.org/article/S0015-0282(10)02696-8/fulltext
http://www.ncbi.nlm.nih.gov/pubmed/21067723?tool=bestpractice.com
HIFU has been found to significantly reduce uterine size in patients with adenomyosis, ranging from 12.7% at 6 months to 54.0% at 12 months in various studies.[168]Cheung VYT. High-intensity focused ultrasound therapy. Best Pract Res Clin Obstet Gynaecol. 2018 Jan;46:74-83.
http://www.ncbi.nlm.nih.gov/pubmed/29074037?tool=bestpractice.com
[169]Fukunishi H, Funaki K, Sawada K, et al. Early results of magnetic resonance-guided focused ultrasound surgery of adenomyosis: analysis of 20 cases. J Minim Invasive Gynecol. 2008 Sep-Oct;15(5):571-9.
http://www.ncbi.nlm.nih.gov/pubmed/18657480?tool=bestpractice.com
It also shows promise in reducing heavy menstrual bleeding, with a 25% to 66% reduction at 12 months reported in studies using MRI-guided focused ultrasound and 48% to 65% reduction in studies using ultrasound-guided focused ultrasound.[171]Ferrari F, Arrigoni F, Miccoli A, et al. Effectiveness of magnetic resonance-guided focused ultrasound surgery (MRgFUS) in the uterine adenomyosis treatment: technical approach and MRI evaluation. Radiol Med. 2016 Feb;121(2):153-61.
http://www.ncbi.nlm.nih.gov/pubmed/26349572?tool=bestpractice.com
[172]Fan TY, Zhang L, Chen W, et al. Feasibility of MRI-guided high intensity focused ultrasound treatment for adenomyosis. Eur J Radiol. 2012 Nov;81(11):3624-30.
http://www.ncbi.nlm.nih.gov/pubmed/21719223?tool=bestpractice.com
HIFU may also improve dysmenorrhea associated with adenomyosis, with reported improvement in menstrual pain scores ranging from 25% to 83%.[173]Polina L, Nyapathy V, Mishra A, et al. Noninvasive treatment of focal adenomyosis with MR-guided focused ultrasound in two patients. Indian J Radiol Imaging. 2012 Apr;22(2):93-7.
https://www.thieme-connect.de/products/ejournals/pdf/10.4103/0971-3026.101078.pdf
http://www.ncbi.nlm.nih.gov/pubmed/23162249?tool=bestpractice.com
A desire for future fertility is considered to be a relative but not absolute contraindication to HIFU.[126]Chen J, Porter AE, Kho KA. Current and future surgical and interventional management options for adenomyosis. Semin Reprod Med. 2020 May;38(2-03):157-67.
http://www.ncbi.nlm.nih.gov/pubmed/33152768?tool=bestpractice.com
However, the data to counsel patients regarding timing, risks, efficacy, and fertility outcomes are still lacking. Combining medical treatment such as GnRH agonists or the levonorgestrel intrauterine device (IUD) with HIFU may also be beneficial.[174]Pang LL, Mei J, Fan LX, et al. Efficacy of high-intensity focused ultrasound combined with GnRH-a for adenomyosis: a systematic review and meta-analysis. Front Public Health. 2021 Aug 16;9:688264.
https://www.frontiersin.org/journals/public-health/articles/10.3389/fpubh.2021.688264/full
http://www.ncbi.nlm.nih.gov/pubmed/34485218?tool=bestpractice.com
[175]Zhao TT, Pang LL, Yang LL, et al. Efficacy of high-intensity focused ultrasound combined with LNG-IUS for adenomyosis: a systematic review and meta-analysis. Arch Gynecol Obstet. 2023 Aug;308(2):351-62.
http://www.ncbi.nlm.nih.gov/pubmed/35947146?tool=bestpractice.com
Uterine-sparing excision surgery (for diffuse adenomyosis)
Uterine-sparing excision surgery (adenomyomectomy) may be considered as a last resort for diffuse adenomyosis in patients who wish to preserve fertility. It must be performed by highly skilled and experienced surgeons in specialized centers. The surgical procedure for resection of diffuse disease is significantly more challenging than for focal disease, and carries considerable perioperative risks. It typically requires advanced techniques performed primarily via laparotomy, although minimally invasive approaches have also been reported.[141]Hlinecka K, Mara M, Boudova B, et al. Comparison of clinical and reproductive outcomes between adenomyomectomy and myomectomy. J Minim Invasive Gynecol. 2022 Mar;29(3):392-400.
http://www.ncbi.nlm.nih.gov/pubmed/34670164?tool=bestpractice.com
[176]Moawad G, Feki A, Ayoubi JM, et al. Tips and tricks for robotic management of diffuse adenomyosis. Fertil Steril. 2024 Nov 29 [Epub ahead of print].
http://www.ncbi.nlm.nih.gov/pubmed/39617093?tool=bestpractice.com
Careful counseling is essential regarding the uncertain impact on fertility and pregnancy outcomes and the increased risk of serious complications in any subsequent pregnancy.[2]Dason ES, Maxim M, Sanders A, et al; Society of Obstetricians and Gynaecologists of Canada (SOGC). Guideline no. 437: diagnosis and management of adenomyosis. J Obstet Gynaecol Can. 2023 Jun;45(6):417-29.e1.
https://www.jogc.com/article/S1701-2163(23)00307-9/abstract
http://www.ncbi.nlm.nih.gov/pubmed/37244746?tool=bestpractice.com
The risk of uterine rupture is increased with a greater extent of excision and associated larger size of myometrial defect.[2]Dason ES, Maxim M, Sanders A, et al; Society of Obstetricians and Gynaecologists of Canada (SOGC). Guideline no. 437: diagnosis and management of adenomyosis. J Obstet Gynaecol Can. 2023 Jun;45(6):417-29.e1.
https://www.jogc.com/article/S1701-2163(23)00307-9/abstract
http://www.ncbi.nlm.nih.gov/pubmed/37244746?tool=bestpractice.com
Many experts recommend prelabor cesarean delivery.[126]Chen J, Porter AE, Kho KA. Current and future surgical and interventional management options for adenomyosis. Semin Reprod Med. 2020 May;38(2-03):157-67.
http://www.ncbi.nlm.nih.gov/pubmed/33152768?tool=bestpractice.com
According to one meta-analysis, the rupture rate after surgery for diffuse adenomyosis was found to be 6.8%; however, only 44 cases were included in this analysis.[177]Tan J, Moriarty S, Taskin O, et al. Reproductive outcomes after fertility-sparing surgery for focal and diffuse adenomyosis: a systematic review. J Minim Invasive Gynecol. 2018 May-Jun;25(4):608-21.
http://www.ncbi.nlm.nih.gov/pubmed/29305234?tool=bestpractice.com
Subsequent retrospective studies have shown no cases of uterine rupture following diffuse excisional surgery, based on 75 pregnancy outcomes across two studies.[178]Zhou Y, Shen L, Wang Y, et al. Long-term pregnancy outcomes of patients with diffuse adenomyosis after double-flap adenomyomectomy. J Clin Med. 2022 Jun 17;11(12):3489.
https://www.mdpi.com/2077-0383/11/12/3489
http://www.ncbi.nlm.nih.gov/pubmed/35743559?tool=bestpractice.com
[179]Wu R, Zeng L, Hu Q, et al. Outcome of uterine functional structures protection by fertility preservative PUSH surgery in diffuse adenomyosis. Hum Fertil (Camb). 2023 Oct;26(4):720-32.
https://www.tandfonline.com/doi/full/10.1080/14647273.2023.2260102
http://www.ncbi.nlm.nih.gov/pubmed/37913797?tool=bestpractice.com
Hysteroscopic excision
Hysteroscopic resection of adenomyosis has been described in the literature, but data on its safety, efficacy, and impact on fertility are scarce.[126]Chen J, Porter AE, Kho KA. Current and future surgical and interventional management options for adenomyosis. Semin Reprod Med. 2020 May;38(2-03):157-67.
http://www.ncbi.nlm.nih.gov/pubmed/33152768?tool=bestpractice.com
One case series of 51 patients who underwent hysteroscopic resection of adenomyotic cysts under transabdominal ultrasound guidance found that bleeding symptoms improved in 65%, with partial or complete relief.[180]Xia W, Zhang D, Zhu Q, et al. Hysteroscopic excision of symptomatic myometrial adenomyosis: feasibility and effectiveness. BJOG. 2017 Sep;124(10):1615-20.
http://www.ncbi.nlm.nih.gov/pubmed/28544260?tool=bestpractice.com
At 2-year follow-up, clinical effectiveness was reported in 84.6% of patients, and over 93% of women reported improvement in dysmenorrhea.[180]Xia W, Zhang D, Zhu Q, et al. Hysteroscopic excision of symptomatic myometrial adenomyosis: feasibility and effectiveness. BJOG. 2017 Sep;124(10):1615-20.
http://www.ncbi.nlm.nih.gov/pubmed/28544260?tool=bestpractice.com
No fertility outcomes were reported.