Laparoscopic adenomyomectomy in the single uterus of the Uterine didelphys

Laparoscopic adenomyomectomy in the single uterus of the Uterine didelphys | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Case Report Laparoscopic adenomyomectomy in the single uterus of the Uterine didelphys Jisu Hong, Jeong Soo Lee, Sujin Kim, Jae Young Kwack, Yong-Soon Kwon This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-2497639/v1 This work is licensed under a CC BY 4.0 License Status: Posted Version 1 posted You are reading this latest preprint version Abstract Background Because the surgical approach was in challenging, we adjusted adnomyomectomy in the single uterus of a patient with uterus didelphys and adenomyosis and this is the first reported case that was successfully treated. Case presentation A 31-year-old primipara woman with dysmenorrhea and hypermenorrhea and a known history of uterus didelphys and left kidney agenesis was referred to our hospital. Ultrasonography revealed adenomyosis in the right uterus. She had a plan to have another child and wanted to preserve fertility. Laparoscopic adenomyomectomy was performed in the right uterus with conservation of the original structure of uterine didelphys. Transient occlusion of uterine artery (TOUA) was employed in the right uterine artery, which inhibited the circulation to the right uterus. The incised adenomyosis lesion weighed 20 g, operative time was 55 min, and estimated blood loss was 50 mL. After 5-day hospitalization, the patient was discharged without any notable complications. After 7 months of the surgery, symptoms, and adenomyoma on ultrasonography were improved. To the best of our knowledge, this is the first report of laparoscopic adenomyomectomy surgery performed on a patient with uterine didelphys. Conclusion Laparoscopic adenomyomectomy can be considered as a feasible treatment option for patients with uterus didelphys who want to preserve fertility. Uterine adenomyosis Laparoscopy Uterine Didelphys Adenomyomectomy Figures Figure 1 Figure 2 Figure 3 Introduction Adenomyosis is characterized by the presence of endometrial tissue within the myometrium ( 1 ). Its symptoms are usually dysmenorrhea or menorrhagia, and it can cause infertility. Symptoms except infertility can be managed by medical treatments such as gonadotropin-releasing hormone agonists (GnRHa), contraceptive medication, or hysterectomy ( 2 ). However recently, there has been an increase in the patients with adenomyosis who want to relieve their symptoms and preserve fertility. For these patients, uterus-sparing adenomyomectomy surgery could be an available option. Some reports demonstrate the safety, efficacy, and pregnancy outcomes of adenomyomectomy surgery ( 3 – 5 ). Congenital malformations of the female genital tract occur because of embryonic maldevelopment of the müllerian or paramesonephric ducts, with the mean prevalence rate of 5.5% in the general population. Uterus didelphys is one of these, and manifests as two separate uterine bodies and two separate cervices with or without longitudinal vaginal septum; the vagina can be either one or two ( 6 ). Previously, only a few papers reported uterus didelphys with adenomyosis. Because the surgical approach was in challenging there were a few reports that had tried uterine artery embolization ( 7 ) or hysterectomy ( 8 ). And a report of hemi-hysterectomy in case of uterine didelphys for reasons other than adenomyosis, could be suggested ( 9 ). However, we adjusted adnomyomectomy in the single uterus of a patient with uterus didelphys and adenomyosis and this is the first reported case that was successfully treated. Case Report A 31-year-old primipara woman with dysmenorrhea and hypermenorrhea, and a known history of uterus didelphys with single vagina and left kidney agenesis was referred to our hospital. She had taken oral contraceptives because of the symptoms. In the ultrasonography, uterus didelphys was confirmed and adenomyosis was noted in the right uterus. (Fig. 1 ) She had a history of preterm delivery at 23-weeks by cesarean delivery due to preterm labor and chorioamnionitis and had a plan to have another child. The patient stopped using oral-pill and agreed to uterus conserving surgery. This study was approved by the Institutional Review Board of Nowon Eulji Medical Center (IRB No. 2019-07-023). Informed consent to participate was obtained. On December 15, 2020, surgery was performed by following the procedure. A uterine manipulator was inserted into the right side of the uterus, which was affected by adenomyosis. Left adnexa was identified to adhere to the posterior pelvic wall and the left uterosacral ligament was thickened. Laparoscopy revealed pelvic endometriosis with adhesion. After adhesiolysis and excision of the endometriosis lesion, the right ureter was identified and the umbilical artery was isolated by blunt-tip suction to find out the branching site of the uterine artery from the internal iliac artery crossing the ureter. The right-side uterine artery was occluded with an endoscopic transient bulldog clamp (Aesculap, Inc. Center Valley, PA, USA). This procedure is called transient occlusion of uterine artery (TOUA), which intends to reduce intraoperative blood loss to ensure a stable operation field ( 10 ). It is usually performed on both the uterine arteries. However, in this case, only the right uterine artery was clamped which led to inhibition of circulation to the right uterus. A horizontal incision was made on the fundus to reveal the endometrium. Using a hook-shaped monopolar electrode, wedge resection was performed to reduce the size of the uterus and ease the procedure of uteroplasty. After that, the adenomyotic lesion between the endometrial side to the serosa was more deeply excised for further complete excision. For reconstruction of the uterus, it was sutured layer by layer with continuous barbed suture. The endometrium was closed by suturing the adjacent myometrium without penetration into the endometrium itself. (Fig. 2 ) Excised adenomyotic lesion was removed by using the port site. Incised adenomyosis lesion weighed 20 g, operative time was 55 min, and estimated blood loss was 50 mL. The baseline hemoglobin level 2-weeks before the surgery was 13.8 g/dL and the postoperative hemoglobin level was 11.0 g/dL at Postoperative day (POD) 3. After 5-day hospitalization, the patient was discharged without any notable complications, and her condition was assessed as Clavien-Dindo’s classification grade I ( 11 ). The patient visited the clinic for 3 cycles of gonadotropin-releasing hormone agonist (GnRHa) subcutaneous injection at a monthly interval. With regular check-up visits after 7 months and 13 months from the surgery, symptoms, and adenomyoma on ultrasonography were improved and continued to the resolving state. (Fig. 3 ) Discussion There is a continuous increase in the demand for uterus-sparing surgery in women with adenomyosis, who not only want to resolve the symptoms but also conserve fertility. There exist reports that demonstrate uterine artery embolization or hysterectomy in patients with uterus didelphys and adenomyosis; however, this is the first report of laparoscopic adenomyomectomy in a patient with uterus didelphys. In patients with uterus didelphys and adenomyosis, it is exactly not known which surgery or procedure will help improve fertility. In a systemic review that demonstrated the reproductive outcome of various congenital uterine anomalies, unification defects, such as the bicornuate, unicornuate, and didelphic uterus, did not appear to reduce fertility but were identified to be associated with aberrant outcomes throughout pregnancy. Women with unicornuate uteri have an increased risk of miscarriage, preterm birth, and fetal malpresentation while women with uterus didelphys seem to have only a modestly increased risk of preterm labor ( 12 ). Considering this finding that unicornuate uterus does not show a better outcome compared to uterus didelphys, there is no evidence that adenomyomectomy with preservation of the structure of uterus didelphys, might be inferior to hemi-hysterectomy. Particularly, in the case of the present patient with uterus didelphys, the exact uterus was not known, but she had experienced her first pregnancy and childbirth before. In planning the next pregnancy, it was judged that it would be better to proceed with conservative surgery that maintains the original uterine structure rather than hemi-hysterectomy in terms of conserving fertility. Compared to uterine artery embolization, it is advantageous to perform adenomyomectomy as other possible causes such as combined endometriosis can be treated simultaneously. Besides, a study confirmed decreased ovarian function after uterine artery embolization, with an interpretation that the uterine artery embolism could cause deterioration of ovarian function and infertility ( 13 ). Another study in myoma patients (a randomized group of 121 women who were followed up for 2 years) revealed that surgical myomectomy showed a better reproductive outcome compared to uterine artery embolization ( 14 ). From this point of view, it can be assumed that the effects of preserving fertility (ovarian function) might be minimized when applying the surgical method using TOUA that temporarily blocks the blood vessels and results in re-perfusion of the uterine artery. The absorbable barbed suture materials make the suturing faster and stronger compared to earlier times. Suturing the three anatomic layers in the uterus would be helpful to maintain the structure of the uterus. However, concerning adenomyomectomy surgery, there have been arguments on the risk of uterine rupture during pregnancy. There exist some case reports about uterine rupture after adenomyomectomy ( 15 ). A single-institution study involving a review of 224 patients who underwent laparotomic or laparoscopic adenomyomectomy reported the pregnancy outcomes as 14 conceptions, 3 missed abortions, and the rest delivered safely by cesarean section without uterine rupture ( 16 ). Another retrospective cohort study showed 10 cases of delivery without uterine rupture after adenomyomectomy ( 17 ). After adenomyomectomy, the patients should be informed about the possible complications, and precise antenatal care should be provided. The imaging methods such as three-dimensional (3D) sonography or magnetic resonance imaging (MRI) can be considered to determine uterine wall thickness during pregnancy. Our patient also received individualized counseling, and it is proposed that close observation is necessary during pregnancy. In conclusion, although it was necessary to continuously monitor the development of problems with subsequent pregnancy and delivery in the present patient, the patient was discharged without major complications after the operation, and improvement in symptoms has been maintained since the past 13 months after the operation. Thus, it is proposed that through sufficient explanation and consultation, laparoscopic adenomyomectomy can be considered as a feasible treatment option for patients with uterus didelphys who want to preserve fertility. Declarations Ethical Approval and consent to participate This study was approved by the Institutional Review Board of Nowon Eulji Medical Center (IRB No. 2019-07-023) and informed consent was obtained. Consent to Publication The patient signed informed consent for the publication of this case report and any associated images. A copy of the consent form is available for review by the Editor of this journal. Data Availability statement The datasets used and/or analysed during the current study available from the corresponding author on reasonable request. Conflict of interest The authors declare that they have no competing interests. Funding Not applicable. Acknowledgment Not applicable. Author contribution JH wrote the manuscript. JYK and YSK analyzed the data and revised the manuscript. JSL and SK was responsible for acquisition and interpretation of the image. All authors read and approved the final manuscript. References Brosens JJ, Barker FG, de Souza NM. Myometrial zonal differentiation and uterine junctional zone hyperplasia in the non-pregnant uterus. Hum Reprod Update. 1998;4(5):496-502. Soave I, Wenger JM, Pluchino N, Marci R. Treatment options and reproductive outcome for adenomyosis-associated infertility. Curr Med Res Opin. 2018;34(5):839-49. Osada H, Silber S, Kakinuma T, Nagaishi M, Kato K, Kato O. Surgical procedure to conserve the uterus for future pregnancy in patients suffering from massive adenomyosis. Reprod Biomed Online. 2011;22(1):94-9. Kwack JY, Kwon YS. Conservative surgery of diffuse adenomyosis with TOUA: Single surgeon experience of one hundred sixteen cases and report of fertility outcomes. Kaohsiung J Med Sci. 2018;34(5):290-4. Kishi Y, Yabuta M, Taniguchi F. Who will benefit from uterus-sparing surgery in adenomyosis-associated subfertility? Fertil Steril. 2014;102(3):802-7 e1. Chan YY, Jayaprakasan K, Zamora J, Thornton JG, Raine-Fenning N, Coomarasamy A. The prevalence of congenital uterine anomalies in unselected and high-risk populations: a systematic review. Hum Reprod Update. 2011;17(6):761-71. Kim JY, Kim MD, Cho JH, Park SI, Lee MS, Lee MS. Uterine artery embolization for symptomatic adenomyosis in a patient with uterus didelphys. J Vasc Interv Radiol. 2011;22(10):1489-91. Yang CC, Tseng JY, Chen P, Wang PH. Uterus didelphys with cervical agenesis associated with adenomyosis, a leiomyoma and ovarian endometriosis. A case report. J Reprod Med. 2002;47(11):936-8. Kai K, Kawano Y, Yano M, Okamoto M, Hori E, Nasu K, et al. Two cesarean deliveries after hemi-hysterectomy due to gestational trophoblastic neoplasia. Taiwan J Obstet Gynecol. 2018;57(2):315-8. Kwack JY, Kwon YS. Laparoscopic Surgery for Focal Adenomyosis. JSLS. 2017;21(2). Clavien PA, Barkun J, de Oliveira ML, Vauthey JN, Dindo D, Schulick RD, et al. The Clavien-Dindo Classification of Surgical Complications: Five-Year Experience. Annals of Surgery 2009;250(2): 187-196 Chan YY, Jayaprakasan K, Tan A, Thornton JG, Coomarasamy A, Raine-Fenning NJ. Reproductive outcomes in women with congenital uterine anomalies: a systematic review. Ultrasound Obstet Gynecol. 2011;38(4):371-82. Hehenkamp WJ, Volkers NA, Broekmans FJ, de Jong FH, Themmen AP, Birnie E, et al. Loss of ovarian reserve after uterine artery embolization: a randomized comparison with hysterectomy. Hum Reprod. 2007;22(7):1996-2005. Mara M, Maskova J, Fucikova Z, Kuzel D, Belsan T, Sosna O. Midterm clinical and first reproductive results of a randomized controlled trial comparing uterine fibroid embolization and myomectomy. Cardiovasc Intervent Radiol. 2008;31(1):73-85. Kwack JY, Jeon SB, Kim K, Lee SJ, Kwon YS. Monochorionic twin delivery after conservative surgical treatment of a patient with severe diffuse uterine adenomyosis without uterine rupture. Obstet Gynecol Sci. 2016;59(4):311-5. Kwack JY, Im KS, Kwon YS. Conservative surgery of uterine adenomyosis via laparoscopic versus laparotomic approach in a single institution. J Obstet Gynaecol Res. 2018;44(7):1268-73. Sugiyama M, Takahashi H, Baba Y, Taneichi A, Suzuki H, Usui R, et al. Perinatal outcome of pregnancy after adenomyomectomy: summary of 10 cases with a brief literature review. J Matern Fetal Neonatal Med. 2020;33(24):4145-9. Additional Declarations No competing interests reported. Cite Share Download PDF Status: Posted Version 1 posted You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. We do this by developing innovative software and high quality services for the global research community. Our growing team is made up of researchers and industry professionals working together to solve the most critical problems facing scientific publishing. Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-2497639","acceptedTermsAndConditions":true,"allowDirectSubmit":true,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":176294719,"identity":"b6b8e956-ac45-4e11-bdd5-cf9a052d4558","order_by":0,"name":"Jisu Hong","email":"","orcid":"","institution":"Nowon Eulji Medical Center, Eulji University","correspondingAuthor":false,"prefix":"","firstName":"Jisu","middleName":"","lastName":"Hong","suffix":""},{"id":176294720,"identity":"3a8cd078-c092-491a-8657-5f1443e40d9b","order_by":1,"name":"Jeong Soo Lee","email":"","orcid":"","institution":"Nowon Eulji Medical Center, Eulji University","correspondingAuthor":false,"prefix":"","firstName":"Jeong","middleName":"Soo","lastName":"Lee","suffix":""},{"id":176294721,"identity":"ba1bbb8a-0cfc-44e0-baba-927022eeb9f0","order_by":2,"name":"Sujin Kim","email":"","orcid":"","institution":"Nowon Eulji Medical Center, Eulji University","correspondingAuthor":false,"prefix":"","firstName":"Sujin","middleName":"","lastName":"Kim","suffix":""},{"id":176294722,"identity":"50c68d86-6660-44ab-b33f-a90f73e5bccd","order_by":3,"name":"Jae Young Kwack","email":"","orcid":"","institution":"Nowon Eulji Medical Center, Eulji University","correspondingAuthor":false,"prefix":"","firstName":"Jae","middleName":"Young","lastName":"Kwack","suffix":""},{"id":176294723,"identity":"b26fabb0-f808-4a81-96a6-98827c2b3aa4","order_by":4,"name":"Yong-Soon Kwon","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAA6ElEQVRIiWNgGAWjYBACCQkwdYCBgb0HzOLhI14LzxmIFjYonwgtEjkQEYJaJGc3H3v4peZO4nbJtwc/F+bYybAx8B58/AGPFmmZY+nGMseeJe6cnZcsPXNbMtBhfMkG+GyRk8gxk5ZsOJy44XaOgTTvNmagFh4zCeK03Dxj/Jt3Wz1Ii/kPfFqkgVokP4K03OAxA9pyGGwLfu/POZYmzXDssPGGMzlm1rzbjvOwMfMYS5zBo0XidvMxyR81h2U3HD9jfJt3W7U9P3uP4YcKPFpAgJkHlUtAOQgw/iBC0SgYBaNgFIxgAADHIkpphLnTKAAAAABJRU5ErkJggg==","orcid":"","institution":"Nowon Eulji Medical Center, Eulji University","correspondingAuthor":true,"prefix":"","firstName":"Yong-Soon","middleName":"","lastName":"Kwon","suffix":""}],"badges":[],"createdAt":"2023-01-20 03:59:15","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-2497639/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-2497639/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":33049879,"identity":"f2effafd-b9c6-438c-9319-cd3eed09ea9d","added_by":"auto","created_at":"2023-02-16 20:14:01","extension":"png","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":1259577,"visible":true,"origin":"","legend":"\u003cp\u003ePre-operative trans-vaginal ultrasonogram of the patient. A coronal view of the uterus presents uterus didelphys with adenomyosis on the right uterus at the time of the visit.\u003c/p\u003e","description":"","filename":"figure1.png","url":"https://assets-eu.researchsquare.com/files/rs-2497639/v1/7cdbeb2e774bab1861fc8d55.png"},{"id":33049877,"identity":"9278f38d-843a-4960-9d37-27313ce199dd","added_by":"auto","created_at":"2023-02-16 20:14:01","extension":"png","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":1625095,"visible":true,"origin":"","legend":"\u003cp\u003eThe final ultrasonogram after adenomyomectomy of the right uterus.\u003c/p\u003e","description":"","filename":"figure2.png","url":"https://assets-eu.researchsquare.com/files/rs-2497639/v1/8f1ee4f2f0509b99475490d1.png"},{"id":33050133,"identity":"ab1ff156-246f-40c3-9f2e-78c8e912bca3","added_by":"auto","created_at":"2023-02-16 20:22:01","extension":"png","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":1331358,"visible":true,"origin":"","legend":"\u003cp\u003eA transvaginal ultrasonogram shows the resolving state of the adenomyosis on the right side 7 months after the surgery.\u003c/p\u003e","description":"","filename":"figure3.png","url":"https://assets-eu.researchsquare.com/files/rs-2497639/v1/4171f7bb6202f34e23f1b896.png"},{"id":33822672,"identity":"15ca92ba-ea0d-4238-aa83-1dbc5feb59db","added_by":"auto","created_at":"2023-03-06 07:59:47","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":2499366,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-2497639/v1/aa07040d-161f-48d7-94df-17f653eac171.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"Laparoscopic adenomyomectomy in the single uterus of the Uterine didelphys","fulltext":[{"header":"Introduction","content":"\u003cp\u003eAdenomyosis is characterized by the presence of endometrial tissue within the myometrium (\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e). Its symptoms are usually dysmenorrhea or menorrhagia, and it can cause infertility. Symptoms except infertility can be managed by medical treatments such as gonadotropin-releasing hormone agonists (GnRHa), contraceptive medication, or hysterectomy (\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e). However recently, there has been an increase in the patients with adenomyosis who want to relieve their symptoms and preserve fertility. For these patients, uterus-sparing adenomyomectomy surgery could be an available option. Some reports demonstrate the safety, efficacy, and pregnancy outcomes of adenomyomectomy surgery (\u003cspan additionalcitationids=\"CR4\" citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eCongenital malformations of the female genital tract occur because of embryonic maldevelopment of the m\u0026uuml;llerian or paramesonephric ducts, with the mean prevalence rate of 5.5% in the general population. Uterus didelphys is one of these, and manifests as two separate uterine bodies and two separate cervices with or without longitudinal vaginal septum; the vagina can be either one or two (\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e). Previously, only a few papers reported uterus didelphys with adenomyosis. Because the surgical approach was in challenging there were a few reports that had tried uterine artery embolization (\u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e) or hysterectomy (\u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e). And a report of hemi-hysterectomy in case of uterine didelphys for reasons other than adenomyosis, could be suggested (\u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e). However, we adjusted adnomyomectomy in the single uterus of a patient with uterus didelphys and adenomyosis and this is the first reported case that was successfully treated.\u003c/p\u003e"},{"header":"Case Report","content":"\u003cp\u003eA 31-year-old primipara woman with dysmenorrhea and hypermenorrhea, and a known history of uterus didelphys with single vagina and left kidney agenesis was referred to our hospital. She had taken oral contraceptives because of the symptoms. In the ultrasonography, uterus didelphys was confirmed and adenomyosis was noted in the right uterus. (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003e) She had a history of preterm delivery at 23-weeks by cesarean delivery due to preterm labor and chorioamnionitis and had a plan to have another child. The patient stopped using oral-pill and agreed to uterus conserving surgery. This study was approved by the Institutional Review Board of Nowon Eulji Medical Center (IRB No. 2019-07-023). Informed consent to participate was obtained.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003eOn December 15, 2020, surgery was performed by following the procedure. A uterine manipulator was inserted into the right side of the uterus, which was affected by adenomyosis. Left adnexa was identified to adhere to the posterior pelvic wall and the left uterosacral ligament was thickened. Laparoscopy revealed pelvic endometriosis with adhesion. After adhesiolysis and excision of the endometriosis lesion, the right ureter was identified and the umbilical artery was isolated by blunt-tip suction to find out the branching site of the uterine artery from the internal iliac artery crossing the ureter. The right-side uterine artery was occluded with an endoscopic transient bulldog clamp (Aesculap, Inc. Center Valley, PA, USA). This procedure is called transient occlusion of uterine artery (TOUA), which intends to reduce intraoperative blood loss to ensure a stable operation field (\u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e). It is usually performed on both the uterine arteries. However, in this case, only the right uterine artery was clamped which led to inhibition of circulation to the right uterus. A horizontal incision was made on the fundus to reveal the endometrium. Using a hook-shaped monopolar electrode, wedge resection was performed to reduce the size of the uterus and ease the procedure of uteroplasty. After that, the adenomyotic lesion between the endometrial side to the serosa was more deeply excised for further complete excision. For reconstruction of the uterus, it was sutured layer by layer with continuous barbed suture. The endometrium was closed by suturing the adjacent myometrium without penetration into the endometrium itself. (Fig.\u0026nbsp;\u003cspan refid=\"Fig2\" class=\"InternalRef\"\u003e2\u003c/span\u003e) Excised adenomyotic lesion was removed by using the port site.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003eIncised adenomyosis lesion weighed 20 g, operative time was 55 min, and estimated blood loss was 50 mL. The baseline hemoglobin level 2-weeks before the surgery was 13.8 g/dL and the postoperative hemoglobin level was 11.0 g/dL at Postoperative day (POD) 3. After 5-day hospitalization, the patient was discharged without any notable complications, and her condition was assessed as Clavien-Dindo\u0026rsquo;s classification grade I (\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eThe patient visited the clinic for 3 cycles of gonadotropin-releasing hormone agonist (GnRHa) subcutaneous injection at a monthly interval. With regular check-up visits after 7 months and 13 months from the surgery, symptoms, and adenomyoma on ultrasonography were improved and continued to the resolving state. (Fig.\u0026nbsp;\u003cspan refid=\"Fig3\" class=\"InternalRef\"\u003e3\u003c/span\u003e)\u003c/p\u003e \u003cp\u003e \u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eThere is a continuous increase in the demand for uterus-sparing surgery in women with adenomyosis, who not only want to resolve the symptoms but also conserve fertility. There exist reports that demonstrate uterine artery embolization or hysterectomy in patients with uterus didelphys and adenomyosis; however, this is the first report of laparoscopic adenomyomectomy in a patient with uterus didelphys.\u003c/p\u003e \u003cp\u003eIn patients with uterus didelphys and adenomyosis, it is exactly not known which surgery or procedure will help improve fertility. In a systemic review that demonstrated the reproductive outcome of various congenital uterine anomalies, unification defects, such as the bicornuate, unicornuate, and didelphic uterus, did not appear to reduce fertility but were identified to be associated with aberrant outcomes throughout pregnancy. Women with unicornuate uteri have an increased risk of miscarriage, preterm birth, and fetal malpresentation while women with uterus didelphys seem to have only a modestly increased risk of preterm labor (\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e). Considering this finding that unicornuate uterus does not show a better outcome compared to uterus didelphys, there is no evidence that adenomyomectomy with preservation of the structure of uterus didelphys, might be inferior to hemi-hysterectomy. Particularly, in the case of the present patient with uterus didelphys, the exact uterus was not known, but she had experienced her first pregnancy and childbirth before. In planning the next pregnancy, it was judged that it would be better to proceed with conservative surgery that maintains the original uterine structure rather than hemi-hysterectomy in terms of conserving fertility.\u003c/p\u003e \u003cp\u003eCompared to uterine artery embolization, it is advantageous to perform adenomyomectomy as other possible causes such as combined endometriosis can be treated simultaneously. Besides, a study confirmed decreased ovarian function\u003c/p\u003e \u003cp\u003eafter uterine artery embolization, with an interpretation that the uterine artery embolism could cause deterioration of ovarian function and infertility (\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e). Another study in myoma patients (a randomized group of 121 women who were followed up for 2 years) revealed that surgical myomectomy showed a better reproductive outcome compared to uterine artery embolization (\u003cspan citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e). From this point of view, it can be assumed that the effects of preserving fertility (ovarian function) might be minimized when applying the surgical method using TOUA that temporarily blocks the blood vessels and results in re-perfusion of the uterine artery.\u003c/p\u003e \u003cp\u003eThe absorbable barbed suture materials make the suturing faster and stronger compared to earlier times. Suturing the three anatomic layers in the uterus would be helpful to maintain the structure of the uterus. However, concerning adenomyomectomy surgery, there have been arguments on the risk of uterine rupture during pregnancy. There exist some case reports about uterine rupture after adenomyomectomy (\u003cspan citationid=\"CR15\" class=\"CitationRef\"\u003e15\u003c/span\u003e). A single-institution study involving a review of 224 patients who underwent laparotomic or laparoscopic adenomyomectomy reported the pregnancy outcomes as 14 conceptions, 3 missed abortions, and the rest delivered safely by cesarean section without uterine rupture (\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e). Another retrospective cohort study showed 10 cases of delivery without uterine rupture after adenomyomectomy (\u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e). After adenomyomectomy, the patients should be informed about the possible complications, and precise antenatal care should be provided. The imaging methods such as three-dimensional (3D) sonography or magnetic resonance imaging (MRI) can be considered to determine uterine wall thickness during pregnancy. Our patient also received individualized counseling, and it is proposed that close observation is necessary during pregnancy.\u003c/p\u003e \u003cp\u003eIn conclusion, although it was necessary to continuously monitor the development of problems with subsequent pregnancy and delivery in the present patient, the patient was discharged without major complications after the operation, and improvement in symptoms has been maintained since the past 13 months after the operation. Thus, it is proposed that through sufficient explanation and consultation, laparoscopic adenomyomectomy can be considered as a feasible treatment option for patients with uterus didelphys who want to preserve fertility.\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003eEthical Approval and consent to participate\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThis study was approved by the Institutional Review Board of Nowon Eulji Medical Center (IRB No. 2019-07-023) and informed consent was obtained.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConsent to Publication\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe patient signed informed consent for the publication of this case report and any associated images. A copy of the consent form is available for review by the Editor of this journal.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eData Availability statement\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe datasets used and/or analysed during the current study available from the corresponding author on reasonable request.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConflict of interest\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe authors declare that they have no competing interests.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eFunding\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNot applicable.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAcknowledgment\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNot applicable.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAuthor contribution\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eJH wrote the manuscript. JYK and YSK analyzed the data and revised the manuscript. JSL and SK was responsible for acquisition and interpretation of the image. All authors read and approved the final manuscript.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n\u003cli\u003eBrosens JJ, Barker FG, de Souza NM. Myometrial zonal differentiation and uterine junctional zone hyperplasia in the non-pregnant uterus. Hum Reprod Update. 1998;4(5):496-502.\u003c/li\u003e\n\u003cli\u003eSoave I, Wenger JM, Pluchino N, Marci R. Treatment options and reproductive outcome for adenomyosis-associated infertility. Curr Med Res Opin. 2018;34(5):839-49.\u003c/li\u003e\n\u003cli\u003eOsada H, Silber S, Kakinuma T, Nagaishi M, Kato K, Kato O. Surgical procedure to conserve the uterus for future pregnancy in patients suffering from massive adenomyosis. Reprod Biomed Online. 2011;22(1):94-9.\u003c/li\u003e\n\u003cli\u003eKwack JY, Kwon YS. Conservative surgery of diffuse adenomyosis with TOUA: Single surgeon experience of one hundred sixteen cases and report of fertility outcomes. Kaohsiung J Med Sci. 2018;34(5):290-4.\u003c/li\u003e\n\u003cli\u003eKishi Y, Yabuta M, Taniguchi F. Who will benefit from uterus-sparing surgery in adenomyosis-associated subfertility? Fertil Steril. 2014;102(3):802-7 e1.\u003c/li\u003e\n\u003cli\u003eChan YY, Jayaprakasan K, Zamora J, Thornton JG, Raine-Fenning N, Coomarasamy A. The prevalence of congenital uterine anomalies in unselected and high-risk populations: a systematic review. Hum Reprod Update. 2011;17(6):761-71.\u003c/li\u003e\n\u003cli\u003eKim JY, Kim MD, Cho JH, Park SI, Lee MS, Lee MS. Uterine artery embolization for symptomatic adenomyosis in a patient with uterus didelphys. J Vasc Interv Radiol. 2011;22(10):1489-91.\u003c/li\u003e\n\u003cli\u003eYang CC, Tseng JY, Chen P, Wang PH. Uterus didelphys with cervical agenesis associated with adenomyosis, a leiomyoma and ovarian endometriosis. A case report. J Reprod Med. 2002;47(11):936-8.\u003c/li\u003e\n\u003cli\u003eKai K, Kawano Y, Yano M, Okamoto M, Hori E, Nasu K, et al. Two cesarean deliveries after hemi-hysterectomy due to gestational trophoblastic neoplasia. Taiwan J Obstet Gynecol. 2018;57(2):315-8.\u003c/li\u003e\n\u003cli\u003eKwack JY, Kwon YS. Laparoscopic Surgery for Focal Adenomyosis. JSLS. 2017;21(2).\u003c/li\u003e\n\u003cli\u003eClavien PA, Barkun J, de Oliveira ML, Vauthey JN, Dindo D, Schulick RD, et al. The Clavien-Dindo Classification of Surgical Complications: Five-Year Experience. Annals of Surgery 2009;250(2): 187-196\u003c/li\u003e\n\u003cli\u003eChan YY, Jayaprakasan K, Tan A, Thornton JG, Coomarasamy A, Raine-Fenning NJ. Reproductive outcomes in women with congenital uterine anomalies: a systematic review. Ultrasound Obstet Gynecol. 2011;38(4):371-82.\u003c/li\u003e\n\u003cli\u003eHehenkamp WJ, Volkers NA, Broekmans FJ, de Jong FH, Themmen AP, Birnie E, et al. Loss of ovarian reserve after uterine artery embolization: a randomized comparison with hysterectomy. Hum Reprod. 2007;22(7):1996-2005.\u003c/li\u003e\n\u003cli\u003eMara M, Maskova J, Fucikova Z, Kuzel D, Belsan T, Sosna O. Midterm clinical and first reproductive results of a randomized controlled trial comparing uterine fibroid embolization and myomectomy. Cardiovasc Intervent Radiol. 2008;31(1):73-85.\u003c/li\u003e\n\u003cli\u003eKwack JY, Jeon SB, Kim K, Lee SJ, Kwon YS. Monochorionic twin delivery after conservative surgical treatment of a patient with severe diffuse uterine adenomyosis without uterine rupture. Obstet Gynecol Sci. 2016;59(4):311-5.\u003c/li\u003e\n\u003cli\u003eKwack JY, Im KS, Kwon YS. Conservative surgery of uterine adenomyosis via laparoscopic versus laparotomic approach in a single institution. J Obstet Gynaecol Res. 2018;44(7):1268-73.\u003c/li\u003e\n\u003cli\u003eSugiyama M, Takahashi H, Baba Y, Taneichi A, Suzuki H, Usui R, et al. Perinatal outcome of pregnancy after adenomyomectomy: summary of 10 cases with a brief literature review. J Matern Fetal Neonatal Med. 2020;33(24):4145-9.\u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":true,"highlight":"","institution":"","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true},"keywords":"Uterine adenomyosis, Laparoscopy, Uterine Didelphys, Adenomyomectomy","lastPublishedDoi":"10.21203/rs.3.rs-2497639/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-2497639/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003eBackground\u003c/p\u003e\n\u003cp\u003eBecause the surgical approach was in challenging, we adjusted adnomyomectomy in the single uterus of a patient with uterus didelphys and adenomyosis and this is the first reported case that was successfully treated.\u003c/p\u003e\n\u003cp\u003eCase presentation\u003c/p\u003e\n\u003cp\u003eA 31-year-old primipara woman with dysmenorrhea and hypermenorrhea and a known history of uterus didelphys and left kidney agenesis was referred to our hospital. Ultrasonography revealed adenomyosis in the right uterus. She had a plan to have another child and wanted to preserve fertility. Laparoscopic adenomyomectomy was performed in the right uterus with conservation of the original structure of uterine didelphys. Transient occlusion of uterine artery (TOUA) was employed in the right uterine artery, which inhibited the circulation to the right uterus. The incised adenomyosis lesion weighed 20 g, operative time was 55 min, and estimated blood loss was 50 mL. After 5-day hospitalization, the patient was discharged without any notable complications. After 7 months of the surgery, symptoms, and adenomyoma on ultrasonography were improved. To the best of our knowledge, this is the first report of laparoscopic adenomyomectomy surgery performed on a patient with uterine didelphys.\u003c/p\u003e\n\u003cp\u003eConclusion\u003c/p\u003e\n\u003cp\u003eLaparoscopic adenomyomectomy can be considered as a feasible treatment option for patients with uterus didelphys who want to preserve fertility.\u003c/p\u003e","manuscriptTitle":"Laparoscopic adenomyomectomy in the single uterus of the Uterine didelphys","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2023-02-16 20:13:56","doi":"10.21203/rs.3.rs-2497639/v1","editorialEvents":[{"type":"communityComments","content":0}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"b8237a62-d374-4213-bf27-c34da301a30e","owner":[],"postedDate":"February 16th, 2023","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"posted","subjectAreas":[],"tags":[],"updatedAt":"2023-03-06T07:59:35+00:00","versionOfRecord":[],"versionCreatedAt":"2023-02-16 20:13:56","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-2497639","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-2497639","identity":"rs-2497639","version":["v1"]},"buildId":"B-jG_2CBjPDmsCi4Wdhf-","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}