Retroperitoneal leiomyoma: case report of a rare case of multiple retroperitoneal leiomyomas

Retroperitoneal leiomyoma: case report of a rare case of multiple retroperitoneal leiomyomas | 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 Research Article Retroperitoneal leiomyoma: case report of a rare case of multiple retroperitoneal leiomyomas Agathe Rabattu, Manon Magdelyns, Alix Danon, Frédéric Buxant This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-4745694/v1 This work is licensed under a CC BY 4.0 License Status: Under Review Version 1 posted 5 You are reading this latest preprint version Abstract Background : Uterine leiomyomas represent the most prevalent benign solid pelvic tumors observed in women. Their primary location is within the uterus; however, they can occasionally occur in uncommon sites, although their presence in the retroperitoneum is extremely rare. Due to their rarity in the retroperitoneum, where most tumors are malignant, establishing a preoperative diagnosis is often challenging. Clinical presentation tends to be nonspecific. Radiological examinations can reveal the presence of a retroperitoneal tumor, but the imaging findings lack specificity. Therefore, the definitive diagnosis typically requires histopathological examination. Case presentation : We report here a case of multiple retroperitoneal leiomyomas diagnosed after a laparoscopic exploration in a 30-year-old poorly symptomatic patient, who had a previous history of an open myomectomy in 2012. We review the difficulties encountered for diagnosis and different treatment options. Conclusion Retroperitoneal leiomyoma should be considered as a differential diagnosis of retroperitoneal masses in women with previous history of leiomyoma removal or hysterectomy. case report retroperitoneal leiomyoma leiomyoma myoma diagnostic imaging pathology uterine myomectomy surgery Figures Figure 1 Figure 2 Figure 3 Figure 4 Figure 5 Figure 6 Background Uterine leiomyomas are a very common pathology affecting 20 to 30% of women over 35 years old. They are the most common gynecologic and uterine neoplasms, being benign tumors composed of smooth muscle tissue[ 4 ]. While these tumors are primarily found in the uterus, they can occasionally present with unusual growth patterns or occur in atypical locations. Examples of leiomyomas with unusual growth patterns include disseminated peritoneal leiomyomatosis, benign metastatic leiomyomas, intravenous leiomyomatosis, retroperitoneal leiomyomas, and parasitic leiomyomas. Unusual sites are typically confined to the genitourinary tract, such as the bladder, urethra, vulva, and ovaries [ 6 ]. Retroperitoneal leiomyomas are extremely rare, with a literature review in 2008 revealing just over 100 cases [ 21 ]. Preoperative diagnosis of this neoplasm can be challenging due to its rarity and nonspecific clinical and imaging presentations. In this paper, we present a case of multiple retroperitoneal leiomyomas and their treatment. Additionally, we conducted a review of the scientific literature on this topic. Case Presentation A 30-year-old woman visited our service for the follow-up of a leiomyoma. She reported no specific symptoms other than a feeling of pelvic heaviness. She had a history of an open myomectomy in 2012 and a cesarean section in 2013. During a vaginal examination, a right adnexal mass was detected. A transvaginal ultrasound revealed a small uterus measuring 70x30x26 mm, a 61x27 mm homogenous (hypoechoic and well-circumscribed) solid mass with low vascularization in the left adnexa, and two right adnexal masses measuring 35x26 mm and 35x25 mm (Fig. 1 ). The ultrasound conditions were suboptimal, and the ovaries were not visualized. Given that the ovaries were not identified, it is probable that the tumors originated from the ovaries. The differential diagnosis included pedunculated subserosal leiomyomas, classified as type 7 according to FIGO classification[ 19 ]. For ovarian masses, the IOTA score indicated a 70.9% chance of being benign. MRI revealed bilateral adnexal masses, homogenous and well-delimited with homogenous enhancement, displaying a type 2 enhancement curve, categorized as ORADS4. The right mass was lobulated and measured 83x33x64 mm, while the left mass measured 51x23x63 mm. Radiologists could not exclude the possibility of Brenner’s tumor or ovarian metastases. Based on these findings, a laparoscopic exploration was proposed to the patient. The Ca-125 marker was normal. During the procedure, the uterus, ovaries, and fallopian tubes appeared normal, but two 4–5 cm retroperitoneal masses were found in the ovarian fossa. Additionally, two other masses approximately 8 cm each were visualized along the vena cava and the aorta (Fig. 2 ). These masses were renitent, liquid, supple, and without induration. Since the nature of these masses could not be determined through MRI or ultrasound, it was unclear whether they were lymphatic, vascular, or lymph nodes. As the patient was not ready for potential vascular or digestive surgery, a more comprehensive radiological assessment was planned, with further discussion on the diagnosis to follow. An abdominal CT and MRI were performed, confirming the two retroperitoneal masses and similar lesions in the upper retroperitoneum and along the aortocaval chain, on both sides of the emergence of the renal vessels (Figs. 3 and 4 ). A PET scan showed the known masses to be slightly hypermetabolic with no other masses detected. To ascertain the lymphatic nature of the masses, a pelvic lymphoscintigraphy was conducted, revealing bilateral iliac lymph nodes and right lombo-aortic lymph nodes. The patient later mentioned having had leukemia at age four, treated with chemotherapy. An internal medicine assessment showed no signs of leukemia relapse. Another laparoscopic intervention aimed at removing one of the retroperitoneal masses to obtain a definitive pathological diagnosis was performed. The pathological report identified the mass as a 4 cm benign leiomyoma, with estrogen receptor positivity suggesting a gynecological origin. Since the patient was asymptomatic and opted against surgery, we recommended clinical follow-ups every six months and an annual MRI. If the masses grew, surgical removal would be considered. Nine months later, the patient returned with abdominal pain. A new MRI showed an increase in the size of the retroperitoneal fibroma but no new masses (Fig. 5 ). A laparotomy was performed, and all retroperitoneal masses were removed (Fig. 6 ). The patient had no surgical complications. Discussion Leiomyomas are common tumors that can originate from any location where smooth muscle exists[ 4 ]. While the most common site is the uterus, they have also been described in other locations[ 2 , 21 ]. The retroperitoneum is a rare site for leiomyomas [ 1 , 5 , 12 , 26 ], typically occurring in the pelvis but occasionally extending into the upper retroperitoneum [ 6 ]. In a 2008 review by Poliquin et al. [ 21 ], 105 cases of retroperitoneal leiomyomas were identified. In 2020 Sabrine et al [ 23 ], reported approximately 100 cases in English literature to date. Since then, no other systematic review of the literature has been conducted. A recent PubMed search using the terms “intraperitoneal” and “leiomyoma” revealed additional cases [ 3 , 7 , 9 , 10 , 14 , 15 , 17 , 18 , 20 , 24 , 25 , 27 – 30 ]. More than 40% of women with retroperitoneal leiomyomas have a history of hysterectomy for uterine leiomyoma treatment or have concurrent uterine leiomyomas [ 21 ]. The etiology and pathogenesis of retroperitoneal leiomyomas are not well understood. It is unclear whether these lesions are metastatic or synchronous primary lesions and whether they arise from hormonally sensitive smooth muscle cells or embryonal remnants of the Müllerian or Wolffian ducts. Estrogen is believed to play a significant role in the development and maintenance of uterine leiomyomas, as evidenced by their frequent occurrence during reproductive age, reduction in size after menopause, and growth during pregnancy [ 1 , 5 ]. Stutterecker et al. [ 26 ] speculated that embryonal remnants or local vessel musculature could be the tumor’s origin. Zaitoon et al. [ 31 ] proposed the theory of parasitic leiomyomas, where uterine leiomyomas that adhere to surrounding structures acquire an auxiliary blood supply and detach from the uterus. A newer type of parasitic myoma, called iatrogenic parasitic myoma, has been associated with the use of electric tissue morcellators in laparoscopy, which can produce small myoma fragments that implant on peritoneal and omental tissue and grow [ 8 ]. Several hypotheses have been proposed to explain the metastasizing theory [ 12 ]. One suggests benign tumors spread hematogenously, another proposes surgically induced hematogenous spread with tumor gaining venous access from surgical trauma (such as hysterectomy or myomectomy), and the last suggests a low-grade metastatic leiomyosarcoma. Retroperitoneal leiomyoma can also be isolated, without concurrent uterine leiomyoma or history of leiomyoma treatment, suggesting they could also represent primary extrauterine leiomyomatosis [ 11 ]. Retroperitoneal leiomyomas can be asymptomatic even if they are large and may be detected incidentally during routine check-ups or autopsies [ 5 , 6 ]. Common symptoms include abdominal discomfort, fatigue, urinary symptoms, weight loss, back pain and pelvic pain. Almost 90% of patients with retroperitoneal leiomyomas present with a palpable pelvic mass [ 21 ]. Despite modern imaging techniques, retroperitoneal leiomyomas are often misdiagnosed as malignant tumors due to their unusual location. MRI is the most reliable technique for evaluating retroperitoneal masses, while ultrasound and CT scans can also be helpful. The differential diagnosis includes common benign and malignant neoplasms of neurogenic origin (such as paraganglioma, schwannoma, ganglioneuroma, extra-renal pheochromocytoma), as well as teratoma, desmoid tumor, hemangioma, extra-adrenal angiomyolipoma, sarcoma, lymphoma, and metastatic tumors. Most reported cases of retroperitoneal leiomyomas were not correctly diagnosed preoperatively, with the definitive diagnosis requiring pathological examination. US-guided percutaneous biopsy of the mass is useful for determining its histologic composition preoperatively, though sometimes a complete resection of the mass is necessary for a full histologic examination [ 16 ]. In current scientific literature, the only treatment for smooth muscle tumors of the retroperitoneum, whether benign or malignant, is surgical resection [ 11 ]. Most of the previous reported cases of retroperitoneal leiomyomata were treated by laparotomy but the laparoscopic approach is also feasible and provides many advantages for the patient, even for obese women. Surgical excision is invasive and requires familiarity and experience with this area. The goals of resection are to rule out any malignancy and offer pain or symptom relief if present [ 1 ]. The prognosis is good. Indeed, Poliquin et al. [ 21 ] found that surgery was mostly curative with only five reported cases of recurrence out of the 105 cases. Although surgery is currently the only recommended treatment, two cases have reported the use of medical treatment in conjunction with surgery. Kortekaas et al. [ 13 ] used ulipristal acetate after a relapse one-year post-surgery. The hypothesis is that ulipristal acetate and more recently relugolix, blocks the progesterone receptors in lesions, thereby restricting their growth. It can be used to reduce the size of fibroids before surgery or as a long-term treatment for patients who are not candidates for surgery. This should be balanced with the potential significant side effects, mostly hepatic. The patient showed multiple nodules in the lungs, omentum, mesenterium and retroperitoneum. With ulipristal acetate treatment (16 month total), the nodules decreased and the patient remained asymptomatic 8 month after the treatment ended. Rodriguez et al. [ 22 ] started treatment with aromatase inhibitors following incomplete surgical resection, leading to the patient remaining healthy and the residual tumor stable 23 months later. Conclusions Retroperitoneal leiomyomas are rare. Despite their histologically benign nature, they can mimic malignant tumors on imaging, posing a diagnostic challenge. In this paper, we presented an uncommon case of multiple retroperitoneal leiomyomas. Imaging alone was insufficient for an accurate diagnosis, necessitating tumor resection for histological examination. This case underscores the importance of considering retroperitoneal leiomyomas as a differential diagnosis for retroperitoneal masses, especially in women with a history of leiomyoma removal or hysterectomy. Abbreviations FIGO International Federation of Gynecology and Obstetrics MRI Magnetic Resonance Imaging CT Computed Tomography PET Positron Emission Tomography ORADS Ovarian-Adnexal Imaging-Reporting-Data System CA Cancer Antigen US Ultra Sound Declarations Ethics approval and consent to participate The ethics committee of the Hopitaux Iris Sud (HIS) approved the research. Consent for publication Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for the review by the Editor-in-Chief of this journal. Availability of data and materials The datasets used and/or analyzed during the current study are available from the corresponding author on reasonable request. Competing interests The authors declare that they have no competing interest. Funding N/A Authors Contribution AR and MM analyzed the data, conducted a scientific literature search, and drafted the article. AD and FB participated in the writing of the article and provided their expert opinion in gynecology. All authors have read and approved the manuscript. Acknowledgments We thank our colleagues of gynecology and radiology team for their help and support. References Arab M, Farzaneh F, Ghavami B, Mohajeri S, Marzban Rad Z, Ashori A (2016) A Rare Case of a Retroperitoneal Leiomyoma in the Obturator Fossa. Iran Red Crescent Med J 18. 10.5812/ircmj.28475 Barnaś E, Raś R, Skręt-Magierło J, Wesecki M, Filipowska J, Książek M, Skręt A, Widenka K (2019) Natural history of leiomyomas beyond the uterus. 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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-4745694","acceptedTermsAndConditions":true,"allowDirectSubmit":false,"archivedVersions":[],"articleType":"Research Article","associatedPublications":[],"authors":[{"id":344593305,"identity":"17ba5228-5225-4f06-815f-2850a4939da7","order_by":0,"name":"Agathe Rabattu","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAA8klEQVRIiWNgGAWjYJACZjApASZtGPgkGNgI6WBshmphbDjAkMbARqqWw4S18Pcvfv64oIIhn3928/PHH9vOJ7ZJN7A9+IBHi8SNZ4bNM84wWM64c8yw4WDb7cQ2mQPshjPwWXPjgGEzbxuDAcONBKgWiQQ2aR48OuRvHP/YzPuPwUD+RvpHoJZzEC1/8GgxON8DtKWBwcDgRg7IlgMQLfjcZXiDp3A2zzEJA8M7ZwpnnDmXbNwmc7DdsAePFrnzxzd85qmxMZC73b7hQ0WZnWy/dPOxBz/wWSORACaRhRgb8GkARswB/PKjYBSMglEwChgA6+ZU2x3u57gAAAAASUVORK5CYII=","orcid":"https://orcid.org/0009-0005-6774-8392","institution":"Université Libre de Bruxelles: Universite Libre de Bruxelles","correspondingAuthor":true,"prefix":"","firstName":"Agathe","middleName":"","lastName":"Rabattu","suffix":""},{"id":344593306,"identity":"95feca25-5a90-41c7-b104-74fe1c93aef0","order_by":1,"name":"Manon Magdelyns","email":"","orcid":"","institution":"Université Libre de Bruxelles: Universite Libre de Bruxelles","correspondingAuthor":false,"prefix":"","firstName":"Manon","middleName":"","lastName":"Magdelyns","suffix":""},{"id":344593307,"identity":"52190ac1-2a95-4a2e-8bee-590a24fbab1e","order_by":2,"name":"Alix Danon","email":"","orcid":"","institution":"Université Libre de Bruxelles: Universite Libre de Bruxelles","correspondingAuthor":false,"prefix":"","firstName":"Alix","middleName":"","lastName":"Danon","suffix":""},{"id":344593308,"identity":"95b5881f-c0cc-4013-b4ee-003938d67a13","order_by":3,"name":"Frédéric Buxant","email":"","orcid":"","institution":"Université Libre de Bruxelles: Universite Libre de Bruxelles","correspondingAuthor":false,"prefix":"","firstName":"Frédéric","middleName":"","lastName":"Buxant","suffix":""}],"badges":[],"createdAt":"2024-07-15 22:03:41","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-4745694/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-4745694/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":66766249,"identity":"a72bf0a1-60c5-42f2-b55f-093aab44c2ce","added_by":"auto","created_at":"2024-10-16 09:25:06","extension":"jpg","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":76715,"visible":true,"origin":"","legend":"\u003cp\u003eEndovaginal ultrasound: A. 61x27mm left adnexal mass. B. 35x26mm right adnexal mass. C. 35x25mm right adnexal mass\u003c/p\u003e","description":"","filename":"figure1.jpg","url":"https://assets-eu.researchsquare.com/files/rs-4745694/v1/5eaee0e6120a2ba898dd956c.jpg"},{"id":66766250,"identity":"c7cfb11e-51e8-4a39-bdbe-2003e5b5e852","added_by":"auto","created_at":"2024-10-16 09:25:06","extension":"jpg","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":55986,"visible":true,"origin":"","legend":"\u003cp\u003eLaparoscopic images: A. Right retroperitoneal mass. B. Left retroperitoneal mass. C. Periaortic masses.\u003c/p\u003e","description":"","filename":"figure2.jpg","url":"https://assets-eu.researchsquare.com/files/rs-4745694/v1/0f3d9d2a79ca4cdde48b6e76.jpg"},{"id":66766254,"identity":"ba50d997-d2de-481b-b449-65ea71e4c611","added_by":"auto","created_at":"2024-10-16 09:25:06","extension":"jpg","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":80418,"visible":true,"origin":"","legend":"\u003cp\u003eComputed Tomography: A. coronal plane after contraste portal phase acquisition; we can distinguish the solid tumors with homogenous enhancement at the pelvic area as well at the retroperitoneal space. B. axial plane after contrast portal phase acquisition; solid masses with homogenous enhancement at the pelvic area - both iliac fossae\u003c/p\u003e","description":"","filename":"Figure3.jpg","url":"https://assets-eu.researchsquare.com/files/rs-4745694/v1/d638c87c4905a944869e62d0.jpg"},{"id":66766255,"identity":"e41c4589-ca5a-4a1d-acf6-1b18653dc1bf","added_by":"auto","created_at":"2024-10-16 09:25:07","extension":"jpg","order_by":4,"title":"Figure 4","display":"","copyAsset":false,"role":"figure","size":99148,"visible":true,"origin":"","legend":"\u003cp\u003eMagnetic resonance Imaging: A. Axial plane T1 fat suppressed after gadolinium contrast shows the bilateral homogenous tumors on both iliac fossae. B. Axial T2 weighted image indicates the homogenous well circumscribed lesions.\u003c/p\u003e","description":"","filename":"Figure4.jpg","url":"https://assets-eu.researchsquare.com/files/rs-4745694/v1/8488c40ded884881d592e11c.jpg"},{"id":66766251,"identity":"5206eb4a-92a9-4c65-b47e-5ea7170d928e","added_by":"auto","created_at":"2024-10-16 09:25:06","extension":"jpg","order_by":5,"title":"Figure 5","display":"","copyAsset":false,"role":"figure","size":116024,"visible":true,"origin":"","legend":"\u003cp\u003eMRI follow up image- T2 weighted image on coronal plane. Multiple coalescent homogeneous masses on the retroperitoneal space and at both iliac fossae.\u003c/p\u003e","description":"","filename":"Figure5.jpg","url":"https://assets-eu.researchsquare.com/files/rs-4745694/v1/c1794c009c78dd18effd54f4.jpg"},{"id":66766252,"identity":"ac15cc4d-e091-4369-a03c-0bc1967456a8","added_by":"auto","created_at":"2024-10-16 09:25:06","extension":"jpg","order_by":6,"title":"Figure 6","display":"","copyAsset":false,"role":"figure","size":653557,"visible":true,"origin":"","legend":"\u003cp\u003eLaparotomy images: A: Para-aortic mass. B. Ilio-obturator right fossa mass.C. Masses removed\u003c/p\u003e","description":"","filename":"figure6.jpg","url":"https://assets-eu.researchsquare.com/files/rs-4745694/v1/a1f817c8493cd548da093650.jpg"},{"id":66766919,"identity":"254048d0-bbbc-41d0-a0aa-82efd657737a","added_by":"auto","created_at":"2024-10-16 09:33:06","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":1348558,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-4745694/v1/eac743c9-4fd1-484e-b004-9f5568502943.pdf"},{"id":66766253,"identity":"4f24ae25-76d3-4c99-a4c9-15089aceaa33","added_by":"auto","created_at":"2024-10-16 09:25:06","extension":"pdf","order_by":10,"title":"","display":"","copyAsset":false,"role":"supplement","size":1718861,"visible":true,"origin":"","legend":"","description":"","filename":"CAREchecklist.pdf","url":"https://assets-eu.researchsquare.com/files/rs-4745694/v1/7e5b29d00d86348d751958bf.pdf"}],"financialInterests":"","formattedTitle":"Retroperitoneal leiomyoma: case report of a rare case of multiple retroperitoneal leiomyomas","fulltext":[{"header":"Background","content":"\u003cp\u003eUterine leiomyomas are a very common pathology affecting 20 to 30% of women over 35 years old. They are the most common gynecologic and uterine neoplasms, being benign tumors composed of smooth muscle tissue[\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eWhile these tumors are primarily found in the uterus, they can occasionally present with unusual growth patterns or occur in atypical locations. Examples of leiomyomas with unusual growth patterns include disseminated peritoneal leiomyomatosis, benign metastatic leiomyomas, intravenous leiomyomatosis, retroperitoneal leiomyomas, and parasitic leiomyomas. Unusual sites are typically confined to the genitourinary tract, such as the bladder, urethra, vulva, and ovaries [\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e]. Retroperitoneal leiomyomas are extremely rare, with a literature review in 2008 revealing just over 100 cases [\u003cspan citationid=\"CR21\" class=\"CitationRef\"\u003e21\u003c/span\u003e].\u003c/p\u003e \u003cp\u003ePreoperative diagnosis of this neoplasm can be challenging due to its rarity and nonspecific clinical and imaging presentations. In this paper, we present a case of multiple retroperitoneal leiomyomas and their treatment. Additionally, we conducted a review of the scientific literature on this topic.\u003c/p\u003e"},{"header":"Case Presentation","content":"\u003cp\u003eA 30-year-old woman visited our service for the follow-up of a leiomyoma. She reported no specific symptoms other than a feeling of pelvic heaviness. She had a history of an open myomectomy in 2012 and a cesarean section in 2013. During a vaginal examination, a right adnexal mass was detected. A transvaginal ultrasound revealed a small uterus measuring 70x30x26 mm, a 61x27 mm homogenous (hypoechoic and well-circumscribed) solid mass with low vascularization in the left adnexa, and two right adnexal masses measuring 35x26 mm and 35x25 mm (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003e). The ultrasound conditions were suboptimal, and the ovaries were not visualized. Given that the ovaries were not identified, it is probable that the tumors originated from the ovaries. The differential diagnosis included pedunculated subserosal leiomyomas, classified as type 7 according to FIGO classification[\u003cspan citationid=\"CR19\" class=\"CitationRef\"\u003e19\u003c/span\u003e]. For ovarian masses, the IOTA score indicated a 70.9% chance of being benign.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003eMRI revealed bilateral adnexal masses, homogenous and well-delimited with homogenous enhancement, displaying a type 2 enhancement curve, categorized as ORADS4. The right mass was lobulated and measured 83x33x64 mm, while the left mass measured 51x23x63 mm. Radiologists could not exclude the possibility of Brenner\u0026rsquo;s tumor or ovarian metastases. Based on these findings, a laparoscopic exploration was proposed to the patient. The Ca-125 marker was normal.\u003c/p\u003e \u003cp\u003eDuring the procedure, the uterus, ovaries, and fallopian tubes appeared normal, but two 4\u0026ndash;5 cm retroperitoneal masses were found in the ovarian fossa. Additionally, two other masses approximately 8 cm each were visualized along the vena cava and the aorta (Fig.\u0026nbsp;\u003cspan refid=\"Fig2\" class=\"InternalRef\"\u003e2\u003c/span\u003e). These masses were renitent, liquid, supple, and without induration. Since the nature of these masses could not be determined through MRI or ultrasound, it was unclear whether they were lymphatic, vascular, or lymph nodes. As the patient was not ready for potential vascular or digestive surgery, a more comprehensive radiological assessment was planned, with further discussion on the diagnosis to follow.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003eAn abdominal CT and MRI were performed, confirming the two retroperitoneal masses and similar lesions in the upper retroperitoneum and along the aortocaval chain, on both sides of the emergence of the renal vessels (Figs.\u0026nbsp;\u003cspan refid=\"Fig3\" class=\"InternalRef\"\u003e3\u003c/span\u003e and \u003cspan refid=\"Fig4\" class=\"InternalRef\"\u003e4\u003c/span\u003e). A PET scan showed the known masses to be slightly hypermetabolic with no other masses detected. To ascertain the lymphatic nature of the masses, a pelvic lymphoscintigraphy was conducted, revealing bilateral iliac lymph nodes and right lombo-aortic lymph nodes.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003eThe patient later mentioned having had leukemia at age four, treated with chemotherapy. An internal medicine assessment showed no signs of leukemia relapse.\u003c/p\u003e \u003cp\u003eAnother laparoscopic intervention aimed at removing one of the retroperitoneal masses to obtain a definitive pathological diagnosis was performed. The pathological report identified the mass as a 4 cm benign leiomyoma, with estrogen receptor positivity suggesting a gynecological origin.\u003c/p\u003e \u003cp\u003eSince the patient was asymptomatic and opted against surgery, we recommended clinical follow-ups every six months and an annual MRI. If the masses grew, surgical removal would be considered. Nine months later, the patient returned with abdominal pain. A new MRI showed an increase in the size of the retroperitoneal fibroma but no new masses (Fig.\u0026nbsp;\u003cspan refid=\"Fig5\" class=\"InternalRef\"\u003e5\u003c/span\u003e). A laparotomy was performed, and all retroperitoneal masses were removed (Fig.\u0026nbsp;\u003cspan refid=\"Fig6\" class=\"InternalRef\"\u003e6\u003c/span\u003e). The patient had no surgical complications.\u003c/p\u003e "},{"header":"Discussion","content":"\u003cp\u003eLeiomyomas are common tumors that can originate from any location where smooth muscle exists[\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e]. While the most common site is the uterus, they have also been described in other locations[\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e, \u003cspan citationid=\"CR21\" class=\"CitationRef\"\u003e21\u003c/span\u003e]. The retroperitoneum is a rare site for leiomyomas [\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e, \u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e, \u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e, \u003cspan citationid=\"CR26\" class=\"CitationRef\"\u003e26\u003c/span\u003e], typically occurring in the pelvis but occasionally extending into the upper retroperitoneum [\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eIn a 2008 review by Poliquin et al. [\u003cspan citationid=\"CR21\" class=\"CitationRef\"\u003e21\u003c/span\u003e], 105 cases of retroperitoneal leiomyomas were identified. In 2020 Sabrine et al [\u003cspan citationid=\"CR23\" class=\"CitationRef\"\u003e23\u003c/span\u003e], reported approximately 100 cases in English literature to date. Since then, no other systematic review of the literature has been conducted. A recent PubMed search using the terms \u0026ldquo;intraperitoneal\u0026rdquo; and \u0026ldquo;leiomyoma\u0026rdquo; revealed additional cases [\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e, \u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e, \u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e, \u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e, \u003cspan citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e, \u003cspan citationid=\"CR15\" class=\"CitationRef\"\u003e15\u003c/span\u003e, \u003cspan citationid=\"CR17\" class=\"CitationRef\"\u003e17\u003c/span\u003e, \u003cspan citationid=\"CR18\" class=\"CitationRef\"\u003e18\u003c/span\u003e, \u003cspan citationid=\"CR20\" class=\"CitationRef\"\u003e20\u003c/span\u003e, \u003cspan citationid=\"CR24\" class=\"CitationRef\"\u003e24\u003c/span\u003e, \u003cspan citationid=\"CR25\" class=\"CitationRef\"\u003e25\u003c/span\u003e, \u003cspan additionalcitationids=\"CR28 CR29\" citationid=\"CR27\" class=\"CitationRef\"\u003e27\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR30\" class=\"CitationRef\"\u003e30\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eMore than 40% of women with retroperitoneal leiomyomas have a history of hysterectomy for uterine leiomyoma treatment or have concurrent uterine leiomyomas [\u003cspan citationid=\"CR21\" class=\"CitationRef\"\u003e21\u003c/span\u003e]. The etiology and pathogenesis of retroperitoneal leiomyomas are not well understood. It is unclear whether these lesions are metastatic or synchronous primary lesions and whether they arise from hormonally sensitive smooth muscle cells or embryonal remnants of the M\u0026uuml;llerian or Wolffian ducts. Estrogen is believed to play a significant role in the development and maintenance of uterine leiomyomas, as evidenced by their frequent occurrence during reproductive age, reduction in size after menopause, and growth during pregnancy [\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e, \u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e]. Stutterecker et al. [\u003cspan citationid=\"CR26\" class=\"CitationRef\"\u003e26\u003c/span\u003e] speculated that embryonal remnants or local vessel musculature could be the tumor\u0026rsquo;s origin. Zaitoon et al. [\u003cspan citationid=\"CR31\" class=\"CitationRef\"\u003e31\u003c/span\u003e] proposed the theory of parasitic leiomyomas, where uterine leiomyomas that adhere to surrounding structures acquire an auxiliary blood supply and detach from the uterus. A newer type of parasitic myoma, called iatrogenic parasitic myoma, has been associated with the use of electric tissue morcellators in laparoscopy, which can produce small myoma fragments that implant on peritoneal and omental tissue and grow [\u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e]. Several hypotheses have been proposed to explain the metastasizing theory [\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e]. One suggests benign tumors spread hematogenously, another proposes surgically induced hematogenous spread with tumor gaining venous access from surgical trauma (such as hysterectomy or myomectomy), and the last suggests a low-grade metastatic leiomyosarcoma. Retroperitoneal leiomyoma can also be isolated, without concurrent uterine leiomyoma or history of leiomyoma treatment, suggesting they could also represent primary extrauterine leiomyomatosis [\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eRetroperitoneal leiomyomas can be asymptomatic even if they are large and may be detected incidentally during routine check-ups or autopsies [\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e, \u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e]. Common symptoms include abdominal discomfort, fatigue, urinary symptoms, weight loss, back pain and pelvic pain. Almost 90% of patients with retroperitoneal leiomyomas present with a palpable pelvic mass [\u003cspan citationid=\"CR21\" class=\"CitationRef\"\u003e21\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eDespite modern imaging techniques, retroperitoneal leiomyomas are often misdiagnosed as malignant tumors due to their unusual location. MRI is the most reliable technique for evaluating retroperitoneal masses, while ultrasound and CT scans can also be helpful. The differential diagnosis includes common benign and malignant neoplasms of neurogenic origin (such as paraganglioma, schwannoma, ganglioneuroma, extra-renal pheochromocytoma), as well as teratoma, desmoid tumor, hemangioma, extra-adrenal angiomyolipoma, sarcoma, lymphoma, and metastatic tumors. Most reported cases of retroperitoneal leiomyomas were not correctly diagnosed preoperatively, with the definitive diagnosis requiring pathological examination. US-guided percutaneous biopsy of the mass is useful for determining its histologic composition preoperatively, though sometimes a complete resection of the mass is necessary for a full histologic examination [\u003cspan citationid=\"CR16\" class=\"CitationRef\"\u003e16\u003c/span\u003e].\u003c/p\u003e \u003cp\u003eIn current scientific literature, the only treatment for smooth muscle tumors of the retroperitoneum, whether benign or malignant, is surgical resection [\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e]. Most of the previous reported cases of retroperitoneal leiomyomata were treated by laparotomy but the laparoscopic approach is also feasible and provides many advantages for the patient, even for obese women. Surgical excision is invasive and requires familiarity and experience with this area. The goals of resection are to rule out any malignancy and offer pain or symptom relief if present [\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e]. The prognosis is good. Indeed, Poliquin et al. [\u003cspan citationid=\"CR21\" class=\"CitationRef\"\u003e21\u003c/span\u003e] found that surgery was mostly curative with only five reported cases of recurrence out of the 105 cases. Although surgery is currently the only recommended treatment, two cases have reported the use of medical treatment in conjunction with surgery. Kortekaas et al. [\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e] used ulipristal acetate after a relapse one-year post-surgery. The hypothesis is that ulipristal acetate and more recently relugolix, blocks the progesterone receptors in lesions, thereby restricting their growth. It can be used to reduce the size of fibroids before surgery or as a long-term treatment for patients who are not candidates for surgery. This should be balanced with the potential significant side effects, mostly hepatic. The patient showed multiple nodules in the lungs, omentum, mesenterium and retroperitoneum. With ulipristal acetate treatment (16 month total), the nodules decreased and the patient remained asymptomatic 8 month after the treatment ended. Rodriguez et al. [\u003cspan citationid=\"CR22\" class=\"CitationRef\"\u003e22\u003c/span\u003e] started treatment with aromatase inhibitors following incomplete surgical resection, leading to the patient remaining healthy and the residual tumor stable 23 months later.\u003c/p\u003e"},{"header":"Conclusions","content":"\u003cp\u003eRetroperitoneal leiomyomas are rare. Despite their histologically benign nature, they can mimic malignant tumors on imaging, posing a diagnostic challenge. In this paper, we presented an uncommon case of multiple retroperitoneal leiomyomas. Imaging alone was insufficient for an accurate diagnosis, necessitating tumor resection for histological examination. This case underscores the importance of considering retroperitoneal leiomyomas as a differential diagnosis for retroperitoneal masses, especially in women with a history of leiomyoma removal or hysterectomy.\u003c/p\u003e"},{"header":"Abbreviations","content":"\u003cdiv class=\"DefinitionList\"\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eFIGO\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eInternational Federation of Gynecology and Obstetrics\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eMRI\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eMagnetic Resonance Imaging\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eCT\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eComputed Tomography\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003ePET\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003ePositron Emission Tomography\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eORADS\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eOvarian-Adnexal Imaging-Reporting-Data System\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eCA\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eCancer Antigen\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eUS\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eUltra Sound\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003c/div\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003eEthics approval and consent to participate\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe ethics committee of the Hopitaux Iris Sud (HIS) approved the research.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConsent for publication\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eWritten informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for the review by the Editor-in-Chief of this journal.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAvailability of data and materials\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe datasets used and/or analyzed during the current study are available from the corresponding author on reasonable request.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCompeting interests\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThe authors declare that they have no competing interest.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eFunding\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eN/A\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAuthors Contribution\u0026nbsp;\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eAR and MM analyzed the data, conducted a scientific literature search, and drafted the article. AD and FB participated in the writing of the article and provided their expert opinion in gynecology. All authors have read and approved the manuscript.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAcknowledgments \u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eWe thank our colleagues of gynecology and radiology team for their help and support.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\u003cli\u003e\u003cspan\u003eArab M, Farzaneh F, Ghavami B, Mohajeri S, Marzban Rad Z, Ashori A (2016) A Rare Case of a Retroperitoneal Leiomyoma in the Obturator Fossa. Iran Red Crescent Med J 18. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003e10.5812/ircmj.28475\u003c/span\u003e\u003cspan address=\"10.5812/ircmj.28475\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBarnaś E, Raś R, Skręt-Magierło J, Wesecki M, Filipowska J, Książek M, Skręt A, Widenka K (2019) Natural history of leiomyomas beyond the uterus. 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J Urol 135:130\u0026ndash;131. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003e10.1016/s0022-5347(17)45543-1\u003c/span\u003e\u003cspan address=\"10.1016/s0022-5347(17)45543-1\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"highlight":"","institution":"","isAcceptedByJournal":true,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"bulletin-of-the-national-research-centre","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"bnrc","sideBox":"Learn more about [Bulletin of the National Research Centre](https://BNRC.springeropen.com)","snPcode":"42269","submissionUrl":"https://submission.springernature.com/new-submission/42269/3","title":"Bulletin of the National Research Centre","twitterHandle":"","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"stoa","reportingPortfolio":"Springer Open","inReviewEnabled":true,"inReviewRevisionsEnabled":true},"keywords":"case report, retroperitoneal leiomyoma, leiomyoma, myoma, diagnostic imaging, pathology, uterine myomectomy, surgery","lastPublishedDoi":"10.21203/rs.3.rs-4745694/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-4745694/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003e\u003cstrong\u003eBackground\u003c/strong\u003e: Uterine leiomyomas represent the most prevalent benign solid pelvic tumors observed in women. Their primary location is within the uterus; however, they can occasionally occur in uncommon sites, although their presence in the retroperitoneum is extremely rare. Due to their rarity in the retroperitoneum, where most tumors are malignant, establishing a preoperative diagnosis is often challenging. Clinical presentation tends to be nonspecific. Radiological examinations can reveal the presence of a retroperitoneal tumor, but the imaging findings lack specificity. Therefore, the definitive diagnosis typically requires histopathological examination.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCase presentation : \u003c/strong\u003eWe report here a case of multiple retroperitoneal leiomyomas diagnosed after a laparoscopic exploration in a 30-year-old poorly symptomatic patient, who had a previous history of an open myomectomy in 2012. We review the difficulties encountered for diagnosis and different treatment options.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConclusion\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eRetroperitoneal leiomyoma should be considered as a differential diagnosis of retroperitoneal masses in women with previous history of leiomyoma removal or hysterectomy.\u003c/p\u003e","manuscriptTitle":"Retroperitoneal leiomyoma: case report of a rare case of multiple retroperitoneal leiomyomas","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2024-10-16 09:25:01","doi":"10.21203/rs.3.rs-4745694/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Reject after peer review","date":"2024-12-29T03:26:38+00:00","index":"","fulltext":""},{"type":"reviewerAgreed","content":"","date":"2024-12-23T10:31:40+00:00","index":0,"fulltext":""},{"type":"reviewersInvited","content":"","date":"2024-08-24T15:10:23+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2024-08-21T07:10:48+00:00","index":"","fulltext":""},{"type":"submitted","content":"Bulletin of the National Research Centre","date":"2024-08-18T08:44:30+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"bulletin-of-the-national-research-centre","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"bnrc","sideBox":"Learn more about [Bulletin of the National Research Centre](https://BNRC.springeropen.com)","snPcode":"42269","submissionUrl":"https://submission.springernature.com/new-submission/42269/3","title":"Bulletin of the National Research Centre","twitterHandle":"","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"stoa","reportingPortfolio":"Springer Open","inReviewEnabled":true,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"a537ba45-7914-48bf-94c1-b315cce38676","owner":[],"postedDate":"October 16th, 2024","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"under-review","subjectAreas":[],"tags":[],"updatedAt":"2024-12-22T17:30:31+00:00","versionOfRecord":[],"versionCreatedAt":"2024-10-16 09:25:01","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-4745694","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-4745694","identity":"rs-4745694","version":["v1"]},"buildId":"_2-kVJe1T_tPrBINL-cwx","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}