Ureteral endometriosis post-hysterectomy for adenomyosis: a case report and literature review

A 48-year-old female was admitted on March 12, 2023, due to moderate anemia and an ultrasound finding of uterine enlargement. The patient reported dysmenorrhea and menorrhagia but no dyspareunia. Laboratory tests revealed a hemoglobin (Hb) level of 69 g/L and a CA125 level of 53.2 U/ml. Pelvic ultrasound revealed a 7.1 × 4.8 × 7.0 cm mixed echogenic mass with an “onion-skin” appearance, as shown in Fig. 1 . She was diagnosed with adenomyosis and moderate anemia and underwent laparoscopic total hysterectomy and bilateral salpingectomy. Intraoperative exploration of the pelvic cavity revealed no signs of endometriotic lesions. Postoperative pathology revealed adenomyosis combined with leiomyoma. The surgery was successful, and the postoperative recovery was smooth. However, no long-term pharmacological management was provided after the surgery. Fig. 1 Vaginal ultrasound image of the uterus Vaginal ultrasound image of the uterus On April 25, 2024, the patient was admitted to the Urology Department due to right lumbar discomfort. CT imaging revealed a soft tissue density nodule in the distal right ureter near the entrance to the bladder, measuring approximately 24.5 × 16.5 × 18 mm, with unclear margins and associated hydronephrosis and dilation of the ureter proximal to the lesion (Fig. 2 ). On April 28, 2024, the patient underwent a right ureteral biopsy and transurethral right ureteral stent placement. Intraoperatively, the ureteral mucosa appeared smooth, with no visible neoplasms. Postoperative pathology revealed proliferative fibrous connective tissue. Fig. 2 CT image of ureteral endometriosis CT image of ureteral endometriosis A multidisciplinary consultation involving the Gynecology Department revealed the patient’s history of adenomyosis, raising suspicion of ureteral endometriosis. After discussion with the patient and her family, she was admitted to the Gynecology Department on July 12, 2024. MRI revealed an abnormal nodular signal in the pelvic segment of the right ureter near the bladder, suggesting a possible neoplastic lesion. On July 15, 2024, the patient underwent laparoscopic excision for deep ureteral endometriosis and ureterolysis. The surgery was successful, and the postoperative recovery was uneventful. Pathology confirmed ureteral endometriosis with glandular cystic expansion (Fig. 3 ). There were no significant diagnostic challenges encountered in this case. The patient was subsequently treated with gonadotropin-releasing hormone agonist (GnRH-a) therapy (3.6 mg of goserelin via subcutaneous injection every 28 days, six doses). The patient adhered well to the prescribed GnRH-a therapy and reported no discomfort or adverse effects during the treatment course. Fig. 3 a  Pathological examination (HE staining x200) b  Immunohistochemical ER (+) c  Immunohistochemical PR (+) a  Pathological examination (HE staining x200) b  Immunohistochemical ER (+) c  Immunohistochemical PR (+) She also expressed satisfaction with the treatment outcome and reported improvement in symptoms following the intervention. On August 15, 2024, the patient underwent removal of the right ureteral stent via ureteroscopy in the Urology Department. Intraoperatively, no neoplasms, fistulas, or strictures were observed in the ureter. The procedure was successful. Follow-up ultrasound five months later revealed no abnormalities in the kidneys, ureters, or bladder. The patient has remained under follow-up, and no recurrence has been detected to date. To clarify the diagnostic and treatment sequence, a chronological summary of the patient’s clinical course is presented in Table  1 . This timeline outlines the key symptoms, surgical procedures, departmental transitions, and clinical outcomes from March 2023 through August 2024. Additional narrative details are provided below. Table 1 Chronological summary of diagnosis and treatment Date Clinical Presentation Surgery Date Procedure Department Outcome 2023-03-12 Dysmenorrhea, heavy menstrual bleeding 2023-03-24 Laparoscopic total hysterectomy + bilateral salpingectomy + pelvic adhesiolysis Gynecology Recovered and discharged 2024-04-25 Right-sided lumbar pain 2024-04-28 Right ureteral biopsy + transurethral right ureteral stent placement Urology Recovered and discharged 2024-07-12 CT: Soft tissue nodule in lower right ureter near bladder entrance, suspected ureteral endometriosis 2024-07-15 Laparoscopic excision of deep endometriosis + right ureterolysis + bladder repair Gynecology Postoperative GnRH-a therapy initiated (goserelin 3.6 mg every 28 days, total of 6 doses) 2024-08-15 Removal of ureteral stent 2024-08-15 Right ureteral stent removal Urology (Outpatient) Recovered Chronological summary of diagnosis and treatment

This case highlights a potential need to re-evaluate the role of long-term hormonal therapy in patients with adenomyosis who undergo total hysterectomy, particularly in preventing deep infiltrating recurrence such as ureteral endometriosis. While causality cannot be established from a single case, this observation supports further investigation into risk-stratified postoperative management strategies. Additional clinical studies are warranted to clarify patient subgroups who may benefit from continued pharmacological therapy despite definitive surgery. The patient reported satisfaction with the overall treatment process and was pleased with the improvement in her symptoms following surgery and hormonal therapy. She expressed gratitude for the timely diagnosis and multidisciplinary care provided.

UE refers to the implantation of active endometrial tissue around or directly infiltrating the ureter. Its onset is insidious, with 30% of patients presenting no clinical symptoms, whereas others may experience nonspecific symptoms such as dysmenorrhea, dyspareunia, or nonmenstrual pelvic pain [ 9 ]. These characteristics often lead to diagnostic challenges and delays, potentially causing silent kidney damage [ 6 ]. As the disease progresses, approximately 25% of patients develop lumbar pain, and 15% experience gross hematuria [ 10 ]. Histological biopsy and pathological confirmation remain the gold standards for diagnosing UE. Initial evaluation involves a detailed medical history, physical examination, and auxiliary tests. Currently, transvaginal ultrasonography (TVS) combined with urinary system ultrasound is considered a first-line imaging modality for diagnosing DIE because of its noninvasive nature, affordability, and reproducibility [ 11 , 12 ]. In cases of hydronephrosis, further pelvic MRI and CT imaging can pinpoint the site and severity of ureteral obstruction, whereas renal scintigraphy can be used to assess kidney function [ 13 ]. The choice of treatment for UE depends on the patient’s age, fertility intentions, extent of ureteral involvement, and renal function. Surgery is currently considered the gold standard for treating UE. Its main goals are to completely excise endometriotic lesions, relieve ureteral obstruction, and preserve renal function. The secondary goals include obtaining a pathological diagnosis, enabling long-term management to prevent recurrence and ureteral restenosis [ 14 ]. Some researchers believe that ureteral stenosis in UE patients is caused primarily by fibrosis and suggest that, even in patients with hydronephrosis, ureterolysis alone is effective for most patients and should be considered a fundamental treatment method for UE [ 15 ]. In this case, the patient developed lumbar pain more than a year after undergoing total hysterectomy for adenomyosis. Considering the patient’s history of adenomyosis, a multidisciplinary consultation with the Gynecology Department led to a diagnosis of ureteral endometriosis. The patient subsequently underwent laparoscopic excision of the deep ureteral endometriosis and ureterolysis. Postoperative recovery was uneventful. Timely diagnosis and intervention prevent further complications, such as renal failure. For patients with UE, surgery remains the cornerstone of treatment. However, postoperative recurrence continues to pose a major clinical challenge. Delaying or reducing recurrence is a critical objective in the long-term management of endometriosis. Several studies have shown that combining postoperative hormonal therapy (e.g., GnRH agonists) with surgical treatment significantly reduces the recurrence rate of UE compared to surgery alone [ 16 – 18 ]. In a study by Di Maida et al., the recurrence rate in the conservative surgery group without postoperative hormonal therapy reached as high as 28.6%, highlighting the importance of medical intervention, particularly in patients with incompletely excised lesions [ 19 ]. Similarly, a retrospective study by Ceccaroni et al. involving 160 patients emphasized that radical lesion excision is a key factor in preventing UE recurrence [ 20 ]. These findings, along with others in the literature, are summarized in Table  2 . The table provides a comparative overview of reported UE case series, including sample size, surgical methods, use of postoperative hormonal therapy, and recurrence outcomes. It reinforces the importance of individualized treatment strategies and highlights how factors such as surgical completeness, patient age, and autoimmune status contribute to recurrence risk. Table 2 Comparative summary of reported UE cases and recurrence management Study (Author, Year) Sample Size ( n ) Time to Diagnosis Surgical Approach Postoperative Hormonal Therapy Recurrence Rate Follow-up Duration Key Findings/Notes Jia et al., 2022 [ 16 ] 28 Median: 17.8 months Laparoscopic ureterolysis ± ureteral reimplantation 19 patients received GnRH-a (3–6 months)and 5 patients Gestrinone (3–6 months) 0% 5-72months Emphasized individualized treatment approach Liu et al., 2019 [ 17 ] 16 Mean: 8.3 ± 2.7 months Laparoscopic ureterolysis ± excision ± ureteral reimplantation 15 patients received GnRH-a (4–6 months) 0% 3-24months Confirmed safety and efficacy of laparoscopy in UE Hung et al., 2020 [ 18 ] Case series Median: 27.8 months Robot-assisted ureteral reconstruction All patients received GnRH-a (4–6 months) 0% 12-31months Robotic surgery feasible for ureteral reconstruction Di Maida et al., 2022 [ 19 ] 105 Not specified Open surgical Approach (24 cases), laparo-Scopic (30 cases), robot-assisted approach(51cases) 52 patients received no hormonal therapy 28.6% 22–51 months Recurrence linked to absence of hormonal therapy, age, autoimmunity, and incomplete resection Ceccaroni et al., 2019 [ 20 ] 160 Not specified Laparoscopic ureteroneocystostomy 119patients received postoperative hormone therapy with oral progestin or combined estrogen-progestin 1.2% 1–60 months Radical excision essential to reduce recurrence risk Comparative summary of reported UE cases and recurrence management Therefore, regardless of the surgical technique used, efforts should be made to ensure complete excision of ectopic lesions, and postoperative hormonal therapy should be considered based on individual risk profiles. In the present case, the lesion was completely excised intraoperatively, and the patient received six months of postoperative GnRH agonist therapy. No recurrence has been observed during follow-up, aligning with the literature’s recommendations for individualized postoperative management in high-risk patients. This case raises the question of whether patients with adenomyosis should receive long-term pharmacological management after definitive hysterectomy to reduce the recurrence rate of endometriosis. Total hysterectomy is the definitive treatment for AM in women who no longer desire fertility. However, there is currently no clear consensus on whether hormonal therapy is necessary following hysterectomy, especially in the absence of visible endometriotic lesions. In clinical practice, postoperative management varies significantly, and cases like the one presented here raise important questions about whether a more individualized, risk-based approach may be warranted. The necessity of postoperative hormonal therapy following hysterectomy for adenomyosis remains a topic of debate. Several studies have demonstrated that adenomyosis shares estrogen-dependent pathophysiological mechanisms with endometriosis. Thus, even after hysterectomy, residual endometriotic foci may continue to progress under the influence of endogenous estrogen. For patients undergoing uterus-sparing surgery for AM, postoperative management of endometriosis, or excision of deep infiltrating lesions (DIE), long-term hormonal therapy is commonly recommended to reduce recurrence rates and delay disease progression [ 21 , 22 ]. However, opposing viewpoints exist: in patients without visible lesions or in whom lesions have been completely excised, postoperative hormonal therapy may represent overtreatment. In premenopausal women, such therapy may also induce menopausal side effects, including hot flashes and decreased bone density [ 23 ]. Endometriosis is associated with a relatively high postoperative recurrence rate, with an average 5-year recurrence of up to 50% [ 24 ]. Several studies have explored risk factors for recurrence. For example, Seo et al. found that patients aged 40–45 years had significantly lower recurrence rates compared to those under 40, potentially due to lower ovarian activity and estrogen levels, which reduce the stimulation of residual endometriotic lesions [ 25 , 26 ]. In addition, the severity of dysmenorrhea prior to surgery is positively associated with recurrence risk, with more severe pain indicating a higher likelihood of recurrence [ 27 , 28 ]. A high American Society for Reproductive Medicine (ASRM) score, particularly ≥ 40, is also considered a significant risk factor [ 29 , 30 ]. For such patients, meticulous intraoperative exploration and complete excision of lesions, combined with postoperative adjuvant hormonal therapy, may help reduce recurrence and improve outcomes. Incomplete excision of deep infiltrating endometriosis (DIE) lesions is another key contributor to recurrence [ 31 ]. Due to the extensive and dense adhesions commonly seen in DIE, surgical planning should aim to preserve pelvic nerves in order to avoid iatrogenic injury and minimize postoperative pelvic organ dysfunction. Recent studies suggest that nerve-sparing surgical approaches can significantly improve postoperative pelvic organ function, particularly urinary and bowel function [ 32 ]. In light of this literature, we hypothesize that the absence of postoperative hormonal therapy in this case may have contributed to the development of ureteral endometriosis. We propose that a risk-stratified management approach may be beneficial. Patients with factors such as age < 40, intraoperative DIE involvement, suspected residual lesions, or severe symptoms may still require postoperative hormonal therapy, even after total hysterectomy, to prevent recurrence in deep pelvic organs. Conversely, for patients without high-risk features and with complete lesion excision, close monitoring may be a reasonable alternative to routine pharmacological intervention.

Endometriosis (EM) refers to the presence, growth, and infiltration of endometrial tissue (glands and stroma) outside the uterine cavity, causing symptoms such as dysmenorrhea, pelvic pain, and infertility. Its prevalence among women of reproductive age ranges from 5 to 15% [ 1 ]. Adenomyosis (AM), considered a distinct but related condition, involves the invasion of endometrial tissue into the myometrium and has a prevalence of up to 70% [ 2 ]. AM and EM are both estrogen-dependent conditions, but they differ in pathophysiology and clinical presentation. AM is characterized by the infiltration of endometrial glands and stroma into the myometrium, typically leading to uterine enlargement, heavy menstrual bleeding, and dysmenorrhea [ 3 ]. In contrast, EM involves the ectopic growth of endometrial tissue outside the uterus—such as in the ovaries, peritoneum, or deep pelvic structures—and commonly presents with chronic pelvic pain and infertility [ 4 ]. Despite these differences, both are considered chronic inflammatory diseases that may share a common origin. EM can lead to debilitating symptoms such as cyclic pelvic pain, infertility, and bowel or urinary dysfunction, significantly impairing quality of life and psychosocial well-being. Studies have reported that up to 30–50% of women with EM may experience infertility, and many suffer from years of delayed diagnosis due to overlapping symptoms with other gynecologic or gastrointestinal disorders [ 5 ]. Urinary tract endometriosis, including ureteral involvement, is a rare subtype of deep infiltrating endometriosis (DIE), with ureteral cases comprising approximately 10% of urinary tract involvement [ 6 ]. Its occurrence after hysterectomy for AM is particularly rare. Ureteral endometriosis (UE), a rare manifestation, occurs in only 0.1–1.7% of EM cases [ 7 ]. It is often secondary to other forms of endometriosis, with 52–68% of cases associated with ovarian endometriotic cysts (chocolate cysts) and 10–56% involving lesions affecting the uterosacral ligaments, cardinal ligaments, and rectovaginal septum, among other paracervical structures. Isolated or solitary UE is rare [ 8 ]. This report presents a case of UE identified more than one year after total hysterectomy for AM.

Supplementary Material 1. Supplementary Material 1.