Resolution of unilateral obstructive uropathy from a bladder adherent large exophytic cervical leiomyoma following selective inferior vesical artery embolization.

This patient is a 51-year-old female with a history of solid and pseudopapillary tumor of the pancreas status post central pancreatectomy with Roux-en-Y pancreatojejunostomy and symptomatic large uterine leiomyoma. At an outside hospital she underwent supracervical abdominal hysterectomy with bilateral salpingectomy and intra-operatively was found to have an extra-uterine mass adherent to and growing into the urinary bladder, concerning for a bladder mass. Urology was consulted intraoperatively and the mass was biopsied with a diagnosis of extra-uterine leiomyoma. She was found to have right-sided obstructive hydronephrosis secondary to compression of the urinary bladder by the extra-uterine leiomyoma. Pelvis MRI with contrast showed a large soft-tissue pelvic mass measuring approximately 12.3 x 8.7 x 10.6 arising from the junction of remnant lower uterine segment/cervix abutting the rectosigmoid colon and severe right hydronephrosis and hydroureter secondary to the bladder mass ( Fig. 1 ). She was offered partial cystectomy or complete cystectomy with urinary conduit creation at an outside hospital and presented to our Urology department for a second opinion with the goal of bladder preservation. Fig. 1 Large bladder adherent cervical leiomyoma causing right obstructive uropathy. (A) Axial T2-weighted non-fat saturated MR image of pelvis shows large predominantly hypointense cervical leiomyoma (white asterisk) with significant mass effect on the urinary bladder (white arrowhead) and marked dilatation of the distal right ureter (white arrow). (B) Coronal maximum intensity projection T2 weighted Half-Fourier-Acquired Single-shot Turbo Spin Echo (HASTE) MR image of abdomen and pelvis shows marked right hydroureteronephrosis (white arrows) to the level of the pelvic mass. Fig. 1 Large bladder adherent cervical leiomyoma causing right obstructive uropathy. (A) Axial T2-weighted non-fat saturated MR image of pelvis shows large predominantly hypointense cervical leiomyoma (white asterisk) with significant mass effect on the urinary bladder (white arrowhead) and marked dilatation of the distal right ureter (white arrow). (B) Coronal maximum intensity projection T2 weighted Half-Fourier-Acquired Single-shot Turbo Spin Echo (HASTE) MR image of abdomen and pelvis shows marked right hydroureteronephrosis (white arrows) to the level of the pelvic mass. Patient reported rare urinary incontinence but otherwise no lower urinary tract symptoms. Serum creatinine was 0.99 mg/dL with an estimated glomerular filtration rate of 66 ≥ 60ml/min/BSA. She underwent renal scintigraphy with Tc-99m MAG-3 which showed asymmetric, reduced and delayed perfusion to the right kidney with differential renal function of 85.5 % for the left kidney and 14.5 % for the right kidney. Overall there was severe right hydronephrosis with minimal excreted radiotracer into the right renal collecting system and proximal ureter, findings consistent with obstruction. Cystoscopy was performed which showed a large submucosal mass primarily on the right side of the bladder and the trigone and posterior wall with minor telangiectasia at the 9 o'clock position of the bladder neck just inside the bladder and contiguous with inferior aspect of the bladder wall ( Fig. 2 ). Fig. 2 Cystoscopy demonstrates (A) mass effect from the submucosal bladder mass and (B) significant submucosal vascularity. Fig. 2 Cystoscopy demonstrates (A) mass effect from the submucosal bladder mass and (B) significant submucosal vascularity. Vascular Interventional Radiology was consulted. On the review of an outside pelvis CTA, there was candidate arterial supply to the mass from branches of the right superior and/or inferior vesical arteries. After multidisciplinary discussion, the patient presented for trans-arterial embolization of the cervical leiomyoma with the goal of shrinking the leiomyoma to relieve the right sided obstructive uropathy and bladder preservation. Patient underwent preprocedural epidural catheter placement by the regional anesthesia team. A foley catheter was placed. Subsequently, via a transfemoral approach, diagnostic pelvic angiography with intraprocedural sub-selective CTA were performed which demonstrated arterial supply to the entire mass from the right inferior vesical artery with no arterial supply from the right superior vesical artery. The right inferior vesical artery was embolized to stasis with 200 μm spherical embolic (HydroPearl®, Terumo, Japan) ( Fig. 3 ). She was admitted for overnight observation and discharged on post-procedure day 1 following a successful voiding trial. She had approximately 10 days of urinary urgency and frequency that was managed with Oxybutynin. Otherwise, no peri-procedural or post-procedural complications. Fig. 3 Selective right vesical artery embolization treating the bladder adherent leiomyoma. A) Selective right internal iliac artery angiogram in steep lateral anterior oblique projection demonstrates a common vesical artery trunk giving right superior vesical artery branches and a hypertrophied right inferior vesical artery branch (white asterisk). B) Selective right superior vesical artery angiogram demonstrates no supply to the hypervascular mass (white arrowhead). C) Selective right inferior vesical artery angiogram demonstrates supply to the entirety of the hypervascular mass (white arrow). D) Intraprocedural selective right inferior vesical artery CT angiogram confirmed supply to the entirety of the hypervascular mass and no vascular supply beyond the mass (white arrow). Hypervascular mass was embolized to stasis with particles. E) Post-embolization non-contrast CT through the pelvis demonstrates embolic material throughout the entire mass (white arrow) and F) completion right internal iliac artery angiogram demonstrates hemostasis in the embolized mass (white asterisk). Fig. 3 Selective right vesical artery embolization treating the bladder adherent leiomyoma. A) Selective right internal iliac artery angiogram in steep lateral anterior oblique projection demonstrates a common vesical artery trunk giving right superior vesical artery branches and a hypertrophied right inferior vesical artery branch (white asterisk). B) Selective right superior vesical artery angiogram demonstrates no supply to the hypervascular mass (white arrowhead). C) Selective right inferior vesical artery angiogram demonstrates supply to the entirety of the hypervascular mass (white arrow). D) Intraprocedural selective right inferior vesical artery CT angiogram confirmed supply to the entirety of the hypervascular mass and no vascular supply beyond the mass (white arrow). Hypervascular mass was embolized to stasis with particles. E) Post-embolization non-contrast CT through the pelvis demonstrates embolic material throughout the entire mass (white arrow) and F) completion right internal iliac artery angiogram demonstrates hemostasis in the embolized mass (white asterisk). At 6-month follow-up, pelvis MRI was performed which demonstrated approximately 40 % reduction in the overall size of the leiomyoma with a large area of central necrosis and persistent but decreased now moderate right hydronephrosis (not shown). At 1-year follow-up, abdomen/pelvis MRI was performed which showed resolution of the right hydronephrosis with continued reduction in size of the mass with greater than 50 % of the mass non-enhancing, non-viable tissue (not shown). At 2-year follow-up renal scintigraphy was repeated with Tc-99m MAG-3 which showed increased right kidney function from 14.5 % to 23.3 % and renal ultrasound showed mild right renal cortical thinning but no hydronephrosis. At 3-year follow-up patient endorsed no lower urinary tract symptoms and repeat pelvis MRI showed decreased size of the mass measuring 8.3 × 6.7 cm from 12.3 × 8.7 cm with more than two-thirds of the mass non enhancing, non-viable ( Fig. 4 ). Creatinine was stable at 1.0 mg/dL. Fig. 4 3-year follow-up after selective right inferior vesical artery embolization for treatment of bladder adherent cervical leiomyoma with resolution of right sided obstructive uropathy. A-B) Grayscale ultrasound of the A) right and B) left kidneys demonstrate long-term resolution of the right hydronephrosis with decreased right kidney size and cortical thickness compared to the left kidney. C-D) Coronal and axial post-gadolinium enhanced T1-weighted MR images of the pelvis demonstrates greater than 50 % reduction in volume of the bladder adherent mass with central necrosis and a rim of viable enhancing tissue (white arrow). Fig. 4 3-year follow-up after selective right inferior vesical artery embolization for treatment of bladder adherent cervical leiomyoma with resolution of right sided obstructive uropathy. A-B) Grayscale ultrasound of the A) right and B) left kidneys demonstrate long-term resolution of the right hydronephrosis with decreased right kidney size and cortical thickness compared to the left kidney. C-D) Coronal and axial post-gadolinium enhanced T1-weighted MR images of the pelvis demonstrates greater than 50 % reduction in volume of the bladder adherent mass with central necrosis and a rim of viable enhancing tissue (white arrow).

Sydney Whalen: Conceptualization, Writing – original draft, Writing – review & editing. R. Jeffrey Karnes: Writing – review & editing. Scott M. Thompson: Conceptualization, Data curation, Formal analysis, Investigation, Supervision, Writing – original draft, Writing – review & editing.

In summary, herein we describe successful treatment of a bladder adherent large exophytic cervical leiomyoma with selective inferior vesical artery embolization resulting in resolution of unilateral obstructive uropathy from the mass, improvement in split kidney function and preservation of the bladder.

Uterine leiomyoma are common benign tumors most commonly arising from the uterine body. Uterine leiomyoma can grow into and become adherent to the urinary bladder resulting in obstructive uropathy in upwards of 11 % of patients. 2 Uterine artery embolization is a safe and effective treatment option for primary and secondary post-partum hemorrhage as well as symptomatic uterine fibroids and adenomyosis. 4 , 5 , 6 Moreover, prior studies have shown that uterine artery embolization in patients with leiomyoma-associated hydronephrosis is a safe and effective treatment option with resolution of hydronephrosis in more than 80 % of patients. 7 , 8 While the majority of leiomyoma arise from the uterine body, extra-uterine leiomyoma arising from the cervix are more rare occurring in less than 1 % of patients. 3 Uterine artery embolization has also been shown to be safe and effective treatment option for symptomatic fibroids arising from the cervix. 9 The present case is a rare example of a cervical leiomyoma adherent to and growing into the urinary bladder resulting in unilateral obstructive uropathy with reduced kidney function. Given the prior hysterectomy with ligation of the uterine arteries, the dominant arterial supply to the mass was from the right inferior vesical artery. Vesical artery embolization has been shown to be a safe and effective treatment option for intractable hematuria from both bladder tumors and radiation cystitis. 10 Given embolization of the inferior vesical artery with small particles to achieve tumor necrosis, the patient did experience transient urinary frequency and urgency that was managed with Oxybutynin. This is an expected but transient side-effect of embolization of the bladder. At long-term follow-up the mass has decreased in overall volume by greater than 50 % with more than two-thirds of the mass non-viable, non-enhancing with a large area of central necrosis that will continue to involute over time. Importantly, the patient has no lower urinary tract symptoms, resolution of the right-sided obstructive uropathy with improved with kidney function to almost 25 % and preservation of her bladder. There are limitations to this study, primarily in it being a single case report. Furthermore, the optimal embolic agent for a bladder adherent leiomyoma is not known.

Uterine leiomyoma are common benign tumors that can present in women with both urologic and gynecologic symptoms including lower urinary tract symptoms, sexual dysfunction, menstrual irregularities, pelvic pain, infertility and sexual dysfunction. 1 Uterine leiomyoma can grow into and become adherent to the urinary bladder resulting in obstructive uropathy in up to 10.5 % of patients. 2 While the majority of leiomyoma arise from the uterine body, extra-uterine leiomyoma arising from the cervix or vagina are more rare. 3 This report describes a case of a patient who underwent hysterectomy for symptomatic large uterine leiomyoma and intra-operatively was found to have a biopsy-proven extra-uterine exophytic cervical leiomyoma adherent to and growing into the urinary bladder causing right-sided obstructive uropathy.