CASE REPORT Adult Ureterocele Presenting with Ureteral Obstruction and Urosepsis during Pregnancy Catherine R Harris,1 Amjad Alwaal,1,2* Glen Yang,1 Michael L Eisenberg,1 Benjamin N Breyer1 Keywords: pregnancy; ureterocele; adult; female; hydronephrosis; pathology. INTRODUCTION Ureteroceles are cystic dilatations of the distal ureter that occur due to congenital ureteric wall weakness.(1) They can be orthotopic, occurring in normal ureteric locations and most commonly seen in adults. On the other hand, heterotopic ureteroceles are located in ectopic ureters or in ectopic duplex renal systems, and are more common in children.(2) Ureterocele causing obstruction in adults is less commonly reported. There are few case reports and small case series in the adult population that describe ureteral obstruction from ureteroceles,(3-5) and only one published report of a ureterocele presenting as prolapsed mass containing stones during pregnancy.(6) We describe a report of an adult presenting with an obstructing ureterocele and urosepsis during pregnancy. The ureter- ocele was successfully extirpated cystoscopically without radiation exposure to mother or fetus. We also review the varied presentations and management of obstructing ureteroceles in adults who have presented at our institution. CASE REPORT A 35 year-old woman gravida 5 para 2 at 22 weeks gestation presented to the emergency room with fevers and right flank pain. She was febrile to 39.0oC, tachycardic at 120/min, with stable blood pressure. On physical examination she had right costovertebral angle tenderness. Laboratory results revealed a leukocytosis of 19 × 109/L and creati- nine of 0.5 mg/dL. Her urinalysis with microscopy was positive for pyuria and bacteriuria. Renal-bladder ultra- sound showed moderate right hydroureter (grade 3) proximal and distal to the gravid uterus as well as a ureterocele at the ureterovesical junction (Figure 1). The patient was otherwise healthy, with a history of a single urinary tract infection 2 years ago while not pregnant. Surgical Technique The patient was taken emergently to the operating room for endoscopic ureteral decompression. Cystoscopy was performed which showed a 3 cm right ureterocele. A ureteral orifice was not apparent, and a guidewire was unable to be passed into the ureter. Urine efflux was not visualized. An attempt to puncture the thick walled ureterocele with the Bugbee electrode (Medline Industries, Mundelein, IL, USA) was unsuccessful. A Collins knife (STORZ, Tuttlingen, Germany) was then used to incise the ureterocele. After extirpation with the Collins knife, we were able to visualize what appeared to be a ureteral orifice at the lateral extend of the ureterocele (Figure 2). A guidewire and 5-French ureteral exchange catheter passed through the ureteral orifice easily. We placed then a temporary 1 Department of Urology, University of California, San Francisco, USA. 2 Department of Urology, King Abdul Aziz University, Jeddah, Saudi Arabia. *Correspondence: 400 Parnassus Avenue A633, San Francisco, CA 94143-0738, USA. Tel: +1 415 206 8805. Fax: +1 415 206 5153. E-mail: amjadwal@yahoo.com. Received February 2015 & Accepted June 2015 Abbreviations: M, male; F, female; CT, computed tomography; IVP, intravenous pyelography; DMSA, dimercaptosuccinic acid; UTI, urinary tract infection. Age (years) Sex Presenting Diagnostic Laterality Type of Associated Surgical Symptom Imaging System Stone Technique 35 F Pyelonephritis Ultrasound Right Single, orthotopic No Incision 61 M Pyelonephritis CT, IVP Bilateral Single, orthotopic Yes Excision, laser lithotripsy, stent 27 F Flank pain CT, DMSA Left Duplicated, ectopic No Heminephrectomy 26 F Recurrent UTI Ultrasound Left Single, orthotopic No Incision 68 F Recurrent UTI CT Left Single, orthotopic No Incision 22 M Recurrent UTI CT Left Single, orthotopic No Open excision, ureteral reim plantation Table. San Francisco general hospital experience with adult ureteroceles 1996-2015. Case Report 2285 ureteral stent that was removed 4 weeks later. Postoperative Course The patient defervesced and her pain resolved after the procedure. Her urine culture grew pansensitive Escheri- chia coli. She was discharged on post-operative day two with a two-week course of amoxicillin. One month after her procedure she had complete resolution of her symp- toms and was afebrile with a white blood cell count of 8 × 109/L. Repeat ultrasound showed stable hydrone- phrosis and hydroureter bilaterally consistent with a 7-month pregnancy. Her creatinine was 0.4 mg/dL at baseline. DISCUSSION Our experience with adult ureteroceles is extremely varied, as shown in Table. In the pediatric population, symptomatic ureterocele is related to a variety of com- plex congenital anomalies, such as duplex kidney, ec- topic ureter and bladder outlet obstruction.(7) In contrast, adult ureteroceles are usually intravesical, small, and single system. Another difference between ureterocele according to age is the presence of calculi within the ureterocele. Calculi are commonly reported in adults, but are rare in children. It is likely that the formation of calculi over time from urinary stasis in an otherwise small asymptomatic ureterocele is the cause of delayed presentation of ureteroceles into adulthood. Increased estimated glomerular filtration rate and hy- dronephrosis from ureteral compression by the gravis uterus are well known physiologic and anatomic man- ifestations of pregnancy.(8) Physicians must therefore rely on clinical judgment to differentiate between phys- iologic and pathologic obstruction of the urinary sys- tem. The concern for fetal radiation also limits the use of more sensitive diagnostic imaging techniques such as computed tomography scan in determining the cause of obstruction. As this report demonstrates, it is important to carefully consider non-pregnancy related causes of obstruction in the pregnant population, such as ureter- ocele. CONCLUSIONS In conclusion, this is the first report of an obstructing ureterocele in a pregnant woman. Therefore, uretero- cele should be considered as a potential cause for ob- struction in pregnant women. CONFLICT OF INTEREST None declared. REFERENCES 1. Neufang KF MU, Beyer D. The roentgen diagnosis of ureterocele-with special reference to the simple orthotopic ureterocele in adults. Rontgenblatter. 1981;34:9-14. 2. Diard F CL, Bondonny JM, Elie G. Simple orthotopic ureteroceles in children. J Radiol. 1980;61:387-95. 3. Sinha RK, Singh S, Kumar P. Prolapsed ureterocele, with calculi within, causing urinary retention in adult female. BMJ Case Reports. 2014;2014. 4. Prakash J, Goel A, Kumar M, Sankhwar S. Stone in ureterocele peeping through ureteric orifice. BMJ Case Reports. 2013;2013. 5. Westesson K, Goldman H. Prolapse of a single-system ureterocele causing urinary retention in an adult woman. Int Urogynecol J. 2013;24:1761-3. 6. Scovell JM, Chan RC, Khavari R. Prolapse of a Single System Large Ureterocele Containing Multiple Stones in a Pregnant Woman. Urology. 2014;83:e3-e4. 7. Landi L EA, Adorisio O. Prolapsed vaginal ureterocele as a cause of urinary incontinence in a child. Urol J. 2015;12:1999-2000. 8. Hill CC, Pickinpaugh J. Physiologic Changes in Pregnancy. Surgical Clinics of North America. 2008;88:391-401. Figure 1. Bladder ultrasound showing right ureterocele. Figure 2. Cystoscopy showing the ureterocele pre- and post-extirpation. Vol 12 No 04 July-August 2015 2286 Adult Ureterocele in Pregnancy-Harris et al.