Successful endoscopic management for early manifested postcesarean section uretero-uterine fistula: A case report and literature review

Abstract

We report a rare case of post-cesarian section uretero-uterine fistula (UUF) in a 36 years old female who presented on a postoperative day 5 with paradoxical urine incontinence and occasional normal urethral voiding. After a complete evaluation, Cystoscopy and right internal JJ ureteric stent insertion was done; the patient had a dramatic response; she was completely dry until the stent was removed three months later with complete healing of UUF. To our knowledge, the present case is the 4th reported with successful endoscopic management for post-obstetrics and gynecological procedures Uretero-Uterine fistula.

INTRODUCTION

Obstetric and gynecological procedures associated with iatrogenic urinary tract injuries account for 0.4%–2.5% for benign conditions. Uretero-uterine fistula (UUF) is a rare presentation that constitutes 1.7%–5.1% of all urogenital fistulas.[1]

To our knowledge, there are 54 cases of postobstetric/gynecological UUF reported in the literature up to date.

Iatrogenic UUF was reported postcesarean section in most cases. Management depends mainly on surgical repair as reported in more than two-thirds of cases; however, there is still a role for minimally invasive procedures such as Ureteroscopy Ureteroscopy (URS) and internal JJ ureteric stenting.

We report a rare case of postcesarean section UUF with early presentation and early successful endourological management, reviewing the previously published cases.

CASE REPORT

A 36-year-old female patient p3 + 1 underwent emergency cesarean section for uterus rupture on the 36^th gestational week and delivered a live boy weighing 2810 g.

The postoperative period was uneventful until postoperative day 5 when the patient experienced paradoxical urine incontinence with normal urethral voiding; the abdominal examination was unremarkable; pelvic examination showed a defect in the anterior fornix and urine pooling in the posterior fornix. KUB ultrasound of kidney, ureter, and bladder showed mild right hydroureteronephrosis, so computed tomographic (CT) urography was done and showed mild right hydroureteronephrosis and contrast extravasation at the lower right ureteric segment with right UUF [Figure 1a]; we excluded vesicovaginal and vesicouterine fistula after three swab test.

Figure 1.

Radiological findings of postcesarean section right uretero-uterine fistula. (a) Computed tomographic urography showing mild right hydroureteronephrosis and contrast extravasation at the lower right ureteric segment with right uretero-uterine fistula. (b) Right retrograde ureteropyelography showed a medial deviation of the lower right ureteric segment (fistula site). (c) Successful endoscopic management for right uretero-uterine fistula, retrograde passage of Guidewire up to the right kidney followed by right JJ ureteric stenting. (d) Postright JJ removal, right RGP showed normal course and caliper of right ureter. (e) Follow-up computed tomographic urography 4 weeks poststent removal showing no more backpressure with complete healing of uretero-uterine fistula

Hysteroscopy showed urine coming from cervical Os, Cystoscopy and Right retrograde ureteropyelography showed a medial deviation of the lower Right ureteric segment (fistula site) [Figure 1b], and the diagnosis of the right UUF was confirmed.

Using cystoscopy, right JJ ureteric stent was inserted successfully [Figure 1c]. Day 1 poststent fixation, the patient had a dramatic response, and she was completely dry till the stent removed 3 months later. At that time, right Retrograde Pyelopgraphy (RGP) showed normal course and caliper of the right ureter [Figure 1d]. Follow-up CT urography done 4 weeks poststent removal revealed no more backpressure with complete healing of UUF [Figure 1e].

DISCUSSION

Ureteric injury during obstetric and gynecological procedures is rare and accounts for less than 0.5%–1% and may rise to 2% in radical hysterectomy.[2]

There are many factors suggested predisposing for iatrogenic ureteric injury during obstetric and gynecological procedures, including previous cesarean section due to fibrosis and ureteric displacement, dextro-rotation of the uterus explaining the more frequent injury of the left ureter, and also prolonged labor with disproportion that might result in ureteric wall edema and necrosis.[3] The lower third segment of the ureter can be injured through a low uterine transverse incision or by too far lateral incision extension, inadvertent suture ligation, or hemostatic clip that might result in a hematoma, infection, and fistulous tract formation between the uterus and injured ureter.[1]

UUF can be presented early days postoperative as in our case or a few weeks later. It can be explained by injury mechanism, which would present early in patients with direct ureteric injury, or later with ischemic injury. UUF usually presents with paradoxical urine incontinence and normal urethral voiding.[3]

Diagnosis can be confirmed with urine dribbling from cervical uteri orifice on speculum examination; a three-swab test, and cysto-panendoscopy to rule-out vesicovaginal and vesicouterine fistulae. Sheen et al. modified the swab test by phenazopyridine administration every 8 hours followed by installation of 200 cc of methylene blue into the urinary bladder after 24 hours; yellow urine from the vagina could confirm UUF and exclude vesicovaginal fistula. Intravenous urography and CT urography can identify the location of the ureteric injury, extravasation, or even the fistulous tract and give an idea about renal function. Retrograde ureteropyelography can delineate the lower ureteric segment that not-enhanced in the contrast study and assess ureteric continuity to help in decision making.[2]

UUF management’s rationale depends on the early minimal invasive procedures’ intervention that can guarantee the continuity of the injured ureter, preserve renal function, and prevent urine leakage with subsequent infection and tissue necrosis.[2] Percutaneous nephrostomy can ensure urine diversion with complete healing of UUF as reported in two cases, but the surgical repair was mandatory later in six patients. On the other hand, internal JJ ureteric stent for UUF was successful as a treatment option for UUF in reported four cases, including the current case, and failed in three cases.

Surgical repair is the mainstay in treating uretero-uterine fistula, as reported in > 68% of cases, especially in delayed presentation and failed PCN or endoscopic management. The surgical approach depends mainly on UUF location, length of ureteric segment involved, the extent of fibrosis, and surgeon preference. It includes uretero-ureterostomy, ureteroneocystostomy with psoas hitch and boari flap if indicated, and human dura matter allograft ureteroplasty.[4] Surgical intervention could be open, laparoscopic, or robot-assisted laparoscopic repair.[3]

We reviewed the previously reported cases (n=54) of post-obstetrics and gynecology procedures UUF as shown in Tables 1 and 2; we found that 80 % of patients followed caesarian section, 10 % post-hysterectomy, and 10 % post-abortion (D&C). UUF was left side in 64% of reported cases. Presentation ranged between one day and six months postoperative (median=20 days). The treatment options included spontaneous healing in one case,[5] percutaneous nephrostomy was sufficient for UUF healing in only 25%, endoscopic intervention and JJ stent insertion was successful in 57%, and surgical repair was the gold standard treatment option offered to most patients (n>30) with a success rate > 96%

Table 1.

The previously published cases of post-obstetric and gnecology procedures Uretero-Uterine Fistula

Author	Cases number	Etiology	Time to present	Diagnosis	Side	Management	Outcome
De Aguiar, 1959	1	LSCS	-	-	-	-	-
Zielinski, 1962	1	-	-	-	-	-	-
Claret et al., 1964	2
Truc et al., 1969	1	-	-	-	-	-	-
Mahgoob and Zeniny, 1971	1	LSCS	-	IVU, dye test, HSG	Left	UNC	Cured
Suhler and Saout, 1971	1	LSCS	-	-	-	-	-
Dukhovnikov et al., 1973	1	LSCS	-	-	-	-	-
Jequier and Piper, 1973	1	LSCS	2 weeks	Dye test, CPE, IVU, RGP	Left	PCN for 7 weeks	Cured
Barton et al., 1978	1	Abortion, D and C	-	IVU, RGP	-	PCN	Cured
Oumachigui et al., 1980	3	LSCS	9 days 15 days 23 days	IVU, dye test, CPE	Right, left, left	UNC	Cured
Moussu et al., 1980	1	LSCS	-	IVU, RGP	Left	UNC + psoas hitch	Cured
Keegan and Forkowitz, 1982	1	D and C	1 week	IVU	Right	PCN, UNC + psoas hitch	Cured
Docquier, 1986	1	LSCS	-	-	-	UU	Cured
Gorrea et al., 1986	1	LSCS	-	IVU, RGP	-	Spontaneous healing	Cured
Pernin et al., 1993	1	-	-	-	-	-	-
Natta et al., 1993	1	LSCS	-	-	-	-	-
Sharfi et al., 1994	8	LSCS, hysterectomy	-	IVU, dye test, cystoscopy, RGP	-	UNC + psoas hitch (3 cases) + 3 boari flap (3 cases)	All cured
Fernandez et al., 1994	1	AH	-	-	-	-	Cured
Saltutti et al., 1994	1	LSCS	20 days	IVU, CT, cystoscopy	Left	PCN, UNC	Cured
Lodh et al., 1996	1	Abortion, D and C	6 months	IVU, RGP	Right	UNC + boari flap	Cured
Lazarevski and Badiev, 1996	1	LSCS	2 weeks	IVU, dye test, HSG	Left	UU, PCN, UNC	Cured
Wang and Hung, 1997	1	Abortion, D and C	-	IVU, CT nephrostogram	Right	PCN, UNC + boari flap	Cured
Kajbafzadeh, 1997	1	LSCS	-	Cystogram, IVU, RGP, dye test	Right	Ureteric dilatation and DJS	Cured
Sheen et al., 1998	1	LSCS	4 weeks	IVU, dye test, HSG, cystography	Left	UNC (Politano–Leadbetter technique)	Cured

LSCS: Lower-segment cesarean section, AH: Abdominal hysterectomy, IVU: Intravenous urography, HSG: Hysterosalpingogram, CPE: Cystopanendoscopy, RGP: Retrograde pyelogram, CT: Computerized tomography, UNC: Ureteroneucystostomy, PCN: Percutaneous nephrostomy, UU: Ureteroureterostomy, DJS: Double-J stent

Table 2.

The previously published cases of post-obstetric and gynecology procesures Uretero-Uterine Fistula

Author	Cases number	Etiology	Time to present	Diagnosis	Side	Management	Outcome
Nabi et al., 2000	1	LSCS	Week	IVU, CPE, TST	Left	UNC	Cured
Nouira et al., 2000	1	LSCS	-	-	-	URS, exploration of the ureter	-
Singh et al., 2001	1	LSCS	2 weeks	IVU, CPE, dye test, Moir’s test	Left	URS, balloon dilatation, DJS	Cured
Billmeyer et al., 2001	1	LSCS	-	IVU, cystoscopy, TST	Left	Repair of VVF, UNC	Cured
Koziak et al., 2004	1	LSCS	-	IVU	-	Repair with human dura mater allograft for stricture ureter	Cured
Adhikary et al., 2005	1	D and C	1 day	IVU, CPE, RGP	Left	UNC + psoas hitch	Cured
El-Tabey et al., 2006	1	LSCS	-	IVU, cystoscopy	Left	UNC + boari flap	Cured
Eze, 2007	1	LSCS	-	-	-	-	-
Lanary et al., 2008	1	LSCS	18 days	CT/IVU, cystoscopy, URS	Right	PCN, UNC	Cured
Kumar et al., 2011	1	LSCS	Week	IVU, cystoscopy, CT nephrostogram	Left	PCN, laparoscopic UNC	Cured
Levy et al., 2012	1	LSCS	2 days	CT, CPE	Right	PCN	-
Shetty et al., 2016	1	-	-	CT, cystoscopy, RGP	Left	DJS for 6 weeks	Cured
Lo et al., 2016	3	ELSCS	-	IVU, CT, dye test, cystoscopy	Left	UNC	Cured
Yamamoto et al., 2017	1	LSCS	-	IVU, CT, RGP, dye test, CPE	Right	UNC + psoas hitch, hysterectomy	Cured
Sharma and Ratkal, 2019	3	LSCS	2–3 weeks	IVU, CT, RGP	(2) Left	UNC	Cured
Selvaraj et al., 2020	1	LSCS	2 months	CT, CPE, RGP	Right	Robot-assisted laparoscopic bilateral ovary preserving hysterectomy, UNC + boari flap and psoas hitch	Cured
Current case	1	LSCS	5 days	CT, TST, CPE, RGP	Right	DJS stent for 3 months	Cured

LSCS: Lower-segment cesarean section, ELSCS: Emergency LSCS, CPE: Cystopanendoscopy, TST: Three-swab test, HSG: Hysterosalpingogram, RGP: Retrograde pyelogram, UNC: Ureteroneucystostomy, PCN: Percutaneous nephrostomy, UU: Ureteroureterostomy, URS: Ureteroscopy, DJS: Double-J stent, IVU: Intravenous urography, CT: Computerized tomography, VVF: Vesicovaginal fistula

The present case of right UUF presented early days postcesarean section. It was managed successfully with cystoscopy and internal JJ ureteric stent for 3 months, obviating the need for percutaneous nephrostomy and/or surgical repair with its morbidity and complications.

CONCLUSION

Endoscopic management for postobstetric and gynecological UUF is a feasible and less invasive option and can be offered before surgical repair, especially to early presented cases.

Consent

We obtained written informed consent for publication from the patient; a copy is available for review.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that her name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.