UROLOGY JOURNAL Vol. 11 No. 04 July - August 2014 1844
Abdominal Endometriosis Arising in an Exstro-
phy Patient
Michael S Floyd (Jr),1 Rahul Mistry,2 Charles T Burrows,2 Andrew
David Baird1
Departments of Urology 1 and Pa-
thology 2, Aintree University Hos-
pital, Lower Lane, Liverpool, L9
7AL, United Kingdom.
Corresponding Author:
Michael S Floyd (Jr), Specialist
Registrar, Department of Urology,
Aintree University Hospital, Low-
er Lane, Liverpool L9 7AL, United
King-dom.
Tel: +44 151 5293775
E-mail: nilbury@gmail.com
Received August 2013
Accepted February 2014
Keywords: abdomen; surgery; endometriosis; etiology; complications; bladder exstrophy.
INTRODUCTION
Bladder exstrophy is a rare congenital anomaly. Associations with complete duplication of the genitourinary and gynecological systems have been reported. We describe a case of an abdominal swelling in an exstrophy patient which revealed endometriosis.
CASE REPORT
A 29 years old nulliparous female presented to the urology outpatients with a four months history of an in-
termittently enlarging abdominal swelling located over a scar. Her past history was remarkable for bladder
exstrophy for which she underwent primary bladder closure on the third day of life. This had been followed
by a succession of urological procedures over seven years. She had initiated self-intermittent catheteriza-
tion following this. Her menarche had been at the age of thirteen. On presentation she described episodic
swelling over her inferior abdominal wall beneath her laparotomy scar. It was unrelated to her menses and
no systemic upset occurred.
On examination a firm indurated area was noted beneath the skin within the rectus sheath. Subsequent
magnetic resonance imaging (MRI) confirmed the presence of a 3.5 cm × 2.7 cm × 3.9 cm enhancing mass
within her abdominal wall which was separate from the peritoneum (Figure 1).
Under general anesthesia an excisional biopsy was performed. Intraoperatively a mass was found within
the reconstructed rectus sheath. The peritoneum was opened and a segment from the dome of the bladder
was removed as the mass was attached to it inferiorly. The remainder of the bladder was unremarkable.
CASE REPORT
skin covered double bladder exstrophy has been reported along with
complete duplication of the mullerian structures(4) and also separately
with colonic sequestration with a normal hindgut.(5)
Following progression into adulthood the reconstructed female ex-
strophy patient may face problems with parturition, sexual health and
gynecological concerns. Krisoloff and colleagues have found that
multiple surgical procedures with scarring in the abdominopelvic area
may have a detrimental effect on body image and sexual function.(6)
Successful pregnancies have been reported in patients who have un-
dergone exstrophy repair(7) but caesarean delivery is recommended(8)
Gynecological complications of exstrophy repair include mucocolpos
and vaginal stone formation.(9,10) Burbige and colleagues studied fe-
male exstrophy patients and found that none had endometriosis.(11) Our
patient was nulliparous and denied dyspareunia or endometriosis.
We describe an abdominal mass in a corrected female exstrophy pa-
tient, which when excised, revealed endometriosis. Reports exist of
dual pathologies in exstrophy patients who underwent reconstruction
in adulthood.(12) Kitajima and colleagues reported a case of scar en-
dometriosis is a 26 years old exstrophy patient who underwent repair
as an infant.(13) Our case differs, however, as the ectopic deposit was
painless and unrelated to her menstrual cycle.
CONCLUSION
We believe this to be the second reported case of scar related abdom-
inal endometriosis in an exstrophy patient. As the long term follow
up of this population is predominantly urological, female exstrophy
patients with gynecological problems may present to the urologist.
Therefore, the differential diagnosis of an abdominal mass in a female
exstrophy patient should include gynecological pathologies such as
ectopic endometrial tissue.
CONFLICT OF INTEREST
None declared.
REFERENCES
1. Marshall VF, Meucke EC. Variations in exstrophy of the bladder. J Urol.
1962:88;776-84.
2. Sheldon GA, McLorie GA, Khoury A, Churchill BM Duplicate bladder
exstrophy: a new variant of clinical and embryological significance. J
Urol. 1990;144;334-6.
3. Ahmed S, Abu Daia J. Exstrophic abdominal wall defect without bladder
exstrophy (pseudoexstrophy). Br J Urol. 1998;81:762-3.
4. Berkowitz J, Warlcik C, North A, Gearhart JP. Duplicate bladder exstro-
phy with complete duplication of Mullerian Structures. Urology.
2007;70:811.e15-7.
5. Kajbafzadeh AM, Aghdas FS, Tajik P. Complete covered duplication
of the bladder, urethra, vagina, uterus and visceral sequestration. Int J
Urol. 2006;13:1129-31.
6. Krisiloff M, Puchner PJ, Tretter W, Macfarlane MT, Lattimer JK. Preg-
She was discharged after 48 hours following removal of a suprapubic
catheter. Outpatient review at 2 months revealed no recurrence and she
remained asymptomatic.
Final histopathological examination confirmed a 40 g specimen con-
sisting of fibrous tissue containing glands typical of endometriosis
(Figure 2).
DISCUSSION
Variants of classical bladder exstrophy have been described by Mar-
shall and colleagues(1) and include male and female epispadias with or
without incontinence, superior vesical fissures, duplicated exstrophy
and cloacal exstrophy. Duplicate bladder exstrophy consists of either
an anterior-posterior or side by side alignment.(2) Pseudoexstrophy
patients have normal functioning bladders and external genitalia but
have the skeletal and abdominal findings of exstrophy.(3) In adults,
Figure 1. Abdominal magnetic resonance image showing a mass located on
the inferior abdominal wall which enhanced following the administration of
contrast.
Figure 2. Photomicrographs of a section from the excised mass demonstrating
endometrial glands and stroma in fibrous tissue (Hematoxylin and Eosin; left
× 40, right × 80.
1845 Case Report
UROLOGY JOURNAL Vol. 11 No. 04 July - August 2014 1846
Abdominal Endometriosis-Floyd et al
nancy in women with bladder exstrophy. J Urol. 1978:119;478-9.
7. Sharma D, Singhal SR, Singhal SK. Successful pregnancy in a patient
with previous bladder exstrophy. Aust N Z J Obstet Gynaecol.
1998;38:227-8.
8. Mathews RI, Gan M, Gearhart JP. Urogynaecological and obstetric
issues in women with the exstrophy-epispadias Complex. BJU Int.
2003;91:845-9.
9. Zorn KC, Spiess PE, Salle JL, Jednak R. Mucocolpos associated with
bladder exstrophy; a case report. Can J Urol. 2005;12:2614-5.
10. Eyk NA, Grover S, Fink AM. Vaginal calculus as a late complication of
bladder Exstrophy. J Pediatr Adolesc Gynecol. 2003;16:285-7.
11. Burbige KA, Hensle TW, Chambers WJ, Leb R, Jeter KF. Pregnancy and
sexual function in women with bladder exstrophy. Urology. 1986;28:12-
4.
12. Quiroz-Guerrero J, Badillo M, Munoz N, Anaya J, Rico G, Maldona-
do-Valadez R. Bladder augmentation in a young adult female exstrophy
patient with associated omphalocele; An extremely unusual case. J Pedi
atr Urol. 2009;5:330-2.
13. Kitajima T, Inoue M, Uchida K, Otake K, Kusunoki M. Scar endometri-
osis in a patient with bladder exstrophy. Int Surg. 2013;98:145-8.