Keywords transverse vaginal septum - hysteroscopy - hematocolpos - laparoscopy - vaginal anomaly
Palavras-chave septo vaginal transverso - histeroscopia - hematocolpos - laparoscopia - anomalia
vaginal
Introduction
Transverse vaginal septum (TVS) is an uncommon anomaly of the female genital tract
whose incidence ranges from 1:2100 to 1:84000.[1 ]
[2 ]
[3 ]
The etiology of this embryologic anomaly is still unknown; this anatomical condition
is linked to autosomal recessive transmission, but, in most cases, a genetic origin
cannot be found.[4 ]
Physiologically, the sinovaginal bulbs invaginate from the urogenital sinus and meet
the Müllerian tubercle on the caudal end of the Müllerian ducts to form the vaginal
plate that is then canalized to form the lower part of the vagina.[5 ]
Transverse vaginal septa are thought to result from a failure in the canalization
of the vaginal plate at the point where the urogenital sinus meets the Müllerian duct.[1 ]
Genitourinary and gastrointestinal tract anomalies might be associated with TVS, such
as: imperforate anus, mal-rotation of the gut, ectopic ureter with hypoplastic kidney,
hydronephrosis, vesicovaginal fistula, and bicornuate uterus.[6 ]
[7 ] Rare defects include musculoskeletal defects, aorta coarctation and atrial septal
defect.[7 ]
Transverse vaginal septum can develop anywhere in the vagina, and its more common
locations are the upper part of the vagina, the junction between the vaginal plate
and the caudal end of the fused Müllerian ducts.[1 ]
[2 ] Rock et al[7 ] observed that 46% of septa were located in the upper vagina, 35%, in the middle,
and 19%, in the lower vagina.
Imperforate septa usually present during adolescence with symptoms such as cyclical
abdominal pain, obstructed menstruation, hematocolpos, and, occasionally, urinary
retention.[1 ] Hematocolpos is defined as a distended vagina filled with menstrual blood; hematometra
is a term used to describe the distended uterus filled with blood caused by extreme
accumulation of menstrual products.[8 ]
The diagnosis is suspected when an abdominal or pelvic mass is palpated, or when a
foreshortened vagina is encountered and the cervix cannot be identified.[1 ] Clinical examination, ultrasound, and magnetic resonance imaging (MRI) are all used
in the diagnosis, and the MRI is useful before the surgery to determine the thickness
and depth of the septum.[9 ]
[10 ]
A review of the literature suggests that ultrasound is still the primary diagnostic
tool used, while the MRI is particularly useful to delineate more complex gynecologic
anatomies.[11 ]
[12 ]
In the literature, several approaches are described, but none seems to be better than
the others.[1 ]
It is deeply important to recognize as early as possible the disease to prevent patient
complications, including endometriosis, hydronephrosis, possible pyelonephritis and
superinfection that will cause sepsis. Moreover, the surgery should be performed by
an expert and educated team with the goal of restoring the regular anatomy and physiology
of the patient.[13 ]
We present a case of transverse vaginal septum in which the patient and her family
expressed the strong desire to preserve hymenal integrity due to a sociocultural belief.
We propose the management of the septum with vaginoscopic resection using a hysteroscope,
under laparoscopic control, followed by the introduction of a Foley catheter in the
vagina.
Case Description
A 14-year-old virgin girl presented to the emergency department complaining of severe
hypogastric pain. She presented suprapubic fullness, but did not present abdominal
distension or other symptoms.
Despite the presence of secondary sexual features, she had never had menarche, and
had a history of cyclical pelvic pain.
Upon examination, she had a tender suprapubic mass, palpable almost 2 cm under the
umbilicus. A rectal examination revealed an anterior mass. An abdominal ultrasound
revealed a distended uterus and a vagina filled with a homogenous thick fluid; a diagnosis
of hematocolpometra was made ([Fig. 1 ]).
Fig. 1 Abdominal ultrasound showing a distended uterus.
An MRI was performed, and it showed proximal vagina and uterine distension, especially
involving the cervix. The vagina appeared to terminate in a low complete transverse
septum, ∼ 3 cm from the vaginal introitus, with a 7-mm thickness. No other abnormalities
were detected ([Fig. 2 ]).
Fig. 2 (A) Sagittal view of the magnetic resonance imaging scan: hematocolpometra and level
of the septum (arrow); (B) transversal view of the septum (arrow).
The patient and her family refused vaginal surgery to preserve hymen integrity due
to a sociocultural belief. After counselling about performing the vaginoscopic resection
of the septum using a hysteroscope under laparoscopic control, the patient and her
family provided written informed consent for the procedure.
During laparoscopy, the uterus looked anteverted and enlarged. Both ovaries were normal.
No endometriosis was detected.
The vagina appeared grossly distended, obliterating the Douglas pouch. A 5-mm office
hysteroscope (Bettocchi Office Hysteroscope, Karl Storz GmbH & Co., Tuttlingen, Germany)
was introduced through the hymenal opening according to the vaginoscopic approach
technique.[14 ]
[15 ] The vaginoscopy revealed a bulging complete TVS. A 5-F bipolar electrode (Versapoint
Bipolar Electrosurgical System, Gynecare, Ethicon, Inc., Somerville, NJ) was introduced
through the operative channel of the hysteroscope, a bipolar electroresection of the
septum was performed, and 300 mL of dark, old blood was evacuated. A Foley catheter
was introduced from the introitus toward the septal perforation, and the balloon of
the catheter was insufflated with 15 ml of fluid. The patient recovered well from
the surgery, and was placed on continuous oral contraceptive pills to suppress her
menses and prevent hematocolpos reaccumulation.
She was dismissed three days after the procedure; a vaginoscopy was scheduled two
months later, when the Foley catheter was removed. At the vaginoscopic examination,
a fibrotic ring narrowing the vaginal canal was detected and completely resected with
the bipolar electrode, without hymenal damage ([Fig. 3 ]).
Fig. 3 (A-B-C-D) 5-F bipolar electrode introduced in the operative channel of the hysteroscope
and bipolar electroresection of the septum performed with dark old blood evacuated;
(E) Foley catheter introduced from the introitus toward the septal perforation and
the balloon of the catheter insufflated with 15 ml of fluid; (F) fibrotic ring resection
with bipolar electrode without damage to the hymen.
The patient was discharged on the same day, and underwent regular follow-up. The oral
contraceptive pill was stopped four months later. The patient experienced regular
menses and no other symptoms or discomfort 6 and 12 months after the last procedure.
An abdominal ultrasound scan showed a normal genital tract ([Fig. 4 ]).
Fig. 4 Normal uterus on ultrasound after 12 months.
Discussion
One of the main causes of hematocolpometra is a complete vaginal septum.[16 ] A transverse vaginal septum results from either incomplete canalization of the vaginal
plate or failure of the paramesonephric ducts to meet the urogenital sinus.[7 ]
[17 ]
Very few data are available in the literature about the surgical management. The studies
report on septum location, thickness and perforation.[1 ]
[3 ]
[7 ]
The location is also based on the distance from the vaginal introitus to the distal
end of the septum, and it can be low (< 3 cm), mid (3–6 cm), or high (> 6 cm). About
the thickness, the septum can be thin (< 1 cm) or thick (> 1 cm). In the presence
of perforation, the thickness can usually be determined upon vaginal examination.
In cases of imperforate septum, the thickness of the septum is measured via MRI.[1 ]
According to Dennie's et al[18 ] experience, the septum has to be removed when the girl reaches the menarche age,
and the operative intervention is easier if the patient presents with hematocolpos
before its drainage. Williams et al[1 ] described 46 patients affected by TVS treated in 3 different ways: abdominoperineal
vaginoplasty via laparotomy, simple excision using the vaginal approach, and laparoscopic
resection of the vaginal septum.
Another surgical approach is the Grünberger method, which consists of a cross-shaped
incision on the caudal part of the septum, a cruciate incision on the cranial part,
and transverse closure.[19 ]
[20 ] Wierrani et al[21 ] described good results in 13 patients treated with Grünberger modification of the
Garcia Z plasty.
Van Bijsterveldt et al[22 ] proposed two novel techniques for the treatment of the vaginal septum: the push
through and pull through techniques. The first one requires a combined abdominal-vaginal
approach, and it is used in patients presenting higher restenosis risk after surgery.
The pull through technique is reserved for patients with a simple vaginal obstruction.[22 ]
A modification of this technique was performed by Layman et al[23 ] with a pull through of a proximal distended vagina using an Olbert balloon catheter
to facilitate the surgical management and to limit the postoperative narrowing of
the vagina.
Vaginal septum may not be the only anatomical defect in a patient, but part of a more
complex syndrome. Tug et al[24 ] reported the case of a young patient affected by obstructed hemivagina and ipsilateral
renal anomaly (OHVIRA) syndrome. In this case, the vaginal septum is completely incised
by CO2 laser with hysteroscopy without hymenotomy, and the distal part of the septum
is simply cut and sutured.[24 ] Tehrani et al,[25 ] also reported a single case of septum removal in a 19-year-old girl by resectoscope
followed by hymenorraphy.
Sardesai et al[2 ] described double cross plasty/Z plasty for the management of TVS after a 20-year
experience as a better technique compared with the other surgical methods.
Several surgical techniques have been described, but Kansagra et al[26 ] proposed an alternative approach. A method of serial balloon dilation over a transvaginally
inserted guide wire to create a durable outflow tract from the uterus to the lower
vagina, without the immediate need of surgery.[26 ]
Postoperative complications, such as vaginal stenosis and reobstruction, can occur,
especially when the septum is thick.[3 ] To decrease the chance of stenosis, vaginal molds can be used; however these require
the cooperation of the young patient, who may not be emotionally mature enough to
use dilators faithfully.[7 ]
In our case, since the patient and her family rejected any vaginal approach to preserve
hymenal integrity, which represents virginity according to the sociocultural values
and traditions of their society, we developed an alternative method that would not
damage the hymen.
The vaginoscopic approach allowed us to preserve the integrity of the hymen. The risk
of stenosis may still be present, but the pressure applied by the balloon of the catheter
on the septal perforation is expected to decrease the closure of the septum. After
12 months of follow-up, there was no septal closure, and the menstrual outflow was
effective. The patient did not complain of other symptoms or of discomfort after 12
months.
Conclusion
In selected cases, with an intact outflow tract, vaginoscopy using a hysteroscope
followed by the insertion of a Foley catheter could represent an effective method
for the resection of vaginal septa, even in the case of virgin patients, because it
is safe, effective, and easy to perform. The vaginoscopic approach should be used
when performing hysteroscopy, but, above all, it should be considered as an alternative
treatment in societies composed of ethnic groups that place importance on the integrity
of the hymen.