Laparoscopic, Endoscopic and Robotic Surgery 2 (2019) 21e24

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Laparoscopic, Endoscopic and Robotic Surgery

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laparoscopic-endoscopic-and-robotic-surgery

Septate uterus with cervical duplication and longitudinal vaginal

septum: A two case series
Noa Ndoua Claude Cyrille a, b, Ayissi Gre 
gory a, *, Mendibi Sandrine b, Belinga Etienne a, b
,
Metogo Junie a, b, Kasia Onana Yves Bertrand a, b, Kasia Jean-Marie a, b
a
Departement of Gynecology and Obstetrics, Faculty of Medicine and Biomedical Science, University of Yaounde I, Yaounde, Cameroon
b
Hospital Center for Research and Application in Endoscopic Surgery and Human Reproduction, Yaounde, Cameroon

a r t i c l e i n f o a b s t r a c t

Article history: Müllerian duct anomalies consist of a set of structural malformations resulting from abnormal devel-
Received 5 September 2018 opment of the paramesonephric or Müllerian ducts. Failures in lateral fusion may result in uterus
Received in revised form didelphys, bicornuate uterus or arcuate uterus while, reabsorption failure results in a uterus with a
2 November 2018
partial or complete septum. We reported a series of two cases of a woman with a septate uterus, cervical
Accepted 12 November 2018
Available online 22 November 2018
duplication and longitudinal vaginal septum undergoing an in vitro fertilization procedure. Septate
uterus with double cervix and longitudinal vaginal septum is a rare anomaly. We reported its endoscopic
management in two infertile women. Hysteroscopic metroplasty which is a safe and simple approach for
Keywords:
Septate uterus
the removal of the uterine septum can improve reproductive outcomes but in vitro fertilization can be an
Cervical duplication alternative approach for these patients.
Metroplasty © 2018 Sir Run Run Shaw Hospital affiliated to Zhejiang University School of Medicine. Published by
Hysteroscopy Elsevier B.V. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/
licenses/by-nc-nd/4.0/).

1. Introduction clinical symptoms depend on the presentation of the anomalies.

The treatment for Müllerian anomalies varies according to the
Müllerian duct anomalies consist of a set of structural malfor- specific type of malformation found in each patient.
mations, resulting from abnormal development of the para- Therefore, we sought to report the management of two women
mesonephric or Müllerian ducts.1 The prevalence of these anomalies with septate uterus, cervical duplication, and longitudinal vaginal
range to 4%e7% in the general population, and even higher in septum undergoing infertility investigations at the Hospital Center
selected populations such as recurrent aborters.2,3 for Research and Application in Endoscopic Surgery and Human
Uterine malformations result from failure in organogenesis or Reproduction (CHRACERH) in Yaounde-Cameroon.
from fusion or reabsorption of the Müllerian ducts.1 Fusion defects
result from incomplete merging of the caudal portion with the 2. Case report
Müllerian ducts (lateral fusion) or incomplete merging of the
structures of the urogenital sinus with the Müllerian tubercle 2.1. Case 1
(vertical fusion). Failures in lateral fusion may result in uterus
didelphys, bicornuate uterus or arcuate uterus. Following caudal A 48 years old female patient, Gravida 6 Para 0060 presented in
fusion of the ducts, the remaining portion of the central septum is the Outpatient Department of Obstetrics and Gynecology at our
reabsorbed. Reabsorption failure results in a uterus with a partial or hospital, with a history of recurrent miscarriage, menorrhagia and
complete septum. cyclic dysmenorrhea since the age of 13. A longitudinal vaginal
Müllerian anomalies are frequently asymptomatic, and are often septum, was first discovered at 27 years old, following the first
missed in routine gynecological examinations. Nevertheless, miscarriage and pelvic ultrasound showed a polymyomatous
didelphys uterus, cervical duplicity and longitudinal vaginal
septum. In 2012 she had an incomplete uterine plasty and myo-
* Corresponding author: Department of Gynecology and Obstetrics, Faculty of
mectomy by laparotomy at the Yaounde Gyneco-Obstetric and
Medicine and Biomedical Sciences, University of Yaounde  I, Yaounde, Cameroon. Pediatric Hospital and she came to consult at CHRACERH for the
gory).
E-mail address: ayissigregory@yahoo.fr (A. Gre desire to have children. The clinical examination of the genitalia

https://doi.org/10.1016/j.lers.2018.11.002
2468-9009/© 2018 Sir Run Run Shaw Hospital affiliated to Zhejiang University School of Medicine. Published by Elsevier B.V. This is an open access article under the CC BY-
NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
22 N.N. Claude Cyrille et al. / Laparoscopic, Endoscopic and Robotic Surgery 2 (2019) 21e24

Fig. 1 Vaginal septum.

Fig. 3 Double cervix.

Fig. 2 Skene cyst. Fig. 4 Uterine septum.

found two vaginal orifices divided by a vertical septum (Fig. 1), a and a notion of unicorn uterus diagnose at a pelvic ultrasound done
cyst of the Skene gland (Fig. 2) at the anterior wall of the vaginal in 2011. She consulted for the desire to conceive and the initial
septum and one cervix in each part of the septum (Fig. 3). A diag- evaluation found that two vaginal orifices were divided by a vertical
nostic hysteroscopy with a saline distention media performed after septum, and one cervix in each part of the septum. The uterus was
the surgical resection of the vaginal septum found two cervical slightly increase clinically. A pelvic ultrasound was done and found
orifices ending in a single uterine cavity, itself divided by longitu- a non-pregnant uterus, 3 small myomas without impact on the
dinal septum interesting the upper third of the cavity. The two ostia cavity, a secretory endometrium of 8e9 mm and normal ovaries.
were visualized in the uterine cavity on both sides of the longitu- The lack of concordance between clinic and pelvic ultrasound
dinal septum, and the left ostium was higher situated than the right motivated a diagnostic laparoscopy associated with hysteroscopy. A
one (Fig. 4). Intraoperative procedures consisted of a cystectomy slightly increased uterus was found at the laparoscopic assessment
and vaginoplasty initially, and the second step consisted of an with a sagittal white band giving the impression of a septate uterus
operative hysteroscopy for the septum resection under laparo- (Fig. 5), ovaries and fallopian tubes were normal. There were no
scopic control at one month intervals. pelvic adhesions. A diagnostic hysteroscopy with a saline distention
media performed after the surgical resection of the vaginal septum
2.2. Case 2 found two cervical orifices (Fig. 6) ending in a single uterine cavity,
itself divided by a longitudinal septum interesting the total cavity.
A 38 years old patient, G0 P0, presented in the Outpatient The two ostia were visualized in the uterine cavity on both sides of
Department of Obstetrics and Gynecology at our hospital with a the longitudinal septum. The indication of a hysteroscopic metro-
history of infertility of male and female origin evolving since 8 plasty with laparoscopic control was made in a second operative
years ago, with a notion severe oligospermia in the husband in 2011 time at one month intervals.
 N.N. Claude Cyrille et al. / Laparoscopic, Endoscopic and Robotic Surgery 2 (2019) 21e24 23

uterus with longitudinal vaginal septum and a complete septum is

defined by the proximity of the septum with the internal orifice of
the cervix.1 Therefore it seems like our patientsinitially had total
failure in resorption between the two müllerian ducts, resulting in
a septate uterus with a longitudinal vaginal septum, and because
the septum was complete and very close with the external orifice of
the cervix, that led to the formation of a double cervix.
The two patients were seeking for children and the first one had
a history of recurrent miscarriage, menorrhagia and cyclic
dysmenorrhea while the second was asymptomatic. Uterine
septum are usually asymptomatic, but they can lead to repeated
miscarriage and cause infertility among women who are affected.1
Fedele et al demonstrated that the abnormal septa endometrium
was responsible of recurrent miscarriages with a reduced number
of glandular ostia, irregular nonciliated cells with rare microvilli,
incomplete ciliogenesis on ciliated cells, and decrease in the cili-
ated/nonciliated cell ratio (1:52 ± 11 versus 1:21 ± 8).7 However
successful pregnancy have been report in women with septate
uterus after in vitro fertilization.8
Laparoscopy combined with hysteroscopy is thought to be the
gold standard in the investigation of women with congenital mal-
formations and especially the uterine ones, which is currently used
Fig. 5 Sagittal white band. in our setting. However, the diagnosis is mainly based on the
subjective impression of the performing clinician, and this is
thought to be a limitation in the objective estimation of the
anomaly.9
The treatment is based on surgical resection of the septum with
or without cervical plasty. Hysteroscopic metroplasty is a simple
approach for the removal of the uterine septum and is performed
worldwide to improve reproductive outcomes.10 Many studies
represented the progression of spontaneous pregnancy rate after
the hysteroscopic removal of septate uterus, such as Esmaeilzadeh
et al who reported that the hysteroscopy septoplasty resulted in an
overall pregnancy rate of 67% and a live birth 57.5% in 106 infertile
women and the pregnancy rate for patients who had not male
infertility was 92.1%.11 However a systematic review performed by
Rikken et al to determine whether hysteroscopic septum resection
in women of reproductive age with a septate uterus improves live
birth rates showed no evidence to support this surgical interven-
tion in these women.12
The hysteroscopic resection of the cervical septum may be
related to cervical incompetence and secondary infertility. How-
ever, a randomized controlled trial performed to evaluate the
Fig. 6 Vaginal septum. safety and efficacy of resection of the cervical septum during
hysteroscopic metroplasty showed that this procedure was safer
and easier with resection than with preservation of the cervical
3. Discussion septum.13 The use of estrogen therapy or an intrauterine device
are postsurgical alternatives for minimizing formation of uterine
Septate uterus with double cervix and longitudinal vaginal adherences which remain the main causes of infertility after the
septum is a rare anomaly.4 Saravelos et al found that the prevalence septum resection.14
of congenital uterine anomalies is approximately 7.3% (95% CI,
6.7%e7.9%) in the infertile population and approximately 16.7%
(95% CI, 14.8%e18.6%) in the recurrent miscarriage population and 4. Conclusion
the septate uterus was the most common anomaly in the infertile
population.5 The exact prevalence of septate uterus with double Septate uterus with double cervix and longitudinal vaginal
cervix and longitudinal vaginal septum remain unknown, but septum is a rare anomaly. We reported its endoscopic management
published reports suggest that the true incidence of the anomaly is in two infertile women in our country at CHRACERH. Hysteroscopic
more common than initially believed.6 metroplasty which is a safe and simple approach for the removal of
In our cases, the first patient, due to the previous partial the uterine septum can improve reproductive outcomes but in vitro
resection, presented a partial uterine septum, a cervical duplication fertilization can be an alternative approach for this patients.
and a longitudinal vaginal septum. And the second one had a total
sagittal septum, cervical duplication and longitudinal vaginal
septum. It is known that the septate uterus develops from a defect References
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