Case Report: Successful Vaginal Delivery of Naturally Conceived Dicavitary Twin in Didelphys Uterus: A Rare Reported Case

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Case Reports in Obstetrics and Gynecology


Volume 2017, Article ID 7279548, 4 pages
https://doi.org/10.1155/2017/7279548

Case Report
Successful Vaginal Delivery of Naturally Conceived Dicavitary
Twin in Didelphys Uterus: A Rare Reported Case

Houda Nasser Al Yaqoubi and Nishat Fatema


Department of Obstetrics and Gynaecology, Ibri Regional Hospital, Ministry of Health, Ibri, Oman

Correspondence should be addressed to Nishat Fatema; [email protected]

Received 2 June 2017; Accepted 30 July 2017; Published 27 August 2017

Academic Editor: Giampiero Capobianco

Copyright © 2017 Houda Nasser Al Yaqoubi and Nishat Fatema. This is an open access article distributed under the Creative
Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the
original work is properly cited.

Didelphys uterus, or double uterus, is an embryological developmental malformation of the müllerian ducts with the incidence
of approximately 8.3% of all müllerian duct abnormalities (MDAs). Didelphys uterus accompanying dicavitary twin gestation is
encountered as a very rare entity with overall incidence of about 1 in 1,000,000. We report a rare case of didelphys uterus, diagnosed
since her first pregnancy, and during her fourth pregnancy she conceived dicavitary twin naturally without any infertility treatment.
Though, the pregnancy course was complicated by preterm labour at 34-week gestation and she delivered simultaneously both
fetuses with the cephalic presentation by spontaneous vaginal delivery with good maternal and neonatal outcomes.

1. Introduction without any fertility treatment has not been researched


extensively in the literature, so the exact rate of occurrence
Didelphys uterus is an embryological developmental mal- is unknown in general population [1, 4].
formation of the müllerian or Wolffian ducts, characterized Dicavitary twin in a double uterus was first described
by complete failure of the müllerian ducts to fuse, resulting in 1927. The overall incidence of dicavitary twin gestation
in two separate uterine cavities and cervices. Sometimes a (ART/Natural conception) in uterus didelphys has been
longitudinal or transverse vaginal septum varying from thin reported approximately 1 in 1,000,000 [1, 3].
and easily displaceable to thick and inelastic may also be Like other MDAs, uterine didelphys is associated with
associated with didelphys uterus [1, 2]. various obstetric complications like spontaneous miscar-
Among müllerian duct anomalies (MDAs), the septate riages, malpresentation, preterm delivery, preterm rupture of
uterus is the most common (35%) followed by bicornuate membrane, intrauterine growth restriction, and the need for
uterus (25%), arcuate uterus (20%), then unicornuate (9.6%), operative delivery [4, 5].
and complete agenesis (3%). The occurrence of didelphys We are discussing an extremely rare case of a naturally
uterus is the second least common with the incidence of conceived dicavitary twin pregnancy in didelphys uterus,
approximately 8.3% of all MDAs. The prevalence of didelphys who had a successful vaginal delivery of both fetuses simulta-
uterus is reported to be 1 in 1000–1 in 30,000 women [1]. neously without any complications at 34 weeks of gestation.
These uterine anomalies are associated with delayed nat-
ural conception and subfertility. In case of infertile women, 2. Case Presentation
uterus didelphys has been found around 0.2%. For such
cases, successful pregnancies can be achieved by artificial A 30-year-old G4P1Ab2 woman was diagnosed by MRI to
reproductive technique and embryo transfer [2, 3]. have uterus didelphys with two cervices and longitudinal
Natural twin conception in each cavity of didelphys upper vaginal septum since her first pregnancy. During her
uterus is a very rare entity and only a handful of cases are fourth pregnancy, she was booked with us at 10 weeks of
reported in the literature to date. Because of its rarity, didel- gestation with a singleton fetus in each cavity (dicavitary
phic uterus accompanying natural dicavitary twin conception twin) of the uterus didelphys.
2 Case Reports in Obstetrics and Gynecology

artery Doppler for both fetuses was normal (Figure 3).


Cardiotocograph tracing for both fetuses was satisfactory.
Two days later she was discharged and plan of delivery was
discussed with the couple in detail; if both babies remained
cephalic and no complications arise, she will be allowed for
the trial of vaginal delivery. If there was malpresentation or
any element of fetal distress of any one of the fetuses, then the
cesarean section would be considered for both fetuses. She
and her husband agreed with our plan.
One week after discharge from the hospital, at 34 + 3
weeks of gestation, she was presented with preterm labour.
On physical examination, she was vitally stable and was
getting the strong uterine contraction and vaginal exami-
nation revealed that she was in the second stage of labour.
Immediately after admission, the first twin was delivered and
after delivery of first twin amniotomy was done for the second
twin. Around 11 minutes after the delivery of first twin, the
Figure 1: Thick septum separating the uterine cavities showing in second twin was delivered from the other uterine cavity. Both
2D and 3D ultrasound mode. The arrows showed thick septum placentas were removed smoothly from the separate uterine
separating the uterine cavities showing in 2D and 3d ultrasound cavities. The neonatal outcomes were good with Apgar scores
mode. for both babies of 9 in 1 minute and 10 in 5 minutes. The first
baby was a male with weight 1.6 kg, and the second baby was
female with weight 2 kg. Neonates were kept in the neonatal
Her past obstetric history revealed that she had two intensive care unit for preterm care and observation. Neither
first trimester miscarriages followed by IUI (intrauterine the patient nor the neonates have experienced any other
insemination) conception in her third pregnancy. In her third complications. Patient’s postnatal course was uneventful and
pregnancy, induction of labour (IOL) was done at 36 weeks of on the second postnatal day she was discharged in good
gestation in view of intrauterine growth restriction (IUGR). condition with her healthy babies.
Following IOL, she delivered vaginally an alive preterm baby
with the weight of 2 kg. Her medical history is significant for
hypothyroidism and she is on Tab. Thyroxin 25 microgram
3. Discussion
daily. We presented a case of known uterus didelphys with naturally
During her current pregnancy, she conceived naturally conceived dicavitary twin pregnancy which is an extremely
without any fertility treatment. After booking, she was rare occurrence.
referred for routine antenatal care at Maternal-Fetal Medicine
The failure of fusion of the müllerian ducts results
(MFM) clinic due to twin gestation and uterine anomaly and
in uterus didelphys. It is a developmental abnormality of
for the risks associated with it. On subsequent follow-up in
müllerian ducts comprised the double uterus with completely
MFM clinic, she was diagnosed as dicavitary twin pregnancy
developed independent horns including endometrium,
in didelphys uterus by USG (Figure 1). Her blood group was
myometrium, and serosal layers; two cervices; and lon-
AB +ve and booking hemoglobin was 11.7 gm/dl.
gitudinal or transverse vaginal septum (Figure 2(a)). The
She was started on low dose aspirin (75 mg) in view of the
etiology of uterus didelphys is not known exactly with the
previous history of IUGR baby. She followed up biweekly at
frequency from 1 in 1000 to 1 in 30,000 women [4, 5]. Didel-
the MFM clinic. Detailed anatomical analysis of both fetuses
phys uterus accompanying naturally conceived twin preg-
was done by ultrasound during second trimester. The analysis
nancy with each fetus in the separate cavity (Figure 2(b)) is a
revealed normal amniotic fluid and anteriorly placed placenta
rare entity. Only a few number of cases have been reported,
for both fetuses. The length of both cervices was within
but no large series exist in the literature [1, 6].
normal limit.
During 33 + 3 weeks of gestation at the time of her To the best of our knowledge, not more than 20 cases of
routine antenatal follow-up, she complained of intermittent dicavitary twin or multiple gestation in didelphys uterus have
premature contraction, so vaginal examination was per- been researched to date [6, 7].
formed which revealed left cervix 1.5 cm long os 2 cm dilated The exact incidence of the condition is unknown. The
membrane intact and station at −3 and the other cervix was overall incidence of dicavitary twin gestation conceived either
closed. spontaneous or by the artificial reproductive technique is esti-
She was admitted for observation; two doses of dexam- mated approximately 1 in 1,000,000 [1, 6]. It is hypothesized
ethasone (12 mg) were given 12 hours apart for fetal lung that these twins are biovular in all cases, where the two ova
maturation. Fetal growth for both fetuses by USG: first fetus might come from the two follicles of the same ovary or in both
(right uterus) was with estimated weight 1.7 kg and normal ovaries ovulation may occur during the same cycle [8].
amniotic fluid, and the second fetus (left uterus) estimated Our patient was diagnosed to have didelphys uterus
weight was 1.9 kg and normal amniotic fluid. Umbilical since her first pregnancy and conceived dicavitary twin,
Case Reports in Obstetrics and Gynecology 3

(a) (b)

Figure 2: (a) Schematic diagram of nongravid didelphys uterus and (b) schematic diagram of gravid didelphys uterus with dicavitary twin.

Figure 3: Umbilical artery Doppler for both fetuses at 33 weeks of gestation.

naturally without any infertility treatment. Unfortunately, the a 66 days’ interval at 35 weeks of gestation. They described
pregnancy course was complicated by preterm labour at 34 that in didelphys uterus as the uterine horns are individually
weeks of gestation and she delivered simultaneously both functioning so the initiation of labour could be local rather
fetuses with the cephalic presentation by spontaneous vaginal than systemic control [7].
delivery. The time interval between deliveries of both fetuses Maki et al. described another case of dicavitary twins in
was only 11 minutes. didelphys uterus that were conceived after fertility treatment,
Similar to our case, Allegrezza reported a case of natural where at 37 weeks of gestation the woman had preterm
dicavitary twin pregnancy in didelphys uterus, in which the premature rupture of the membrane of right horn of uterus
patient had premature rupture of membrane followed by followed by progression of labour with simultaneous contrac-
preterm labour at 31 weeks of gestation, both fetuses were tions of both horns of the uterus. The fetus in the right horn
cephalic and delivered vaginally without any complications was delivered by spontaneous vaginal delivery and the second
[4]. twin was delivered by cesarean section in view of abnormal
The contractions of both uteri may not begin simultane- cardiotocograph (CTG). They analyzed the synchronized
ously. There are reported cases where the delivery interval contractions of both horns of the didelphys uterus and
between the twins varies from several hours or even several commented that the primary uterine contractions are caused
weeks [8]. by the individual rhythms of the bilateral pacemaker sites
One case is reported by Nohara et al. in which one surrounding the uterotubal junction and subsequently the
twin was delivered by cesarean section at 25 weeks of help of the gap junctions in between both uterine sides
gestation due to fetal distress followed by premature rupture resulted in synchronized uterine contractions to expel the
of membrane and another one was delivered vaginally with uterine contents [6].
4 Case Reports in Obstetrics and Gynecology

In our case, luckily the CTG tracing of both fetuses Conflicts of Interest
was reactive, and both were delivered vaginally without any
difficulty within an 11-minute time interval. The authors declare that they have no conflicts of interest.
Only a few cases of twin gestation with didelphys uterus
that had spontaneous vaginal delivery are mentioned in the Acknowledgments
literature [1, 8].
Didelphys uterus is associated with varieties of obstetric The authors would like to thank Dr. Tanima Roy (Bangladesh)
complications including early and late miscarriages, malpre- for her great effort and support by drawing the schematic
sentation, intrauterine growth restriction, preterm delivery, diagrams (Figure 2).
and preterm rupture of membrane [4, 5, 9].
Cervical incompetence is not commonly occurred with References
didelphys uterus so cervical cerclage is not routinely recom-
mended unless there is an evidence of cervical incompetence [1] O. Ozyuncu, M. Turgal, A. Yazicioglu, and A. Ozek, “Sponta-
or dilation either by clinical examination or ultrasonography neous twin gestation in each horn of uterus didelphys compli-
during early second trimester. A case of didelphys uterus with cated with unilateral preterm labor,” Case Reports in Perinatal
dicavitary twin was reported with the short cervix at 30 weeks Medicine, vol. 3, no. 1, 2014.
of gestation with uterine contractions. They managed the case [2] S. Rezai, P. Bisram, I. Lora Alcantara, R. Upadhyay, C. Lara, and
by tocolytic therapy with nifedipine until 34 weeks and then M. Elmadjian, “Didelphys uterus: a case report and review of
at 37 weeks of gestation cesarean section was done for both the literature,” Case Reports in Obstetrics and Gynecology, vol.
fetuses due to fetal distress of one twin. They did not observe 2015, Article ID 865821, 5 pages, 2015.
any adverse effects of tocolytic therapy [1, 2]. [3] M. Yang, J. Tseng, C. Chen, and H. Li, “Delivery of double
The overall obstetric outcome of uterus didelphys is poor singleton pregnancies in a woman with a double uterus, double
cervix, and complete septate vagina,” Journal of the Chinese
but still better than the other MDAs like the septate or
Medical Association, vol. 78, no. 12, pp. 746–748, 2015.
bicornuate uterus. The reason behind this occurs is that
[4] D. M. Allegrezza, “Uterus didelphys and dicavitary twin preg-
in didelphys uterus the blood supply through the collateral
nancy,” Journal of Diagnostic Medical Sonography, vol. 23, no. 5,
circulation in between two horns is better in comparison to
pp. 286–289, 2016.
other MDAs. The successful pregnancy rate with didelphys
[5] C. Magudapathi, “Uterus didelphys with longitudinal vaginal
uterus is 57%, and the fetal survival rate is documented
septum: normal deliver—case report,” Journal of Clinical Case
around 64% [2, 4, 10]. Reports, vol. 2, article 13, 2012.
In these cases, no specific route of termination of preg-
[6] Y. Maki, S. Furukawa, H. Sameshima, and T. Ikenoue, “Inde-
nancy is recommended in the literature, though both vaginal pendent uterine contractions in simultaneous twin pregnancy
and cesarean delivery have been mentioned in the previous in each horn of the uterus didelphys,” Journal of Obstetrics and
studies. The incidence of cesarean section is documented Gynaecology Research, vol. 40, no. 3, pp. 836–839, 2014.
about 82%. If both fetal presentations are cephalic and there [7] M. Nohara, M. Nakayama, H. Masamoto, K. Nakazato, K.
are no other associated risk factors, then vaginal delivery can Sakumoto, and K. Kanazawa, “Twin pregnancy in each half of a
be considered as the mode of delivery [2, 5, 10]. uterus didelphys with a delivery interval of 66 days,” BJOG: An
If the cesarean section is indicated then a low midline International Journal of Obstetrics & Gynaecology, vol. 110, no.
longitudinal incision is preferable for proper exposure of both 3, pp. 331-332, 2003.
uterine cavity to facilitate the delivery of the fetuses [3]. [8] R. Kekkonen, M. Nuutila, and T. Laatikainen, “Twin pregnancy
with a fetus in each half of a uterus didelphys,” Acta Obstetricia
4. Conclusion et Gynecologica Scandinavica, vol. 70, no. 4-5, pp. 373-374, 1991.
[9] J. R. Jackson, B. Williams, and J. Thorp, “Spontaneous triplets
Most of the previous studies regarding didelphys uterus carried in a uterus didelphys,” Case Reports in Women’s Health,
with twin gestation had the history of fertility treatment, vol. 3-4, pp. 1-2, 2014.
and the termination of pregnancy was required by cesarean [10] S. Bhattacharya and P. K. Mistri, Twin Pregnancy in a Woman
section either due to fetal malpresentation or fetal distress. with Uterus Didelphys, 2011, http://cogprints.org/7271/.
Our presented case is the dicavitary twin with didelphys
uterus, which was conceived naturally, and although the
pregnancy was complicated by preterm labour, both fetuses
were delivered vaginally with good maternal and neonatal
outcomes.
Didelphys uterus is associated with a twin pregnancy is a
high-risk pregnancy. The early detection of this anomaly of
the uterus and accompanying pregnancy by ultrasonography
is of great value. Close monitoring of fetal growth, biophysical
profile, and the cervical condition is recommended through-
out the pregnancy. The time and mode of delivery should
be planned and discussed in detail with the couple during
antenatal follow-up [4, 5, 8].
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