A 29-year-old Para 2 was admitted to the emergency department with increasing lower abdominal pain. The patient had undergone an uncomplicated elective repeat caesarean section 7 days before being admitted to the emergency department. An emergency laparotomy revealed a uterus didelphys with a torsion of one of the uteri

Elisabeth D Van Oudgaarden

Lucio M A Cipullo

Slobodan Milosavljevic

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Case Reports in Obstetrics and Gynecology
Volume 2012, Article ID 190167, 3 pages
doi:10.1155/2012/190167
Case Report
Uterus Didelphys: Report of a Puerperal Torsion and
a Review of the Literature
Lucio M. A. Cipullo, Slobodan Milosavljevic, and Elisabeth D. van Oudgaarden
Department of Obstetrics and Gynaecology, Slotervaart Medisch Centrum, Louwesweg 6, 1066 EC Amsterdam, The Netherlands
Correspondence should be addressed to Lucio M. A. Cipullo, luciocipullo@hotmail.com
Received 25 April 2012; Accepted 2 July 2012
Academic Editors: M. G. Porpora and Y. Purwosunu
Copyright © 2012 Lucio M. A. Cipullo et al. 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.
A 29-year-old Para 2 was admitted to the emergency department with increasing lower abdominal pain. The patient had undergone
an uncomplicated elective repeat caesarean section 7 days before being admitted to the emergency department. An emergency
laparotomy revealed a uterus didelphys with a torsion of one of the uteri.
1. Introduction
Uterine torsion is the twist of the uterus between the
cervix and uterine body. A minor degree of rotation of the
pregnant uterus is fairly common during the third trimester
of pregnancy but is deemed rather negligible. In contrast,
an axial rotation of more than 45 degrees is quite unusual
and its occurrence is defined as uterine torsion. The unusual
occurrence of this latter condition during the puerperal
period is often associated with predisposing factors altering
the shape or position of the uterus or adnexa.
A severe torsion of the uterus can cause irreversible
ischemic injury or serious thrombotic accidents. Hence, it is
crucial to diagnose this condition quickly.
In this paper we describe a case of a uterine torsion in a
woman carrying a Müllerian anomaly (uterus didelphys). The
torsion involved one of the uteri and occurred on the seventh
day after an uncomplicated caesarean section for which it was
necessary to perform a surgical reintervention.
2. Case Presentation
A 29-year-old woman Para 2 was admitted to the emergency
department with increasing lower abdominal pain. The
patient had no history of systemic disease or chronic
abdominal tenderness. Her past obstetric history revealed
a caesarean section performed at 38 weeks because of a
breech presentation three years earlier.
The patient had undergone an uncomplicated elective
repeat caesarean section 7 days before being admitted to the
emergency department. Indication for CS was breech pre-
sentation at term. The operation was without complications
except some heavy bleeding occurred after the incision of
the lower uterine segment; the bleeding was controlled by
means of haemostatic stitches. The postoperative course was
uneventful and the patient was discharged in good clinical
conditions four days after CS.
Three days after CS she reported a significant decrease of
postpartum discharge (lochia) as well as a sudden complete
stop of vaginal bleeding and discharge on day 4 after SC.
On admission, patient was alert, conscious, and well-
oriented. She complained of severe low abdominal pain.
Blood pressure was 140/80 mm Hg, HR 105 b/min, and temp.
37.4. The pain started the day before the admission as an
abdominal discomfort with a sudden increase during the
following day. At the time of her admission the patient
described a constant low abdominal/pelvic pain with colicky
exacerbations. Examination at this stage found no vaginal
bleeding. Palpation revealed a painful swelling on the right
side just above the pelvis. There was no sign of peritoneal
irritation or abdominal distension. The wound seemed to be
healing well. Lab tests at admission: Hb 12,8 g/dL, Hct 35,
PLT 280, and WBC 10, 5× 109/L.
2 Case Reports in Obstetrics and Gynecology
Routine urine examination was normal. Ultrasound
investigation showed a strongly involuted uterus (LD 7.5 cm,
TD 4.2 cm, APD 3.7 cm) which was localized centrally in the
pelvis and also an oval-shaped mass on the right side of the
lower abdomen (25 cm × 8 cm), showing thickened walls
with complex internal echo patterns apparently suggesting
a organized haematoma. In the pouch of Douglas, there
were no signs of active bleeding or clots. Despite of the
administration of meperidine hydrochloride 100 mg IM, a
reassessment after two hours showed increased symptoms
with additional signs of peritoneal irritation and restlessness
whereupon an explorative laparotomy was decided.
Access was gained through the previous Pfannenstiel
incision. At the opening of the abdomen a large amount
of coagulated, dark red blood was evident amongst the
intestinal loops. A further exploration of the pelvis showed
an involuted uterus without signs of recent hysterotomy. On
the right side, at the level of right iliac fossa, there was an oval,
reddish, soft structure with a maximum diameter 25 cm.
Mobilisation and further inspection revealed an enlarged
uterus situated on the right side in respect to the small one
(uterus didelphys). This second uterus showed a torsion of
90◦ on its axis and had a congested appearance.
The uterine cavity was filled with blood (lochia) coming
out the tubae by squeezing of the uterus. There were no signs
of uterine weakening. During the inspection before detorsion
it was noted a transverse hysterotomy suture on the right
side of the uterus which was not bleeding. The other smaller
uterus appeared firmly fixed to the anterior pelvic fascia and
the lower segment of the left anterolateral wall of the other
uterus (Figure 1). Both adnexa were normal at inspection.
All adhesions needed to be removed before attempting a
complete emptying and detorsion of the organ (Figure 2).
Postoperative recovery was uneventful and the patient could
be discharged 5 days after the operation in good clinical
condition. She got prescribed enoxaparin 20 mg per day for
6 weeks to prevent thromboembolism.
3. Discussion
Uterine torsion is the rotation of the uterus at the level of
the lower uterine segment of more than 45 degrees along
its axis. Dextrorotation occurs in two-thirds of the cases and
laevorotation is found in the other one-third [1].
Labbe published the first case of uterine torsion in
1876 [2]. There have been very few cases since this first
publication, all nearly exclusively regarding torsion of the
uterus occurring during pregnancy. The torsion of the uterus
during the puerperal period is even rarer with only three
cases reported in literature to date.
The risk factors for uterine torsion during the puer-
perium include fixation of the uterus by adhesions, ovar-
ian tumor, uterine myomas, large neoplasms, and uterine
Müllerian anomalies. According to some authors intrinsic
intrapelvic pathology is responsible for 66% of the cases
during pregnancy [3]. A study conducted with MRI evalu-
ation of patients following low transverse caesarean section
suggested that, occasionally, poor healing of the hysterotomic
Figure 1: Uterus before detorsion.
Figure 2: Uterus after detorsion.
scar may result in suboptimal restoration of normal cervical
length and strength predisposing the uterus to torsion in
those cases [4]. In the case presented here, the association of
the two factors that were described determined the torsion:
uterus didelphys together with iatrogenic adhesion between
one of the uteri and the pelvic wall. The clinical presentation
of puerperal uterine torsion is nonspecific and may differ
from the symptoms of torsion in pregnancy. Symptoms
at presentation could suggest an adnexal torsion or other
colicky abdominal pain. The most common symptom is
abdominal pain varying from mild abdominal tenderness
through to symptoms of an acute abdomen making diagnosis
difficult especially in the absence of MRI imaging. The
sudden cessation of the physiological vaginal discharge
associated with abdominal pain in puerperal period is
strongly suggestive of uterine torsion. In about 11 percent
of cases torsion is asymptomatic [5]. In case of torsion,
a prompt diagnosis is crucial to start surgery as soon as
possible to avoid the risk of ischemic and thromboembolic
complications. Ultrasound is not specific for this kind
of diagnosis. In some cases, if previous ultrasound scans
revealed fibroids that have changed position, a torsion of a
myomatous uterus may be suspected. Use of CT scan or MRI
is a valid diagnostic tool when available in an emergency
situation. MRI provides an accurate evaluation. The wall of
the upper vagina changes from normal H configuration to an
x-shaped configuration in uterine torsion [6, 7].
In the case discussed here, the differential diagnosis
was made between an adnexal tumor, a partially organised
bleeding or a retroperitoneal mass. Existence of a Müllerian
anomaly was ignored. Only the direct examination revealed
the presence of a double-uterus of large proportions twisted
on its axis but also rotated behind the small uterus. Most
likely, the first caesarean section had been carried out in
the left uterus causing adhesions between the left side of
Case Reports in Obstetrics and Gynecology 3
the uterus and the anterior pelvic wall. Both the anomalous
position of the uterus due to the firm adherences and the
Müllerian anomaly increased the risk of torsion. During
inspection of the rotated uterus, the hysterotomy commonly
performed in a different area, appeared to be positioned
rather abnormally on the right side of the organ. This impor-
tant detail suggested that the uterus had undergone the
torsion after caesarean section had been performed. The
subsequent torsion determined both the abrupt cessation
of the postpartum discharge of haematic lochia and over
distension of the uterine body. Rotation of the uterus on
its axis had partially involved the uterine vessels causing
a general congestion of the organ. Accurate exploration of
uterus excluded the presence of any irreversible ischemic
damage, a condition which would require a hysterectomy.
It is, however, very difficult to determine whether the
ischemic injury affecting the uterus is reversible or not,
because puerperal torsion is a rare pathological condition.
Hysterectomy needs to be considered for women who
have fulfilled reproductive wishes with uterine necrosis
resulting from prolonged torsion [8]. Bilateral plication of
the round ligaments can be done to prevent immediate
postpartum recurrence of uterus torsion [9]. This may help
to keep uterus in its natural position and reduce the effect of
iatrogenic uterine adhesion. Bilateral plication of uterosacral
ligaments is also described, which may provide resistance to
torsion and prevent long-term recurrence of this condition
[10].
Conflict of Interests
The authors declare no conflict of interests.
Consent
Patient has given her consent for the case report to be
published.
References
[1] H. R. K. Barber and E. A. Graber, “Uterine Torsion during
pregnancy,” in Surgical Disease in Pregnancy, pp. 387–388, WB
Saunders, Philadelphia, Pa, USA, 1974.
[2] D. Jovanovic, A. Del Granado, and A. Stiller, “Torsion of the
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[3] A. L. Robinson and H. M. Duvall, “Torsion of the pregnant
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[4] S. Kawakami, K. Togashi, T. Sagoh et al., “Uterine deformity
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[8] A. Dua, K. Fishwick, and B. Deverashetty, “Uterine torsion in
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[9] M. A. Pelosi and M. A. Pelosi, “Managing extreme uterine
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[10] M. S. Mustafa, F. Shakeel, and B. Sporrong, “Extreme torsion
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Case Report Uterus Didelphys: Report of a Puerperal Torsion and a Review of the Literature

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Case Report Uterus Didelphys: Report of a Puerperal Torsion and a Review of the Literature

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