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Special Report

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Postpartum hemorrhage
caused by uterine artery
pseudoaneurysm and uterine
rupture after vaginal delivery
Expert Rev. Obstet. Gynecol. 7(4), 313319 (2012)

Massimo Origoni*, A case of massive hemoperitoneum determined by uterine rupture and uterine artery
Chiara Gelardi, pseudoaneurysm dissection after vaginal delivery, requiring an emergency laparotomy with
Federica Pasi, arterial ligation, is reported. The authors reviewed the published literature, finding 12cases of
uterine artery pseudoaneurysm and associated postpartum hemorrhage after vaginal delivery,
StefanoSalvatore and
but no cases with uterine rupture have ever been published. Diagnosis is based upon imaging,
Massimo Candiani and management options include arterial embolization, arterial ligation and hysterectomy in
Department of Gynecology and particularly severe and life-threatening situations. Vaginal bleeding is associated with favorable
Obstetrics, Vita Salute San Raffaele
outcomes and conservative treatment, while the absence of external blood loss seems to be
University School of Medicine,
SanRaffaele Scientific Institute, Via correlated with a worsened prognosis and the need for an invasive treatment option. Uterine
Olgettina 58, 20132 Milano, Italy artery pseudoaneurysm rupture after vaginal delivery is a very rare condition of life-threatening
*Author for correspondence: postpartum hemorrhage, being more frequently associated with cesarean section. Uterine artery
Tel.: +39 2 2643 2653
Fax: +39 2 2643 2759
pseudoaneurysm dissection should be suspected in any patient presenting with early or delayed
massimo.origoni@hsr.it postpartum hemorrhage after a vaginal delivery and can be associated with uterine rupture.

Keywords : postpartum hemorrhage PPH uterine pseudoaneurysm uterine rupture

Postpartum hemorrhage (PPH) is defined as the available literature on similar clinical expe-
a blood loss >500 ml after vaginal delivery riences to draw final considerations on the
or >1000 ml after cesarean delivery [1] . PPH management of such a rare event.
occurs in fewer than 5% of all deliveries and is
one of the major causes of maternal mortality Case report
worldwide, accounting for 15% of all maternal A 34-year-old woman, gravida1 para1, on day5
deaths and for a casefatality rate of 1% [2] . postpartum, presented with diffuse abdominal
Early PPH occurs within 24h of delivery, while pain irradiated to the lower quadrants, dysuria
late PPH occurs from 24 h to 6 weeks post- and urinary frequency persisting for several
partum. Primary causes of PPH include uter- days. The patient had been discharged after
ine atony (70% of cases), followed by retained an uncomplicated vaginal delivery of a single
placental/membrane fragments, lower genital healthy fetus 5 days earlier in good general
tract lacerations, uterine rupture and hereditary condition. The use of a single fundal pressure
coagulopathies [1,2] . (Kristellers maneuver) during the second stage
Uterine artery pseudoaneurysm rupture is a rare of labor was recorded and postpartum recov-
cause of PPH after uterine surgical procedures, ery had been reported as normal in her hospital
such as cesarean section or dilation and curettage record. No use of oxytocic agents was reported.
in the great majority of cases [3,4] . This event is Aside from the symptoms referred, she was
almost exceptional following a vaginal delivery [3] . healthy and took no medication. Personal history
We report a case of delayed intraperitoneal was negative and no other significant element
PPH after vaginal delivery secondary to subclini- was reported; in particular, no g ynecological
cal uterine rupture and uterine artery pseudoa- surgical procedures emerged.
neurysm, describing the diagnostic process, the Vital signs on admission body tempera-
management and treatment. We also reviewed ture, cardiac frequency, arterial pressure and

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Special Report Origoni, Gelardi, Pasi, Salvatore & Candiani

oxygen saturation were all normal. A physical examination right cornual region to the uterosacral ligament was recognized.
revealed regular heart sounds and clear lung fields; the abdo- After removal of the hematoma, a complete dissection of the
men was tender with doubtful peritoneal defense reaction at deep ascending branch of the uterine artery very close to its origin
palpation (Blumbergs sign). No cervical or vaginal lacerations from the main uterine branch was apparent; the artery dissec-
were seen after speculum introduction and accurate inspection. tion was adjacent to the uterine rupture but outside the linear
Pelvic examination revealed a normal uterus size and a closed scar of the uterine wall. Upon visual inspection of the uterine
cervix, with regular serous lochia. No adnexal mass was palpated. artery, the pseudoaneurysm could not be recognized, suggest-
Transvaginal ultrasonography demonstrated a thin and regular ing that the arterial dissection occurred in the pseudoaneurysm
endometrial layer, the absence of retained fragments or any patho- site of the uterine artery. This aspect supports the hypothesis of
logical findings in the uterine cavity; a minimal fluid pouring the origin of the bleeding being exactly at the pseudoaneurysm
was documented bilaterally in the paraovarian regions (Figure 1) . site. A full-thickness single-stich Vycril suture was placed on
Laboratory tests revealed anemia (hemoglobin: 8.3 g/dl; the posterior uterine wall and a right uterine artery ligation to
hematocrit: 24.3%), but no other hematological, enzymatic or control the bleeding was performed. Procedure-related blood
inflammatory alterations. loss was 1000ml, while the estimated hemoperitoneum blood
A total of 3h later, still in the emergengy unit under observa- loss was 4000ml; for this reason the patient received both red
tion, the patient complained of dizziness, weakness and worsening cells and plasma transfusions intraoperatively.
of abdominal pain, and vital signs showed worsening: tachycardia In order to exclude further bleeding, an internal iliac artery
and hypotension were recorded. An ECG and an arterial blood selective computed angiography was performed at the end of the
count were immediately obtained, detecting a hemoglobin level surgical procedure, showing marked hypertrophy of the arterial
of 7.4g/dl. Two peripheral intravenous catheters were placed and vessels of the right uterine side (Figure 4) ; owing to the absence of
500ml of crystalloid infusion was started. A subsequent con- active bleeding no selective embolization was performed.
trasted CT scan revealed a 9-mm pseudoaneurysmatic dilation The patient was postoperatively followed up with blood counts,
of the right uterine artery distal portion associated with active coagulation analysis and pelvic sonography until day 5 post-
bleeding of the same vessel into the peritoneal cavity, indicating laparotomy, when she was discharged from hospital in a good
a significant hemoperitoneum (Figures2&3) . general condition.
Owing to the worsening anemia, symptoms and CT imag-
ing, the patient underwent an emergency explorative lapa- Discussion
rotomy. After abdominal wall incision, the peritoneal cavity Pseudoaneurysm of the uterine artery is a rare cause of PPH and is
appeared to be filled up with a massive hemoperitoneum and defined as a dilation of the artery that differs from true aneurysm
large blood clots, and, at the basis of the right cardinal liga-because of the presence of a single loose connective tissue layer,
ment, a 10-cm hematoma was seen. Massive active bleeding while true aneurysms have a three-layer wall [1] . It is usually sec-
was observed originating from the posterior-lateral right uterine
ondary to a vascular trauma, which alters the arterial wall layers,
wall, where an 8-cm linear uterine rupture extending from the resulting in a reduction of resistance to shear stress forces and
predisposing to vessel rupture. This risk is
increased by the development of a turbulent
flow, causing the false aneurysm to enlarge,
with the consequence that the artery wall
becomes thinner and weaker.
Doppler sonography is a rapid and nonin-
vasive tool that has proven to be very useful
for the diagnosis of these conditions with a
high sensitivity rate (95%) [14] . Upon sono-
graphic evaluation pseudoaneurysms are
often visualized as anechoic or hypoechoic
vascular enlargements, and color Doppler
flowmetry may show the yin and yang sign
in the body and the to-and-fro pattern in
the neck of the pseudoaneurysm, indicating
turbulent arterial flow [5] .
CT scan and MRI can confirm the diag-
nosis and rule out other more common
causes of delayed PPH; selective angiog-
raphy remains the d iagnostic gold stan-
Figure 1. Ultrasound scan before acute onset. Arrow indicates small pouring in dard and may provide definitive therapy
thepelvis. indications.

314 Expert Rev. Obstet. Gynecol. 7(4), (2012)


Uterine artery pseudoaneurysm & postpartum hemorrhage Special Report

In the past, and primarily in emergency


situations, the standard treatment for PPH
secondary to uterine artery pseudoaneu-
rysm rupture was postpartum hysterec-
tomy, with or without hypogastric artery
ligation [6,7] . In recent years, selective uter-
ine artery embolization has become the
accepted treatment of choice [8,9] .
Embolization was first performed in 1979
in a case of PPH [10] , and actually repre-
sents a safe and successful fertility-sparing
technique for the management of pseudo
aneurysmatic lesions correlated with deliv-
ery. Approximately 200 cases of selective
arterial embolization for the treatment of
severe PPH have been described to date,
with an overall success rate of 85100% [11] .
Recently, Kovo et al. described an
alternative treatment option using direct
thrombin injection into the pseudoaneu-
rysm under sonographic guidance [12] . It
consists of thrombin injection into the
pelvic pseudoaneurysm via a percutaneous
or transvaginal approach. Thrombin acts
by converting inactive fibrinogen to fibrin,
Figure 2. Preoperative CT scan. (A) Uterus; (B) right-side hematoma.
which subsequently promotes thrombus
formation. In 6580% of cases of femoral artery pseudoaneu- as uncomplicated vaginal delivery presented aspects that may
rysm, a single injection of 100300U of thrombin is required change their definition: in the paper by Marnela etal., retention
for thrombus formation and bleeding control. A second, third of membranes was described, thus a uterine curettage is likely to
or fourth injection following the initial
treatment session has been reported to be
required in 15, 5 and 1% of these cases,
respectively, with an overall success rate of
91100%.
We performed a Medline search in
English and French for the terms uterine
pseudoaneurysm, postpartum hemor-
rhage and PPH, and, to our knowledge,
12 cases of PPH following vaginal deliv-
ery and determined by pseudoaneurysm
rupture have been reported so far (Table 1) .
In terms of PPH onset, almost half of
the reported cases showed an early mani-
festation. This was observed in six out
of 13 cases including the present case
(46.15%), while the remaining seven
cases (53.84%) had a clinical onset later
in the puerperium, ranging from 5days to
8weeks after delivery.
Delivery was an uncomplicated vaginal
delivery in nine cases (69.23%), while in
four cases (30.76%) an operative deliv-
ery was performed: three cases of forceps
and one of vacuum extractor application.
Of note, two of the nine cases reported Figure 3. Preoperative CT scan. Arrow indicates right uterine artery pseudoaneurysm.

www.expert-reviews.com 315
Special Report Origoni, Gelardi, Pasi, Salvatore & Candiani

bleeding (15.38%) both required laparotomy and uterine artery


ligation, and one case also required hysterectomy.
The unique aspect of our case relates to the coexistence of the
complete uterine artery dissection and uterine rupture; no other
case with this clinical feature is reported in the literature. To state
that vessel rupture preceded and caused uterine rupture or vice
versa remains an open question. In terms of pathophysiology, it
is reasonable to hypothesize that the pseudoaneurysm rupture
could have been the first step towards the final condition of mas-
sive hemoperitoneum; in fact, owing to the vessel wall weakness,
rupture may occur after minor injuries or even without any acute
event (e.g., an arterial pressure increase). By contrast, uterine
rupture would require strong and heavy forces to occur; in our
case, the uterine artery dissection could have determined a type
of uterine hemorrhagic infarction, predisposing the lower uterine
segment to full-thickness rupture. We speculate that the fundal
pressure (Kristellers maneuver) could have acted as a secondary
causal factor for traumatic injury to the uterine lower segment,
contributing to both final complications. In terms of future
pregnancies, we believe that this patient could safely afford a
spontaneous labor and a new vaginal delivery.
In conclusion, uterine artery pseudoaneurysm rupture after
vaginal delivery is a very rare condition of life-threatening PPH.
A traumatic delivery-correlated origin may be recognized, mainly
forceps/vacuum extractor application or uterine cavity curettage
for retained placental/membrane fragments, but an uncompli-
cated vaginal delivery cannot exclude it. In our case, a uterine
Figure 4. Postoperative angiography. Arrow indicates
fundal pressure in the second stage of labor was the only operative
right-side pelvic vessels. procedure performed and, in this case, may have represented a risk
factor for injury to the lower uterine segment and may be advo-
have been performed [5] . In our case, a single Kristellers maneuver cated as a cofactor together with the vascular trauma; consistent
was performed during the second stage of labor; considering these with this interpretation, several reports correlate this common
two cases as complicated vaginal deliveries, the rates change as procedure with maternal complications [1517] . Despite the absence
follows: 53.84% for uncomplicated and 46.15% for operative of specific signs and symptoms, it should be suspected any time
deliveries. patients present in the postpartum with massive vaginal bleeding,
The obstetric history of the patients revealed a minority four uterine atony, acute abdominal pain and hemoperitoneum, with or
out of 13 cases (30.76%) of dystocic or traumatic deliver- without hemodynamic instability. In any case, aggressive resuscita-
ies, while the great majority nine out of 13 cases (69.23%) tion is required. If vaginal bleeding is the only sign and imaging
reported a previous uncomplicated vaginal delivery. (ultrasonography, CT scanning or angiography) is suggestive, uter-
Eleven out of the 13 cases (84.61%) presented significative ine arterial embolization represents the treatment of choice with
postpartum vaginal bleeding, in four cases (30.76%) associated favorable outcomes. In case of hemoperitoneum and/or hemo
with uterine atony. The remaining two cases (15.38%) did not dynamic instability, an emergency surgical procedure is required
show vaginal bleeding but had the worst outcomes of the series: and diagnosis is always confirmed at laparotomy. When feasible
the case of Chung Fat etal. was characterized by maternal death in terms of time and patients conditions, a preoperative CT scan
and our case by massive hemoperitoneum needing emergency or MRI can be discriminant. In these latter events, uterine artery
laparotomy [13] . ligation is reported to be necessary and effective, after accurate
As for the laterality of the pseudoaneurysm, no difference arose inspection of the uterus and ligaments, in order to exclude inju-
in the reported cases, as six (46,15%) had a right uterine ori- ries to these structures. In particularly severe and life-threatening
gin, five (38.46%) a left origin and one (7.69%) had bilateral situations, hysterectomy may be required.
pseudoaneurysms.
In terms of management, it is interesting to underline that all Expert commentary
of the cases associated with vaginal bleeding (76.92%) with Rupture of a pseudoaneurysm of the uterine artery is a rare cause
the exception of the case reported by McGonegle etal. [14] , for of PPH that is very frequently associated with uterine surgical
which no information is available were successfully managed procedures or pregnancy-related interventions, such as dilation
with arterial embolization, while the two cases with no vaginal and curettage, hysteroscopy, previous myomectomy, cesarean

316 Expert Rev. Obstet. Gynecol. 7(4), (2012)


Table 1. Published cases of uterine artery pseudoaneurysm and postpartum hemorrhage after vaginal delivery.
Study (year) Onset Mode of delivery Obstetric Uterine Size of pseudo Location of Vaginal Treatment Ref.
history atony aneurysm (mm) pseudoaneurysm bleeding
Zimon etal. Late (8 days) UVD Uterine Yes 2530 Left Yes Arterial embolization [18]
(1999) perforation

www.expert-reviews.com
Pelage etal. Late (45days) UVD None NA NA NA Yes Arterial embolization [19]
(1999)
Wald etal. Late (8weeks) UVD Manual removal No 25 Right Yes Arterial embolization [6]
(2003) of placenta
McGonegle Late (6weeks) UVD None No 20 Bilateral Yes NA [14]
etal. (2006)
Fargeaudou Early UVD Surgical Yes 6 Left Yes Arterial embolization [8]
etal. (2008) abortion
Early Forceps None No 6 Left Yes Arterial embolization
Early UVD Forceps No 10 Right Yes Arterial embolization
Early Forceps None Yes 8 Left Yes Arterial embolization
Late (10days) Forceps None No 7 Right Yes Arterial embolization
Nagayama etal. Early VE None Yes 25 Left Yes Arterial embolization [9]
(2011)
Chung Fat etal. Early UVD None No NA Right No Arterial ligation + [13]
(2008) hysterectomy (maternal
death)
Marnela etal. Late (3weeks) UVD/placental None No 17 Right Yes Arterial embolization [5]
(2010) fragments retention
Present case Late (5days) UVD/Kristeller None No 9 Right No Arterial ligation [OrigoniM
et al.;
Unpublished
Data]
NA: Not available; UVD: Uncomplicated vaginal delivery; VE: Vacuum extractor.
Uterine artery pseudoaneurysm & postpartum hemorrhage
Special Report

317
Special Report Origoni, Gelardi, Pasi, Salvatore & Candiani

section, placental accretion and/or retained gestational fragments. recently, PPH has almost always been approached and managed
Very rarely the condition can occur after vaginal delivery, even in with hysterectomy, which in many cases represented too high a
uncomplicated situations and in risk-factor-free patients. price for patients.
In the great majority of cases the clinical presentation is In recent years, alternative and conservative treatment options
characterized by significant vaginal bleeding at early or late such as selective arterial embolization have received great con-
onset in the postpartum. Of note, in this case report it is the sideration in clinical practice, mainly for the safety and efficacy
unusual clinical pattern massive intra-abdominal bleeding of these procedures.
and coexisting uterine rupture after an uncomplicated vaginal Diagnostic improvement principally relies on modern imaging
deliverythat calls for particular attention in the differential techniques such as CT and angiography that, for the future,
diagnosis of usual PPH. In fact, no similar cases have been will hopefully become standard procedures to arrive at the correct
found to have been published in the literature. The exact role diagnosis and plan the most effective treatments.
of the pseudoaneurysm and of the uterine rupture in the forma-
tion of the hemoperitoneum is not entirely clear, and which of Ethical conduct of research
the two conditions needs to be identified as the primary cause Being a totally anonymous report with no personal identifying descriptions
of PPH is still to be determined. Our hypothesis is in favor of or images, the Institutional Review Board gave exemption to ethical
a greater responsibility being attributed to the arterial pseu- approval. The authors have followed the principles outlined in the
doaneurysm rupture and secondarily to the uterine rupture. Declaration of Helsinki for all human or animal experimental investiga-
Another interesting point, consistent with reports in the litera- tions. Informed consent for the use of clinical, imaging or laboratory data
ture, is the association with the absence of vaginal bleeding in for scientific or teaching purposes is routinely obtained at the authors
more complex cases that required aggressive treatment options, institution for all patients.
such as laparotomy with arterial ligation or even hysterectomy.
According to our experience and the literature, we believe that Financial & competing interests disclosure
uterine artery pseudoaneurysm must be extensively considered The authors have no relevant affiliations or financial involve-
and ruled out in any case of PPH both after cesarean sec- ment with any organization or entity with a financial interest in or
tion and vaginal delivery and that uterine rupture may be an financialconflict with the subject matter or materials discussed in the
associated complication. manuscript. This includes employment, consultancies, honoraria, stock
ownership or options, expert testimony, grants or patents received or
Five-year view pending, or royalties.
PPH represents one of the major causes of maternal mortality No writing assistance was utilized in the production of this
worldwide, especially in low-resource healthcare settings. Until manuscript.

Key issues
Postpartum hemorrhage (PPH) represents a common life-threatening condition for women.
Uterine artery pseudoaneurysm is a rare cause of PPH; it is more frequently correlated with cesarean section but cannot be excluded
after spontaneous vaginal delivery.
In the case of PPH a detailed imaging evaluation should be obtained.
Selective angiography is the diagnostic gold standard and is decision-guiding for the treatment approach.
Uterine artery embolization is the standard treatment for PPH secondary to pseudoaneurysm.
In case of massive hemoperitoneum, uterine rupture should be excluded and hysterectomy may be necessary.

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