Key words
ECMO - ARDS - uterine rupture - vaginal birth after 2 Caesarean sections - TRALI -
SIRS
Schlüsselwörter
ECMO - SARS - Uterusruptur - vaginale Entbindung nach 2 Kaiserschnitten - TRALI -
SIRS
Introduction
Obstetrical patients rarely end up at intensive care units (ICU). Excessive postpartal
bleeding and hypertensive disorders are the most common reasons for ICU admissions.
Almost 10 to 15 % of ICU patients and up to 20 % of mechanically ventilated patients
meet criteria for acute respiratory distress syndrome (ARDS) [1]. Large blood loss and pre-existing bad pulmonary conditions represent two risk factors
for ARDS. This acute, diffuse, inflammatory lung injury often ends up in long-term
intensive care stays. Numerous other complications may follow. We describe the outcome
and emerging problems of a complicated delivery with postpartal ARDS which needed
emergency admission to ICU and a long-term therapy with extracorporeal membrane oxygenation
(ECMO) for a total of 50 days.
Case Report
A 34-year old female patient from Somalia (gravida 6, para 5) presented at 38 weeks
of gestation at our hospital for birth registration. She reported three vaginal deliveries
in Somalia and had two Caesarean sections (CS) in Germany. She roughly mentioned “lung
problems” in her medical past which were supposed to be resolved. Her exercise capacity
seemed unaffected. Exact details of the medical history and previous operations were
blurred by the patient (no clear information about which hospitals had been involved
were given). The ultrasound examination showed a normal fetus growth with normal Doppler-findings
and a normal amniotic fluid index. During the general examination she presented signs
of preeclampsia (increased blood pressure (> 160/100 mmHg), protein/creatinine ratio
of 900 mg/g). Therefore the medical indication for induction of labour was explained
to the patient. Partly due to religious convictions the patient planned to have more
children in the future, so spontaneous labour instead of third Caesarean section was
discussed. Aware of increased risk for uterine rupture the patient decided for spontaneous
delivery and gave her informed consent.
The vaginal examination showed no female genital mutilation, a soft cervix, approximately
1 cm long and with a uterine orifice open to 2 cm, the childʼs head fixed. First an
induction trial with an extraamniotic Foley balloon catheter was made. As the balloon
did not stay in place however, 1 mg of prostaglandin gel was used intravaginally at
Bishopscore 5.
During the first stage of labour at 3 cm cervical dilatation a prolapse of the childʼs
arm occurred. While the patient was getting prepared for CS fetal bradycardia lead
into emergency CS. Intraoperatively a large rupture of the anterior uterine wall became
apparent. After safe delivery of an initially limp baby (Apgar 2/10/10, pH 6.99) with
direct transfer to the paediatricians this rupture could be sutured with single stiches
in the area of uterotomia and finally with 2 layers of continuous stitches. Due to
excessive intrapartum bleeding (haemoglobin 5.4 mg/dl, blood loss approximately 1.5
litres) the patient was transferred to intensive care unit (ICU). There she was stabilized
with blood transfusion. A perioperative antibiotic prophylaxis (cefuroxime) was initiated.
The patient was transferred to the obstetric ward 24 hours after CS. On day 2 after
CS the patient suffered more and more from pronounced dyspnea. The cardiologic examination
showed normal results and pulmonary embolism was excluded by CT scan. However, the
CT showed bipulmonary infiltrates and shadows. On suspicion of pneumonia the patient
was treated with a broad-spectrum antibiotic (imipenem). In the further course her
general condition worsened so that at one point when the patient suffered from massive
dyspnea the resuscitation team was alerted. When the team arrived the patient was
unconscious and with low heart rate which became an asystolia during the intubation
procedure and lead to resuscitation. CPR was performed for 2 minutes during the intubation
procedure. Saturation at this time was not documented. The blood gas analysis afterwards
showed pH 7.375, pO2 171.9 mmHg, BE 2.2 and lactate 0.9 mmol/l. Again we admitted the patient to ICU at
day 5 after CS.
During the following days the patient underwent lung protective ventilation with low
tidal volumes (4–6 ml/kgBW) in relation to the patientʼs ideal body weight, plateau
pressures were below 30 cm2 H2O. PEEP was adjusted between 10 and 16 cm2 H2O. Prone positioning of the patient was tried several times, but oxygenation was not
improved (non responder/recruiter). Prone positioning was kept for 14–16 h; during
that period the patient required four cardiopulmonary resuscitations due to recurrent
bradycardia of uknown origin. Bradycardia and asytolia occurred reflectory since the
first day in our ICU for example after coughing of the patient, after mouth care or
when changing tubus position or when performing prone positioning. At this point the
patient was not hypoxic. FiO2 was between 0.5 to 0.7 and pO2 never dropped below 70 mmHg. The patient was treated with atropin and/or epinephrine
when this happened and an external pacing was placed and changed to a transjugular
pacer on day 6.
At day ten decarboxylation became suddenly worse under prone positioning (pH 6.965,
pCO2 182 mHg, pO2 111.2 mmHg) and we deciced to perform a VV-ECMO and a tracheotomy. We used a VV-ECMO
for the main problem of decarboxylation and oxygenation. VV ECMO flow was up to 5 l/min
and this was sufficient for oxygenation, too. A VA ECMO was not used because of a
good cardiac function seen in repeated TEEs and TTEs.
During the installation of ECMO-cannulas the transjugular pacer was removed because
this vascular access was needed for the ECMO canula und we switched again to an external
pacer. The external pacer was needed until day 24 after ICU admission (day 14 after
VV ECMO). Then the patientʼs condition became more stable and no more bradycardias
came to pass. Anticoagulation was sustained with a continuous infusion of unfractionated
heparin, titrated to the plasma PTT (1.5 times normal). Until then, multiple microbiological
probes from blood and bronchoalveolar fluid showed no evidence of any bacterial, viral
or fungal infection.
At day 19 after starting ECMO therapy the microbiological testing of blood cultures
revealed for the first time pathogens (klebsiella oxytoca). We hypothesized a bacterial
contamination of the ECMO circuit because of a fibrinogen loss and spontaneous bleeding
from the bladder and from vascular punctures in the previous days. The infection was
treated with antibiotics (first piperacillin plus tazobactam, then meropenem plus
vancomycin) and by changing the oxygenator and the ECMO circuit, except the cannulas.
The strong bladder bleeding required a cystoscopic-guided laser coagulation. Overall,
the patient needed 58 units of stored blood. During the ECMO treatment the maximum
creatininkinase level was 250 U/l. ECG did not show abnormalities at any time. After
50 days the patient could be weaned from ECMO. The patient received assisted spontaneous
breathing for a few more days and the tracheostoma was closed at day 88 after ICU
admission. The patient was discharged home after 115 days of ICU treatment without
cardiopulmonary or neurological deficits.
During her ICU stay, the medical documentation could be provided. Years ago the patient
had undergone multiple pulmonary problems such as atypic pneumonia, chronic bronchitis,
and a lobectomy of the lower lobe of one lung one year before the delivery. Furthermore,
we found out that our patient required intensive care during her second last pregnancy.
A community acquired pneumonia during the third trimester of pregnancy ended in ARDS
together with renal failure and necessitated intubation, dialysis and emergency CS
at 34 weeks of gestation. The last pregnancy showed a very similar course. After an
induction of labour because of oligohydramnios the patient developed a restrictive
ventilation disorder. It ended in secondary Caesarean section at 40th week of pregnancy
and a transfer to ICU for invasive ventilation therapy in total for 11 days again
for ARDS of unknown origin.
The patient was seeking intentionally to mislead us by providing false information
regarding the aforementioned complications as a new pregnancy was not advised after
the second last birth due to maternal health reasons.
Further family planning was discussed with the patient and her husband. We strongly
recommended sterilisation or safe contraception. Medical visit at 6 months after discharge
was without pathological findings. Mother and baby are in good health.
Discussion
This case reports on four rare complications during labour and postpartum period:
the occurrence of arm prolapse, uterus rupture after two CS, postpartum haemorrhage
(PPH) followed by ARDS. And finally we report the successful use of ECMO for circulatory
support and for the treatment of acute respiratory distress in a postpartum situation.
Previous studies have reported a general rate of 1/5700 to 1/20 000 for uterus rupture
in unscarred pregnant uterus [2], [3], [4]. The overall incidence of uterine rupture in women with a prior Caesarean delivery
varies between 0.3 to 1 percent [5], [6], [7]. A recent large meta-analysis found an incidence of 0.7 % for uterus rupture/dehiscence
in women attempting a vaginal birth after one CS (VBAC-1) [7]. Furthermore, several series showed a general rate of 1.36 % of uterine rupture
during/after vaginal birth after two CS (VBAC-2) [8], [9], [10], [11], [12]. Although the uterine rupture rate of VBAC-2 is statistically higher than VBAC-1,
it should not be forgotten that the absolute rate is very low. This should be kept
in mind for patient counselling. In addition, the overall success rate of vaginal
delivery after one or two CS is high so that an achievement is possible in 76.5 %
and 71.1 %, respectively [13].
The greatest concern about a trial of labour after a previous CS is a rupture of the
scarred uterus. Whereby it should be mentioned that women with induction of labour
were more likely to experience uterine rupture or dehiscence than women with spontaneous
labour. Therefore, a labour induction indication after CS must be strictly defined.
Some factors are important for a high success rate: A previous history of vaginal
delivery and a favourable Bishop score are significant. The success rate is between
91 % for women with favourable Bishop score and a prior vaginal delivery, and 45 %
for women with no prior vaginal delivery and an unfavourable Bishop score. For women
with no prior previous vaginal delivery but favourable Bishop score a general rate
of 69 % successful induction was found [14]. The rupture rate of previously scarred uterus in a large prospective study is 0.4 %
with spontaneous labour versus 1 % with labour induction [15]. These results show that labour induction after CS is possible and clinically justifiable
– but, it should only be attempted in appropriately equipped institutions.
ICU admissions during pregnancy and peripartal period have a low incidence of < 1 %
[16], [17]. Among those patients, the most common causes are PPH and hypertensive disorders.
Respiratory failure represents a very rare complication. Differential diagnosis includes
pulmonary edema (mostly caused by tocolytic therapy), pneumonia, aspiration, and pulmonary
embolism.
In this case, none of the above mentioned pathologies were responsible for the postpartum
ARDS. To our opinion, ARDS in this patient was the result of serious tissue injury
caused by the uterus rupture with massive blood loss and blood cell transfusion. In
our case review the PPH was amplified by the large uterine rupture. The general rate
of 1–2.9 % of PPH is set in literature [18].
Like in this case serious tissue injury can cause systemic inflammatory response syndrome
(SIRS). This is a life threatening posttraumatic complication which is characterized
by fever, tachycardia, and tachypnea. These symptoms may resemble a sepsis.
Cellular lesion releases mitochondrial endogenous “damage/danger associated molecular
patterns” (DAMPs) that activate the immune system. Mitochondria are evolutionary endosymbionts
that were derived from bacteria and so bear bacterial molecular motifs. Zhang et al.
[19] found that mitochondrial DAMP is released by severe trauma in a similar way that
bacterial DNA (PAMPs, pathogen-associated molecular patterns) is released following
sepsis. Both use the toll-like receptor-9 (TLR9) to activate neutrophils through activation
of p38 MAP kinase (MAPK) enzyme. Similarly, formylated peptides released from bacteria
and mitochondria in these settings attract neutrophils by the process of chemotaxis
to sites of inflammation and injury through formyl peptide receptor-1 (FPR1) [20]. In both cases, the outcome may be acute lung injury, as part of the SIRS.
On the other hand, the most frequent and severe complication of multiple blood transfusions
is the transfusion-related acute lung injury (TRALI). The current pathogenic concepts
hold that proinflammatory mediators present in transfused blood products are responsible
for the initiation of TRALI [21]. Lee and collegues [22] hypothesized that mitochondrial (mt)DNA DAMPs are present in blood transfusion products,
which may be important in the initiation of TRALI. They extracted DNA from samples
of packed red blood cells, fresh frozen plasma (FFP), and platelets procured from
their local blood bank. The results showed a range of mtDNA DAMPs in all blood components
measured, with FFP displaying the largest variation and they concluded that mtDNA
DAMPs are present in packed red blood cells, FFP, and platelets may be mediators of
TRALI.
So in this case, the combination of tissue injury by the uterine rupture and the postpartum
haemorrhage, which occurs generally in 1 to 5 % of deliveries [23], has led to a severe ARDS in a patient with pre-existing bad pulmonary status.
ECMO often represents the last resort for serious acute respiratory distress, although
a long-term therapy can lead to life-threatening complications such as haemorrhage
and infections. In literature we found little mention of ECMO being used to manage
peripartum patients with massive bleeding [24], [25]. Both cases describe the use of ECMO for a short duration (150 respectively 72 hours).
In our case the use of ECMO lasted for 50 days which represents an exceptionally long
duration compared to literature [24], [25], [26], [27].
Others reasons which can lead to ECMO in obstetrical patients are peripartum cardiomyopathy
[28], amniotic fluid embolism [29] and transfusion-related acute lung injury [30]. In these cases ECMO lasted 40 to 150 hours and ended each time in a complete recovery
of the patient. Main complications of ECMO are bleeding, thromboembolism, cannulation
related problems and heparin-induced thrombocytopenia. Bleeding is the most common
complication (30 to 40 percent) of ECMO. In our case even a urological intervention
was necessary.
In conclusion, this case report includes an extraordinary sequence of unfortunate
circumstances: Beginning with the need of induction of labour due to preeclampsia
(with 2 previous Caesarean sections), arm prolapse during delivery in a multiparous
patient, fetal bradycardia most likely due to uterine rupture followed by emergency
Caesarean section with postpartum haemorrhage. This all followed by deterioration
of maternal pulmonary and general condition resulting in ICU admission, consequently
several manoeuvres of cardiopulmonary resuscitations and ECMO therapy for almost 2
months. Each of these risk factors is associated with an increased risk of mortality.
Nevertheless, our patient could be dismissed in good general condition.
This particular case shows that induction of labour after two prior Caesarean deliveries
might be dangerous although newer data showed that labour induction outcomes are similar
regardless of whether women have had one or two CS [31]. An early clinical registration of the future mother should be gold standard. It
allows obstetricians to identify high-risk pregnancies, take the necessary precautions
and might improve the general outcome of mother and child. And finally we can demonstrate
that ECMO support for severe respiratory failure in postpartum patients is possible
and allows a full recovery even after a long-term therapy with complications.
