Abdominal apoplexy during pregnancy

  1. Jasmeet Kumari and
  2. Rosemary Harkin
  1. Obstetrics and Gynaecology, Our Lady of Lourdes, Drogheda, Ireland
  1. Correspondence to Dr Jasmeet Kumari; Jasmeet2k58@yahoo.com

Publication history

Accepted:13 Oct 2020
First published:30 Oct 2020
Online issue publication:30 Oct 2020

Case reports

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Abstract

We report a case of idiopathic spontaneous intraperitoneal haemorrhage (ISIH) in a 31-year-old patient at 37 weeks gestation in her second pregnancy. The patient presented to the labour ward with abdominal pain and uterine contractions. The initial complain was of sudden onset, severe sharp pain in left iliac fossa. She started having uterine contractions within 30 min of her presentation. Examination confirmed early labour with a footling breech presentation. Urgent caesarean section was performed that confirmed peritoneal bleeding of unknown origin with safe delivery of the baby. Mother and baby were safely discharged on day 5. ‘Abdominal apoplexy’ (ISIH), is a rare obstetric emergency with increased risk of fetal and maternal morbidity and mortality. With various clinical presentations as a possibility, diagnosis is challenging. High index of suspicion with prompt management of suspected cases can be pivotal life saving measure for the fetus and mother.

Background

Idiopathic spontaneous intraperitoneal haemorrhage (ISIH) also known as abdominal apoplexy in pregnancy is a rare clinical entity for majority of obstetricians across the globe. It is characterised by spontaneous non-traumatic haemorrhage in peritoneal cavity from unidentified source. The pathophysiology of ISIH during pregnancy is uncertain. Emerging evidence suggests that factors such as endometriosis and nulliparity, can predispose to this condition but the aetiology is still obscured in majority of reported cases.1

Chronic inflammation secondary to endometriosis is responsible for fragility of utero-ovarian vessels and the resultant adhesions may give further tension to these vessels during pregnancy. These changes with endometriosis are culprit for spontaneous haemoperitoneum in vast majority.2 3

In vitro fertilisation in patients with severe endometriosis may be a risk factor for ISIH.4

Clinical presentation of ISIH in pregnancy can vary from mild abdominal pain of uncertain nature to acute circulatory collapse compromising fetal and maternal life. High maternal and fetal mortality is observed in severe cases. Any culprit lesion is unidentified in majority of cases. High index of clinical suspicion in selected case with multidisciplinary team approach is the integral part of patient management.

We describe a rare case of ISIH in pregnancy that lead to acute circulatory collapse during caesarean section. With few case reports available to date to the best of our knowledge, we highlight the importance of its possibility in pregnancy.

Case presentation

This is a case report of a 31-year-old, gravida 2 para 1, Caucasian white woman who was diagnosed with abdominal apoplexy during lower segment caesarean section (LSCS).

The patient presented to labour ward at 37 weeks with regular painful contractions which had quickly followed on from an initial sharp, twitching pain in the left iliac fossa associated with nausea and three episodes of vomiting. She denied any history of trauma or physical or sexual activity before the onset of pain. This was her second pregnancy with spontaneous conception. She had a previous uncomplicated vaginal birth at term. She had no history of subfertility or dysmenorrhoea. The patient was booked in this pregnancy at 12 weeks under midwifery care with no significant medical, surgical or gynaecological history. She attended all of her routine antenatal visits without any event.

On general physical examination, she appeared pale and distressed with pains. Her vital signs confirmed blood pressure of 121/78 mm Hg, pulse rate of 110 beats/min, temperature of 36.5°C, SaO2 (arterial oxygen saturation) of 98% on ambient air and respiratory rate of 16 breaths per min.

Contractions were regular about 3 in 10 min, moderate in intensity. Vaginal examination confirmed her to be in early labour. A transabdominal ultrasound confirmed footling breech presentation. After written informed consent, urgent LSCS was performed in view of fetal positioning.

A massive haemoperitoneum was identified during caesarean section, with intact uterus. A total of ~3 L of fresh blood and clots were drained from the abdominal cavity.

The liquor was clear. The baby was delivered as a breech presentation with Apgar scores of 6 and 10 at 0 and 5 min. Arterial pH was 7.01 and venous pH was 7.04. The baby did not require resuscitation and was transferred to the ward with mother. The time between presentation and delivery was 50 min. Spinal anaesthesia failed, requiring general anaesthesia.

Treatment/management

Expert obstetrical and surgical opinion was obtained with an urgent laparotomy, secondary to massive haemoperitoneum. During laparotomy, patient became haemodynamically unstable with a blood pressure of 40/20 mm Hg and a heart rate of 125 bpm. Arterial line was secured by the senior anaesthetist and massive transfusion protocol was activated as per local transfusion guidelines. She received 6 units of red cell concentrate, 4 units of fresh frozen plasma, 4 gm of fibrinogen and 2 gm of tranexamic acid.

Inspection of her ovaries, fallopian tubes, broad ligament, omentum, large and small intestine, spleen and liver did not identify any source of bleeding. After resuscitation, patient became haemodynamically stable with no further active bleeding. As bleeding had stopped spontaneously, vascular surgical input was not required. Peritoneal washout was performed. A drain was left in situ and the abdomen closed routinely. The patient was monitored in high dependency unit for next 24 hours. A CT scan including thorax, abdomen and pelvis was performed 12 hours post caesarean section to determine any potential source of active bleeding. The scan did not reveal any source of bleeding or haematomas.

Outcome and follow-up

The patient was debriefed about the events and was transferred to postnatal ward after 24 hours of monitoring in high dependency unit. She was discharged home on day 5 in well condition with haemoglobin of 10 gm/dL. A postnatal follow-up appointment was arranged to review the patient and to address her concerns. She was fortunate to survive as early identification of the problem resulted from the coincidental footling breech presentation. Urgent caesarean section was performed because of the footling breech presentation in early labour due to the risk of cord prolapse.

Discussion

The term abdominal apoplexy first described in 1931 by Green and Power after reporting case series of intraperitoneal haemorrhage due to rupture of gastroepiploic arteries.5 It is a broad term used for all cases of spontaneous haemoperitoneum. Abdominal apoplexy in pregnancy can be called as ‘spontaneous haemoperitoneum in pregnancy’. It is associated with a high maternal mortality rate, ranging from 3.6% to 49% with highest mortality observed during perinatal period.6 7

The first case of ISIH in labour was reported in 1909 by Maurice,8 in which a patient underwent laparotomy at day 2 post vaginal delivery due to worsening abdominal distention and haemodynamic instability. He concluded that haemoperitoneum was secondary to spontaneous rupture of an unidentified blood vessel.

ISIH is associated with gynaecological and non-gynaecological causes. The main differentials are uterine rupture, abdominal pregnancy and placental causes.

Among gynaecological causes, spontaneous rupture of tubo-ovarian vessels secondary to physiological dilatation or acute rise in intra-abdominal pressure can be rare dramatic causes.9 10 Involution of decidualised endometriotic spots may lead to rupture of fragile tubo-ovarian vessels secondary to tension of enlarged uterus.1

Non-gynaecological causes include rupture of liver or spleen with or without its vasculature. Other rare causes of ISIH include perforated peptic ulcer, acute appendicitis, haemolysis, coeliac artery aneurysm, haemangioma and ruptured abdominal viscera.11 It is hypothesised that spontaneous rupture of arteriosclerotic vessels in hypertensive patients secondary to weakness of tunica media may be the cause of abdominal apoplexy.12 In obscure bleeding cases like the one described above; an expert vascular surgical opinion can be of significant help.

Due to non-specific presentation and numerous differential diagnosis, surgical intervention can be challenging especially during second half of pregnancy. In these cases caesarean delivery has to be performed to achieve haemostasis as conservative management can be associated with risk of recurrence and development of infected haematomas.2

ISIH is an abrupt, life-threatening situation and is a diagnosis of exclusion. In literature, the maximum known blood loss in patient with spontaneous haemoperitoneum in pregnancy was 9 L, the patient survived but unfortunately intrauterine fetal death at 26 weeks gestation was reported.13

Abdominal apoplexy in pregnancy primarily occurs spontaneously in the third trimester, as was the case in our patient. Presentation can vastly differ from subacute abdominal pain with reassuring fetal heart rate to hypovolaemic shock and fetal distress. The primary diagnosis of utero-ovarian rupture of vessels is rare and can be easily missed. The delay in surgical treatment because of missed diagnosis is usually associated with poor prognosis.14 Prompt resuscitation and timely surgical intervention should be considered in cases with worsening abdominal pain along with nausea or vomiting and with haemodynamic compromise or fetal distress.

Similarly, in the postpartum period there should be a high index of suspicion in a patient exhibiting signs of unexplained abdominal pain or abdominal distension with cardiovascular instability. Since preoperative diagnosis is difficult, involvement of senior clinical colleagues and multidisciplinary team approach should be considered promptly.

This case describes a rare critical situation which if was not managed appropriately would have led to severe adverse outcome.

Patient’s perspective

This is patient statement received on 22nd April 2020.

“On the 27th January 2020 at approximately 6 p.m., I was sitting down resting and when I got up from the chair. I started experiencing a pain on my left side of my abdomen which felt like a stitch.

Within a short time the pain became extremely intense. I then became clammy, nauseated and generally unwell. I knew it did not feel pregnancy related as this was my second pregnancy, my gut feeling was to go to the Hospital as soon as possible. Although I did not know what was causing the pain, I knew there was something seriously wrong.

I was very conscious of the safety of my unborn child as I was told that my Baby was footling breech position. On the journey to the Hospital the pain was becoming worse in which I started to vomit quite a lot and I also began to feel pressure which I knew was the start of my labour as it was a similar pain to my first pregnancy. My labour started to progress rapidly from this point onwards.

On reaching the Hospital I was met by the Midwife who took me into the Labour Ward where the registrar was waiting to assess and scan me. I was then assessed and it was confirmed that my baby was still in breech position and was informed that I would need to have emergency C-section, every member of the team there acted so quickly.

I was taken to Theatre and from this moment on I have vague memories of what happened. It was a very traumatic experience but at the same time I was grateful for every staff member that was taking care of me at the time. They ensured that my Baby arrived safely and healthy which I will be eternally grateful for.

When I woke up after the C-section I was informed by the consultant of the events which they felt was a very rare case of events that had happened. I was frightened at the time but felt lucky to be here with my baby”.

Learning points

  • Abdominal pain during pregnancy is not uncommon and may be potentially serious. Encourage patients to seek medical attention and expert advice.

  • Clinical presentation of abdominal apoplexy may vary, causing hypovolaemic shock without any overt vaginal bleeding.

  • Always consider abdominal apoplexy as a possible differential diagnosis in cases of severe abdominal pain/sudden collapse, during pregnancy or in early postpartum period.

  • Despite reassuring cardiotocography in haemodynamically unstable cases in third trimester of pregnancy, consider idiopathic spontaneous intraperitonealhaemorrhage, as a rare possibility with early multidisciplinary team involvement.

Footnotes

  • Contributors JK is the corresponding author: conceptualised the case report and basic manuscript writing (primary surgeon involved in the case). RH is the co-author: helped in editing and final approval of the version.

  • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

  • Competing interests None declared.

  • Patient consent for publication Obtained.

  • Provenance and peer review Not commissioned; externally peer reviewed.

References

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