Unusual cause of intraoperative haemorrhage: a lesson for patient counselling

Hepatocellular carcinoma (HCC) is a well-known malignant neoplasm of the liver associated with spontaneous haemorrhage in 3%–15% of cases. This complication is life threatening and has a mortality rate of 33%–100%. Despite the frequency and severity of spontaneous haemorrhage, the importance of patient education about this complication has not been highlighted before. There is currently no information available on the NHS UK website, and no publications have addressed the effect of patient education. We present this case report describing a patient who developed classical symptoms of haemorrhage the day before her elective HCC resection, but was unaware of its importance, and thus did not seek medical attention. She was subsequently found to have a large volume haemoperitoneum, anaemia and a ruptured HCC intraoperatively. This case illustrates the significant importance of counselled regarding the symptoms and risk of spontaneous rupture of HCC to prompt early presentation to medical services.

Surgical oncology, Hepatic cancer

The incidence of hepatocellular carcinoma (HCC) is rising and is responsible for approximately 700 000 deaths worldwide every year.1–4 Up to 15% of HCCs are associated with spontaneous haemorrhage,5 6 and it is the third leading cause of death in patients with HCC following direct cancer deaths and liver failure.7 8 Several articles have highlighted that early diagnosis and intervention in cases of HCC haemorrhage is key to reduce the mortality associated with it.9–12 Furthermore, several case reports described haemorrhage as the first presentation of HCCs,12–15 and the majority of these cases occur in patients who are already aware of their diagnosis.7 However, no literature is available on the effect of preventive patient education on HCC haemorrhage related mortality. The NHS UK website does not warn patients about this severe preoperative complication.16 Similarly, the need for education has not been highlighted in the literature. In other conditions carrying a high risk of haemorrhage, such as abdominal aortic aneurysms (AAA), patient education is well established.17 There are clear descriptions of the symptoms associated with a ruptured AAA on the NHS UK website, with instructions on how to proceed should they occur.18 Case presentation We report the case of a woman in her 60s undergoing an elective right inferior liver resection for a large exophytic liver tumour, radiologically presumed to be an HCC. The patient was initially referred from her general practioner (GP) with a 1-year history of a right-sided, painless, palpable abdominal mass. Relevant medical history included a laparotomy, terminal ileum resection and ileocolic anastomosis for bowel obstruction secondary to endometriosis in 1988. At the time of presentation, the patient took no regular medications, had no allergies and was a non-smoker, with an estimated alcohol consumption of 4 units/week. Serological tests for hepatitis B and C were negative. On examination, she had a large palpable right-sided abdominal mass measuring approximately 10×15 cm. A contrast CT scan of her abdomen and pelvis revealed a 12 cm large, rounded exophytic, hypoattenuating heterogenous lesion arising from segments V and VI of her liver (figure 1). The lesion is separate from surrounding structures such as the duodenum, pancreas, kidney and gallbladder. There was no evidence of cirrhosis and biochemical liver function tests were normal. Preoperatively, on the day of her procedure, the patient did not exhibit any signs of haemodynamic instability. She was complaining then of mild pain in her abdomen, and she only exhibited mild tenderness without peritoism on examining her abdomen. An elective right inferior hepatectomy (segments V and VI) and cholecystectomy was commenced. On entry to the abdomen, a large volume haemoperitoneum and a ruptured HCC with active bleeding was found (figures 2 and 3). Intraoperative arterial blood gas showed a haemoglobin (Hb) concentration of 85 g/L. Preoperative Hb 5 days prior to the operation showed an Hb concentration of 131 g/L .Two units of packed red cells were transfused intraoperatively. Initially haemostasis was achieved followed by complete resection of the lesion with a combination of the Cool-tip radiofrequency ablation system (Medtronic) and Calvitron ultrasonic surgical aspirator. Postoperatively, her Hb concentration recovered to 110 g/L. Her total preoperative and perioperative blood loss was estimated to be 1500 mL. Treatment The management of a spontaneous HCC haemorrhage is determined by several factors including the fitness of the patient, their Child-Pugh score, the degree of haemorrhage and the underlying extent of disease. A variety of strategies have been employed including conservative management, transarterial embolisation (TAE),7 10 11 19 20 transarterial chemoembolisation,5 21 emergency resection and delayed resection following successful haemostasis by other means such as TAE, packing or suture plication.19 20 In this case, the patient was already under general anaesthesia, and a laparotomy was already performed; thus, the haemorrhage was controlled by pressure and packing. Simultaneously, the tumour was resected using standard methods of liver resection; however, complicated by the need of continuous pressure and packing of the rupture. An intermittent Pringle’s manoeuvre was performed to aid control of bleeding. Most of the intraperitoneal haematoma was evacuated, and the abdominal cavity was irrigated by large volume of warm water. Outcome and follow-up On discussing the intraoperative findings with the patient, she reported sudden onset of acute abdominal pain with nausea and vomiting and an episode of syncope 1 day prior to her elective operation. She did not present herself to medical services as she attributed these to anxiety about her imminent operation. Her postoperative recovery was complicated with a postoperative paralytic ileus likely exacerbated by the intraperitoneal haematoma. This resolved with conservative management by day 5. Repeat measurements of her Hb concentration remained satisfactory, and she was discharged on day 12 due to social reasons. She was seen in the clinic 3 weeks after her discharge, which was unremarkable, indicating she had recovered well from the operation. The histopathology report confirmed a fully excised, poorly differentiated HCC with evidence of small vessel vascular invasion (T2 R0). She had a CT scan 6 weeks following her procedure, then an MRI every 4 months for the first year and every 6 months the year after. She continues to be followed-up as an outpatient with no evidence of recurrence to date.

There were approximately 800 000 new HCCs worldwide in 2018, with the highest incidence in Asia.1 2 In the United Kingdom, the number of cases is increasing and the incidence of HCCs is predicted to continue to rise during the next decade.1 22 This is due to a number of factors including a rise in hepatitis C infections and increasing rates of liver cirrhosis secondary to alcohol and fatty liver disease.3 4 23 Therefore, are more likely to encounter such cases and should be aware of the risk of spontaneous haemorrhage. Several mechanisms have been proposed to explain the propensity for HCCs to undergo spontaneous haemorrhage. Hypotheses include that rapid tumour growth causes splitting of the surrounding parenchyma or that rapidly enlarging tumours undergo central necrosis resulting in haemorrhage.7 10 11 21 24 Large tumours are believed to result in venous congestion causing high intratumoral pressure, predisposing to rupture.11 24 Tumour size has been attributed as the most important risk factor for spontaneous rupture of HCC. Two studies report the greatest risk lies with tumours over 5 cm, and they described the relationship between the size and risk of rupture to be linear.25–27 Moreover, exophytic tumours also appear to be at greater risk of undergoing spontaneous haemorrhage than those which do not protrude over the liver margin. The location of the HCC has also been linked to an increased risk of rupture. Peripheral tumours tend to rupture more often, likely secondary to having a shallow layer of overlying parenchyma being at greater risk of spontaneous haemorrhage.26 Other proposed risk factors include liver cirrhosis, portal vein thrombosis, systemic hypertension and impaired liver function.5 9 11 25 28 Recent case reports have focused on haemorrhage as the first indication of an underlying HCC;12–15 however, as in our case, the majority of cases occur in those with previously diagnosed HCCs.7 Spontaneous haemorrhage of HCC is a potentially life-threatening complication and often occurs in patients with limited hepatic functional reserve and extensive disease.27 The most common symptom in patients with spontaneous haemorrhage from an HCC is abdominal pain (66%–100%), followed by abdominal distension, anorexia, vomiting, syncope and shock.9 10 12 29 30 Survival is dependent on several factors such as underlying disease extent, physiological reserve of the patient and treatment modality. However, in those patients suitable for intervention, long-term survival is possible. However, there is little information available for patients about spontaneous HCC haemorrhage which is both a relatively frequent and potentially life-threatening complication. There is currently no information available on the NHS UK website, while similar conditions such ruptured AAAs have well-established patient education initiatives, aimed at reducing mortality.17 18 As recent studies have indicated, early diagnosis and intervention is key to reducing mortality in cases of ruptured HCCs, and if our patient had undergone surgery at a later date, her outcome could have been much worse.5 9 11 24 Careful consideration for establishing an information leaflet to be given to HCC patients preoperatively, due to the heavy impact this risk poses to the patient’s life. Patient counselling regarding the risk, common signs and symptoms, and survival of HCC rupture is important and should be done after diagnosis. Patients must be warned to seek emergency medical care should they become symptomatic. In addition, our case highlights the need for clinicians to be aware of this complication, particularly in light of the rising incidence of HCC. Learning points. Spontaneous haemorrhage of hepatocellular carcinoma (HCC) is a potentially life-threatening complication, especially those larger than 5 cm. Patients diagnosed with HCCs should be educated and counselled about the signs and symptoms of haemorrhage to facilitate early presentation to medical services, as rapid diagnosis and treatment reduces mortality. It is important for clinicians to be aware of this complication, particularly those in regions in which the incidence of HCCs is rising. Footnotes Contributors: All authors have contributed to the planning, conduct and reporting of the work described in the article. Main contributors of project idea and design are DS and MF. Literature review and data collection were mainly contributed by LA and OE. Design framework by DS and MF. Overview of literature review and abstract are DS and MF.Analysis and learning points contributed by LA, OE, MF and DS. 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. Case reports provide a valuable learning resource for the scientific community and can indicate areas of interest for future research. They should not be used in isolation to guide treatment choices or public health policy. Competing interests: None declared. Provenance and peer review: Not commissioned; externally peer reviewed. Ethics statements Patient consent for publication Consent obtained directly from patient(s)

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