Managing a spontaneously ruptured giant hepatic hemangioma nonoperatively

Toni Green, DO

General Surgery Resident

Department of Surgery

John D?Emilia, MD

Surgical Oncology

Attending

Department of Surgery

University of Medicine

and Dentistry of

New Jersey

Stratford, NJ

Introduction: Hemangiomas are the most common benign liver masses. They are often diagnosed as incidental findings on imaging studies of the abdomen or during exploratory surgeries. These lesions can produce symptoms when they enlarge to more than 4 cm.

Results and discussion: Although hemangiomas are rare, spontaneous rupture of these liver lesions has been well-described. Of the 32 cases of spontaneous rupture reported in the literature, 4 were treated with transcatheter hepatic arterial embolization and eventual resection. We describe a case of spontaneous rupture of a giant hemangioma that was treated solely with embolization.

Conclusion: Transcatheter hepatic arterial embolization should be considered before undertaking exploratory laparotomy to treat patients who have a hemorrhagic hemangioma.

Disclosure

The authors have no relationship with any commercial entity that might represent a conflict of interest with the content of this article and attest that the data meet the requirements for informed consent and for the Institutional Review Boards.

Hemangiomas are the most common benign liver masses, with an incidence between 2% and 7% in the general population.¹ They are commonly diagnosed as incidental findings on imaging studies or during exploratory surgeries. Hemangiomas measuring 4 cm or larger are considered "giants" and may produce symptoms. We report a case of spontaneous rupture of a giant hemangioma that was treated nonoperatively with angiographic embolization.

Case report

A 56-year-old woman presented to the emergency department after experiencing sudden chest and epigastric pain with no associated trauma. She was hypotensive and required vasopressor support. Hematology studies showed a hemoglobin count of 7.6 g/dL (normal, 12.0?15.0 g/dL). The hemoglobin test was repeated after aggressive fluid resuscitation, and her count had dropped to 5.0 g/dL.

Figure 1—Initial CT scan showing free fluid in the abdomen and pelvis and a low-attenuation liver mass.

Figure 2—Repeat CT scan showing an increase in fluid, with some extravasation of contrast from the inferior right lobe of the liver.

On physical examination, the patient appeared thin and mildly distressed. Her abdomen was distended, soft, and without guarding, but epigastric tenderness was elicited on palpation. Computed tomography (CT) scanning showed a 10 x 6-cm, low-attenuation lesion in the right lobe of the liver and a significant amount of free fluid throughout her abdomen and extending into the pelvis (Figure 1). A CT scan performed 7 hours later showed an increase in the amount of fluid as well as active extravasation of contrast from the inferior right lobe of the liver (Figure 2). The CT scan was reviewed by a radiologist, and the lesion was determined to be a hemangioma.

Because of the patient's deteriorating condition, angiography was perfomed in an attempt to control the hemorrhage. This showed active extravasation from the right hepatic artery, at which point a transcatheter hepatic arterial embolization was undertaken (Figure 3). The patient tolerated the procedure well and was admitted to the intensive care unit for observation. Her hemoglobin count remained stable, and she was treated conservatively for the remainder of her hospital stay. She was administered hydromorphone for pain and pantoprazole, which inhibits stomach acid production.

The patient failed to return for a scheduled follow-up examination, including a percutaneous liver biopsy and testing for carcinoembryonic antigen and alpha fetal protein tumor markers. However, she came back to the hospital 4 months later describing shortness of breath and right-sided chest pain. A CT scan at that time revealed a subdiaphragmatic fluid collection with a tract into the right chest. Using interventional radiology, a drain was placed into the subdiaphragmatic collection, and the chest was drained through a thoracostomy. Cytology was consistent with abscess, and no malignancy was noted. The patient had an uneventful recovery and was discharged from the hospital. Although surveillance chest radiographs and CT scans were planned, the patient has not returned for follow up.

Discussion

When a hemangioma measures more than 4 cm, it is classified as a "giant." Giant hemangiomas may become symptomatic, generally producing poorly localized and vague abdominal pain or discomfort. Symptoms attributed to their mass effect have been reported, including abdominal fullness, early satiety, nausea, and vomiting. Although spontaneous rupture of hemangiomas is rare, it is often discussed in the literature.

Radiographic findings

On ultrasonography studies, hemangiomas typically appear as well-circumscribed lesions that have a homogenous hyperechoic pattern and no posterior shadowing. Classic CT findings include a well-defined lesion that is hypodense when compared with the rest of the liver, demonstrates early enhancement during the contrast phase of the study, and shows centripetal contrast enhancement with complete isodense opacification on delayed images.¹ Hemangiomas are often discovered incidentally on general abdominal screening CT scans, and they frequently do not meet all of these criteria. This has led to the establishment of dedicated protocols to assist in diagnosing them. When ultrasonography or CT scanning fails to confirm the diagnosis, magnetic resonance imaging (MRI) is used.¹ The characteristic findings of hemangiomas on MRI include a well-defined lesion with low signal intensity on T1-weighted images, very high signal intensity on T2- weighted images, and peripheral nodular enhancement on dynamic gadolinium contrasted images.¹

A

B

C

D

Figure 3—Angiography images showing active extravasation of contrast from the liver (A and B), after which transcatheter hepatic arterial embolization was performed (C and D).

Treatment

Suspected hemangiomas are generally treated only when they become symptomatic, show evidence of hemorrhage, or have diagnostic uncertainty. Transcatheter arterial embolization has been used to manage symptomatic giant hemangiomas.^2,3 Of the 32 cases of spontaneous hemangioma rupture reported in the literature, 84% were classified as giant hemangiomas.⁴ Four of these were treated with transcatheter angiographic embolization and required subsequent hepatic resections.⁴ Yamamoto and colleagues reported the first such case in 1991, describing a hemangioma that was treated initially with embolization, which was repeated 6 days later due to rebleeding.⁵ Resection was undertaken 2 weeks after the second embolization. In 1995, Mazziotti and associates reported the case of a patient who presented in shock, secondary to liver hemorrhage.⁶ This patient initially had been treated with transcatheter arterial embolization, but a CT scan 3 weeks after the procedure showed persistence in the intrahepatic fluid collection, necessitating a lobectomy. Soyer and colleagues described the case of a patient who underwent embolization for a ruptured hemangioma that subsequently re-bled and required repeat embolization. The patient experienced a second rebleeding, and hepatic resection was undertaken.⁷ Corigliano and associates reported the final case in 2003.⁴ Their patient underwent transcatheter arterial embolization but needed a resection the following day.

In addition to these cases, there is one report of a woman who was 18 weeks pregnant when she suffered an intrahepatic hemorrhage, which was treated by embolization and observation.⁸ She did not develop hemoperitoneum and tolerated the procedure well. The hemangioma was no longer visible when the patient underwent an elective primary Cesarean section at 39 weeks' gestation.

Embolization has become a more common treatment in cases of blunt or penetrating liver trauma. The use of embolization has improved overall patientoutcomes, although a relatively high rate of morbidity has been noted.⁹ Reported liver-related complications include delayed liver hemorrhage, hepatic necrosis, biloma, bile leak, intrahepatic abscess, and perihepatic abscess.⁹ Four months after our patient's embolization, she experienced fever and shortness of breath. The workup at that time was consistent with an intra-abdominal abscess and a reactive pleural effusion, which are the known complications of liver embolization.

Conclusion

Prior to this report, there were no other reported cases of a ruptured hemangioma with associated hemoperitoneum that were successfully treated with angiography alone. Although a few clinicians had attempted to do so, their patients experienced rebleeding and underwent subsequent resections.^5,7 It is worth considering transcatheter hepatic arterial embolization as therapy for a hemorrhagic hemangioma before performing exploratory laparotomy. A patient in whom a giant hemangioma has ruptured is likely to be hemodynamically marginal at best; embolization allows the hemorrhage to be controlled so that subsequent resuscitation can be provided in a more controlled environment. While patients may ultimately require resection, surgery can then be performed on a more stable, optimized patient.

References

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