Spontaneous internal hemorrhage of a giant hepatic hemangioma: A case report.

RATIONALE: Hepatic hemangioma, a benign liver tumor, can rarely spontaneously rupture and hemorrhage, which is then associated with significant mortality. The diagnosis of internal hemorrhage is challenging and the management is disputed. PATIENT CONCERNS: We describe the case of a 52-year-old female patient with chief complaints of fever and anemia, with no history of recent trauma. DIAGNOSES: Ultrasound suggested the possibility of a liver abscess and computed tomography (CT) examination revealed a giant space occupying lesion (SOL) (approximately 16 cm) in the right hepatic lobe, indicating a hepatic tumor or abscess. The patient did not respond to antibiotics and blood transfusion. Liver needle biopsy revealed blood clots suggestive of intratumoral hemorrhage, possibly of a hepatic hemangioma.
INTERVENTIONS: Interventional radiologic approach revealed active bleeding at the phrenic artery that supplied the liver SOL. Thus, embolization was performed and re-angiography showed no active bleeding from the embolized vessel. The patient became afebrile, but fever recurred the next day. Hence, an exploratory open right hemihepatectomy was undertaken and the intraoperative frozen biopsy confirmed hepatic hemangioma with internal hemorrhage, but malignancy could not be excluded for some focal tissues. Postoperative pathology report confirmed the diagnosis of hepatic hemangioma with internal hemorrhage and excluded malignancy. OUTCOMES: The fever subsided and the patient was discharged in good health. A follow-up CT performed three months, postoperatively, indicated compensatory growth of the left hepatic lobe. LESSONS: This case demonstrates that the diagnosis of hepatic hemangioma with internal hemorrhage can be confirmed by needle biopsy and surgical resection is the optimal treatment for such a lesion.

Introduction

Hepatic hemangioma is the most common type of benign liver tumor, with the larger tumors being cavernous hemangiomas,[ and rarely transform into malignancies.[ Although the reported incidence of hepatic hemangioma with ruptured bleeding was found to be low (1–4%),[ the mortality is rather high (36–39%).[ Meanwhile, the incidence of a hepatic hemangioma with internal hemorrhage is even rarer, with only sporadic cases reported to date[; therefore, there are limited experiences that we can refer to for their management. Surgical treatment is the predominant therapeutic approach for a hepatic hemangioma with ruptured bleeding[ but there are certain risks involved.[ Recently, it was reported that interventional embolization can improve or even control hepatic hemangioma with ruptured bleeding, which may avoid or prepare for further surgical treatment[; however, the therapeutic efficacy is still unclear.[ In the current report, we describe the findings and management approaches in a female patient with a giant liver space occupying lesion (SOL), confirmed as internal hemorrhage of the lesion via liver needle biopsy. She was managed by 2 interventions—initial embolization of the feeder vessel, and surgical resection undertaken later finally resolved her symptoms and cured the patient.

Case report

A 52-year-old female was admitted on October 24, 2016 with a chief complaint of “fever for over 20 days.” She provided a negative history for any medical condition (including hepatitis), recent trauma, or oral contraceptive use. Twenty days prior to the admission, the patient suffered from chills and fever with no obvious cause, with a body temperature between 38.5 and 39.0°C. The fever usually began from the afternoon, every day, with all the other vital signs remaining stable. Physical examination indicated no abdominal pain, distension, or tenderness, and no percussion pain in the hepatic area. Color Doppler ultrasound had been done elsewhere and showed a 15 × 13 cm area of nonhomogeneous echo in the right hepatic lobe; the area was unclearly demarcated from the nearby tissues, showed no clear capsule, and contained a few liquefied spots; thus, the possibility of a liver abscess was considered. Computed tomography (CT) examination revealed a 16 × 12 cm mass with mixed densities in the right hepatic lobe (Fig. 1), irregular enhancement during the arterial phase (Fig. 2), and no significant changes during the balance and delayed phase (Fig. 3). Laboratory testing results showed a hemoglobin (Hb) level of 6.3 g/L, a neutrophil percentage of 86.8, a normal total white blood cell count, normal hepatic function, no history of hepatitis B, and a normal alpha fetoprotein level. We believed that anemia was a result of the fever in a patient with liver abscess. However, after the administration of antibiotic treatment, there was no improvement of fever (Fig. 4). Although blood transfusion was provided, the anemia was not resolved. Ultrasound-guided liver needle biopsy tissue specimens were blood clots. Based on the biopsy results, we considered a diagnosis of intratumoral hemorrhage. Since the lesion was huge (involving the secondary porta hepatis) and the duration of fever and anemia was long, we presumed direct surgery might pose a great risk and we decided to adopt a relatively less invasive interventional therapy first after deliberations with colleagues.

Figure 1

Preoperative computed tomography.

Figure 2

Preoperative computed tomography arterial phase.

Figure 3

Preoperative computed tomography venous phase.

Figure 4

Body temperature before surgery.

Interventional therapy

Five days after hospital admission, we performed the intervention therapy. The hepatic arteriography indicated a giant SOL in the right hepatic lobe supplied by the right phrenic artery. Obvious contrast agent overflow was observed around the phrenic artery after angiography (Fig. 5); thus, a possibility of ruptured bleeding of the phrenic artery was considered and coil embolization was performed. Reangiography indicated no obvious contrast agent outflow (Fig. 6). Patients were administered with routine postoperative treatments and no fever occurred on the day of the surgery. On day 1, postoperatively, the peak body temperature was 37.5°C and the Hb level rose to 8.1 g/L; however, on day 3, her body temperature increased to 39.0°C. Following a 2-day period of steady body temperature, her body temperature again rose to around 39.0°C (Fig. 7) and Hb level also gradually decreased and was maintained at about 7.4 g/L. After consultation with colleagues, we concluded that the interventional embolization was incomplete and a vascular recanalization was likely to occur; therefore, we had to consider surgical treatment.

Figure 5

DR before embolism. DR = digital radiography.

Figure 6

DR after embolism. DR = digital radiography.

Figure 7

Body temperature report after intervention.

Surgical treatment

On November 21, 2016, we performed the surgical exploration. An experienced surgeon conducted the procedure under general anesthesia with tracheal intubation. A 15-cm oblique subcostal incision was made along the right rib and the abdominal cavity was opened in a layer-by-layer manner. The abdominal findings indicated no effusion or hemorrhage in the abdominal cavity and a giant mass in the right hepatic lobe (approximately 16 × 10 cm), mainly located at segments VII and VIII and involved the majority of segments V and VI (Couinaud classification) was observed. Local texture of the mass that occupied nearly the entire right hepatic lobe was rather hard and the surface was grey in color. No obvious exudation or adhesion was seen at the tissues surrounding the mass and the right hepatic lobe was significantly enlarged. Thus, we decided to perform a right hemihepatectomy. The operating time was 3 h and the intraoperative hemorrhage amount was 200 mL. After resection, we dissected the occupying mass and found solid tissues and blood clots inside. Intraoperative frozen biopsy confirmed the diagnosis of hepatic hemangioma but could not rule out the possibility of malignancy transformation of the consolidated tissues (intraoperative frozen biopsy was performed by an experienced pathologist) (Fig. 8). The postoperative secondary pathology report confirmed the diagnosis of hepatic hemangioma with internal hemorrhage and excluded malignant transformation (Fig. 9). Routine treatments were administered postoperatively and the patient's body temperature returned to normal on the day of the surgery (Fig. 10). The fever did not recur and her anemia gradually improved. No hemorrhage, bile leakage, or other complications were seen. Nine days after the operation, the patient recovered enough to be discharged. One month postoperatively, significant enlargement and growth of the resected liver were observed (Fig. 11) and indices of the blood routine test were restored to their normal levels.

Figure 8

Intraoperative freezing report.

Figure 9

Postoperative pathology report.

Figure 10

Postoperative temperature report.

Figure 11

Postoperative computed tomography.

Discussion

Hepatic hemangioma is the most common benign liver tumor, with the majority being cavernous hemangiomas,[ with little chance of malignant transformation.[ In general, a hepatic hemangioma with a diameter <4 cm is asymptomatic and prone to be neglected, being discovered only as an incidental imaging finding. The diagnosis of a hepatic hemangioma is dependent on ultrasound, CT, and magnetic resonance imaging, all of which provide a diagnostic accuracy of >90%.[ Hepatic hemangiomas with a diameter >4 cm are regarded as giant hemangiomas[ and can cause symptoms such as abdominal discomfort[ and other catastrophic complications including rupture, internal hemorrhage, coagulation disorder, etc. If not promptly and appropriately managed, all the above complications are associated with increased risk of mortality.[ Reports on hepatic hemangioma with internal hemorrhage are rather rare; only a few are available worldwide.[ In general, the ultrasound manifestations of hemangiomas are slightly enhanced echoes and have relatively clear demarcation unless complicated with hemorrhage, fibrosis, or calcification when hypoechoic changes are observed.[ In nonhemorrhagic hemangiomas, the typical features of enhanced CT are swift enhancement and slow expurgation and when complicated with hemorrhage, the images may be manifested as medium hyperintensity. However, since this disease is rare and little experience is available regarding its clinical diagnosis, it is difficult for us to differentiate it from liver abscess, hepatic malignancies, or other diseases. Through liver needle biopsy, we confirmed the diagnosis of internal hemorrhage in this patient, which shows that liver needle biopsy is not contradicted for hepatic internal hemorrhage.

Surgical treatment is the most definitive and effective therapeutic approach for hepatic hemangioma, but is associated with a high risk for morbidity and mortality.[ It is reported that the operative mortality rate of a ruptured hemangioma is about 36.5%.[ For hemangiomas with spontaneously ruptured bleeding, interventional therapies are reported to achieve auxiliary or ideal therapeutic effects[; therefore, we first adopted interventional approaches and found that the right phrenic artery was the blood supply vessel of the hemangioma. This condition, never seen in any previous literature, might share the same underlying pathogenesis of the hepatic carcinoma in which the huge and fast-growing tumor opens and dilates the collaterals between the phrenic and hepatic arteries.[ After embolization of the phrenic artery, repeated observations all indicated no active internal hemorrhage while the patient experienced no fever and her symptoms of anemia was also slightly improved within a short period of time. However, these favorable changes did not last long, necessitating the option of surgical resection for the tumor. After tumor resection, we could neither visually confirm the characteristics of the tumor nor by the intraoperative frozen specimen. Finally, the hepatic hemangioma was confirmed pathologically, postoperatively.

Based on the above information, we have reached the following conclusions: hepatic hemangioma with internal hemorrhage is extremely rare; abnormal blood supply may arise when the tumor grows rapidly; liver needle biopsy facilitates the diagnosis when there is a dilemma; and the efficacy of interventional approach is not adequate for hepatic hemangioma with internal hemorrhage, while surgical resection is still the most reliable therapeutic approach.