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ADOMINAL SURGERY

Official Journal of the American Society of Abdominal Surgeons, Inc.

This article originally appeared in the Winter 20011 / Spring 2012 issue of the Journal.

Giant Cavernous Hemangioma of the Liver: A Case Report and Review of Literature

Dr. Ali Bendjaballah
Dr. M. Taieb

Surgical Department
Ain Taya Hospital
Algiers, Algeria

Address for correspondence:
Dr. Ali Bendjaballah
Department of General Surgery
Ain Taya Hospital
Algiers, Algeria
Tel: +213-551-764-640
E-mail: DJABA6MED@hotmail.com


ABSTRACT

Hemangiomas are the most common benign tumors of the liver. Less than 10 % of cases become large enough to cause symptom. When hemangioma becomes symptomatic, it can be managed by surgery, arterial embolisation or radiofrequency.

We report here a case of giant hemangioma in a patient with chronic renal failure simulating abdominal tumor treated successfully by surgical resection (left atypical lobectomy of the liver). We also review the literature about the management of symptomatic hepatic hemangioma.

BACKGROUND

Hemangioma is a congenital vascular malformation and it is the most common benign tumor of the liver. Hemangiomas are mesenchymal in origin but some authorities consider them to be benign congenital hamartomas.

Malignant transformation has not been reported in hepatic hemangiomas, they may be left safely alone. Most hepatic Hemangiomas are small and asymptomatic at the time of diagnosis; only less than 10% of cases undergoes enlargement and some of them become huge enough to cause symptom.

We present here a case of a giant hepatic hemangioma developed at the left lobe of the liver in patient with chronic renal failure.

CASE REPORT

A 53-year old woman, followed for chronic renal failure since 10 years. A hepatic hemangioma was incidentally discovered in a physical examination by ultrasonography for 15 years ago. At that time the lesion size was smaller (3-4 cm) and it kept growing gradually for attaining a size of 22 cm and became localized in the pelvis.

Figure 1

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Figure 2

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Figure 3

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She was complaining of diffuse abdominal pain since several months and there was no history of any functional sign. On physical examination the patient was slightly pale without jaundice; bilateral lower extremities were free from edema. The abdomen was mildly distended by an isolated anterior mass which was oblong, mobile and slightly painful. There was no ascites and no collateral venous circulation.

Laboratories investigations revealed a mild anemia with moderate renal failure; liver function tests were normal. Hepatitis B and C virus markers were negative and alpha feto-protein level was in normal value.

Ultrasonography examination revealed a huge hyperechoic mass (about 22 cm of diameter) developed from the left lobe of the liver with areas of necrosis inside it and which was nearly occupying the whole abdominal cavity.

Computed tomography scan showed a huge hypodense mass of the left lobe of the liver measuring about 22 cm prolonged to the pelvis with a lot of areas of necrosis and thrombosis.

The diagnosis of giant hepatic hemangioma was established on the followed criteria:

  • there is no history of malignancy
  • no history of chronic disease of the liver
  • the ultrasonography finding and CT scan images were highly suspecting a diagnosis of hepatic hemangioma.

Laboratories liver investigations were normal. There was a discrete renal failure.

Patient underwent laparotomy and atypical left lhepatectomy was performed, she passed a smooth post operative period and she received a 900 cc of blood to restitute blood loss during operation.

She was discharged on seventh post operative day and was oriented to nephrology outpatient for her renal failure.

Patient was followed up on surgery OPD for 3 years and was doing well; all the previous pains were disappear.

The histopathology result confirms the diagnosis of giant cavernous hepatic hemangioma.

DISCUSSION

Hemangiomas of the liver are uncommon. In 2400 autopsies reviewed by Oschsner4 the incidence was found to be 2%; Henson5 of Mayo Clinic collected 35 hepatic hemangiomas from 1907–1954 inclusive. They are more common in the right lobe of the liver than in the left lobe.3

The lesion is predominantly found in female, about 4.5 times higher than the male.6 There was no specific age distribution4 but those presented clinically were usually around 50 years of age5 as noted in our patient.

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Photo 2

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Some authorities admit that hepatic hemangiomas are harmatomatous malformation.There is a high incidence of multiparity in association with these types of lesions; female sex hormones may have a role in their development.7

It is not frequent that large hemangiomas sequester and destroy platelets causing consumptive coagulopathy, symptomatic thrombocytopenia, known as Kasabach Merritt syndrome.8 Hepatic hemangiomas can occur as part of well-defined clinical syndromes.3

The hemangioma may ruptures spontaneously, or with minimal trauma and present as hemoperitoneum, especially when the tumor is close to the surface; the incidence is about 1-4 % and has been described in about 32 cases in the literature with a high mortality rate (36-69% of cases).9,10

The modalities used to aid in the diagnosis of hepatic hemangiomas include ultrasonography; dynamic contrastenhanced computed tomography; nuclear medicine studies using technetium (Tc) 99m-labeled RBCs, magnetic resonance imaging, hepatic arteriography and digital subtraction angiography.3

In general, the finding on ultrasonography of a suspected hemangioma should be diagnostically integrated with CT scan or MRI to insure a correct diagnosis.3

The authors continue to regard MRI as the diagnostic test of choice for hepatic hemangioma; Nuclear medicine studies may be used to confirm the diagnosis when a probable hemangioma is diagnosed on ultrasonography; and may also help to clarify the nature of lesion when the diagnosis is equivocal on CT or MRI.3

Multiple hemangiomas of the liver can occur in about 10% of cases.2 A giant hemangioma is considered when its size is more than 4 cm. Most of them remain stable and no symptom over time. But some of them may become symptomatic and the most common symptom was abdominal pain or discomfort.1

Goodman noted that symptoms are experienced by 40% of patient with 4 cm hemangiomas and by 90% of patient with 10 cm hemangiomas.11

Photo 3

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Photo 4

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Ishak reviewed 89 cases and found only 13.5% with clinical symptom.7 Right upper quadrant pain or fullness is the most common complain. In some cases pain is explained by thrombosis and infarction of the lesion, hemorrhage into the lesion, or compression of adjacent tissues or organs. In other cases pain is unexplained.3

For the symptomatic patients it is advocated that they should undergo a thorough evaluation to find out any other cause for the symptoms. If the hemangiomas are the cause of pain, these patients are candidates for surgical treatment.8 The size of lesion is not a criterion for resection during management of giant liver hemangioma. However the smaller giant hemangioma also can cause symptomatic manifestation.1

In our case the indication for surgical resection was: rapid increase in size; pain and discomfort for long duration despite analgesic drugs; high risk of thrombosis inside the tumor; high risk of rupture with hemoperitoneum.

Regarding liver biopsy, since 2001, hepatologists and surgeons have been increasingly resistant to include liver biopsy in the diagnostic workup of suspected hepatocellular carcinoma. The diagnosis of most lesions can be made by using combination of CT and MRI.12 Liver biopsy is only used when radiologic study result and alpha fetoprotein testing are equivocal.3

After surgery a few patients have post operative wound pain but for the majority of patients (96% of cases) surgical treatment can achieve the best result of symptom relief.1

The size and location of a lesion will influence the surgeon’s decision to perform either a segmental resection or an enucleation of the hemangioma. These procedures can be performed using an open approach but laparoscopic surgery can be performed in some cases. In the case of large lesions hepatic lobectomy can be indicated3 as in our case; we perform an atypical left hepatic lobectomy.

In general, both of two methods (surgical resection & enucleation) are safe and well tolerated by patients. Post operative morbidity is minimal, mortality rates of O% have reported in large series and the average length of hospital stay is 6 days.13

Surgical resection may not be possible in certain cases because of the massive or diffuse nature of lesion or the patient’s comorbidities.1

Besides surgical resection there are other modalities available to manage symptomatic hemangioma in the literature. Arterial embolisation is an option in such circumstances. Branches of hepatic artery can be embolized with polyvinyl alcohol and other substances.14,15

Embolisation results in shrinking of the tumor, thereby minimizing the risk of complications.3

There are several reports in the literature that arterial embolisation decreases tumor sizes and results in symptom relief. In a series of 98 patients who underwent arterial embolisation, the diameter of tumors was successfully decreased after 6 months of treatment. The clinical symptoms were relieved in all 53 symptomatic patients. But transient impairment of liver function was noted in 77 cases.15

In patient experiencing acute rupture or intratumoral hemorrhage arterial embolisation may be an appropriate initial step of therapy. Once the patient is stabilized formal surgical resection of the hepatic hemangioma can be performed.3 But some drawbacks with this technique were reported; Huang and all report in his series six cases of severe biliary and hepatic complications such as liver abscess, gallbladder necrosis, gangrene and bile duct structure after hepatic artery embolisation for hepatic hemangioma. Tumor size of these patients ranged from 3 to 6 cm in diameter. He concludes that regarding this severe destructive biliary damage this technique should be prohibited.16

Suzuki et al. suggested the use of preoperative arterial embolisation in patients with consumption coagulopathy related to intravascular coagulation in the hemangioma.17

Radiofrequency ablation: using both percutaneous and laparoscopic way has been performed successfully to improve abdominal pain in small numbers with symptomatic hepatic hemangioma.18-19 In a series of 12 patients with 15 hepatic cavernous hemangiomas (diameter was between 2.5cm and 9.5cm) were treated with radiofrequency ablation.23

Other techniques were mentioned in the literature

  • surgical ligation of feeding vessels: transhepatic compression sutures using polytetrafluoroethylene Pledgets and selective ligation of large feeding vessels have been described. In one case this technique successfully reduced intratumoral shunting that otherwise would have led to intractable cardiac failure.20
  • hepatic irradiation: with a dose of 30 Gy in 15 fractions over 3 weeks has been reported to produce complete regression of hepatic hemangioma with minimal morbidity.21
  • orthotopic liver transplantation: is occasionally offered to symptomatic patients with large or diffuse lesion. Several cases have now been reported in the literature.22

Finally, for huge symptomatic giant hemangioma the management by combination of both non-surgical and surgical modalities may reduce the size of lesion and make surgical procedure easier.

CONCLUSIONS

Hemangiomas are a congenital vascular malformations and the most common benign tumor of the liver. Most of them are smaller and asymptomatic. Ablation of large hemangiomas is to be undertaken with great caution and requires much experience in hepatic surgery. Surgical treatment is not indicated in the majority of stable or asymptomatic cases. In the contrary; for symptomatic lesions either early surgical or non surgical methods should be performed. Surgery can definitely relieve the symptoms and it should be preferred in patients without definite surgical contraindications and without high risk factors for surgery.

REFERENCES

1. Wen- Yao- Yin et al.: Early treatment for symptomatic giant hepatic hemangioma report of three cases and literature review. Medwell surgery journal 2(4):45-49,2007

2. K H Chew, A Rauff, Ailien Wee, W C Foong: cavernous hemangioma of the liver – A case report. Singapore Medical Journal, vol 23,No 1:49-51,1982

3. David C Wolf, MD, FACP, FACG, AGAF – Unnithan V Raghuraman, MD, FCRP, FACG, FACP Hemangioma, Hepatic: emedecine < Gastroenterology < Liver, Dec 22, 2008

4. Oschsner J L and Halpert B: cavernous hemangioma of the liver. Surgery 1958; 43:577-582.

5. Henson S W JR, Gray H K and Dockerly M B: Benign tumors of the liver. Surgery Obst Gyn. 1956; 103:327-331.

6. Schumacker H B and Baltimore: Hemangioma of the liver. Surgery 1942; 42:209-220.

7. Ishak K G and Rabin L: Benign tumors of the liver. Med. Clinic N America 1975; 59:995-1013.

8. Courtney, M, JR. Townsend, R. Daniel Beauchamp and B. Mark Evers et al. Sabiston Textbook of surgery 2004. The Basis of Modern Surgical Practice 17TH edition. Philadelphia: W B. Saunders Company, pp:1547.

9. Corigliano N, P. Mercantini and P.M. Amodio et al. Hemoperitoneum from a spontaneous rupture of a giant hemangioma of the liver. Report of a case. Surg. Today 2003; 33:459-463.

10. Griffa, B, V. Basilico and R. Belloti et al. Spontaneous rupture of giant subcapsular hemangioma of the liver with hemoperitoneum and hemorrhagic shock. A case report Chirur. Itali, 2005,57:389-392.

11. Goodman Z. Benign tumors of the liver. In: Okuda K, Ishak K G. Neoplasms of the liver. Tokyo: Springer-Verlag; 1987:105-125.

12. Bruix J, Sherman M, Llovet JM et al. Clinical management of hepatocellular carcinoma. Conclusion of the Barcelona -2000 EASL conference. European Association for the Study of the Liver. Journ. Hepatology. Sep. 2001; 35(35):421-430.

13. Arnoletti JP, Brodsky, Surgical treatment of benign hepatic mass lesions. Am. Surg. May 1999; 65(5): 431-433.

14. Zeng Q, Li Y, Chen Y, et al. Gigantic cavernous hemangioma of the liver treated by intra-arterial embolisation with pingyangycin-lipidol emulsion: a multi center study. Cardiovascular Intervention Radiol. Sept-Oct 2004; 27(5):481-5.

15. Srivastava DN, Ghandi D, Seith A, et al. Transcatheter arterial embolisation in the treatment of symptomatic cavernous hemangioma of the liver: a prospective study. Abdom Imaging. Sep-Oct 2001; 26(5)/510-4

16. Huang, X Q., Z Q. Huang and W D. Duan et al. 2002. Severe biliary complications after hepatic artery embolisation. World. J. Gastroenterol. 8:119-123

17. Suzuki, H, Y. Numira and J. Kamiya et al. 1997. Preoperative Transcatheter arterial embolisation for giant cavernous hemangioma of the liver with consumption coagulopathy. Am J. Gastroenterol. 92:688-691.

18. Tak WY, Park SY, Jeon SW et al. Ultrasonography – guided percutaneous radiofrequency ablation for treatment of a huge symptomatic cavernous hemangioma. J Clin Gastroenterol. Feb 2006; 40(2):167-170. Fan R F, Chai FL, He GX, et al. Laparoscopic radiofrequency ablation of hepatic cavernous hemangioma. A preliminary experience with 27 patients. Surg. Endosc. Feb 2006; 20(2)281-5.

19. Rokitansky AM, Jakl RJ, Gopfrich H, et al. Special compression suture: a new surgical technique to achieve a quick decrease in shunt volume caused by diffuse hemangiomatosis of the liver. Pediatr Surg Int. Nov 1998; 14(1-2):119-121.

20. Biswal MB, Sandhu M, Lal P, et al. Role of radiotherapy in cavernous hemangioma of the live. Indian J Gastroenterol. Jul. 1995; 14(3):95-98

21. Tepetes K, Selby R, Webb M, et al. Orthotopic liver transplantation for benign hepatic neoplasms. Arch Surg. Feb 1995; 130(2):153-156.

22. Cui, Y, L.Y. Zhou and M K Dong et al. 2003 Ultrasonography guided percutaneous radiofrequency ablation for hepatic cavernous hemangioma. World J. Gastroenterol. 9:2132-2134.



Journal CoverOFFICIAL PUBLICATION OF:
The American Board of Abdominal Surgery
The American Society of Abdominal Surgeons, Inc.
Foundation for Abdominal Surgery

Louis F. Alfano, Jr., M.D., Editor-in-Chief
Diane M. Pothier, Executive Editor and
Director of Continuing Medical Education


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Figure 1: showed a giant hemangioma of the left lobe of liver with big areas of necrosis and thrombosis.

Figure 2: showed a giant hemangioma of the left lobe of liver with big areas of necrosis and thrombosis.

Figure 3: showed a giant hemangioma of the left lobe of liver with big areas of necrosis and thrombosis.

Photo 1:
Showed a giant hepatic hemangioma exteriorized from the abdominal cavity.

Photo 2:
The same hemangioma from another angle; see the big area of necrosis and thrombosis on the surface.

Photo 3:
Giant hepatic hemangioma attached to the liver by a thin layer of hepatic tissue.

Photo 4:
Total removal of a giant hepatic hemangioma.