Open Access

Hepatic Sclerosed Hemangioma: a case report and review of the literature

  • Shunsuke Miyamoto1,
  • Akihiko Oshita1, 2Email author,
  • Yutaka Daimaru3,
  • Masaru Sasaki1,
  • Hideki Ohdan2 and
  • Atsushi Nakamitsu1
BMC Surgery201515:45

https://doi.org/10.1186/s12893-015-0029-x

Received: 21 May 2014

Accepted: 28 March 2015

Published: 17 April 2015

Abstract

Background

Although cavernous hemangioma is one of the most frequently encountered benign hepatic neoplasms, hepatic sclerosed hemangioma is very rare. We report a case of hepatic sclerosed hemangioma that was difficult to distinguish from an intrahepatic cholangiocarcinoma by imaging studies.

Case presentation

A 76-year-old male patient with right hypochondralgia was referred to our hospital. Abdominal ultrasonography revealed a heterogeneously hyperechoic tumor that was 59 mm in diameter in segment 7 of the liver. Dynamic computed tomography showed a low-density tumor with delayed ring enhancement. Gadolinium-ethoxybenzyl-diethylenetriamine pentaacetic acid-enhanced magnetic resonance imaging (EOB-MRI) demonstrated a low-signal intensity mass with ring enhancement on T1-weighted images. The mass had several high-signal intensity lesions on T2-weighted images. EOB-MRI revealed a hypointense nodule on the hepatobiliary phase. From these imaging studies, the tumor was diagnosed as intrahepatic cholangiocarcinoma, and we performed laparoscopy-assisted posterior sectionectomy of the liver with lymph node dissection in the hepatoduodenal ligament. Histopathological examination revealed a hepatic sclerosed hemangioma with hyalinized tissue and collagen fibers.

Conclusion

Hepatic sclerosed hemangioma is difficult to diagnose preoperatively because of its various imaging findings. We report a case of hepatic sclerosed hemangioma and review the literatures, especially those concerning imaging findings.

Keywords

Hepatic Sclerosed Hemangioma US CT MRI FDG-PET

Background

The preoperative diagnosis of hepatic sclerosed hemangioma is very difficult, even with recent developments in radiological modalities, because it is an extremely rare benign disorder and its radiological features resemble those of hepatic malignancies such as cholangiocarcinoma and metastatic liver cancer [1,2]. We report a case of a hepatic sclerosed hemangioma, that had been preoperatively misdiagnosed as an intrahepatic cholangiocarcinoma and been resected, and review the relevant literature, especially summarizing the imaging findings of hepatic sclerosed hemaigioma.

Case presentation

A 76-year-old male patient had consulted a doctor for upper abdominal pain 16 months before being referred to us and had been followed up. Because plain computed tomography (CT) revealed a space-occupying lesion in the liver, he was referred to our hospital. A laboratory workup on admission showed that total bilirubin, aspartate aminotransferase, alanine aminotransferase, alkaline phosphatase, gamma-glutamyl transpeptidase, albumin, and creatinine were all within normal ranges. Tumor markers including alpha-fetoprotein, protein induced by vitamin K absence or antagonist-II, carcinoembryonic antigen, and carbohydrate antigens 19–9 were also within the normal limits (Table 1).
Table 1

Review of imaging features for Hepatic Sclerosed Hamangioma

Case

Year

Author

Age

Gender

Location

Size (mm)

US

Plain CT

Dynamic CT

MRI (T1/T2)

PET-CT

Preoperative diagnosis

Final diagnosis

1

1995

Ishii

81

F

S3

25

Low

Low

Ring enhanced

Low/Low

Not determined

sclerosed hemangioma

2

1995

Haratake

64

F

S8

26

Low

Ring enhanced

Metastatic liver cancer

sclerosed hemangioma

3

1996

Kobayashi

49

F

S7

22

High

Low

Ring enhanced

Low/High

Not determined

sclerosed hemangioma

4

1998

Ukai

66

F

S6

10

Low

Low

Ring enhanced

Low/High

Hepatocellular carcinoma

sclerosed hemangioma

5

2000

Yamashita

67

F

S4

50

Low

Low

Ring enhanced

High/High

Metastatic liver cancer

sclerosing hemangioma

6

2001

Okada

77

M

S8

23

High

Low

Not enhanced

Metastatic liver cancer

sclerosed hemangioma

7

2001

Aibe

67

F

S4

40

Low

Not enhanced

High/High

Metastatic liver cancer

sclerosed hemangioma

8

2003

Hayakawa

70

F

S2

30

Low

Low

Ring enhanced

Low/Iso-High

Not determined

sclerosed hemangioma

9

2005

Morikawa

66

M

S8

50

Iso

Low

Ring enhanced

Low/High

Not accumulated

Not determined

sclerosed hemangioma

10

2005

Lee

65

F

S6

53

Ring enhanced

Low/High

Hepatocellular carcinoma

sclerosing hemangioma

11

2005

Okamoto

50

F

S3

30

Low

Low

Ring enhanced

sclerosed hemangioma

sclerosed hemangioma

12

2006

Hamatsu

59

M

S8

25

High

Low

Ring enhanced

Metastatic liver cancer

sclerosed hemangioma

13

2006

Hayashi

82

F

S2/3

55

High

Low

Not enhanced

Low/High

Gastric submucosal tumor

sclerosed hemangioma

14

2006

Iida

77

F

S2

39

High

Low

Ring enhanced

Low/Low-High

Not accumulated

Not determined

sclerosing hemaigioma

15

2007

Sawai

67

F

Right robe

145

Low

Low

Ring enhanced

Low/High

Not determined

sclerosed hemangioma

16

2008

Kaji

65

F

S5

25

Low

Low

Ring enhanced

Low/Iso-High

Cholangiocarcinoma

sclerosed hemangioma

17

2008

Tsumaki

70

F

S8

47

Low

Low

Ring enhanced

Low/High

Liver sclerosed hemangioma

sclerosed hemangioma

18

2008

Mori

77

F

S6

100

High

Low

Not enhanced

Low/High

Cholangiocarcinoma

sclerosed hemangioma

19

2010

Yoshida

75

F

S5/6

37

High

Low

Ring enhanced

Low/High

Not accumulated

Cholangiocarcinoma

sclerosing hemaigioma

20

2010

Usui

57

F

S2

17

Low

Low

Ring enhanced

Low/High

Metastatic liver cancer

sclerosed hemangioma

21

2010

Jin

52

M

S6/7

38

Ring enhanced

Low/High

Hepatocellular carcinoma

sclerosed hemangioma

22

2010

Hida

75

F

S5/6

30

High

Ring enhanced

Low/High

Metastatic liver cancer

sclerosed hemangioma

23

2011

Miyaki

60’s

F

S3

30

Low

Low

Low/High

Liver sclerosed hemangioma

sclerosed hemangioma

24

2011

Kitami

72

F

S3

55

Low

Low

Ring enhanced

Low/High

Cholangiocarcinoma

sclerosed hemangioma

25

2011

Tanaka

71

M

S6

15

High

Low

Ring enhanced

Hepatocellular carcinoma

sclerosed hemangioma

26

2011

Mikami

74

F

S2

22

Low

Low

Ring enhanced

Low/High

Not accumulated

Not determined

sclerosed hemangioma

27

2011

Shin

50

M

Right robe

100

Iso-Low

Low

Ring enhanced

Low/High

Not accumulated

Liver sclerosing hemangioma

sclerosing hemangioma

28

2012

Wakasugi

61

F

S2, S5

25,5

Low

Ring enhanced

Low/High

Metastatic liver cancer

sclerosed hemangioma

29

2012

Yamada

75

M

S8

11

Low

Ring enhanced

Low/High

Not accumulated

Metastatic liver cancer

sclerosed hemangioma

30

2013

Song

63

F

S2/3

91

Low

Ring enhanced

Not determined

sclerosing hemangioma

31

2013

Shimada

63

M

S8

10

Low

Ring enhanced

Low/High

Atypical hemangioma

sclerosed hemangioma

32

2015

OUR CASE

76

M

S6/7

59

High

Low

Ring enhanced

Low/High

Cholangiocarcinoma

sclerosed hemangioma

Abdominal ultrasonography (US) revealed a well-defined, heterogeneously hyperechoic mass that was 59 mm in diameter in segment 7 of the liver (Figure 1). Plain CT revealed a low-density 60-mm sized mass with an irregular margin. Dynamic CT revealed early ring enhancement in the peripheral part on the arterial phase and internal heterogeneous enhancement on the delayed phase (Figure 2). Gadolinium-ethoxybenzyl-diethylenetriamine pentaacetic acid-enhanced magnetic resonance imaging (EOB-MRI) showed that the tumor had low-signal intensity on T1-weighted images and that the mass had some high-signal intensity foci in the tumor on T2-weighted images. EOB-MRI showed no uptake in the corresponding area on the hepatobiliary phase and ring enhancement in the peripheral part on the arterial phase and the portal phase (Figure 3).
Figure 1

Abdominal ultrasonography (US). US showed a heterogeneously hyperechoic mass in segment 7 of the liver.

Figure 2

Abdominal computed tomography (CT). (a) plain, (b) arterial phase, (c) portal venous phase, and (d) delayed phase. Plain CT showed a low-density mass. Dynamic CT showed the ring enhancement in the peripheral part on the arterial phase (arrow).

Figure 3

Magnetic resonance imaging (MRI). (a) T1-weighted image, (b) T2-weighted image, and (c) ethoxybenzyl (EOB)-MRI on the hepatobiliary phase. The tumor had low-signal intensity on T1-weighted and some high-signal intensity nodules in the tumor on T2-weighted images. EOB-MRI showed no uptake in the corresponding area on the hepatobiliary phase.

Laparoscopy-assisted posterior sectionectomy and cholecystectomy including lymph node dissection in the hepatoduodenal ligament were performed for a preoperative diagnosis of intrahepatic cholangiocarcinoma. The resected specimen revealed a white solid mass, sized 61 × 46 mm. The cut surface of the tumor was elastic, soft, and homogeneous with the smooth margin including some faint red spots up to 10 mm in size (Figure 4a).
Figure 4

Resected specimen. (a) Surface of slice. The cut surface of the tumor reveals a white solid mass (61 × 46 mm in size) that was elastic, soft, and homogeneous with a smooth margin including some faint red spots, sized 1 cm. (b) Hematoxylin and eosin staining (magnification, ×100). The tumor was composed of fibrous connective tissue highlighted with collagen fiber and various sizes of cavernous hemangioma tissue with some hyaline degeneration secondary to thrombus, necrosis, or cicatrization.

Histopathological examination showed that the tumor was composed of fibrous connective tissue highlighted with collagen fibers and various sizes of cavernous hemangioma tissue with some hyaline degeneration secondary to thrombus, necrosis, or cicatrization, resulting in a hepatic sclerosed hemangioma (Figure 4b).

The postoperative course was uneventful. The patient was discharged on postoperative day 6.

Discussion

Hepatic sclerosed hemangioma, first reported by Ishii in 1995 [1], is a rare disease, detected and reported in only 2 out of 1000 cases on autopsy [3]. We found only 9 cases in PubMed by manual searching for the terms “hepatic, sclerosed, hemangioma” and “hepatic, sclerosing, hemangioma” from January 1983 to January 2015. Additionally, we found 22 cases in ICHUSHI, a bibliographic database established in 1903 and being updated by the Japan Medical Abstracts Society, contains bibliographic citations and abstracts from more than 2500 biomedical journals and other serial publications published in Japanese. The 32 cases, including our case, are summarized in Table 1 [1,4-33].

Hepatic sclerosed hemangioma is caused by degenerative changes such as thrombus formation, necrosis, and scar formation of liver cavernous hemangioma, but the mechanism for degenerative changes in the hepatic cavernous hemangioma has not been well clarified at present [34].

Concerning the imaging studies, Doyle et al. summarized the imaging findings of 10 hepatic sclerosed hemangioma lesions and found the characteristic features to include a geographic pattern, capsular retraction, decrease in size over time, loss of previously seen regions of enhancement [2]. And additional characteristic, features included the presence of transient hepatic attenuation difference, ring enhancement, and nodular regions of intense enhancement as seen in typical hemangioma. In our series reviewed the average size of the hepatic sclerosed hemangiomas was 42.3 mm, ranging from 10 to 145 mm. Abdominal US showed a hyperechoic mass in 11 cases and a hypoechoic tumor in 13 cases. Plain CT was likely to show a low-density mass, and dynamic CT showed ring enhancement, resembling metastatic liver cancer or intrahepatic cholangiocarcinoma, in 27 of 31 reported cases. MRI showed a low-intensity signal in 24 of 26 reported cases on T1-weighted images and a high-intensity signal in 22 of 26 reported cases on T2-weighted images. The radiological features revealed by dynamic CT and MRI resembled those of hepatic malignancies, leading to preoperative misdiagnosis. Whereas, [18F]-fluorodeoxyglucose positron emission tomography (FDG-PET), performed in just 6 cases, showed no accumulation of [18F]-FDG (Table 1). FDG-PET could be helpful in preoperative diagnosis to distinguish benign sclerosed hemangioma from malignant tumors such as intrahepatic cholangiocarcinomas or metastatic liver cancers. We may have had to perform FDG-PET preoperatively.

Surgical resection for hepatic sclerosed hemangioma is controversial. Most of the tumors reported were resected due to a preoperative misdiagnosis of malignancy (Table 1). To make a definite diagnosis of such hepatic tumors, percutaneous needle biopsy is not acceptable because of the possibility of dissemination of the cancer cells if the tumor is malignant. Therefore we would suggest that hepatic resections are chosen for the management of hepatic sclerosed hemangioma at present.

Makhlouf and Ishak compared the findings of sclerosed hemangioma and sclerosing cavernous hemangioma. According to their theory, recent hemorrhages and hemosiderin deposits rich in mast cells are present in the sclerosing hemangioma. While, fibrosis, increased elastic fibers, and dystrophic or psammomatous calcifications with a decreased number of mast cells can be observed in the sclerosed hemangioma [35]. Our case showed a fibrous connective tissue highlighted with collagen fibers and various sizes of cavernous hemangioma tissue with some hyaline degeneration. These findings are consistent with features of hepatic sclerosed hemangioma, resulting in the final diagnosis.

Conclusion

We report a case with a hepatic sclerosed hemangioma. Although it is a rare disease, it is important to distinguish hepatic sclerosed hemangioma from hepatic malignancies. However, it is extremely difficult to diagnose precisely from imaging studies. If the possibility of a malignant tumor cannot be ruled out, hepatic resection might be selected for diagnostic therapy.

Consent

Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor of this journal.

Abbreviations

EOB-MRI: 

Gadolinium-ethoxybenzyl-diethylenetriamine pentaacetic acid-enhanced magnetic resonance imaging

CT: 

Computed tomography

US: 

Ultrasonography

FDG-PET: 

[18F]-fluorodeoxyglucose positron emission tomography

Declarations

Authors’ Affiliations

(1)
Department of Surgery, JA Hiroshima General Hospital
(2)
Department of Gastroenterological and Transplant Surgery, Applied Life Sciences, Institute of Biomedical and Health Sciences, Hiroshima University
(3)
Department of Pathology, JA Hiroshima General Hospital

References

  1. Ishii T, Takahara O, Sano I, Taniguchi H, Nakao S, Eida K, et al. Sclerosing Hemangioma of the liver. Nagasaki Med J. 1995;70(1):23–6.Google Scholar
  2. Doyle DJ, Khalili K, Guindi M, Atri M. Imaging features of sclerosed hemangioma. Am J Roentgenol. 2007;189(1):67–72.View ArticleGoogle Scholar
  3. Berry CL. Solitary “necrotic nodules” of the liver: a probable pathogenesis. J Chin Pathol. 1985;38(11):1278–90.View ArticleGoogle Scholar
  4. Haratake J, Arai K, Makino H. Hemangioma and hyalinized hemangioma. Jpn J Clin Med. 1995;7:336–8.Google Scholar
  5. Kobayashi S, Demachi H, Akakura Y, Miyata S, Konishi K, Tsuji M, et al. A case of sclerosing cavernous hemangioma of the liver. Jpn J Clin Radiol. 1996;41:567–70.Google Scholar
  6. Ukai K, Onidera H, Mikuni J, Ouchi K. A case of sclerosed hemangioma of the liver. Liver. 1998;39(9):638–42.Google Scholar
  7. Yamashita Y, Shimada M, Taguchi K, Gion T, Hasegawa H, Utsunomiya T, et al. Hepatic sclerosing hemangioma mimicking a metastatic liver tumor: report of a case. Surg Today. 2000;30:849–52.View ArticlePubMedGoogle Scholar
  8. Okada S, Tsuyama H, Kurita K, Onchi H, Inoue T, Kinoshita K, et al. A case of sclerosing hemangioma with cancer of the desending colon. J Jpn Surg Assoc. 2001;62(3):757–60.View ArticleGoogle Scholar
  9. Aibe H, Honda H, Kuroiwa T, Yoshimitsu K, Irie H, Tajima T, et al. Sclerosed hemangioma of the liver. Abdom Imaging. 2001;26:496–9.View ArticlePubMedGoogle Scholar
  10. Hayakawa T, Itou M, Taniguchi S, Ideno M, Konno T, Hatayama J. A case of sclerosing hemangioma of the liver. J Med Assoc South Hokkaido. 2001;38:180–3.Google Scholar
  11. Lee VT, Magnaye M, Tan HW, Thng CH, Ooi LL. Sclerosing haemangioma mimicking hepatocellular carcinoma. Singapore Med J. 2005;46(3):140–3.PubMedGoogle Scholar
  12. Morikawa M, Ishida M, Iida A, Katayama K, Yamaguchi A, Imamura Y. Sclerosed hemangioma of the liver mimicking metastatic liver cancer. J Jpn Surg Assoc. 2005;66(7):1698–702.View ArticleGoogle Scholar
  13. Okamoto E, Satou S, Takahashi Y, Ohshima N, Azumi T, Furuta K, et al. A case of hepatic sclerosed hemangioma in patient with fatty liver. J Shimane Med Assoc. 2005;25(3):60–4.Google Scholar
  14. Hamatsu T, Kuroda Y, Hunahashi S, Shima I, Iso Y. Sclerosed hemangioma of the liver mimicking metastatic liver tumor of colon cancer. J Jpn Surg Assoc. 2006;67(4):856–60.View ArticleGoogle Scholar
  15. Hayashi K, Ohara H, Kitajima Y, Tanaka S, Takada H, Imai E, et al. A case of hepatic sclerosed hemangioma mimicking gastric submucosal tumor. Kanzo. 2006;47(10):474–81.View ArticleGoogle Scholar
  16. Iida H, Tango Y, Tsutamoto Y, Harimura T, Tanaka K, Takao T, et al. A resected case of Sclerosed hemangioma. Jpn J Gastroenterol Surg. 2006;39(9):1493–7.View ArticleGoogle Scholar
  17. Sawai K, Ueda N, Senda K, Kimura T, Sawa T. A case of sclerosing hemangioma of the liver. J Jpn Surg Assoc. 2007;68(9):2293–8.View ArticleGoogle Scholar
  18. Kaji S, Koike N, Suzuki S, Harada N, Suzuki M, Hanyu F. A case of hepatic sclerosed hemangioma mimicking cholangiocell carcinoma. J Jpn Surg Assoc. 2008;69(5):1181–5.View ArticleGoogle Scholar
  19. Tsumaki N, Waguri N, Yoneyama O, Hama I, Kawahisa J, Yokoo K, et al. A case of hepatic sclerosed hemangioma with a significant morphological change over a period of 17 years. Kanzo. 2008;49(6):268–74.View ArticleGoogle Scholar
  20. Mori H, Ikegami T, Imura S, Shimada M, Morine Y, Kanemura H, et al. Sclerosed hemangioma of the liver: report of a case and review of the literature. Hepatol Res. 2008;38:529–33.View ArticlePubMedGoogle Scholar
  21. Yoshida T, Sugimachi K, Gion T, Soejima Y, Aijima S, Takatomi S, et al. A case of sclerosing hemangioma mimicking a malignant liver tumor. J Clin Surg. 2010;65(3):451–5.Google Scholar
  22. Usui T, Shiozawa S, Kim DH, Tsuchiya A, Kuhara K, Yokomizo H, et al. A case of sclerosed hemangioma mimicking a metastatic liver tumor. J Jpn Coll Surg. 2010;35(1):89–93.Google Scholar
  23. Jin S-Y. Sclerosed hemangioma of the liver. Korean J Hepatol. 2010;16(4):410–3.View ArticlePubMedPubMed CentralGoogle Scholar
  24. Hida T, Nishie A, Tajima T, Taketomi A, Aishima S, Honda H. Sclerosed hemangioma of the liver: possible diagnostic value of diffusion-weighted magnetic resonance imaging. Jpn J Radiol. 2010;28:235–8.View ArticlePubMedGoogle Scholar
  25. Miyaki D, Aikata H, Waki K, Murakami E, Hashimoto Y, Nagaoki Y, et al. Significant regression of a cavernous hepatic hemangioma to a sclerosed hemangioma over 12 years: a case study. Nihon Shokakibyo Gakkai Zasshi. 2011;108(6):954–61.PubMedGoogle Scholar
  26. Kitami C, Kawachi Y, Nishimura A, Makino S, Kawahara M, Nikuni K. Sclerosed hemangioma of the liver -report of a case-. J Jpn Surg Assoc. 2011;72(12):3120–4.View ArticleGoogle Scholar
  27. Tanaka T, Moriya T, Fukuda S, Yoshifuku Y, Fujino H, Miwata T, et al. A case of sclerosed hemangioma of the liver was difficult to differentiate hepatocellular carcinoma was found during examination of chronic hepatitis C. HBP Imaging. 2011;13(6):646–52.Google Scholar
  28. Mikami J, Tominaga M, Sendo H, Maeda Y, Fujino Y, Koma Y, et al. A case of hepatic sclerosing hemangioma mimicking a malignant liver tumor. J Jpn Surg Assoc. 2011;72(4):965–71.View ArticleGoogle Scholar
  29. Shin YM. Sclerosing hemangioma in the liver. Korean J Hepatol. 2011;17(3):242–6.View ArticlePubMedPubMed CentralGoogle Scholar
  30. Wakasugi M, Ueshima N, Akamatsu D, Tori M, Tsujimoto M, Nishida T. A case of multiple sclerosed hemangioma mimicking a metastatic liver tumor. J Clin Surg. 2012;67(12):1461–5.Google Scholar
  31. Yamada S, Shimada M, Utsunomiya T, Morine Y, Imura S, Ikemoto T, et al. Hepatic sclerosed hemangioma which was misdiagnosed as metastasis of gastric cancer : report of a case. J Med Invest. 2012;59:270–4.View ArticlePubMedGoogle Scholar
  32. Song JS, Kim YN, Moon WS. A sclerosing hemangioma of the liver. Clin Mol Hepatol. 2013;19(4):426–30.View ArticlePubMedPubMed CentralGoogle Scholar
  33. Shimada Y, Takahashi Y, Iguchi H, Yamazaki H, Tsunoda H, Watanabe M, et al. A hepatic sclerosed hemangioma with significant morphological change over a period of 10 years: a case report. J Med Case Rep. 2013;28(7):139.View ArticleGoogle Scholar
  34. Goodman ZD. Benign tumors of the liver. In: Okuda K, Ishak KG, editors. Neoplasm of the liver. Tokyo: Springer; 1987. p. 105–25.View ArticleGoogle Scholar
  35. Makhlouf HR, Ishak KG. Sclerosed hemangioma and sclerosing cavernous hemangioma of the liver: a comparative clinicopathologic and immunohistochemical study with emphasis on the role of mast cells in their histogenesis. Liver. 2002;22(1):70–8.View ArticlePubMedGoogle Scholar

Copyright

© Miyamoto et al.; licensee BioMed Central. 2015

This article is published under license to BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

Advertisement