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.
Hinweise
Competing interests
The authors declare that they have no competing interests.
Authors’ contributions
The first two authors contributed equally to this work. SM drafted the paper and collected date and reviewed the text. AO performed the operation, helped SM to draft the paper and made the final revision. YD diagnosed this disease. MS and AN assisted the operation. HO made the expert assistance. All authors read and approved the final manuscript.
Abkürzungen
EOB-MRI
Gadolinium-ethoxybenzyl-diethylenetriamine pentaacetic acid-enhanced magnetic resonance imaging
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
Anzeige
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).
×
×
×
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).
×
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.
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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.
Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made.
The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder.
The authors declare that they have no competing interests.
Authors’ contributions
The first two authors contributed equally to this work. SM drafted the paper and collected date and reviewed the text. AO performed the operation, helped SM to draft the paper and made the final revision. YD diagnosed this disease. MS and AN assisted the operation. HO made the expert assistance. All authors read and approved the final manuscript.
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