British Journal of Radiology (2008) 81, e37-e39
© 2008 British Institute of Radiology
doi: 10.1259/bjr/36041107
Multicystic cavernous haemangioma of the liver: ultrasonography, CT, MR appearances and pathological correlation
E-Y CHA, MD
1
K W KIM, MD
1
Y J CHOI, MD
1
J S SONG, MD
2
K J CHO, MD
2 and
M-G LEE, MD
1
1 Department of Radiology and Research Institution of Radiology, 2 Department of Pathology, University of Ulsan College of Medicine–Asan Medical Centre, 388-1 Pungnap-2 dong, Songpa-gu, Seoul 138-736, Korea
Correspondence: Kyoung Won Kim, Department of Radiology, University of Ulsan-Asan Medical Centre, 388-1 Pungnap-2 dong, Songpa-gu, Seoul 138-736, Korea. E-mail: kimkw{at}amc.seoul.kr
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Abstract
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Among the hepatic haemangiomas with atypical features when studied radiologically, the multicystic type is extremely rare. We report a case of multicystic hepatic haemangioma in a 62-year-old woman, which was found incidentally during ultrasound screening. Because the tumour showed atypical features on ultrasonography, CT and MRI, the correct diagnosis was not made until the surgery. In this report, the atypical radiological findings are illustrated and correlated with pathological findings.
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Introduction
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In most cases, radiological studies are sufficient for a definitive diagnosis of typical cavernous haemangiomas without referring to the pathological examination. However, hepatic haemangiomas may frequently exhibit atypical radiological features, and they may lead to misdiagnosis and confusion with other lesions. Of those atypical haemangiomas, the multicystic type is extremely rare, and, to our knowledge, fewer than five cases have been reported previously [1–4]. In this report, we present a case of multicystic hepatic haemangioma, comprising the multilocular cystic part in the periphery along with the core-like solid part in the centre. The atypical radiological features on ultrasonography (US), CT and MRI are illustrated and also correlated with pathological findings.
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Case report
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A 62-year-old woman was referred to our hospital for further evaluation of an incidentally detected hepatic mass on US screening at a local clinic. On admission, she presented with no symptoms and signs, and the physical examination showed no abnormalities. The laboratory data were within the reference ranges. US, CT and MRI were performed (Figure 1
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Figure 1. A multicystic hepatic haemangioma in a 62-year-old woman incidentally found on ultrasound screening. (a) Transverse sonogram reveals a well-demarcated complex mass composed of a multilocular cystic part in the periphery and a stellate echogenic part in the centre (arrow). One of the loculi is filled with high-echoic material (arrowhead). (b, c) Contrast-enhanced CT scans during the late arterial (b) and the delayed (c) phases show that while the multilocular cystic part in the periphery shows no contrast enhancement, the stellate central part (arrows) demonstrates gradual enhancement. One of the loculi filled with echogenic material on US also appeared as a hyperdense area on non-contrast CT scan (not shown). (d) The peripheral cystic part shows bright signal intensity and the central part (arrow) shows intermediate to high signal intensity on fat-suppressed T2 weighted MR image (TR/TE, 6356/104). (e) One of the loculi (arrowhead) is seen as slightly high signal intensity on T1 weighted image (TR/TE, 224/2.5), suggesting previous internal haemorrhage. It corresponds to an echogenic area on the sonogram. The central stellate area is isointense with hepatic parenchyma. (f) On cut section of a gross specimen, the tumour macroscopically consists of multicystic loculi separated by multiple septa with variable thickness. The central stellate area is reddish in colour (asterisk). (g) On low-power photomicrograph, the peripheral part consists of variable sized multicystic loculi, empty or filled with necrotic material. The loculi are encircled with a single layer of endothelium, compatible with haemangioma (H&E stain, original magnification, x1.25). (h) On low-power photomicrograph, the central stellate area consists of typical cavernous structures (H&E stain, original magnification, x40).
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US was performed on a commercially available scanner (HDI 5000 system; Philips Medical Systems, Bothell, WA) and revealed a 5.7x5.7x6.3 cm complex mass, comprising a multilocular cystic part in the periphery along with a stellate echogenic part in the centre. Although the majority of the cystic part appeared as echolucent areas with posterior acoustic enhancement, one of the loculi was filled with high-echoic material. Colour Doppler US failed to demonstrate intratumoral vascularity of the lesion.
Non-contrast and post-contrast CT scans were obtained with a multidetector row CT scanner (Sensation 16; Siemens, Erlangen, Germany). Post-contrast CT scans were obtained during the late arterial, the portal venous and the delayed phases. On CT scans, while the well-demarcated, multilocular cystic part in the periphery of the lesion showed no contrast enhancement, the stellate central part of the lesion demonstrated gradual contrast enhancement on the portal venous and delayed phase CT scans. One of the loculi filled with echogenic material on US also appeared as a hyperdense area on the non-contrast CT scan.
MR imaging was performed on a 1.5 T unit (Avanto; Siemens, Erlangen, Germany). On a fat-suppressed T2 weighted MR image (repetition time/echo time (TR/TE), 6356/104), the multilocular cystic part in the periphery of the lesion showed bright signal intensity and the stellate central part of the lesion showed intermediate to high signal intensity. On a T1 weighted image (TR/TE, 224/2.5), one of the loculi was seen as high signal intensity and corresponded to an echogenic area on US and hyperdense area on a non-contrast CT scan. The central stellate area was isointense with hepatic parenchyma.
An extended right hepatic lobectomy was performed under the tentative diagnosis of biliary cystadenoma or cystadenocarcinoma. On the cut section, the tumour measured 5.8x6.0x5.0 cm, was well demarcated without a capsule, had a very thin wall less than 1 mm in thickness, and was filled with necrotic material, serous fluid or thrombi. On histopathological examination, the periphery of the tumour consisted mainly of multicystic loculi, variable in size, encircled with a single layer of endothelium. The central stellate area consisted of typical cavernous structures. The histological diagnosis was multicystic cavernous haemangioma.
The patient's postoperative course was uneventful, and she was discharged 13 days after surgery.
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Discussion
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Because of widespread application of routine US screening and advances in cross-sectional imaging technology, hepatic haemangiomas are being detected more frequently than before. Of paramount importance is the differentiation of these tumours from other hepatic malignancies. Pathologically, haemangiomas are composed of many endothelium-lined vascular spaces separated by fibrous septa, resembling a sponge-like architecture, and the collective size of their constituent vascular spaces may vary [5]. They derive blood supply from the hepatic artery. In cases of typical haemangiomas, cross-sectional imaging findings, especially with dynamic contrast-enhanced studies, usually reflect the pathological characteristics of these tumours and suffice for a definitive diagnosis without referring to a real pathological examination. But there are many kinds of hepatic haemangiomas showing atypical features that make differential diagnosis difficult. These findings include non-enhancing central core and septa usually seen in large haemangiomas, calcifications, and fluid–fluid levels; these findings are presumably due to central fibrosis, central thrombosis, haemorrhage and hyalinization [6–10]. Of these atypical features of haemangiomas, multicystic appearance is extremely rare, and fewer than five cases have been reported before [1–4] to our knowledge.
In our case, US, CT, and MR imaging demonstrated a complex mass composed of a multilocular cystic part in the periphery along with a core-like solid part in the centre. One of the loculi was filled with high echoic material on US, which was also seen as high signal intensity on T1 weighted MR imaging, suggesting previous internal haemorrhage. The central core-like solid part showed gradual contrast enhancement on dynamic CT, but it was recognized as delayed contrast enhancement usually seen in fibrotic stroma rather than centripetal fill-in enhancement typical of haemangioma. The solid part did not show bright signal intensity on fat-suppressed T2 weighted MR imaging. The tumour was preoperatively misdiagnosed as biliary cystadenoma or cystadenocarcinoma, but it was also somewhat different from biliary cystadenoma or cystadenocarcinoma in that the solid part was located in the centre and not in the periphery. Although mesenchymal hamartoma may appear as a multilocular cystic mass with intervening solid septa [11], it was not included in the differential diagnoses because of rarity of this tumour in adults. When retrospectively reviewed, radiological findings corresponded to the macroscopic appearance of this tumour. Pathologically, while the periphery of the tumour consisted mainly of multicystic loculi with very thin walls that filled with necrotic material, serous fluid, or thrombi, the central stellate area was made of cavernous structures typical of hepatic haemangioma.
Among the previously reported cases of cystic cavernous haemangiomas of liver, one appeared as a unilocular cystic mass [1], and the other three as a mixture of solid and multilocular cystic masses [2–4], similar to our case. The pathogenesis of the cystic change of cavernous haemangioma is not clearly understood. Cystic degeneration by necrotic changes from infarction developing as a result of the occlusion of vessels or haemorrhage [2–4], coexistence of components of cavernous haemangioma and lymphangioma [2], and cystic degeneration induced by apoptosis [1] have been proposed as the cause.
In conclusion, the possibility of atypical haemangioma should be included even in the differential diagnoses of a predominantly cystic mass when some part of the cystic mass is enhanced on post-contrast CT and/or MR imaging.
Received for publication July 9, 2006.
Revision received September 28, 2006.
Accepted for publication November 20, 2006.
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Abstract
Introduction
