Reprint requests to: Dr. Jiunn-Ming Lii Department of Radiology, Taipei City Hospital. No. 10, Sec. 4, Ren Ai Road, Taipei 106, Taiwan, R.O.C.

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1 中華放射醫誌 Chin J Radiol 200; 35: Coexisting Hepatic Hemangioma Treated Incidentally with Percutaneous Ethanol Injection and Transarterial Chemoembolization in a case of Hepatocellular Carcinoma Tom Chen Ran-Chou Chen,2 Wei-Tsung Chen Chou-Shian Li Shyi-Jye Duh Jiunn-Ming Lii Department of Radiology, Taipei City Hospital Department of Biomedical Imaging and Radiological Sciences 2, National Yang-Ming University Hepatic hemangiomas are not routinely treated except for large symptomat ic lesions. Transarterial chemo-embolization (TACE) and percutaneous ethanol injection (PEI) are often used to treat hepatocellular carcinoma (HCC). We report a patient with underlying HCC and a coexisting small hemangioma that was inadvertently treated with both PEI and TACE because of misdiagnosis as an additional HCC. The hemangioma persisted with no visible imaging changes, nor complications. It is important to identify hemangiomas in HCC patients to avoid unnecessary treatment. Hemangioma is the most common benign tumor affecting the liver. They rarely cause any symptoms unless progressive growth causes pressure effect or rupture. Symptomatic lesions are generally treated successfully by surgical resection [], and some also claim satisfactory result with embolization [2]. However, Chen et al. also reported incidental transarterial chemoembolization (TACE) of six cases of hemangiomas with concurrent hepatocellular carcinoma (HCC), which appeared to have no effect on the hemangiomas [3]. The other treatment option for the hepatic hemangioma includes radiofrequency ablation [4, 5]. To the best of our knowledge, the combined PEI and TACE for a hepatic hemangioma were not reported previously. We hereby present a rare case of HCC coexisting with an undiagnosed small hemangioma, which was inadvertently treated with one course of percutaneous ethanol injection therapy (PEI) and three sessions of TACE. The hemangioma persisted despite these therapies, and its radiographic features appear to be unaltered. Case Report Reprint requests to: Dr. Jiunn-Ming Lii Department of Radiology, Taipei City Hospital. No. 0, Sec. 4, Ren Ai Road, Taipei 06, Taiwan, R.O.C. A 63-year-old female patient was diagnosed with hepatocellular carcinoma (HCC), on a background of hepatitis C related liver cirrhosis (Childs A). Her past history includes hyperthyroidism treated with thyroidectomy, and pelvic inflammatory disease. Screening ssonography revealed a.5cm nodular lesion in the left hepatic lobe (segment 2, S2). The tumor was then confirmed by CT scan. The tumor received biopsy and histological examination, which was proved to be a well-differentiated hepatocellular carcinoma (HCC), and PEI was performed.

2 26 Hemangioma treated with TACE & PEIT A total of 2 ml pure ethanol over four sessions was injected under ultrasound guidance. Follow up sonography revealed a previously undetected hypoechoic tumor (.2cm in diameter) in S2 in addition to the treated tumor in the same segment, and this tumor enhanced strongly in postcontrast CT images (Fig. a). PEI was then arranged for the treatment of this second S2 tumor, at that time thought to be a recurrent HCC. It was injected with a total of ml of pure ethanol under ultrasound guidance over four sessions. Patient remained well during and after the PEI treatments. Follow up CT af ter PEI showed post-pei changes with hypodensity covering the entire nodule and its surrounding liver parenchyma in the preand postcontrast images, which is compatible with post-pei changes (Fig. b). However the lesion still enhanced well in post-contrast CT images (Fig. b), which indicated tumor viability. In addition, new hypoechoic hepatic tumors (0.6cm and.cm) were also found in right anterior superior segment (S8). TACE was ordered to treat what was thought to be a viable tumor in S2 and the newly detected S8 lesions. TACE was performed via the proper hepatic artery. A total of 20mg of doxorubicin, 6mg of mitomycin, and 5mL of lipiodol was injected. Small pieces of Gelfoam sponge was also injected until stasis of the feeding arteries. The patient remained stable throughout and after the procedure. Non-contrast CT after the TACE showed heterogeneous lipiodol uptake by the S2 tumor (Fig. 2). Her liver cirrhosis remained Childs A at that stage. Three months later, the follow-up triphasic CT showed that the S2 tumor still remained, and lipiodol retention was no longer present. In addition, it was noted that its enhancement was gradual, diffuse, persistent and without portovenous washout. These are atypical imaging features for HCC and hence an alternative diagnosis of hemangioma was considered. MRI revealed that the tumor was very bright in T2W images (Fig. 3a), that it was hypointense in TW images (Fig. 3b), and that it displayed gradual enhancement as well as progressive contrast pooling (Fig. 3c) in delayed images. These are classic MR characteristics of hemangioma and the diagnosis was made. No further treatment was ordered for it. The patient underwent follow-up dynamic CT or MR imaging at three-monthly intervals for one and a half years thereafter, during which viable and recurrent HCCs were discovered and further TACE were required. Two of her TACE included the left lobe and thereby inadvertently treated the then known hemangioma. Subsequent images reveal no permanent change of the hemangioma s radiographic appearance, although temporary lipiodol retention was noted at the follow up CTs after each TACE (Fig. 4). The small hemangioma remained the same after a total of three sessions TACE and one course of PEI. Discussion Ty pical radiog raph ic feat u res of hepat ic hemangiomas include hypo-attenuating lesions that en hance unifor mly (if small) or cent r ipetally (if large) in the arterial phase, and retain a Figure. A 63 year old female with Childs A cirrhosis and HCC. a. The tumor (a hemangioma) in segment 2 (S2) enhanced strongly in post-contrast CT scan. b. Post-contrast arterial CT showed that the persistent enhancement of the lesion, with adjacent hypodensity compatible with post-pel changes. b

3 Hemangioma treated with TACE & PEIT 27 the enhancement in the portal venous phase [6]. Radiog raphic diag nosis of hemangioma does not usually constitute a challenge for a qualified radiologist. However confusion with small or welldifferentiated HCC can occur [7, 8], especially if the hemangioma has other atypical imaging features such as hypoechogeneity. Hemangiomas are typically hyperechoic, however they can appear hypoechoic in patients with hepatic steatosis. Hemangiomas can also become at least partially hypoechoic as they become larger and undergo internal hemorrhage, thrombosis, or myxomatous change [9, 0], and according to some authors approximately 20-40% of hemangiomas have hypoechoic components [0, ]. This confusion happened in our case, and the small hemangioma was not identified by the first Figure 2. Non-contrast CT after TACE showed the retention oflipiodol within the nodule (arrow). 3a 3b 3c Figure 3. a. The nodule has very high signal intensity in T2W (turbo spin echo, TR 2000, TE 00) MR imaging, which is highly suggestive of a hemangioma. b. Precontrast Tl weighted image (gradient echo, TR 220, TE.5), the nodule is hypointense. c. Delayed phase images (gradient echo, TR 24, TE 2.8, aquired approximately five minutes after contrast injection) show contrast pooling, which confirms the nodule to be a hemangioma.

4 28 Hemangioma treated with TACE & PEIT Figure 4. Dynamic CT two and half months after her third TACE showed the retention of lipiodol within the nodule (white ar row). Note the other lipiodol retention tumor in S8 near IVC (black arrow). The lipiodol retained in the hemangioma was no longer present in the next follow up CT. CT. It was then detected as a hypoechoic tumor by sonography and presented as a hypervascular tumor in the second CT and this lead to its misdiagnosis as HCC. In contrast to CT, MRI is both more sensitive (up to 00%) and specific (up to 92%) for hepatic hemangiomas and can be relied on to differentiate hemangiomas from other lesions, especially when heavily T2 weighted sequences are deployed [2]. Uncommonly, HCCs can display bright T2 weighted signals suggestive of a hemangioma [3], in which case other characteristics such as enhancement pattern has to be relied on in its differentiation from hemangioma. MRI confirmed this patient s hepatic hemangioma, with the lesion displaying typical MRI characteristics of hemangiomatous lesion. Hepatic hemangiomas are not routinely treated except for large symptomatic lesions. Common t reat ment options include su rger y [], t ransarterial embolization and radiofrequency ablation [4, 5]. Surgical removal of the hemangioma usually involves enucleation if the lesion is small; or segmentectomy if the lesion is relatively large. If the hemangioma is massive or diffuse in nature, then TAE is the preferred option. Embolic agents include polyvinyl ethanol, absolute alcohol, lipiodol, Gelatin sponge or steel coil [2, 3]. Both TACE and PEI are well-established treatment modalities for HCCs, and combination therapy with both have been shown to be more effective than PEI or TACE monotherapy for patients with HCC who are poor surgical candidates [4]. Although pure ethanol injection has been used to treat hemangioma in other anatomical locations, currently there is no clear documentation of attempts to treat hemangioma in liver with pure ethanol injection or TACE. Although many radiologists have found TAE to be adequate treatment for hepatic hemangiomas, some have found otherwise [3]. Given that hemangiomas have been treated with some degrees of success with TAE [5], we would expect this patient s hemangioma to show some degree of susceptibility to TACE, even if it is resistant to PEI. Therefore it was indeed a little surprising that this patient s hemangioma seemed to have survived both treatment modalities unscathed, exhibiting only transient changes in its imaging characteristics. After the PEI there was significant amount of low-density liver parenchyma surrounding the hemangioma, indicating a good amount of pure ethanol was administered into and around the lesion. It did retain significant amount of lipiodol after the TACE, and it can be reasoned that it must have uptaken reasonable amount of adriamycin and mitomycin. The treatment effect did not last and the hemangioma remained intact. This particular case provides us with a unique experience through the incidental treatment of hemangioma together with HCC, using combination treatment of PEI and TACE. This lesion has puzzled us with its resilience and longevity, and perhaps when what previously thought was a small HCC survives multiple treatment modalities without obvious imaging changes, another possible diagnosis should be considered. Fortunately for the patient, no complication has occurred secondary to the inadvertent treatment of her hemangioma. It is important to identify hemangiomas in HCC patients to avoid unnecessary treatment. Bibliography. Herman P, Costa ML, Machado MA, et al. Management of hepatic hemangiomas: a 4-year experience. J Gastrointest Surg 2005; 9: Zeng Q, Li Y, Chen Y, et al. Gigantic cavernous hemangioma of the liver treated by intra-arterial embolization with pingyangmycin-lipiodol emulsion: a multi-center study. Cardiovasc Intervent Radiol 2004; 27: Chen RC, Lii JM, Chen WT, et al. Transcatheter arterial chemoembolization in patients with hepatocellular carcinoma and coexisting hepatic cavernous hemangioma. Eur Radiol 2006; 6:

5 Hemangioma treated with TACE & PEIT Zagoria RJ, Roth TJ, Levine EA, et al. Radiofrequency ablation of a symptomatic hepatic cavernous hemangioma. AJR Am J Roentgenol 2004; 82: Cui Y, Zhou LY, Dong MK, et al. Ultrasonography guided percutaneous radiofrequency ablation for hepatic cavernous hemangioma. World J Gastroenterol 2003; 9: Vilgrain V, Boulos L, Vullierme MP, et al. Imaging of atypical hemangiomas of the liver with pathologic correlation. Radiographics 2000; 20: Leslie DF, Johnson CD, MacCarty RL, et al. Single-pass CT of hepatic tumors: value of globular enhancement in distinguishing hemangiomas from hypervascular metastases. AJR Am J Roentgenol 995; 65: Yan WQ, Zou LQ, Zhao Y, et al. Triple-phase helical CT in the differential diagnosis between small hepatocellular carcinoma and small hepatic cavernous hemangioma. Zhonghua Zhong Liu Za Zhi 2005; 27: Mirk P, Rubaltelli L, Bazzocchi M, et al. Ultrasonographic patterns in hepatic hemangiomas. J Clin Ultrasound 982; 0: Gibney RG, Hendin AP, Cooperberg PL. Sonographically detected hepatic hemangiomas: absence of change over time. AJR Am J Roentgenol 987; 49: Moody AR, Wilson SR. Atypical hepatic hemangioma: a suggestive sonographic morphology. Radiology 993; 88: McFarland EG, Mayo-Smith WW, Saini S, et al. Hepatic hemangiomas and malignant tumors: improved differentiation with heavily T2-weighted conventional spin-echo MR imaging. Radiology 994; 93: Ohtomo K, Itai Y, Matuoka Y, et al. Hepatocellular carcinoma: MR appearance mimicking cavernous hemangioma. J Comput Assist Tomogr 990; 4: Tanaka K, Nakamura S, Numata K, et al. The long term efficacy of combined transcatheter arterial embolization and percutaneous ethanol injection in the treatment of patients with large hepatocellular carcinoma and cirrhosis. Cancer 998; 82: Moon WK, Choi BI, Han JK, et al. Iodized-oil retention within hepatic hemangioma: characteristics on iodizedoil CT. Abdom Imaging 996; 2:

6 220 Hemangioma treated with TACE & PEIT 與原位肝細胞癌並存之肝臟血管瘤經酒精注射及經肝動脈栓塞治療後的影像變化 陳韻正 陳潤秋,2 陳慰宗 李兆祥 杜錫杰 李俊銘 臺北市立聯合醫院放射科 國立陽明大學生物醫學影像暨放射科學系 2 有症狀之肝臟血管瘤普遍以經血管栓塞, 經皮穿肝酒精注射, 或射頻消融術來治療均為文獻所認可 在此報告一個六十三歲女性患有肝硬化, 原發性肝癌, 及肝血管瘤 經過經皮穿肝酒精注射及經血管栓塞之後其肝血管瘤仍然存在