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Case Reports
. 2022 Dec 2:9:1022017.
doi: 10.3389/fmed.2022.1022017. eCollection 2022.

Case report: Successful treatment with the combined therapy of interferon-alpha 2b and anlotinib in a patient with advanced hepatic epithelioid hemangioendothelioma

Affiliations
Case Reports

Case report: Successful treatment with the combined therapy of interferon-alpha 2b and anlotinib in a patient with advanced hepatic epithelioid hemangioendothelioma

Xiaolei Liu et al. Front Med (Lausanne). .

Abstract

Hepatic epithelioid hemangioendothelioma (HEH) is a very rare tumor originating from vascular endothelial cells, with unpredictable malignancy. At present, there is no standard treatment protocol yet established. Both surgical resection and liver transplantation have been reported to be effective treatments for HEH; however, multiple intrahepatic lesions or extrahepatic metastasis make these procedures unsuitable to most patients. Systematic therapy has also been investigated, but the results are undetermined due to the limited cases. Interferon-alpha 2b (IFN-a 2b) has also been used for the treatment of HEH. Based on our previous study, the rate of tumor regression with IFN-a 2b monotherapy was more than 50%. Here, we reported a patient with advanced HEH, who achieved a partial response with the combined therapy of anlotinib and IFN-a 2b. The tumor stayed stable for 2 years with anlotinib monotherapy and regressed 3 months after the combined therapy of anlotinib and IFN-a 2b. The synergistic effect of combined therapy with anlotinib and IFN-a 2b provided promising guidance for future clinical study.

Keywords: anlotinib; case report; epithelioid hemangioendothelioma; interferon; liver.

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Conflict of interest statement

The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

Figures

Figure 1
Figure 1
Unenhanced CT scan showed multiple intrahepatic lesions. (A) Contrast-enhanced CT scan showed heterogenous enhancement of the lesions with coalescence and target sign in the portal phase [(B,C), marked with arrow].
Figure 2
Figure 2
Multiple small lesions were detected in the lung (marked with arrows).
Figure 3
Figure 3
Hematoxylin and eosin and immunohistochemical staining of the liver biopsy, which showed spindle-shaped tumor cells and epithelioid tumor cells [(A), hematoxylin and eosin, ×200], and positive for CD 31, CD34, and ERG [(B–D), respectively, ×200]. The index rate of Ki-67 was 20% [(E), × 200].
Figure 4
Figure 4
The intrahepatic lesions [(A,B), marked with white arrows] were stable after 2 years of treatment with anlotinib monotherapy [(C,D), marked with yellow arrows], while the lesions regressed or disappeared after 3 months of combined therapy with anlotinib and IFN-a 2b [(E,F), marked with red arrows].

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