Pelvic skeletal metastasis of hepatocellular carcinoma with sarcomatous change: a case report
© Chen et al; licensee BioMed Central Ltd. 2010
Received: 21 March 2010
Accepted: 25 May 2010
Published: 25 May 2010
Sarcomatoid hepatocellular carcinoma (HCC) is a very rare histologic variant of HCC. The characteristic of skeletal metastatic sarcomatoid hepatocellular carcinoma has never been reported. We reported a patient with sarcomatoid hepatocellular carcinoma pelvic metastasis who presented with huge pelvic metastasis that had relatively small osteolytic lesion centrally located accompanied by huge bipeduncular invasive expansile lesions into surrounding soft tissue. The lesion showed almost non-isotope uptake in 99mTc-methylene diphosphonate bone scintigraphy study. He underwent radiotherapy and tumor excision but the tumor rapidly recurred. In addition, serum α-fetoprotein level was never elevated beyond normal limit (< 20 ng/mL) through the whole course of treatment. We considered sarcomatoid hepatocellular carcinoma bone metastasis a highly aggressive lesion with unusual metastatic pattern. Surgical treatment with adequate safe margin in such a huge tumor with hypervascularity and extensive invasion in the pelvis was difficult; and radiotherapy maybe refractory regarding the sarcomatous nature. Therefore, debulking operation with local symptoms control may provide a better quality of life. And the clinical course suggests sarcomatoid hepatocellular carcinoma is derived from the transition of an ordinary hepatocellular carcinoma.
Sarcomatoid hepatocellular carcinoma (HCC) is a very rare histologic variant of HCC with an incidence of 1.8% in surgically resected cases and 3.9% to 9.4% in autopsied cases [1–4]. Although sarcomatous transformation of HCC had been discussed in a few reports [3, 5]; it was never been reported in patients with bone metastasis.
The skeletal metastasis of sarcomatoid HCC have specific features of expansile, destructive nature, accompanied by large, bulky soft tissue masses; and these lesions often showed non-isotope uptake in 99mTc-methylene diphosphonate (MDP) bone scintigraphy (BS) study [4, 6–8].
We report a patient with sarcomatoid HCC pelvic metastasis. The patient presented with huge pelvic metastasis that had relatively small osteolytic lesion centrally located accompanied by huge bipeduncular invasive expansile lesions into surrounding soft tissue. He underwent radiotherapy and tumor excision. Unfortunately, the tumor rapidly recurred in short period. On the other hand, serum α-fetoprotein (AFP) level was never elevated beyond normal limit (< 20 ng/mL) after diagnosis of skeletal metastasis.
The patient was a 61 year-old male, who was a hepatitis B virus (HBV) carrier. He had hepatoma since November 2006 and underwent right hepatectomy. Pathologic analysis showed typical grade II to III HCC. Serum AFP level was greater than 3,000 ng/mL at that time. Abdominal sonography in July 2008 did not detect any viable liver tumor. However, hepatic tumor recurrence was noted since November 2008 and was treated 3 times with transarterial embolization (TAE) therapy from November 2008 to July 2009.
Since July 2008, serum AFP level was always within normal limit (< 20 ng/mL). Even with large bone lesions and systemic disease spreading, serum AFP level before each operation was only 13.92 and 15.39 ng/mL respectively.
Hepatocellular carcinoma with a spindle cell component has been referred to as sarcomatoid or sarcomatous HCC, spindle cell carcinoma, pseudosarcoma, and carcinosarcoma . According to previous reports, 2% to 27% of HCC have a sarcomatous appearance, and the incidence is increasing with the use of more aggressive treatments [2, 9].
The pathogenesis of the sarcomatous appearance of HCC has not yet been clarified because of a debate on whether it is derived from the transition of an ordinary HCC to a sarcomatous appearance or it is a double cancer of HCC and hepatic sarcoma [2, 3, 5, 10]. Most investigators thought that the sarcomatous component is derived from a dedifferentiation of anaplasia from an ordinary HCC rather than double cancer [1, 5]. Our pathologic analyses showed two patterns of tumor cells and the clinical course also suggested dedifferentiation of a metastatic HCC with sarcomatous change.
The typical findings of sarcomatoid HCC of liver is reported to be of massive expanding or multinodular confluent type with partial encapsulation . Intrahepatic metastasis and adjacent organ invasion were relatively more common with sarcomatoid HCC than with ordinary HCC . The metastatic lesions of our patient presented with bulky expansile dumbbell-shaped soft tissue masses, with relatively small bony destruction located at central portion; and often showed cold lesions on 99mTc-MDP BS [4, 6–8]. These characteristics suggested that the lesions originated from skeleton and gradually expanded as soft tumor masses almost without any bone related activity [4, 6–8, 12]. To our knowledge, only one previous report had metastatic HCC presented as sarcoma-like change with peritoneal dissemination . Actually, the larger soft-tissue portion of the metastasis behaved more like a sarcoma.
Radiation induced sarcoma is a rare sequela of radiation therapy, and often occurred more than 10 years after radiotherapy [14, 15]. The radiation dose in patients with radiation induced sarcoma is usually greater than 3,000 cGy . Although we have no direct evidence that this sarcomatous change was associated with radiotherapy, but our patient received 5,250 cGy radiation and primary hepatic lesion revealed a typical HCC. These findings suggested the possibility of radiation induced sarcomatous transformation of HCC, although the interval between radiation and sarcomatous transformation in the case herein presented is a few months rather than the expected duration greater than 10 years.
On the other hand, serum AFP level never exceeded normal limit (< 20 ng/mL) in our patient since diagnosis of bone metastasis. Even with large tumor burden before both bone tumor excisions, the highest recorded serum AFP level was 15.39 ng/mL. This finding was compatible with the 11-patient series by Koo HR et al . In their series, serum AFP level was more than 20 ng/mL in 7 patients (64%) and was more than 500 ng/mL in only 1 patient (9%).
In conclusion, we considered sarcomatoid HCC bone metastasis a highly aggressive lesion with unusual metastatic pattern. Surgical treatment with adequate safe margin in such a huge tumor with hypervascularity and extensive invasion in the pelvis was difficult; and radiotherapy maybe refractory regarding the sarcomatous nature. Therefore, debulking operation with local symptoms control may provide a better quality of life. 99mTc-MDP BS often has false-negative results and serum AFP level is not an appropriate marker for monitoring of bone metastasis of sarcomatoid HCC. Besides, the clinical course suggests sarcomatoid HCC is derived from the transition of an ordinary HCC.
Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
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