Clin J Gastroenterol (2013) 6:134–138 DOI 10.1007/s12328-012-0341-2
CASE REPORT
Primary squamous cell carcinoma of the liver concomitant with primary colon cancer: report of a case Kiyoto Morito • Keita Kai • Atsushi Miyoshi • Hiroshi Kubo • Takao Ide • Shinya Azama • Hiroyuki Irie • Yasuo Koga • Kenji Sumi • Osamu Ikeda • Kenji Kitahara • Hirokazu Noshiro
Received: 6 August 2012 / Accepted: 18 October 2012 / Published online: 26 February 2013 Ó Springer Japan 2013
Abstract A 55-year-old Japanese female was admitted to our hospital to treat colon cancer. Computed tomography revealed a 2.6 9 2.0 cm liver mass considered to be liver metastasis. She synchronously underwent right colectomy with D3 lymph node dissection and subsegmentectomy 8 under the diagnosis of advanced colon cancer with liver metastasis. The pathology examination revealed the liver nodule was pure squamous cell carcinoma (SCC), whereas histology of colon cancer was a well differentiated tubular adenocarcinoma containing no squamous component. The patient underwent intensive checkup by imaging for a primary site of SCC. However, no lesion considered as possible primary site of SCC was found. Therefore, the liver nodule was finally diagnosed as a primary hepatic SCC. Primary SCC of the liver is a rare and high-grade malignant tumor. Recurrent multiple liver nodule was found at 13 months after surgery and the patient died of cancer 17 months after surgery. Keywords Primary liver cancer Squamous cell carcinoma Adenosquamous carcinoma Colon cancer Double cancer
K. Morito (&) A. Miyoshi H. Kubo T. Ide Y. Koga K. Sumi O. Ikeda K. Kitahara H. Noshiro Department of Surgery, Saga University Faculty of Medicine, Nabesima 5-1-1, Saga, Saga 849-8501, Japan e-mail: [email protected] K. Kai Department of Pathology and Microbiology, Saga University Faculty of Medicine, Saga, Japan S. Azama H. Irie Department of Radiology, Saga University Faculty of Medicine, Saga, Japan
123
Introduction Primary squamous cell carcinoma (SCC) of the liver is very rare. It has been reported that primary SCC of the liver is often associated with hepatic teratoma [1], non-parasitic hepatic cyst [2], or hepatolithiasis [3]. For the definite diagnosis of primary liver SCC, exclusion of the possibility of metastasis from primary SCC of other organs is very important. We have experienced a unique case of primary SCC of the liver which did not have a history of prior liver insult or pre-existing hepatic cysts and presented as synchronous double cancer of ascending colon. We herein report the case and the process of discussion about the diagnosis.
Case report A 55-year-old Japanese female was admitted to our hospital to receive treatment for colon cancer found by examination for progressing anemia. She was not a smoker and had no history of alcohol abuse. She had a history of chronic rheumatism, pachyderma and hypertension and she took betamethasone 11 mg/day. She had undergone appendectomy 20 years ago, but has no history of blood transfusion. Laboratory data on admission revealed anemia (red blood cell count 3.67 9 106/ll, hemoglobin concentration 9.2 g/dl, hematocrit 30.7 %) and the presence of inflammation (white blood cell count, 10700/ll and C-reactive protein, 1.48 mg/dl) while platelet counts were within normal range. Other serology and coagulation tests showed no abnormalities. All of the examined tumor markers, the carcinoembryonic antigen (CEA), alphafetoprotein (AFP), and carbohydrate antigen 19-9 (CA19-9) were within normal range. Serology finding for hepatitis B virus was negative but hepatitis C virus was positive.
Clin J Gastroenterol (2013) 6:134–138
Fig. 1 Enhanced-abdominal computed tomography (CT). A mass lesion measured 2.6 9 2.0 cm in diameter showing ring-shaped enhancement is showed at segment 8 of the liver (arrow)
Fig. 2 Pathological findings of primary colon cancer. a Gross findings. The type 1 tumor measuring 4.4 9 3.1 cm was found at the cecum of the resected colon. b Microscopic findings. A well differentiated tubular adenocarcinoma was proliferated but no squamous cell carcinoma component was found in the tumor of the colon
The total colonoscopy revealed a type 1 tumor approximately 3 cm in diameter at the cecum. Enhanced-abdominal computed tomography (CT) revealed a 2.6 9 2.0 cm mass lesion showing ring-shaped enhancement at segment 8 of the liver (Fig. 1). Under the diagnosis of advanced colon cancer with liver metastasis, right colectomy with D3 lymph node dissection and subsegmentectomy 8 of the liver were synchronously performed.
Pathological findings The type 1 tumor measuring 4.4 9 3.1 cm was found at the cecum of the resected colon (Fig. 2a). Histologically, well
135
Fig. 3 Pathological findings of tumor of the liver. a Gross findings. A solitary nodule measuring 2.3 9 2.0 cm in diameter was found. b Microscopic findings. A seat-like proliferation of atypical cells showing an obvious keratinization was found whereas no portion of adenocarcinoma component was observed
differentiated tubular adenocarcinoma was proliferated and invaded the muscularis propria (Fig. 2b). Lymph node metastasis (1/15) and lymphatic vessel invasion were detected while no venous invasion was observed. The final stage according to the Japanese classification of colo-rectal cancer [4] was Stage 3a (T2, N1, M0). On the other hand, the cut surface of the liver specimen, which resected as metastasis of colon cancer, shows a solitary nodule measuring 2.3 9 2.0 cm in diameter (Fig. 3a). Microscopic study revealed the seat-like proliferation of atypical cells showing an obvious keratinization (Fig. 3b) and no portion of adenocarcinoma component was observed. Therefore, this nodule was diagnosed as SCC. It is very important to determine if the liver nodule was a metastasis of colon cancer or not. Entire sections of both of the tumors were intensively re-examined but no squamous cell component was found in the colon cancer and no adenocarcinoma component was found in the liver nodule. Immunohistochemical study revealed differentiating characteristics between the colonic adenocarcinoma and SCC of the liver. The colonic adenocarcinoma expressed cytokeratin (CK) CK20 and MUC 2 (colonic-secreted mucin) whereas it lacked the expression of CK 7 and p63 (a marker of the squamous cell) (Fig. 4a–d). In the liver tumor, almost all tumor cells expressed a squamous cell marker of p63 and a small number of tumor cells expressed CK 7 and 19 which is usually expressed in the bile duct epithelium while the tumor cells lacked the expression MUC 2 and CK 20 (Fig. 4e–h). From these findings, we considered that the liver nodule was primary SCC if any other primary lesion of SCC which could metastasize to the liver was not found. The background liver was almost normal and both chronic
123
136
Clin J Gastroenterol (2013) 6:134–138
Fig. 4 Comparison of immunohistochemical phenotype between colon tumor (a–d) and liver tumor (e–h). The colon tumor shows no expression of cytokeratin (CK) 7 (a magnification 9100), diffuse expression of CK 20 (b magnification 9100) and MUC 2 (c magnification 9100). No nuclear expression of p63 (d magnification 9100)
was observed. The liver tumor shows partial expression of CK 7 (e magnification 9100), no expression of CK 20 (f magnification 9100) and MUC 2 (g magnification 9100). The nuclear expression of p63 (h magnification 9400) was observed
inflammation and squamous metaplasia of bile duct epithelium was not found.
hepatic-foregut cysts, or other benign non-parasitic cysts including epidermoid cysts were noted [1–3, 6–19]. Other reports note the presence of primary squamous liver carcinomas in association with teratoma [5], Caroli’s disease [20], hepatolithiasis [21] and liver cirrhosis [22]. Although the precise steps of the development of the SCC are not exactly clear, proposed etiology is that secondary squamous metaplasia has developed in longstanding biliary-lined cyst or duct due to chronic inflammation and subsequent malignant transformation may arise in metaplastic squamous epithelium [3, 6–9, 23]. On the other hand, a few cases of solid primary liver SCC which did not have a history of prior liver insult or pre-existing hepatic cysts have been reported [24–26]. Interestingly, all these solid-tumor cases (including our case) expressed biliary CK 19 suggesting bile duct origin. One of the possible etiologies of these cases is that it is entirely squamous metaplasia from cholangiocarcinoma. Generally, the cancer containing both SCC and adenocarcinoma in the same lesion are categorized as adenosquamous carcinoma (ASC) and squamous metaplasia from cholangiocarcinoma is also considered as one of the etiologies of ASC [27]. Therefore, ASC and solid-SCC could be regarded as a relative tumor of the metaplasia from cholangiocarcinoma. In fact, both ASC and SCC in the liver
Postoperative course The patient underwent intensive checkup for a primary site of SCC by imaging studies including a CT scan of the chest, head and neck and upper gastroesophageal endoscopy. However, no lesion considered as a possible primary site of SCC was found. Therefore, we finally diagnosed the liver nodule as a primary hepatic SCC. The adjuvant chemotherapy was not done at the patient’s request. Recurrent multiple liver nodule was found at 13 months after surgery. Palliative therapy was done and she died of cancer 17 months after surgery; no autopsy was permitted.
Discussion The first case of SCC of the liver was reported by Imai in 1934 [5]. Since then, cases of hepatic SCC have been sporadically reported. In a majority of these reports, a preexistence of chronic hepatic cysts, either congenital
123
Clin J Gastroenterol (2013) 6:134–138
are categorized as histological variants of intrahepatic cholangiocarcinoma in WHO classification [28]. The present case was synchronous double cancer of SCC of the liver and adenocarcinoma of the colon. Although it is very rare, the cases of SCC or ASC of the colon and rectum have been reported [29–31]. Therefore, we performed intensive pathology investigation to exclude the possibility of metastasis from colon cancer. The review of English literature revealed two cases of metachronous double cancer of nasopharyngeal cancer and liver SCC [17, 32] while no synchronous case was found. Most cases of primary SCC of the liver are found in advanced stage and prognosis of these cases are dismal with an overall survival of less than 1 year [9–11]. Surgical resectability plays an important role in the prognosis of most hepatic malignancy. However, our case followed poor outcome despite the fact that the tumor was found at a relatively early stage (23 mm) and curative surgery was performed. A case of complete remission of SCC of the liver after systemic chemotherapy consisting of cisplatin (CDDP) and 5-fluorouracil (5-FU) and surgery was reported [33]. A few good prognosis cases of hepatic SCC treated with hepatic arterial injection of low dose anti-cancer drugs (CDDP and 5-FU) [26], adjuvant systemic chemotherapy with 5-FU and/or CDDP [17, 19] and radiation [25] were reported. Although there are no guidelines for adjuvant and palliative chemotherapy for primary SCC of the liver at present, these good prognosis cases suggest that radiation therapy and/or chemotherapy with 5-FU and/or CDDP should be recommended in combination with surgical resection if hepatic tumors are pathologically confirmed as primary SCC. This report presented a very rare case of primary SCC of the liver overlapped with primary colon cancer. The primary SCC of the liver is high-grade malignant tumor and strongly influences the prognosis of the other coexistent malignancy. Our experience is consistent with high malignant potential of primary liver SCC despite the fact that the tumor was found in small size and complete surgical resection was performed.
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4.
5.
6.
7.
8.
9.
10. 11.
12.
13.
14.
15.
16. 17.
18. Acknowledgments We would like to thank the Dr. Osamu Nakashima (Department of Pathology, Kurume University), for valuable comments for pathological diagnosis. Conflict of interest of interest.
19.
The authors declare that they have no conflict 20.
References 21. 1. Caratozzolo E, Massani M, Recordare A, Ciardo L, Antoniutti M, Jelmoni A, et al. Squamous cell liver cancer arising from an epidermoid cyst. J Hepatobiliary Pancreat Surg. 2001;8:490–3. 2. Lombardo FP, Hertford DE, Tan LK, Kazam E, Ramirez de Arellaro E. Epidermoid cyst of the liver complicated by
22.
microscopic squamous cell carcinoma: CT, ultrasound, and pathology. J Comput Assist Tomogr. 1995;19:131–4. Weimann A, Klempnauer J, Gebel M, Maschek H, Bartels M, Ringe B, et al. Squamous cell carcinoma of the liver originating from a solitary non-parasitic cyst case report and review of the literature. HPB Surg. 1996;10:45–9. Japanese Society for Cancer of the Colon and Rectum. General rules for clinical and pathological studies on cancer of the colon, rectum and anus, 7th edn. Tokyo: Kanehara-shuppan; 2006 (in Japanese). Imai T. Ein Fall von zystischem Teratom der Leber, in welchem Plattenepithelkrebs entstand. Trans Soc Pathol Jpn. 1934;24:578– 80. Banbury J, Conlon KC, Ghossein R, Brennan MF. Primary squamous cell carcinoma within a solitary nonparasitic hepatic cyst. J Surg Oncol. 1994;57:210–2. Nieweg O, Slooff MJ, Grond J. A case of primary squamous cell carcinoma of the liver arising in a solitary cyst. HPB Surg. 1992; 5:203–8. Roediger WE, Dymock RB. Primary squamous carcinoma of the liver. Clinical and histopathological features. Aust N Z J Surg. 1991;61:720–2. Yagi H, Ueda M, Kawachi S, Tanabe M, Aiura K, Wakabayashi G, et al. Squamous cell carcinoma of the liver originating from non-parasitic cysts after a 15 year follow-up. Eur J Gastroenterol Hepatol. 2004;16:1051–6. Clements D, Newman P, Etherington R, Lawrie BW, Rhodes J. Squamous carcinoma in the liver. Gut. 1990;31:1333–4. Bloustein PA, Silverberg SG. Squamous cell carcinoma originating in an hepatic cyst: case report with a review of the hepatic cystcarcinoma association. Cancer. 1976;38:2002–5. Lynch MJ, Mcleod MK, Weatherbee L, Gilsdorf JR, Guice KS, Eckhauser FE. Squamous cell cancer of the liver arising from a solitary benign nonparasitic hepatic cyst. Am J Gastroenterol. 1988;83:426–31. Monteagudo M, Vidal G, Moreno M, Bella R, Diaz M, Colomer O, et al. Squamous cell carcinoma and infection in a solitary hepatic cyst. Eur J Gastroenterol Hepatol. 1998;10:1051–3. Greenwood N, Orr WM. Primary squamous-cell carcinoma arising in a solitary non-parasitic cyst of the liver. J Pathol. 1972; 107:145–8. Vick DJ, Goodman ZD, Ishak KG. Squamous cell carcinoma arising in a ciliated hepatic foregut cyst. Arch Pathol Lab Med. 1999;123:1115–7. Furlanetto A. Dei Tos AP. Squamous cell carcinoma arising in a ciliated hepatic foregut cyst. Virchows Arch. 2002;441:296–8. Hsieh CB, Chen CJ, Yu JC, Chang TM, Gao HW, Liu YC. Primary squamous cell carcinoma of the liver arising from a complex liver cyst: report of a case. Surg Today. 2005;35:328–31. Zhang X, Wang Z, Dong Y. Squamous cell carcinoma arising in a ciliated hepatic foregut cyst: case report and literature review. Pathol Res Pract. 2009;205:498–501. Iimuro Y, Asano Y, Suzumura K, Yada A, Hirano T, Iijima H, et al. Primary squamous cell carcinoma of the liver: an uncommon finding in contrast-enhanced ultrasonography imaging. Case Rep Gastroenterol. 2011;5:628–35. Spaggiari M, Di Benedetto F, Ballarin R, Losi L, Cautero N, De Ruvo N, et al. Primary squamous cell carcinoma of the liver associated with Caroli’s disease: a case report. Onkologie. 2011; 34:193–5. Song E, Kew MC, Grieve T, Isaacson C, Myburgh JA. Primary squamous cell carcinoma of the liver occurring in association with hepatolithiasis. Cancer. 1984;53:542–6. Arase Y, Endo Y, Hara M, Kumada H, Ikeda K, Yoshiba A. Hepatic squamous cell carcinoma with hypercalcemia in liver cirrhosis. Acta Pathol Jpn. 1988;38:643–50.
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138 23. Gresham GA 3rd. Squamous cell carcinoma of the liver. Hum Pathol. 1985;16:413–6. 24. Lee HL, Liu YY, Yeh CN, Chiang KC, Chen TC, Jan YY. Primary squamous cell carcinoma of the liver: a successful surgically treated case. World J Gastroenterol. 2006;12:5419–21. 25. Naik S, Waris W, Carmosino L, Mehrishi A, Saif MW. Primary squamous cell carcinoma of the liver. J Gastrointestin Liver Dis. 2009;18:487–9. 26. Kaji R, Sasaki N, Tateishi I, Nagata E, Okabe Y, Yoshida T, et al. A case report of primary hepatic squamous cell carcinoma that remarkably responded to low dose arterial injection of anti-cancer drugs. Kurume Med J. 2003;50:71–5. 27. Suzuki E, Hirai R, Ota T, Shimizu N. Primary adenosquamous carcinoma of the liver: case report. J Hepatobiliary Pancreat Surg. 2002;9:769–73. 28. Bosman FT, Carnerio F, Hruban RH, Theise ND. WHO classification of tumors of the digestive system. Int Agency Res Cancer. 2010; 217–27.
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Clin J Gastroenterol (2013) 6:134–138 29. Yitta S, Liang MK, Berman R, Carter JJ, Yee HT, Marks JL. Primary squamous cell carcinoma of the colon associated with hypercalcemia and hyperleukocytosis: report of a case. Dig Surg. 2005;22:371–4. 30. Schneider TA 2nd, Birkett DH, Vernava AM 3rd. Primary adenosquamous and squamous cell carcinoma of the colon and rectum. Int J Colorectal Dis. 1992;7:144–7. 31. Lundquest DE, Marcus JN, Thorson AG, Massop D. Primary squamous cell carcinoma of the colon arising in a villous adenoma. Hum Pathol. 1988;19:362–4. 32. Chou YY, Lee WJ, Su CT, Hsu HC. Case report: primary cystic keratinizing squamous cell carcinoma of the liver in a patient with treated nasopharyngeal carcinoma. J Gastroenterol Hepatol. 1997;12:229–32. 33. Boscolo G, Jirillo A, Da Pian P. Complete remission of poorly differentiated squamous liver carcinoma after systemic chemotherapy and surgery: a case report. Tumori. 2005;91:71–2.
CASE REPORT
Primary squamous cell carcinoma of the liver concomitant with primary colon cancer: report of a case Kiyoto Morito • Keita Kai • Atsushi Miyoshi • Hiroshi Kubo • Takao Ide • Shinya Azama • Hiroyuki Irie • Yasuo Koga • Kenji Sumi • Osamu Ikeda • Kenji Kitahara • Hirokazu Noshiro
Received: 6 August 2012 / Accepted: 18 October 2012 / Published online: 26 February 2013 Ó Springer Japan 2013
Abstract A 55-year-old Japanese female was admitted to our hospital to treat colon cancer. Computed tomography revealed a 2.6 9 2.0 cm liver mass considered to be liver metastasis. She synchronously underwent right colectomy with D3 lymph node dissection and subsegmentectomy 8 under the diagnosis of advanced colon cancer with liver metastasis. The pathology examination revealed the liver nodule was pure squamous cell carcinoma (SCC), whereas histology of colon cancer was a well differentiated tubular adenocarcinoma containing no squamous component. The patient underwent intensive checkup by imaging for a primary site of SCC. However, no lesion considered as possible primary site of SCC was found. Therefore, the liver nodule was finally diagnosed as a primary hepatic SCC. Primary SCC of the liver is a rare and high-grade malignant tumor. Recurrent multiple liver nodule was found at 13 months after surgery and the patient died of cancer 17 months after surgery. Keywords Primary liver cancer Squamous cell carcinoma Adenosquamous carcinoma Colon cancer Double cancer
K. Morito (&) A. Miyoshi H. Kubo T. Ide Y. Koga K. Sumi O. Ikeda K. Kitahara H. Noshiro Department of Surgery, Saga University Faculty of Medicine, Nabesima 5-1-1, Saga, Saga 849-8501, Japan e-mail: [email protected] K. Kai Department of Pathology and Microbiology, Saga University Faculty of Medicine, Saga, Japan S. Azama H. Irie Department of Radiology, Saga University Faculty of Medicine, Saga, Japan
123
Introduction Primary squamous cell carcinoma (SCC) of the liver is very rare. It has been reported that primary SCC of the liver is often associated with hepatic teratoma [1], non-parasitic hepatic cyst [2], or hepatolithiasis [3]. For the definite diagnosis of primary liver SCC, exclusion of the possibility of metastasis from primary SCC of other organs is very important. We have experienced a unique case of primary SCC of the liver which did not have a history of prior liver insult or pre-existing hepatic cysts and presented as synchronous double cancer of ascending colon. We herein report the case and the process of discussion about the diagnosis.
Case report A 55-year-old Japanese female was admitted to our hospital to receive treatment for colon cancer found by examination for progressing anemia. She was not a smoker and had no history of alcohol abuse. She had a history of chronic rheumatism, pachyderma and hypertension and she took betamethasone 11 mg/day. She had undergone appendectomy 20 years ago, but has no history of blood transfusion. Laboratory data on admission revealed anemia (red blood cell count 3.67 9 106/ll, hemoglobin concentration 9.2 g/dl, hematocrit 30.7 %) and the presence of inflammation (white blood cell count, 10700/ll and C-reactive protein, 1.48 mg/dl) while platelet counts were within normal range. Other serology and coagulation tests showed no abnormalities. All of the examined tumor markers, the carcinoembryonic antigen (CEA), alphafetoprotein (AFP), and carbohydrate antigen 19-9 (CA19-9) were within normal range. Serology finding for hepatitis B virus was negative but hepatitis C virus was positive.
Clin J Gastroenterol (2013) 6:134–138
Fig. 1 Enhanced-abdominal computed tomography (CT). A mass lesion measured 2.6 9 2.0 cm in diameter showing ring-shaped enhancement is showed at segment 8 of the liver (arrow)
Fig. 2 Pathological findings of primary colon cancer. a Gross findings. The type 1 tumor measuring 4.4 9 3.1 cm was found at the cecum of the resected colon. b Microscopic findings. A well differentiated tubular adenocarcinoma was proliferated but no squamous cell carcinoma component was found in the tumor of the colon
The total colonoscopy revealed a type 1 tumor approximately 3 cm in diameter at the cecum. Enhanced-abdominal computed tomography (CT) revealed a 2.6 9 2.0 cm mass lesion showing ring-shaped enhancement at segment 8 of the liver (Fig. 1). Under the diagnosis of advanced colon cancer with liver metastasis, right colectomy with D3 lymph node dissection and subsegmentectomy 8 of the liver were synchronously performed.
Pathological findings The type 1 tumor measuring 4.4 9 3.1 cm was found at the cecum of the resected colon (Fig. 2a). Histologically, well
135
Fig. 3 Pathological findings of tumor of the liver. a Gross findings. A solitary nodule measuring 2.3 9 2.0 cm in diameter was found. b Microscopic findings. A seat-like proliferation of atypical cells showing an obvious keratinization was found whereas no portion of adenocarcinoma component was observed
differentiated tubular adenocarcinoma was proliferated and invaded the muscularis propria (Fig. 2b). Lymph node metastasis (1/15) and lymphatic vessel invasion were detected while no venous invasion was observed. The final stage according to the Japanese classification of colo-rectal cancer [4] was Stage 3a (T2, N1, M0). On the other hand, the cut surface of the liver specimen, which resected as metastasis of colon cancer, shows a solitary nodule measuring 2.3 9 2.0 cm in diameter (Fig. 3a). Microscopic study revealed the seat-like proliferation of atypical cells showing an obvious keratinization (Fig. 3b) and no portion of adenocarcinoma component was observed. Therefore, this nodule was diagnosed as SCC. It is very important to determine if the liver nodule was a metastasis of colon cancer or not. Entire sections of both of the tumors were intensively re-examined but no squamous cell component was found in the colon cancer and no adenocarcinoma component was found in the liver nodule. Immunohistochemical study revealed differentiating characteristics between the colonic adenocarcinoma and SCC of the liver. The colonic adenocarcinoma expressed cytokeratin (CK) CK20 and MUC 2 (colonic-secreted mucin) whereas it lacked the expression of CK 7 and p63 (a marker of the squamous cell) (Fig. 4a–d). In the liver tumor, almost all tumor cells expressed a squamous cell marker of p63 and a small number of tumor cells expressed CK 7 and 19 which is usually expressed in the bile duct epithelium while the tumor cells lacked the expression MUC 2 and CK 20 (Fig. 4e–h). From these findings, we considered that the liver nodule was primary SCC if any other primary lesion of SCC which could metastasize to the liver was not found. The background liver was almost normal and both chronic
123
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Fig. 4 Comparison of immunohistochemical phenotype between colon tumor (a–d) and liver tumor (e–h). The colon tumor shows no expression of cytokeratin (CK) 7 (a magnification 9100), diffuse expression of CK 20 (b magnification 9100) and MUC 2 (c magnification 9100). No nuclear expression of p63 (d magnification 9100)
was observed. The liver tumor shows partial expression of CK 7 (e magnification 9100), no expression of CK 20 (f magnification 9100) and MUC 2 (g magnification 9100). The nuclear expression of p63 (h magnification 9400) was observed
inflammation and squamous metaplasia of bile duct epithelium was not found.
hepatic-foregut cysts, or other benign non-parasitic cysts including epidermoid cysts were noted [1–3, 6–19]. Other reports note the presence of primary squamous liver carcinomas in association with teratoma [5], Caroli’s disease [20], hepatolithiasis [21] and liver cirrhosis [22]. Although the precise steps of the development of the SCC are not exactly clear, proposed etiology is that secondary squamous metaplasia has developed in longstanding biliary-lined cyst or duct due to chronic inflammation and subsequent malignant transformation may arise in metaplastic squamous epithelium [3, 6–9, 23]. On the other hand, a few cases of solid primary liver SCC which did not have a history of prior liver insult or pre-existing hepatic cysts have been reported [24–26]. Interestingly, all these solid-tumor cases (including our case) expressed biliary CK 19 suggesting bile duct origin. One of the possible etiologies of these cases is that it is entirely squamous metaplasia from cholangiocarcinoma. Generally, the cancer containing both SCC and adenocarcinoma in the same lesion are categorized as adenosquamous carcinoma (ASC) and squamous metaplasia from cholangiocarcinoma is also considered as one of the etiologies of ASC [27]. Therefore, ASC and solid-SCC could be regarded as a relative tumor of the metaplasia from cholangiocarcinoma. In fact, both ASC and SCC in the liver
Postoperative course The patient underwent intensive checkup for a primary site of SCC by imaging studies including a CT scan of the chest, head and neck and upper gastroesophageal endoscopy. However, no lesion considered as a possible primary site of SCC was found. Therefore, we finally diagnosed the liver nodule as a primary hepatic SCC. The adjuvant chemotherapy was not done at the patient’s request. Recurrent multiple liver nodule was found at 13 months after surgery. Palliative therapy was done and she died of cancer 17 months after surgery; no autopsy was permitted.
Discussion The first case of SCC of the liver was reported by Imai in 1934 [5]. Since then, cases of hepatic SCC have been sporadically reported. In a majority of these reports, a preexistence of chronic hepatic cysts, either congenital
123
Clin J Gastroenterol (2013) 6:134–138
are categorized as histological variants of intrahepatic cholangiocarcinoma in WHO classification [28]. The present case was synchronous double cancer of SCC of the liver and adenocarcinoma of the colon. Although it is very rare, the cases of SCC or ASC of the colon and rectum have been reported [29–31]. Therefore, we performed intensive pathology investigation to exclude the possibility of metastasis from colon cancer. The review of English literature revealed two cases of metachronous double cancer of nasopharyngeal cancer and liver SCC [17, 32] while no synchronous case was found. Most cases of primary SCC of the liver are found in advanced stage and prognosis of these cases are dismal with an overall survival of less than 1 year [9–11]. Surgical resectability plays an important role in the prognosis of most hepatic malignancy. However, our case followed poor outcome despite the fact that the tumor was found at a relatively early stage (23 mm) and curative surgery was performed. A case of complete remission of SCC of the liver after systemic chemotherapy consisting of cisplatin (CDDP) and 5-fluorouracil (5-FU) and surgery was reported [33]. A few good prognosis cases of hepatic SCC treated with hepatic arterial injection of low dose anti-cancer drugs (CDDP and 5-FU) [26], adjuvant systemic chemotherapy with 5-FU and/or CDDP [17, 19] and radiation [25] were reported. Although there are no guidelines for adjuvant and palliative chemotherapy for primary SCC of the liver at present, these good prognosis cases suggest that radiation therapy and/or chemotherapy with 5-FU and/or CDDP should be recommended in combination with surgical resection if hepatic tumors are pathologically confirmed as primary SCC. This report presented a very rare case of primary SCC of the liver overlapped with primary colon cancer. The primary SCC of the liver is high-grade malignant tumor and strongly influences the prognosis of the other coexistent malignancy. Our experience is consistent with high malignant potential of primary liver SCC despite the fact that the tumor was found in small size and complete surgical resection was performed.
137
3.
4.
5.
6.
7.
8.
9.
10. 11.
12.
13.
14.
15.
16. 17.
18. Acknowledgments We would like to thank the Dr. Osamu Nakashima (Department of Pathology, Kurume University), for valuable comments for pathological diagnosis. Conflict of interest of interest.
19.
The authors declare that they have no conflict 20.
References 21. 1. Caratozzolo E, Massani M, Recordare A, Ciardo L, Antoniutti M, Jelmoni A, et al. Squamous cell liver cancer arising from an epidermoid cyst. J Hepatobiliary Pancreat Surg. 2001;8:490–3. 2. Lombardo FP, Hertford DE, Tan LK, Kazam E, Ramirez de Arellaro E. Epidermoid cyst of the liver complicated by
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