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  Table of Contents    
CASE REPORT  
Year : 2021  |  Volume : 64  |  Issue : 5  |  Page : 140-142
Primary hepatic adenosquamous carcinoma: A rare case report


1 Department of General Surgery, Zhejiang Putuo Hospital, Zhoushan, China
2 Department of Endocrinology, Zhejiang Putuo Hospital, Zhoushan, China

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Date of Submission03-Dec-2018
Date of Decision06-Sep-2019
Date of Acceptance12-Sep-2019
Date of Web Publication7-Jun-2021
 

   Abstract 


Primary hepatic adenosquamous carcinoma (ASC) is an extremely rare primary hepatic malignant tumor, which is easily misdiagnosed as hepatocellular carcinoma before surgery. It has both the histologic features—adenocarcinoma and squamous cell carcinoma. Primary hepatic ASC incidence rate in intrahepatic cholangiocarcinoma is 2% to 3%.[1] Patients with hepatic ASC may experience recurrence or metastasis after surgery. The epidemiology, clinical diagnosis, etiology, and treatment of the disease remain challenging. It is important to improve the recognition of hepatic ASC because of its bad prognosis. Here, we report a man who had complained of an upper stomachache for 2 months and was diagnosed with primary hepatic ASC by histology and immunohistochemistry (IHC) after laparoscopic hepatectomy. The patient remained asymptomatic and survived well for 2 years post surgery and long-term follow-up was continued till now. In addition, we offer a brief discussion about the histopathological features, clinical behavior, and treatment of hepatic ASC and review the relevant literature.

Keywords: Adenosquamous carcinoma, hepatic tumor, immunohistochemistry, laparoscopic hepatectomy, pathology

How to cite this article:
Wei D, Lu L, Ying C, Qingsong K, Dongbo L, Feibo L. Primary hepatic adenosquamous carcinoma: A rare case report. Indian J Pathol Microbiol 2021;64:140-2

How to cite this URL:
Wei D, Lu L, Ying C, Qingsong K, Dongbo L, Feibo L. Primary hepatic adenosquamous carcinoma: A rare case report. Indian J Pathol Microbiol [serial online] 2021 [cited 2021 Jun 13];64:140-2. Available from: https://www.ijpmonline.org/text.asp?2021/64/5/140/317924





   Introduction Top


Primary hepatic adenosquamous carcinoma (ASC) is an extremely rare primary hepatic malignant tumor, only 59 cases have been reported since Pianzola and Durtfirst described it in 1971.[2] It has both the histologic features—adenocarcinoma and squamous cell carcinoma.[2],[3] To our knowledge, there were only a few cases of hepatic ASC, and the disease has nonspecific symptoms and imaging characteristics. The preoperative diagnosis and treatment of the disease remain challenging. To improve the recognition and treatment of hepatic ASC, we describe a 71-year-old male patient with hepatic ASC and review the relevant literature.


   Case Report Top


A 71-year-old male with a 2-month history of pain in the upper abdominal quadrant was admitted to our hospital for treatment. The discomfort and pain increased after meals, but it could not be related to defecation or change in position and was tolerable. He had a fever with a sometimes a maximum body temperature 38°C. The patient had a weight loss of 3 kg. He had no past or family history of liver tumor. On physical examination, he had no jaundice, but upper abdominal tenderness was significant.

Laboratory test on admission showed that the blood tests were all within normal ranges; serum tumor markers: CA 19-9 was 361.60 U/mL, CA-125 was 65.82 U/mL and HBsAg was positive. A computed tomography (CT) imaging scan of the abdomen at our institution revealed a 6 × 7 cm nodule with low-density shadow and blurry boundary, but CT during arterial of hepatic contrast enhancement demonstrated a large heterogeneous mass posteriorly in the left hepatic lobe [Figure 1]a. It was diagnosed as a kind of primary hepatic carcinoma. A laparoscopic hepatectomy was then performed not only for the purpose of curing the disease but also for histopathologic diagnosis.
Figure 1: Multidetector spiral CT during portal phase (a) show a tumor in the left hepatic lobe; (b) The Cross-section of the tumor specimen is variably gray to light-brown (the size 7 cm)

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The tumor was well-encapsulated and demarcated with a largest diameter of 7 cm and the cross-section of the specimen were variably gray to light-brown [Figure 1]b. Then, a histopathologic diagnosis was conducted. Microscopically, a part of the tumor cells showed irregular glandular tubules with a round nucleus, large volume, abundant cytoplasm, red staining, and keratin pearl. Additionally, there was a large necrosis area between tumor tissues, showing the structure of adenosquamous cell carcinoma [Figure 2]. The immunohistochemical staining was positive for CK7, P63, CK19, and Ki-67 (80%) and negative for CK20. The diagnosis of hepatic ASC was established. The patient remained asymptomatic and survived well for 2 years post surgery and long-term follow-up was continued till now.
Figure 2: (a) The tumor cells showed irregular glandular tubules with round nucleus, large volume, abundant cytoplasm, red staining, keratin pearl (H and E, ×200). Immunohistochemical staining was positive for CK7 (b) , P63 (c), CK19 (d), Ki-67 (80%) (e) and negative for CK20 (f)

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   Discussion Top


Primary hepatic ASC's histological feature is special , its general shape is a single tumor nodule, and the cross-section of the specimen is variably offwhite, its center texture is hard with cyst cavity. It was generally thought to be one rare member of intrahepatic cholangiocarcinoma. The pathogenesis of the disease is still unclear. It is usually connected with liver teratoma, intrahepatic bile duct stone, and simple hepatic cysts. Chronic cholangitis and bile duct cyst are easilily stimulated to produce epithelialization or tumor changes in the bile duct by infection, the cuboidal or columnar epithelium of the bile duct occur epithelial squamous dysplasia on the basis of adenocarcinoma, it developed into inadenosquamous cell carcinoma eventually,[4] Intrahepatic bile duct cells have the multipotential differentiation ability of stem cells, they can develop into adenosquamous carcinoma under the action of inflammation stimulation and biliary tract infection gradually.

Laboratory blood tests usually showed within normal ranges for alpha-fetoprotein (AFP) and serum CA-199. The squamous cell carcinoma antigen (SCC) rose a little in our report. Hepatic ASC has nonspecific symptoms, the patient may suffer from stomachache and fever when the tumor is large. The patient may have a history of intrahepatic cholangiopathy and chronic cholangitis but without hepatitis B antigen and liver cirrhosis.[5] The hepatic ASC imaging findings can be used as the basis for the diagnosis. In a CT scan, it may appear as a big mass with unclear border, but it may sometimes also have a liquefied central content with intrahepatic stone.[6] A magnetic resonance imaging (MRI) scan may show low density in the portal venous phase and delayed phase. Thus, it is also easily misdiagnosed with intrahepatic cholangiocarcinoma (ICC) on the basis of images; therefore, they need to be carefully identified before surgery.[7] Hepatic ASC should identify with the following diseases:

  1. Collision carcinoma: Two different tumors arise in the same area, which occur in gastroesophageal junction, lung, and anorectal junction, and maybe the collision of adenocarcinoma and sarcoma[8]
  2. Hepatapostema: It's CT imagine has a typical target sign, and patients have the symptoms of fever and abdominal pain[9]
  3. Single liver metastatic tumor: The patient has a history of primary malignancy and the CT imagine has a typical Bull eye sign.


After a laparoscopic hepatectomy, the patient was diagnosed with primary hepatic ASC by histopathology and immunohistochemistry. Before surgery, it was misdiagnosed as ICC. Pathology is thought as the only definite diagnostic criteria. Hepatic ASC has a typical pathological feature, the tumor cells were arranged in the nest bulk microscopically, part of them had keratin pearl with plenty cytoplasm, nuclear atypia, and nuclear division. Atypical hyperplasia of bile ducts was seen around the liver tissue, and it can prove the primary tumor with chronic cholangitis and hepatolithiasis. The immunohistochemical staining of hepatic ASC cell is positive for CK7 and CK19,but positive for C, HCC is usually positive for negative for and negative for CK7, the expression of CK19, P63 could be helpful for the diagnosis.

Hepatic ASC is a malignant tumor, resection is the first choice for its treatment and laparoscopic hepatectomy is the feasible and invasive way. Most patients have lost the chance of surgery for the malignancy and misdiagnosis,[10] the patient would have lymph node metastasis, liver recurrence, and metastasis in a short time even if the operation is implementation. The prognosis is very poor, it is reported that the mean survival time of patients after surgery is 2 to 12 months. Suzuki found that lymph node dissection can improve the postoperative survival time of patients. Other methods like transcatheter arterial chemoembolization (TACE), liver transplantation, and liver resection combined with radiotherapy and chemotherapy can be used for the treating of hepatic ASC.


   Conclusion Top


This case report together with the previous literature review alert clinicians about primary hepatic ASC. Early diagnosis and right treatment are important. Although it is still difficult to diagnose Hepatic ASC, we should summarize the experiences and study the disease at genetic and molecular level to improve the diagnosis and treatment of the disease.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Acknowledgements

This study is supported by the Department of Pathology, the Zhejiang Putuo Hospital.

Financial support and sponsorship

This study was funded by the Graduate Student Innovation Special Fund Project of Nanchang University awarded to Wei Dong, No. cx2015190.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Nakajima T, Kondo Y. A clinicopathologic study of intrahepatic cholangiocarcinoma containing a component of squamous cell carcinoma. Cancer 1990;65:1401-4.  Back to cited text no. 1
    
2.
Pianzola LE, Drut R. Mucoepidermoid carcinoma of the liver. Am J Clin Pathol 1971;56:758-61.  Back to cited text no. 2
    
3.
Barr RJ, Hancock DE. Adenosquamous carcinoma of the liver. Gastroenterology 1975;69:1326-30.  Back to cited text no. 3
    
4.
Zhao R, Zhu K, Wang R. Primary squamous cell carcinoma of the liver: A case report and review of the literature. Oncol Lett 2012;4:1163-6.  Back to cited text no. 4
    
5.
Takeda T, Murata K, Ikeda M, Mizokami M. Primary adenosquamous carcinoma of the liver presenting with hematochezia due to tumor invaion of the colon. Nisshoshi 2013;110:1959-67.  Back to cited text no. 5
    
6.
Tan Y, Xiao EH. Rare hepatic malignant tumors: Dynamic CT, MRI, and clinicopathologic features: With analysis of 54 cases and review of the literature. Abdom Imaging 2013;38:511-26.  Back to cited text no. 6
    
7.
Asayama Y, Tajima T, Okamoto D, Nishie A, Ishigami K, Ushijima Y, et al. Imaging of cholangiolocellular carcinoma of the liver. Eur J Radiol 2010;75:120-5.  Back to cited text no. 7
    
8.
Coupland SE, Dodson A, Liu H, Du MQ, Angi M, Damato BE. Intraocular collision tumour: A case report and literature review. Graefes Arch Clin Exp Ophthalmol 2013;251:1383-8.  Back to cited text no. 8
    
9.
Li B, Zhang Y, Chen S, Hou J, Shi H. (18)F-FDG PET/CT in a case of primary hepatic adenosquamous carcinoma. Rev Esp Med Nucl Imagen Mol 2016;35:344-6.  Back to cited text no. 9
    
10.
Yeh CN, Jan YY, Chen MF. Adenosquamous carcinoma of the liver: Clinicopathologic study of 10 surgically treated cases. World J Surg 2003;27:168-72.  Back to cited text no. 10
    

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Correspondence Address:
Li Feibo
Department of General Surgery, Zhejiang Putuo Hospital, 19 Wenkang Street, Zhoushan - 316 100, Zhejiang
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/IJPM.IJPM_785_18

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