Journal of Pathology and Translational Medicine

Case Study

Isolated Mass-Forming IgG4-Related Cholangitis as an Initial Clinical Presentation of Systemic IgG4-Related Disease: Seokhwi Kim, Hyunsik Bae, Misun Choi, Binnari Kim, Jin Seok Heo, Ho Seong Kim, Seung Hee Choi, Kee-Taek Jang; J Pathol Transl Med. 2016;50(4):300-305. Published online January 11, 2016; DOI: https://doi.org/10.4132/jptm.2015.12.01

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IgG4-related disease (IgG4-RD) may involve multiple organs. Although it usually presents as diffuse organ involvement, localized mass-forming lesions have been occasionally encountered in pancreas. However, the same pattern has been seldom reported in biliary tract. A 61-year-old male showed a hilar bile duct mass with multiple enlarged lymph nodes in imaging studies and he underwent trisectionectomy under impression of cholangiocarcinoma. Gross examination revealed a mass-like lesion around hilar bile duct. Histopathologically, dense lymphoplasmacytic infiltration and storiform fibrosis were identified without evidence of malignancy. Immunohistochemical stain demonstrated rich IgG4-positive plasma cell infiltration. Follow-up imaging studies disclosed multiple enlarged lymph nodes with involvement of pancreas and perisplenic soft tissue. The lesions have been significantly reduced after steroid treatment, which suggests multi-organ involvement of systemic IgG4-RD. Here, we report an unusual localized mass-forming IgG4-related cholangitis as an initial presentation of IgG4-RD, which was biliary manifestation of systemic IgG4-related autoimmune disease.

Citations

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Pathologic interpretation of endoscopic ultrasound–guided fine needle aspiration cytology/biopsy for pancreatic lesions
Haeryoung Kim, Kee-Taek Jang
Journal of Pathology and Translational Medicine.2020; 54(5): 367. CrossRef
Multivisceral IgG4-related disease presenting as recurrent massive gastrointestinal bleeding: a case report and literature review
Xuexue Deng, Ronghua Fang, Jianshu Zhang, Rongqiong Li
BMC Gastroenterology.2018;[Epub] CrossRef
Recent advances in understanding and managing IgG4-related disease
Anna R. Wolfson, Daniel L. Hamilos
F1000Research.2017; 6: 185. CrossRef

Original Articles

Newly Formed Hepatic Masses in Children with Biliary Atresia after Kasai Hepatic Portoenterostomy.: Hye Jong Song, Yeon Lim Suh; Korean J Pathol. 2011;45(2):160-169.; DOI: https://doi.org/10.4132/KoreanJPathol.2011.45.2.160

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Abstract PDF

BACKGROUND
This report describes the clinicopathologic findings of six hepatic masses that developed after Kasai hepatic portoenterostomy (HPE) in six patients with longstanding biliary atresia (BA).
METHODS
Hepatic masses were found in six of 55 pediatric patients who underwent liver transplantation for BA after Kasai HPE from 1997 to 2009. Clinicopathologic analysis was performed and immunohistochemical staining was carried out for CD34, smooth muscle actin (SMA) and cytokeratin 7.
RESULTS
Of the six hepatic masses, two were diagnosed as focal nodular hyperplasia (FNH)-like lesions, two were large regenerative nodules (LRN), one was a mesenchymal hamartoma (MH) and one was a cholangiocarcinoma. The immunohistochemical staining findings for SMA and CD34 were more prominent for the FNH-like nodules than for the cirrhotic background liver. Dysplastic biliary epithelium arising from intestinal metaplasia was found in the cholangiocarcinoma.
CONCLUSIONS
Our findings suggest that FNH-like lesions, LRNs and MH are the results of vascular hemodynamic changes after Kasai HPE and that cholangiocarcinoma is due to recurrent cholangitis after BA. All the lesions in this series must be included in the differential diagnosis of a newly formed hepatic mass in patients after portoenterostomy.

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Features of Nodules in Explants of Children Undergoing Liver Transplantation for Biliary Atresia
Ana M. Calinescu, Anne-Laure Rougemont, Mehrak Anooshiravani, Nathalie M. Rock, Valerie A. McLin, Barbara E. Wildhaber
Journal of Clinical Medicine.2022; 11(6): 1578. CrossRef
Biliary Atresia Patients With Successful Kasai Portoenterostomy Can Present With Features of Obliterative Portal Venopathy
Kalyani R. Patel, Sanjiv Harpavat, Zahida Khan, Sadhna Dhingra, Norma Quintanilla, Mihail Firan, John Goss
Journal of Pediatric Gastroenterology and Nutrition.2020; 71(1): 91. CrossRef

IMP3 Expression of the Cholangiocarcinoma in Cytology Specimen and Its Diagnostic Value.: Jung Wook Yang, Jong Sil Lee, Dong Chul Kim, Dae Hyun Song, Gyung Hyuck Ko, Jeong Hee Lee; Korean J Pathol. 2010;44(6):642-650.; DOI: https://doi.org/10.4132/KoreanJPathol.2010.44.6.642

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Abstract PDF

BACKGROUND
Pathologists occasionally have difficulty in distinguishing cholangiocarcinoma from benign reactive cells on the biliary cytology. We estimated the diagnostic value of insulinlike growth factor II mRNA binding protein 3 (IMP3) on biliary cytology specimens.
METHODS
We selected 40 patients who had undergone a biliary cytology examination and whose diagnoses were confirmed by biopsy. The IMP3 expression was evaluated by immunohistochemical staining in both the tissue and the cytology specimens. Their histologic diagnoses were cholangiocarcinoma (n = 34), high grade dysplasia (n = 2), low grade dysplasia (n = 1) and non-biliary tumor or no tumor (n = 3).
RESULTS
On the histology, all the cases of cholangiocarcinoma or high grade dysplasia were positive for IMP3 (36/36). The low grade dysplasia or the normal bile ducts in the other 4 cases were negative for IMP3 (0/4). Of the 36 cases diagnosed as cholangiocarcinoma or high grade dysplasia histologically, 29 cases were positive for IMP3 on the cytology (29/36; sensitivity, 80.6%). Of the 4 cases without cholangiocarcinoma or high grade dysplasia, 1 case was weakly positive for IMP3 on the cytology. The histologic diagnosis of this case was hepatocellular carcinoma. Except for this hepatocellular carcinoma case, the specificity of IMP3 on the cytology was 100%.
CONCLUSIONS
IMP3 is a useful diagnostic marker for cholangiocarcinoma on cytology.

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Insulin-like Growth Factor II mRNA-Binding Protein 3 Expression in Benign and Premalignant Lesions and Carcinomas of the Stomach
Dae Hyun Song, Jung Wook Yang, Dong Chul Kim, Jong Sil Lee, Jeong Hee Lee, Sang Ho Jeong, Gyung Hyuck Ko
The Korean Journal of Pathology.2011; 45(4): 379. CrossRef

Expression and Prognostic Significance of Serum Response Factor in Cholangiocarcinoma.: Shin Young Park, Kyu Yun Jang, Yo Na Kim, Hee Jin Kim, Ho Sung Park, Myoung Ja Chung, Hee Chul Yu, Baik Hwan Cho, Kyoung Ryul Kim, Woo Sung Moon; Korean J Pathol. 2009;43(6):517-522.; DOI: https://doi.org/10.4132/KoreanJPathol.2009.43.6.517

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Abstract PDF

BACKGROUND
Serum response factor (SRF) is a transcriptional factor that plays an important role in cell growth and differentiation for several types of cells. The expression of SRF in cholangiocarcinoma (CC) and its potential role has not been examined. The aim of this study was to determine the relationship between the expression of SRF in CC and the clinicopathological parameters, as well as patient survival.
METHODS
We analyzed the expression of SRF in 84 surgically resected cases of CC (33 cases of intrahepatic CC [ICC] and 51 cases of extrahepatic CC [ECC]) by using immunohistochemistry. RESULTS: The positive expression of SRF was detected in 48.8% of the cases of CC (42.4% in ICC, 52.9% in ECC). SRF was predominantly expressed in the CC cells with intense labeling in the nucleus. A SRF expression was significantly associated with the cell proliferation rate (Ki-67 labeling index, p=0.046) and poor patient survival (p=0.002). The tumor differentiation (p=0.038), the T category (p<0.001), lymph node and distant metastasis (p<0.001, p=0.009) and nerve and vessel invasion (p=0.010, p=0.012) were also found to be significantly associated with a poor CC prognosis. CONCLUSIONS: These results suggest that the SRF may play a role in the tumor cell proliferation of CC, and its expression in tumor cells can provide additional prognostic information.

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Serum response factor induces epithelial to mesenchymal transition with resistance to sorafenib in hepatocellular carcinoma
JUN SANG BAE, SANG JAE NOH, KYOUNG MIN KIM, KYU YUN JANG, MYOUNG JA CHUNG, DAE GOHN KIM, WOO SUNG MOON
International Journal of Oncology.2014; 44(1): 129. CrossRef
Clinicopathologic significance of serum response factor expression in colorectal adenocarcinomas
Se Min Jang, Young Jin Jun, Hulin Han, Kang Hong Lee, Ki-Seok Jang, Seung Sam Paik
Basic and Applied Pathology.2011; 4(2): 46. CrossRef

Case Report

Intraductal Variant of Peripheral Cholangiocarcinoma of the Liver A report of three cases.: Won Mi Lee, Seok Hoon Jeon, Eun Kyung Hong, Moon Hyang Park, Jung Dal Lee; Korean J Pathol. 1998;32(3):222-225.

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Abstract PDF: Intraductal variant of peripheral cholangiocarcinoma is extremely rare. This variant shows intraductal growth and intraluminal extension without any infiltrative growth. The mode of intraductal growth is not known. The prognosis of this variant is better than that of usual cholangiocarcinoma. We report three cases, one of which is associated with Clonorchis sinensis (CS) infection. The tumors were entirely confined within the dilated peripheral tributaries of the intrahepatic bile duct. Microscopically, the tumors were well to moderately well differentiated, with a papillary or a micropapillary growth pattern. Focal clear cytoplasmic change and mucin production were noted. The tumors showed intraductal spreading without any invasion to the liver parenchyme. Mucosal hyperplasia and dysplasia were noted in the adjacent ducts. The authors assume that intraductal cholangiocarcinoma is a distinct subtype, and persistent irritation, such as, CS infection may undergo a malignant transformation through mucosal dysplasia.

Original Articles

Expression of Cytokeratins 7 and 20 in Cholangiocarcinoma and Metastatic Colonic Adenocarcinoma of the Liver.: Cheol Keun Park, Mi Kyung Kim; Korean J Pathol. 1999;33(1):42-47.

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Abstract PDF: The distinction between cholangiocarcinoma (CC) and metastatic colonic adenocarcinoma of the liver (MCA) is often difficult, particularly in needle biopsy and fine-needle aspiration specimens, if histologic features alone are used. To examine the differences in the expressions of the cytokeratin (CK) 7 and 20 in the CCs and MCAs, we performed immunohistochemical studies on surgically resected 19 CCs and 23 MCAs. We used monoclonal antibodies against CK 7 and CK 20, and applied microwave antigen retrieval technique on formalin-fixed, paraffin-embedded tissue. We interpreted diffuse cytoplasmic reactivity found in > or =5% of tumor cells as positive. CCs showed CK 7+/CK 20- immunophenotype in 63%, CK 7+/CK 20+ in 32%, CK 7-/CK 20+ in 5%, and CK 7-/CK 20- in 0%. MCAs exhibited CK 7-/CK 20+ immunophenotype in 87%, CK 7+/CK 20+ in 9%, CK 7-/CK 20- in 4%, and CK 7+/CK 20- in 0%. CK 20-reactive cells in CCs were frequently columnar in shape (p<0.05). In conclusion, the CK 7/CK 20 immunophenotype was useful in the differentiation of CCs from MCAs: the CK 7+/CK 20- immunophenotype strongly suggested CCs, whereas the CK 7-/CK 20+ immunophenotype strongly suggested MCAs.

The Observation of Histologic Changes of Major Intrahepatic Bile Duct Epithelium in the Resected Liver Tissue with Hepatolithiasis.: Woon Sup Han, Sae Kyung Choi, Sun Hee Sung; Korean J Pathol. 2001;35(1):20-25.

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Abstract PDF: BACKGROUND
The relationship between hepatoliths and cholangiocarcinoma is etiologically unclear. However, histogenetic sequencing with hyperplasia, dysplasia and carcinoma can occur in the bile ducts of hepatolithiasis.
METHODS
We studied 55 cases of hepatolithiasis and examined the specimens of resected liver tissue with a microscope. The growth patterns of bile duct epithelium were divided into four types: flat, tufting, micropapillary and papillary. The dysplasia was also divided into low-grade dysplasia (LGD) and high-grade dysplasia (HGD).
RESULTS
Of 55 cases of hepatolithiasis, 30 cases (54.6%) were of the flat pattern, 13 cases (23.6%) the micropapillary pattern, and 11 cases (20%) the tufting pattern. Epithelial hyperplasia was noted in only 36 cases (65.5%) in the large bile ducts, but dysplastic changes were found in 19 cases. Of 19 cases of dysplasia, LGD was present in 14 cases (25.5% of total 55 cases) an HGD in 5 cases (9% of total 55 cases). The epithelial hyperplasia showed histologic growth of the flat pattern in 29 cases out of 36 cases. But LGD (14 cases) had 6 cases of the tufting pattern and 7 cases of the micropapillary pattern. HGD (5 cases) revealed 4 cases of the micropapillary pattern with one case of the tufting pattern.
CONCLUSION
This study suggests that sequences of hyperplasia, low-grade dysplasia and high-grade dysplasia can play a role in the carcinogenesis of bile duct epithelium in hepatolithiasis with the histologic pattern changing from flat to micropapillary growth.

Expression of Cytokeratin 7 and 20 According to The Anatomical Location of Colon Cancer and The Differential Diagnosis with Cholangiocarcinoma.: Yoon Kyung Jeon, Sun Lee, Byoung Kwon Kim, Woo Ho Kim, Gyeong Hoon Kang; Korean J Pathol. 2002;36(3):146-153.

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Abstract PDF: BACKGROUND
Colonic adenocarcinoma usually shows CK7 negativity and CK20 positivity, which helps to differentiate it from cholangiocarcinoma usually showing a reverse immunohistochemical profile. We immunohistochemically investigated the pattern of CK7 and 20 expressions according to the anatomical location of colon cancer to refine the usefulness of CK expression in differential diagnosis.
METHODS
Immunohistochemical staining was done on 90 cases of surgically resected colon cancers and 84 cases of cholangiocarcinomas.
RESULTS
When the cases of colon cancer were divided into CATD (from the cecum to the descending colon) (32), sigmoid (26), and rectum (32), the positivity of CK7 was 41%, 15% and 28%, respectively, and the negativity of CK20 was 25%, 0 and 9% (p=0.013), respectively. In sigmoid colon cancers, 22 cases (85%) exhibited CK7-/CK20+ immunophenotype. However, the percentage decreased to 63% in the rectum and 47% in CATD. The CK7+/CK20- immunophenotype was found only in cancers in the cecum and ascending colon. The expression of CK7 was related to histologic differentiation (p=0.017).
CONCLUSIONS
The aberrant expressions of CKs were frequent in cancers of the rectum and ascending colon which are located in the transition site from the anus and small bowel, respectively. If adenocarcinoma in the liver were CK7+/CK20+ or CK7-/CK20-, the possibility of metastatic adenocarcinoma from CATD and rectum should be considered.

Expression of Proliferating Cell Nuclear Antigen (PCNA) of Major Intrahepatic Bile Duct Epithelium in Resected Liver Tissue with Hepatolithiasis and Hepatolithiasis Associated with Cholangiocarcinoma.: Shi Nae Lee, Sun Hee Sung, Woon Sup Han; Korean J Pathol. 2002;36(4):232-237.

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Abstract PDF: BACKGROUND
Histologic progressive changes of bile duct epithelium with hyperplasia, dysplasia and cholangiocarcinoma could be caused by hepatolithiasis. To be clarified as a neoplastic process, this histologic process should be evaluated with various aspects of cell biology.
METHODS
Immunohistochemical study of proliferating cell nuclear antigen (PCNA) was performed on 45 cases (10; normal, 15; hyperplasia, 14; low-grade dysplasia:LGD, 6; high-grade dysplasia: HGD) of hepatolithiasis and 10 cases (all HGD) of hepatolithiasis with cholangiocarcinoma.
RESULTS
In the hepatolithiasis, mean PCNA labelling indices (LI) of normal, hyperplasia, LGD and HGD of major intrahepatic bile duct epithelium were 24.5+/-4.3, 51.5+/-0.1, 62.0+/-.4 and 84.7+/-.3, respectively and gradually increased. Mean LI of PCNA in HGD of major intrahepatic bile duct epithelium of hepatolithiasis with cholangiocarcinoma was 68.7+/-.7, which was similar to that of LGD in hepatolithiasis without cholangiocarcinoma.
CONCLUSIONS
Histologic transformation through hyperplasia, dysplasia and carcinoma in major intrahepatic bile duct epithelium of hepatolithiasis may be a neoplastic process if these histologic changes are evaluated in the cellular proliferation aspect.

Case Report

A Case of Combined Hepatocellular Carcinoma-Cholangiocarcinoma with Pseudosarcomatous Features.: Ok Ran Shin, Jae Young Park, Hae Kyung Lee, Young Jin Choi, Kee Hwan Kim, Young Mi Ku, Keun Woo Lim; Korean J Pathol. 2006;40(4):303-305.

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Abstract PDF: Combine hepatocellular-cholangiocarcinoma with pseudosarcomatous features is a rare primary liver tumor. We report a 46-year-old man who is a carrier of HBV and has presented intermittent severe abdominal pain on right upper quadrant for 6 days. Tumor markers, alpha-fetoprotein, CEA and CA19-9 were within normal ranges. Abdominal computed tomogram revealed a large heterogeneous mass in the posteroinferior segment of right liver without associated biliary dilatation and conglomerated, enlarged portocaval and peripancreatic lymph nodes. Right hepatectomy was performed with local lymph node dissection. The tumor, measuring 7.5 x 5.5 cm showed an ill demarcated round to oval shape and a variegated whitish yellow solid cut surface with necrosis. Microscopically, the tumor consisted of three components, hepatocellular carcinoma, cholangiocarcinoma and undifferentiated sarcomatoid tumor cells. Immunohistochemically, hepatocellular carcinoma portion reacted positively with alpha-fetoprotein, cholangiocarcinoma portion reacted positively with bile duct-type cytokeratins (cytokeratin 7 and 19) and EMA. The pseudosarcomatous portion reacted with bile duct type cytokeratins and EMA. These findings suggested that pseudosarcomatous tumor cells may be with closely, associated primitive stem cells that can differentiate into the ductal epithelium.

Original Article

Expression Patterns of Tumor Related Proteins for Differential Diagnoses of Intrahepatic Adenocarcinomas.: Ghil Suk Yoon, Mi Sung Kim, Young Hwa Li, Ji Hyun Uhm, Sun Ju Kim, Tae Sook Kim, In Su Suh, Han Ik Bae; Korean J Pathol. 2005;39(4):229-235.

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Abstract PDF: Background
: Differential diagnoses of intrahepatic adenocarcinomas (IHAC) play an important role in the detecting primary sites and the determining type of treatment and overall prognosis of the patient. However, histopathologic findings alone have limitations of differential diagnoses of IHAC. Methods : To clarify which tumor related proteins (TRP) are useful for differential diagnoses of IHAC, TRP expression were investigated immunohistochemically, using MUC5AC, MUC2, mAb 91.9H, MUC1, and pS2, and by high iron diamine (HID) staining in 61 clinically confirmed IHACs. Results : MUC5AC (9/18, p<0.05) and MUC1 (17/18, p>0.05) displayed the most frequent expression in cholangiocarcinomas, and MUC2 (11/18, p<0.05), mAb 91.9H (16/18, p<0.05), and HID (16/18, p<0.05) in colorectal adenocarcinomas. pS2 (3/11, p>0.05) was expressed more often in pancreatic adenocarcinomas than other IHAC, while MUC2 and 91.9H were not expressed at all in pancreatic adenocarcinomas. The positivity of several TRP did not correlate with tumor differentiation. Conclusions : MUC5AC, MUC2, mAb 91.9H, and HID may be useful in differentiating cholangiocarcinomas from colorectal adenocarcinomas.

Case Report

A Case of Combined Hepatocellular and Cholangiocarcinoma with Neuroendocrine Differentiation and Sarcomatoid Transformation: A Case Report.: Mi Jung Kim, Hyun Lyoung Koo, Seung Kyu Lee, Jae Y Ro, Eunsil Yu; Korean J Pathol. 2005;39(2):125-129.

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Abstract PDF: We report here on a case of combined hepatocellular and cholangiocarcinoma (CHC) with neuroendocrine differentiation and sarcomatoid transformation. A 59-year-old male who had had HBV-associated chronic liver disease presented with hepatic masses. The explanted liver showed three small masses, two in the right lobe and one in the left lobe. The largest one in the right lobe was a 2.0 cm sized binodular mass,consisting of a yellowish tan nodule and an abutting reddish brown nodule. Microscopically, the reddish brown nodule was a cholangiocarcinoma (CC) showing neuroendocrine differentiation and sarcomatoid tranformation. The yellowish tan nodule and the remaining two masses were hepatocellular carcinoma (HCC)s. On immunohistochemistry, both the adenocarcinoma and spindle sarcomatoid cells were positive for pancytokeratin, but only the adenocarcinoma cells were positive for chromogranin and carcinoembryonic antigen (CEA). Mitotic and Ki67 labeling indices as well as p53 immunopositivity were significantly increased only in the CC component. We report here on the first case of CHC in which the CC displayed neuroendocrine differentiation and sarcomatoid transformation with high mitotic and Ki67-labeling indices, as well as having p53 overexpression.

Original Articles

Microsatellite Instability and Mismatch Repair Protein (hMLH1, hMSH2) Expression in Intrahepatic Cholangiocarcinoma.: Yun Kyung Kang, Woo Ho Kim; Korean J Pathol. 2005;39(1):9-14.

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Abstract PDF: BACKGROUND
To clarify the role of the mismatch repair (MMR) system in the carcinogenesis of intrahepatic cholangiocarcinoma (ICC), we investigated the microsatellite instability (MSI) status and MMR protein expression in ICC.
METHODS
Thirty-six cases of ICCs were examined by microsatellite analysis for 55 markers that encompassed all of the chromosomal arms and BAT26. An immunohistochemical study for hMLH1 and hMSH2 was also performed.
RESULTS
Widespread MSI (MSI-H) accompanied with a loss of hMLH1 expression was found in one case (2.8%). This MSI-H case was an adenosquamous carcinoma showing intraductal tubulopapillary adenocarcinoma and invasive adenosquamous carcinoma component. Loss of hMLH1 was noted in both components while the frequency and shifted band patterns of MSI were not identical between the components. Another 10 ICCs (27.8%) revealed low level MSI with preserved MMR gene expression.
CONCLUSIONS
Our data suggested that a genetic defect in the MMR system and MSI is not a major pathway in the carcinogenesis of ICC.

Combined Hepatocellular-Cholangiocarcinoma: Analysis of 8 cases with special reference to their growth patterns.: Yong Il Kim, Geun Kook Lee, Woo Ho Kim, Eun Sil Yu, Ghee Young Choe; Korean J Pathol. 1992;26(3):229-241.

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Abstract PDF: Eight cases of combine hepatocellular-cholangiocarcinoma(HCC-CC) of the liver were analysed along with their growth patterns and histologic subtypes to draw a possible implication in understanding of their histogenesis. The relative incidence of combined HCC-CC among the surgically resected primary carcinomas of the liver(485 cases) was 1.6%. The combination patterns varied and were classified as follows; the multinodular tumor, each consisting of HCC or CC element(type 1) was found in 1 case, the single tumor mass with two distinct compartments of HCC and CC(type 2) in 3, and the single tumor with random mixture of two elements(type 3) comprised the remaining 4 cases. Of the 7 cases of single tumor mass(type 2 and 3) HCC and CC comprised the major component in 5 and 2 cases, respectively. The histologic features of both HCC and CC elements were not different from those of isolated HCC and CC, except for two of CC element in type 3 which disclosed the intermediated differentiation or socalled cholangiolocellular carcinoma in part. We conclude that there is no significant difference in the relative incidence of combined HCC-CC among primary carcinomas of the liver and their subtypes compared to that in other countries, regardless of high incidence of both HCC and CC in Korea. Also, we discussed a possible histogenisis along a hypothesis that some of the combined HCC-CC be the consequence of interposition of different cell type from a new subclone into the growth of an initial single cell type of primary carcinoma of the liver.

Cystandenoma and Primary Cystadenocarcinoma of the Liver.: Kyoung Ho Kim, Chan Il Park; Korean J Pathol. 1989;23(2):263-268.

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Abstract PDF: Primary cystic neoplasia of the liver is rare. We report a cystadenoma with mesenchymal stroma (CMS) and a cystadenocarcinoma, and make a review of literature with particular reference to their histogenesis. The CMS has many similarities to the ovarian mucinous cystadenoma; occurring almost exlusively in female, being lined by mucus-secreting epithelial cells, and containing dense ovarian-like stroma. These features suggest that CMS may arise from the ectopic ovarian tissue within the liver. Cystadenocarcinoma may have its origin in CMS or cystadenoma without mesenchymal stroma of CMS. Cholangiocarcinoma arising from the congenital hepatic cysts can be differentiated only when it contains benign epithelia.

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