Carcinosarcoma of the Gallbladder with Chondrosarcomatous Differentiation and Intracytoplasmic Eosinophilic Hyaline Globules (Thanatosomes): A Report of a Case and Review of the Literature

Table 1

Cases of Carcinosarcoma of Gallbladder with Chondrosarcomatous Differentiation (1925-2018).

No	Author; Country; Year (Reference)	Sex	Age	Microscopic findings	IHC carcinoma	IHC sarcoma	Metastasis and Outcome
1	Kritsch; Germany; 1925 [17]	F	59	Carcinoma and sarcoma containing cartilage and bone.	Not done	Not done	Autopsy diagnosis.
2	Billi; Italy; 1964 [18]	F	59	Carcinoma with myxochondrosarcoma.	Not done	Not done	Alive four months after surgery.
3.	Edmondson; USA; 1967 [19]	?	?	Squamous cell carcinoma with foci of adenocarcinoma. Undifferentiated sarcoma with islands of malignant cartilage.	Not done	Not done	One of two cases of CSGB reported in AFIP fascicle on tumors of gallbladder without clinical data.
4	Sagi and Gyori; Hungary; 1972 [20]	F	79	Adenocarcinoma and fibro-chondromyxosarcoma.	Not done	Not done	Autopsy diagnosis
5	Higgs et al.; USA; 1973 [5]	M	77	Neoplastic glandular structures. Mucinocarminophilia was demonstrated. Neoplastic mesenchymal spindle-shaped cells, malignant cartilaginous and metaplastic osseous tissue.	Not done	Not done	Patient had tumor spread within bile duct. He developed infection of biliary tract by Klebsiella, and died on 29th postoperative day. Metastatic spread to liver and bile ducts with fibrosarcoma pattern predominated over all other neoplastic elements.
6	Mansori and Cho; USA; 1980 [6]	M	81	Most of the tumor is composed of bizarre pleomorphic cells. Foci of malignant gland formation. Spindly sarcomatous component with areas of malignant cartilage.	Not done	Not done	Tumor invaded adjoining liver tissue. Patient died within four weeks postoperatively. He had leukocytosis, suspected for sepsis and was placed on chemotherapeutic agents with no effect.
7	Von Kuster and Cohen; USA; 1982; Case 1 [7]	F	91	Well to poorly differentiated adenocarcinoma. Cellular stroma with spindle cells in herringbone pattern. Foci of malignant cartilage One focus of malignant osteoid.	Not done	Not done	Exploratory laparotomy showed a. mass in the gallbladder, with extensive metastases to the liver. Patient died on the third post-operative day of massive pulmonary embolism. Autopsy showed metastases to liver, left adrenal gland and para-aortic lymph nodes.
8	Von Kuster and Cohen; USA; 1982; Case 2 [7]	F	77	Moderately to poorly differentiated adenocarcinoma, contained mucin. Fibrosarcomatous stroma with small focus of malignant cartilage, and rhabdomyoblasts.	Not done	Not done	Cholecystectomy was done. No evidence of spread of the tumor. No chemotherapy was given. Patient was alive 31 months following surgery.
9	Hasegawa et al.; Japan; 1983 [21]	F	73	Solid nests of carcinoma with signet ring cells. Extensive areas of chondrosarcoma. Osteoid formation is present.	Not done	Not done	Patient had also adenocarcinoma of sigmoid colon. Patient had metastases to the liver. She died of peritoneal metastases 3 months postoperatively.
10	Miyamoto et al.; Japan; 1983 [22]	F	61	Adenocarcinoma and squamous cell carcinoma with malignant osteoid and cartilage.	Not done	Not done	Patient died 3 months after surgery.
11	Born et al.; USA; 1984 [23]	F	90	Malignant glandular structures in multilayered fashion. Malignant cartilaginous tissue.	Not done	Not done	Tumor was invading duodenum. Patient died on the third postoperative month from intractable pulmonary sepsis. Permission for autopsy was not granted.
12	Inoshita et al.; Japan; 1986 [24]	M	53	Adenocarcinoma, papillary and cribriform pattern. Sarcomatous component with interlacing bundles of highly atypical spindle cells admixed with malignant cartilage, osteoid, and foci of rhabdomyoblastic cells.	Positive for EMA	Rhabdomyoblast cells were positive for myoglobin	Died 17 months postoperative resection. Metastases to liver (Carcinomatous and RMS), and to the pancreas, diaphragm, and regional lymph nodes (RMS component only)
13	Uesaka et al; Japan; 1995 [25]	F	54	Carcinomatous glandular and ductal structures, nests of squamous cell carcinoma. Sarcomatous component with non-calcified osteoid differentiation and a small island of malignant cartilaginous tissue.	Positive for CEA, EMA, and Cytokeratin	Positive for EMA, CK, and S-100 protein in chondroid foci. Negative for vimentin and CEA	Portal tumor thrombus of sarcomatous elements, and osteoid differentiation.; Metastatic adenocarcinoma to one lymph node. Patient died of liver metastases 3 months after surgery.
14	Yavuz et al.; Turkey; 2000 [26]	F	50	Adenocarcinoma with tubular and cribriform structures. Sarcomatous component shows interlacing bundles of spindle cells with small islands of malignant cartilage and many rhabdomyoblasts.	Positive for cytokeratin and EMA	Diffusely positive for vimentin. Rhabdomyoblasts positive for desmin.	Patient died three months after diagnosis due to disseminated spread of the tumor.
15	Hotta et al; Japan; 2002 [13]	F	53	Carcinomatous component with tubular formations. Spindly and pleomorphic sarcomatous component with few foci of chondroid formation	Positive for cytokeratin, EMA and CEA	Positive for vimentin. Some tumor cells positive for cytokeratin	Patient underwent three surgical procedures. Developed leakage at anastomosis site and severe infection. Developed many liver metastases, and died of liver failure on 61st post-operative day.
16	Ajiki et al.; Japan; 2002 [27]	F	69	Well and poorly differentiated tubular adenocarcinoma. Sarcomatoid tissue with chondroid differentiation.	Positive for Keratin	Focally positive vimentin and cytokeratin. Positive for S-100 protein	Metastatic sarcoma with chondroid differentiation in the lesser omentum. The tumor had spread to the liver. The left kidney showed a mass (clear cell carcinoma). He died 7 months later with peritoneal dissemination.
17	Sakurai et al.; Japan; 2006 [28]	F	54	Poorly differentiated tubular adenocarcinoma and spindle-cell sarcoma with differentiation to cartilage and partly bone	Not detailed	Positive cytokeratin staining in malignant cartilage..	Tumor was advanced involving the transverse colon and the duodenum. Patient received chemotherapy. He developed liver metastases and died 15 months following surgery.
18	Okamura et al.; Japan; 2006 [29]	F	60	Adenocarcinoma Sarcomatous tumor cells with a spindle shape and partial chondroid differentiation.	Positive for epithelial markers.	Chondroid areas are positive for S100 protein.	Patient had long survival and was alive 54 months following surgical resection.
19	Oberoi et al.; India; 2006 [30]	M	68	Well differentiated adenocarcinoma and poorly differentiated sarcoma with chondroid areas.	Positive for cytokeratin	Positive for vimentin and cytokeratin	Direct invasion of the liver and metastatic deposits to the omentum, mostly adenocarcinomatous component. No data on outcome.
20	Zhang et al.; USA; 2008 [4] 4	M	65	Squamous cell carcinoma, with mesenchymal spindle cell component and malignant chondroid elements.	Not available.	Not available.	Alive at three months following surgery.
21	Kohtani et al.; Japan; 2009 [31]	M	84	Adenocarcinoma admixed with malignant mesenchymal elements including bone and cartilage.	Positive for cytokeratin and EMA but negative for CD68 and desmin	Positive for CD68 but negative for cytokeratin and EMA	Cholecystectomy only was done, followed by low dose chemotherapy. Patient survived four years postoperatively.
22	Krishnamurthy et al. India; 2011 [32]	M	83	Undifferentiated carcinoma spindle and giant cell type with osteoclastic giant cells. Focal chondrosarcoma.	Positive for Cytokeratin and CD68 (in osteoclastic giant cells)	Not documented	Gallbladder was friable necrotic and removed piecemeal. Died on the postoperative day of atrial fibrillation and cardiogenic shock.
23	Coetzee et al.,; South Africa; 2011 [33]	F	38	Malignant glands with serrated architecture, focal adenoca in-situ. Pleomorphic and atypical stromal cells showed a myxoid background with chondroid appearance, focal osteoid was present.	Positive pancytokeratin AE1/3 in dysplastic lining epithelium	FNA specimen: Positive MSA in stromal cells.	Patient had cholecystectomy. He returned nine months later with dyspnea and jaundice. Ultrasound-guided FNA of mass under diaphragm: biphasic tumor, stromal component predominated.
24	Parreira et. al.,; Brazil; 2012 [34]	F	58	Well-differentiated adenocarcinoma, papillary and tubular, with foci of squamous cell carcinoma. Chondrosarcomatous, osteoid and atypical tissues.	Not done	Not done	Patient died six months following surgery due to multiple metastases.
25	Kim et al.,; South Korea; 2012; Case 1 [35]	F	72	Foci of malignant glands. Widespread crossing bundles of mesenchymal components with chondroid differentiation.	Positive for: Cytokeratin, CK20, CK7, CEAE-Cadherin, Ki67 and p53	Positive for vimentin, osteocalcin, S-100 protein, Ki67 and p53 and SMA. Negative for myoglobin, and desmin.	Tumor progressed with direct invasion of the colonic loop, stomach, and liver.
26	Sadamori et al.; Japan; 2012 [36]	M	80	Moderately to poorly differentiated adenocarcinoma. Sarcoma with osteosarcomatous and chondrosarcomatous elements.	Not done	Not done	Died due to multiple liver metastases 13 months after surgery.
27	Aleaga et al.; Cuba; 2013 [37]	F	72	Moderately differentiated adenocarcinoma. Sarcomatoid stroma with pleomorphic cellular areas and malignant chondroid matrix.	Positive CAM 5.2, focally positive for CEA.	Positive for vimentin and S-100 protein.	Patient died 7 months following surgery with multiple liver metastases.
28	Kishino et al.; Japan; 2014 [38]	F	70	Adenocarcinoma. Sarcomatous element of malignant spindle cells, Foci of malignant cartilage and bone.	Positive for cytokeratin (AE1/AE3).	Positive for vimentin, myogenin and SMA.	Liver metastasis developed 19 months after cholecystectomy, and partial hepatectomy was performed. No tumor recurrence after two years following operation.
29	Faujdar et al.; India; 2015 [39]	F	60	Carcinomatous element with moderate anaplasia. Sarcomatous elements in the form of spindle cells arranged in bundles and fascicles, and heterologous elements of cartilage and mature bone.	Positive for cytokeratin.	Positive for vimentin	Not documented. Patient was lost for follow up.
30	Al Ratroot et al.; Saudi Arabia; 2018; (Current Case)	F	52	Solid malignant epithelial nests within highly malignant sarcomatous stroma with scattered islands of malignant cartilage. Intracytoplasmic eosinophilic hyaline globules present.	Positive for cytokeratin. Negative for vimentin.	Positive for vimentin and desmin. Cartilaginous islands positive for S100 protein.	Metastases to the liver, omentum, peri-colic and peri-gastric fatty tissue and lymph node.; Patient lost for follow up after seven months.