Brief reports

embedded material, using the avidin-biotin complex method on 3 pm sections showed cytoplasmic staining for factor VIII-related antigen (factor VIII-rAg) and staining with Ulex europaeus lectin (Dako, UK).

Discussion The histological features of the thyroid tumour and pulmonary metastases were those of a malignant haemangioendothelioma. The endothelial origin of the tumour was endorsed by the positive staining reaction for factor VIII-rAg and the positive Ulex europaeus binding. Few reports of thyroid haemangioendothelioma have objectively demonstrated endothelial differentiation. Pfaltz et d 3found 1 5 of their 36 tumours were positive for factor VIII and Eusebi et d . 4 were able to demonstrate an endothelial origin in four cases using electronmicroscopy and immunohistochemistry. Many tumours, however, originally classified as malignant thyroid haemangioendothelioma have yielded negative reactivity for endothelial cell markers on subsequent analysis. Thus, factor VIII rAg and Weibel-Palade bodies could be demonstrated in only two of Egloff 's 6 5 casesi. This case provides further evidence for the existence of malignant haemangioendothelioma as an entity in the thyroid. It is notable that the tumour is almost entirely restricted to regions of endemic goitreh and very unusual in thyroid pathology in the UK. The occurrence in

541

association with thyrotoxicosis has not, as far as the author is aware, been reported previously.

Acknowledgements I am grateful to Professor E.D.Williams for confirmation of the diagnosis, to Mrs C.Price for typing the manuscript and to Mr B.Sims for photographic assistance. I am also indebted to Dr CEdwards for help in the preparation of this report.

References 1. Vollenweider I. Hedinger C, Saremaslani P. Pfaltz M. Malignant haemangioendothelioma of the thyroid. iminunohistochemical evidence ofheterogeneity. Patho/. Rus. Pract. 1989: 184; 376-381. 2. Krisch K. Holzner JH, Kokoschka R. Jakesz R. Niederle B. Roka R. Haemangioendotheljoma of the thyroid gland-true endothelioma or anaplastic carcinoma! Pothol. Hes. Pract. 1980: 170; 2 30-242. 3 . Pfaltz M. Hedinger C. Saremasloni P, Egloff B. Malignant haemangioendothelioma of the thyroid and factor VIII-related antigen. 17itc~howsArch. / A ] I 9 8 3: 401 : I 77- I 84. 4. Eusehi V. Carcangiu ML. Dina R. Rosai J. Keratin-positive epithelioid angiosarcoma of thyroid A report of four cases. Am. /, Surg. Patho/. 1990: 14; 737-747. 5. Egloff B. 'The haemangioendothelioma of the thyroid. Virchows Arch. 1.11 1983: 400; 119-142. 6 . Ruchti C. Gerber H. Schaffner T. Factor VIII-related antigen in malignant haemangioendothelioma of the thyroid: Additional evidence for the endothelial origin of this tumour. Am, 1. Cliri. Pathol. 1984; 82: 474-480.

Histopat/i~i/ogg 1992. 20, 541-544

BRIEF REPORT

Hepatoid adenocarcinoma of the papilla of Vater G.W.GARDINER, G.LAJOIE & K.KEITH Departmerits of Pathology and Surgery, St. Michael's Hospital and Uriiversity of Toronto, Toronto, Ontario, Canada Date of submission 22 August 1991 Accepted for publication 14 January 1992

An adenocarcinoma of the papilla of Vater showing hepatoid differentiation similar to that described for hepatoid adenocarcinoma of the stomach is rep0rtedI.l. The tumour was a poorly differentiated adenocarcinoma with clear cells containing occasional hyaline droplets

and exhibiting bile secretion. Alpha-fetoprotein (AFP). zl-antitrypsin (a1-AT) and carcino-embryonic antigen (CEA) were demonstrated by immunoperoxidase staining. This appears to be the first report of a hepatoid adenocarcinoma at this location.

Address for correspondence: Dr G. W.Gardiner. Department of Pathology. St. Michael's Hospital. 3 0 Bond Street, Toronto. Ontario. M5B 1WX. Canada.

Keywords: adenocarcinoma*ampulla of Vater. tumour differentiation, hepatic tissue

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Brief reports

Case report A 61-year-old Caucasian woman presented with a 2month history of painless jaundice and fatigue. Abdominal ultrasound suggested a mass in the head of the pancreas with no evidence of liver metastases. Endoscopic retrograde cholangiopancreatography revealed an obstructive tumour at the papilla of Vater requiring the insertion of a stent in the common bile duct. A Whipple’s procedure was performed.

PATHOLOGICAL F I N D I N G S

A 6 cm length of gastric antrum with a n attached 1 2 cm length of duodenum and jejunum and a small 2 cm portion of the head of pancreas were resected. The papilla of Vater was polypoid with a slightly haemorrhagic granular mucosal surface and measured about 1 x 1 x 1 cm. On cut section, tumour was present

obstructing the distal ampulla and spreading to involve adjacent muscularis propria. Microscopically, the tumour consisted of marginal areas of dysplastic glands and well-differentiated adenocarcinoma of intestinal type that blended into solid, poorly differentiated adenocarcinoma which formed the bulk of the tumour (Figure 1).In such areas the tumour exhibited large clear cells in medullary nests with fine strands of outlining fibrocollagen bundles. Tumour cell nuclei were large and vesicular with central eosinophilic nuclei. Cell cytoplasm was clear and empty with scattered eosinophilic hyaline globules of variable size, and occasional yellow-brown bile droplets were present in cell cytoplasm and between cells (Figure 2). A van Gieson stain identified bile as bright green while stains for iron and lipofuschin were negative. Mitotic figures were frequent. PAS-diastase staining for glycogen was focally weakly positive and acid mucin was present in glandular areas with alcian blue-PAS-diastase staining. Immunoperoxidase stains demonstrated strong CEA positivity (Zymed, San Francisco. CA, USA) in glandular areas and a canalicular

Figure 1. Ampullary tumour showing a well-differentiated adenocarcinoma and b adjacent clear cell areas with bile plugs (arrows).

pattern in undifferentiated areas. Strong diffuse cytoplasmic staining for AFP (Dakopatts, Denmark) was observed in patches of clear cells in solid sheets (Figure 3 ) . Most hyaline droplets stained positive for c(l-AT.

Discussion

Figure 2. Higher power view of medullary sheets of clear cells with bile plugs (arrows)and areas of ductular formation.

Gastrointestinal tumours that show hepatocellular carcinoma-like features are rare and have been described in the stomach as hepatoid adenocarcinoma by Ishikura et nl. 1,2. In their series of seven cases, hepatoid differentiation was confirmed by morphological similarity to hepatocellular carcinoma, immunohistochemical identification of liver synthesized proteins, bile production and markedly elevated serum AFP levels. Only a single case in their series had evidence of bile production and two exhibited a clear cell pattern that caused diagnostic difficulty. Our case was a n adenocarcinoma with undifferentiated areas composed predominantly of clear cells that exhibited bile secretion, diagnostic of hepatoid differentiation. The clear cell component of this tumour raised the possibility of a metastatic clear cell tumour. The clear cells formed poorly differentiated tumour sheets but the transition to more typical intestinal glandular areas with mucin production suggested that this was part of a

Figure 3. a Patchy cytoplasmic staining of tumour cells for cc-fetoprotein. b Diffuse intense cytoplasmic staining of tumour cells for r ,-antitrypsin.

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Brief reports

primary tumour. Tumour cell cytoplasm may be optically clear in less differentiated neoplasms and may cause confusion with germ cell turn our^^,^. In fact two of the seven cases of hepatoid adenocarcinoma reported by Ishikura et aL2 were initially diagnosed as embryonal carcinoma. However, clear cell patterns are also well known to occur in hepatocellular carcinoma. The histogenesis of hepatoid foci in adenocarcinomas is of considerable interest and has been reported in a variety of tumours including stomach, ovary, pancreas and germ cell tumours2,3.Ishikura et aL2 concluded that the hepatoid elements in gastric adenocarcinoma arose from the differentiation of uncommitted somatic cells. Experimental work by Makino et aL4 in regenerating hamster pancreas in adult animals has shown the remarkable plasticity of pancreatic ductal cells with the potential to differentiate into hepatocytes-transdifferentiation. This phenomenon has also been suggested in a report by Hruban et aL5 of a pancreatic neoplasm exhibiting both pancreatic and hepatocellular differentiation. Because pancreatic ducts, large bile ducts, and the ampulla of Vater have an identical epithelial lining transdifferentiation could be a histogenetic explanation in our case. Finally, since stomach, liver and pancreas are derived from foregut endoderm in direct continuity

with yolk sac, it would not be surprising for some tumours at this site to share morphological features with hepatocellular carcinoma.

Acknowledgements The authors wish to thank Ms Sandra Cohen for photographic assistance.

References 1. Ishikura H. Fukasawa Y. Ogasawara K. Natori T. Tsukada Y, Aizawa M. An AFP-producing gastric carcinoma with features of hepatic differentiation, a case report. Cariccr 1985: 56; 840-848. 2. Ishikura H. Kirimoto K. Shamoto M et a/. Hepatoid adenocarcinomas of the stomach, a n analysis of seven cases. Cancer 1986; 58; 119- 126. 3 . Nakashima N , Fukatsu T. Nagasaka T. Sobue M. Takeuchi J. The frequency and histology ofhepatic tissue in germ cell tumours. Am. j . Surg. Pothol. 1987: 11; 682-692. 4. MakinoT. IJsudaN. Rao S. Reddy JK, Scarpelli DG. Transdifferentialion of ductular cells into hepatocytes in regenerating hamster pancreas. Lub. Irivcst. 1990; 62; 552-561. 5. Hruban RH. Molina JM. Reddy MN. Boitnott JK. A neoplasm with pancreatic and hepatocellular differentiation presenting with subcutaneous fat necrosis. Am. J. Clin. Pathol. 1987: 88: 639-645.

Hepatoid adenocarcinoma of the papilla of Vater.

An adenocarcinoma of the papilla of Vater showing hepatoid differentiation similar to that described for hepatoid adenocarcinoma of the stomach is rep...
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