Bile duct adenoma with oncocytic features

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ling. In contrast to HSV infections of squamous epithelium, characteristic viropathic changes of the colonic epithelium were not apparent. The interface of viable colonic mucosa and ulcer tissue was sharply delineated and did not show viropathic cell changes. Large areas had the low-power appearance of an infiltrative process with significant nuclear atypia, similar to the case presented by Farrell et al.,1 features which contributed to an initial working diagnosis of haematolymphoid neoplasm in our case also. Additional immunoperoxidase studies for HSV and electron microscopy ultimately helped to arrive at the correct diagnosis. H Blaszyk N H Hyman1 K Cooper Departments of Pathology and 1Surgery, University of Vermont College of Medicine, Burlington, VT, USA

1. Farrell DJ, Birchley DW, Edwards CM. Lesson of the month: histiocytic infiltration in ulcerative colitis, simulating malignancy. Histopathology 2004; 45; 638–640. 2. Goodell SE, Quinn TC, Mkrtichian E et al. Herpes simplex virus proctitis in homosexual men: clinical, sigmoidoscopic, and histopathologic features. N. Engl. J. Med. 1983; 308; 868–871. 3. Naik HR, Chandrasekar PH. Herper simplex virus (HSV) colitis in a bone marrow transplant recipient. Bone Marrow Transplant. 1996; 17; 285–286. 4. Daley AJ, Craven P, Holland AJ et al. Herpes simplex virus colitis in a neonate. Ped. Infect. Dis. J. 2002; 21; 887–888. 5. Delis S, Kato T, Ruiz P et al. Herpes simplex colitis in a child with combined liver and small bowel transplant. Ped. Transplant. 2001; 5; 374–377. 6. El-Serag HB, Zwas FR, Cirillo NW et al. Fulminant herpes colitis in a patient with Crohn’s disease. J. Clin. Gastroenterol. 1996; 22; 220–223. 7. Ruther U, Nunnensiek C, Muller HA et al. Interferon alpha (IFN alpha 2a) therapy for herpes virus-associated inflammatory bowel disease (ulcerative colitis and Crohn’s disease). Hepato-Gastroenterology 1998; 45; 691–699. 8. Caserta L, Riegler G. Cytomegalovirus and herpes simplex virus antibodies in patients with idiopathic ulcerative colitis. Am. J. Gastroenterol. 2001; 96; 3036–3037.

times show a central depression. Most of the lesions are less than 10 mm in diameter and are located subcapsularly. Here we describe the first case of a bile duct oncocytic adenoma found incidentally at postmortem in a 57-year-old HIV+ female. The lesion macroscopically appeared as an intraparenchymal white mass 3 mm in diameter located in the right hepatic lobe. The mass did not show any infiltrative pattern beyond the surrounding parenchyma. Histology showed small to medium tubular structures with secondary cystic dilation of some affected bile ducts (Figure 1A). The tubular epithelium was composed of a homogeneous population of cells with abundant, granular and eosinophilic cytoplasm. The nuclei were small to medium in size and centrally located. Nucleoli were absent. No mitotic activity was found. Occasional binucleated cells were seen. A lymphocytic infiltrate was present in the stroma (Figure 1B–D). Immunohistochemical studies showed that the tumour was positive for carcinoembryonic antigen, epithelial membrane antigen and cytokeratin 7 (Figure 1E) and negative for Hepar-1 (Figure 1F), confirming the biliary origin of the lesion. Oncocytes were first described by Hamperl in 1931 as epithelial cells, which showed abundant, finely granular, eosinophilic cytoplasm by light microscopy due to an increased number of mitochondria. Oncocytic differentiation of the biliary epithelium is extremely rare and has been described only in cases of intrahepatic biliary cystadenocarcinoma.3,4 A variant of bile duct adenoma composed of clear cells has been described5 as well as a type with a prominent fibrous stroma6 and recently Spector et al.7 described the first case of an intraductal oncocytic papillary neoplasm of the extrahepatic biliary tree, but, to the best of our knowledge, this is the first report of intrahepatic bile duct adenoma with oncocytic features. V Arena E Arena E Stigliano A Capelli Istituto di Anatomia Patologica, Universita` Cattolica del Sacro Cuore, Rome, Italy

Bile duct adenoma with oncocytic features DOI: 10.1111/j.1365-2559.2006.02441.x

Sir: Tumours with oncocytic differentiation may occur in a variety of organs, but are extremely rare in the biliary system.1,2 Bile duct adenomas appear as wellcircumscribed wedge-like white masses, which some-

1. Wolf HK, Garcia JA, Bossen EH. Oncocytic differentiation in intrahepatic biliary cystadenocarcinoma. Mod. Pathol. 1992; 5; 665–668. 2. Sudo Y, Harada K, Tsuneyama K, Katayanagi K, Zen Y, Nakuma Y. Oncocytic biliary cystadenocarcinoma is a form of intraductal

 2006 The Authors. Journal compilation  2006 Blackwell Publishing Ltd, Histopathology, 49, 309–328.

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A

B

C

D

E

F

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Figure 1. Biliary adenma with oncocytic features. See text for details. A, B, C, Stained with H&E. D, Stained with P.A.S. E, Immunohistochemical staining for Cytokeratin 7. F, Immunohistochemical staining for Hepar-1. oncocytic papillary neoplasm of the liver. Mod. Pathol. 2001; 14; 1304–1309. 3. Martin RCG, Klimstra DS, Schwartz L, Yilmaz A, Blumgart LH, Jarnagin W. Hepatic intraductal oncocytic papillary carcinoma. Cancer 2002; 95; 2180–2187.

4. Terada T, Taniguchi M. Intaductal oncocytic papillary neoplasm of the liver. Pathol. Int. 2004; 54; 116–123. 5. Albores-Saavedra J, Hoang MP, Murakata LA, Sinkre P, Yaziji H. Atypical bile duct adenoma, clear cell type: a previously undescribed tumour of the liver. Am. J. Surg. Pathol. 2001; 25; 956–960.

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6. Varnholt H, Vauthey JN, Cin PD et al. Biliary adenofibroma: a rare neoplasm of bile duct origin with an indolent behavior. Am. J. Surg. Pathol. 2003; 27; 693–698. 7. Spector SA, Bejarano PA, Amortegui JD, Renfrow MR, Livingstone AS. Intraductal oncocytic papillary neoplasm of the extrahepatic biliary tree: first report. Am. Surg. 2004; 70; 55–58.

Pulmonary histiocytosis X in a patient with epithelioid mesothelioma DOI: 10.1111/j.1365-2559.2006.02391.x

Sir: Histiocytosis X (reviewed1,2) is a rare disease characterized by the proliferation of Langerhans cells forming nodular lesions, sometimes with cavitation. The Langerhans cells are derived from the monocyte lineage and have a probable role in antigen presentation. They are identified by their expression of CD1a and S100 by immunohistochemistry or by the presence of Birkbeck granules on electron microscopy. In the adult form the condition is often restricted to the lung (pulmonary histiocytosis X or pulmonary eosinophilic granuloma) but may also involve bone and lymph nodes, in contrast to paediatric cases where the disease often involves multiple systems and the prognosis is poorer. Pulmonary histiocytosis X (PHX) has been described in association with lung cancer3–5 and rare reports of associations with carcinomas at other sites and with haematological malignancies have been documented.2,6 We have recently encountered a case of PHX in association with a pleural malignant mesothelioma. A 49-year-old male smoker was referred with a persistent cough. He had a significant history of asbestos exposure. There were no other respiratory symptoms, weight was stable and chest examination revealed only a dull right base. Chest radiography showed a prominent right hilum and a calcified left hemidiaphragm. Histology from a video- assisted thoracoscopic biopsy of parietal pleura showed epithelioid mesothelioma. The patient underwent staging investigations (bronchoscopy and mediastinoscopy), which did not show any metastasis, and chest magnetic resonance imaging, which showed no involvement of the pericardium or crura. He therefore underwent a pleuropneumonectomy. Examination of the resection specimen confirmed the diagnosis of epithelioid mesothelioma (pT2 N1 Mx) (Figure 1). In addition, the cut surface of the lung showed evidence of focal emphysema, particularly of the upper lobe. Histological examination of the lung parenchyma demonstrated stellate nodular scars (Figure 2) containing a mixed inflammatory cell infiltrate including numerous large Langerhans cells that showed immunoreactivity for

Figure 1. Macroscopic view of a para-saggital slice taken through the resection specimen following formalin fixation. The parietal and visceral pleurae are both studded with numerous nodules of pale tumour that histologically were confirmed to be epithelioid mesothelioma. The upper lobe shows evidence of focal emphysema.

Figure 2. Histology taken from the upper lobe shows rather stellate nodular scars with some associated enlargement of the adjacent airspaces. Immunohistochemical staining with a monoclonal antibody to CD1a demonstrated the presence of large numbers of Langerhans cells consistent with the diagnosis of pulmonary histiocytosis X (insert).

 2006 The Authors. Journal compilation  2006 Blackwell Publishing Ltd, Histopathology, 49, 309–328.