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Bizarre stromal cells mimicking malignancy in chondro‐osseous respiratory epithelial adenomatoid hamartoma

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To the Editor: Chondro-osseous respiratory epithelial adenomatoid hamartoma (COREAH), is a rare entity, with only 21 cases having been reported in the literature so far. We report the case of… Click to show full abstract

To the Editor: Chondro-osseous respiratory epithelial adenomatoid hamartoma (COREAH), is a rare entity, with only 21 cases having been reported in the literature so far. We report the case of a 64-year-old female patient who presented with a one-year history of left sided nasal obstruction, non-responsive to medical treatment with antihistamines. Clinical examination with anterior rhinoscopy and flexible nasendoscopy revealed a smooth, polypoid mass, occupying almost entirely the left nasal cavity. CT scan of the facial structures in axial and coronal planes demonstrated a mixed density mass in the left nasal cavity, being in contact with the nasal septum and measuring 5.0 3.4 1.0 cm. The patient underwent endoscopic excision of the lesion under general anesthesia. Intraoperatively a smooth, pedunculated elastic lesion was found, originating with a stalk from the olfactory cleft area and was easily excised. There were no postoperative complications and the patient was discharged two days later. One year followup has not shown any recurrence. The patient has been symptom-free and no further intervention has been required. Microscopy revealed nasal mucosa with intense edema and moderate chronic inflammatory infiltrates of the lamina propria, as well as focal presence of lobules of proliferating hamartomatous glands consisting of respiratory epithelium. The stroma consisted of a fibrous tissue with lobules of moderately cellular chondroid tissue (Fig. 1a). The chondrocytes were located in lacunae and presented mild variation in shape and size. Mitotic activity was absent. In the fibrous stroma there were numerous bizarre stromal cells, in no direct relationship with the chondroid tissue, some of which were multinucleated. These cells were spindle or asteroid in shape and contained vesicular nuclei with moderate variation in shape and size, but without evident mitotic activity (Fig. 1b). In addition, we performed morphometric quantity assessment of the bizarre stromal cells by using the ImageJ image processing software, in 10 high power field photographs of the stroma, containing approximately 6,764 cells. The total number of bizarre stromal cells was assessed at approximately 2,380 cells, a percentage of about 35% of the total cell population of the stroma. On immunohistochemichal stains, the bizarre stromal cells showed strong positivity for vimentin while a minority of them was positive for CD68 antigen, S-100 protein and, interestingly, for glial fibrillary acidic protein (Fig. 1c,d). Stains for keratins, smooth muscle actin, desmin, myogenin, CD34 and CD1a antigens were negative. Cells in the areas of chondroid differentiation showed diffuse positivity for S-

Keywords: respiratory epithelial; stromal cells; osseous respiratory; bizarre stromal; chondro osseous; epithelial adenomatoid

Journal Title: Pathology International
Year Published: 2017

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