Myoepithelial carcinoma: first case reported in the trachea

Myoepithelial carcinoma displaying exclusively myoepithelial differentiation mainly occurs in the salivary glands and breasts, and is considered extremely rare in the trachea. We present the first documented case of a primary myoepithelial carcinoma in the trachea. The patient was a 23-year-old man who presented with shortness of breath and cough for four months. Bronchoscopy and computed tomographic (CT) scan revealed an intraluminal mass in the trachea. A standard sleeve of trachea resection with end-to-end reconstruction was performed. The tumor was mainly composed of spindle cells and epithelioid cells which exhibited relatively uniform nuclei with finely distributed chromatin and inconspicuous nucleoli, suggesting that it may arise from benign myoepithelioma. Moreover, the tumor displayed marked cytologic atypia and an infiltrative tumor border in some areas, suggesting that it was a malignant tumor. Immunohistochemically, the tumor cells were diffusely positive for AE1/AE3, Vimentin and myoepithelial makers (Calponin, P63 and GFAP). The patient's postoperative course was uneventful with no evidence of recurrence at six months after surgery.     

Esophageal tuberculosis: an unusual localization in an immunocompetent patient

Oesophageal tuberculosis is a rare localisation for extrapulmonary tuberculosis. This report describes the case of a 30-year-old immunocompetent woman with dysphagia, fever and weight loss. Endoscopic examination revealed an ulcerated lesion in the third part of the oesophagus strongly suggesting oesophageal carcinoma. Histologic samples showed epithelioid cells granuloma in the submucosa. Diagnosis was later confirmed by acid-fast bacilli positive gastric aspirates and a positive Mantoux test using the tuberculin reaction. Barium swallow revealed a narrowing in the third oesophagus. A chest x-ray revealed a infiltrated lesion and a mediastinal lymph node. The patient was put on antitubercular treatment. On follow-up after 3 months, dysphagia, fever, weight loss had disappeared and the ulcerated lesion of oesophagus had improved. The subsequent course of the patient is not known, since she was lost to follow-up.     

An unusual lesion of the nasopharynx: oncocytic metaplasia

Oncocytic metaplasia of the nasopharynx is an exceptional lesion which exact etiopathogenesis, although largely discussed, still remains controversial. The purpose of this paper is to present the epidemiological characteristics and clinical signs of this lesion and to study its pathogenesis and its therapeutic modalities. We report two cases that occurred respectively in a 53- and 60-year-old woman. The first presented with pharyngeal dysesthesia and otalgia. The endoscopic examination revealed an irregularity of the posterior wall of the nasopharynx. The second patient presented with tinnitus, discomfort of the left ear and bilateral hearing loss. Endoscopic exam revealed a bilateral structural abnormality to the eardrum. Microscopy showed focal oncocytic metaplasia of the nasopharynx mucosa in both cases. There was a positive outcare for both patients after excisional biopsy. Oncocytic metaplasia seems to be in relation to the stimulation of sympathic neuropeptidergic nerve fibers which target epithelial, connective, endothelial and lymphoid cells.     

Myoepithelial cells in needle aspirations of two cases of unusual breast lesions: an aid in differential diagnosis

The observation of myoepithelial cells bordering abnormal epithelial cells in fine-needle aspiration smears of two unusual breast lesions helped to prevent misdiagnosis of these lesions as malignant. The presence of myoepithelial cells has been a well-established criterion for histologically benign lesions. Their presence or absence appears to be equally valuable in fine-needle aspiration smears of breast lesions.     

Myoepithelial carcinoma arising in an adenomyoepithelioma of the breast: case report with immunohistochemical and mutational analysis

Adenomyoepithelioma (AME) of the breast is an uncommon tumor characterized by biphasic proliferation of both epithelial and myoepithelial cells. In rare instances, the epithelial, the myoepithelial or both components of an AME may become malignant. Described herein is the case of a 69-year-old woman who presented with myoepithelial carcinoma of the breast in an AME. Malignancy of myoepithelial component (MEC) was evidenced by the presence of cytological atypia, high mitotic rate, necrosis and local invasion. Immunohistochemical study demonstrated strong expression of P53 and phosphorylated extracellular signal-regulated kinase 1/2 in MEC. Laser capture microdissection technique and mutational analysis further revealed point mutation of the p53 gene (T-->G transversion at codon 270) in this population, but not in glandular epithelial cells or adjacent normal ductal epithelium. No mutations in exons 1 and 2 of the K-, H-, and N-ras genes were identified in any of the neoplastic component. To the authors' knowledge this is the first report of a mutation in the p53 gene in a malignant AME of the breast.     

Myoepithelial carcinoma of soft tissue: a case report

Myoepithelioma of soft tissue is a recently categorized entity and myoepithelial carcinoma is extremely rare. We describe a case of myoepithelial carcinoma of soft tissue in a 30-year-old male patient, who presented with a painless mass located at the back of left leg involving popliteal fossa that was present since childhood. A wide local excision was performed. The distinct histopathological features included infiltrative margins, cytologically moderate to severely atypical epithelioid/spindled cells with prominent nucleoli, 3-4 mitoses/10HPF, tumor necrosis and lymphovascular invasion. No heterologous elements were identified. The myoepithelial origin was confirmed by positive immunohistochemical staining for S100 protein, epithelial membrane antigen and smooth muscle actin. Mib-1 (Ki-67) proliferation index was 20-25%. These carcinomas have variable clinical presentation and can have an indolent course for several years. Recognition of myoepithelial carcinoma is clinically significant because compared to its benign counterpart, this has increased frequency of local recurrences and metastases that warrants a close clinical follow-up.     

Myoepithelial carcinoma of the orbit: a clinicopathological and histopathological study

Tran TL, Broholm H, Daugaard S, Fugleholm K, Poulsgaard L, Prause JU, Kennedy SM, Heegaard S. Myoepithelial carcinoma of the orbit: a clinicopathological and histopathological study. APMIS 2010; 118: 324–30. Two cases of invasive myoepithelial carcinoma arising from the paranasal sinuses and invading the orbit are presented. Patient 1, a 53‐year‐old man, had a 3‐month history of proptosis, pain and epiphora of the right eye. The second patient, a 24‐year‐old man, had for a week been complaining of protrusion of his left eye and of orbital pain. Computed tomography scan and magnetic resonance imaging revealed tumour masses in the frontal, ethmoidal and maxillary sinuses with invasion of the orbit and the frontal lobe. Biopsies from both cases showed spindle and epithelioid tumour cells. Mitotic figures were frequent. Immunohistochemical staining showed positive reaction for bcl‐2, calponin, cytokeratins, CD99, S100, muscle‐specific antigen, smooth muscle antigen and vimentin. The Ki‐67 index was between 30–50% and 5–25%, respectively. Ultrastructurally, intermediate filaments, perinuclear tonofilaments and desmosomes were present. Based on these findings, a diagnosis of myoepithelial carcinoma of mixed cell type in both cases was evident. Both patients died shortly after the diagnosis was made even though both underwent radical surgery. Myoepithelial carcinoma of the paranasal sinuses is very rare and only six cases have been reported previously. We present the first two cases of myoepithelial carcinoma in the paranasal sinuses with invasion of the orbit. This is also the first report of myoepithelial carcinoma arising in the ethmoidal sinus.     

Myoepithelial cell layer integrity in canine mammary carcinoma

The aim of this study was to determine whether the myoepithelial (ME) cell marker calponin could be used to analyze the integrity of the ME cell layer as a means of identifying canine mammary carcinoma in situ. Tissue from 74 canine mammary lesions was evaluated (two dysplasia, eight benign tumours and 64 carcinomas including one carcinoma in situ). The 63 carcinomas included examples of histological grade 1 (n=32), grade 2 (n=23) and grade 3 (n=8). Expression of calponin was determined by immunohistochemistry. The percentage of proliferating cells surrounded by a single layer of calponin-positive cells formed the basis of classification as type I (≥ 90%), type II (70-90%) and type III (≤ 70%). Expression of Ki67 was used to determine the proliferation index (PI). The malignant tumours comprised of an approximately equal mixture of type I, II and III lesions. The two examples of dysplasia, the carcinoma in situ and two thirds of the benign tumours were classified as type I lesions. Some overlap in the level of calponin expression was observed between benign and malignant tumours. Positive correlations between the degree of calponin expression and the type of lesion (i.e. benign versus malignant; R=+0.3, P=0.08) and the histological grade of malignancy (R=+0.54, P=0.000001) were found. A negative correlation between the degree of calponin expression and PI (R=+0.027, P=0.016) was found. The ME cell marker calponin may be used as an aid in the identification of canine carcinoma in situ, but the study of the ME cell layer integrity is not definitive for the diagnosis of malignancy in canine mammary tumours.     

Cutaneous metastases from gastric carcinoma: an unusual presentation

We report the case of a 71-year-old gentleman who intially developed cutaneous metastases from gastric carcinoma on his chin and cheek resembling sebaceous cysts.     

Myoepithelial carcinoma in pleomorphic adenoma of salivary gland type, occurring in the mandible of an infant

A rapidly growing mandibular tumor occurred in a 17 month old female infant. Tumor outgrowth showing a periosteal reaction was radiographically seen on the lower surface (base) of the mandible. Under the biopsy diagnosis of osteosarcoma, high-dose chemotherapy with methotrexate was performed, resulting in little effect. The right hemiman-dibulectomy specimen disclosed intraosseous infiltrative growth of pleomorphic adenoma of salivary gland type, associated with chondroid stroma and reactive bone formation. The highly proliferative small-sized cells retained immunohistochemical features of myoepithelial cells, with positive reactivity of cytokeratin, vimentin, S-100 protein, a-smooth muscle actin, epithelial membrane antigen, CA15–3, type IV collagen, laminin and p53 protein. No heterotopia of the salivary gland was identified within the bone tissue. The tumor recurred 2 months later. Due to uncontrollable local growth, the patient died 8 months after operation. At autopsy, reactive ossification was closely associated with malignant myoepithelial proliferation. No distant metastasis was noted. This osteosarcoma-like tumor can be regarded as myoepithelial carcinoma in pleomorphic adenoma, originating from intramandibular heterotopic salivary gland tissue.     

A case of hepatocellular carcinoma in an elder man with metastasis to the nasopharynx

The lung, bone, brain, etc are common metastatic sites of hepatocellular carcinoma. Nasopharynx metastasis of hepatocellular carcinoma rarely occurs and has not been reported yet. Here we report one case of nasopharynx metastasis from liver in a 50-year-old male patient who was diagnosed with primary hepatocellular carcinoma (nodular and diffuse-type) in 2014. 6 and underwent interventional therapy for two times afterward. However, he suffered from severe headache in 2014. 8, and head contrast-enhanced MRI scan did not show clues for brain or skulls metastasis. Then the lumbar puncture was performed to examine his cerebrospinal fluid (CSF). It showed that cerebrospinal fluid protein (AFP) was extremely higher than the normal level. Then, he developed left blepharoptosis, eye opening obstacle, impaired vision and dysphagia. Positron emission tomography CT (PET-CT) showed that there was multiple bone destruction in skull base, indicating the nasopharyngeal cancer which was proven to be the metastatic tumor from liver histologically by biopsy. Finally, this patient underwent radiotherapy (RT) of nasopharyngeal metastatic tumor and the local symptoms changed for the better.     

Myoepithelial carcinoma of pharynx expressing KIT and PDGFRA

KIT and PDGFRA expression has rarely been examined in myoepithelial carcinoma (MC) of the salivary glands. An 89-year-old Japanese woman presented with a pharyngeal mass. Gross and imaging examinations revealed an elevated mass in the middle pharynx next to the oral cavity. A biopsy revealed atypical cells, and tumorectomy was performed. The tumor was composed of atypical epithelioid cells arranged in solid nests, cords, and vague acinar patterns. Mitotic figures were recognized in 3 per 50 high power fields. Immunohistochemically, the tumor cells were positive for myoepithelial markers including cytokeratin (CK) 14, α-smooth muscle antigen, S100 protein, and p63. They were also positive for KIT, PDGFRA, pancytokeratin AE1/3, CK34βE12, CK5/6, vimentin, p53, and Ki-67 (labeling=28%). They were negative for neuron-specific enolase, CD45, CD34, CD56, chromogranin, synaptophysin, melanosome, desmin, epithelial membrane antigen, CK18, CK20, pancytokeratin (CAM5.2). A pathologic diagnosis of myoepithelial carcinoma arising from minor salivary glands was made. No metastatic lesions were found by various imaging techniques. The patient is now receiving palliative radiation therapy 2 months after the operation. The present case showed that MC can express KIT and PDGFRA.     

Necrotizing (malignant) otitis externa: an unusual localization of mucormycosis

Malignant otitis externa (MOE) is a severe infection of external auditory canal and skull base. A 17-year-old diabetic girl was admitted with diabetic ketoacidosis. Cellulitis of her right ear occurred on the second day of hospitalization and a black necrotic scar in the same region appeared on the next day. The lesion rapidly invaded to right side of neck and surrounding tissue of the patient. Therefore, antimycotic therapy was started. Unfortunately the patient died on seventh day of hospitalization because of probably extensive fungal invasion. Physicians should suspect MOE connected to mucormycosis especially in patients with cutaneous lesions of ear unresponsive to antibiotic therapy.     

Paucicellular infiltrating ductal carcinoma of pancreas: an unusual variant

A 65-year-old man presented with obstructive jaundice from a 3-cm mass in the head of the pancreas. A Whipple resection was performed. Histologic examination of the tumor showed scanty tumor cells arranged in a single-file manner set within a desmoplastic sclerotic stroma. In other areas, the tumor was arranged around preexisting ducts in a targetoid fashion. Very focal ductal and signet-ring cell differentiation was noted. This unusual variant of pancreatic cancer was characterized by paucicellularity and a pattern of invasion resembling lobular carcinoma of breast.     

An unusual hepatocellular carcinoma: an ultrastructural and immunohistochemical study

This paper describes a 50-year-old woman with an unusual malignant tumour of the liver. Poorly differentiated liver cells, tubular structures and spindle cells in abundant intercellular ground substance characterized the tumour. Extremely high serum concentration of alpha-fetoprotein was present. The spindle cells showed strong immunoreactivity against this antigen and vimentin. Both the spindle and epithelial cells showed negative immunoreactivity against desmin, epithelial membrane antigen and keratin. Markers for low molecular cytokeratin (MAK-6, CAM 5.2) were demonstrated in both cell types. Ultrastructurally the neoplastic epithelial cells showed frequent intercellular spaces and well-formed brush borders suggestive of differentiation towards hepatocytes and bile duct epithelium. The immunohistological findings established the epithelial nature of the spindle cells thus distinguishing the tumour from hepatoblastoma.