Villo-glandular adenoma or polypoid dysplasia of esophagus: a case report

Gastrointestinal adenomas are neoplasms of glandular epithelium containing dysplasia of varying degrees. They are rare in esophagus. A case of villous tumor of the esophagus in a 71-year-old man is described. Histologically, this polypoid, villous lesion was developed on Barrett's esophagus and contained dysplastic epithelium and small adenocarcinomatous foci. This case illustrates that esophagus adenoma may be a premalignant lesion like other adenomas of the gastrointestinal tract. The term of polypoid dysplasia has therefore been recommended. This lesion, usually developed in gastric heterotopy or Barrett's esophagus, is often associated with high-grade dysplasia or carcinoma.     

Infiltrating granular cell tumor of the esophagus: a description of two cases

Granular cell tumor of the esophagus is an unusual tumor. It presents usually as a small and well limited lesion, localized in the mucosa or the submucosa. We report two cases of granular cell tumor of the esophagus, remarkable for their infiltrative growth. The tumor invaded the esophageal muscularis propria in one case and went through the adventitia in the other. There was no recurrence 1 year and 7 years after surgery, despite an incomplete resection in the second case. Thirteen cases of infiltrative granular cell tumors of the esophagus have been published. They are usually responsible for dysphagia. They can invade the muscularis propria and the adventitia as well as the periesophageal organs. There is no recurrence, even after an incomplete resection. The infiltrative feature of the granular cell tumors of the esophagus, by itself, cannot be considered as a malignant feature. The diagnosis of malignant granular cell tumor of the esophagus lies on the discover of metastases.     

Granular-cell tumor of the esophagus: Report of a case with a cytologic diagnosis based on esophageal brushing

We report a case of granular-cell tumor (GCT) in the esophagus. This is a rare location for this tumor and only 80 cases have been described. The tumor was diagnosed in a 24-yr-old woman with epigastralgia. Endoscopic examination revealed a submucosal ulcerated lesion. The smears derived from esophageal brushings contained clusters of granular cells. Histologic examination of the endoscopic biopsy provided diagnostic confirmation of GCT. To our knowledge, this is the first reported case of esophageal GCT in which the characteristic tumor cells were seen in the endoscopic brushing material. This case demonstrates that a cytologic diagnosis of GCT is possible if the lesion is ulcerated and/or brushing is subsequent to taking the biopsy. Although rare in the esophagus, GCT should be considered in the differential diagnosis of esophageal neoplasms. Diagn. Cytopathol. 1998;19:455–457. © 1998 Wiley-Liss, Inc.     

Esophageal anthracosis: lesion mimicking malignant melanoma

A case of anthracosis of the esophagus is reported. The patient was a previously healthy 69-year-old Japanese woman. A black and slightly elevated lesion was detected in her esophagus by upper gastroesophageal fiberoscopic examination. Endoscopically, the lesion looked like malignant melanoma. Thoracic esophagotomy was then performed. Histological examination revealed a pigmented lesion beneath the mucosal epithelial layer. The lesion consisted of an aggregation of histiocytes containing an abundance of tiny black pigments. A few mature lymphocytes and plasma cells were also evident in the periphery of the lesion. Histologically, these findings looked like lymph nodes in the pulmonary hilus; however, no lymph nodal structure was evident in the esophageal wall. Traction diverticula were also noted in the pigmented lesion. The patient has remained well without disease for 9 months since the surgery. Although anthracosis is a rare condition in the esophagus, the present case gave warning to pathologists and clinicians that it does indeed occur. Endoscopists and pathologists should differentiate anthracosis from malignant melanoma because the treatment and outcome are quite different for each.     

Malignant glomus tumor with oncocytic features: an unusual presentation of dysphagia

Glomus tumors in the gastrointestinal tract are unusual, as the previous series in the literature have been mainly limited to the stomach. Less than 10 cases of esophageal glomus tumors have been described in the literature. Oncocytic glomus tumors are a recently identified, rare variant of the glomus tumor. We report a 47‐year‐old female who presented with an approximately 3‐month history of dysphagia and weight loss. Upper gastrointestinal endoscopy showed a black‐purple, hypervascular, protruding lesion measuring approximately 65 mm at the 37th cm of the esophagus. The patient underwent an Ivor Lewis operation via open thoracotomy. The resected specimen had a protuberant, ulcerated mass measuring 80 × 35 mm in the posterior wall of the esophagus. Based on the histopathological, immunohistochemical and electron microscope findings, the final diagnosis was a malignant glomus tumor with oncocytic features. To our knowledge, this is the first report of a malignant glomus tumor with oncocytic features in an esophageal location.     

Sialadenoma papilliferum of the bronchus: Rare tracheobronchial tumor of salivary gland type

Presented herein is the second case of sialadenoma papilliferum of the bronchus in a 75-year-old man. The bronchial tumor had an exophytic papillary lesion protruding into the lumen of the ostial region of the right superior lobe bronchus on chest CT and bronchoscopy. Grossly, the resected tumor was pedunculated, 5 × 3 × 3 mm in size and consisted of both an exophytic papillary lesion protruding into the bronchial lumen and a glandular component in the bronchial submucosa. Histologically, the papillary structures were lined by ciliated or non-ciliated columnar epithelium and metaplastic non-keratinizing stratified squamous epithelium. The submucosal glandular components were well circumscribed beneath and in continuity with the papillary lesion. The glandular components of cysts and duct-like structures were lined by a double-layer epithelium composed of luminal cells and basal cells. Sialadenoma papilliferum is a rarely recognized salivary gland tumor, mostly occurring at intraoral mucosal sites including the hard and soft palate and buccal mucosa. There have been sporadic cases of sialadenoma papilliferum of the esophagus, nasopharynx and nasal cavity. To the authors' knowledge only one case of sialadenoma papilliferum of the bronchus has been reported previously.     

Hepatoid adenocarcinoma in Barrett's esophagus associated with achalasia: first case report

We report an unusual hepatoid adenocarcinoma in Barrett's esophagus with achalasia, which developed in a 44-year-old Japanese woman. The patient received an esophago-gastrectomy after diagnosis of the tumor and achalasia at the lower esophagus, 4 months before her death due to multiple metastatic tumors of the liver. The main granular tumor removed surgically was a hepatoid adenocarcinoma, mainly composed of clear cancer cells (alpha-1 antitrypsin, albumin and alpha-fetoprotein positive), with elements of choriocarcinoma and tubular adenocarcinoma. Non-neoplastic specialized columnar epithelium was present extensively near the oral side of the tumor edge in the esophagus, indicating Barrett's esophagus. This unusual tumor was therefore considered to have originated in Barrett's esophagus. The gastroesophageal reflux was presumed to have occurred for a long period, as there was a well-preserved fundic gland in the stomach and a history of frequent vomiting from the patient's youth, accounting for the appearance of achalasia.     

Double muscularis mucosae in Barrett's esophagus.

To clarify the histology and morphogenesis of the double muscularis mucosae in Barrett's esophagus, eight specimens resected from patients with Barrett's esophagus were compared histopathologically with 352 specimens resected from patients without Barrett's esophagus. A double muscularis mucosae was observed in seven (87.5%) of the eight cases with Barrett's esophagus, but in none of the 352 cases without Barrett's esophagus. The mucosa in the segment of Barrett's esophagus consisted of columnar epithelium, a superficial lamina propria, a superficial muscularis mucosae, a deep lamina propria, and a deep muscularis mucosae. The distal end of the superficial muscularis mucosae was connected to the deep muscularis mucosae at the esophagogastric junction, and its proximal end was located in fibrous tissue below the squamocolumnar junction of the mucosal epithelium or the distal edge of the erosive lesion. The deep muscularis mucosae in the portion with Barrett's esophagus was continuous with the original muscularis mucosae of the proximal esophagus and muscularis mucosae of the stomach. Barrett's esophagus is considered to be not merely a metaplastic lesion within the epithelium, but a newly developed lesion containing columnar epithelium, lamina propria, and a superficial muscularis mucosae on the lamina propria of the esophageal mucosa.     

Dysphagia in a patient with a history of large B-cell lymphoma: esophageal disease with negative biopsy findings

A patient with a previous diagnosis of lymphoma showed signs of dysphagia. Endoscopy found a lesion of the esophagus. Brush cytology and biopsy sampling were accomplished. The biopsy showed inflammation and granulation tissue but no tumor. The cytology specimen, however, was diagnostic of lymphoma. This case emphasizes the need for obtaining cytologic specimens concurrently with biopsies of esophageal lesions.     

Brunner gland hamartoma with predominant adipose tissue and ciliated cysts

We report an unusual case of Brunner gland hamartoma of the duodenal bulb with multiple ciliated cysts in a 43-year-old man. The patient presented with regurgitation. An endoscopy of the upper digestive tract revealed the presence of a pedunculated tumor of the first duodenum. Surgical resection of the antrum and first duodenum was performed. The polypoid lesion measured 3.5 cm and was located in the submucosa. It was composed of predominant adipose tissue containing hyperplastic lobules of Brunner gland and cystic ducts lined by ciliated cells. Brunner gland hamartoma is a rare benign duodenal lesion; it is usually composed of mature but disorganized tissues and is found most commonly in the duodenum. The presence of ciliated cells in the gut mucosa is a rare phenomenon, which has already been described in the esophagus and stomach. We report an unusual case of ciliated metaplasia in a duodenal Brunner gland hamartoma.     

Esophageal squamous cell carcinoma characterized by extensive chondroid differentiation

A rare case of carcinoma characterized by extensive chondroid elements at a site of primary esophageal and metastatic lesion is reported. The patient was a 67-year-old man complaining of dysphagia due to an ulcerative lesion at the lower middle esophagus. He underwent irradiation treatment prior to surgery. Histologically, the tumor consisted of both carcinomatous and chondroid elements and had invaded deeply into the esophageal wall. The carcinomatous cells had gradually become chondroid cells embedded within an extensive extracellular matrix. In addition, the metastatic lesion showed findings similar to those of the primary lesion. Immunohistochemistry revealed that both carcinomatous and chondroid elements were immunostained with cytokeratin and epithelial membrane antigen, suggesting an epithelial nature to the chondroid cells. Conversely, only chondroid cells were positively stained for S-100 protein. Furthermore, bone morphogenetic proteins (BMP) were positive for chondroid cells and their surrounding carcinomatous cells. Given the apparent transition between carcinomatous and chondroid cells based on microscopy and immunohistochemical findings in the present case, we concluded that the chondroid cells were derived from carcinomatous cells. In addition, our findings suggest that BMP produced by carcinomatous cells lead to chondroid differentiation of the carcinoma cells.     

A cooled intraesophageal balloon to prevent thermal injury during endocardial surgical radiofrequency ablation of the left atrium: a finite element study

Recent clinical studies on intraoperative monopolar radiofrequency ablation of atrial fibrillation have reported some cases of injury to the esophagus. The aim of this study was to perform computer simulations using three-dimensional finite element models in order to investigate the feasibility of a cooled intraesophageal balloon appropriately placed to prevent injury. The models included atrial tissue and a fragment of esophagus and lung linked by connective tissue. The lesion depth in the esophagus was assessed using a 50 degrees C isotherm and expressed as a percentage of thickness of the esophageal wall. The results are as follows: (1) chilling the esophagus by means of a cooled balloon placed in the lumen minimizes the lesion in the esophageal wall compared to the cases in which no balloon is used (a collapsed esophagus) and with a non-cooled balloon; (2) the temperature of the cooling fluid has a more significant effect on the minimization of the lesion than the rate of cooling (the thermal transfer coefficient for forced convection); and (3) pre-cooling periods previous to RF ablation do not represent a significant improvement. Finally, the results also suggest that the use of a cooled balloon could affect the transmurality of the atrial lesion, especially in the cases where the atrium is of considerable thickness.     

So-called carcinosarcoma of the esophagus: report of a case

The carcinosarcoma of the esophagus is a rare malignant neoplasm consisting of both carcinomatous and sarcomatous components. A case of so-called carcinosarcoma of the esophagus is described herein. A 69-year-old man presented with dysphagia and was admitted to our hospital. Imaging studies revealed a localized ulcerative tumor in the middle intrathoracic esophagus without any invasion or metastasis. The patient was initially thought to have squamous cell carcinoma and underwent subtotal esophagectomy with lymphadenectomy. Final diagnosis of the tumor was so-called carcinosarcoma of the esophagus. The patient had an uneventful postoperative course and showed no evidence of recurrence or metastasis in the 4-year postoperative period.     

Intra-abdominal esophageal duplication cyst in an adult

Esophageal duplication cysts are congenital anomalies of the foregut that are rarely found in the abdomen. An accurate preoperative diagnosis is not always possible, so the definitive diagnosis can be made by histologic examination of the surgical specimen. We experienced a case of Intra-abdominal esophageal duplication cyst in a 52-year-old female, who initially presented with an esophageal submucosal tumor on upper gastrointestinal endoscopy. She did not have any gastrointestinal symptoms. Barium esophagography, chest computed tomography scan and endoscopic ultrasonography demonstrated the cystic lesion in the intra-abdominal esophagus. Transhiatal enucleation of the lesion was performed successfully via the abdominal approach with no postoperative complications. Histologic study showed that the cyst wall contained a two-layered muscle coat and the surface of the lumen was lined by pseudo-ciliated columnar epithelium. The patient has been doing well without any complaints for 3 months of follow-up period.     

Sclerosing mucoepidermoid carcinoma with eosinophilia of the thyroid: Case report of a rare lesion with novel genetic mutation

Sclerosing mucoepidermoid carcinoma with eosinophilia (SMECE) is a rare primary cancer of the thyroid. This tumor is analogous to other primary tumors of the salivary glands, breast, pancreas, and esophagus. We present a case of this rare tumor with characteristic clinical features, ultrasound images, cytopathology, histopathology, and a heretofore undocumented somatic gene mutation. Additionally, we provide a succinct review of the controversial literature for this uncommon lesion.     

Sebaceous glands in the esophagus.

We report a case of sebaceous glands in the esophagus diagnosed by endoscopic biopsy. The patient was a 47-year-old Korean man presented with postprandial pain of several months duration. An endoscopic examination disclosed an early gastric carcinoma in the gastric antrum and a 0.4 x 0.4 cm sized irregular lobulated nodule in the middle esophagus. Microscopically, the lobule was proven to be sebaceous glands in the submucosa. Possible histogenesis of this lesion is discussed.     

A case of primary esophageal B-cell lymphoma of MALT type,presenting as a submucosal tumor

The primary esophageal lymphoma is extremely rare, and shows various morphologic characteristics. Only a single case of mucosa-associated lymphoid tissue (MALT) type lymphoma confined to the esophagus has been reported in the literature. A 61-yr-old man was referred to our hospital for evaluation of an esophageal submucosal tumor (SMT) that had been detected incidentally by endoscopy. He had a history of pulmonary tuberculosis with long-term anti-tuberculosis medication 15 yr before, and also had a history of syphilis, which had been treated one year before. He had been taking a synthetic thyroid hormones for the past 10 months because of an autoimmune thyroiditis. Endoscopy showed a longitudinal round and tubular shaped smooth elevated lesion, which was covered with intact mucosa and located at the mid to distal esophagus, 31 cm to 39 cm from the incisor teeth. Endoscopic ultrasonography (EUS) showed a huge longitudinal growing intermediate- to hypo-echoic mass located in the submucosal layer with internal small, various sized honeycomb-like anechoic lesions suggesting germinal centers. Subsequently, he underwent a surgery, which confirmed the mass as a primary esophageal low-grade B-cell lymphoma of MALT type.     

Multifocal presentation of gangliocytic paraganglioma in the mediastinum and esophagus

Gangliocytic paraganglioma (GP) is a rare, typically benign tumor that shows neuroectodermal (neurosustentacular or Schwannian and neuronal) and neuroendocrine differentiation. Once thought to arise exclusively in the periampullary region as a solitary lesion, recent reports have documented both origin of GP in a variety of extra-duodenal sites as well as synchronous multifocal presentation of the tumor. Herein, we describe the first reported case of simultaneous occurrence of GP in the superior mediastinum and esophagus. A mass in the mid-distal esophagus and a separate mass in the superior mediastinum at the thoracic inlet were found in a 58-year-old woman by computed tomography scan. Subsequent biopsy of the superior mediastinal mass showed nests of epithelioid tumor cells coexisting with ganglioneuromatous elements, whereas biopsy of the esophageal mass showed nests of epithelioid cells with interspersed ganglion cells. The epithelioid tumor cells showed diffuse immunohistochemical expression of keratin (CAM 5.2), chromogranin, and synaptophysin supporting true neuroendocrine differentiation; ganglion cells expressed S-100 protein and neurofilament protein; and the spindled elements expressed S-100 protein, neurofilament protein, and glial fibrillary acidic protein indicating Schwannian differentiation. The finding of another GP occurring outside the periampullary region bolsters the argument for a stem cell origin of this unusual tumor.