Simultaneous Manifestation of Gangliocytic Paraganglioma and Heterotopic Pancreas of Ampulla of Vater Treated by Endoscopic Resection / 대한췌담도학회지

Gangliocytic paraganglioma is an uncommon tumor of digestive system that is usually found in the second portion of duodenum. It is generally considered benign tumor, although few reports of local recurrences and regional lymph node metastases have been made. Gangliocytic paraganglioma is characterized by its histologic pattern including ganglion cells, spindle cells and epithelioid cells. Heterotopic pancreas, also known as ectopic pancreas, is a pancreatic tissue appeared outside of its normal location lacking anatomic or vascular connection with the pancreas. In duodenum, it is a relatively unusual lesion that may be found incidentally during surgery or endoscopy. We present a case of 39-year-old woman with gangliocytic paraganglioma combined with heterotopic pancreas in the ampulla of Vater successfully treated by endoscopic resection.

Simultaneous Manifestation of Gangliocytic Paraganglioma and Heterotopic Pancreas of Ampulla of Vater Treated by Endoscopic Resection / 대한췌담도학회지

Gangliocytic paraganglioma is an uncommon tumor of digestive system that is usually found in the second portion of duodenum. It is generally considered benign tumor, although few reports of local recurrences and regional lymph node metastases have been made. Gangliocytic paraganglioma is characterized by its histologic pattern including ganglion cells, spindle cells and epithelioid cells. Heterotopic pancreas, also known as ectopic pancreas, is a pancreatic tissue appeared outside of its normal location lacking anatomic or vascular connection with the pancreas. In duodenum, it is a relatively unusual lesion that may be found incidentally during surgery or endoscopy. We present a case of 39-year-old woman with gangliocytic paraganglioma combined with heterotopic pancreas in the ampulla of Vater successfully treated by endoscopic resection.

Clinical characteristics of acute Q fever in Daegu area / 대한내과학회지

BACKGROUND/AIMS: Although only a few sporadic cases of Q fever have been reported in Korea, a total of 13 cases have been seen in our area. We performed this study to evaluate the clinical characteristics of these cases of acute Q fever. METHODS: Demographic features, clinical manifestations, laboratory and radiologic findings, and therapeutic outcomes of all cases were evaluated. Q fever was diagnosed using an indirect micro-immunofluorescence assay (MIFA) and polymerase chain reaction (PCR). RESULTS: A total of 13 patients with acute Q fever seen from January 2006 to August 2008 at three teaching hospitals in the Daegu Metropolitan City area were enrolled. The mean age was 49 years old (range, 24~76), and the male to female ratio was 11:2. Six (46.2%) cases had a history of animal contact. Fever (100%) was the most common manifestation, followed by myalgia (84.6%), headache (61.5%), anorexia (61.5%), and chills (61.5%). All cases were diagnosed with high titers of anti-phase II antibody (IgM> or = 1:50, IgG> or =1:200) and positive nested PCR for the 27-kDa OMP com-1 gene of Coxiella burnettii in the blood. In three cases, liver biopsies revealed the presence of compact fibrin-ring granulomas. No characteristics of pneumonia were diagnosed on chest X-rays. The predominant presentation was acute febrile illness with hepatitis, including three cases (27.3%) of severe cholestatic hepatitis. The most frequently used antimicrobial agent was doxycycline (84.6%), followed by azithromycin (7.7%). CONCLUSIONS: Acute Q fever may be added to the list of differential diagnosis of patients with acute febrile illness and hepatitis in the Daegu Metropolitan City area.

A Case of Sclerosed Hemangioma Mimicking Intrahepatic Cholangiocarcinoma / 대한소화기학회지

Hemangioma is one of the most frequently encountered benign hepatic neoplasm which can develop secondary degeneration. Sclerosed hemangioma is a rare disease histologically characterized by large amount of collagen and elastic fibril between sclerosed small vessels. Its differential diagnosis is very difficult. It should be included in the differential diagnosis of other hepatic lesions such as hepatocellular carcinoma, intrahepatic cholangiocarcinoma, and metastatic hepatic tumor. A 77-year old male was admitted with upper abdominal discomfort. Abdominal ultrasonography revealed GB stone, dilated common bile duct with bile duct stone, and a 4.6 cm sized hyperechoic mass at segment 5 and 6 of the liver. Abdominal dynamic computed tomography demonstrated dilated intrahepatic bile ducts and a 5x5 cm sized mass which showed minimally delayed enhancement. Abdominal magnetic resonance imaging revealed the mass with low signal intensity in T1 weighted image, high signal intensity and focal low signal in T2 weighted image which showed minimal enhancement. We removed common bile duct stone with endoscopic retrograde cholangiopancreatography then decided to undergo right lower segmentectomy of liver due to possibility of cholangiocarcinoma. Histopathological examination of hepatic mass showed large amount of fibrous tissue with occasional residual vascular channels. We describe one case of sclerosed hemangioma mimicking cholangiocarcinoma.

Spindle Cell/Pleomorphic Lipoma of the Oropharynx

We report a rare case of spindle cell/pleomorphic lipoma of the oropharynx. A 45-year-old woman presented with a 9-month history of a lump in 2001. A well demarcated polypoid, rubbery mass was found in the left vallecula and was surgically removed. The mass was diagnosed as a spindle cell lipoma. She revisited with the same complaint in 2008. Examination revealed another polypoid mass at the left aryepiglottic fold, near the previous excision site. The excised mass histologically consisted of mature fat cells, numerous bizarre giant cells, and bland spindle cells, features of a typical pleomorphic lipoma. This is the first case of recurrent oropharyngeal spindle cell/pleomorphic lipoma, showing histologic changes during the recurrence. Complete removal and follow-up are necessary for the treatment of this uncommon neoplasm.

Calcifying Fibrous Pseudotumor of the Stomach That Was Diagnosed by Endoscopic Submucosal Dissection / 대한소화기내시경학회지

Calcifying fibrous pseudotumor (CFPT) is very rare and especially at an intrinsic visceral location such as the stomach. The CFPT is generally located in the subcutaneous or deep soft tissue and the subserosal area of organs and it is mainly diagnosed during childhood or young adulthood. The etiology and pathophysiology of CFPT are unclear. Herein we describe a case of gastric CFPT and we review the related literature. A thirty-year-old asymptomatic female patient visited our hospital for the evaluation of a submucosal tumor that was seen on previous endoscopy. On the endoscopic ultrasonography (EUS), a subepithelial lesion was found at the gastric angle and it was a slightly hypoechoic, homogenous mass located at the submucosal layer of the gastric wall. Endoscopic submucosal dissection was performed under the suspicion of carcinoid. On the microscopic findings, most of the tissue was composed of hyalinized collagenous tissue. Mild lymphocytic infiltration and several dystrophic calcifications were also seen. Immunohistochemically, bcl-2 and CD34 staining was negative, so we diagnosed the mass as CFPT.

Six Cases of Congenital Tufted Angioma / 대한피부과학회지

Tufted angioma is a rare slowly progressive vascular tumor, characterized by histopathologic findings of numerous angiomatous lobules of "cannonball" appearance within the dermis. It affects children but occurs infrequently at birth. It appears as a clinical imitator of hemangioma of infancy (HOI), so it needs to be differentiated from HOI. Clinically, lesions most often present as red to purple, indurated or nodular plaque, and are usually located on the neck, upper trunk or proximal limb. We report six cases of tufted angioma of congenital onset. We undertook immunohistochemical study using CD31, D2-40 and GLUT1 to rule out other vascular tumors or malformations, particularly HOI. These cases may demonstrate the keys of differential diagnosis between HOI and tufted angioma, which share common features clinically and rarely histopathologically.

Pedunculated subpleural lipoma with incomplete torsion: A case report / 대한내과학회지

Lipoma is a common benign neoplasm, but lipoma arising from the pleura is rare. Most pleural lipomas are asymptomatic and are discovered incidentally on chest radiographs. Here we report an unusual case of a pedunculated subpleural lipoma with incomplete torsion in a 29-year-old woman who presented with flank pain. On chest radiography, there was a well-defined, ellipsoidal, homogeneous, radioopaque mass in the left lower lobe. An intrathoracic tumor was suspected, and video-assisted thoracic surgery was performed to establish the final diagnosis. The resected tumor was a 10 X 5 cm sized pedunculated pleural mass. The cut surface showed a yellow, well-circumscribed mass with areas of hemorrhage. Histologically, the tumor was composed of mature adipose tissue with fibrous septae composed of fibroblasts. The outer surface of the tumor was covered by mesothelial cells. Some areas were hypercellular, and occasional mitoses were found, but neither lipoblasts nor atypical mitoses were identified.

A Case of Exogenous Lipoid Pneumonia after Ingestion of Squalene in Patients with Achalasia / 결핵및호흡기질환

Exogenous lipoid pneumonia (ELP) is a chronic inflammatory reaction of the lungs resulting from the aspiration of vegetable, animal or mineral oils. Squalene, is a derivative of shark liver oil that is taken as a traditional remedy in some Asian countries, and is used widely also in cosmetics. Similar to the symptoms in most cases of oil aspiration, the symptoms of squalene-induced lipoid pneumonia are either absent or nonspecific. Hence, the disease is generally detected incidentally. Although many cases with predisposing factors have been reported, ELP with achalasia is quite rare. We report a 47-year old woman with achalasia who developed ELP after ingesting squalene. The patient was treated successfully by supportive care and surgical treatment of the achalasia.

Surgical Resection of Hepatic Metastasis of Thymoma

The thymoma with extrathoracic metastasis is an exceedingly rare malady. We report here on the surgical resection and the pathologic findings of liver metastasis from an invasive thymoma in a 57-year-old female. In 1998, a benign thymoma in the anterosuperior mediastinum was removed from the patient and then postoperative radiation therapy was given. After 6 years, the patient complained of dyspnea, dry cough and abdominal pain. Abdominal CT and MRI showed an encapsulated and septated 19 x 14 cm sized cystic mass in the right liver. Right hepatectomy was done and it was shown to be a metastatic thymoma. An extensive literature review has revealed only a few cases of surgical resection of liver metastasis in a patient with invasive thymoma.

Hepatic Metastasis of Thymoma: A Case Report

Thymoma is the most common neoplasm in the anterior mediastinum, and extrathoracic involvement is rare. Moreover, cystic liver metastasis is extremely rare; few cases have been reported in the literature to date. We report here on a case of cystic liver metastasis of thymoma treated with surgical resection, describing the ultrasonography, CT and MRI findings.

Massive bleeding caused by bronchoscopic biopsy of an abnormal bronchial artery / 대한내과학회지

A 39 years old woman was admitted to the hospital because of recurrent hemoptysis. On a bronchoscopic inspection, nodular shaped mass-like lesions were found on the orifice of right middle and lower lobar bronchus and a biopsy was performed. This was immediately followed by massive bleeding into the airways. The bleeding could not be controlled by nonsurgical treatment. Immediately she underwent a right middle and lower bilobectomy in order to control of bleeding. Biopsy revealed the bleeding to have been caused by a biopsy injury of an abnormal artery that had run superficially in the bronchial mucosa.

Massive bleeding caused by bronchoscopic biopsy of an abnormal bronchial artery / 대한내과학회지

A 39 years old woman was admitted to the hospital because of recurrent hemoptysis. On a bronchoscopic inspection, nodular shaped mass-like lesions were found on the orifice of right middle and lower lobar bronchus and a biopsy was performed. This was immediately followed by massive bleeding into the airways. The bleeding could not be controlled by nonsurgical treatment. Immediately she underwent a right middle and lower bilobectomy in order to control of bleeding. Biopsy revealed the bleeding to have been caused by a biopsy injury of an abnormal artery that had run superficially in the bronchial mucosa.

Sarcomatoid Cholangiocarcinoma of the Liver; a case study / 한국간담췌외과학회지

A 71 year-old man with persistent fever, right pleuritic pain and discomfort for 2 months was referred to hospital. An imaging study using ultrasonography, computed tomography and magnetic resonance imaging showed a huge hepatic mass, which was revealed to be a sarcoma-like mass of the right lobe of the liver. On laparotomy, 5, 6 and 7 segmentectomies were performed. The tumor was 18x16x15 cm in size. On cross section the tumor was a yellowish- fanning, diffusely soft, friable and clearly demarcated mass. Microscopically, the tumor cells were contained in the sarcomatous component (63%), tumor necrosis (36%) and adenocarcinoma component (0.6%) on histological mapping. Histologically, the tumor was composed of malignant osteoid tumor cells. Immunohistochemical staining for cytokeratin was positive in the sarcomatous components. These findings favored the possible epithelial origin of the sarcomatous cells. Thus, the diagnosis confirmed a sarcomatoid cholangiocarcinoma, with 2 osteosarcomatous components. After surgery, the pyrexia subsided, but early peritoneal and intrahepatic recurrence occurred 2 months postoperatively. He received systemic chemotherapy 6 times over 8 months, and partially responded. From our experienced the characteristics of this tumor have been found to be very aggressiveness, with poor prognosis.

Steatocystoma Multiplex in a Family

Steatocystoma multiplex is a rare, inherited disorder that is characterized by multiple, asymptomatic, variably sized dermal cysts. The condition is transmitted in an autosomal dominant fashion; although sporadic cases have been documented. Keratin 17 has been proposed to be an important factor in inherited steatocystoma. In this study, a 29-year old man has a 4-year history of asymptomatic, movable, skin-colored nodules on his face, neck, scalp, anterior chest and back. His father and elder-brother have similar lesions. Histologically, the cysts show a thin stratified squamous epithelium with sebaceous glands arising from its wall and an absence of the granular cell layer. Generally, there are two treatments-medical treatment and surgical treatment. In case of non- inflamed lesions, surgical excision or drainage is regarded as the best treatment. We tried excisional biopsy and until now there has been no recurrence in the operation area over the past 12 months following the operation.

Pulmonary Cryptococcosis: Report of 2 cases / 대한흉부외과학회지

Cryptococcus neoformans is a ubiquitous fungus found worldwide, particularly in soil contaminated by pigeon droppings. Pulmonary cryptococcosis occurs rarely in immunocompetent individuals. The risk of cryptococcal infection increases with the degree of immunal compromise, in human immunodeficiency virus infection especially. Pulmonary cryptococcosis is most frequently encountered as asymptomatic single or multiple pulmonary nodules found by routine chest x-ray examination. The diagnosis is most often made in these situations by the histology of the resected lesion. Acute progressive pneumonia may occur, with symptoms of cough, sputum production, fever, and weakness. The clinical picture is not pathognomonic. The yeasts are stained well by any of the special stains for fungi. Treatment is now indicated for all cases given a diagnosis of cryptococcosis, even if the diagnosis has been made by resecton of a solitary, asymptomatic pulmonary nodule.

Elastofibroma Dorsi: A case report / 대한흉부외과학회지

Elastofibroma dorsi, a rare, noncapsulated benign entity is characterized by the proliferation of fibrous tissue with elastin and occurs most often in the infrascapular area of elderly women. It is a relatively slowly growing lesion and no reports of malignant transformation exist. Which is overlooked easily because it rarely causes symptoms such as tenderness, pain, or restriction of movement. The diagnosis of elastofibroma is established by typical histopathologic findings. Radiographic evaluation may lead to a presumptive diagnosis. We experienced a case of elastofibroma dorsi in a 48-year-old woman and report this case with a review of the literature.

Desmoplastic Infantile Astrocytoma

Desmoplastic infantile astrocytoma is a huge cystic tumor, typically occurring in the cerebral hemispheres of infants. The authors report a 4-month-old baby presented with increased head circumference, bulging fontanel, and the setting-sun sign. Magnetic resonance imaging revealed a round mass with huge cystic component in the right cerebral hemisphere. Microsurgical gross total resection of the tumor was performed and pathological examination of the specimen showed features of desmoplastic infantile astrocytoma. The postoperative course was complicated by subdural hygroma which was managed by the placement of subduroperitoneal shunt. Although desmoplastic infantile astrocytomas are rare, it must be distinguished from other hemispheric tumors in infancy or childhood because of good prognosis.

A case of lipomatous hemangiopericytoma of the nasal cavity / 대한이비인후과학회지

Hemangiopericytoma (HPC) is an uncommon vascular tumor and arises from the proliferation of special cells called pericytes. In 1995, a unique variant of HPC, containing some mature fat, has been first described by Nielsen and was termed lipomatous hemangiopericytoma (LHPC). Clinically, LHPC appears to be indolent, with no recurrences or metastatic spreads. Histologically, LHPC is composed of a variable amount of benign lipomatous and hemangiopericytomatous components. Since the original description, there had been only 2 reports of these tumors, and none in Korea. We report a recent case of lipomatous hemangiopericytoma of the nasal cavity treated by endoscopic excision.

A Case of Adenosquamous Carcinoma of the Larynx / 대한이비인후과학회지

Malignant laryngeal tumors include a variety of histologic types with different biologic features, each calling for a different therapeutic approach and giving rise to a different prognosis. We experienced a case of laryngeal adenosquamous carcinoma which is an extremely rare neoplasm in larynx. The lesion is histologically distinctive because of the presence of mixed areas of unequivocal adenocarcinoma and squamous cell carcinoma. Differential diagnosis include adenoid squamous cell carcinoma and mucoepidermoid carcinoma. It is found that combined surgery and additional radiation therapy is necessary because the biologic behavior and prognosis of this neoplasm are considered as more aggressive and worse than that of conventional squameus cell carcinoma.