• Journal of the Korean Association of Oral and Maxillofacial Surgeons
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• v.49 no.6
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• pp.360-364
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• 2023

Xanthogranulomatous inflammation (XGI) is an uncommon type of chronic inflammation and is histologically characterized by foamy histiocytes and giant cells. The most common sites of occurrence are kidneys and gallbladder. The etiology remains controversial. Involvement of the lower jaw bone is rare. In this study, we report a case of XGI presenting in the lower jaw.

• Pediatric Gastroenterology, Hepatology & Nutrition
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• v.23 no.1
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• pp.110-114
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• 2020

Gastric xanthoma is frequently an incidental finding on upper endoscopy in adults. Gastric xanthomas (GX) can be mistaken for malignancies and warrant prompt histologic diagnosis. The underlying etiology is not fully understood; however, it has been linked to Helicobacter pylori gastritis and gastric cancer. GX in the pediatric population is largely unreported in the literature. Because of the relative rarity, documentation with case reports are essential to provide as much data as possible to see if there is a correlation between GX and malignant potential in the pediatric population. Our group is reporting two cases, a 10-year-old male and a 7-year-old male, both who presented with chronic dysphagia, upper abdominal pain, nausea, vomiting, and loss of appetite. Upper endoscopies for both patients revealed small polypoid lesions located in the antrum with foamy histiocytes on histology, leading to the diagnosis of gastric xanthoma.

• Tuberculosis and Respiratory Diseases
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• v.64 no.3
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• pp.224-229
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• 2008

Erdheim-Chester disease (ECD) is a rare disease that is characterized by multi-organ involvement of foamy histiocytes. It causes systemic inflammation, and also demonstrates various clinical manifestations and has a poor prognosis. We encountered a case of ECD in a patient that had been treated for underlying polycythemia vera. As far as we know, this is the first reported case worldwide where ECD developed in association with polycythemia vera. A 59-year-old man visited our hospital due to pleuric pain at the right side of the chest. Pleural tissue that was obtained following a thoracoscopic biopsy showed non-Langerhan's cell histiocytosis, suggesting the presence of ECD. The histiocytes stained positively for CD68, but were negative for S-100 and CD1a. The patient also complained of pain at both hips and the right shoulder area. An X-ray and magnetic resonance image demonstrated that the lesion showed sclerosis and osteolysis in both the proximal femur and right humerus. Treatment was started with predinisolone, and subsequently cyclophosphamide was added. ECD is a very rare multi-systemic disease, and its cause and therapeutic options have not yet been defined. ECD has a poor prognosis. Therefore, we believe that additional case studies are needed prior to the determination of a novel therapy for ECD.

• Journal of Genetic Medicine
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• v.4 no.2
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• pp.200-203
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• 2007

Niemann-Pick type C is an inborn error of metabolism that affects lipid degradation and storage, which is characterized by hepatosplenomegaly and progressive neurological symptoms. A 7-month-old girl with jaundice was presented cholestasis and hepatosplenomegaly. Laboratory study showed elevated acid phosphatase, angiotensin converting enzyme and mild decrease of cholesterol. Characteristic foamy cell and sea-blue histiocytes in bone marrow biopsy consistent with Niemann-Pick disease. Niemann-Pick type C was suspected by past medical history and findings of physical examination. Therefore, molecular analysis was performed and found mutations of NPC1 gene. We report the first Korean case of type C Niemann-Pick disease confirmed by mutation analysis.

• The Korean Journal of Cytopathology
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• v.9 no.1
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• pp.45-54
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• 1998

Fine needle aspiration cytology is considered as a useful diagnostic procedure in management of patients with breast lesions. This study was undertaken to evaluate the scoring system of Masood in the interpretation of breast aspirates, to establish the most useful cytologic criteria for the diagnosis of breast lesions, and to subclassify the benign breast diseases. To assess the feasibility of a cytologic grading system, 57 cases of benign breast disease, 61 cases of malignant breast disease were studied, respectively. The aspirates were evaluated for the cellular arrangement, the degree of cellular pleomorphism and anisonucleosis, and the presence of myoepithelial cells and nuceoli. Values ranging from 1 to 4 were assigned to each criterion and the sum of the individual values was made for each case. The presence of stroma, apocrine metaplasia, foamy histiocytes and inflammatory cells, background of the smear, and cellularity were also evaluated. Cut-off value of the scoring system of Masood between benign and malignant lesion was 15. Among the cytologic criteria, cellular arrangement, presence of myoepithelial cells, nucleoli, and stroma, status of chromatin pattern, and background of smear were useful criteria in the differentiation between benign and malignant lesions. Application of the scoring system of Masood does not always make the accurate diagnosis and the subclassification of benign breast disease.

• The Korean Journal of Cytopathology
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• v.17 no.1
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• pp.63-68
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• 2006

Inflammatory myofibroblastic tumor (IMT), normally referred to as inflammatory pseudotumor, is a fairly rare condition. Fine needle aspiration cytology (FNAC) of IMT has only rarely been reported. Here, we describe one such case of pulmonary inflammatory myofibroblastic tumor. A 30-year-old man presented with a 2.8cm-sized mass in his lung. Chest CT revealed a well defined, poorly enhancing mass. FNAC showed some fascicular or swirled clusters of spindle cells, admixed with occasional inflammatory cells and foamy histiocytes. The majority of the tumor cells evidenced bland, elongated nuclei, but infrequent pleomorphic nuclei. Some of the tumor cells evidenced nuclear grooves and intranuclear inclusions. Although the cytological differentiation of IMT from malignant lesions is not immensely problematic, due to the general paucity of cytological and nuclear atypia, a definite cytological diagnosis of IMT cannot be rendered simply by FNAC. Therefore, a diagnosis of IMT may be suggested via exclusive diagnosis.

• Journal of Yeungnam Medical Science
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• v.15 no.2
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• pp.359-370
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• 1998

Eight cases of chonproblastoma were studied by analyzing the clinical and pathologic findings. The age of eight cases ranged from 17 to 38 years old(median age, 22.7 years old). The tumors developed in the femur(3 cases), patella(2 cases), tibia(1 case), fibula(l case), and ulna(1 case). The mean diameter of tumors was 4.0cm (range, 1.5 to 8.0cm). Grossly, tumors showed grayish brown solid area with foci of secondary aneurysmal bone cyst. Histologically, the tumor cells were round or polygonal in shape with nuclear groove. And there were chondroid differentiation(7 cases), mitosis(3 cases), calcific deposits(3 cases), secondary aneurysmal bone cyst(4 cases), hemosiderin deposits(4 cases), necrosis(3 cases), vascular invasion(1 caes), and foamy histiocytes and cholesterol cleft(l cases). All cases showed no metastasis to lymph node and distant organ. Seven cases (87.5%) were immunoreactive for S-100 protein. None was immunoreactive for cytokeratin.

• Journal of The Korean Ophthalmological Society
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• v.59 no.11
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• pp.1071-1076
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• 2018

Purpose: To report a case of immunoglobulin G4 (IgG4)-related ophthalmic disease associated with adult xanthogranulomatous disease. Case summary: A 38-year-old male with a history of cholecystectomy visited our clinic for bilateral periorbital swelling. Histopathology of the orbital biopsy showed diffuse infiltration of foamy histiocytes with Touton giant cells and lymphoid follicles, with a diagnosis of adult-onset xanthogranuloma. After excisional biopsy, he was treated with azathioprine and prednisolone. Four years after treatment, he again visited the clinic due to bilateral, yellowish eyelid masses. Serological examinations were all nonspecific findings, except for elevation of IgG and IgG4 levels. Magnetic resonance imaging showed bilateral symmetric soft tissue enlargement with slightly heterogeneous T1/T2 isosignal intensity, with contrast enhancement at the superolateral aspect of extraconal spaces. Excisional biopsy and blepharoplasty were performed. Immunohistochemical sections showed that the IgG4+/IgG plasma cell ratio was 10-20% and the IgG4 plasma cell count was 22/high power field (HPF). His past sections of 2013 from the pathology department were again stained and showed that the IgG4+/IgG plasma cell ratio was 40-50% and the IgG4 plasma cell count was 59/HPF. Thus, he was definitely diagnosed with IgG4-related ophthalmic disease. Conclusions: If there is recurrent eyelid swelling, IgG4-related ophthalmic disease should be considered as a differential diagnosis. And the patient with adult xanthogranulomatous disease can be diagnosed with IgG4-related ophthalmic disease.