Purpose: To report a case of keratitis caused by Brevundimonas diminuta/vesicularis.Case summary: A 20-year-old man presented with decreased visual acuity and glare in his right eye from 2 days earlier. At the first visit, slit lamp examination revealed a 1.5 × 1.5 mm2-sized stromal infiltration, an edematous cornea and endothelial deposits in the center of the right eye. Microbiological examinations were performed from corneal scrapings. Treatment was started with topical eye drops (fortified cefazolin, fortified tobramycin) as empirical antibiotics. After several days, the cultures revealed Brevundimonas diminuta/vesicularis. After treatment with empirical antibiotic eye drops, the symptoms and visual acuity improved. Conclusions We report a case of infectious keratitis caused by Brevundimonas diminuta/vesicularis, which has never previously been reported in the Republic of Korea.

Purpose: To report two cases of adult inclusion conjunctivitis masquerading as chronic conjunctivitis.Case summary: (Case 1) A 34-year-old man presented with a foreign body sensation in his right eye for 2 months. At the first visit, slit lamp examination showed follicles on the inferior palpebral conjunctiva. Chlamydia trachomatis immunoglobulin M (IgM) antibody and polymerase chain reaction (PCR) tests were positive. The patient was prescribed doxycycline for 1 week, which improved the symptoms, followed by gentamicin and levofloxacin for an additional 5 days, which further improved the clinical symptoms and conjunctivitis. (Case 2) A 39-year-old man presented with right eye pain and redness for 5 months. At the first visit, slit lamp examination showed follicles on the inferior palpebral conjunctiva. Chlamydia trachomatis IgM antibody and PCR tests were positive. After 1 week of doxycycline treatment, the clinical symptoms and conjunctivitis improved. Conclusions Because adult inclusion conjunctivitis caused by chlamydial infection has non-specific symptoms, it can be misdiagnosed as chronic conjunctivitis. A detailed medical history and appropriate diagnostic tests are necessary to make this diagnosis.

PURPOSE: We aimed to analyze the frequency of eosinophilia-associated diseases and to search for possible markers that may be useful for their differential diagnosis. METHODS: We retrospectively reviewed the medical records of 148 patients with peripheral blood eosinophil count of more than 500/µL who visited the Allergy Department of Chonnam National University Hospital for the first time from January to December 2016. Blood eosinophilia was categorized as mild (<1,500/µL), moderate (1,500–5,000/µL), and severe (>5,000/µL). RESULTS: Blood eosinophilia was mostly caused by allergic diseases (41.9%), parasitic infestation (23.6%), and drug allergy (19.6%). Eosinophil count was higher in patients with parasitic infestation (P<0.01), drug allergy (P<0.01), hypereosinophilic syndrome (HES, P<0.001), or eosinophilic granulomatosis with polyangiitis (EGPA, P<0.001) than in those with allergic diseases. The eosinophilic cationic protein level was higher in patients with HES than in those with allergic diseases (P<0.05) and parasitic infestation (P<0.05). The total IgE level was lower in patients with HES than in those with parasitic infestation (P<0.05) and EGPA (P<0.05). The vitamin B12 level was higher in patients with HES than in those with parasitic infestation (P<0.05). There was no statistically significant difference in tryptase levels between the groups. The most common cause of mild eosinophilia was allergic diseases (59.8%), followed by parasitic infestation (22.7%) and drug allergy (13.4%). The common causes of moderate eosinophilia were drug allergy (37.8%), parasitic infestation (29.7%), and allergic diseases (10.8%). The common causes of severe eosinophilia were EGPA (28.6%), HES (21.4%), parasitic infestation (14.3%), and drug allergy (14.3%). CONCLUSION: Common causes of blood eosinophilia in patients who visit the allergy department are allergic diseases, parasitic infestation, and drug allergy. Several markers, including eosinophil count, total IgE, and vitamin B12, may be useful for the differential diagnosis of eosinophilia-associated diseases.
Diagnosis, Differential ; Drug Hypersensitivity ; Eosinophilia ; Eosinophils ; Granulomatosis with Polyangiitis ; Humans ; Hypereosinophilic Syndrome ; Hypersensitivity ; Immunoglobulin E ; Jeollanam-do ; Medical Records ; Parasitic Diseases ; Retrospective Studies ; Tryptases ; Vitamin B 12

Chronic urticaria may often be associated with interleukin (IL)-1-mediated autoinflammatory disease, which should be suspected if systemic inflammation signs are present. Here, we report a case of Schnitzler's syndrome without monoclonal gammopathy treated successfully with the IL-1 receptor antagonist anakinra. A 69-year-old man suffered from a pruritic urticarial rash for 12 years. It became aggravated episodically and was accompanied by high fever, arthralgia, leukocytosis, and an elevated C-reactive protein and erythrocyte sedimentation rate. The episodes each lasted for over one week. Neutrophilic and eosinophilic inflammation was found on skin biopsy. However, serum and urine electrophoresis showed no evidence of monoclonal gammopathy. The cutaneous lesions were unresponsive to various kinds of anti-histamines, systemic glucocorticoids, colchicine, cyclosporine, dapsone, and methotrexate, which were administered over a span of 3 years immediately preceding successful treatment. A dramatic response, however, was observed after a daily administration of anakinra. This observation suggests that the correct diagnosis of this case is Schnitzler's syndrome without monoclonal gammopathy. For an adult patient with refractory chronic urticaria and systemic inflammation, Schnitzler's syndrome could be considered as a possible differential diagnosis. Although the typical form of Schnitzler's syndrome exhibits the presence of monoclonal gammopathy as a diagnostic criterion, monoclonal gammopathy may be absent in an atypical form. In such a situation, an IL-1 antagonist should be effective for the management of chronic urticaria.
Aged ; Blood Sedimentation ; C-Reactive Protein/metabolism ; Chronic Disease ; Humans ; Interleukin 1 Receptor Antagonist Protein/therapeutic use ; Leukocytes/metabolism ; Male ; Paraproteinemias/complications ; Schnitzler Syndrome/blood ; Schnitzler Syndrome/drug therapy ; Urticaria/complications

Sarcoidosis is a systemic disease of unknown cause involving multiple organs and is characterized by noncaseating granuloma. Immune thrombocytopenia (ITP) is an autoimmune disease characterized by increased peripheral platelet destruction due to the presence of an antibody to the platelet and abnormal platelet production. There is no known pathogenesis that occurs concurrently with ITP and sarcoidosis. However, considering together of 2 known pathogenesis, abnormal immune response triggers either ITP or sarcoidosis. The disease that develops first stimulates secondary disease. After development of secondary disease, they stimulate each other. A few cases of ITP associated with sarcoidosis are well documented in English; however, the disease has rarely been reported in Korea. Here, we report on a case of ITP with sarcoidosis in a 29-year-old man. He suffered from easy bruising. The chest X-ray and the contrast-enhanced computed tomography scan showed bihilar lymphadenopathy and reticulonodular infiltrates. Bone marrow study and fluoroscopy-guided percutaneous needle biopsy were performed and the patient was diagnosed with sarcoidosis and ITP. He was put on 400 mg/kg of intravenous immunoglobulin for 5 days and administered oral steroids and further follow-up will be carried out. He has shown a good response without significant bleeding event. However, administration of more oral steroid and additional follow-up is required than for single disease, whether sarcoidosis or ITP.
Adult ; Autoimmune Diseases ; Biopsy, Needle ; Blood Platelets ; Bone Marrow ; Granuloma ; Hemorrhage ; Humans ; Immunoglobulins ; Korea ; Lymphatic Diseases ; Sarcoidosis* ; Steroids ; Thorax ; Thrombocytopenia*

Type 1 neurofibromatosis (von Recklinghausen's disease, NF-1) is an autosomal-dominant neurocutaneous-disorder characterized by systemic cafe'-au-lait spots, multiple cutaneous neurofibromas, axillary or inguinal freckling, and Lisch nodules (pigmented iris hamartomas). Approximately 10-25% of NF1 patients have gastrointestinal neoplasms. Gastrointestinal stromal tumor (GIST) in patients with neurofibromatosis is most commonly found in the small bowel and the stomach, and approximately 60% of such patients have multiple tumors or multiple tumor sites. Although, the increased incidence of GIST in patients with neurofibromatosis is well documented in pathology literature in English, but has rarely been documented in Korea. Here, we report a case of multiple GISTs in a 48-year-old woman accompanied by NF1. She was admitted to Yeungnam University Hospital with complaints of melena and dyspnea. A contrast-enhanced computed tomography (CT) scan revealed that multiple soft tissue masses were occupying the entire peritoneal cavity. An ultrasonogram- guided biopsy was performed and the tumors were found to have been composed of tumor cells that were positive for c-kit protein. The patient was put on Imatinib mesylate treatment, and further follow-up will be carried out.
Biopsy ; Dyspnea ; Female ; Follow-Up Studies ; Gastrointestinal Neoplasms ; Gastrointestinal Stromal Tumors* ; Humans ; Incidence ; Iris ; Korea ; Melena ; Mesylates ; Middle Aged ; Neurofibroma ; Neurofibromatoses ; Neurofibromatosis 1* ; Pathology ; Peritoneal Cavity ; Proto-Oncogene Proteins c-kit ; Stomach ; Imatinib Mesylate

Objectives: . In subset of patients, acinic cell carcinoma (AcCC) exhibits aggressive features such as recurrence, distant metastasis, and mortality. This study aimed to investigate clinicopathologic factors influencing patients’ prognosis and to identify adverse features predictive of an unfavorable prognosis. Methods: . Between January 2000 and December 2016, 59 patients with AcCC were enrolled in this study. Results: . The patients’ 5-year overall survival rate was 93.3%, and their 5-year recurrence-free survival rate was 80.5%. During the study period, recurrence occurred in 10 patients. The mean time to recurrence after surgery was 26 months (range, 5–60 months). During the study period, three patients died from the disease. Univariate analysis showed that sex, surgical extent, extranodal extension, T classification, and TNM stage were significantly associated with disease recurrence. Multivariate analysis showed that, among the clinicopathologic factors included in the analysis, only TNM stage displayed a statistically significant correlation with disease recurrence. Conclusion . Surgical treatment alone yielded good results for AcCC, and additional treatment did not affect the recurrence-free survival rate or the overall survival rate, even when the resection margin was less than 1 mm. Other pathologic factors did not show prognostic significance for disease recurrence or death.

Background: Acute pulmonary thromboembolism (APTE) is often confused with myocardial infarction. Previous studies have shown that patients with APTE exhibit lower initial and peak cardiac troponin I (CTI) levels, but higher D-dimer (DD) levels, than patients with myocardial infarction. The present study aimed to reaffirm the tree model algorithm using an entirely new set of data. Methods: We reviewed retrospective clinical and laboratory data from patients who were diagnosed with APTE or non-ST-elevation myocardial infarction (NSTEMI) between 2015 and 2016. Subjects who were not classified with a diagnosis or did not have their CTI or DD levels assessed were excluded. We categorized patients according to the previous algorithm and compared the outcomes with the previous test dataset. Results: The analysis involved data from 156 patients with APTE and 363 patients with NSTEMI. In the validation data set, the APTE group showed higher initial DD levels (9.80±10.84 μg/mL) and lower initial CTI levels (0.17±0.54 μg/mL) than the NSTEMI group. The accuracy rate for the test dataset and the validation set were similar. The test set accuracy rate was 91.0%, while the accuracy rate in the validation set improved to 88.6%. Conclusions Patients with APTE exhibited lower initial and peak CTI levels, but higher DD levels than NSTEMI patients. The accuracy rate estimates were similar between the test set obtained from the tree model algorithm and the validation set. The study findings demonstrate that the assessment of cardiac biomarkers can be useful for differentiating between APTE and NSTEMI.