Aged ; Female ; Humans ; Lingual Thyroid ; complications ; drug therapy ; physiopathology ; Oral Hemorrhage ; diagnosis ; etiology ; physiopathology

We report a case of hypereosinophilia causing multiple areas of cerebral infarcts. A 52-year-old Korean man presented with dysarthria and weakness in both arms. A brain MRI revealed multiple acute infarcts in the distal border zone with focal intracerebral hemorrhage, whereas a cerebral angiogram was not remarkable. The eosinophil count was 5,500/microLiter and was accompanied by elevated cardiac enzyme levels. The pattern of cerebral infarcts and laboratory results suggest a thromboembolic infarction associated with hypereosinophilia.
Cerebral Hemorrhage/diagnosis/*etiology ; Cerebral Infarction/diagnosis/*etiology ; Diagnosis, Differential ; Eosinophilia/*complications/drug therapy ; Humans ; Magnetic Resonance Imaging ; Male ; Middle Aged

OBJECTIVEAntiphospholipid antibody (APL) is a particularly important laboratory diagnostic criterion for antiphospholipid syndrome (APS). The significances of positive APL in childhood are seldom reported nor fully understood. The purpose of this study was to analyze 13 cases with positive APL seen in our hospital and to study the relationship between the positive rates of APL and various clinical diseases especially systemic lupus erythematosus (SLE) in order to improve the clinical diagnoses and treatment level of APS in children.

METHODSThe clinical data collected from 2000 to 2002 of 13 hospitalized children with positive APL were retrospectively evaluated. Enzyme linked immunosorbent assay (ELISA) and indirect immunofluorescence technique were used respectively to detect APL and antineutrophil cytoplasmic autoantibodies (ANCA) of sera from those children. Other various indexes were also detected according to different characteristics of different diseases.

RESULTSEight cases had SLE; 2 had acute post-streptococcal infections. The other 3 cases did not show any evidences of primary diseases; they probably had primary APS. SLE was the most common primary diseases to cause development of APL and the cases with SLE showed more severe cutaneous vasculitis than SLE patients who were negative for APL. There was no significant relationship between the positive rates of APL and that of ANCA. Eight APL positive cases complicated with thrombocytopenia and bleeding were treated with high dosage of immunoglobulin [400 mg/(kg.d), for 3 - 5 d] intravenously; the clinical conditions of these cases were ameliorated soon. While the 5 cases who had thrombotic vasculitis and thromboembolism were treated with anticoagulant and antithrombotic therapy with low molecular weight heparin [50 - 100 U/(kg.d)], which led to good clinical effects.

CONCLUSIONSThe clinical manifestations of children positive for APL were somehow different from those of adults. Positive APL itself may be nonspecific, it can occur from different causes of diseases. APL detection may be useful to suggest anticoagulant and/or antithrombosis therapy. Treatments for APS should be variable according to different causes and severity of diseases, in the cases of thrombocytopenia and bleeding, high dose intravenous immunoglobulin should be given as soon as possible, while in the cases of thrombotic vasculitis and thromboembolism, anticoagulant and antithrombotic therapy should be given soon.

Adult ; Antibodies, Antineutrophil Cytoplasmic ; blood ; Antibodies, Antiphospholipid ; blood ; immunology ; Anticoagulants ; therapeutic use ; Antiphospholipid Syndrome ; blood ; complications ; diagnosis ; therapy ; Child ; Fibrinolytic Agents ; therapeutic use ; Hemorrhage ; etiology ; therapy ; Humans ; Immunoglobulins, Intravenous ; therapeutic use ; Lupus Erythematosus, Systemic ; immunology ; Streptococcal Infections ; immunology ; Thrombocytopenia ; etiology ; therapy ; Thromboembolism ; drug therapy ; etiology ; Thrombosis ; drug therapy ; etiology ; Vasculitis ; drug therapy ; etiology

Paragonimiasis is caused by ingesting crustaceans, which are the intermediate hosts of Paragonimus. The involvement of the brain was a common presentation in Korea decades ago, but it becomes much less frequent in domestic medical practices. We observed a rare case of cerebral paragonimiasis manifesting with intracerebral hemorrhage. A 10-yr-old girl presented with sudden-onset dysarthria, right facial palsy and clumsiness of the right hand. Brain imaging showed acute intracerebral hemorrhage in the left frontal area. An occult vascular malformation or small arteriovenous malformation compressed by the hematoma was initially suspected. The lesion progressed for over 2 months until a delayed surgery was undertaken. Pathologic examination was consistent with cerebral paragonimiasis. After chemotherapy with praziquantel, the patient was monitored without neurological deficits or seizure attacks for 6 months. This case alerts practicing clinicians to the domestic transmission of a forgotten parasitic disease due to environmental changes.
Animals ; Anthelmintics/therapeutic use ; Brain/parasitology/pathology ; Cerebral Hemorrhage/*etiology ; Child ; Dysarthria/etiology ; Facial Paralysis/etiology ; Female ; Humans ; Magnetic Resonance Imaging ; Paragonimiasis/*diagnosis/drug therapy/parasitology ; Paragonimus/isolation & purification ; Praziquantel/therapeutic use ; Tomography, X-Ray Computed ; Vascular Malformations/etiology

Paragonimiasis is caused by ingesting crustaceans, which are the intermediate hosts of Paragonimus. The involvement of the brain was a common presentation in Korea decades ago, but it becomes much less frequent in domestic medical practices. We observed a rare case of cerebral paragonimiasis manifesting with intracerebral hemorrhage. A 10-yr-old girl presented with sudden-onset dysarthria, right facial palsy and clumsiness of the right hand. Brain imaging showed acute intracerebral hemorrhage in the left frontal area. An occult vascular malformation or small arteriovenous malformation compressed by the hematoma was initially suspected. The lesion progressed for over 2 months until a delayed surgery was undertaken. Pathologic examination was consistent with cerebral paragonimiasis. After chemotherapy with praziquantel, the patient was monitored without neurological deficits or seizure attacks for 6 months. This case alerts practicing clinicians to the domestic transmission of a forgotten parasitic disease due to environmental changes.
Animals ; Anthelmintics/therapeutic use ; Brain/parasitology/pathology ; Cerebral Hemorrhage/*etiology ; Child ; Dysarthria/etiology ; Facial Paralysis/etiology ; Female ; Humans ; Magnetic Resonance Imaging ; Paragonimiasis/*diagnosis/drug therapy/parasitology ; Paragonimus/isolation & purification ; Praziquantel/therapeutic use ; Tomography, X-Ray Computed ; Vascular Malformations/etiology

Adult-onset Still's disease (AOSD) is an inflammatory disease that presents with a variety of clinical symptoms. Pulmonary involvement is well-known in AOSD and is seen in up to 53% of AOSD cases, with the most common pulmonary diseases being pleural effusion and transient pulmonary infiltrates. We present the first case of chronic AOSD complicated with diffuse alveolar hemorrhage during the acute flare of the disease.
Adult ; Ankylosis/diagnosis ; Anti-Inflammatory Agents/administration & dosage ; Diagnosis, Differential ; Female ; Fever of Unknown Origin/diagnosis ; Hemorrhage/*diagnosis/drug therapy/etiology ; Humans ; Lung Diseases/*diagnosis/drug therapy/etiology ; Methylprednisolone/administration & dosage ; *Pulmonary Alveoli ; Still's Disease, Adult-Onset/complications/*diagnosis/drug therapy/radiography ; Tomography, X-Ray Computed

Hemangiomas are the most common benign tumors of the liver. They are generally asymptomatic, but giant hemangiomas can lead to abdominal discomfort, bleeding, or obstructive symptoms. Kasabach-Merritt syndrome is a rare but life-threatening complication of hemangioma, characterized by consumptive coagulopathy with large vascular tumors. More than 80% of Kasabach-Merritt syndrome cases occur within the first year of life. However, there are few reports of Kasabach-Merritt syndrome with giant hepatic hemangioma in adults and, as far as we know, no reports of Kasabach-Merritt syndrome with hepatic hemangioma treated with first line medical treatment only. The most important treatment for this syndrome is removal of the large vascular tumor. However, surgical treatment entails risk of bleeding, and the patient's condition can mitigate against surgery. We herein present a case of unresectable giant hepatic hemangioma with disseminated intravascular coagulopathy. The patient was a 60-year-old woman who complained of hematochezia, ecchymosis, and abdominal distension. She refused all surgical management and was therefore treated with systemic glucocorticoids and beta-blockers. After two weeks of steroid therapy, she responded partially to the treatment. Her laboratory findings and hematochezia improved. She was discharged on hospital day 33 and observed without signs of bleeding for three months.
Abdomen/diagnostic imaging ; Ecchymosis/etiology ; Female ; Gastrointestinal Hemorrhage/etiology ; Hemangioma/complications/*diagnosis ; Humans ; Kasabach-Merritt Syndrome/complications/*diagnosis/drug therapy ; Middle Aged ; Prednisone/therapeutic use ; Propranolol/therapeutic use ; Tomography, X-Ray Computed

PURPOSE: To evaluate the efficacy of intravitreal triamcinolone acetonide (IVT) for the management of postvitrectomy diabetic vitreous hemorrhage. METHODS: The authors conducted a retrospective study of patients with postvitrectomy diabetic vitreous hemorrhage who were administered 4 mg (0.1 cc) of triamcinolone acetonide ophthalmic suspension. Ocular history, adverse events, BCVA, intraocular pressure, external eye examination, slit-lamp biomicroscopy, fundus examination, B-scan ultrasonography, and fundus photography were assessed on day 1, weeks 1, 2, and 4 and months 2 and 3. RESULTS: There were 19 eyes of 18 consecutive patients with mean follow-up after IVT injection of 28 weeks. Of the 19 eyes, 17 eyes (89%) experienced clearing of vitreous hemorrhage within 1 to 5 weeks (mean, 1.7 weeks) with visible triamcinolone precipitates along with blood clot in the inferior aspect of fundus. Of these 17 eyes, 12 eyes (63%) maintained vitreous hemorrhage-free condition at last follow-up with a mean visual acuity of 20/63 (range, 20/320 20/25), whereas 5 (29%) developed recurrent vitreous hemorrhage after clearing of vitreous hemorrhage. Vitreous hemorrhage was not cleared in 2 eyes, which required surgical procedures. CONCLUSIONS: IVT injection may be beneficial for clearing recurrent postvitrectomy Diabetic Vitreous Hemorrhage.
Adult ; Aged ; Diabetic Retinopathy/*complications/diagnosis ; Female ; Follow-Up Studies ; Glucocorticoids/*administration & dosage ; Humans ; Injections ; Male ; Microscopy, Acoustic ; Middle Aged ; Postoperative Hemorrhage/diagnosis/*drug therapy/etiology ; Retrospective Studies ; Time Factors ; Treatment Outcome ; Triamcinolone Acetonide/*administration & dosage ; Visual Acuity ; Vitrectomy/*adverse effects ; Vitreous Body ; Vitreous Hemorrhage/diagnosis/*drug therapy/etiology

Systemic sclerosis (SSc) is a chronic systemic disease that affects the skin, lungs, heart, gastrointestinal tract, kidneys, and musculoskeletal system. Although up to 90% of patients with scleroderma have been estimated to have gastrointestinal involvement, liver disease has been reported only rarely. A 51-year-old woman was hospitalized due to esophageal variceal bleeding. Her serum was positive for anti-nuclear antibody and anti-centromere antibody. Sclerodactyly was noted on both hands, and she had recently developed Raynaud's syndrome. Punch biopsy of the hand showed hyperkeratosis, regular acanthosis, and increased basal pigmentation in the epidermis, and thick pale collagenous bundles in the dermis. Liver biopsy showed chronic active hepatitis with bridging fibrosis. Consequently, she was diagnosed with liver cirrhosis due to autoimmune hepatitis (AIH) combined with SSc. AIH had subsided after administration of prednisolone at 40 mg per day. She received 5-10 mg/day of prednisolone as an outpatient, and her condition has remained stable. Patients with either AIH or SSc should be monitored for further development of concurrent autoimmune diseases. The early diagnosis of AIH combined with SSc will be helpful in achieving optimal management.
Anti-Inflammatory Agents/therapeutic use ; Antibodies, Antinuclear/blood ; Esophageal and Gastric Varices ; Female ; Gastrointestinal Hemorrhage ; Hepatitis, Autoimmune/complications/*diagnosis/drug therapy ; Humans ; Liver Cirrhosis/*diagnosis/etiology/pathology ; Middle Aged ; Prednisolone/therapeutic use ; Raynaud Disease/diagnosis ; Scleroderma, Systemic/complications/*diagnosis ; Skin/pathology

CMV infection may occur anywhere in the gastrointestinal tract. Among the small intestine, ileum is the most common site of CMV disease and infection of jejunum is a rare one in patients with CMV gastroenteritis. Although rare, the reason why the recognition of this diagnosis is important is that it cause the lethal hemorrhage and perforation of gastrointestinal tract when its diagnosis and treatment was delayed. Rapid diagnosis are able to using the immunohistochemical stain in shell vial culture of infected specimen or peripheral neutrophils preparation in viremic patients within 8 to 36 hours. The treatment of choice is antiviral agent or surgical resection. We experienced a case of CMV disease of jejunum in patient with non-Hodgkin's lymphoma who showed severe ulceration in jejunum and massive intestinal hemorrhage, and he survived after successful treatment with segmental resection of jejunum and intravenous ganciclovir.
Adult ; Antiviral Agents/therapeutic use ; Cytomegalovirus Infections/drug therapy ; Cytomegalovirus Infections/diagnosis ; Cytomegalovirus Infections/complications* ; Disease-Free Survival ; Enteritis/virology ; Enteritis/surgery ; Enteritis/complications ; Ganciclovir/therapeutic use ; Gastrointestinal Hemorrhage/therapy ; Gastrointestinal Hemorrhage/etiology* ; Gastrointestinal Hemorrhage/diagnosis ; Human ; Jejunal Diseases/virology ; Jejunal Diseases/surgery ; Jejunal Diseases/complications* ; Lymphoma, Non-Hodgkin/drug therapy ; Lymphoma, Non-Hodgkin/diagnosis ; Lymphoma, Non-Hodgkin/complications* ; Male ; Opportunistic Infections/drug therapy ; Opportunistic Infections/diagnosis ; Opportunistic Infections/complications* ; Substances: Ganciclovir ; Substances: Antiviral Agents