Churg-Strauss syndrome (CSS) is a rare systemic vasculitis that affect small and medium-sized blood vessels and is accompanied by asthma, eosinophilia, and peripheral neuropathy. This report describes a case of a 52-year-old man who had a history of sinusitis, asthma, and thymus cancer and who had complained of bilateral lower extremity paresthesia and weakness for a month. Peripheral neuropathy was detected by electrodiagnostic studies. Resection of a mediastinal mass, which was diagnosed as thymic neuroendocrine carcinoma, was performed five months before his visit. After thymectomy, peripheral blood tests revealed a gradual increase in eosinophils. Two months after surgery, he was admitted to the hospital for dyspnea, and nodules of focal consolidation were found in his chest X-ray. One month later, pyoderma occurred in the right shin, and the skin biopsy showed extravascular eosinophilic infiltration. He was diagnosed with CSS after thymectomy, and we report a very rare case of CSS presented with thymic neuroendocrine carcinoma.
Asthma ; Biopsy ; Blood Vessels ; Carcinoma, Neuroendocrine* ; Churg-Strauss Syndrome* ; Diagnosis* ; Dyspnea ; Eosinophilia ; Eosinophils ; Hematologic Tests ; Humans ; Lower Extremity ; Middle Aged ; Paresthesia ; Peripheral Nervous System Diseases ; Polyneuropathies ; Pyoderma ; Sinusitis ; Skin ; Systemic Vasculitis ; Thorax ; Thymectomy ; Thymus Neoplasms

Kimura's disease is a rare, benign, slow-growing chronic inflammatory swelling with a predilection for the head and neck region and is almost always with peripheral blood eosinophilia and elevated serum IgE levels. It is endemic in Asian males and rare in Western people. Surgical excision of the lesion is the first line therapy. Drug and radiation therapy have to be considered for the refractory lesions.
Angiolymphoid Hyperplasia with Eosinophilia ; diagnosis ; Asian Continental Ancestry Group ; Eosinophilia ; pathology ; Humans ; Immunoglobulin E ; blood ; Inflammation ; pathology ; Male ; Neck ; pathology

PURPOSE: The authors of the present study describe a rare case of angiolymphoid hyperplasia with eosinophilia (ALHE) of the eyelid. CASE SUMMARY: A 63-year-old male who was diagnosed with ALHE based on biopsy of an inguinal mass presented with an eyelid mass of 1 month duration. A light brown, solitary, 1.0 x 0.5 cm-sized mass involved the right upper eyelid. There was no lymphadenopathy, but eosinophilia was present. An excisional biopsy of the mass was performed for diagnosis and management. Macroscopic examination of the excised mass revealed a well-defined, smooth, firm, yellowish-red colored lesion measuring 1.0 x 0.6 x 0.5 cm. Histopathology showed the proliferation of small blood vessels, many of which were lined by enlarged endothelial cells with uniform ovoid nuclei and intracytoplasmic vacuoles. The distinctive endothelial cells were described as having a cobblestone appearance. In addition, a perivascular and interstitial infiltrate composed primarily of lymphocytes and eosinophils was present. ALHE was finally confirmed with clinical and microscopic examination. CONCLUSIONS: The authors of the present study report a rare case of ALHE of the eyelid and suggest that a differential diagnosis should be considered.
Angiolymphoid Hyperplasia with Eosinophilia ; Biopsy ; Blood Vessels ; Diagnosis, Differential ; Endothelial Cells ; Eosinophilia ; Eosinophils ; Eyelids ; Humans ; Light ; Lymphatic Diseases ; Lymphocytes ; Male ; Middle Aged ; Vacuoles

OBJECTIVE: To improve the diagnosis and treatment of Kimura's disease (KD) by investigating its clinical characteristics, pathological features and complications. METHOD: The clinical data of 33 cases of KD were analyzed retrospectively. RESULT: Of 33 cases, 22 showed the mass on head and neck, while in the other cases, the mass distributed in the region of groin, axillary fossa, hilum of lung and mesentery. Regional lymph nodes were involved in 21 cases and major salivary glands were invaded in 8 cases. Twenty-three cases had typical peripheral eosinophilia, although only in 2 patients the quantity of serum total IgE increased markedly. Urine abnormalities happened to 7 cases, such as massive proteinuria (3 cases) and hematuria (2 cases). Among 6 cases which underwent bone marrow aspiration, 2 showed eosinophilia. Two cases were complicated with nephritic syndrome. Six cases were combined with local inflammation on head and neck and 2 cases were combined with malignant tumor. CONCLUSION Mass on the head and neck is the typical clinical manifestation in KD, with regional lymph nodes and major salivary glands involved most. Serum total IgE and histopathologic examination should always be done to confirm KD, especially in the cases with unknown eosinophilia increasing.
Adolescent ; Adult ; Aged ; Angiolymphoid Hyperplasia with Eosinophilia ; diagnosis ; pathology ; therapy ; Child ; Female ; Head ; pathology ; Humans ; Immunoglobulin E ; blood ; Lymph Nodes ; pathology ; Male ; Middle Aged ; Neck ; pathology ; Retrospective Studies ; Salivary Glands ; pathology ; Young Adult

Seroprevalence of the IgG antibodies for Clonorchis sinensis, Paragonimus westermani, Taenia solium metacestode (cysticercus), and Spirometra erinacei plerocercoid (sparganum) was measured using enzyme-linked immunosorbent assay (ELISA) in sera of patients in Korea from 1993 to 2006. A total of 74,448 specimens referred nationwide from 121 hospitals revealed an IgG positive rate of 7.6% for the 4 parasites. The IgG positive rate (18.7%) for the 4 parasites in 1993 decreased gradually to 6.6% in 2006. Individual positive rate decreased from 5.2% (1993) to 1.6% (2006) for C. sinensis, from 2.8% (1993) to 1.1% (2006) for P. westermani, from 8.3% (1993) to 2.2% (2006) for cysticercus, and from 2.6% (1993) to 1.6% (2006) for sparganum. The positive rate was highest (21.2%) in the group of patients who ranged in age from 50-59 yr old, and in the group that was referred from the Seoul area (55.9%). In conclusion, our results suggest that tissue invading parasitic infections should always be included in differential diagnosis for patients with eosinophilia associated lesions of the central nervous system, liver, and lungs in Korea.
Adolescent ; Adult ; Age Factors ; Aged ; Aged, 80 and over ; Animals ; Antibodies, Helminth/*blood ; Child ; Child, Preschool ; Clonorchiasis/diagnosis/*epidemiology ; Clonorchis sinensis/immunology/isolation & purification ; Cysticercosis/diagnosis/*epidemiology ; Cysticercus/immunology/isolation & purification ; Diagnosis, Differential ; Enzyme-Linked Immunosorbent Assay ; Eosinophilia/immunology ; Female ; Humans ; Immunoglobulin G/blood ; Infant ; Male ; Middle Aged ; Paragonimiasis/diagnosis/*epidemiology ; Paragonimus westermani/immunology/isolation & purification ; Republic of Korea/epidemiology ; Seroepidemiologic Studies ; Sparganosis/diagnosis/*epidemiology ; Sparganum/immunology/isolation & purification

Toxocariasis is one of the causes of pulmonary eosinophilic infiltrate that is increasing in Korea. This study was designed to identify the prevalence of toxocara seropositivity in patients with unexplained pulmonary patchy infiltrate and to evaluate associated factors. We evaluated 102 patients with unexplained pulmonary patchy infiltrate on chest computed tomography (CT) scan. As a control set, 116 subjects with normal chest CT were also evaluated. History of allergic disease, drug use, parasitic disease and raw cow liver intake were taken. Blood eosinophil count and total IgE level were measured. Specific serum IgG antibody to Toxocara canis larval antigen and specific IgG antibodies to 4 other parasites were measured by enzymelinked immunosorbent assay (ELISA). In the infiltrate group, 66.7% subjects were toxocara seropositive whereas 22.4% of the control group were seropositive (p< 0.001). In the infiltrate group, patients with a history of eating raw cow liver (odds ratio [OR], 7.8) and patients with eosinophilia (OR, 5.2) had a higher incidence of toxocara seropositivity. Thirty-five percent of toxocara seropositive patients with infiltrate exhibited migrating infiltrate and 48% had decreased infiltrate on the follow- up CT. We recommend that toxocara ELISA should be performed in patients with unexplained pulmonary patchy infiltrate, and that the eating of raw cow liver should be actively discouraged.
Adult ; Aged ; Animals ; Case-Control Studies ; Female ; Humans ; Immunoglobulin E/blood ; Korea ; Leukocyte Count ; Liver/parasitology ; Male ; Middle Aged ; Pulmonary Eosinophilia/diagnosis/*etiology/immunology ; Seroepidemiologic Studies ; Tomography, X-Ray Computed ; Toxocara/immunology ; Toxocariasis/*complications/diagnosis/epidemiology

Eosinophilic myocarditis usually results from myocardial damage as a result of drugs or parasites, and is generally associated with increased peripheral eosinophil count. This form of myocarditis is difficult to diagnose clinically. A 25 year-old previously healthy woman was transferred from a local clinic because of hypotension and dyspnea with sudden cardiogenic shock after a three day history of gastrointestinal illness. Echocardiography revealed concentric left ventricular wall thickening with moderate pericardial effusion. Biopsy of endomyocardial tissue from the right ventricle showed diffuse infiltration of inflammatory cells, mostly eosinophils, even though the patient had a peripheral eosinophil count that was normal at the time of biopsy. The patient was treated with corticosteroids for the symptoms of pericarditis, and she recovered without cardiac sequelae, clinically and echocardiographically. We here report a case of acute eosinophilic myopericarditis, with cardiogenic shock, diagnosed by endomyocardial biopsy with normal peripheral eosinophil count at the time of biopsy, and complete recovery without sequelae.
Shock, Cardiogenic/blood/*etiology ; Pericarditis/blood/*diagnosis ; Myocarditis/blood/*diagnosis ; Leukocyte Count ; Humans ; Female ; *Eosinophils ; Eosinophilia/blood/*diagnosis ; Adult ; Acute Disease

Allopurinol, a commonly prescribed medicine for the management of gout and hyperuricemia, may induce life-threatening hypersensitivity characterized by fever, eosinophilia, hepatitis, renal failure, and skin eruptions such as Stevens-Johnson syndrome or toxic epidermal necrolysis. Stevens-Johnson syndrome may rarely affect the gastrointestinal tract, associated with a poor prognosis. We have experienced a patient having allopurinol hypersensitivity syndrome (AHS) with esophageal ulcer bleeding. A 64-year-old man was admitted with ten-day history of widespread rash and fever. Six weeks before admission, he had symptoms of gouty arthritis, and he was treated with allopurinol and colchicine for 10 days. Complete blood count showed leukocytosis with eosinophilia and blood biochemistry showed impaired renal and hepatic function. The diagnosis of an AHS with Stevens-Johnson syndrome was made from the history and the typical clinical feature. Despite adequate hydration, steroid and immunoglobulin therapy, severe esophageal ulcer bleeding, sepsis and disseminated intravascular coagulation had been developed and the patient died 33 days after admission. Until now, there is no specific treatment for the AHS. The only means of minimizing the incidence of AHS is to limit the allopurinol therapy to accepted indications and to adjust the dosage for the patient's renal function.
Allopurinol* ; Arthritis, Gouty ; Biochemistry ; Blood Cell Count ; Colchicine ; Diagnosis ; Disseminated Intravascular Coagulation ; Eosinophilia ; Exanthema ; Fever ; Gastrointestinal Tract ; Gout ; Hemorrhage* ; Hepatitis ; Humans ; Hypersensitivity* ; Hyperuricemia ; Immunization, Passive ; Incidence ; Leukocytosis ; Middle Aged ; Prognosis ; Renal Insufficiency ; Sepsis ; Skin ; Stevens-Johnson Syndrome ; Ulcer*

We report here a case with hypereosinophilia and peripheral artery occlusion. A 32-yr-old Korean woman presented to us with lower extremity swelling and pain. Angiography revealed that multiple lower extremity arteries were occlusive. The biopsy specimen showed perivascular and periadnexal dense eosinophilic infiltration in dermis and subcutaneous adipose tissue. Laboratory investigations revealed a persistent hypereosinophilia. She was prescribed prednisolone 60 mg daily. Her skin lesion and pain were improved and the eosinophil count was dramatically decreased. After discharge, eosinophil count gradually increased again. Cyanosis and pain of her fingers recurred. She had been treated with cyclophosphamide pulse therapy. Her eosinophilia was decreased, but the cyanosis and tingling sense were progressive. The extremity arterial stenoses were slightly progressed. Skin biopsy showed perivascular eosinophilic infiltration in the dermis and CD40 ligand (CD40L) positive eosinophilic infiltration. The serum TNF-alpha was markedly increased. These results suggest that CD40L (a member of TNF-alpha superfamily) could play a role in the inflammatory processes when eosinophil infiltration and activation are observed. We prescribed prednisolone, cyclophosphamide, clopidogrel, cilostazol, beraprost and nifedipine, and she was discharged.
Adult ; Arterial Occlusive Diseases/*diagnosis/etiology ; CD40 Ligand/analysis ; Cyanosis/etiology ; Diagnosis, Differential ; Eosinophilia/*diagnosis/etiology ; Female ; Gangrene/etiology ; Humans ; Hypereosinophilic Syndrome/blood/complications/*diagnosis ; Immunohistochemistry ; Peripheral Vascular Diseases/*diagnosis/etiology ; Skin/chemistry/pathology ; Tumor Necrosis Factor-alpha/metabolism ; Vasculitis/*diagnosis/etiology

OBJECTIVETo explore the role of eotaxin in the pathogenesis of bronchial asthma and the clinical value in the diagnosis of asthma.

METHODSSerum eotaxin were measured by ELISA in 38 patients with asthma, 28 patients with non-asthma allergy, and 30 healthy controls.

RESULTSThe levels of serum eotaxin in the asthma group were higher than those in the non-asthma allergic and control group (P<0.01). Furthermore, eotaxin levels in patients with acute asthma were significantly higher than those in patients with stable asthma (P<0.001). It was also found that the eotaxin levels of the acute asthma group were positively correlated to the amounts of eosinophils in peripheral blood (r=0.4196, P<0.001), and inversely correlated to the forced expiratory volume in one second (FEV1) (r=-0.3746, P<0.001).

CONCLUSIONIt suggests that eotaxin may play a crucial pathogenic role in the asthmatic process possibly by activating the allergic inflammatory cells and controlling the recruitment of eosinophils from blood to bronchial epithelium of the airway. The concentration of eotaxin is significantly associated with the attack of acute asthma and its severity. Eotaxin may be a potential therapeutic target in patients with asthma.

Adult ; Asthma ; diagnosis ; physiopathology ; Cell Count ; Chemokine CCL11 ; Chemokines, CC ; blood ; physiology ; Eosinophilia ; pathology ; Female ; Forced Expiratory Volume ; Humans ; Male ; Middle Aged