[Pulmonary Sequestration Associated with Increased Serum Tumor Markers;Report of a Case].

A 51-year-old woman visited our hospital with chief complaints of cough and fever. A chest X-ray detected an abnormal shadow in the right lung field. A chest computed tomography scan showed solid consolidation at S10 of the right lung. A blood test revealed elevated levels of the tumor markers, CEA(12.1 ng/ml), SLX (134 U/ml) and CA19-9 (76.2 U/ml). Fluorodeoxyglucose-positron emission tomography (FDG-PET) showed abnormally increased 18F-FDG uptake with an SUV max of 11.29. Lung cancer was strongly suspected, and the surgery was performed. Abnormal blood vessels were found within the pulmonary ligament. Intraoperative rapid pathology indicated no malignancy, and the final diagnosis was pulmonary sequestration.

Adenovirus pneumonia in an immunocompetent adult.

Adenovirus pneumonias are reported relatively commonly in pediatric or immunocompromised patients, but the clinical presentation of adenovirus pneumonia in immunocompetent hosts is not well known. We treated an immunocompetent 42-year-old man with mild adenovirus pneumonia following pharyngitis and conjunctivitis. Diagnosis was established on the basis of chest radiologic findings, detection of adenovirus type 7 DNA by PCR assay in material obtained from bronchoalveolar lavage (BAL), and a greater than fourfold rise in adenovirus-specific antibody titers during the course of illness. The patient's self-limiting symptoms improved within 2 weeks, and chest radiologic findings improved within 4 weeks. PCR assay of material obtained by BAL was useful for the rapid diagnosis of adenovirus pneumonia.

[A case of pulmonary dirofilariasis with a single nodule on an apical lesion of the right upper lobe].

A 75-year-old man with chronic gastritis and gastro-esophageal regurgitation syndrome was admitted to our hospital complaining of continuous productive cough, and an abnormal shadow was noted on a chest X-ray film. Chest computed tomography revealed a nodule 2.8 cm in diameter with a regular margin on the right apex. Fiberoptic bronchoscopy was performed, but did not yield a definitive diagnosis. We suspected primary lung cancer, and therefore, lung partial resection was performed under thoracoscopic surgery. The intraoperative pathological findings revealed inflammatory granuloma with necrosis, and pulmonary dirofilariasis was finally diagnosed. Pulmonary dirofilariasis is an important differential diagnosis in elderly patients with a chest abnormal nodular shadow. Lesions have been reportedly observed in the peripheral portion of the right lower lobe in many previous reports. However, in this case, a nodular lesion was noted in the right apex.

[A case of Ehlers-Danlos syndrome suspected from pulmonary hematoma due to disruption of the lung].

A 20-year-old man was referred to our hospital due to hemoptysis. Chest CT showed a ground-glass opacity, suggesting pulmonary bleeding; however, a diagnosis was not obtained. At a follow-up examination after 2 months, Chest CT showed improvement of the ground-glass opacity, however a cavitary nodule had newly appeared. Four months later, another new nodule was found on chest X-ray film. Video-assisted thoracoscopic lung biopsy was performed for pathological diagnosis; disruption of the pleural, lung and blood vessels, and pulmonary hematoma were found. We thought of the fragility of the pleuro-pulmonary connective tissue and also thought of the probability of Ehlers-Danlos syndrome (EDS). A biochemical analysis of cultured dermal fibroblasts and molecular biological examination revealed decreased production of type III collagen in fibroblasts and COL3A1 mutation. We diagnosed this case as vascular EDS. EDS is one of the differential diagnoses in patients presenting hemoptysis and pulmonary hematoma due to disruption of the lung.

Ehlers-Danlos syndrome with recurrent spontaneous pneumothoraces and cavitary lesion on chest X-ray as the initial complications.

A 17-year-old-man developed left-sided pneumothorax in 1995. Chest computed tomography (CT) showed a thick-walled cavity in the left lower lobe. Video-assisted thoracic surgery was performed, and pathologic findings of the resected lung showed a cavity, organizing hematoma, and a fibrous nodule. Fragility of connective tissue was suspected, and biochemical and molecular analysis showed reduction of type III collagen production and point mutation of the COL3A1 gene. The patient was diagnosed as having vascular-type Ehlers-Danlos syndrome (EDS). From 2002, the patient developed hemoptysis and bloody sputum once a year. Chest CT detected several nodules and cavities, which were regarded as hematomas with or without excretion. Several vascular changes including aneurysmal formations have been found since 2002, and an aneurysm of the left ulnar artery was resected. The patient continues to be followed regularly on an outpatient basis. We report a rare case of vascular-type EDS who developed pulmonary symptoms as an initial complication.

[Case of pneumonia caused by beta-lactamase-producing and amoxicillin/clavulanate resistant strains of H. influenzae].

An 80-year-old woman presenting with fever and cough was given a diagnosis of community-acquired pneumonia. She was hospitalized and treated with ampicillin/sulbactam (ABPC/SBT) and clarithromycin (CAM). Gram stain images and sputum culture results led us to believe that the causative agent was Haemophilus influenzae. Drug sensitivity testing indicated that the H. influenzae was a beta-lactamase-positive, ABPC-resistant (BLPAR) strain. Treatment with ABPC/SBT was not clinically effective. We considered the possibility of beta-lactamase-positive amoxicillin/clavulanate-resistant (BLPACR) strains. Further testing revealed that the MIC of ABPC was 128 microg/ml, that of SBT/ABPC was 8 microg/ml, and that of AMPC/CVA was 4 microg/ml. Furthermore, genetic analysis indicated the H. influenzae to be a BLPACR-I strain. The poor clinical course eventually led to a diagnosis of BLPACR. When beta-lactamase-producing H. influenzae is cultured, the possibility of a BLPACR strain resistant to ABPC/SBT and AMPC/CVA must be considered.

Development of sarcoidosis during etanercept therapy.

This report describes a 65-year-old woman who developed granulomatous lesions consistent with sarcoidosis during etanercept therapy for rheumatoid arthritis. Hilar and mediastinal lymphadenopathy and multiple nodules in both lung fields developed 21 months after administration of etanercept. Noncaseating epithelioid cell granulomas consistent with sarcoidosis were detected in a lung biopsy specimen and in the parietal pleura obtained via thoracotomy. Diseases showing similar histologic changes were excluded, and a diagnosis of sarcoidosis was made. Etanercept was discontinued, which resulted in symptomatic relief, improvement of oxygenation and radiologic findings. There is substantial evidence of tumor necrosis factor-alpha involvement in the induction and maintenance of granuloma formation; however, we should keep in mind that granulomatous disease, such as sarcoidosis, can develop during treatment with a tumor necrosis factor-alpha blocking agent, such as etanercept.

[Long-term CT findings of X-linked agammaglobulinemia with bronchiectasis diagnosed in an adult].

We report a case of X-linked agammaglobulinemia who presented with bronchiectasis. The patient had suffered pneumonia about every five years since childhood until he presented to our hospital at age 34 years old. CT showed bronchiectasis predominantly in the right middle lobe, lingula, and lower lobes. Administration of antibiotics resulted in symptomatic relief. Episodes of recurrent pulmonary infection and bronchiectasis indicated congenital immunodeficiency disorder. Investigation of lymphocyte subsets and serum immunoglobulin values showed remarkable reduction of B cells, IgG 772 mg/dl, IgA 216 mg/dl, and IgM 29 mg/dl. Flow cytometric assessment combined with genetic analysis was performed, and the results showed decreased expression of monocyte Bruton's tyrosine kinase (BTK) and missense mutation of Btk gene. We diagnosed X-linked agammaglobulinemia. IgG remained above 600 mg/dl in this case, we have not administered immunoglobulin after discharge. He suffered from pneumonia in 2004 and 2006 and bronchiectasis has progressed. In this report, we present a case including CT findings over a period of 8 years.

Desquamative interstitial pneumonia with a remarkable increase in the number of BAL eosinophils.

A 57-year old man with desquamative interstitial pneumonia (DIP) showed a marked increase in eosinophils in the bronchoalveolar lavage (BAL) fluid. The patient was referred to our hospital for abnormal shadows on his chest X-ray with no symptoms in May 2007. Computed tomography (CT) showed patchy, peripheral predominate ground-glass opacity. The BAL fluid revealed an increase of the total number of cells, including markedly elevated levels of eosinophils (62.1%), in contrast with only a slight increase of peripheral blood eosinophils, or minimal eosinophils in the alveolar spaces and interstitium of the thoracoscopic lung biopsy specimen. Since the specimens showed findings compatible with a DIP pattern, we diagnosed the patient with DIP. Although it is a rare entity, we should therefore consider DIP in the differential diagnosis when we encounter patients with a marked increase in the number of BAL eosinophils.

Pulmonary Schizophyllum commune infection developing mucoid impaction of the bronchi.

A 54-year-old woman was admitted for cough, sputum, and an abnormal chest X-ray shadow. Bronchoscopy showed mucoid impaction of the bronchi (MIB). Histopathologic evidence of mucous plugs was consistent with one component of allergic bronchopulmonary mycosis. Schizophyllum commune (S. commune) was identified. Two attempts at removal of the mucous plugs were unsuccessful. Itraconazole was then administered, and the mucous plugs disappeared. There are few reports of MIB due to S. commune; we herein report a case of MIB due to S. commune infection.