Sj?gren's syndrome with solitary nodular pulmonary amyloidosis]

We describe a case of limited pulmonary amyloidosis with Sj?gren syndrome. A 58-yr-old woman was referred to our hospital because of an abnormal chest radiograph (solitary small nodule) that was examined to investigate the cause of a persistent cough. A chest CT revealed a solitary small nodule in the left lower lung field. The specimens obtained by thoracoscopic surgery showed AL (kappa) amyloid deposits with lymphoplasmacytic infiltrate. Immunofixation of the serum and concentrated urine failed to demonstrate monoclonal immunoglobulins, and no amyloid deposits in the stomach were detected. She was subsequently diagnosed as having primary Sj?gren's syndrome. Nodular pulmonary amyloidosis with Sj?gren syndrome is very rare condition, and most cases present multiple nodules. As far as is known, this is the first report of a solitary nodule in pulmonary amyloidosis with Sj?gren syndrome.     

FDG-PET findings of nodular pulmonary amyloidosis with a long-term observation]

A 60-year-old woman was referred to our hospital because of an abnormal chest radiograph in May, 2000. She was found to have rheumatoid arthritis in March, 1998, and pharmacologic therapy with anti-rheumatic drug was started. The chest CT scan revealed bilateral multiple lung nodular lesions of various sizes up to 30 mm. F-18 fluorodeoxyglucose positron emission tomography (FDG-PET) demonstrated a lesion with intense FDG activity in the right lung with a standardized uptake value (SUV) of 10.1. Fiberoptic bronchoscopy revealed no endobronchial lesions. Video-assisted thoracoscopic surgery was done to ascertain the pathological diagnosis. Histological examination showed that the pulmonary nodules were composed of amyloid A (AA) protein. Secondary Sj?gren syndrome was subsequently diagnosed. A diagnosis of localized nodular pulmonary amyloidosis with AA type amyloid protein was made, and therapy with anti-rheumatic drugs was continued. After six years of therapy, the size of pulmonary amyloidoma was reduced, and the accumulation of FDG returned to normal. We reported this interesting case in which FDG-PET apparently reflected the disease activity of pulmonary amyloidosis.     

Successful thoracoscopic resection of pulmonary arterio-venous fistula]

A 65-year-old female was admitted for further examination of an abnormal shadow on a chest roentgenogram. Chest computed tomography revealed a lobular nodule with blood vessels, which was diagnosed as pulmonary arterio-venous fistula (PAVF). Thoracoscopic resection was performed under general anesthesia. The fistula was situated directly below the pleura of the left lower lobe. Partial resection of the left lung including the PAVF was performed with an autosuture device dedicated to endoscopic surgery (Endo GIA II). The patient was discharged from our hospital 3 days after surgery without major complications. Mild hypoxemia was corrected after the surgery. Thoracoscopic resection is considered to be safe and useful for a solitary PAVF existing a peripheral field of the lung.     

Pulmonary histoplasmosis exhibiting solitary pulmonary nodule resected by thoracoscopic surgery: a case report and review of the Japanese literature]

We reported a case of pulmonary histoplasmosis showing solitary nodular shadow. A 43-year-old man was referred to our hospital because of an abnormal shadow on chest X-ray films during a routine checkup. He had traveled to Honduras for 7 days. Chest computed tomographic (CT) scans showed a 13 x 12 mm nodular shadow with unclear margin in the left upper lobe (S3). Both transbronchial lung biopsy and CT guided transcutaneous needle biopsy failed to yield a definitive diagnosis. Thoracoscopic resection of the nodule was performed due to suspicion of lung cancer. Pathologically, the nodule displayed central caseous necrosis with many round yeast-like fungi. The fungi measured 3 to 4 microns in diameter and were well-stained by Grocott stain. Immunohistochemical staining was positive for anti-Histoplasma capsulatum antibody, resulting in the final diagnosis of pulmonary histoplasmosis. The patient's postoperative course was uneventful, and no recurrence was observed. Histoplasmosis is a rare disease in Japan. However, it is important to keep imported infectious diseases in mind when examining and treating patients who have a history of travel abroad.     

Spontaneous resolution of multiple nodular pulmonary AA amyloidosis

A 62-year-old man presented with a two-week history of dry cough. A chest computed tomography (CT) showed three nodular masses of soft tissue density without calcification or cavitary formation in the right lung. F-18 fluorodeoxyglucose PET/CT scan revealed high FDG uptake in two out of three pulmonary nodules. Transbronchial lung biopsy specimens consisted of amorphous eosinophilic deposits that were demonstrated to be amyloid because they were positive for Congo Red staining. After oxidation with permanganate solution, the Congo Red staining disappeared, indicating that this amyloid was amyloid A protein-derived type. There was no evidence of any systemic diseases. We diagnosed the patient as having multiple nodular pulmonary AA amyloidosis. The patient was conservatively managed without treatment, and the pulmonary nodules disappeared spontaneously three months later.     

Pulmonary eosinophilic granuloma presenting as a solitary pulmonary nodule]

A symptomless 60 year old man with a heavy smoking history presented with a nodule in the right lung, which was found by an annual chest X-ray. Chest CT showed a 1 cm speculated nodule in the right S8. Bronchoscopic transbronchial biopsy to the nodule yielded no definite diagnosis. Since the nodule was highly suggestive of lung cancer, wedge resection was performed by video-assisted thoracoscopic surgery. Pathological findings of the specimen showed that the nodule consisted of a mixed population of histiocytoid cells with eosinophils. The nuclei and cytoplasms of the histiocytoid cells were stained positively for S-100 protein. Pulmonary eosinophilic granuloma (PEG) was diagnosed. Common radiographic findings of PEG should present with a mixture of multiple nodular shadows and cystic lesions. PEG presenting as a solitary nodule is rare.     

Nodular Pulmonary Amyloidosis Associated with Sjögren's Syndrome

Amyloidosis is a rare disease characterized by the deposition of abnormal proteins in extracellular tissues. We herein report a case with instructive radiologic features of nodular pulmonary amyloidosis associated with Sjögren''s syndrome. A 67-year-old woman was referred to our department because of an abnormal chest radiograph. Chest computed tomography revealed multiple round cysts accompanied by calcified nodules. The patient was clinically diagnosed with primary Sjögren''s syndrome and pathologically diagnosed with nodular pulmonary amyloidosis (light chain, kappa). Although multiple lung cysts have many etiologies, the presence of calcified nodules associated with multiple lung cysts is useful for narrowing down the differential diagnosis.     

An intra-pulmonary lymphnode resembling typical pulmonary primary adenocarcinoma on high-resolution computed tomographic (HR-CT) chest images]

A 40-year-old man was admitted to our hospital for further evaluation of a pulmonary nodule in chest radiographs. The 8-mm nodular lesion was located in the right anterior basal lobe on a plain chest radiograph, and showed 1) spiculation, 2) pleural indentation and 3) a converging vessel formation in high-resolution computed tomography of the chest. The radiographic findings were highly suggestive of primary pulmonary adenocarcinoma and the patient underwent video-assisted thoracoscopic surgery (VATS) to obtain a precise diagnosis. The nodule was diagnosed histopathologically as an intrapulmonary lymph node. In cases with such radiographic findings, careful attention should be paid in the differential diagnosis to distinguish intrapulmonary lymph nodes from primary pulmonary adenocarcinoma.     

A case of multiple nodular pulmonary amyloidosis complicated with primary Sjogren syndrome]

A 79-year-old woman was admitted to the Department of Orthopedics Surgery for treatment of osteoarthritis in her knee. Multiple pulmonary nodular lesions were found on preoperative chest x-ray film screening. Metastatic lung tumor was suspected, but no tumorous lesions were detected in other organs. CT guided lung biopsy was performed. Histopathological examination revealed amyloid consisting of homogenous eosinophilic materials. No amyloid deposits were detected in other organs, so we diagnosed localized nodular pulmonary amyloidosis. She was subsequently given a diagnosis of primary Sj?gren syndrome. We believe that such a case of multiple nodular pulmonary amyloidosis with Sj?gren syndrome is rare, and the case showed interesting radiological findings, such as mimicking metastatic lung tumor.     

Four cases of inflammatory pseudotumor]

We report four cases that were pathologically diagnosed, after surgical resection, as inflammatory pseudotumors (IPT) of the lung in patients admitted to Fukuoka University Hospital between 1985 and 2001. On chest radiographs, one patient had a solitary nodular shadow, while the other three had multiple nodular shadows. Chest computed tomography (CT) was performed in 3 patients. All had at least one nodule attached to the pleura. In one case, multiple nodules that had been noted five years before and had disappeared later without treatment, had reappeared two months before admission. The tentative preoperative diagnosis was primary or metastatic lung cancer. Precise diagnosis of IPT totally depends on histological examination after surgical resection. IPT sometimes tends to grow aggressively, although it is histologically benign. Complete resection is the only treatment to avoid relapse.     

Three cases of the nodular pulmonary amyloidosis with a longterm observation

Longterm observation with chest radiograph and computed tomography (CT) scan was performed for pulmonary amyloidosis. There are few reports of primary pulmonary amyloidosis with a longterm observation. We encountered three cases of nodular pulmonary amyloidosis observed by intermittent chest radiograph or CT for 5 years or more. The patients were a 54-year-old man, and 67- and 68-year old women. For diagnosis, transbronchial biopsy and percutaneous lung biopsy were performed. Amyloid nodules grew slowly and two cases showed findings of cavity and calcification.     

A clinicopathological study of pulmonary cryptococcosis--chest CT and pathologic correlation]

We reviewed the clinicopathological features in 12 patients (7 males and 5 females; mean age 54 yr) with pulmonary cryptococcosis. Eleven of the patients were asymptomatic and the disease was detected by chest radiograph abnormalities. The underlying systemic disease had been diagnosed as diabetes mellitus in two. Chest CT scans showed a solitary nodule in 9 of the 12 patients, multiple nodules in 2, and infiltration in 1. The nodular diameter was less than 2 cm in 10 of the 12. All nodules were located in the subpleural region. On the chest CT, cavitary nodules, scattered nodules, or both, and spiculated nodules were difficult to distinguish from pulmonary tuberculosis and primary lung cancer, respectively. According to McDonnell's pathological classification of pulmonary cryptococcosis, the resected 8 lungs revealed peripheral pulmonary granuloma in 5 and granulomatous pneumonia in 3. It is important to perform a pathological examination for the diagnosis of pulmonary cryptococcosis to avoid misdiagnosis as lung cancer or pulmonary tuberculosis.     

Pulmonary AL amyloidosis in a patient with primary Sjögren syndrome

The condition of a 29-year-old woman with primary Sjögren syndrome (SS) was complicated by amyloid light chains- (AL-) type amyloidosis in the paranasal sinus. She had not complained of respiratory symptoms, but her chest computed tomography (CT) scan revealed bilateral multiple nodular shadows. Lung biopsy specimens using video-associated thoracoscopy showed amyloidoma in a subpleural nodular lesion and amyloid deposits in the interstitial parenchymal walls and pulmonary vessels. Pulmonary AL amyloidosis, presumably related to a chronic inflammatory lymphoproliferative process in SS, has rarely been reported.     

Pulmonary amyloidoma. Histologic proof yielded by transthoracic coaxial fine needle biopsy.

Nodular pulmonary amyloidosis was diagnosed by percutaneous transthoracic fine needle biopsy specimen in an 88-year-old woman. Congo red staining should be performed whenever band-like hyalinized material is obtained on aspiration of a solitary nodule. Dense calcifications can occur in pulmonary amyloidomas. In selected cases, fine needle biopsy appears to be preferable to transbronchial forceps biopsy since the risk of a possibly life-threatening pulmonary hemorrhage may be lower.     

Two cases with hepatic amyloidosis suspected of having primary sclerosing cholangitis

Sclerogenic biliary changes in hepatic amyloidosis are seldom observed. Here, we report two recent cases initially suspected as primary sclerosing cholangitis (PSC), which were later diagnosed as hepatic amyloidosis (AL type). Case 1: On the basis of magnetic resonance cholangiopancreatography (MRCP) findings, PSC was suspected in a 41‐year‐old woman with jaundice. Computed tomography (CT) showed nodular pulmonary lesions and swollen cervical, mediastinal and para‐aortic lymph nodes, the cause of which was unknown despite detailed examinations. Because of rapid deterioration in the patient's liver function, living donor liver transplantation was performed. She was then diagnosed with hepatic amyloidosis, but died of heart failure due to cardiac amyloidosis 74 days after surgery. Case 2: On the basis of MRCP findings, PSC was suspected in a 49‐year‐old woman with jaundice. CT showed multiple cystic pulmonary lesions, and hypogammaglobulinemia was also observed (immunoglobulin G, 481 mg/dL). After a biliary plastic stent was placed, liver and lung biopsy confirmed the presence of amyloid deposition. These two cases indicate that it is important to consider hepatic amyloidosis as a differential diagnosis of PSC. The presence of atypical extrahepatic lesions may be useful clues for confirming the diagnosis.     

Growth of a solitary pulmonary nodule after 6 years diagnosed as oncocytic carcinoid tumour with a high 18‐fluorodeoxyglucose (18F‐FDG) uptake in positron emission tomography‐computed tomography (PET‐CT)

Introduction: Pulmonary carcinoid tumour is low‐grade neuroendocrine malignancy that is seen 1%–2% of all lung neoplasms. Oncocytic carcinoid type is a rarely seen variant of pulmonary carcinoids. As carcinoid tumours have hypometabolic activity, they usually have lower 18‐fluorodeoxyglucose (18F‐FDG) uptake than expected for lung carcinoma on positron emission tomography (PET). Case Report: A 45‐year‐old non‐smoking man had a stable solitary pulmonary nodule followed for 6 years; the tumour remained the same size (1.5 × 2.4 cm) during this period. The patient was admitted to the hospital with complaints of repetitive sneezing and rhinorrhoea. He also experienced flushing and bronchospasm. His chest X‐ray revealed a minimal increase in the size of the solitary pulmonary nodule (2.0 × 2.8 cm). In PET‐computed tomography (CT), the parenchymal nodule in the anterior segment of the right lung had a standard uptake value of 38.0 mg/mL, which was interpreted as a malignant nodule. He underwent fibre‐optic bronchoscopy, but cytology showed no evidence of malignancy. Right upper and middle bilobectomy was performed, and a pulmonary carcinoid tumour with an oncocytic subgroup was diagnosed. The diagnosis of carcinoid syndrome was further confirmed by an elevated 24‐h urinary excretion of 5‐hydroxyindoleacetic acid. Conclusion: We present a rare case of an oncocytic carcinoid tumour with an increase in the size of a solitary pulmonary nodule after 6 years' follow‐up. In addition, PET‐CT showed a very high 18F‐FDG uptake in this patient, which is an unexpected finding with a pulmonary carcinoid tumour. Please cite this paper as: Turan O, Ozdogan O, Gurel D, Onen A, Kargi A and Sevinc C. Growth of a solitary pulmonary nodule after 6 years diagnosed as oncocytic carcinoid tumour with a high 18F‐FDG uptake in positron emission tomography‐computed tomography (PET‐CT). Clin Respir J 2013; 7: e1–e5.     

A case of lung adenocarcinoma presenting a solitary, predominantly solid pulmonary nodule that showed slow growth over 7 years]

A small, solitary, predominantly solid pulmonary nodule (7 x 6 mm) was found in a 63-year-old woman during a CT screening for lung cancer. After 7 years, another chest CT examination revealed that the lesion had grown to a size of 15 x 10 mm. The patient then underwent surgery to remove the nodule, because primary lung cancer was strongly suspected. The resected specimen proved to be a poorly differentiated adenocarcinoma of type D according to the criteria of Noguchi et al. The tumor doubling time (TDT) in this case was estimated to be 661 days, which was longer than in other reported cases of Noguchi type D adenocarcinoma. High-resolution CT (HRCT) of the nodule revealed a predominantly solid lesion with a polygonal shape. No further changes were observed in a one-year follow-up CT, suggesting a benign tumor. We therefore suggest that the follow-up of small, solitary pulmonary nodules is of diagnostic value.     

Improvement of extrapulmonary lesions of eosinophilic granuloma after cessation of smoking]

We encountered a case of pulmonary eosinophilic granuloma complicated with pneumothorax. A 24-year-old man was admitted to our department because of respiratory difficulty. Chest radiography on admission showed a right pneumothorax. A thoracic catheter was therefore inserted, and the pneumothorax was improved. A chest radiograph obtained after treatment showed diffuse linear reticular shadows; and a chest CT scan showed starlike nodular lesions and multiple cysts in the lungs, and a large nodule in the left rib. Because an ulcer had been diagnosed in the oral cavity 1 year previously, a mandible biopsy was performed, and a granulomatous lesion consisting of eosinophils was recognized. Taken together with the pulmonary findings, an eosinophilic granuloma was diagnosed. The patient was instructed not to smoke and the clinical course was observed. The pulmonary, mandibular, and costal lesions improved.     

A case of micronodular pneumocyte hyperplasia diagnosed through lung biopsy using thoracoscopy

We present a case of a 39-year-old woman with sporadic tuberous sclerosis (TSC), whose chest radiograph demonstrated bilateral diffuse nodular shadowing. A transbronchial lung biopsy specimen revealed the possibility of multiple atypical adenomatous hyperplasia (AAH), which had not been reported in TSC. Thoracoscopic lung biopsy was, therefore, performed. The specimens revealed the characteristic histological and immunohistochemical features of micronodular pneumocyte hyperplasia, which has been reported as an extremely rare pulmonary manifestation of TSC. In addition, no evidence of AAH or any other pulmonary involvements of TSC including lymphangioleiomyomatosis were detected in biopsy specimens obtained at thoracoscopy.     

Nodular pulmonary amyloidosis in a patient with rheumatoid arthritis

We describe a 67-year-old white woman with a long-standing active rheumatoid arthritis who refused treatment. Chest roentgenograms performed in 2000 revealed a pulmonary nodule in the mid-left lung. Progression of the nodule was followed annually by computerized tomography (CT). In the last CT in 2002, we observed multiple nodules in both lungs in the absence of lymph gland involvement. The patient was operated by videothoracoscopy to resect one of the pulmonary nodules. Pathological examination of the excised tissue revealed amyloid A-type (AA) amyloidosis. Although pulmonary amyloidosis is rare in patients with systemic AA amyloidosis, we recommend that this possibility be considered when confronted with a patient with these characteristics.