Two cases of pulmonary eosinophilic granuloma with multiple nodular shadows]

We encountered two rare cases of pulmonary eosinophilic granuloma with multiple nodular shadows in both lungs. The patient in case 1 was a 54-year-old man complaining of dry cough and chest pain. He had smoked 20 cigarettes a day for 36 years. The patient in case 2 was a 37-year-old woman complaining of dry cough. She had smoked 15 cigarettes a day for 20 years. Chest radiography and CT revealed multiple nodular shadows in both lungs. Diagnosis was made by open lung biopsy in case 1 and by percutaneous lung biopsy in case 2. After smoking cessation, symptoms improved markedly and the shadows in the chest radiographs and CT disappeared. In cases of bilateral multiple nodular shadows, other than metastatic lung tumor cases, pulmonary eosinophilic granuloma should be considered.     

肺部原发性小血管炎的临床表现和诊断

目的 研究肺脏受累的原发性小血管炎的临床特点和早期诊断要点，减少其误诊率。方法 对26例原发性小血管炎伴肺脏受累患者的临床资料进行回顾性分析。结果 26例中，显微镜下多血管炎(MPA)15例，韦格纳肉芽肿(WG)9例，变应性肉芽肿性血管炎(CCS)2例。抗中性粒细胞胞浆抗体(ANCA)阳性率61．5％。肺脏受累的临床表现有咯血、呼吸困难、干咳、胸痛、胸闷等，胸部X线多表现为多发斑片状或点状阴影、大片高密度阴影或团块影、结节影、网格状影，可有空洞形成。疾病早期多误诊为肺部感染、肿瘤、肺间质病变等。应用肾上腺皮质激素和环磷酰胺治疗肺部病变可明显吸收，临床危重症得以改善。结论 伴肺脏受累的原发性小血管炎临床表现复杂多样，诊断困难，误诊率高，易延误病情，须引起临床医师的重视。     

Rapid remission of pulmonary eosinophilic granuloma in a young male patient after cessation of smoking]

A 24-year-old asymptomatic man was referred to our hospital in October 7, 1997 for further examination of abnormal shadows detected on chest X-ray films that had been obtained during an annual health examination on September 12, 1997. The patient had suffered productive cough since the end of August 1997. A chest X-ray film obtained one year earlier showed no abnormalities. The patient had smoked 20 cigarettes per day since the age of 14. The findings of a physical examination and laboratory tests were unremarkable. A chest X-ray films showed micronodules 2-5 mm in size that were predominantly distributed in the upper and middle fields of both lungs. A chest computed tomogram (CT) showed multiple cysts measuring about 10 mm in diameter, with thick walls (2 to 3 mm) in addition to small nodular shadows. A transbronchial lung biopsy and a thoracoscopic lung biopsy revealed several stellate nodular fibrotic lesions containing some S-100 protein-positive, large mononuclear cells and lymphoid cells. An electron microscopic study found several Langerhans' cells with Birbeck's granules in their cytoplasm. A chest CT scan obtained 2 months after the patient stopped smoking (the day of admission) showed marked regression of the cystic shadows. Evidence suggests the pathogenesis of the disease is closely linked to smoking, and some case reports have documented clinical and radiographic improvements after patients stop smoking. Pulmonary small nodular lesions and cystic lesions with thick walls tend to undergo regression. Although surgical lung biopsy specimens disclosed several stellate nodular fibrotic lesions of eosinophilic granuloma that seemed to be histologically irreversible, our patient experienced remarkable regression of his pulmonary lesions within 2 months after the cessation of smoking.     

Multiple cavitary pulmonary metastases from ovarian cancer: a case report]

Cavitation in pulmonary metastases is thought to be uncommon. To date, few cases of pulmonary metastases originating from ovarian cancer and showing cavitation have been reported. We report a patient with multiple cavitation in pulmonary metastases from ovarian mucinous cystadenocarcinoma. A 28-year-old woman was admitted to our hospital presenting with cough and fever. The patient had undergone right ovariectomy for ovarian mucinous cystadenocarcinoma at the age of 23 years. Her chest radiograph on admission showed multiple cavities associated with infiltration in both lungs. Histological sections obtained by transbronchial lung biopsy revealed mucus-secreting adenocarcinoma, and a diagnosis of metastatic lung cancer from the ovary was made. Computed tomographic (CT) scans of the chest demonstrated various findings, including multiple thick-walled cavities, thin-walled cavities, air-space consolidations, ground glass opacities, and centrilobular nodular shadows formed by aspiration of the mucinous secretions. It is important to recognize that cavitation can occur in pulmonary metastases from ovarian cancer.     

Multiple intrapulmonary rheumatoid nodules]

A 66-year-old woman who had been treated at a nearby hospital since 1977 for rheumatoid arthritis complained of cough. Chest X-ray films disclosed multiple nodular shadows with cavitation in the fields of both lungs. The patient was admitted to our hospital and a thoracoscopic lung biopsy was performed. Histologically, the nodule consisted of necrotizing granuloma, indicating a necrobiotic nodule. Rheumatoid nodule was diagnosed because the patient exhibited rheumatoid arthritis. The chest X-ray shadow disappeared without medication. Rheumatoid nodules without coniosis are uncommon, but should be considered in the differential diagnosis of lung nodular lesions in patients with rheumatoid arthritis.     

A case of proliferative sparganosis associated with PIE syndrome and pulmonary embolism

A 48-year-old man was admitted because of ischialgia. The CT scanning revealed tumors in the right pelvic cavity and the gluteal muscle. The bodies of Sparganum proliferum and granulation tissues were revealed by the tumor biopsy. In this hospital course, the chest roentgenogram revealed patchy infiltrative shadows in both lungs and left pleural effusion while chest CT scanning revealed multiple nodular shadows. Eosinophils increased in the peripheral blood and transbronchial lung biopsy (TBLB) showed non-destructive vasculitis with eosinophilic infiltration. The pulmonary lesion was diagnosed as intrapulmonary migration of Sparganum proliferum and PIE syndrome caused by this worm. Thiabendazole and prednisolone were administrated, but he died of pulmonary embolism.     

Case report of multiple myeloma associated with diffuse pulmonary calcinosis]

A case of multiple myeloma with diffuse metastatic calcinosis of the lung is presented. The patient was a 60-year-old male with IgA-kappa-myeloma who developed renal failure and hypercalcemia. Multiple small nodular shadows were observed both in plain chest films and CT films. The patient died of progressive respiratory failure. Postmortem examinations showed pulmonary infiltrations and massive pulmonary calcifications. Small nodular shadows were due to diffuse calcium deposits which were observed in and around the alveolar basement membranes of both the bronchioles and the blood vessels. It is generally believed that pulmonary calcinosis may not be detected by routine chest films; however, the nodular shadows observed in our patient seem to be pathognomonic and may indicate the severity of calcinosis.     

肺脏受累的原发性小血管炎的诊断和治疗

目的了解肺脏受累的原发性小血管炎的临床特点。方法对1993～1998年间13例肺脏受累的原发性小血管炎进行回顾性分析。结果13例中7例为显微镜下多血管炎（MPA）,6例为Wegener肉芽肿（WG）。发病年龄17～68岁,平均48．8岁。抗中性粒细胞胞浆抗体（ANCA）阳性率69％（9／13）例,MPA的ANCA阳性率为100％（7／7）,WG为33％（2／6）。肺部症状包括咯血占69％（9／13）、呼吸困难占23％（3／13）、干咳占15％（2／13）、胸痛占15％（2／13）。胸部X线在MPA多表现为多发斑片状影（3／7）或大片状阴影（2／7）,WG则主要是单发或多发团块影或结节影（5／6）,可有空洞形成。MPA的肺部表现多误诊为肺部感染71％（5／7）。WG多误诊为肿瘤对％（4／6）。使用肾上腺皮质激素及环磷酰胺（CTX）治疗有效,尤其肺部损害可明显吸收。结论伴肺脏受累的原发性小血管炎诊断困难,须引起注意。ANCA对MPA的诊断有重要价值。激素及CYX对该组疾病疗效显著。     

A case of endobronchial sarcoidosis presenting as a polypoid lesion]

A 51-year-old woman was admitted to our hospital because of deterioration of abnormal chest shadows. Though the shadows had been pointed out at another hospital about two years before, no evaluation had been made at that time. A chest CT scan showed multiple small nodular shadows and thickening of the bronchovascular bundles bilaterally, and marked swollen lymph nodes in the axilla, mediastinum, and bilateral the hili of both lungs. Bronchoscopic evaluation revealed bronchial mucosal hypervascularity and a polypoid lesion at the orifice of the right B8b. The transbronchial biopsy specimen of the polypoid lesion showed non-caseating epithelioid cell granulomas in the bronchial mucosa. The bronchoalveolar lavage revealed a increase in the total number of cells including high levels of lymphocytes. Therefore, a diagnosis of sarcoidosis was made. This is a very rare case of endobronchial sarcoidosis. However, biopsy specimens of normal mucosa in sarcoidosis often show a microscopic sarcoid process, so a diagnosis of endobronchial sarcoidosis in this case should be given prudently.     

A case of multiple pulmonary fibroleiomyomatous hamartoma]

A 50-year-old woman had been followed up at another hospital since 1996 because of multiple nodular shadows in both lung fields on chest radiography and CT. She was admitted to our hospital on 27 July 1999 to undergo further examination for the chest shadows, which had become enlarged since 1998. One of them was surgically removed by using video-assisted thoracoscopy. Its pathological features showed a mass composed of spindle-shaped smooth muscle cells, located adjacent to the bronchiolar wall and connected with the bronchiolar smooth muscle. Moreover, the mass had cystic spaces covered only with monolayered bronchiolar epithelium. Therefore, the mass was considered to be a hamartomatous proliferation of bronchiolar smooth muscle rather than a metastatic tumor of uterine leiomyoma. This case was thought to be important, since there are few reports concerning the pathological features of multiple pulmonary fibroleiomyomatous hamartomas.     

A case of primary pulmonary MALT lymphoma as a nodular shadow on CT scan, and relapsed with diffuse micronodular shadows after surgical resection at 7 years ago]

A 76-year-old man without symptoms was admitted to our hospital for investigation of an abnormal chest shadow in 1994. His chest radiograph showed a nodular shadow in the lingual lobe. Segmentectomy was performed and the histological diagnosis was MALT (mucosa-associated lymphoid tissue) lymphoma. In June 2001, there were no abnormal shadows on the chest radiograph or in 10 mm slice CT. However, high-resolution CT with 2-mm slice thickness revealed diffuse micronodular shadows in both lungs. A relapse of MALT lymphoma was diagnosed by immunohistochemical analysis of tissue specimens obtained by transbronchial lung biopsy. It was observed that the patterns of radiological findings in primary pulmonary MALT lymphoma cases may differ between the initial state and a relapse.     

Pulmonary nocardiosis with bilateral diffuse granular lung shadows in a patient with subcutaneous panniculitic T-cell lymphoma

A 40-year-old woman undergoing prednisolone and cyclosporine therapy for subcutaneous panniculitic T-cell lymphoma complained of a cough for a few weeks. A chest X-ray revealed bilateral diffuse granular shadows. Additionally, the patient was discovered to have multiple subcutaneous abscesses. Gram-stained smears of sputum and pus from the abscess showing branched gram-positive rods led to a diagnosis of pulmonary nocardiosis with dissemination to the lungs and subcutaneous tissues. Combination therapy consisting of sulfamethoxazole/trimethoprim and panipenem/betamipron produced rapid improvement of radiographic abnormalities. It is suggested that pulmonary nocardiosis should be considered in the differential diagnosis of diffuse granular shadows on chest X-rays, especially in immunocompromised patients.     

非免疫缺陷原发性肺隐球菌病21例临床分析

目的总结肺隐球菌病的临床资料,提高其诊疗水平。方法收集确诊的21例原发性肺隐球菌病患者的临床资料,对临床表现、实验室检查、影像学检查、病理学检查及治疗进行回顾。结果男性15例,女性6例。有基础疾病4例,无基础疾病17例。肺部症状不典型,有咳嗽、咳痰、发热、咯血、胸痛等;其影像学表现常见结节影、块影、片影等,可伴有空洞、毛刺等;孤立结节影6例,病灶累及双肺15例。初期误诊为肺癌行手术切除7例,误诊为结核4例,误诊为普通肺炎7例。17例抗真菌治疗有效,4例抗真菌治疗后行肺叶切除。结论肺隐球菌病临床及影像学表现不特异,无明显免疫缺陷者也可发病,常误诊为肺癌、肺结核、肺炎,确诊多依赖病理活检。多数患者抗真菌治疗有效,少数需手术治疗。     

Septic pulmonary embolism associated with periodontal disease: reports of two cases and review of the literature

We report two cases of septic pulmonary embolism associated with periodontitis. Chest CT revealed multiple nodular shadows with features characteristic of septic pulmonary embolism in both patients. Both patients had toothache, fever, and chest pain, and showed findings of periodontitis at initial presentation. Antimicrobial agents combined with dental surgery were successful in treatment. While septic pulmonary embolism from the lesions of periodontitis appears to be rare, periodontitis remains important in the differential diagnosis of septic pulmonary embolism.     

A case of chronic pulmonary paracoccidioidomycosis]

In a 43-year-old Japanese Brazilian who came to Japan in 2001, since subjective symptoms such as cough, sputum, and dyspnea on exertion had become severe, he was referred to our hospital because of suspicion of pulmonary tuberculosis in chest radiography and CT findings. A chest radiograph of initial examination showed interstitial shadows in both lungs with nodular, infiltrative or cavitary changes. No Mycobacterium tuberculosis was found. The mycetocyte with multipolar budding resembling the steerage of a ship, which was characteristic of Paracoccidioides was observed in sputum and transbronchial lung biopsy specimens. We cultured a fungus to show dimorphism of temperature dependency, and a diagnosis of chronic lung paracoccidioidomycosis was arrived at. By administration of ITCZ 200 mg/day, the chest radiography findings and clinical manifestations were improved. This case seems to be worthy of reporting in Japan since the affected site or organ was limited to the two lungs with multiple cavitary lesions and fibrotic changes on radiographic examination, and final diagnosis was made by cytology of sputum and pathology of TBLB specimens.     

Minute pulmonary meningothelial-like nodules: a case report]

The presence of multiple small nodular shadows on a routine chest radiograph was noticed in a 56-year-old woman who had undergone a left mastectomy on the diagnosis of breast cancer 8 years before. Chest CT films revealed small nodules scattered beneath the pleura mainly in both lower lobes. A biopsy was performed during video-assisted thoracoscopy to rule out metastasis from breast cancer. Biopsy specimens showed spindle--or oval-shaped cells arranged in nests associated with a dedicated network of capillaries. These findings were compatible with minute meningothelium-like nodules (MN). The pathogenesis of MN is still unknown. It is common in elderly women, and the prognosis is excellent without any treatment. MN is an important disease in the differential diagnosis of multiple nodular shadows found on chest CT.     

Pulmonary Mycobacterium fortuitum infection with multiple nodular shadows in a healthy man

We report a case of pulmonary Mycobacterium fortuitum infection with multiple nodular shadows. A 52 year-old male was admitted complaining of fever and chest abnormal shadows. He didn't have pulmonary or systemic underlying diseases. Chest radiograph and computed tomography scan showed multiple nodular shadows in the both lung fields. Isoniazid, rifampicin and ethambutol were administered based on the presumptive diagnosis of tuberculosis. Cultures of the sputum and bronchial washing fluid were repeatedly positive for M. fortuitum, and the case was diagnosed as pulmonary M. fortuitum infection. Although the in vitro susceptibility was resistant to isoniazid, rifampicin and ethambutol, abnormal shadows on the X-ray showed improvement by the combined use of INH, RFP and EB. There are no signs of recurrence after completion of the treatment for 12 months.     

Acute eosinophilic pneumonia with fine nodular shadows

A 19-year-old woman presented with acute onset of cough and dyspnea. She started smoking two weeks before the appearance of symptoms. On admission, arterial blood gas analysis on room air breathing revealed PaO2 55 Torr. Chest roentgenogram and high resolution computed tomograms showed localized fine nodular shadows at the right lower lung field. Bronchoalveolar lavage fluid revealed a high eosinophil count. Eosinophil infiltration was also observed in transbronchial lung biopsy specimens. The final diagnosis was acute eosinophilic pneumonia (AEP). Although few reports have demonstrated diffuse fine nodular shadows in AEP, localized fine nodular shadows on chest roentgenogram and CT may sometimes be the sign of AEP especially in the early phase of the clinical course.     

A case of Beh?et disease with multiple nodular shadows and aneurysm of the brachiocephalic trunk caused by necrotizing vasculitis]

A 36-year-old man was admitted to our hospital in 1994 because of fever, and abdominal CT showed multiple low-density areas in the liver. Although granulomas were found in a liver biopsy specimen, a definitive diagnosis could not be established. With complaints of oral and genital ulcerations and erythema nodosum, Beh?et disease was diagnosed in 1995 and he was treated with colchicine and cyclosporin. In May 1997 he had fever, leg edema, and proteinuria, and a renal biopsy revealed secondary amyloidosis. Cavitary lesions were found on a chest X-ray for the first time, but these later disappeared spontaneously. In October 2002, nodular shadows, cavitary lesions, and a mediastinal tumor appeared on a chest X-ray film. The nodular shadows in the lung fields had transformed into cavity lesions, resulting in the disappearance of the shadows. Specimens obtained from an open lung biopsy showed necrotizing granulomas and destructive vasculitis of the lung, and aneurysm of the brachiocephalic trunk caused by destructive vasculitis. Because multiple nodular shadows with cavitary lesions in Beh?et disease, as in this case, have never been reported, we think this is a rare case.     

Wegener's granulomatosis in a woman with asymptomatic primary biliary cirrhosis

A 60-year-old woman was admitted to our hospital in June 1985, complaining of fever, cough and right lower chest pain, with a five-year history of asymptomatic primary biliary cirrhosis. Chest X-ray on admission showed an infiltrative shadow in the right lower lung field. She was first treated with various antibiotics unsuccessfully. Hemoptysis continued. Dyspnea and anemia appeared. Chest X-ray 17 days after admission showed multiple infiltrative shadows in the both lung fields. She was treated with steroid pulse therapy successfully. During prednisolone treatment decreasing nodular shadows with cavities appeared on chest X-ray. An open lung biopsy was performed in March 1986. The histologic findings showed a necrotizing vasculitis with granuloma and perivascular fibrosis. She was treated with prednisolone and prednisolone-azathioprine therapy unsuccessfully, but successfully with prednisolone-cyclophosphamide therapy. This case was a rare case of Wegener's granulomatosis with transition from fulminant type to granulomatous type. No similar case of Wegener's granulomatosis with asymptomatic primary biliary cirrhosis has been reported in the literature.