Two cases of inflammatory pseudotumor of the lung

Two cases of inflammatory pseudotumor of the lung were reported. Case 1. A 68 year-old man was referred to our hospital because of an abnormal shadow on chest X-ray. He had a history of pneumonia in the right upper lobe five months before. The chest X-ray film revealed a coin lesion in the right upper lung field (S1), the same segment as the previous pneumonia. Although RPF and INH were administered for three months, the shadow did not change, and cough and sputum continued. In order to confirm the diagnosis, open thoracotomy was performed and microscopic findings of the resected tumor showed inflammatory pseudotumor; proliferation of fibrous tissue with infiltration by inflammatory cells (plasma cells, lymphocytes and a few neutrophils). Case 2. A 35 year-old man was admitted to our hospital because of an abnormal shadow on chest X-ray, i.e. a coin lesion with vascular indentation in the left lower lung field (S8). A wedge resection including the mass was performed, and histopathologic examination revealed inflammatory pseudotumor, or plasma cell granuloma; proliferation of fibrous connective tissue with infiltration by predominantly mature plasma cells. Both patients have been doing well after the operation. We also reviewed 46 cases reported in the Japanese literature and discussed various aspects of this disease. Of 10 patients who had a history of previous respiratory tract infection, 8 had histopathologic features with various inflammatory cells, including many lymphocytes, as in our Case 1. Although the common etiology of inflammatory pseudotumor is obscure, we suppose that in some cases the lesion may be a result of post-inflammatory repair process.     

Inflammatory pseudotumor of the lung]

The patient was a 76-year-old man whose chief complaint was a dry cough. His chest X-ray film revealed a large hazy shadow with unclear margin in the left upper lobe. Bronchiolitis obliterans organizing pneumonia was initially diagnosed because transbronchial lung biopsy (TBLB) specimens showed organizing pneumonia with no evidence of malignancy. However, because the hazy shadow increased gradually in size despite steroid therapy. TBLBs were performed several more times to confirm the diagnosis. The last TBLB specimen showed proliferation of fibroblasts and mononuclear cells, with marked infiltration mainly of plasma cells 12 months after the initiation of steroid therapy. Because we were unable to obtain a histological diagnosis by bronchofiberscopy, a left upper lobectomy was preformed and the lesion resected. Histology disclosed inflammatory pseudotumor of a lymphoplasmacytic type with organizing pneumonia. The results of an immuno-histochemical examination confirmed that the proliferating plasma cells were polyclonal. These findings suggest that inflammatory pseudotumors should be taken into account by differential diagnoses of cases of organizing pneumonia that are resistant to steroid therapy.     

口服糖皮质激素治疗肺部浆细胞肉芽肿1例病例报告及文献复习

目的浆细胞性肉芽肿是炎性假瘤的一种亚型,是一种特发性的炎性假瘤,肺部多发,临床较为罕见,通过病例报告及文献复习提高对该病的认识。方法我们报道了1例39岁男性患者,以进行性胸闷、活动后气喘、活动耐量较平时减低为主要临床表现。胸部CT提示右肺多灶斑片影。给予充分抗感染治疗后复查胸部CT无明显变化,遂予CT引导经皮肺穿刺活检。HE染色显示肺泡间隔明显增厚及多量浆细胞及散在中性粒细胞侵润。肺间质局灶区胶原纤维增生和淋巴细胞聚集,未见明确上皮样肉芽肿,考虑浆细胞性肉芽肿。确诊后患者拒绝手术,随即给予口服糖皮质激素治疗。结果口服糖皮质激素治疗半年后,复查胸部CT提示肺部病灶得到吸收。结论对于临床多发斑片或者占位性病变,在抗感染治疗效果不佳后应积极通过活检取得病理帮助明确诊断,在活检病理提示炎症伴浆细胞浸润时要考虑浆细胞性肉芽肿疾病的可能性,并通过给予口服糖皮质激素取得满意疗效,避免误诊为吸收不佳的普通肺炎而延误治疗。     

48例肺部炎性假瘤患者内科治疗体会

目的总结肺部炎性假瘤的临床诊治经验。方法分析我院呼吸内科收治48例肺部炎性假瘤临床资料。结果48例肺部炎性假瘤病人中,最终外科手术治疗23例(47.9%),放弃治疗8例,内科治疗17例(35.4%),其中病灶吸收明显者8例,病灶略有吸收者5例,病灶增大者4例。结论外科手术应为肺部炎性假瘤首选治疗方案;内科治疗可能有效,对于拒绝手术或无手术指征的患者可能为一种有效的选择。     

Inflammatory Pseudotumor of the Liver Associated with Acute Myelomonocytic Leukemia

A patient with inflammatory pseudotumor of the liver associated with acute myelomonocytic leukemia (M4) is reported. He had spiking fever, epigastralgia, and elevated levels of serum C-reactive protein (CRP) and alkaline phosphatase (ALP). Ultrasonography showed a hypoechoic mass in the liver, and ultrasonically guided fine needle aspiration biopsy of the mass revealed that it was composed of fibrous connective tissue infiltrated with plasma cells, eosinophils, and neutrophils. Accordingly, a diagnosis of inflammatory pseudotumor of the liver was made. Marked reduction in the size of the lesion and a decrease of the levels of the CRP and ALP occurred without specific treatment. We emphasize the importance of ultrasonically guided aspiration biopsy in diagnosis of inflammatory pseudotumor of the liver without the need for surgery.     

Immunoglobulin G4-related gastrointestinal diseases,are they immunoglobulin G4-related diseases?

In immunoglobulin G4(IgG4)-related disease(RD),organ enlargement or nodular lesions consisting of abundant infiltration of lymphocytes and IgG4-positive plasma cells and fibrosis are seen in various organs.Although infiltration of many IgG4-positive plasma cells is detected in the gastric and colonic mucosa and major duodenal papilla of patients with autoimmune pancreatitis,it cannot be diagnosed as a gastrointestinal lesion involved in IgG4-RD,because none of the following is observed in these lesions:a mass-like formation;dense fibrosis;or obliterative phlebitis.Based on our review of the literature,there appear to be two types of IgG4-related gastrointestinal disease.One is a gastrointestinal lesion showing marked thickening of the wall of the esophagus and stomach,consisting of dense fibrosis with abundant infiltration of IgG4-positive plasma cells,which usually show submucosal spreading.The other is an IgG4-related pseudotumor occurring in gastrointestinal regions such as the stomach,colon,and major duodenal papilla,showing polypoid or mass-like lesions.Most solitary IgG4-related gastrointestinal lesions that are not associated with other IgG4-RD appear to be difficult to diagnose.It is of utmost importance to rule out malignancy.However,these lesions may respond to steroid therapy.To avoid unnecessary resection,IgG4-related gastrointestinal diseases should be considered in the differential diagnosis.     

Recurrence of inflammatory pseudotumor of the lung with endobronchial lesion after resection]

A 74-year-old man was admitted to our hospital because of an abnormal shadow in the left middle lung field on chest radiography. Since a transbronchial biopsy was not diagnostic, a left lower lobectomy was performed. Histopathologically, the patient was diagnosed as having an inflammatory pseudotumor of the lung. Four months after the surgery, the patient was readmitted with hemosputum. Bronchoscopically, an endobronchial lesion was found in the left main bronchus. Transbronchial biopsy of the lesion showed proliferation of inflammatory cells compatible with inflammatory pseudotumor. Therefore, a recurrent inflammatory pseudotumor was diagnosed. Treatment with corticosteroid resulted in the disappearance of the endobronchial lesion and the erythrocyte sedimentation rate (ESR) returned to normal. The patient showed a prolonged elevation of ESR even after the resection, suggesting that the ESR may be a good indicator of the activity of this disease.     

Diverse roentgenographic manifestations of the rare pulmonary involvement in myeloma.

Roentgenographic manifestations of two cases of pulmonary involvement with myeloma were presented. One showed a nodular mass lesion extending from an extramedullary mediastinal plasmacytoma into the lung parenchyma while the other showed diffuse reticulonodular infiltrate by myeloma cells in association with alveolar septal amyloidosis. These two cases demonstrate the extreme variability of pulmonary involvement by myeloma, which can mimic a solitary tumor or an inflammatory infiltrate.     

肺炎性假瘤的CT表现及误诊分析

目的探讨肺炎性假瘤的CT征象,分析其误诊原因。方法对38例经手术病理证实为肺炎性假瘤患者的CT征象及诊断进行回顾性分析。结果肺炎性假瘤较典型的CT表现为：病变多位于肺外周部分,平直征、桃尖征、空泡征、支气管充气征、毛刺与棘突征、均匀强化是其常见征象。肺炎性假瘤容易误诊为肺癌。结论全面分析CT征象能提高肺炎性假瘤的正确诊断率,减少误诊。     

Inflammatory pseudotumor of the right ventricle in a 35-year-old woman with Behçet's disease: a case report

Inflammatory pseudotumor is a rare benign neoplasm. It is common in children and has been reported in various locations throughout the body but rarely in the heart. Behçet's disease is a multisystemic, recurrent, inflammatory disorder. We report a 35‐year‐old Behçet's disease patient with pseudotumor of the right ventricle and multiple pulmonary emboli. Transthoracic echocardiography showed dilation of right atrium, right ventricle, and a mass in the right ventricle. Multislice computed tomography revealed a large, poorly defined mass infiltrating the groove and multiple pulmonary emboli. Surgery was performed with diagnostic and therapeutic intent. Following sternotomy, we identified a mass in the anterior wall of right ventricle, the outflow tract, and the inflow tract of right ventricle. The histopathologic analysis identified an inflammatory infiltrate composed of lymphocytes, plasma cells, and other inflammatory cells, without mitosis. And there were also some thrombus on top of the mass. (Echocardiography 2012;29:E134‐E136)     

A case of pulmonary sarcoidosis with usual interstitial pneumonia-like lesions distributed predominantly in the lower lung fields]

In 2000, in a 75-year-old man, nodular and reticular opacities were detected in both lower lung fields. He was admitted to our hospital for further examination of these abnormal shadows. Bronchoscopic examination revealed pulmonary sarcoidosis. Prednisolone was prescribed because cardiac sarcoidosis was diagnosed as a clinical complication. In April 2002, the patient visited our hospital for dyspnea on effort. Chest radiography and computed tomography showed nodular and reticular opacities in the right upper lobe, and video-assisted thoracoscopic surgery was performed on the basis of a histological diagnosis. The histological findings of the biopsied specimens revealed a lesion of the type seen in usual interstitial pneumonia, whereas non-caseous granulomas were not detected. His symptoms and chest radiographic findings improved and stabilized with prednisolone and azathioprine. In the present case of pulmonary sarcoidosis, the reticular and nodular opacities predominantly distributed in both lower lung fields, and the histological findings obtained by video-assisted thoracoscopic surgery showed a usual interstitial pneumonia-like lesion. These findings may assist in the understanding of the process of development of pulmonary sarcoidosis.     

Primary pulmonary cryptococcosis with endobronchial lesion]

A 19-year old man was admitted to our hospital with fever and productive cough. Chest X-ray films revealed multiple nodular shadows accompanied by a fusing tendency in the right middle field. A diagnosis of bacterial pneumonia was indicated, and the patient was treated with antibiotics. However, because this failed to resolve the nodular shadows, bronchofibroscopy was performed. Examination with a fiberoptic bronchoscope revealed an elevated lesion in the truncus intermedius. Histopathologic examination of a specimen obtained by transbronchial biopsy revealed cryptococcal organisms with the infiltration of monocytes. These findings yielded a diagnosis of pulmonary cryptococcosis with endobronchial lesion. After 6 months of treatment with fluconazole, the multiple nodular shadows disappeared. Comparison of bronchoscopic findings before and after fluconazole treatment clearly demonstrated the efficacy of the therapy.     

Magnetic resonance imaging of inflammatory lung disorders: preliminary studies in children

Magnetic resonance(MR) scans were performed on 14 children with a variety of inflammatory disorders of the lungs. MR successfully identified disease in all of the children and accurately localized the disease within the lung fields. In patients with pneumonia and lung abscesses the acute inflammation was associated with a marked increase in signal intensity on T2 when compared with T1 weighted images. One patient with an inflammatory pseudotumor demonstrated a smaller increase in signal intensity on T2 weighted images than was seen in acute pneumonia. Abscess cavities were well identified along with the wall thickness. In patients with diffuse lung disease (diffuse histoplasmosis, miliary tuberculosis, Letterer-Siwe disease, and allergic alveolitis), each disease appeared different on the MR images. These preliminary studies indicate that magnetic resonance imaging is effective for identifying pulmonary disease in children and may improve the radiologist's ability to differentiate pulmonary disorders.     

A case of splenic inflammatory pseudotumor]

A 74-year-old woman underwent abdominal echography at a local clinic and a splenic mass was found. She was hospitalized for detailed examinations and treatment. Splenectomy was performed to make a definite diagnosis and for treatment because a definitive diagnosis could not be made, despite various examinations. Histopathological examination revealed that the lesion was infiltrated by polyclonal lymphoid cells and contained proliferating spindle-shaped fibroblasts without any atypical cells, so the splenic mass was diagnosed as an inflammatory pseudotumor. Because some cases of inflammatory pseudotumor can be diagnosed from the clinical course and imaging findings, this possibility should also be considered in the differential diagnosis of a splenic mass.     

Rapid recurrence of an inflammatory myofibroblastic tumor in the right ventricular outflow tract

We describe an unusual case of rapid recurrence of a previously excised inflammatory myofibroblastic tumor of the right ventricular outflow tract in a 5-month-old infant. The infant is asymptomatic 18 months after the second surgery. The very rare cardiac involvement, and the early relapse of the inflammatory pseudotumor, to the best of our knowledge, is a unique combination. The inflammatory myofibroblastic tumor, as known as a pseudotumor or plasma cell granuloma is an uncommon reactive lesion with unknown aetiology. It is found most commonly in the lung and a number of visceral organs, such as the spleen, liver, ileum, salivary glands, urinary bladder, larynx or brain or in the retroperitoneum and lymph nodes. To our knowledge only 9 cases have been published of such tumor arising within the heart.     

Inflammatory pseudotumor of the liver diagnosed by needle liver biopsy under ultrasonographic tomography guidance

Inflammatory pseudotumor of the liver is a rare benign lesion, but exploratory laparotomy and a hepatectomy are often performed unnecessarily after various misdiagnoses, including liver abscess, hepatocellular carcinoma, metastatic liver tumor, and cholangiocarcinoma. We present a case of hepatic inflammatory pseudotumor in a 17-year-old man in whom diagnosis was confirmed by liver needle biopsy under ultrasonographic tomography (UST) guidance. He had complained of fever and right hypochondralgia 2 months after being operated for appendicitis. He was admitted to our hospital because of the persistence of these symptoms and the presence of a hepatic mass lesion detected by UST. He had hepatomegaly, with tenderness; leukocytosis and elevated erythrocyte sedimentation rate and C-reactive protein level were noted. UST showed a hypoechoic mass in the liver and pre-contrast computerized tomography (CT) revealed a low-density area with an ill defined margin, which was barely enhanced by the contrast medium. On the basis of the patient's clinical symptoms and the laboratory data and imaging studies, the presence of a liver abscess was suspected and antibiotics were administered. One month after the initiation of the antibiotic therapy, UST demonstrated that the portal vein had dilated serpiginously and penetrated into the mass. As the heterogeneous appearance displayed by post-enhanced CT indicated the need for a differential diagnosis of the hepatic mass lesion to rule out hepatocellular carcinoma, percutaneous needle biopsy was performed, under UST guidance. Histopathological examination demonstrated marked infiltration of plasma cells and fibrosis, findings which were consistent with those of hepatic inflammatory pseudotumor. There was a spontaneous reduction of the hepatic pseudotumor without continuous antibiotics and this reduction was documented on follow-up UST and CT. Received: June 30, 1999 / Accepted: December 17, 1999     

肺炎性假瘤的诊断与手术治疗

目的探讨肺炎性假瘤的诊断与手术治疗。方法回顾52例肺炎性假瘤的检查和手术进行分析。结果1例术后并发支气管胸膜瘘死亡外,余均治愈。结论手术是治疗肺炎性假瘤的有效方法。     

A case of gallbladder cancer associated with a common bile duct neuroma,and a cystic lesion of the liver with histologic findings similar to those of an inflammatory pseudotumor

We report a rare case of gallbladder cancer associated with a common bile duct neuroma, and a cystic liver lesion with histologic findings similar to an inflammatory pseudotumor, in a patient who had had no previous abdominal surgery. The patient was a 62-year-old man whose major complaint was fever. Ultrasonography and a computed tomography scan revealed gallstones, an elevated lesion in the gallbladder, and a cystic liver lesion. Endoscopic retrograde cholangiopancreatography demonstrated stenosis of the common bile duct. Cultures of the cystic fluid and gallbladder bile were positive forStaphylococcus aureus. The patient underwent hepatectomy (inferior S4, S5, and S6), cholecystectomy, resection of the common bile duct, and right hemicolectomy. The resected specimens revealed gallbladder cancer with the microscopic appearance of a papillary adenocarcinoma, and a 12×4.5×3.5 cm cystic liver lesion with a wall 7 mm thick. Histologic studies of the wall of the cystic liver lesion revealed infiltration by histiocytes and plasma cells, and the presence of fibrous connective tissue, which findings are characteristic of inflammatory pseudotumors. A 9×6 mm elevated lesion, with the microscopic appearance of a neuroma, was resected from the common bile duct.     

肺炎性假瘤外科诊治体会

目的探讨肺部炎性假瘤的临床特点、诊断和外科治疗效果。方法对70例外科手术治疗肺部炎性假瘤患者的临床资料进行分析。结果 70例手术治疗,术后病理均证实为炎性假瘤。1例因术后胸液多再次手术,1例切口感染,均痊愈出院。楔形切除患者术后有4例在同肺叶复发,再次行肺叶切除,2例恶变伴远处转移死亡。结论肺部炎性假瘤在临床症状和影像学上与肺癌鉴别困难,对肺部占位性病变宜积极手术治疗,外科手术切除预后良好。手术以局部肺切除和肺叶切除为主。肺部炎性假瘤存在复发、恶变可能,术后须加强随访。     

Inflammatory pseudotumor of the liver successfully treated with nonsteroidal anti-inflammatory drugs: a challenge diagnosis for one not so rare entity

Inflammatory pseudotumor of the liver is a rare, benign lesion characterized by a well-circumscribed mass of chronic inflammatory cell infiltration and proliferating fibrous tissue. Its etiology remains unclear, although inflammatory processes have been proposed. It is often misdiagnosed as a malignant tumor, and the management has been traditionally surgical. We report the case of a 16-year-old boy who was referred from another hospital with a fever of >38 degrees C with rigor and right upper quadrant pain which he had suffered from for 5 days. The ultrasonographic computed tomography and MRI findings were not diagnostic, and we performed a needle biopsy from the lesion that was consistent with inflammatory pseudotumor (of liver, mixed fibrous tissue and chronic inflammatory cell infiltration). The patient was treated with nonsteroidal anti-inflammatory drugs and had an uneventful clinical course. During follow-up, the lesion subsequently shrank to completely vanish 1 year later.