CT-based radiomics for differentiating peripherally located pulmonary sclerosing pneumocytoma from carcinoid.

BACKGROUND: Pulmonary sclerosing pneumocytoma (PSP) and pulmonary carcinoid (PC) are difficult to distinguish based on conventional imaging examinations. In recent years, radiomics has been used to discriminate benign from malignant pulmonary lesions. However, the value of radiomics based on computed tomography (CT) images to differentiate PSP from PC has not been well explored. PURPOSE: We aimed to investigate the feasibility of radiomics in the differentiation between PSP and PC. METHODS: Fifty-three PSP and fifty-five PC were retrospectively enrolled and then were randomly divided into the training and test sets. Univariate and multivariable logistic analyses were carried to select clinical predictor related to differential diagnosis of PSP and PC. A total of 1316 radiomics features were extracted from the unenhanced CT (UECT) and contrast-enhanced CT (CECT) images, respectively. The minimum redundancy maximum relevance and the least absolute shrinkage and selection operator were used to select the most significant radiomics features to construct radiomics models. The clinical predictor and radiomics features were integrated to develop combined models. Two senior radiologists independently categorized each patient into PSP or PC group based on traditional CT method. The performances of clinical, radiomics, and combined models in differentiating PSP from PC were investigated by the receiver operating characteristic (ROC) curve. The diagnostic performance was also compared between the combined models and radiologists. RESULTS: In regard to differentiating PSP from PC, the area under the curves (AUCs) of the clinical, radiomics, and combined models were 0.87, 0.96, and 0.99 in the training set UECT, and were 0.87, 0.97, and 0.98 in the training set CECT, respectively. The AUCs of the clinical, radiomics, and combined models were 0.84, 0.92, and 0.97 in the test set UECT, and were 0.84, 0.93, and 0.98 in the test set CECT, respectively. In regard to the differentiation between PSP and PC, the combined model was comparable to the radiomics model, but outperformed the clinical model and the two radiologists, whether in the test set UECT or CECT. CONCLUSIONS: Radiomics approaches show promise in distinguishing between PSP and PC. Moreover, the integration of clinical predictor (gender) has the potential to enhance the diagnostic performance even further.

18F-Labeled Fluoro-2-Deoxyglucose Positron Emission Tomography and Computed Tomography in a Large Pulmonary Sclerosing Pneumocytoma with Contralateral Lung Metastasis.

Pulmonary sclerosing pneumocytoma is an exceedingly rare neoplasm of the lung. These tumors are usually slow growing with a benign disease course but can easily be mistaken for carcinoid tumors or adenocarcinoma in cytology or histopathology specimens. Rare occurrences of metastases have been reported in the literature making 18F-labeled fluoro-2-deoxyglucose positron emission tomography and computed tomography useful for the evaluation of these tumors.

Huge Pulmonary Sclerosing Pneumocytoma with Endobronchial Invasion: A Case Report with a Literature Review.

Pulmonary sclerosing pneumocytoma (PSP) is a tumor of pneumocytic origin that is classified as a benign neoplasm. To date, aggressive behavior of this tumor has rarely been reported. Here, we describe a case of a 56-year-old woman with a huge, 19-cm PSP that resulted in mediastinal shift and showed microscopic endobronchial invasion and necrosis. The differential diagnosis included malignant mesenchymal tumors, such as solitary fibrous tumor; however, PSP was confirmed based on the characteristic thyroid transcription factor 1 positivity and membranous expression of Ki-67 on immunohistochemical staining of tumor cells.

Large pulmonary sclerosing pneumocytoma in a young female: A rare lung tumor.

We report a case of a 28-year-old female who presented with a solid mass lesion in the right middle lobe (RML). A chest computed tomography (CT) scan showed a 3.5 cm sized round and solid mass between the medial and lateral segment of the RML. The patient underwent a percutaneous lung biopsy with CT scan guidance and pathological examination showed pulmonary sclerosing pneumocytoma. RML lobectomy was performed for definitive treatment. Here, we describe this rare lung disease which presented as a large homogeneous lesion. Pulmonary sclerosing pneumocytoma should be considered in the differential diagnosis of solitary lung tumor, even if the patient is young.

[Analysis of Clinical Characteristics of 35 Cases 
of Pulmonary Sclerosing Pneumocytoma].

BACKGROUND: Pulmonary sclerosing pneumocytoma (PSP) is rare benign lung tumor which usually develops in middle-aged women without typical clinical and imaging findings. PSP consists of two basic cell types (surface cubic epithelial cells and round mesenchymal cells) and four histological types (hemorrhagic, sclerotic, solid and papillary). It grows slowly, but it can metastasize to distant organs. The pathology before surgery is easily misdiagnosed. This study aims to improve clinicians' understanding of PSP by discussing the clinical characteristics of the disease. METHODS: This represents a retrospective study of thirty-five patients diagnosed with pulmonary sclerosing pneumocytoma by pathological examination from January 2011 to December 2019. RESULTS: A total of 35 patients in this study, 12 cases were male and 23 cases were female, the average age is 51 years old. 7 cases were discovered accidentally by physical examination or routine chest computed tomography (CT), and 28 cases were found due to symptoms such as cough, sputum, hemoptysis and chest pain. The imaging changes is mainly featured with isolated or clear circular or round-like single nodule and lump in the lungs. In this group, 12 cases underwent percutaneous lung biopsy, only 7 cases were diagnosed with PSP. A total of 28 patients underwent surgery, 24 cases underwent rapid frozen pathological biopsy, only 5 cases diagnosed with PSP. Postoperative pathological examination results shows that 1 case was diagnosed with keratotic squamous cell carcinoma with partial PSP, and the rest were diagnosed with PSP. The surgical and non-surgical patients were followed up for 1 to 8 years after discharge, and the overall recovery was good. The patients were no recurrence and metastasis on chest CT review. CONCLUSIONS: PSP is a clinically rare benign lung tumor, which is more common in middle-aged women. The clinical manifestations and imaging features are lack of significance. Percutaneous lung puncture pathological examination and intraoperative rapid frozen pathological sections often leads to misdiagnosis. Final diagnosis relies on postoperative pathological work-up for most cases.

Multiple sclerosing hemangioma of the right lung in a 23-year-old female patient: A case report and review of the literature.

Pulmonary sclerosing hemangioma (PSH) is a relatively uncommon benign tumor of the lung, predominantly affecting young and middle-aged women. In the majority of the patients, PSH is incidentally found on physical examination and typically presents as a solitary nodule with smooth borders, as it is generally asymptomatic or lacks typical symptoms. In the present case, a 23-year-old woman was incidentally diagnosed with pulmonary nodules during routine physical examination and reported suffering from intermittent fevers for >2 months. The patient received antituberculosis therapy for 1 year; however, a computed tomography imaging examination revealed that the lesions had progressed. Finally, the patient underwent thoracoscopic lung biopsy followed by histopathological examination and the lesions were diagnosed as multiple sclerosing hemangioma. The aim of the present study was to review the relevant literature in order to improve our understanding of PSH.

Analysis of Clinical Characteristics of 35 Cases of Pulmonary Sclerosing Pneumocytoma / 中国肺癌杂志

BACKGROUND@#Pulmonary sclerosing pneumocytoma (PSP) is rare benign lung tumor which usually develops in middle-aged women without typical clinical and imaging findings. PSP consists of two basic cell types (surface cubic epithelial cells and round mesenchymal cells) and four histological types (hemorrhagic, sclerotic, solid and papillary). It grows slowly, but it can metastasize to distant organs. The pathology before surgery is easily misdiagnosed. This study aims to improve clinicians' understanding of PSP by discussing the clinical characteristics of the disease.@*METHODS@#This represents a retrospective study of thirty-five patients diagnosed with pulmonary sclerosing pneumocytoma by pathological examination from January 2011 to December 2019.@*RESULTS@#A total of 35 patients in this study, 12 cases were male and 23 cases were female, the average age is 51 years old. 7 cases were discovered accidentally by physical examination or routine chest computed tomography (CT), and 28 cases were found due to symptoms such as cough, sputum, hemoptysis and chest pain. The imaging changes is mainly featured with isolated or clear circular or round-like single nodule and lump in the lungs. In this group, 12 cases underwent percutaneous lung biopsy, only 7 cases were diagnosed with PSP. A total of 28 patients underwent surgery, 24 cases underwent rapid frozen pathological biopsy, only 5 cases diagnosed with PSP. Postoperative pathological examination results shows that 1 case was diagnosed with keratotic squamous cell carcinoma with partial PSP, and the rest were diagnosed with PSP. The surgical and non-surgical patients were followed up for 1 to 8 years after discharge, and the overall recovery was good. The patients were no recurrence and metastasis on chest CT review.@*CONCLUSIONS@#PSP is a clinically rare benign lung tumor, which is more common in middle-aged women. The clinical manifestations and imaging features are lack of significance. Percutaneous lung puncture pathological examination and intraoperative rapid frozen pathological sections often leads to misdiagnosis. Final diagnosis relies on postoperative pathological work-up for most cases.

Symptomatic pulmonary sclerosing hemangioma: a rare case of a solitary pulmonary nodule in a woman of advanced age.

BACKGROUND: Pulmonary sclerosing hemangioma (PSH) is a rare tumor that usually develops in middle-aged Asian women. PSH has four histological types (hemorrhagic, sclerotic, solid, and papillary) and often grows slowly in a lower lobe of the lung. Preoperative misdiagnosis frequently occurs because of the absence of specific clinical manifestations and imaging findings. Few reports have described PSH in women of advanced age. Case presentation: A 75-year-old woman presented to our hospital in China with a 5-day history of productive cough and intermittent hemoptysis. Computed tomography indicated bronchiectasis and a large mass in the left inferior lobe of the lung. Treatment of the bronchiectasis provided no symptom relief. The hemoptysis resolved following left lower pulmonary lobectomy, and PSH was pathologically diagnosed following surgery. At the time of this writing (after 6 months of follow-up), the tumor had not recurred, no metastases had been detected, and close follow-up was ongoing. CONCLUSIONS: Both bronchiectasis and PSH can cause hemoptysis. This case demonstrates that PSH should be included as a differential diagnosis of hemoptysis in women of advanced age. For patients with chronic hemoptysis, the diagnosis of PSH should be considered if the therapeutic effect of bronchiectasis is poor.

Pulmonary sclerosing pneumocytoma presenting as slow-growing multiple nodules over a long period.

Pulmonary sclerosing pneumocytoma is an uncommon slow-growing benign tumor that usually occurs in middle-aged women and generally presents as a solitary well-defined nodule. An 18-year-old woman was incidentally detected to have multiple lung nodules on chest radiography that slowly increased in size over a period of 7 years. Computed tomography images showed multiple well-defined nodules surrounded by numerous smaller nodules with a maximum diameter of 3 cm in the left lung. A percutaneous core needle biopsy was performed, but malignancy could not be excluded because of the high proportion of papillary structures. A video-assisted partial wedge resection was performed and the pathologic diagnosis was pulmonary sclerosing pneumocytoma. Pulmonary sclerosing pneumocytoma presenting as multiple lung nodules is a rare but very important condition to include in the differential diagnosis of multiple lung nodules. There is a possibility of misdiagnosis of another type of tumor or malignancy on preoperative biopsy. We should be aware not only of the clinical, radiologic, and pathologic features of pulmonary sclerosing pneumocytoma but also of the potential pitfalls in its diagnosis and management.

18F-FDG PET/CT characteristics of pulmonary sclerosing hemangioma vs. pulmonary hamartoma.

The radiological features of pulmonary sclerosing hemangioma (PSH) and pulmonary hamartoma are poorly specified. Thus, the present study aimed to compare and analyze the characteristics of fluorodeoxyglucose positron emission tomography/computed tomography (18F-FDG PET/CT) in PSH versus pulmonary hamartoma. 18F-FDG PET/CT characteristic findings of 12 patients with PSH and 14 patients with pulmonary hamartoma were retrospectively reviewed. A total of 12 lesions were detected from the 12 patients with PSH, of which 3 masses exhibited calcification. The mean diameter and standardized maximum uptake value (SUVmax) were 1.9±0.7 cm and 2.6±1.0, respectively, and there was no significant correlation between the lesion size and SUVmax (P>0.05). For the 14 patients with pulmonary hamartoma, 14 lesions were found, of which 4 exhibited calcification. The mean diameter and SUVmax were 1.7±0.8 cm and 1.5±0.6, respectively, and there was a significant correlation between the size and SUVmax (r=0.625, r2=0.391, P<0.05). Although there was no significant difference between the size of PSH and pulmonary hamartoma (P>0.05), the SUVmax of PSH was significantly higher than that of pulmonary hamartoma (P<0.05). Moreover, the SUVmax of 1.95 was applied as a cutoff for the diagnosis of PSH, and the resulting sensitivity and specificity for PET/CT to differentiate PSH from pulmonary hamartoma were 83.3 and 78.6%, respectively. Although the morphological features were not specific, PSH showed significantly higher FDG accumulation than pulmonary hamartoma on PET/CT imaging, which may aid the differential diagnosis. Further studies with larger populations are warranted to confirm these study results.

Pulmonary sclerosing pneumocytoma with mediastinal lymph node metastasis.

Pulmonary sclerosing pneumocytoma is a rare tumour with 23 cases reported to have metastasis to lymph nodes, but to date, only 6 had involved mediastinal nodal stations. Our patient was a 40-year-old Asian female with a 2.5 cm nodule in her right lower lobe. Positron-emission tomography suggested no nodal disease; but after resection, the intra-lobular, hilar and subcarinal nodes were all found to be involved. As prognosis of this rare disease is uncertain, we aim to report our findings against available literature and similar cases.

Pulmonary sclerosing hemangioma with a rare symptom: A case report and review of the literature.

Pulmonary sclerosing hemangioma (PSH) is a relatively rare benign tumor. However, as it occurs only rarely, the natural course of the tumor is not well understood. In the present study, a case is presented of a 35-year-old woman who underwent intermittent fevers for more than one year. This case highlighted fever as a rare symptom of PSH. Fever is possibly one of the symptoms of PSH, although it is less likely to occur. Another symptom of the patient was that the tumor grew quickly in two months. The lesion was diagnosed as multiple sclerosing hemangioma of the lung (i.e., PSH), in which papillary, solid and sclerotic patterns appeared. Immunohistochemical evaluation of the lesion revealed positive staining for thyroid transcription factor 1 (TTF1), epithelial membrane antigen (EMA), pancytokeratin (PCK) and cytoskeleton 7 (CK7). In the present case study, the biological activity of PSH was identified to be aggressive. A review of the literature was performed in order to comment further on the clinical and pathological features of this rare disease.

Co-occurrence of bronchial papilloma and pulmonary sclerosing hemangioma in a male.

INTRODUCTION: Bronchial papilloma and pulmonary sclerosing hemangioma (PSH) are rare tumors. The development of PSH combined with bronchial papilloma in lung is extremely rare. We herein presented a case of the co-occurrence of bronchial papilloma and PSH in a male. CASE DESCRIPTION: A 48-year-old man was referred to our department for further treatment of the productive purulent cough and fever. After bronchoscopy, the biopsy of the specimen showed a bronchial papilloma in the lumen of posterior segment of the right upper lobar bronchus. For computed tomography demonstrated a focal lesion with an air bubble in the posterior segment of the right upper lobe, a lobectomy was performed and PSH was diagnosed. DISCUSSION: The report emphasizes the fact that even though some of bronchial papilloma and PSH may have a low prevalence, nonetheless, the low prevalence of both diseases in a male has meant that it has not been possible to explore the depth of association between them. CONCLUSIONS: The paper reports a case of PSH in a male suffering from bronchial papilloma which is the exceptional concurrence of these two extremely infrequent events.

Whole-exome sequencing identifies recurrent AKT1 mutations in sclerosing hemangioma of lung.

Pulmonary sclerosing hemangioma (PSH) is a benign tumor with two cell populations (epithelial and stromal cells), for which genomic profiles remain unknown. We conducted exome sequencing of 44 PSHs and identified recurrent somatic mutations of AKT1 (43.2%) and ß-catenin (4.5%). We used a second subset of 24 PSHs to confirm the high frequency of AKT1 mutations (overall 31/68, 45.6%; p.E17K, 33.8%) and recurrent ß-catenin mutations (overall 3 of 68, 4.4%). Of the PSHs without AKT1 mutations, two exhibited AKT1 copy gain. AKT1 mutations existed in both epithelial and stromal cells. In two separate PSHs from one patient, we observed two different AKT1 mutations, indicating they were not disseminated but independent arising tumors. Because the AKT1 mutations were not found to co-occur with ß-catenin mutations (or any other known driver alterations) in any of the PSHs studied, we speculate that this may be the single-most common driver alteration to develop PSHs. Our study revealed genomic differences between PSHs and lung adenocarcinomas, including a high rate of AKT1 mutation in PSHs. These genomic features of PSH identified in the present study provide clues to understanding the biology of PSH and for differential genomic diagnosis of lung tumors.

Pulmonary sclerosing pneumocytoma mimicking lung cancer: Case report and review of the literature.

Pulmonary sclerosing pneumocytoma (PSP) is a rare benign neoplasm that predominantly affects middle-aged Asian women. PSP is often asymptomatic and demonstrates a solitary pulmonary nodule on radiologic examination. We report a case of PSP initially misdiagnosed as lung cancer because of strong (18)F-fluorodeoxyglucose (FDG) uptake revealed by (18)F-FDG positron emission tomography-computed tomography scan. After surgery, pathology revealed that the tumor cells were immunopositive for epithelial membrane antigen and thyroid transcription factor-1. The patient has been followed up without complication or recurrence.

Sclerosing Pneumocytoma with a Wax-and-Wane Pattern of Growth: A Case Report on Computed Tomography and Magnetic Resonance Imaging Findings and a Literature Review.

Sclerosing pneumocytoma (SP) of the lung is a rare benign neoplasm. Here, we describe an unusual presentation of SP with a wax-and-wane pattern of growth in a 47-year-old woman. Tumor diameter decreased over a 3-year follow-up period and then increased on serial follow-up computed tomography scans. The mass showed high signal intensity on both T1- and T2-weighted chest magnetic resonance imaging (MRI) and early enhancement with a plateau on dynamic MRI. We speculate that intratumoral bleeding and resorption processes accounted for the changes in tumor size.

Pathological features of pulmonary sclerosing hemangioma:an analysis of 120 cases in frozen section / 临床与实验病理学杂志

Purpose To analyze retrospectively the clinicopahological characteristics and immuophenotype in 120 cases of pulmonary selerosing hemangioma ( PSH) . Methods Data of 120 cases of PSH were collected and reviewed, including gross features and frozen diagnosis, histopathological features, morphology in HE and immunohistochemical characteristics of tumor cells. Results PSH pres-ents a well-circumscribed mass with a sense of expansion after cutting, which showed a solid, grey to tan-yellow surface with foci of haemorrhage. Two kinds of tumor cells and four typical patterns with mixed forms, are often shown under light microscope (92/120, 76. 67%). Focal accumulation of histocytes and scattered mast cells were noted in some cases (83/120, 69. 17%). Both surface cells and round cells were TTF-1 and EMA positive. Surface cells expressed SP-A, CK and NapsinA. Round cells expressed vimentin. Conclusion Gross features and typical“two cell types, four patterns” contribute to PSH diagnosis. Meanwhile, foal accumulation of foam histocytes and scattered mast cells show a clue to PSH diagnosis and differential diagnosis.

CT and 18F-FDG PET/CT features of pulmonary sclerosing hemangioma / 中华核医学与分子影像杂志

Objective To evaluate characteristics of CT and 18F-FDG PET/CT in pulmonary sclerosing hemangioma (PSH).Methods A retrospective study involving 12 patients (2 males,10 females;24-80 years old) confirmed as PSH by pathology from May 2012 to July 2014 was investigated.All patients underwent chest CT scan,including enhanced CT;and 5 cases underwent whole-body 18F-FDG PET/CT.All imaging data were collected and analyzed to find out a more effective diagnostic method.Results In 12 PSH patients,9 had single lesion,of which 4 involved left lung and 5 right lung.The rest 3 patients including 1 with two nodules located in the right lower lobe,and 2 with multiple nodules scattered in several lobes.Plain CT showed all lesions had uniformly isodensity,4 with calcification,3 with air meniscus sign.Contrast-enhanced CT examinations showed that the majority lesions were prominent enhancement and few moderate enhancement,all lesions showed vessel marginating sign and 6 with cystic and necrosis area.18F-FDG PET/CT examinations showed that the lesions displayed patchy pattern of mild to moderate uptake with SUVmax 2.2--4.0.Conclusions The characteristic CT features of PSH,especially the contrast-enhanced CT imaging,are important for diagnosis.18F-FDG PET/CT findings are extremely helpful in differentiating PSH from malignant lesions.

Sclerosing Pneumocytoma with a Wax-and-Wane Pattern of Growth: A Case Report on Computed Tomography and Magnetic Resonance Imaging Findings and a Literature Review

Sclerosing pneumocytoma (SP) of the lung is a rare benign neoplasm. Here, we describe an unusual presentation of SP with a wax-and-wane pattern of growth in a 47-year-old woman. Tumor diameter decreased over a 3-year follow-up period and then increased on serial follow-up computed tomography scans. The mass showed high signal intensity on both T1- and T2-weighted chest magnetic resonance imaging (MRI) and early enhancement with a plateau on dynamic MRI. We speculate that intratumoral bleeding and resorption processes accounted for the changes in tumor size.

Multiple pulmonary sclerosing hemangiomas (pneumocytoma) mimicking lung metastasis detected in fluorine-18 fluorodeoxyglucose positron emission tomography/computed tomography.

Pulmonary sclerosing hemangioma (PSH), or the alternative name of "sclerosing pneumocytoma," is a rare benign neoplasm. PSH is often asymptomatic and presents as a solitary or multiple pulmonary nodules on radiologic imaging studies. Few articles have been reported to describe the fluorodeoxyglucose positron emission tomography (FDG PET) findings about PSH. The authors describe an interesting but uncommonly encountered cause of false positive FDG PET scan in the thorax in a 25-year-old woman, a known case of arteriovenous malformation of oral cavity who underwent embolization and presented with incidental detection of bilateral lung nodules. She is asymptomatic and is on follow-up.