Pulmonary Sclerosing Hemangioma Research Articles

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.

Pulmonary sclerosing hemangioma mimicking bronchial malignancy: A case report

Pulmonary sclerosing hemangioma is a rare benign neoplasm of the lung and predominantly occurs in female patients. Commonly, it presents as asymptomatic solitary pulmonary nodule on chest radiograph. We report a case of pulmonary sclerosing hemangioma who presented with fever and chest x-ray and computed tomography revealed cavitary lesion and mediastinal lymphadenopathy . The chest imaging findings were mimicking bronchial carcinoma. On histopathology and immune-cyto-histochemistry, we found her as having pulomonary sclerosisng hemangioma.The patient is now on follow up without any progression of the disease for last two months.
 Birdem Med J 2019; 9(3): 251-253

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

BackgroundPulmonary 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.ConclusionsBoth 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.

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.

Abstract 3385: Genomic landscapes of pulmonary sclerosing hemangioma

Abstract Pulmonary sclerosing hemangioma (PSH) is a benign tumor that usually presents as a solitary, well-defined mass in the lung. The PSH predominantly affects females with a higher incidence in the Far East. Histologically, the tumor cells in PSH consist of two cell types (cuboidal epithelial and polygonal stromal cells). Immunohistochemical and ultrastructural studies have identified that both cells are derived from undifferentiated respiratory epithelium that is the histologic origin of lung adenocarcinoma as well. Previous studies have shown that PSH and adenocarcinoma in the lung share some immunohistochemical and genetic features. However, as for somatic mutations, whereas many driver genes for lung adenocarcinomas have been identified, there have not been any candidate driver mutations identified in PSHs, except for low frequency mutations in β-catenin and TP53. Frequent somatic mutations identified in lung adenocarcinomas such as KRAS and EGFR have not been detected in PSH, suggesting that genomic alterations of these two lung tumors might be different from each other. Also, there is no evidence of PSH progression to lung cancers. These earlier data indicate that despite the common cellular origin of PSH and lung adenocarcinoma, genetic mechanisms for their development may be different. Based on the established concept that PSH is a true tumor, we hypothesize that it may harbor somatic mutations. In this study, we conducted whole exome sequencing of 44 PSHs and identified recurrent somatic mutations of AKT1 (43.2%) and β-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 β-catenin mutations (overall 3/68, 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. Since the AKT1 mutations were not found to co-occur with β-catenin mutations (or any other known driver alterations) in any of the PSHs studied, we speculate that this may be a 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 discovered in the present study provide clues to understanding the biology of PSH and for differential genomic diagnosis of lung tumors. Citation Format: Seung-Hyun Jung, Min Sung Kim, Sun Shin, Mi-Ryung Han, Yu Jin Cho, So Youn Kim, Sug Hyung Lee, Yeun-Jun Chung. Genomic landscapes of pulmonary sclerosing hemangioma [abstract]. In: Proceedings of the American Association for Cancer Research Annual Meeting 2017; 2017 Apr 1-5; Washington, DC. Philadelphia (PA): AACR; Cancer Res 2017;77(13 Suppl):Abstract nr 3385. doi:10.1158/1538-7445.AM2017-3385

Multi-slice spiral CT in the diagnosis of pulmonary sclerosing hemangioma

Objective To analyze the CT imaging features of pulmonary sclerosing haemangioma(PSH), so as to improve the level of the diagnosis of this disease. Methods Retrospectively analyzed the Multi-slice spiral computed tomography(MSCT) imaging data of 13 cases of PSH with who confirmed by surgical pathology, all cases were performed CT with non-enhanced scaning and enhanced. Results 13 patients' average age was 42 years old, including 10 female, accounted for 76.92%(10/13). All the nodules or masses of the 13 cases were single, long diameter was 0.6-5.0cm, all tumors presented as round or oval shaped, clear boundary.1 case with calcifications in 13 cases, 2 cases with air meniscus sign, and 2 cases with halo sign around.Contrast-enhanced: 10 cases were enhanced homogeneously, 3 cases were enhanced heterogeneously and with vessel marginating sign, 1 case of lymph node was enlargement in mediastinal. Conclusion PSH mainly more shows at middle-aged women, it has relative characteristic of CT imaging within non-enhanced scaning and enhanced.Therefore, a comprehensive analysis of these CT features can help to improve the accuracy of diagnosis. Key words: Pulmonary; Selerosing hemangioma; Tomography, computed

Application value of endobronchial ultrasound-guided transbronchial needle aspiration in the diagnosis and treatment of mediastinal and pulmonary diseases

Aim: This study aims to investigate the value of endobronchial ultrasound-guided transbronchial needle aspiration (EBUS-TBNA) in the diagnosis and treatment of mediastinal and pulmonary diseases. Methods: The clinical data of 52 patients who were hospitalized in the Department of Respiratory and Critical Care Medicine at Jinling Hospital from July 2014 to February 2015 with documented EBUS-TBNA was obtained. Results: Positive results of EBUS-TBNA biopsy were statistically compared to the final diagnosis. A total of 82 specimens from 52 patients were obtained through EBUS-TBNA biopsy. Out of 52, 45 patients had concurrent surgical pathology, including 33 malignant lesions and 12 benign lesions. There were 7 patients with false-negative results, including 1 case of pulmonary sclerosing hemangioma, 4 cases of lung cancer, and 2 cases of tuberculosis. The sensitivity of EBUS-TBNA in this study was 85.2%, accuracy of 85.4%, positive predictive value of 100%, and negative predictive value of 7.7%. There were no serious complications. Conclusion: EBUS-TBNA is a safe and minimally invasive method for the diagnosis of mediastinal and pulmonary diseases. It may also have utility as a therapeutic option during the diagnostic process of some diseases.

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

IntroductionBronchial 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 descriptionA 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.DiscussionThe 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.ConclusionsThe 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 β-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 β-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 β-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.

Preoperative Diagnosis in 46 Cases of Pulmonary Sclerosing Hemangioma.

Preoperative Diagnosis in 46 Cases of Pulmonary Sclerosing Hemangioma.

Pulmonary Hilar Tumor: An Unusual Presentation of Sclerosing Hemangioma.

Pulmonary sclerosing hemangioma is an uncommon benign tumor of the lung; however, on rare occasions it can arise from the pulmonary hilar region. Herein, we report a 53-year-old female patient who presented with a round opacity in the right upper lung field on a radiograph. Chest computed tomography scanning revealed a 3.1 cm mass in the right pulmonary hilum. Thoracoscopic tumor excision was subsequently performed. On pathohistologic study, the tumor was well defined and composed of round stromal cells and surface cells arranged in a papillary, sclerotic, solid, and hemorrhagic pattern. In immunochemical study, the round cells were positive for thyroid transcription factor-1 (TTF-1) and epithelial membrane antigen (EMA) and negative for cytokeratin. The surface cells were positive for TTF-1, EMA, and cytokeratin. Therefore, a final diagnosis of sclerosing hemangioma was confirmed. In conclusion, pulmonary sclerosing hemangioma is uncommon and rare in the pulmonary hilar region. CT scanning is useful to determine its benignity, although imaging features are not specific for a definite differential diagnosis from other pulmonary tumors. Therefore, tissue diagnosis is usually necessary, and pulmonary sclerosing hemangioma should be listed in the differential diagnoses of pulmonary hilar tumors.

リンパ節転移を伴った肺硬化性血管腫の1例及び病理学的検討

リンパ節転移を伴った肺硬化性血管腫の1例及び病理学的検討

Sclerosing Hemangioma of the Lung and Concurrent Sarcoidosis of the Hilar Lymph Nodes

Objective: To present chest radiographs, CT and pathological fndings of pulmonary sclerosing hemangioma with concurrent sarcoidosis. To review the literatures regarding lung cancer and associated sarcoidosis and/or sclerosing hemangioma.Materials and Methods: A 50 year-old female patient was screened for health at human dock at Preventive Medicine Research Center of our institution, and a rounded nodule, measuring about 15 mm in diameter, was found in the left lower lung feld medially. Non-enhanced CT showed a round nodule at the left cardiac border. The patient was sent to a clinic of our institution.Results: The patient was further studied by contrast enhanced CT (CECT), which showed marked increase of CT value to 91.8 HU from 50.1 HU on non-enhanced CT. It was difcult to di?erentiate a benign lesion from malignant lesion. CECT also showed enlarged hilar and mediastinal lymph nodes. Cytology at the time of bronchoscopy revealed Class 3 at the left B5b bronchus. Partial resection of the lingual was carried out by video-assisted thoracoscopic surgery. Biopsy of the left hilar nodes (#10, #11) at the same time revealed sarcoidosis.Conclusion: A rare case of pulmonary screlosing hemangioma and concurrent sarcoidosis was reported with imaging results and pathological proof.

CT and 18F-FDG PET/CT features of pulmonary sclerosing hemangioma

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. Key words: Pulmonary sclerosing hemangioma; Tomography, X-ray computed; Tomography, emission-computed; Deoxyglucose

Pulmonary Sclerosing Haemangioma - Case Report

Introduction: Pulmonary sclerosing haemangioma (PSH) is a benign tumour, also known under the term “pneumocytoma”. It is a rare disease, of low grade of malignancy possibly originating from type II pneumocytes or Clara cells. It is mostly notified in older females and is usually solitary. According to the histological appearance, sclerosing haemangioma could be: papillary, sclerotic, solid and haemorrhagic. Case Report: A 40-year old female patient, smoker, was hospitalised because of the surgical curing mostly asymptomatic lesion in the lower right lobe. The lesion has been radiologically diagnosed a few years before, and regularly controlled by a Computed Tomography (CT). During the last control, the growth of the lesion was noted and the surgical curing indicated. After clinical examinations, by using Video Assisted Thoracoscopy (VATS), the exploration and precised identification of the well vascularised lesion was performed and the atypic resection of the whole tumour was done. The tumour was hystologically established as sclerosing pneumocytoma, containing two types of cells cuboidas and ovoid, with the same immunophenotype. The following controls have pointed out neider recidive nor malignant alternation. Conclusion: Sclerosing haemangioma should be considered in patients with radiologically diagnosed “coin– like” lesions of a solid as well as multiple type. The complete surgical resection is considered the only effective treatment for PSH, and the normal pulmonary tissue should be reserved as possible.

The role of video-assisted thoracoscopic surgery in management of the multiple ground-glass nodules.

We investigated the outcomes of patients with multiple ground-glass nodules (GGNs) to identify the role of video-assisted thoracoscopic surgery (VATS) in diagnosis and treatment. We included patients with multiple GGNs who were qualified for thoracoscopic surgery resection and analyzed the statistics. Fifty-one GGNs were detected in 21 patients. There were 40 pure GGNs and 11 part-solid ones. Around 46 of the 51 lesions were resected via VATS. Four pure GGNs <10 mm and deep in the lung were proceeded with continuous follow-up. One pure GGN measuring 16 mm considered as subnodule and also deep in the lung underwent stereotactic ablative radiotherapy. Resection methods included lobectomy (1), segmentectomy (1), lobectomy + segmentectomy (6), lobectomy + wedge resection (10), and segmentectomy + wedge resection (3). Of the 46 resected lesions, 4 (8.7%) were atypical adenomatous hyperplasia (AAH), 23 (50%) were adenocarcinoma in situ(AIS), 15 (32.7%) were minimally invasive adenocarcinoma (MIA), 2 (4.3%) were invasive adenocarcinoma, one was pulmonary sclerosing hemangioma, and one was nonspecific fibrosis. Intersegmental lymph node metastasis was found in one of the 21 patients. No postoperational complication occurred in any of the patients. Multiple GGNs were generally independent primary lung cancers, mainly including AAH, AIS, MIA, rather than intrapulmonary metastasis. VATS was superior to thoracotomy for less invasive and shorter hospital stay.

肺癌多発肺転移と鑑別困難であった多発硬化性血管腫の1症例

肺癌多発肺転移と鑑別困難であった多発硬化性血管腫の1症例

Pulmonary sclerosing hemangioma with lymph node metastasis: A case report and literature review.

Pulmonary sclerosing hemangioma (SH) is an uncommon benign or low-grade malignant tumor. Multicentric SH and SH with lymph node metastasis have rarely been reported. The present report describes a case of pulmonary SH with lymph node metastasis in a middle-aged female. A nodule was found incidentally in the lower left lung. The patient underwent left lower pulmonary lobectomy and lymph node dissection. Histologically, the nodule demonstrated the characteristic features of SH and one of the resected lymph nodes contained a metastasis of this tumor. Thus, pulmonary SH has the potential to metastasize, a potential not suggested by histological features.

進行直腸癌に併存した多発性肺硬化性血管腫の1例

進行直腸癌に併存した多発性肺硬化性血管腫の1例