Solitary Pulmonary Capillary Hemangioma Research Articles

Solitary pulmonary capillary hemangioma – An underrecognized rare tumor. Report of 32 new cases with literature review

ObjectiveTo explore the clinical, imaging, pathologic characteristics and differential diagnosis of solitary pulmonary capillary hemangioma (SPCH). MethodsThirty two cases of SPCH were collected and studied, with literature review. ResultsThis study included 13 males and 19 females, with a male-to-female ratio of 1:1.5. The age ranged from 26 to 70 years (median age of 43 years). All patients were asymptomatic at presentation. Lung nodules were incidentally discovered during chest computed tomography (CT). Imaging features included 21 cases with partial solid nodules (PSN), 7 cases with ground-glass nodules (GGN), and 4 cases with solid nodules (SN). Eleven cases were in the left lung lower basal segment, 11 cases in the right lung lower basal segment, 6 cases in the right lung upper anterior segment, and 4 cases in the right lung middle lateral segment. The lower basal segments of the lungs were involved in 22 (11 in each lung) cases (22/32, 68 %). The tumors ranged from 6 to 18 mm (average 10 mm). Macroscopically, 16 cases had clear boundaries, while 16 cases had unclear boundaries, and gray-red or dark brown on cut surfaces. Intraoperative frozen section was performed in 27 cases, with diagnosis of SPCH in 12 and pneumonia or inflammatory lesion in 15. Microscopically, the nodules were composed of densely proliferated and dilated capillaries. The capillary walls were lined with a single layer of flat endothelial cells, without atypical features. Collapsed alveolar septa were replaced by a large number of capillaries. All cases showed proliferating capillaries spreading into the walls of small veins/arteries and bronchi, with 3 cases showing dilated capillaries protruding into the bronchiolar lumens as polyp-like structures. Twenty-six cases (26/32, 81 %) showed proliferating capillaries passed over the interlobular septa. Twenty-six cases (26/32, 81 %) showed irregular intimal thickening of small muscular arteries in the peripheral areas of the lesions, with the thickened intima being cellular or fibrous. In twenty-seven cases (27/32, 84 %) the lesions were located in the subpleura, with 6 cases involving the pleura. ConclusionSPCH is a rare benign lung tumor that mostly occurs in the lung lower basal segments with predominance in females. It usually appears as a ground-glass nodule on CT and is very similar to early-stage lung cancer. Accurate diagnosis requires collaboration of radiologists, surgeons, and pathologists. SPCH should be regarded as an important differential diagnosis of small incidental lung nodules.

Solitary pulmonary capillary hemangioma: a clinicopathological analysis of 5 cases

Solitary pulmonary capillary hemangioma: a clinicopathological analysis of 5 cases

A Case of Solitary Pulmonary Capillary Hemangioma

Solitary pulmonary capillary hemangioma (SPCH) is a very rare disease that has been reported to be difficult to diagnose preoperatively because the unenhanced computed tomography (CT) findings resemble adenocarcinoma in appearance. On the other hand, the findings of contrast-enhanced (CE) CT have not been reported as far as we could find in PubMed. In this article, we report a case of SPCH with its chest radiograph and dynamic CECT. Dynamic CECT could depict the pathologic features of capillary proliferation, especially dense in the central component of the lesion, edema, and congestion of the alveolar interstitium, and intra-alveolar edema in the periphery of the lesion. Dynamic CECT might be helpful for differentiating SPCH from other solitary pulmonary nodules.

Solitary Pulmonary Capillary Hemangioma: CT and PET-CT Features with Clinicopathologic Correlation.

The aim of this study was to evaluate the CT and PET-CT features of solitary pulmonary capillary hemangioma (SPCH) with clinicopathologic correlations. This retrospective study included 17 patients with histologically proven SPCH from four tertiary institutions. The clinical, pathological and imaging findings of SPCH were reviewed. The CT features assessed included lesion location, size, density, contour, margin, enhancement, presence of air bronchogram, perivascular lucency and pleural retraction, and 18F-fluorodeoxyglucose uptake on PET-CT. Changes in the size during the follow-up period were also evaluated. Imaging features were correlated with the clinicopathologic findings. The mean age of the patients was 47 years (range 30-60 years). All SPCHs were incidentally detected during screening CT examinations (n = 13, 76%) or during cancer work-up (n = 4, 24%). Most SPCHs appeared as part-solid nodules (n = 15, 88%), the remaining appeared as a pure ground-glass nodule or a pure solid nodule, respectively. Most had smooth contours (n = 16, 94%), while one had a lobulated contour. Nine SPCHs (53%) showed ill-defined margins. Air bronchogram was present in ten (59%) SPCHs, and perivascular lucency in two (12%). All SPCHs exhibited hypoattenuation on contrast-enhanced CT and hypometabolism on PET-CT. During the follow-up period (mean 14.8 ± 17.7 months), the lesions showed no change in size or density in ten SPCHs (59%), decreased or fluctuation in size and density in three (18%). SPCH is often incidentally detected in young and middle-aged adults, commonly as an ill-defined part-solid nodule that may accompany air bronchogram, perivascular lucency, and fluctuation in size or density on CT and hypometabolism on PET-CT.

Solitary pulmonary capillary hemangioma of adult: a series of 10 cases

Objective: To examine the clinical features, diagnostic and therapeutic strategy of solitary pulmonary capillary hemangioma (SPCH). Methods: The data of 10 SPCH cases who underwent surgical operations from June 2017 to June 2020 in Shanghai Pulmonary Hospital, Tongji University were retrospectively reviewed. There were 4 males and 6 females, aged (49.8±13.6) years (range: 26 to 66 years). The clinical manifestations, imaging manifestations, treatment and pathological diagnosis were analyzed. Results: All patients were asymptomatic, and all nodules were detected by CT. The size of nodule was (14.9±5.8) mm (range: 8 to 30 mm). Seven of 10 cases showed the mixed ground-glass nodule appearance and 2 cases showed solid nodule and 1 case showed cystic solid nodule appearance in CT findings. The growth speed was very slow. The follow-up time was 4.5(21.5) months before surgery. Histologically, SPCH manifested as a solitary lesion composed of densely proliferating and dilated capillaries without cytologic atypia within the alveolar septa. Immunohistochemically, capillaries of SPCH uniformly expressed endothelial markers, such as CD31, CD34. The patients were followed up for 15.0(22.0) months after surgery and all recovered well. Conclusions: SPCH is probably an unrecognized benign capillary proliferative disease. SPCH lesions mimic early lung cancer on CT as mixed ground-glass nodule, may be misdiagnosed as other nonspecific benign lesions. With careful histologic examination, SPCH can be successfully diagnosed using CD34 or CD31 immunohistochemistry staining.

Solitary pulmonary capillary hemangioma: Clinicopathologic and radiologic characteristics of nine surgically resected cases

BackgroundSolitary pulmonary capillary hemangioma (SPCH) is an extraordinarily rare capillary derived mesenchymal neoplasm. Although routine morphology and immunohistochemistry are adequate for the diagnosis of classical SPCH in surgical specimens, true gross appearance identification of most tumor themselves and diagnosis for some exceptional cases are still very difficult. Furthermore, preoperative imaging and frozen diagnosis remain a challenge. MethodsWe reported nine original cases of solitary pulmonary capillary hemangioma and summarized the clinical characteristics of twenty-one reported lesions. Imaging materials were reviewed by the image experts of our hospital. Quick hematoxylin-eosin stained intraoperative frozen sections and routine histological diagnosis were re-confirmed by 3 specialist pathologists with at least 10 years of diagnostic experience in our department. Immunohistochemistry analysis was performed on formalin fixed archival tissue. The surgical methods, following up information and prognosis were retrospectively analyzed. ResultsIn imaging, three tumors showed solid nodules, three cases displayed mix ground glass nodules, two nodules were pure ground glass density, and one case was a cystic-solid mass. Macroscopically, solitary pulmonary capillary hemangiomas were ill-defined soft hemorrhagic lesion with pale yellow or dark brownish cut surface. Two cases had a clear boundary and seven lesions were poorly demarcated. Typical morphological features were densely proliferating thin-walled capillaries composing of single layer of flatten or cuboidal endothelial cells within the thickened alveolar septa. One case was mistaken for a histiocytogenic lesion during freezing. The cystic-solid lesion showed a hyperplasia capillary network along the submucosal interstitium of bronchioles. Immunohistochemically, tumor endothelial cells were positive for ERG, Fli-1, CD31, CD34 and Vimentin and negative for CK, α-SMA, TTF-1, HMB45, S-100 and CD68. Lobectomy was performed on seven cases, wedge resection and segmentectomy were proceeded in two patients respectively. Follow up information showed no evidence of complication or recurrence. ConclusionsSolitary pulmonary capillary hemangioma has special imaging and various histological features and must be distinguished from small benign lung lesions and preinvasive cancer. Although the prognosis of this tumor is good after surgical resection, the correct interpretation of the gross appearance and radiographic findings are still important. Choosing appropriate resection mode depends on accurate evaluation preoperative and intraoperative.

Solitary pulmonary capillary hemangioma: An under-recognized pulmonary lesion mimicking early lung cancer on computed tomography images

ObjectivesSolitary pulmonary capillary hemangioma (SPCH) is a rare lung tumor typically presenting as pure or part-solid ground-glass nodules (GGNs) on computed tomography (CT), which clinically resembles early lung cancer. Materials and MethodsIn addition to 10 recently diagnosed patients with SPCH, 71 benign lung nodules that were surgically resected between January 2013 and December 2017 were reviewed by thoracic pathologists to identify any previously unrecognized SPCH cases. Finally, 6 tumors (8.5%; 6/71) were determined to be SPCH. Elastic fiber stain (orcein stain) as well as immunohistochemistry for cytokeratin, CD31, and thyroid transcription factor 1 were performed for confirmation. Clinical and radiological data were analyzed. ResultsAll 16 SPCH lesions were unrecognized or misdiagnosed by general pathologists. The SPCH ranged in size from 3 to 15 mm, and a predominance among women (68.8%; 11/16) was noted. Pathologically, all SPCH lesions were nodular with a higher vascular density than the adjacent lung tissue. Decreased cytokeratin staining and disrupted elastic fibers were clearly observed in all SPCH lesions. ConclusionsSPCH lesions mimic early lung cancer on CT; they are largely unrecognized by general pathologists and are diagnosed as other nonspecific benign lesions. With careful histologic examination, SPCH can be successfully diagnosed using cytokeratin/CD31 immunohistochemistry and elastic fiber staining.

Solitary pulmonary capillary hemangioma presents as ground glass opacity on computed tomography indicating adenocarcinoma in situ/atypical adenomatous hyperplasia: A case report.

Solitary pulmonary capillary hemangioma (SPCH) is a rare type of benign lung tumor, which must be distinguished from early lung cancer and precancerous lesions of the lung that manifest in a similar way upon imaging. The current study describes a case of SPCH and a review of the literature is performed. The patient was a 40-year-old Chinese woman who was referred to the Liaoning Cancer Hospital and Institute (Shenyang, China) in October, 2015 with a cough without obvious inducement. Computed tomography (CT) demonstrated pure ground glass opacity (GGO) in the right upper lung. Following systemic anti-inflammatory therapy over 6 months, the lesion did not exhibit any change on CT and was suspected to be an adenocarcinoma in situ (AIS) or atypical adenomatous hyperplasia (AAH). Video-assisted thoracic surgery wedge resection was subsequently performed. Frozen section diagnosis revealed a benign tumor without atypical epithelial cells. Subsequent to surgery, paraffin sections demonstrated that the tumor contained narrow alveolar cavities, thickened alveolar septa and a clear boundary separating it from healthy lung tissue. Furthermore, the proliferation lumens in the alveolar septa were lined with a single layer of flat cells. Immunohistochemical staining revealed that the flat cells were positive for cluster of differentiation CD31 and CD34, and negative for thyroid transcription factor-1 and cytokeratin. The proliferation of capillary vessels lead to the thickened alveolar septa and the tumor was diagnosed as SPCH. When imaging examination demonstrates a GGO in lung, SPCH must be considered in the differential diagnosis of AIS/AAH. As the prognosis of these lesions is entirely different, a pathological examination must be conducted to ensure a correct diagnosis.

画像上高分化型肺腺癌と鑑別困難であった孤立性肺毛細血管腫の1切除例

画像上高分化型肺腺癌と鑑別困難であった孤立性肺毛細血管腫の1切除例

Pulmonary capillary hemangioma diagnosed by needle core biopsy: Case report and review of the literature

Pulmonary hemangiomas are extremely rare. Here we report a case of a 42year old healthy woman with an incidental nodule identified by computed tomography (CT). In consideration of her smoking history and the appearance of irregular borders of the nodule on CT imaging, despite lack of change after a 9-month follow-up, a CT-guided percutaneous needle core biopsy was performed to rule out lung cancer. A diagnosis of solitary pulmonary capillary hemangioma was made histopathologically. The feasibility of using needle core biopsy to diagnose pulmonary hemangiomas is discussed with the clinical, radiologic and pathological features of this case and a review of literature.

Solitary Pulmonary Capillary Hemangioma of Adult Cases: Clinicopathologic Characteristics as an Unrecognized Entity.

Solitary pulmonary capillary hemangioma (SPCH) is a rare disease, first described about autopsy cases in 2000 and about surgically resected cases in 2006. To date, only 9 surgically resected cases have been published in English. Here, we report 7 original cases with surgery (median age, 54 y; 4 females, 2 never-smokers). All patients were asymptomatic, and all nodules were detected by computed tomography (CT). The median (range) size of nodule was 11 (8 to 16) mm. Six of 7 cases showed the part-solid nodule appearance and 1 showed pure ground-glass nodule appearance in CT findings. The growth speed was very slow. No abnormal uptake of fluorine-18 fluorodeoxyglucose was observed in systemic positron-emission CT in all 3 cases we examined. No patients died from SPCH. Histologically, SPCH manifested as a solitary lesion composed of densely proliferating and dilated capillaries without cytologic atypia within the alveolar septa. In addition, capillaries of SPCH spread into the vascular lumen and involved the walls of bronchioles with protrusion into the lumen. Immunohistochemically, capillaries of SPCH uniformly expressed endothelial markers, such as CD31, CD34, and Factor VIII; and α-smooth muscle actin positive cells were also observed. To be accurately diagnosed, especially in intraoperative frozen sections, SPCH should be conceived as an entity that presents as a solitary nodule in CT. We propose that SPCH is an unrecognized benign capillary proliferative disease.

A Resected Case of Solitary Pulmonary Capillary Hemangioma Showing Pure Ground Glass Opacity

A 53-year-old Japanese woman was referred to our hospital for pure ground glass opacity (GGO) in the left upper lung on computed tomography. 18F-fluorodeoxyglucose positron emission tomography revealed no abnormal uptake in the lesion. After 4 months of follow-up, we strongly suspected the lesion to be a bronchioloalveolar carcinoma and decided to perform a surgery for diagnosis. Planning to perform lingual segmentectomy by video-assisted thoracic surgery (VATS) with mini thoracotomy, we finally performed the left upper lobectomy to guarantee the optimal margin by VATS, because the lesion was not identified by palpation. Final pathological diagnosis was a solitary pulmonary capillary hemangioma (SPCH). We herein report a resected case of SPCH showing pure GGO and review all reported SPCH cases.

Case of solitary pulmonary capillary hemangioma: Pathological features based on frozen section analysis

Solitary pulmonary capillary hemangioma (SPCH) is a rare benign lung tumor that must be distinguished from small and early lung cancers. Here, we report a case of SPCH for which we performed frozen section diagnosis. The patient was a 55-year-old Japanese woman. Five years before the operation, mixed ground-glass opacity was detected by computed tomography in the left posterior basal segment of the lower lobe (S10). Because the interior tumor density of the ground-glass opacity increased slightly, video-assisted thoracic surgery wedge resection was performed. Frozen section diagnosis revealed a benign tumor without proliferation of atypical epithelial cells. The tumor had narrow alveolar lumens, thickened alveolar septa and a clear boundary separating it from normal lung tissue. The proliferated lumens varied in size and were lined with single layers of flat cells. After the operation, immunohistochemical staining of a paraffin section revealed that the thickened alveolar septa resulted from the proliferation of capillary vessels, the flat cells of which were positive for CD31 and CD34 and negative for podoplanin; the tumor was diagnosed as SPCH. Here, we discuss the pathological features of SPCH on frozen sections with reference to this case and review previous related reports.