Adult ; Aged ; Female ; Humans ; Lung Neoplasms ; diagnosis ; Male ; Middle Aged ; Pulmonary Sclerosing Hemangioma ; diagnosis ; Tomography, X-Ray Computed ; Young Adult

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.
Female ; Humans ; Lung/pathology/radiography ; Magnetic Resonance Imaging/*methods ; Middle Aged ; Pulmonary Sclerosing Hemangioma/*diagnosis/pathology/radiography ; Tomography, X-Ray Computed/*methods

No abstract available.
Aged ; Biomarkers, Tumor/analysis ; Biopsy, Large-Core Needle ; Bone Neoplasms/chemistry/radiotherapy/*secondary ; Female ; Humans ; Immunohistochemistry ; Lung Neoplasms/chemistry/*pathology/surgery ; Pneumonectomy ; Positron-Emission Tomography ; Pulmonary Sclerosing Hemangioma/chemistry/radiotherapy/*secondary/surgery ; Tomography, X-Ray Computed ; Treatment Outcome

Pulmonary sclerosing hemangioma is a relatively rare neoplasm of the lung with polymorphic histologic features of 2 unifying cellular components including surface cuboidal cells and interstitial round cells. Pulmonary sclerosing hemangioma typically occurs in middle aged women with asymptomatic, peripheral, solitary, well-circumscribed lesions. Although it is pathologically benign, it reveals size growing and chest symptom. We here report a case of pulmonary sclerosing hemangioma in a 72-year-old woman. She presented chest discomfort. A chest radiography and a chest computed tomography scan showed growing size from 3.2x3.1 cm to 6.0x5.3 cm in left upper lung during 10 years' follow-up period. Surgical resection of lung revealed a distinct constellation of findings including 2 epithelial cell types, surface cells, and round cells, which form 4 architectural patterns, papillary, sclerotic, solid, and hemorrhagic. She was diagnosed as pulmonary sclerosing hemangioma and chest discomfort disappeared.
Aged ; Epithelial Cells ; Female ; Follow-Up Studies ; Humans ; Lung ; Lung Diseases ; Middle Aged ; Pulmonary Sclerosing Hemangioma ; Thorax

OBJECTIVETo investigate the histogenesis of pulmonary sclerosing hemangioma (PSH).

METHODSTissue microarray and immunohistochemical technique were used to detect the expression of pan-cytokeratin, epithelial membrane antigen(EMA), vimentin, thyroid transcription factor (TTF)-1, napsin A, synaptophysin, chromogranin A, CD56, E-cadherin, β-catenin, CD117, CD68 and transforming growth factor(TGF)-β1 in 49 cases of PSH.

RESULTSImmunohistochemistry revealed that all cuboidal surface cells expressed pan-cytokeratin, EMA, TTF-1 and napsin A. The polygonal cells expressed EMA, TTF-1, napsin A (positive rate 16.3%, 8/49), but not pan-cytokeratin. Both types of cells were negative for synaptophysin, chromogranin A and CD56. Strong positive staining for E-cadherin and β-catenin appeared on the membrane of cuboidal cells in all PSH, with cytoplasm staining for β-catenin as well. The expression levels of these adhesion molecules decreased in the polygonal cells, with the staining localized to the cytoplasm. E-cadherin staining was not detected or was weak. β-catenin staining was not detected on the cell membrane but partially in the cytoplasm. The polygonal cells stained strongly for vimentin, while only a few cuboidal cells were positive. CD117 and CD68 positive inflammatory cells were scattered between the polygonal cells, which was consistent with the distribution of TGF-β1 positive cells.

CONCLUSIONSPSH originates from the primitive respiratory epithelium, and polygonal stromal cells may be derived from epithelial-mesenchymal transformation of the cuboidal cells. TGF-β1 may play an important role in the formation of sclerosing hemangioma.

Adolescent ; Adult ; Aged ; Antigens, CD ; metabolism ; Antigens, Differentiation, Myelomonocytic ; metabolism ; Aspartic Acid Endopeptidases ; metabolism ; Cadherins ; metabolism ; Female ; Humans ; Immunohistochemistry ; Keratins ; metabolism ; Male ; Middle Aged ; Mucin-1 ; metabolism ; Nuclear Proteins ; metabolism ; Pneumonectomy ; Proto-Oncogene Proteins c-kit ; metabolism ; Pulmonary Sclerosing Hemangioma ; metabolism ; pathology ; surgery ; Thyroid Nuclear Factor 1 ; Transcription Factors ; metabolism ; Transforming Growth Factor beta1 ; metabolism ; Vimentin ; metabolism ; Young Adult ; beta Catenin ; metabolism

OBJECTIVETo investigate the clinicopathological features and surgical treatment of pulmonary sclerosing hemangioma (PSH).

METHODSClinic data of PSH patients admitted by surgical resection from January 1985 to December 2010 was analyzed retrospectively. One hundred and sixty-five patients were enrolled in the study. There were 27 male and 138 female patients with a mean age of (48 ± 13) years. Seventy-nine patients were asymptomatic at the time of diagnosis. Eighty-nine tumors arose in the right lung (27 in right upper lobe, 24 in right middle lobe, 34 in right lower lobe, 2 in right upper lobe with invasion of right middle lobe, 1 in right middle lobe with invasion of right lower lobe, and 1 case with multiple lobe lesions), 75 in the left (33 in left upper lobe, 42 in left lower lobe), and 1 in the bilateral. There were huge mass lesions in 2 cases, endobronchial lesions in 2 cases, and multiple lesions in 6 cases. The mean size of the lesion was (2.6 ± 0.9) cm (ranging from 0.9 to 10.0 cm). Forty-eight cases (29.1%) were misdiagnosed as malignancies preoperatively, and 41 cases (24.8%) were misdiagnosed intraoperatively.

RESULTSResections were performed by means of video-assisted thoracoscopy (n = 53) and thoracotomy (n = 112). Surgical resection included pulmonary wedge excision in 61 patients, lobectomy in 89 patients, right bilobectomy in 5 patients, anatomic segmentectomy in 2 patient, enucleation in 6 patients, and synchronous bilateral pulmonary wedge resection in 1 patient. Operative mortality and morbidity occurred in 0 and 2 (4.3%) patients, respectively. Mean follow-up was 34.7 months (ranging from 6 to 62 months). There was no local recurrence or death from PSH.

CONCLUSIONSPSH is a rare benign lung tumor. It is difficult to make accurate diagnosis preoperatively, and sometimes even intraoperative frozen sections can't differentiate it from malignant tumors. Surgical resection is usually indicated for definite diagnosis and treatment. Partial resection is a sufficient treatment in view of uncommon tumor recurrence. Thoracoscopic surgery is recommended for PSH.

Adolescent ; Adult ; Aged ; Female ; Follow-Up Studies ; Humans ; Male ; Middle Aged ; Pneumonectomy ; Pulmonary Sclerosing Hemangioma ; diagnosis ; surgery ; Retrospective Studies ; Young Adult

BACKGROUND: Pulmonary sclerosing hemangioma is a rare thoracic tumor, and pathophysiology or clinical course of this tumor is not yet fully described. Furthermore, there is no consensus on the standard operative procedure for this tumor. MATERIAL AND METHODS: Medical records of thirty-two patients, who underwent surgical resection of pulmonary sclerosing hemangioma from 1996 to 2007, were retrospectively reviewed. RESULTS: Nineteen patients underwent lobectomy and thirteen patients underwent limited resection. Video-assisted thoracoscopic surgery was performed in 9 patients in the latter group. Lymph node dissection was done in 21 patients, and one patient was found to have lymph node metastasis of the tumor. There was no postoperative complication, no early death and no tumor-related late mortality. The mean follow-up duration was 39.3 months (2 months~129 months), and all patients were free of local recurrence and distant metastasis during this period. There was no significant difference in patient's characteristics between the two groups, except that the mean hospital stay was shorter in limited resection group than in lobectomy group (p=0.0031). CONCLUSION: Pulmonary sclerosing hemangioma usually requires surgical resection for both diagnosis and treatment. Limited resection can decrease hospital stay with a surgical outcome comparable to lobectomy, and may be preferred to lobectomy if sufficient resection margin can be achieved.
Consensus ; Follow-Up Studies ; Hemangioma ; Humans ; Length of Stay ; Lung Neoplasms ; Lymph Node Excision ; Lymph Nodes ; Medical Records ; Neoplasm Metastasis ; Postoperative Complications ; Pulmonary Sclerosing Hemangioma ; Recurrence ; Retrospective Studies ; Surgical Procedures, Operative ; Thoracic Surgery, Video-Assisted

Bronchi ; Female ; Follow-Up Studies ; Humans ; Lymph Node Excision ; Lymph Nodes ; pathology ; Lymphatic Metastasis ; Middle Aged ; Pneumonectomy ; Pulmonary Sclerosing Hemangioma ; pathology ; surgery

OBJECTIVETo study the clonality of polygonal cells and surface cuboidal cells in the so-called pulmonary sclerosing hemangioma (PSH).

METHODS17 female surgically resected PSH were found. The polygonal cells and surface cuboidal cells of the 17 PSH cases were microdissected from routine hematoxylin and eosin-stained sections. Genomic DNA was extracted, pretreated through incubation with methylation-sensitive restrictive endonuclease HhaI or HpaII, and amplified by nested polymerase chain reaction for X chromosome-linked androgen receptor (AR) and phosphoglycerate kinase (PGK) genes. The length polymorphism of AR gene was demonstrated by denaturing polyacrylamide gel electrophoresis and silver staining. The PGK gene products were treated with Bst XI and resolved on agarose gel.

RESULTSAmongst the 17 female cases of PSH, 15 samples were successfully amplified for AR and PGK genes. The rates of polymorphism were 53% (8/15) and 27% (4/15) for AR and PGK genes respectively. Polygonal cells and surface cuboidal cells of 10 cases which were suitable for clonality study, showed the same loss of alleles (clonality ratio = 0) or unbalanced methylation pattern (clonality ratio < 0.25).

CONCLUSIONSThe polygonal cells and surface cuboidal cells in PSH demonstrate patterns of monoclonal proliferation, indicating that both represent true neoplastic cells.

Chromosomes, Human, X ; genetics ; DNA, Neoplasm ; genetics ; Female ; Humans ; Male ; Phosphoglycerate Kinase ; genetics ; Polymerase Chain Reaction ; Polymorphism, Genetic ; Pulmonary Sclerosing Hemangioma ; genetics ; pathology ; Receptors, Androgen ; genetics ; X Chromosome Inactivation

Apoproteins ; analysis ; Epithelium ; chemistry ; ultrastructure ; Humans ; Lung ; chemistry ; pathology ; Nuclear Proteins ; analysis ; Pulmonary Sclerosing Hemangioma ; chemistry ; pathology ; Pulmonary Surfactant-Associated Proteins ; analysis ; Secretory Component ; analysis ; Thyroid Nuclear Factor 1 ; Transcription Factors ; analysis