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An unusual case of malignant melanoma of the pleura in a patient with "bathing-suit" type of giant congenital hairy nevus is presented. The treatment advocated and the outcome are discussed.
We treated a patient with solitary fibrous tumor of the pleura (SFT) whose angiograms demonstrated its feeders and strongly contrasted tumor stain. Although no one has clearly identified the specific features of SFT on angiograms, SFT can be suspected in the clinical setting if a localized chest wall tumor presents with the feeders and strongly contrasted tumor stain shown by angiography.
Thirteen patients with malignant mesothelioma of the pleura have been treated with 24-hour cyclophosphamide infusion (2.5 g/m2) therapy. There were no complete responders. Three patients (23%) had a partial response, and a further three patients who did not fulfil the criteria for objective response, had subjective benefit from therapy with increase in performance status. The overall median survival from the onset of therapy was 6 months (range 2-19 months). High dose cyclophosphamide offers no advantage over standard dose therapy.
BACKGROUND: In nondiabetic women, pregnancy alone rarely causes clinical hypoglycemia. Non-islet cell tumors have recently been shown to be associated with the production of insulin-like growth factor II (IGF-II) and a paraneoplastic syndrome resulting in hypoglycemia. A case report and review of pathophysiologic mechanisms involved is presented. CASE: A 38-year-old multigravida presented suffering from clinical and biochemical hypoglycemia, which was found to be secondary to a mesothelioma of the pleura and ectopic IGF-II production. Tumor resection was performed during the 13th gestational week. The mother became euglycemic immediately after the surgery and remained asymptomatic. Clinical indicators of pregnancy and an ultrasound scan after the surgery were consistent with a normal viable fetus. CONCLUSION: Symptomatic hypoglycemia and other medical conditions occurring during pregnancy require immediate diagnosis and treatment. In addition to the more common causes, documented cases of medical conditions due to paraneoplastic syndromes of ectopic hormone production during pregnancy have been described. This case establishes the non-islet cell tumor with IGF-II-induced hypoglycemia as another such syndrome to be considered when evaluating hypoglycemia in pregnancy.
Solitary fibrous tumors (SFTs) often involve the pleura and also may encompass the peritoneum and nonserosal sites. On occasion SFTs mimics other neoplasms, including desmoplastic mesothelioma. CD-34, initially characterized as a hematopoietic progenitor cell antigen, recently has been identified in a small number of SFTs. Based on this observation, we compared the keratin, vimentin, and CD-34 expression of 19 SFTs and eight desmoplastic mesotheliomas. Fifteen of 19 SFTs (78.9%) expressed CD-34, whereas keratin expression was absent in all SFTs. In contrast, none of the desmoplastic mesotheliomas expressed CD-34 and keratin expression was found in seven of eight (87.5%). Vimentin expression was noted in 18 of 19 SFTs and in seven of eight desmoplastic mesotheliomas. We conclude that CD-34 expression distinguishes SFT from desmoplastic mesothelioma. Additionally, the results of our study support the idea that SFT is not derived from or related to conventional mesothelium.
This report describes three cases of localized fibrous tumor of the pleura (LFTP) with clinical and pathological differences. Case 1 presented with symptoms; the tumor size was 6.5 cm and it was attached to the lung and chest wall; histologically it was composed of spindle cells with pleomorphism, mitoses, hemorrhage and necrosis. Cases 2 and 3 were casual findings and consisted of well-circumscribed tumors of 3 and 8.5 cm, respectively; histologically both showed hypocellularity. All cases exhibited positive stains for vimentin and negative for keratin. These results, added to histological features, suggested a mesenchymal origin. Flow cytometry quantitation of DNA disclosed a diploid pattern in all three cases with a small "near diploid" cell population additionally in Case 1; the S-phase fraction was low in all cases. These findings, that could be considered favourable prognostic signs, and the complete tumoral resection performed in the three tumors, could explain the absence of recurrences after 32, 27 and 19 months, respectively.
Localized fibrous tumors of the pleura are rare. They are often asymptomatic and may have symptoms based on size, bronchial invasion, or hormone production, or a combination of these. Complete resection offers the best chance of cure. However, recurrence is reported in a significant number of patients and can often be treated by repeated resection, albeit with increasing difficulty. We present a case in which delayed recurrence occurred after excision of such a tumor. This required a chest-wall resection and reconstruction after which a second recurrence occurred. Further thoracotomy including a latissimus dorsi free flap procedure was needed for a third-time recurrence.
A case of solitary fibrous tumor of the pleura with the karyotype 46,XY,t(8;12)(p11.2;q24.3) is reported. Although rearrangement of 12q15 approximately 24 is a recurring abnormality in solitary fibrous tumors, rearrangement of chromosome 8 was previously unreported in these tumors.
Primary synovial sarcoma of the pleura is an uncommon entity in the present medical literature. However, the paucity of documented cases may be due to misdiagnoses as a malignant mesothelioma in the past. Early diagnosis with new molecular techniques may improve the outcome of this tumour, which is believed to have a grave prognosis at present.
AIMS: To report a clinicopathological series of cases of solitary fibrous tumor of the pleura from Taiwan. METHODS: Clinical data was collected from a review of medical records and telephone interviewing for follow-up. RESULTS: Eight patients, three men and five women aging from 34 to 71 years, underwent tumor resection and were followed in a period from 7 months to 13.5 years. Six patients underwent standard thoracotomy and two had VATS for tumor excision. Tumors were pathologically benign in seven patients and malignant in one. Patients were all alive with no evidence of tumor recurrence at the time of this report. CONCLUSIONS: One should always consider SFTPs as potentially malignant tumors. Complete resection remains the mainstay of cure. Standard thoracotomy should always be considered when a high suspicion of malignancy is raised, whereas VATS may be a preferred approach for smaller tumors.
This study was performed to add clinical data, to introduce new markers, and to perform syntactic structural analysis on localized fibrous tumors (LFTs) of the pleura. The material comprised clinical data and processed sections obtained from 36 patients. The results achieved from quantitative imaging techniques and syntactic structure analysis were correlated with clinical data, including patients' habits (smoking), asbestos exposure, survival, and tumor recurrence. The disease caused increasing chest pain and dyspnea in 47% of patients. Exposure to asbestos was noted in 13 out of 36 patients, whereas smoking posed no major risk factor. Two patients developed a recurrent tumor after 8 and 42 months, respectively; none of the other patients died of this tumor disease within the follow-up period of maximal 212 months. The cases were clearly discriminated from mesotheliomas by the marker profile. Frequent expression of accessible ligands for endogenous lectins galectins-1 and -3, the expression of the angiogenic macrophage migration inhibitory factor (MIF), and the dense vascularization intimate a functional relationship. The proliferation index (Nv) was computed to be 1.6% in line with the balance of galectin expression. Abnormal p53 was expressed in only 19.4% of the cases. The diagnosis of LFT can be aided by quantitative assessment of vimentin, CD34, MIF, vascularization, and proliferation. Considering the galectin network, differential expression was noted with preference to effectors limiting growth and aggressiveness.
BACKGROUND: Castleman disease of the pleura is unusual, and we present our experience with eight surgically proven cases. METHODS: Between 1980 and 2002, 8 patients (7 women and 1 man; age range, 20 to 53 years; mean, 26.5 years) with surgically proven, pleural Castleman disease (six hyaline vascular type, one plasma cell type, and one mixed type) were encountered. Their clinical, imaging, and surgical findings were reviewed. RESULTS: Five patients were asymptomatic, 1 had dyspnea, 1 had cough, and 1 experienced chest discomfort. Chest radiography showed a well-circumscribed interlobar, cardiophrenic, or paraaortic mass in 6 patients, a massive effusion in 1, and a focal diaphragmatic bulge in 1. Six tumors showed varying degrees of contrast enhancement (10 to 95 HU; mean, 46 HU) on computed tomography. Three cases appeared as well-defined, heterogeneously hyperintense pleural masses on magnetic resonance imaging. The masses varied in size from 3 to 10 cm (mean, 5.2 cm). Five masses greater than 5 cm had prominent pleural arterial blood supply and severe adhesions requiring thoracotomy and resection of nearby structures for radical tumor excision. Blood loss from patients varied between 100 and 850 mL (mean, 620 mL). No tumor recurrence was noted during follow-up (range, 1 to 16 years; mean, 6.5 years). CONCLUSIONS: Pleural Castleman disease predominately affects young women and manifests as a well-circumscribed mass with a varying degree of contrast enhancement on computed tomography and heterogeneity on magnetic resonance imaging. Tumors greater than 5 cm have profuse pleural blood supplies and severe adhesion necessitating open thoracotomy and resection of neighboring structures. Radical resection can produce a satisfactory outcome.
Computed tomography (CT) and plain radiography have been performed on 32 patients with malignant mesothelioma of the pleura, in order to assess the extent of disease and suitability for radical treatment. A comparison of the assessments by the two techniques is reported, with confirmation from either thoracotomy or post-mortem findings available in 16 cases. CT was found to be more sensitive than plain radiology in the detection of pleural effusions, although routine lateral decubitus radiographs has not been performed. The two techniques were equally effective in detecting involvement by tumour of the lateral chest wall, mediastinum and lung, with no false positive assessments by either technique, and few false negative assessments. Disease was shown to be extensive and gross at presentation in mesothelioma, so that the greater sensitivity of CT was of no advantage. Diaphragmatic involvement by tumour was accurately assessed by plain radiology but there were frequent false negative assessments by CT, possibly due to difficulty of interpretation of the curved diaphragm on the cross-sectional CT image. In this study CT offered little advantage over conventional radiology.
Hemangiomatosis involving the pleura is a rare cause of bloody pleural effusion in the neonate. When congestive heart failure and consumption coagulopathy are associated, the prognosis is particularly grave. Corticosteroids and radiation therapy have been reported to shrink hemangiomas. We present such a case in a 2.5-mo-old whose condition did not appear to respond to prednisone or radiation therapy but resolved quickly when cyclophosphamide was given. In this case cyclophosphamide appeared to be life saving.
Malignant mesothelioma of the pleura most commonly occurs in persons with a heavy occupational exposure to asbestos. Some patients, however, have no such history of exposure. Clinical features include initial complaints of nonpleuritic chest pain and dyspnea. The most frequent roentgenographic finding is a unilateral pleural effusion. Thrombocytosis and elevated erythrocyte sedimentation rates are common. Pleural effusions are typically exudates, are often hemorrhagic, and are usually insufficient for diagnosing mesothelioma based on cytology alone. Even pleural biopsy may not produce enough tissue to enable the pathologist to make a firm diagnosis. Thoracotomy and open biopsy will confirm the diagnosis in most cases. Pathologic distinction from metastatic adenocarcinoma may be difficult even after the use of special stains and electron microscopy. Clinical deterioration is primarily attributable to local spread of tumor. Several factors seem to predict prolonged survival: (1) epithelial histologic subtype, (2) performance score, (3) age of the patient at the time of diagnosis, and (4) absence of chest pain. Surgical treatment, chemotherapy, and irradiation, alone or in combination, have been used for malignant mesothelioma. Except for the palliative effect of irradiation, most treatment protocols have not altered the dismal median survival of approximately 11 months seen in untreated patients with malignant mesothelioma.
We present a case of Rosai-Dorfman disease (sinus histiocytosis with massive lymphadenopathy) manifesting primarily as a pleural disease without involvement of the lung parenchyma. The patient is an 81-year-old man who presented with increasing shortness of breath. Radiographic studies revealed a large loculated left-sided pleural effusion and parietal pleural thickening. Video-assisted thoracoscopic biopsy of the pleura showed the characteristic S100-positive histiocytes exhibiting emperipolesis. In this rare case of pleural Rosai-Dorfman disease, the extensive involvement of the pleural lymphatics by the characteristic histiocytes appears to have been responsible for the pleural effusion.
A solitary fibrous tumor of the pleura was studied. Its karyotype was 46,XY,t(6;17) (p11.2;q23),ins(9;12)(q22;q15q24.1),inv(16)(p13.1q24). The rearrangement of 12q13-15 was also described in a subset of hemangiopericytomas of soft tissue and meninges. Because both types of tumors are morphologically and immunophenotypically quite similar, and because some of them share rearrangement of 12q13-15, the possibility of their histogenetical relatedness should be considered.
A 29-year-old woman who underwent evaluation for a 3-month history of left-sided back pain proved to have a left pleural tumor accompanied by a bloody pleural effusion (cytological class II). Three years previously, a chest roentgenogram had been normal. The tumor originated from the parietal pleura at the level of the first three intercostal muscles and was excised completely in continuity with these muscles, including a margin of normal muscle. The tumor measured 15x12 cm and the pathologic diagnosis was benign solitary fibrous tumor; while the tumor invaded the intercostal muscles, no histologically malignant features were present. Long-term follow-up is planned because a possibility of local recurrence exists.