Primary intrapulmonary solitary fibrous tumours
- Authors:
- Published online on: January 15, 2018 https://doi.org/10.3892/ol.2018.7798
- Pages: 3653-3661
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Copyright: © Lin et al. This is an open access article distributed under the terms of Creative Commons Attribution License.
Abstract
Introduction
A solitary fibrous tumour (SFT) is a rare, slow-growing, mesenchymal neoplasm arising from the pleura, which is unrelated to asbestos exposure or cigarette smoking (1) and was initially described in 1931 (2). Over the past 80 years, SFTs have been identified in numerous extrapleural locations, including the nasal cavity (3), breast (4), stomach (5), bronchus (6), head and neck (7), liver (8), oesophagus (9), pelvic (10), pancreas (11), prostate (12), orbit (13), central nervous system (14), parotid gland (15), kidney (16), lung (17), sella turcica (18), heart (19), conus medullaris (20), omentum (21), infratemporal fossa (22), bladder (23), soft tissues of the extremities (24), palatine tonsil (25), diaphragm (26), mesentery (27), lumbar spine (28), thymus (29), oral cavity (30), spermatic cord (31), thyroid (32), rectum (33), salivary glands (34), retroperitoneum (35), larynx (36), trachea (37), adrenal gland (38), female genital tract (39), periosteum of bone (40), mediastinum (41) and hypopharynx (42).
To our knowledge, SFTs are extremely rare in the lung (43). There are few detailed case reports concerning the clinical course, imaging characteristics, diagnosis, treatment and prognosis of primary intrapulmonary SFTs. The main purpose of the present study was to report our experience with the diagnosis and management of primary intrapulmonary SFTs and to systematically review previously reported cases in the literature.
Patients and methods
We retrospectively reviewed the records of 5 patients with primary intrapulmonary SFTs who underwent surgical resection at the Department of Cardiothoracic Surgery, Lishui Center Hospital (Lishui, China), and Clinical College of Yangzhou University, (Yangzhou, China), between January 2000 and January 2016. Age, sex, medical history, clinical presentation, diagnostic methods, intraoperative findings, postoperative complications and outcome were retrieved from hospital records. Meanwhile, relevant studies regarding intrapulmonary SFTs were searched via PubMed from January 1990 to January 2016. The text words and MeSH terms ‘Solitary fibrous tumours’, ‘Intrapulmonary’, and ‘Lung’ were used. Tumour characteristics, clinicopathologic features, therapeutic strategy and survival outcomes were reviewed, and these data were tabulated.
Results
Report of cases
Of the five cases, all were males, with a mean age of 57.6 years (range, 37–68 years). Two patients (nos. 2 and 5) had history of hypertensive disease. One patient (no. 2) had history of diabetes mellitus. One patient (no. 1) had history of bronchial asthma. One patient (no. 3) had history of nodular goitre. The remaining patient (no. 4) had no history of any disease. All patients were asymptomatic, and their tumours were discovered incidentally on routine computed tomography (CT) examination. Contrast-enhanced CT of the chest revealed a lung mass with no calcification or any fatty tissue (Fig. 1). One, one, one and two tumours occurred in the right lower, left upper, right upper and left lower lung, respectively. Three patients (nos. 1–3) were preoperatively diagnosed with spindle cell tumour by CT-guided percutaneous aspiration biopsy. Other examinations, including pulmonary function, echocardiogram, electrocardiogram, coagulation function and blood routine examination, were normal. The serum levels of Na+, K+, Cl−, Ca2+, Mg2+ were all within reference range. Serum carbohydrate antigen, carcino-embryonic antigen, squamous cell antigen and neuron-specific enolase were within normal limits. No evidence of metastasis was found via head magnetic resonance imaging (MRI) and abdominal ultrasound.
One patient underwent tumour enucleation though thoracotomy (no. 1), one patient underwent upper left lobectomy associated with lymph node dissection involving radical dissection of the mediastinum (no. 2), and one patient underwent tumour resection associated with bilateral subtotal thyroidectomy (no. 3). The remaining two patients underwent tumour enucleation via video-assisted thoracoscopic surgery (VATS) (nos. 4 and 5) (Figs. 2 and 3). Data of the clinical features are shown in Table I.
The haematoxylin and eosin (H&E) stain showed a rich variety of spindle cells and amorphous areas of collagen (Fig. 4). Immunohistochemical reactions of the tumour cells were strongly positive for B-cell lymphoma (Bcl)-2 (Fig. 5) and cluster of differentiation (CD)34 (Fig. 6). Tumour cells were positive for vimentin (nos. 4 and 5) (Fig. 7) and CD99 (nos. 2–5) (Fig. 8). The tumour cells were negative for CD117, desmin, smooth muscle actin (SMA), epithelial membrane antigen and S-100. Following an immunohistochemical-analysis-based study, the diagnosis of intrapulmonary SFTs was made.
Figure 7.The immunohistochemical reactions for vimentin protein were positive (magnification, ×100). |
Postoperative haemorrhage occurred in one patient (no. 1), and he received surgical intervention for haemostasis. The average time of intrathoracic drain was 3.6 (1–9) days. The average hospital stay was 15 (4–22) days, and no mortality occurred. All patients were discharged from the hospital following an uneventfully recovery. The mean length of the postoperative follow-up was 37.6 (1–67) months. One patient was lost to follow-up, and four patients were asymptomatic.
Published case study findings
Nineteen articles were identified from the searches of databases (17,43–59) (Table II). They had a total of 45 patients: Twenty-three males and 22 females. The mean age was 59.4 years, ranging from 7 to 81 years. Twelve patients were asymptomatic, and pain and cough were the major symptoms. Five, one, two, four, and seventeen tumours occurred in the right upper lobe, right middle lobe, right lower lobe, left upper lobe and left lower lobe, respectively. Thirty-nine patients underwent surgery, one patient underwent radiotherapy, and one patient underwent radiofrequency ablation. Twenty-two patients were followed up, and the mean length of the postoperative follow-up was 48 (1–168) months. One patient was diagnosed with chest wall metastases. Five patients died (Table III).
Table II.Clinicopathologic features of intrapulmonary solitary fibrous tumours present in the English literature. |
Table III.Characteristics of the primary intrapulmonary solitary fibrous tumors present in the English literature. |
Pathologic and immunohistochemical features
Twenty-eight of 32 cases showed low mitotic counts (0–5/10 HPF), 1 of 32 had a middle mitotic count (0–6/10 HPF), and 3 of 32 showed high mitotic counts (5–10/10 HPF). Immunohistochemical staining analyses were performed in 28 cases. Twenty-four of 28 cases were CD34-positive. Ten of 13 cases were CD99-positive. Fourteen of 15 cases were Bcl-2 positive. Fourteen of 16 cases were vimentin positive. Five of 17 cases were smooth muscle antibody (SMA)-positive. Two of 9 cases were epithelial membrane antigen (EMA) positive. Eight of 9 cases were mib-monoclonal antibody-1 (MIB-1)-positive (Table IV).
Discussion
SFTs are rare, mesenchymal neoplasms initially described in the pleura but have since been discovered in nearly every anatomic location (61). Klemperer and Rabin (62) reported 5 cases of primary pleural neoplasms in 1931 and proposed that SFT was of submesothelial origin. However, in the subsequent decades, on the basis of immunohistochemical analyses and ultrastructural features, it is now recognized that SFTs arise from primitive fibroblast-like cells in connective tissue (61). SFTs, to our knowledge, most often occur in the pleura. They can be rarely found in the lung, central nervous system, kidney and other extrapleural sites. Via searches of databases, a total of 45 patients with intrapulmonary SFTs were found.
Intrapulmonary SFTs are usually found incidentally and may be associated with chest pain and cough. Seventeen patients (5 in our study and 12 in published literature) were asymptomatic and were discovered incidentally. Two patients presented with pain, and two patients presented with cough. Of note, few patients with SFTs present with refractory hypoglycaemia, which is a paraneoplastic syndrome that secretes a prohormone form of insulin-like growth factor-II (IGF-II), referred to as the Doege-Potter syndrome (DPS).
Due to their atypical clinical and radiographic appearance as a common lung tumour, the diagnosis of intrapulmonary SFTs presents unique challenges. Imaging examinations, including chest X-rays, CT and MRI, are used for assessing intrapulmonary SFTs. There are limited data on X-rays and CT imaging features of intrapulmonary SFTs. The available data show that intrapulmonary SFTs are well-defined ovoid or round pulmonary nodules on chest X-rays and CT scanning, but they are non-specific. The PET-CT findings of SFTs have been rarely reported. PET-CT is a useful tool for evaluating the size, regional invasion and distant metastasis of a tumour. Yan et al (63) have reported a malignant SFT with mildly increased FDG uptake. Dong et al (46) have reported a benign SFT with intense FDG uptake. The clinical behaviour of the tumour may be predicted based on PET-CT findings, and FDG uptake degree may be related to the tumour's aggressive behaviour (46). An intrapulmonary SFT may be identified by CT-guided percutaneous aspiration biopsy. Caruso et al (55) operated on a pulmonary mass CT-guided FNA cytology biopsy. The FNA cytologic specimen contained spindle cells. Furthermore, FNA cytology, proper clinical and radiologic findings are helpful in narrowing the diagnostic possibilities and making tentative pathologic diagnoses. However, spindle-shaped cells also appear in fibrosarcoma, leiomyosarcoma, schwannoma and others. Therefore, the diagnosis of SFTs may not be identified without immunohistochemical staining. The differential diagnosis of intrapulmonary SFT includes numerous malignant and benign tumours, including pulmonary adenofibroma, benign neural neoplasms, leiomyoma, leiomyosarcoma, synovial sarcoma, spindle cell thymoma, spindle cell carcinoid tumour, nerve sheath tumour, fibrosarcoma, sarcomatoid carcinoma, and sarcomatoid mesothelioma (54). To our knowledge, positive CD34, CD99, vimentin and Bcl-2 expression are important markers for the diagnosis of an SFT.
Our review of previously reported cases in the literature suggests complete surgical resection is generally accepted as the definitive and effective treatment of choice for intrapulmonary SFTs. Adequate wedge resection, anatomic segmentectomy and lobectomy, according to the location of mass, are common surgical procedures for resection of intrapulmonary tumours. In our study, three patients underwent adequate wedge resection, whereas the other two patients under anatomic lobectomy. In recent decades, with the development of minimally invasive technology, surgical approaches have been radically changed. More and more intrapulmonary tumours can be safely and availably excised via VATS. In our study, two patients underwent VATS. Previous studies have used radiotherapy and radiofrequency ablation for intrapulmonary SFTs. However, these treatments carry a higher risk of death during follow-up. The effectiveness and capability of such treatment has yet to be confirmed.
Due to their deficiency of information and their languages of publication (not English), some studies were not included in our study. Thirty-six of 681 cases and 8 of 88 cases were SFTs of the lung and pleura, respectively, in Choi's et al (64) and Schirosi's et al (65) studies, and these were not included. Gonullu et al (66) reported a case of metastatic breast carcinoma to SFT in the lung, and Strickland et al (67) reported a case of SFT of the uterus presenting with lung metastases. Míguez González et al (68) reported a case of intrapulmonary SFT associated with haemoptysis, and Masuda et al (69) report two cases of intrapulmonary tumours with different growth patterns. However, the articles were not written in English. Radulescu et al (70) reported a case of malignant primary intrapulmonary SFT with haemangiopericytoma-like features.
In conclusion, we retrospectively reviewed the records of 5 patients with primary intrapulmonary SFTs who underwent surgical resection, and we systematically reviewed previously reported cases in the literature. This study has 4 important findings. First, intrapulmonary SFTs can occur in people of all ages and with no sex predominance. The male-to-female ratio was 1.0:0.96. Second, intrapulmonary SFTs are often discovered incidentally. When symptomatic, the common symptoms are chest pain and cough. Third, the left lower lobe was the most common site location. Finally, complete surgical resection is a safe, effective and successful treatment.
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