Pneumonectomy for an endobronchial carcinosarcoma with long-term survival: a case report

Introduction

Carcinosarcoma is a malignant tumor with a poor prognosis (1). It has a predilection for middle-aged and older men, especially in the smoking population. It consists of a mixture of carcinoma and sarcoma, which contain differentiated mesenchymal components such as malignant cartilage, bone, and skeletal muscle. In this report, we present a rare case of endobronchial carcinosarcoma, which appeared as a gloved finger sign, coarse calcifications, and intense uptake of ¹⁸F-fluorodeoxyglucose (¹⁸F-FDG). Importantly, the patient has remained disease-free for 7 years following pneumonectomy without receiving neoadjuvant or adjuvant therapy. We present this article in accordance with the CARE reporting checklist (available at https://acr.amegroups.com/article/view/10.21037/acr-2025-227/rc).

Case presentation

A 48-year-old man (de-identified in this article) presented to The First Affiliated Hospital of Guangzhou Medical University with a 30-year history of chronic productive cough and a 1-month history of shortness of breath. He reported smoking one pack per day for several years. On admission, his physical examination was unremarkable. Laboratory tests revealed an activated partial thromboplastin time (APTT) of 47.2 seconds (normal <42.8 seconds), prothrombin time ratio (PTR) of 1.41 (normal <1.15), and a hypersensitive C-reactive protein (hs-CRP) level of 4.13 mg/L (normal <3.0 mg/L). His neuron-specific enolase (NSE) was elevated at 22.14 ng/mL (normal <16.3 ng/mL), and his partial pressure of oxygen was reduced to 67.5 mmHg (normal >83.0 mmHg).

Computed tomography (CT) revealed a branching tubular mass in the left upper lobe measuring 8.6 cm × 5.7 cm, obstructing the bronchus and displaying a gloved finger sign (Figure 1A,1B). The mass contained multiple coarse calcifications (Figure 1B) and demonstrated moderate enhancement after contrast administration (Figure 1C). A ¹⁸F-FDG positron emission tomography/computed tomography (PET/CT) showed intense uptake of FDG by the tumor [maximum standardized uptake value (SUVmax) =14.7] (Figure 1D). The mass had a clear demarcation from the surrounding lung parenchyma (Figure 1E). No lymphoid or distal metastasis was detected on PET/CT and brain magnetic resonance imaging. Five months later (2 weeks prior to pneumonectomy), CT showed left lung atelectasis and pleural effusion (Figure 2). Eight months post-pneumonectomy, CT indicated a complete excision without tumor recurrence or metastasis (Figure 3).

Figure 1 Images of a 48-year-old male with cough and shortness of breath, obtained 5 months prior to surgery. (A) Coronal MPR showing the mass occupying the bronchus (arrow). (B) Sagittal MPR showing a branching tubular mass in the left upper lobe, resembling a gloved finger sign (arrow), with multiple coarse calcifications (arrowheads). (C) Contrast enhanced CT showing moderate enhancement of the lesion (arrows), with CT values of 48 HU in the pre-contrast phase, 94 HU in the arterial phase, and 78 HU in the venous phase. (D) PET/CT showing intense uptake of ¹⁸F-FDG by the tumor (SUVmax =14.7). (E) The mass (arrowhead) being clearly demarcated from the surrounding lung tissue. CT, computed tomography; FDG, fluorodeoxyglucose; HU, Hounsfield unit; MPR, multiplanar reconstruction; PET/CT, positron emission tomography/computed tomography; SUVmax, maximum standardized uptake value.

Figure 2 MPR obtained 2 weeks prior to the pneumonectomy. (A) Coronal and (B) sagittal MPR showing left lung atelectasis due to occlusion of left main bronchus by the tumor (arrow) and pleural effusion (arrowhead). MPR, multiplanar reconstruction.

Figure 3 Postoperative imaging. Contrast-enhanced coronal MPR showing the post-pneumonectomy changes (arrow) without evidence of tumor recurrence or metastasis 8 months after the pneumonectomy. MPR, multiplanar reconstruction.

A bronchoscopic biopsy performed before pneumonectomy revealed grayish-yellow shredded tissue (Figure 4A). Pathology strongly suggested carcinosarcoma. The patient subsequently underwent pneumonectomy at another hospital 5 months later, and pathology confirmed carcinosarcoma. The mass appeared grayish-white, tough, and partially hard on the cut surface (Figure S1). Microscopic analysis showed a mix of squamous cell carcinoma (80%) (Figure 4B) and sarcoma (20%), primarily composed of osteosarcoma (Figure 4C) and chondrosarcoma. Immunohistochemical staining showed that the squamous carcinoma component was positive for CK, CK5/6, and P40, while the sarcoma component stained positive for Vim and S-100. The Ki-67 proliferative index was approximately 60%. The resected specimen showed negative surgical margins, no lymphovascular or perineural invasion, and no metastasis in eight examined hilar and mediastinal lymph nodes (0/8), consistent with an R0 resection.

Figure 4 Bronchoscopy and biopsy pathology. (A) Bronchoscopy showing piles of greyish-yellow shredded mass occluding the entire lumen. (B) Nested distribution (arrow) of squamous carcinoma cells (hematoxylin and eosin, 400×). (C) Osteosarcoma components (arrow) appearing in the tumor (hematoxylin and eosin, 400×).

We recently conducted a follow-up call with the patient, who reported that he has been undergoing annual check-ups at his local hospital, with no evidence of tumor recurrence or metastasis over the past 7 years. No neoadjuvant or adjuvant therapy was administered from the onset till now because of the patient’s unwillingness.

Diagnostic and therapeutic timeline is shown in the Figure S2.

All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Declaration of Helsinki and its subsequent amendments. Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Discussion

Carcinosarcoma is a bidirectional malignancy containing both carcinomatous and sarcomatous components. Squamous cell carcinoma is the most common epithelial component of lung carcinosarcomas (2,3), especially in centrally located tumors within the airway (4). Sarcomatous components include rhabdomyosarcoma, chondrosarcoma, osteosarcoma, etc. This malignancy primarily affects middle-aged and older men (3), particularly those with a significant history of smoking (4,5), with a male-to-female incidence ratio of 7.25:1 (3).

In this case, the tumor showed a typical gloved finger sign, which is commonly seen in the setting of bronchial obstruction and consequent bronchial dilatation and mucus plugging (6-8). The sign can be identified in a variety of congenital and acquired diseases such as segmental bronchial atresia, cystic fibrosis, allergic bronchopulmonary aspergillosis (ABPA), broncholithiasis, and foreign body aspiration (9). However, they exhibit no enhancement. Benign tumor (bronchial hamartoma, lipoma, and papillomatosis) (9) can also present with a gloved finger sign, but usually shows low ¹⁸F-FDG uptake. For other malignancies (bronchogenic carcinoma, carcinoid tumor, and metastases) (9), the diagnosis should be based on clinical and pathological findings.

Previous studies report that carcinosarcomas of the lung usually present as isolated masses in the upper lobe with an average diameter of 7 cm (3), consistent with this case. Calcifications within the mass are suggestive of a chondrosarcoma or osteosarcoma matrix, while intense ¹⁸F-FDG uptake may indicate sarcomatous components, aligning with prior reports (7,8).

Bronchoscopy can be used to initially rule out diseases that also present with the gloved finger sign such as ABPA, asthma, etc. However, diagnosis is difficult merely based on the small biopsy specimens by bronchoscopy and usually requires excision of the mass (5).

The prognosis for patients with lung carcinosarcoma is generally poor, with a 5-year survival rate of 21.3% due to the tumor’s high potential for early metastasis or local recurrence (3,5). Prognosis largely depends on the tumor location, composition, status of metastasis, and treatment strategy (2,4,10).

There are two clinical subtypes of lung cancer sarcomas: peripheral invasive type and central endobronchial type (10). Previous studies showed the 1-year survival rate of peripheral and endobronchial type was 6.9% and 35.7%, respectively (10). The central endobronchial subtype can cause earlier symptoms from airway obstruction, sometimes allowing diagnosis at a surgically resectable stage (6,10). The gloved finger sign is also associated with the central endobronchial type, resulting from either tumor extension into adjacent bronchi or obstruction of mucus drainage from peripheral airways. Tumors originating in the peripheral distal airways are less likely to cause mucus obstruction and, consequently, are less likely to exhibit this sign.

Surgical resection is the main treatment method since carcinosarcoma is insensitive to chemotherapy and radiotherapy (4). If the tumor is at an advanced stage, combined chemotherapy or radiation therapy may serve as palliative measures (5). Although preoperative imaging 2 weeks before surgery revealed progression, the patient achieved 7-year recurrence-free survival. This may be related to the successful R0 resection.

To date, no standardized treatment protocol has been established for pulmonary carcinosarcoma (11). A recent review confirmed that adjuvant therapy significantly improves survival compared with surgery alone (P=0.0055) (11). Nevertheless, our case achieved an exceptionally rare 7-year recurrence-free survival with surgery alone, far exceeding the longest survival (35 months) reported in that series. Furthermore, a 2024 case series reported that patients with locally advanced disease received neoadjuvant immunotherapy combined with chemotherapy, followed by R0 resection and adjuvant therapy (1). Despite this intensive multimodal regimen, disease-free survival did not exceed 3 years (1). Although neoadjuvant and adjuvant therapies are generally recommended, our case demonstrates that prolonged disease-free survival following R0 resection alone may be possible in some patients (younger age, early diagnosis, absence of distant metastasis, specific genetic mutations, etc.), offering cautious optimism to patients who cannot undergo perioperative systemic therapy. Nevertheless, this case remains an exception rather than the rule, and treatment decisions must be individualized within a multidisciplinary framework.

This report is limited by its single-patient design. The observed outcome may not be generalizable. And genomic profiling (e.g., PD-L1, tumor mutational burden, or driver mutation analysis) was not performed, limiting molecular insights into this favorable outcome. Moreover, multicenter case series are needed to validate the prognostic significance of R0 resection in the absence of neoadjuvant and adjuvant therapy.

Conclusions

Endobronchial carcinosarcoma is a rare malignancy, with the central endobronchial subtype often displaying a characteristic gloved finger sign on CT. Coarse calcifications and high FDG uptake within the mass may indicate sarcomatous components. Despite the typically poor prognosis associated with endobronchial carcinosarcoma, complete surgical resection (R0) may achieve a favorable prognosis even in the absence of neoadjuvant or adjuvant therapy. This provides cautious optimism for patients unable to undergo perioperative systemic therapy. Overall, management must be individualized.

Acknowledgments

None.

Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://acr.amegroups.com/article/view/10.21037/acr-2025-227/rc

Peer Review File: Available at https://acr.amegroups.com/article/view/10.21037/acr-2025-227/prf

Funding: This work was supported by the 2023 Major Project Jointly Funded by the Guangzhou Science and Technology Bureau Basic Research Program and City-University (Institute) Cooperation (No. 2023A03J0359); and the 2024 Medical Imaging Standards and Innovation Development Research Project of the People’s Health Research Institute (No. HX25AII001036).

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://acr.amegroups.com/article/view/10.21037/acr-2025-227/coif). The authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Declaration of Helsinki and its subsequent amendments. Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the editorial office of this journal.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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