Atypical type D extended sleeve resection after neoadjuvant chemoimmunotherapy in a 69-year-old man with locally advanced lung cancer and a tracheobronchial branching anomaly: a case report

Introduction

Recent studies have demonstrated the efficacy of combination therapy with platinum agents plus immune checkpoint inhibitors (ICIs) as perioperative treatment for locally advanced non-small cell lung cancer (NSCLC) (1-3). The benefits of surgery following preoperative therapy include improved surgical safety and complete resection rate due to tumor reduction, improved prognosis by avoiding pneumonectomy, and preservation of lung function. Notably, some reports have demonstrated the usefulness of preoperative treatment with bronchial sleeve resection to avoid pneumonectomy (4-6). However, whether immune-related inflammatory changes after neoadjuvant chemoimmunotherapy can cause severe hilar fibrosis or a “frozen hilum” that is observed after chemoradiotherapy remains unclear. Further reports on operative findings and technical considerations in complex bronchioplasty after chemoimmunotherapy are needed (7). Furthermore, extended sleeve resection (ESL) is considered more challenging than standard sleeve resection due to the need to adjust the caliber difference, distance, and angle at the anastomotic site (8,9).

Tracheal bronchus is a rare congenital tracheobronchial anomaly, with a reported incidence of approximately 0.1–2% (10). Although the tumor did not originate from the anomalous bronchus, this abnormal branching pattern directly influenced the surgical reconstruction. Reports of complex bronchioplastic reconstruction for lung cancer in the setting of a tracheal bronchus are extremely limited (11), underscoring the novelty of the present case.

According to the modified Okada classification, a type D ESL generally refers to resection of the right middle and lower lobes with anastomosis between the right main bronchus and the right upper lobe bronchus. In contrast, our procedure involved anastomosis of the right main bronchus directly to B2+3 because B1 arose independently from the trachea. This atypical type D ESL differs from the standard classification and represents a technically rare form of bronchioplastic surgery. Therefore, we report this rare case and discuss the clinical value of neoadjuvant chemoimmunotherapy, the feasibility of bronchioplasty after immunotherapy, and technical considerations for atypical ESL. We present this article in accordance with the CARE reporting checklist (available at https://tlcr.amegroups.com/article/view/10.21037/tlcr-2026-1-0199/rc).

Case presentation

A 69-year-old man with a history of heavy smoking (1.5 packs per day for 47 years) presented to his previous physician with a chief complaint of bloody sputum. He had a history of atrial fibrillation, for which he was taking anticoagulants. On initial evaluation, his vital signs were within normal limits, and his Eastern Cooperative Oncology Group (ECOG) performance status was 0. Thoracic auscultation revealed no obvious decrease in breath sounds despite middle lobe atelectasis. Laboratory tests showed normal coagulation parameters, including prothrombin time and activated partial thromboplastin time. Serum carcinoembryonic antigen (CEA) and squamous cell carcinoma antigen (SCC-Ag) were elevated to approximately 16 and 12 ng/mL, respectively. Chest X-ray revealed an abnormal shadow in the right hilar area, and a contrast-enhanced computed tomography (CT) scan revealed a mass in the right hilar area with a tumor size of 7.9 cm, and #7 lymph node (LN) was swollen (Figure 1A,1B). The right B1 bronchus had an anomaly with tracheobronchial branching directly from the trachea (Figure 1C), and the tumor had invaded into the intermediate bronchial trunk, and invasion of the right upper lobe bronchus (B2+3) was also suspected, while the middle lobe was completely atelectatic. The middle pulmonary artery trunk was encased centrally by the tumor, and the inferior pulmonary vein was obstructed peripherally by tumor invasion (Figure 1D). Bronchoscopy showed tumor extension to the bronchial orifice of the intermediate bronchial trunk at the same level as the right B2+3 branch (Figure 1E). Transbronchial lung biopsy revealed a diagnosis of squamous cell carcinoma with a high programmed death-ligand 1 (PD-L1) expression and a tumor proportion score of 90% (22HC). Positron emission tomography-computed tomography (PET-CT) showed an abnormal Fluorodeoxyglucose uptake with a maximum standardized uptake value of 18 in the main tumor and uptake in the enlarged #7 LN and #11s LN (Figure 1F), leading to a diagnosis of cT4N2aM0 stage IIIB [Union for International Cancer Control (UICC) 9th]. Respiratory function tests showed mixed ventilatory impairment with a percent vital capacity of 78% and a percentage forced expiratory volume in one second (FEV1.0%) of 67.5%. The treatment strategy was discussed at a multidisciplinary conference involving pulmonologists, thoracic surgeons, and radiation oncologists. Because the mediastinal nodal disease was considered potentially resectable rather than diffusely infiltrative, and the high PD-L1 expression suggested a favorable response to treatment, neoadjuvant chemoimmunotherapy followed by surgery was chosen to achieve complete resection while avoiding pneumonectomy. No specific diagnostic challenges were encountered in this case.

Figure 1 Pretreatment imaging and bronchoscopic findings. (A,B) A chest contrast-enhanced CT scan revealed a 7.9 cm mass in the right lower lobe and hilar region. Enlargement of lymph node 7 was also observed, suggesting metastasis. (C) The yellow arrow indicates the B1 bronchus branches directly from the trachea, revealing a congenital anomaly of tracheobronchial bifurcation (tracheobronchus). (D) The inferior pulmonary vein was obstructed owing to tumor infiltration. (E) Bronchoscopic findings. The yellow arrow indicates the B2+3 bifurcation. The tumor was exposed at the orifice of the intermediate bronchial trunk. (F) PET-CT revealed abnormal uptake with a SUVmax of 17.9 at the primary lesion, with increased uptake also observed in the hilar and mediastinal lymph nodes. No uptake was observed in other lesions. PET-CT, positron emission tomography-computed tomography; SUVmax, maximum standardized uptake value.

Three cycles of chemotherapy with ICIs were administered: cisplatin (75 mg/m²) + gemcitabine (2,000 mg/m²) + nivolumab (360 mg/body). After neoadjuvant treatment, serum CEA and SCC-Ag levels decreased to 12 and 3.0 ng/mL, respectively. CT imaging showed tumor shrinkage, with regression to the level of the middle bronchial trunk and reduction of the #7 LN (Figure 2A-2D). No new lesions were observed, and the response was classified as partial response according to RECIST. There were no adverse events that could affect the surgery. After neoadjuvant treatment, the clinical stage was ycT2bN2aM0 stage IIIA (UICC 9th), right pneumonectomy was deemed avoidable, and right middle and lower lobectomy and LN dissection with possible bronchioplasty was scheduled.

Figure 2 Radiological response after neoadjuvant chemoimmunotherapy and surgical planning. (A) On plain chest radiography, the hilar mass shadow had shrunk compared with the initial examination image. (B,C) Coronal and axial images from chest contrast-enhanced computed tomography. Compared with pre-treatment, tumor reduction was observed. The main bronchus remains patent, and the location of the tumor relative to the pulmonary artery and bronchus was now clearly defined. (D) Three-dimensional reconstructed CT images viewed dorsally before and after neoadjuvant chemoimmunotherapy. The yellow arrow indicates the B1 bronchus, and the yellow arrowhead indicates the B2+3 bronchus. Before neoadjuvant treatment, the tumor extended to the root of the B2+3 bifurcation. However, tumor shrinkage following neoadjuvant treatment suggested the possibility of achieving a safe resection margin away from the B2+3 bronchus. CT, computed tomography.

Regarding perioperative anticoagulation management, anticoagulant therapy was discontinued preoperatively after consultation with a cardiologist and was restarted as early as possible postoperatively. No intraoperative heparin bridging was performed.

Surgery was performed under general anesthesia and epidural anesthesia in the left lateral position. A screening thoracoscopy was performed, followed by a 25 cm posterolateral incision and a 5th intercostal thoracotomy (Video 1). The #7 LN could be detached from the contralateral bronchus and pericardium and was dissected. The inferior pulmonary vein was secured through the intrapericardial space, and segments 4 and 5 of the pulmonary vein (V4+5) were secured extrapericardially. The tumor was in contact with the central vein, and there was relatively little fibrosis around it. Therefore, we were able to dissect it for exposure. Asc.A2 was sacrificed because it was difficult to separate from the swollen #11s LN. The intermediate trunk of the bronchus could be dissected and secured at the proximal end; therefore, we determined that a right pneumonectomy could be avoided at this point. The inferior pulmonary vein, V4+5, and intermediate pulmonary artery trunk were each dissected with a stapler. The tumor extended to the entrance of the middle bronchus trunk, suggesting that simple transection of the bronchus would likely leave residual tumor and cause constriction of B2+3. Therefore, we decided on the right middle and lower sleeve resection. The right main bronchus distal to the B1 tracheal bronchus and B2+3 were transected, and the right middle and lower lobes were removed. To reduce tension at the anastomotic site, the pulmonary ligament was divided, and the pericardium was opened during inferior pulmonary vein processing. Intraoperative frozen section analysis confirmed a negative bronchial margin. Anastomosis between the right main bronchus and B2+3 was performed using a 4-0 polydioxanone (PDS) II RB-1 suture (Ethicon Inc. Johnson & Johnson Company, New Brunswick, NJ, USA) and completed using a telescoping technique. All sutures were individually tied with the knots placed outside the lumen, and the suture pitch between the central and distal bronchial sides was approximately 3:1, with a total of 16 stitches (Figure 3). After completion of the bronchial anastomosis, an air leak test was performed under ventilation. The residual lung expanded and collapsed promptly, and no air leakage from the anastomosis was observed. Free pericardial fat tissue was harvested and interposed between the pulmonary artery and the anastomosis. Operative time was 4 h 53 min, with an estimated blood loss of 415 g, and bronchoscopy immediately after the procedure confirmed good patency of the peripheral anastomosis.

Figure 3 Intraoperative photograph. This image shows sleeve resection of the middle and lower lobes with anastomosis of B2+3 (yellow arrowhead) to the right main bronchus (yellow arrow). The white vessel loop is retracting the central vein (blue arrow) to maintain the surgical field. Using a telescopic technique, all sutures were performed with a single ligation using 4-0 PDS 16 sutures. To prevent bronchial cutting, tension was reduced during ligation by crossing adjacent sutures to minimize the central-peripheral distance. PDS, polydioxanone.

The postoperative course was uneventful, and the patient was discharged as planned. Preoperatively, mixed ventilatory failure was observed; however, as a pneumonectomy was avoided, the introduction of home oxygen therapy was not required, and he was able to maintain his activities of daily living. At the postoperative bronchoscopy performed 1 month postoperatively, there was favorable healing at the anastomotic site (Figure 4A-4C), and chest X-ray showed good lung expansion (Figure 4D). No changes in the planned therapeutic strategy were required during treatment. Adrenal insufficiency due to immune-related adverse events (irAEs) on postoperative day 21 required hospitalization and treatment, but symptoms improved quickly with steroid replacement therapy. Histopathology revealed an overall tumor size of 3.7 cm × 2.5 cm, LN: 1/18 (12L LN), ypT2aN1M0 stage IIB (UICC 9th), EF2, and residual viable tumor of 20%. Microscopic images showed tumor disappearance in the bronchus dissection site and suggested that preoperative induction therapy could have avoided pneumonectomy and allowed for radical resection (Figure 5A-5C). At 10 months postoperatively, serum CEA and SCC-Ag levels had further decreased to 9 and 2.2 ng/mL, respectively, and no recurrence was observed (Figure 6A,6B). The clinical course of this patient is summarized in Table 1.

Figure 4 Postoperative bronchoscopic and radiological findings. (A) Bronchoscopic findings on POD 1. The anastomotic site was unremarkable, and the distal bronchus was patent. (B,C) Bronchoscopic findings 1 month postoperatively. The anastomotic site exhibited favorable coloration, confirming a good healing response. (D) Postoperative chest X-ray at 1 month. The lung expansion was good throughout the peripheral region. POD, postoperative day.

Figure 5 Microscopic pathological images. (A) HE staining of the primary lesion. Histopathological examination demonstrated nests of atypical squamous cells with enlarged nuclei (scale bar: 100 µm). (B) Histopathological examination of the bronchial resection margin (HE staining) demonstrated extensive stromal fibrosis with layered keratinous material and multinucleated giant cells. These findings suggest that the preoperative treatment was effective and that the tumor had disappeared. (C) Highly magnified photograph of bronchial resection margin (HE staining). Keratinized material and foreign body histiocytes are observed. Nuclear atypia is not prominent. HE, hematoxylin and eosin.

Figure 6 Three-dimensional bronchial reconstruction on the 6-month postoperative CT scan. S2+3 was positioned approximately 90° caudally (A), and the distal bronchial anastomosis showed good patency (B). CT, computed tomography.

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 the 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

This case involved a patient with locally advanced lung cancer accompanied by an anomalous tracheobronchial branching pattern (B1) and preexisting mixed ventilatory impairment. Following a favorable response to neoadjuvant chemoimmunotherapy, we performed an atypical type D ESL with anastomosis of B2+3 to the right main bronchus, achieving complete resection while avoiding pneumonectomy and preserving postoperative activities of daily living.

Bronchioplasty after neoadjuvant therapy remains technically challenging, particularly in complex airway reconstruction. Radiotherapy may impair bronchial healing through vascular reduction, fibrosis, and cellular damage (12,13), and previous studies have reported anastomotic complication rates of 5.2–10.8% after bronchioplasty following neoadjuvant chemoradiotherapy (14,15). In contrast, recent reports suggest that sleeve lobectomy after neoadjuvant chemoimmunotherapy can be performed with acceptable postoperative morbidity and favorable outcomes in selected patients (4,16-18).

Although immune-related inflammatory changes may cause hilar fibrosis and make procedures around the pulmonary arteries more difficult (19), this does not necessarily indicate chronic vascular damage of the kind seen following radiotherapy. In the present case, however, severe hilar fibrosis was not observed intraoperatively, and the pulmonary artery could be dissected safely without vascular reconstruction. This may have contributed to preservation of bronchial blood supply and favorable anastomotic healing.

Extended resections after neoadjuvant therapy remain technically demanding, particularly when pulmonary artery reconstruction or bronchial sleeve procedures are required, and careful patient selection and surgical planning are essential (7,20). The present case required an atypical type D ESL, involving anastomosis between the right main bronchus and B2+3, because of both tumor extent and an anomalous tracheobronchial branching pattern. To the best of our knowledge, this type of atypical type D ESL following neoadjuvant chemoimmunotherapy has not been previously reported.

Marked caliber mismatch and abnormal bronchial alignment in such reconstruction may increase the risk of anastomotic complications, including ischemia, granulation tissue formation, and late-onset stenosis (21). To reduce these risks, we focused on three technical principles: preservation of bronchial blood supply, reduction of anastomotic tension, and stepwise size adjustment. The bronchus was resected carefully to preserve distal perfusion, which partly depends on retrograde flow from the pulmonary artery. A longer segment of the right main bronchus distal to the B1 bifurcation was preserved to avoid traction and bronchial angulation. Finally, the caliber mismatch between the right main bronchus and B2+3 was managed using a telescoping technique with a 3:1 central-to-distal suture pitch, rather than trimming the membranous portion. Favorable anastomotic healing was achieved without ischemia, delayed healing, or stenosis during the current follow-up period. Nevertheless, careful long-term bronchoscopic surveillance is warranted to monitor for late anastomotic complications, given the marked caliber mismatch, anastomotic tension, and limited experience with bronchioplasty after neoadjuvant chemoimmunotherapy.

Postoperative adrenal insufficiency represented an important irAE in this case. Symptoms such as fatigue, anorexia, and general malaise are nonspecific and may be difficult to distinguish from normal postoperative symptoms. Because the patient had received ICIs preoperatively, endocrine irAE was considered early, and hormonal evaluation including adrenal and thyroid function tests was promptly performed. This case highlights the importance of maintaining a low threshold for endocrine testing when unexplained fatigue or systemic symptoms occur after surgery in patients treated with neoadjuvant immunotherapy.

Although high PD-L1 expression supported the use of neoadjuvant chemoimmunotherapy in this case, treatment response may also occur in PD-L1-low or -negative tumors. Therefore, patients with potentially resectable locally advanced NSCLC should not be excluded from consideration for neoadjuvant treatment based on PD-L1 status alone; instead, treatment strategy should be determined through multidisciplinary discussion to avoid missing an opportunity for curative resection.

Previous ESL series have reported acceptable long-term outcomes, supporting its role as a lung preservation surgery alternative to pneumonectomy in selected patients (8,12,22). However, evidence regarding atypical ESL after neoadjuvant chemoimmunotherapy remains limited (23). Although this case remains recurrence-free 10 months after surgery, longer follow-up is required to evaluate recurrence-free survival and oncological durability.

This report has several limitations. First, it describes a single case, and the findings cannot be generalized to all patients with locally advanced NSCLC. Second, the follow-up period remains relatively short; therefore, long-term safety and oncological durability require further evaluation. Additional experience is needed to clarify the safety and feasibility of atypical ESL after neoadjuvant chemoimmunotherapy.

Patient perspective

The patient commented that he was able to go about his daily life without experiencing any shortness of breath.

Conclusions

We report a case of locally advanced lung cancer with an anomalous tracheobronchial branching pattern in which neoadjuvant chemoimmunotherapy enabled curative resection through an atypical type D ESL. This case suggests that combining neoadjuvant chemoimmunotherapy with bronchioplasty may help avoid pneumonectomy and preserve pulmonary function in selected patients. In atypical extended sleeve reconstruction with marked caliber mismatch, meticulous tension reduction, preservation of bronchial blood supply, and careful adjustment of bronchial caliber and axial alignment are essential. Further follow-up and additional cases are needed to clarify long-term outcomes.

Acknowledgments

We thank Editage for English language editing. We also thank the patient who provided written informed consent for the publication of this case report. We presented a poster at the Annual Meeting of the Japanese Association for Chest Surgery in 2025.

Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://tlcr.amegroups.com/article/view/10.21037/tlcr-2026-1-0199/rc

Peer Review File: Available at https://tlcr.amegroups.com/article/view/10.21037/tlcr-2026-1-0199/prf

Funding: None.

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://tlcr.amegroups.com/article/view/10.21037/tlcr-2026-1-0199/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 the 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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