Case Report: Pembrolizumab-associated airway-predominant relapsing polychondritis with longitudinal CD8+ T-cell activation and bronchial stenting
- 1Department of Rheumatology, Renji Hospital, Shanghai Jiao Tong University School of Medicine
- 2Department of Respiratory and Critical Care Medicine, Shanghai East Hospital, School of Medicine, Tongji University
Abstract
Immune checkpoint inhibitors (ICIs) can trigger autoimmune and autoinflammatory toxicities through disruption of peripheral immune tolerance. Relapsing polychondritis (RP) after programmed cell death protein 1 (PD-1) blockade is rare and are described mainly in individual reports. We report a 66-year-old man who developed severe airway-predominant RP after 10 cycles of pembrolizumab for lung adenocarcinoma. Chest CT showed new diffuse tracheal and bilateral bronchial wall thickening and calcification, with severe obstructive ventilatory impairment and systemic inflammation. Exploratory serial peripheral-blood immunophenotyping demonstrated increased proportions of CD38+ and HLA-DR+ cells among circulating CD8+ T cells early in the disease course. Serial measurements showed lower activated CD8+ T-cell proportions during immunosuppressive treatment, in parallel with partial stabilization of respiratory symptoms, pulmonary function and imaging. The patient received sequential glucocorticoids, tofacitinib, methotrexate, abatacept and tocilizumab. Despite partial inflammatory control, subsequent pulmonary infection with severe coughing was followed by pneumomediastinum and progressive structural airway compromise requiring bilateral main-bronchial stenting. One year after stenting, the stents remained in situ with no obvious luminal stenosis and pulmonary function remained stable. This case extends the reported spectrum of pembrolizumab-associated RP by documenting an airway-predominant, treatment refractory course with serial immune phenotyping and long-term post-stenting follow-up.
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BibTeX
@article{xia2026case,
title = {{Case Report: Pembrolizumab-associated airway-predominant relapsing polychondritis with longitudinal CD8+ T-cell activation and bronchial stenting}},
author = {Can Xia and Jie Chen and Xia Fang and Changwen Deng and Xiaoping Zhu and Shuang Ye and Runci Wang},
journal = {Frontiers in Immunology},
year = {2026},
month = oct,
doi = {10.3389/fimmu.2026.1999620},
url = {https://www.frontiersin.org/articles/10.3389/fimmu.2026.1999620/full},
note = {CC BY 4.0},
}
RIS
TY - JOUR
TI - Case Report: Pembrolizumab-associated airway-predominant relapsing polychondritis with longitudinal CD8+ T-cell activation and bronchial stenting
AU - Can Xia
AU - Jie Chen
AU - Xia Fang
AU - Changwen Deng
AU - Xiaoping Zhu
AU - Shuang Ye
AU - Runci Wang
PY - 2026
DA - 2026/10/22
JO - Frontiers in Immunology
DO - 10.3389/fimmu.2026.1999620
UR - https://www.frontiersin.org/articles/10.3389/fimmu.2026.1999620/full
AB - Immune checkpoint inhibitors (ICIs) can trigger autoimmune and autoinflammatory toxicities through disruption of peripheral immune tolerance. Relapsing polychondritis (RP) after programmed cell death protein 1 (PD-1) blockade is rare and are described mainly in individual reports. We report a 66-year-old man who developed severe airway-predominant RP after 10 cycles of pembrolizumab for lung adenocarcinoma. Chest CT showed new diffuse tracheal and bilateral bronchial wall thickening and calcification, with severe obstructive ventilatory impairment and systemic inflammation. Exploratory serial peripheral-blood immunophenotyping demonstrated increased proportions of CD38+ and HLA-DR+ cells among circulating CD8+ T cells early in the disease course. Serial measurements showed lower activated CD8+ T-cell proportions during immunosuppressive treatment, in parallel with partial stabilization of respiratory symptoms, pulmonary function and imaging. The patient received sequential glucocorticoids, tofacitinib, methotrexate, abatacept and tocilizumab. Despite partial inflammatory control, subsequent pulmonary infection with severe coughing was followed by pneumomediastinum and progressive structural airway compromise requiring bilateral main-bronchial stenting. One year after stenting, the stents remained in situ with no obvious luminal stenosis and pulmonary function remained stable. This case extends the reported spectrum of pembrolizumab-associated RP by documenting an airway-predominant, treatment refractory course with serial immune phenotyping and long-term post-stenting follow-up.
SN - 1664-3224
ER -