Markov Nikolay S, Esposito Anthony J, Senkow Karolina J, Schleck Maxwell, Cusick Luisa, Yu Zhan, Sokolenko Yuliana V, Diaz Estefani, Jonasson Emmy, Swaminathan Suchitra, Lu Ziyan, Nafikova Radmila, Fenske Samuel, Bunyan Elsie G, Pérez-Leonor Xóchitl G, Abdala-Valencia Hiam, Flozak Annette S, Joshi Nikita, Argento A Christine, Malsin Elizabeth S, Reyfman Paul A, Puchalski Jonathan, Gulati Mridu, Carns Mary, Aren Kathleen, Cooper Phillip, Field Natania S, Mohsin Suror, Shawabkeh Malek, Soriano Alexandra, Gundersheimer Aaron N, Goldberg Isaac A, Damore Bailey, Peltekian Alec, Agrawal Ankit, Cheung Crystal, Perez Stephanie, Teaw Shannon, Williams Alyssa, Page Nicolas, Kujawski Sophia E, Odell William, Gunes Baran Ilayda, Cheng Michelle, Emokpae Morgan, Cumming R Ian, Tighe Robert M, Grudzinski Kevin, Savas Hatice, Rubinowitz Ami N, Kadhim Bashar A, Kurihara Chitaru, Bharat Ankit, Mehta Vikas, Dematte Jane E, Bemiss Bradford C, Makinde Hadijat M, Cuda Carla M, Dapas Matthew, Richardson Carrie, Perlman Harris, Lam Anna P, Gottardi Cara J, Budinger G R Scott, Misharin Alexander V, Hinchcliff Monique E
Division of Pulmonary and Critical Care Medicine, Feinberg School of Medicine, Northwestern University.
Simpson Querrey Lung Institute for Translational Science, Northwestern University.
bioRxiv. 2025 Aug 11:2025.08.07.669006. doi: 10.1101/2025.08.07.669006.
Interstitial lung disease (ILD) is present in over 60% of patients with systemic sclerosis (SSc) and is the leading cause of SSc-related deaths. Profibrotic monocyte-derived alveolar macrophages (MoAM) play a causal role in the pathogenesis of pulmonary fibrosis in animal models where their persistence in the niche requires signaling through Colony Stimulating Factor 1 Receptor (CSF1R). We hypothesized that the presence and proportion of MoAM in bronchoalveolar lavage (BAL) fluid from patients with SSc-ILD may be a biomarker of ILD severity. To test this hypothesis, we analyzed BAL fluid from 9 prospectively enrolled patients with SSc-ILD and 13 healthy controls using flow cytometry and single-cell RNA sequencing. Patients with SSc-ILD had more MoAM and interstitial macrophages in BAL fluid than healthy controls, and their abundance was associated with lung fibrosis severity. We identified changes in the MoAM transcriptome as a function of treatment with mycophenolate, an effective therapy for SSc-ILD. In SSc-ILD lung explants, spatial transcriptomics identified an expanded population of interstitial macrophages spilling into the alveolar space. Our findings suggest that the proportion of profibrotic MoAM and interstitial macrophages in BAL fluid may serve as a biomarker of SSc-ILD and credential them as possible targets for therapy.
间质性肺疾病(ILD)存在于超过60%的系统性硬化症(SSc)患者中,并且是SSc相关死亡的主要原因。促纤维化单核细胞衍生的肺泡巨噬细胞(MoAM)在动物模型的肺纤维化发病机制中起因果作用,在这些模型中,它们在生态位中的持续存在需要通过集落刺激因子1受体(CSF1R)进行信号传导。我们假设,来自SSc-ILD患者的支气管肺泡灌洗(BAL)液中MoAM的存在和比例可能是ILD严重程度的生物标志物。为了验证这一假设,我们使用流式细胞术和单细胞RNA测序分析了9名前瞻性招募的SSc-ILD患者和13名健康对照的BAL液。与健康对照相比,SSc-ILD患者的BAL液中有更多的MoAM和间质巨噬细胞,并且它们的丰度与肺纤维化严重程度相关。我们确定了MoAM转录组的变化是霉酚酸治疗(一种治疗SSc-ILD的有效疗法)的函数。在SSc-ILD肺外植体中,空间转录组学确定了一群扩大的间质巨噬细胞溢出到肺泡空间。我们的研究结果表明,BAL液中促纤维化MoAM和间质巨噬细胞的比例可能作为SSc-ILD的生物标志物,并证明它们是可能的治疗靶点。