TY - JOUR T1 - COVID-19 in patients with pulmonary alveolar proteinosis: a European multicentre study JF - ERJ Open Research JO - erjor DO - 10.1183/23120541.00199-2022 VL - 9 IS - 1 SP - 00199-2022 AU - Spyros A. Papiris AU - Ilaria Campo AU - Francesca Mariani AU - Maria Kallieri AU - Lykourgos Kolilekas AU - Andriana I. Papaioannou AU - Efsun Gonca Chousein AU - Erdogan Cetinkaya AU - Francesco Bonella AU - Raphael Borie AU - Maria Kokosi AU - Thomas Pickworth AU - Maria Molina-Molina AU - Mercè Gasa AU - Elżbieta Radzikowska AU - Justyna Fijolek AU - Stéphane Jouneau AU - Emmanuel Gomez AU - Cormac McCarthy AU - Elisabeth Bendstrup AU - Wojciech J. Piotrowski AU - Rishi Pabary AU - Alice Hadchouel AU - Nathalie Coolen-Allou AU - Tiago Alfaro AU - Carlos Robalo Cordeiro AU - Elvira-Markela Antonogiannaki AU - Ioannis P. Tomos AU - Despoina Papakosta AU - Theodoros Kontakiotis AU - Panagiota Panagiotou AU - Konstantinos Douros AU - Andrea Schams AU - Sara Lettieri AU - Vassiliki Papaevangelou AU - Christina Kanaka-Gantenbein AU - Anna Karakatsani AU - Stelios Loukides AU - Ulrich Costabel AU - Bruno Crestani AU - Cliff Morgan AU - Ryushi Tazawa AU - Andrew Bush AU - Matthias Griese AU - Effrosyni D. Manali Y1 - 2023/01/01 UR - http://openres.ersjournals.com/content/9/1/00199-2022.abstract N2 - Granulocyte–macrophage colony-stimulating factor (GM-CSF) signalling is essential in both alveolar macrophage (AM) differentiation and activation of lung immune cells [1]. Differentiated AMs are crucial in both the elimination of alveolar microbes and surfactant clearance. The disruption of the GM-CSF axis in AMs leads to the development of pulmonary alveolar proteinosis (PAP) [1]. In the majority of patients, this relates to the presence of autoantibodies against GM-CSF (autoimmune (a)PAP) but there are multiple other causes [1–3]. GM-CSF-deficient animals may have impaired lung inflammatory response to commensal microbes and humans with PAP may occasionally develop opportunistic lung infections [4]. The mainstay of pharmacological treatment in aPAP is inhaled GM-CSF (iGM-CSF), which is off-label but increasingly used worldwide [5–9].Adult PAP patients experience similar #COVID19 rates to the general population, and high rates of hospitalisation and deaths, underscoring their vulnerability and the need for measures to prevent infection. The impact of iGM-CSF must be considered. https://bit.ly/3M0wKnZ ER -