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Pulmonary alveolar proteinosis associated with ruxolitinib
Rav Sellahewa1,2, Daniel Tan3,4, Maraed Rosa3
1Department of Respiratory Medicine and Sleep Disorders, The Royal Melbourne Hospital, Melbourne, Victoria, Australia ravindu.sellahewa@mh.org.au.
Abstract:
Pulmonary alveolar proteinosis (PAP) is a rare condition characterised by impaired alveolar macrophage-mediated surfactant clearance, resulting in the accumulation of lipoproteinaceous material within the alveoli. Secondary PAP has been associated with certain medications. Ruxolitinib, a Janus kinase 1/2 inhibitor has recently been implicated in rare cases of PAP.We report the case of a woman in her early 60s treated with ruxolitinib for chronic pulmonary graft-versus-host disease following an allogeneic haematopoietic stem cell transplant for acute lymphoblastic leukaemia, who developed progressive exertional dyspnoea. High-resolution CT demonstrated a 'crazy paving' pattern, and bronchoalveolar lavage revealed periodic acid-Schiff positive granular material consistent with PAP. Microbiological studies were negative. Ruxolitinib was discontinued with subsequent symptomatic, radiological and lung function improvement.This case highlights ruxolitinib as a potential cause of secondary PAP and emphasises the importance of considering this rare complication in patients who develop new respiratory symptoms while receiving ruxolitinib.
Insights
Ruxolitinib, a medication for graft-versus-host disease, may cause pulmonary alveolar proteinosis (PAP). Discontinuing the drug led to patient improvement, highlighting ruxolitinib as a potential cause of this rare lung condition.
Area of Science:
- Pulmonology
- Pharmacology
- Hematology
Background:
- Pulmonary alveolar proteinosis (PAP) is a rare lung disease caused by impaired surfactant clearance.
- Secondary PAP can be linked to certain medications.
- Ruxolitinib, a Janus kinase inhibitor, has been rarely associated with PAP.
Purpose of the Study:
- To report a case of secondary PAP potentially induced by ruxolitinib.
- To emphasize the importance of considering ruxolitinib as a cause of PAP in patients with respiratory symptoms.
Main Methods:
- Case report of a patient treated with ruxolitinib for chronic pulmonary graft-versus-host disease.
- Clinical assessment including high-resolution CT and bronchoalveolar lavage.
- Monitoring of patient's condition after ruxolitinib discontinuation.
Main Results:
- The patient developed progressive exertional dyspnea.
- High-resolution CT showed a 'crazy paving' pattern.
- Bronchoalveolar lavage confirmed PAP; ruxolitinib discontinuation led to symptomatic and radiological improvement.
Conclusions:
- Ruxolitinib is a potential cause of secondary pulmonary alveolar proteinosis.
- Clinicians should consider ruxolitinib-induced PAP in patients presenting with new respiratory symptoms.
