{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,1]],"date-time":"2026-07-01T02:53:00Z","timestamp":1782874380520,"version":"3.54.5"},"update-to":[{"DOI":"10.12688\/f1000research.127299.1","type":"new_version","label":"New version","source":"publisher","updated":{"date-parts":[[2023,8,21]],"date-time":"2023-08-21T00:00:00Z","timestamp":1692576000000}}],"reference-count":8,"publisher":"F1000 Research Ltd","license":[{"start":{"date-parts":[[2023,8,21]],"date-time":"2023-08-21T00:00:00Z","timestamp":1692576000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["f1000research.com"],"crossmark-restriction":false},"short-container-title":["F1000Res"],"abstract":"<ns7:p>\n                    <ns7:bold>Background:<\/ns7:bold>\n                    Pulmonary alveolar proteinosis (PAP) is a very rare diffuse lung disease characterized by the accumulation of amorphous and periodic acid Schiff-positive lipoproteinaceous material in the alveolar spaces due to impaired surfactant clearance by alveolar macrophages. Three main types were identified: Autoimmune, secondary and congenital. PAP has been previously reported to be associated with several systemic auto-immune diseases. Accordingly, we present the first case report of PAP associated with myasthenia gravis.\n                  <\/ns7:p>\n                  <ns7:p>\n                    <ns7:bold>Case:<\/ns7:bold>\n                    A 27-year-old female patient, ex-smoker, developed a dyspnea on exertion in 2020. The chest X-ray detected diffuse symmetric alveolar opacities. Pulmonary infection was ruled out, particularly Coronavirus disease 2019. The chest scan revealed the \u201ccrazy paving\u201d pattern. The bronchoalveolar lavage showed a rosy liquid with granular acellular eosinophilic material Periodic acid-Schiff positive. According to the lung biopsy results, she was diagnosed with PAP. The granulocyte macrophage colony-stimulating factor autoantibodies were negative. Nine months later, she was diagnosed with bulbar seronegative myasthenia gravis, confirmed with the electroneuromyography with repetitive nerve stimulation showing significant amplitude decrement of the trapezius and spinal muscles. She was treated with pyridostigmine, oral corticosteroids and azathioprine. Given the worsening respiratory condition of the patient, a bilateral whole lung lavage was performed with a partial resolution of symptoms. Thus, this previously unreported association was treated successfully with rituximab, including improvement of dyspnea, diplopia and muscle fatigability at six months of follow-up.\n                  <\/ns7:p>\n                  <ns7:p>\n                    <ns7:bold>Conclusions:<\/ns7:bold>\n                    This case emphasizes on the possible association of auto-immune disease to PAP, which could worsen the disease course, as the specific treatment does not exist yet. Hence, further studies are needed to establish clear-cut guidelines for PAP management, particularly when associated to auto-immune diseases.\n                  <\/ns7:p>","DOI":"10.12688\/f1000research.127299.2","type":"journal-article","created":{"date-parts":[[2023,8,21]],"date-time":"2023-08-21T11:05:58Z","timestamp":1692615958000},"page":"1439","update-policy":"https:\/\/doi.org\/10.12688\/f1000research.crossmark-policy","source":"Crossref","is-referenced-by-count":1,"title":["Case Report: The first reported case of pulmonary alveolar proteinosis with myasthenia gravis in a 27-year-old patient"],"prefix":"10.12688","volume":"11","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8296-4182","authenticated-orcid":false,"given":"Islam","family":"Mejri","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7752-0723","authenticated-orcid":false,"given":"Lenda","family":"Ben Hmida","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maroua","family":"Kacem","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mariem","family":"Msalmani","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Houda","family":"Sn\u00e8ne","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hana","family":"Blibech","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aida","family":"Ayadi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jamel","family":"Zaouali","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zied","family":"Moatemri","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"2560","published-online":{"date-parts":[[2023,8,21]]},"reference":[{"key":"ref1","doi-asserted-by":"publisher","first-page":"e057671","DOI":"10.1136\/bmjopen-2021-057671","article-title":"Efficacy and safety of whole-lung lavage for pulmonary alveolar proteinosis: a protocol for a systematic review and meta-analysis.","volume":"12","author":"S Liu","year":"20 avr 2022","journal-title":"BMJ Open"},{"key":"ref2","doi-asserted-by":"publisher","first-page":"389-391","DOI":"10.7417\/CT.2021.2344","article-title":"ANCA Associated Vasculitis and Related Pulmonary Alveolar Hemorrhage in a Patient with Pulmonary Alveolar Proteinosis.","volume":"172","author":"M Mutlu","year":"29 sept 2021","journal-title":"Clin. Ter."},{"key":"ref8","doi-asserted-by":"publisher","first-page":"38-43","DOI":"10.7453\/gahmj.2013.008","article-title":"The CARE Guidelines: Consensus-based Clinical Case Reporting Guideline Development.","volume":"2","author":"J Gagnier","year":"sept 2013","journal-title":"Glob. Adv. Health Med."},{"key":"ref3","doi-asserted-by":"publisher","first-page":"491-498","DOI":"10.1097\/MCP.0b013e32832ea51c","article-title":"Alveolar proteinosis syndrome: pathogenesis, diagnosis, and management.","volume":"15","author":"I Huizar","year":"sept 2009","journal-title":"Curr. Opin. Pulm. Med."},{"key":"ref4","doi-asserted-by":"publisher","first-page":"939-940","DOI":"10.1136\/thx.43.11.939","article-title":"Pulmonary alveolar lipoproteinosis complicating juvenile dermatomyositis.","volume":"43","author":"M Samuels","year":"nov 1988","journal-title":"Thorax."},{"key":"ref5","doi-asserted-by":"publisher","first-page":"77-83","DOI":"10.1620\/tjem.243.77","article-title":"Elevated Serum Anti-GM-CSF Antibodies before the Onset of Autoimmune Pulmonary Alveolar Proteinosis in a Patient with Sarcoidosis and Systemic Sclerosis.","volume":"243","author":"M Yamasue","year":"sept 2017","journal-title":"Tohoku J. Exp. Med."},{"key":"ref6","doi-asserted-by":"publisher","first-page":"1060-1063","DOI":"10.1177\/0961203313498798","article-title":"Systemic lupus erythematosus complicating autoimmune pulmonary alveolar proteinosis that was worsened by immunosuppressive therapy.","volume":"22","author":"J Nagasawa","year":"sept 2013","journal-title":"Lupus."},{"key":"ref7","doi-asserted-by":"publisher","first-page":"1361-1367","DOI":"10.1183\/09031936.00197710","article-title":"An open-label trial of rituximab therapy in pulmonary alveolar proteinosis.","volume":"38","author":"M Kavuru","year":"d\u00e9c 2011","journal-title":"Eur. Respir. 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