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We report a 40-year-old woman who presented with rapidly severe bilateral proptosis, visual impairment, and ocular surface damage. Cranio-orbital imaging demonstrated bilateral extraconal masses and diffuse lytic lesions. Immunoelectrophoresis identified monoclonal IgA kappa, and bone marrow aspiration showed 48% dystrophic plasma cells. Histopathological examination of a conjunctival biopsy confirmed extramedullary plasmacytoma, establishing the diagnosis of IgA kappa MM with bilateral OP. Despite a marked early local response after one month of treatment with high-dose corticosteroids and immunochemotherapy, the patient died shortly after. This case highlights an exceptionally rare and aggressive initial presentation of MM with bilateral OP, underscoring the high tumor burden and poor prognosis associated with this unusual manifestation.<\/jats:p>","DOI":"10.1093\/omcr\/omag176","type":"journal-article","created":{"date-parts":[[2026,9,14]],"date-time":"2026-09-14T11:19:34Z","timestamp":1789384774000},"source":"Crossref","is-referenced-by-count":0,"title":["Bilateral proptosis as the initial manifestation of multiple myeloma with orbital involvement: case report and literature review"],"prefix":"10.1093","volume":"2026","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3681-1406","authenticated-orcid":false,"given":"Sarra","family":"Chadli","sequence":"first","affiliation":[{"name":"Internal Medicine and Clinical Immunology, Ibn Sina University Hospital, Mohammed V University in Rabat , Abderrahim Bouabid Ave., Souissi, 10100, Rabat ,","place":["Morocco"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mouna","family":"Maamar","sequence":"additional","affiliation":[{"name":"Internal Medicine and Clinical Immunology, Ibn Sina University Hospital, Mohammed V University in Rabat , Abderrahim Bouabid Ave., Souissi, 10100, Rabat ,","place":["Morocco"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hajar","family":"Khibri","sequence":"additional","affiliation":[{"name":"Internal Medicine and Clinical Immunology, Ibn Sina University Hospital, Mohammed V University in Rabat , Abderrahim Bouabid Ave., Souissi, 10100, Rabat ,","place":["Morocco"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wafa","family":"Ammouri","sequence":"additional","affiliation":[{"name":"Internal Medicine and Clinical Immunology, Ibn Sina University Hospital, Mohammed V University in Rabat , Abderrahim Bouabid Ave., Souissi, 10100, Rabat ,","place":["Morocco"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Naima","family":"Moatassim","sequence":"additional","affiliation":[{"name":"Internal Medicine and Clinical Immunology, Ibn Sina University Hospital, Mohammed V University in Rabat , Abderrahim Bouabid Ave., Souissi, 10100, Rabat ,","place":["Morocco"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2151-2721","authenticated-orcid":false,"given":"Hamza","family":"Retal","sequence":"additional","affiliation":[{"name":"Radiology, Ibn Sina University Hospital, Mohammed V University in Rabat , Abderrahim Bouabid Ave., Souissi, 10100, Rabat ,","place":["Morocco"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8672-467X","authenticated-orcid":false,"given":"Zoubida","family":"Tazi Mezalek","sequence":"additional","affiliation":[{"name":"Clinical Hematology, Ibn Sina University Hospital, Mohammed V University in Rabat , Abderrahim Bouabid Ave., Souissi, 10100 , Rabat ,","place":["Morocco"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hicham","family":"Harmouche","sequence":"additional","affiliation":[{"name":"Internal Medicine and Clinical Immunology, Ibn Sina University Hospital, Mohammed V University in Rabat , Abderrahim Bouabid Ave., Souissi, 10100, Rabat ,","place":["Morocco"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2026,9,25]]},"reference":[{"key":"2026092500382237300_ref1","doi-asserted-by":"publisher","first-page":"e538","DOI":"10.1016\/S1470-2045(14)70442-5","article-title":"International myeloma working group updated criteria for the diagnosis of multiple myeloma","volume":"15","author":"Rajkumar","year":"2014","journal-title":"Lancet Oncol"},{"key":"2026092500382237300_ref2","doi-asserted-by":"publisher","first-page":"3384","DOI":"10.3324\/haematol.2023.282905","article-title":"Cancer-specific mortality in multiple myeloma: a population-based retrospective cohort study","volume":"108","author":"McCurdy","year":"2023","journal-title":"Haematologica."},{"key":"2026092500382237300_ref3","doi-asserted-by":"publisher","first-page":"vii143","DOI":"10.1093\/annonc\/mdq370","article-title":"Multiple myeloma. 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