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Introduction We described a case of a pseudomonas aeruginosa subperiosteal abscess in a healthy adult, complicated by ophthalmic artery occlusion. Case presentation A 41-year-old woman presented with the chief complaint of a severe painful left eyelid. The visual acuity was limited to light perception. Fundus examiantion showed diffuse retinal edema, papillary swelling and whitened retinal vessels without cherry-red spot. Multimodal imaging confirmed the diagnosis of ophthalmic artery occlusion. Computed tomography study was performed and objectified a pansinusitis complicated by left orbital cellulitis and a 7.4mm × 29.8 mm subperiosteal abscess (SPA). In addition to intravenous antibiotics, surgical drainage of the SPA was performed. The bacterial culture of the abscess has shown growth of Pseudomonas aeruginosa and laboratories studies did not find any cause of immunodeficiency. Although medical and surgical treatment, the retinal damage was irreversible with visual acuity limited to light perception. Clinical discussion The developing of a subperiosteal abscess (SPA) of the orbit is a serious complication that arises usually from bacterial sinusitis and can lead to sight threatening complications. Pseudomonas aeruginosa is not common in healthy adults. An early diagnosis and an adequate treatment are important for the visual prognosis. Conclusion Orbital cellulitis should be diagnosed and treated promptly, even in healthy people, to improve visual prognosis., Highlights • The developing of a subperiosteal abscess of the orbit is a serious complication of bacterial sinusitis. • Orbital cellulitis incriminating Pseudomonas species are very rare. • Nowadays, the severe cases resulting from orbital cellulitis have dropped significantly due to the general use of antibiotics. • Orbital cellulitis are sight threatening, even in immunocompetent people.