Background: Acute bacterial rhinosinusitis is a common condition often affecting children with inflammation of the mucous membrane in one or more nasal sinuses. Isolated sphenoid sinus infections can exhibit atypical or no symptoms before potentially severe suppurative complications develop. Awareness of early signs of complications is crucial to avoid delays in referral, diagnosis, and treatment. We present the first detailed report of a child with sphenoid sinus empyema with the symptoms intermittent unilateral esotropia and limb and gait ataxia. This case emphasizes the need to recognise atypical presentations, aiding early diagnosis and treatment to prevent severe, possibly life-threatening complications. Case presentation: A 7-year-old girl, previously healthy, presented with a months-long decline in health marked by general malaise, significant weight loss, and recurrent upper respiratory infections. Despite multiple primary care visits no antibiotics were prescribed because infection parameters were found to be normal. Subsequently, the emergence of intermittent double vision prompted referral to the pediatric emergency department. Examination revealed a wide-based gait, ataxic finger-nose-finger and knee-heel tests, and intermittent esotropia of the left eye. Neurological examination was otherwise unremarkable. Imaging of the cerebrum was performed on the suspicion of intracranial pathology and found isolated empyema within the left sphenoid sinus. Endoscopic sinus surgery and antibiotic treatment led to a full recovery within a month. The accommodative esotropia, likely triggered by the infection, was identified, and successfully treated with spectacles. Conclusion: This case recognises isolated empyema in the sphenoid sinus as a potential cause of intermittent esotropia and limb and gait ataxia. It serves as a reminder for clinicians to be vigilant for early signs of suppurative complications to commence prompt diagnosis and treatment to prevent delayed recognition and severe complications. It advocates the inclusion of images of the sphenoid sinus to accompany the conventional scans of the cerebrum, as patients risk referral on the suspicion of intracranial pathology on the basis of neurological symptoms.
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