A symptomatic cluster headache (CH), caused by fungal maxillary sinus infection fulfilling the criteria of International Headache Society (ICDH-3) for CH is presented. A 35-year-old man without previous headaches reported episodes of pain in the right fronto-orbitary region, with severe eyelid oedema, photophobia, frontal sweating, lacrimation, conjunctival injection and rhinorrhoea, which lasted for 2–3 h. A brain magnetic resonance imaging (MRI) showed occupation of the right maxillary sinus, a mycetoma due to Aspergillus fumigatus. Although rare, secondary causes must be discarded before the diagnosis of a primary CH is made. Imaging study should always be performed, even though no atypical feature was present and ICDH-3 were fulfilled.
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