Key words: short-lasting unilateral neuralgiform headache with conjunctival injection and tearing, trigeminal autonomic cephalalgia, blink reflex, headache (Headache 2020;60:476-477) We report the case of a 32-year-old man experiencing episodes of strictly unilateral, unbearable, stabbing pain involving the left orbitotemporal region, lasting about 60 seconds, associated with mild ipsilateral cranial autonomic features such as conjunctival injection, tearing, and nasal congestion. The uniqueness of the attack presentation lies in the simultaneous dystonic-like jaw opening. The patient attacks could be triggered by facial skin stimulation, chewing, or mandibular overstretching, and the patient was completely symptom-free during the interictal phase. The patient came to our attention 5 months after the onset of the attacks and had previously a neurologist who suspected a trigeminal neuralgia and started a therapy with carbamazepine up to a dose of 200 mg t.i.d. which did not result in any benefits.Although the reported episodes may fulfill the International Headache Society criteria for both trigeminal neuralgia and short-lasting unilateral neuralgiform headache with conjunctival injection and tearing (SUNCT), 1 the poor response to carbamazepine, the long attack duration, the presence of significant autonomic features, the pain quality, and localization made the diagnosis of trigeminal neuralgia unlikely. Moreover, although facial muscles contractions can be observed during trigeminal neuralgia attacks (the so-called "tic-doloreux"), they are usually shorter in duration, with milder autonomic symptoms and the location of the pain (electric shock-like in quality) interest more frequently the second and/or third trigeminal branches. 2 Physical and neurologic examinations, laboratory investigations, brain MRI, and angio MRI were unremarkable. The blink reflex test, in the interictal period, showed a contralateral delay of the later R2 response during stimulation of the right supraorbital nerve and a bilateral delay of the later R2 responses during stimulation of the left supraorbital nerve. These findings suggest an impairment of the Headache