unilateral headache with orbital pain is a common presenting symptom of several ocular and neurologic complication , and should be differentially diagnosed in the emergency department ( ed ) . herein , we review to our knowledge , the first case of unilateral fronto - temporal headache with ocular pain caused by spontaneous lens dislocation due to zonulysis . lens dislocation ( ectopia lentis ) refers to a displacement of the lens from its normal position . the lens may be completely dislocated , rendering the pupil aphakic ( luxated ) , or partially displaced , still remanding in the pulpillary area ( subluxated ) . a 52-year - old male patient , who had been experiencing left fronto - temporal headache with left ocular pain for 1 day , was admitted to the ed . the day before admission , the man had experienced headache and ocular pain for 3 hours , and then recovered spontaneously . on the day of admission , the headache was dull , and ocular pain was more severe in the lean - forward position . he also complained of blur red vision and lacrimation in the left eye in the supine position . the patient s vital signs at the time of admission into the ed were as follows : blood pressure , 170/105 mmhg ; heart rate , 68 beats / min ; respiratory rate , 18 breaths / min ; body temperature 36.1c . his mental status was clear . 1 ) . consult to neurologist and ophthalmologist was done due to rule out cluster headache and glaucoma . neurologist ordered brain magnetic resonance angiography ( mra ) of the brain to rule out cavernous carotid fistula . the initial ophthalmologic examination revealed normal findings , including normal visual acuity ( visus oculi dextri , 1.0 diopter [ d ] , visus oculi sinistri [ vos ] , 1.0 d ) , and normal intraocular pressure ( normal range , 12 to 22 mmhg ) . we reviewed the ct scan of the brain , and observed lens subluxation in the left eye ( fig . it revealed protrusion of the vitreous body into the anterior chamber , posterior subcapsular opacity , phacodonesis , and lens subluxation toward the nasal area of the left eye , and the ophthalmologist decided him to out - patient department follow up for the possibility of operation . so we recommended him to visit outpatient department and explained the possibility of laser iridectomy in case of aggravation of disease . after 3 days , he visited the ophthalmologic out - patient department , and initial examination showed fluctuation of vision scale ( initial exam vos 0.1 d ; after 1hour exam vos 0.9 d ) in left eye . the intraocular pressure was normal but slit lamp examination revealed further prolapse of the vitreous body into the anterior chamber . there was more progressive inferior dislocation of left lens due to zonulysis in dilated funduscopic examination otherwise normal finding in right eye ( fig . after 2 days , the left eye had been rendered aphakic because of complete dislocation of the lens . left pars plana vitrectomy ( ppv ) and intra ocular lens ( iol ) fixation were recommended by the ophthalmologist . however , the patient wanted to undergo the operation in his hometown and hence , was discharged . the reported causes of dislocation of the crystalline lens of the eye include trauma , heredity , and spontaneous occurrence . among these , however , in our case , the patient had no history of trauma , nor were there any symptoms or signs of trauma . hereditary forms of lens dislocation have been associated with other systemic anomalies ( eg , marfan syndrome , homocystinuria , and weill - marchesani syndrome ) . among them , marfan syndrome is the most common . lens dislocation occurs in about 75% of the patients with marfan syndrome and is usually bilateral , symmetrical , and supertemporal . however in our case the patient had no recurrence on the contralateral side for 2 years , and showed no salient features of marfan syndrome including tall stature , arachnodactyly , joint laxity , mitral valve prolapse , aortic dilatation , axial myopia , increased incidence of retinal detachment , etc . this disease is an inborn error of metabolism with an early age of onset which does not fit in our case . additionally , the patient had no history or symtoms of homocystinuria . according to literature , in homocystinuria , the lens is classically displaced either inferiorly or inferonasally , which was the case with our patient ; however , the average age of homocystinuria patients is below 30 years , which is far below our patients age . in weil - marchesani syndrome , it occurs in children and patients who have skeletal malformations including short stature , brachycephaly , limited joint mobility , and well - developed muscular appearance . therefore , it is hard to assume that systemic anomaly caused the lens dislocation in the case of our patient . this case was one of spontaneous dislocation of the lens with luxation into the vitreous cavity , a very unusual and rare occurrence . unilateral headache with ophthalmalgia can have several other causes such as cluster headache , glaucoma , tolosa - hunt syndrome , cavernous carotid fistula , etc . this case was caused by lens dislocation and movement because of change in the patient s position . the lens was positioned normally when the patient was sitting ; however , in the supine position ( for example , for ct ) , the lens underwent dislocation . generally , we tend to more carefully read a brain parenchyma as reading a brain ct than the other anatomical lesion including orbital lesion . however , as in this case , we should review area other than brain parenchyma carefully , not to miss the important findings .
a 52-year - old man presented to the emergency department ( ed ) for left frontotemporal headache and left eye pain . in the ed , visual exam with extra - ocular movement was performed to rule out ocular pathology and brain imaging to rule out cerebral pathology . 
 no abnormal finding was noted after evaluation of ed . initial exam by an ophthalmologist was also negative with normal intraocular pressure . left lens dislocation 
 was found only after brain imaging . in subsequent second exam by an ophthalmologist with iridodilator , lens dislocation due to spontaneous zonulysis 
 was finally confirmed . 
 hence , ed physicians should carefully review ocular as well as brain anatomy in such cases .