Unilateral Involuntary Eyelid Closure Induced by Diplopia That Did Not Remit With Contact Lens Occlusion

Abstract

We report an unusual case of unilateral blepharospasm secondary to diplopia that did not remit even with contact lens occlusion. A 50-year-old woman had diplopia on right gaze for 3 years. Examination revealed a right face turn, a complete right abduction deficit, and a partial deficit of right adduction and supraduction. The eyelids and pupils were normal. Magnetic resonance imaging revealed a right intracavernous meningioma. During follow-up, diminished supraduction of the OD and synkinetic elevation of the lid on adduction and infraduction developed, as well as absence of right incyclotorsion on attempted downward gaze, attributed to aberrant regeneration of a palsied third cranial nerve (Fig. 1). The face-turn increased and involuntary unilateral closure of the OD lids developed that was attributed to a desire to eliminate diplopia (Fig. 2).FIG. 1: Right third cranial nerve palsy. The patient's lids have been elevated and she has been asked to direct her gaze up, down, left, and right (see arrows). There is limitation principally of supraduction and abduction of the OD.FIG. 2: Involuntary blepharospasm, right. The left panel shows the patient at rest with involuntary closure of the OD lids. This occurred when she was awake and also persisted when the OD was occluded with a contact lens. The right panel shows that she could, with effort, open her OD for short periods. There is moderate ptosis caused by partial weakness of the levator palpebrae superioris.Botulinum toxin injected into the right medial rectus failed to relieve primary position diplopia, so a right occlusive contact lens was fitted. Despite relief of the diplopia, the involuntary OD lid closure persisted. When the occlusive contact lens was removed and she occluded her OS, the OD lids opened briefly, but she experienced vertigo. A right upper lid crutch in conjunction with the right occlusive lens was unsuccessful. She was finally treated with botulinum toxin injection into the right orbicularis muscle. With the occluding contact lens in place in the OD, the OD lids remained open. Several triggers of blepharospasm have been reported. These include bright lights, air, and cold (1). Elston (2) found that 57% of 272 blepharospasm patients had ophthalmological symptoms at the onset, mostly irritation and photophobia, but there was no evidence of ocular disease on examination. Jankovic and Orman (3) reported the lack of associated ocular signs in patients with blepharospasm who described ocular symptoms. Ocular irritation as a result of lagophthalmos from Bell's palsy is thought also to be a precipitating factor in those predisposed to blepharospasm (4). Precipitating triggers have been noted in other dystonias, such as local hand injury in those with writer's cramp (2). It is possible that diplopia-activated brain stem mechanisms subserving unilateral eye closure in our patient resulted in involuntary eye closure that persisted despite contact lens occlusion and eventually required botulinum toxin treatment. Eugene Tay MSc, MRCSEd, MRCOphth Gordon T. Plant MA, MD, FRCP Moorfields Eye Hospital and Royal National Hospital for Neurology and Neurosurgery; London, England; E-mail: [email protected]