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• W2152992004 abstract "This case report describes a 16-year-old female with a unilaterally tonic pupil and contralateral anterior ischemic optic neuropathy (AION) as the ocular features of Takayasu arteritis. A 16-year-old female reported sudden blurring of vision in the left eye. Systemic examination showed the presence of left hemiparesis along with nonpalpable upper limb pulses and weak left carotid pulsations. Magnetic resonance imaging of the brain (Fig. 1A) demonstrated an acute infarct in the right periventricular frontoparietal lobe (the watershed territory of right middle and anterior cerebral artery). Magnetic resonance angiography (MRA) demonstrated complete occlusion of the origins of the right brachiocephalic artery along with its branches and left subclavian artery. There was near total occlusion of the left common carotid artery (Fig. 1B) in keeping with type IIa Takayasu arteritis.1Moriwaki R. Noda M. Yajima M. Sharma B.K. Numano F. Clinical manifestation of Takayasu arteritis in India and Japan—new classification of angiographic findings.Angiology. 1997; 48: 369-379PubMed Google Scholar The best corrected visual acuity was measured as 6/6 in the right eye and 6/9 in the left eye. Examination of the pupil revealed anisocoria, with the right pupil measuring 5 mm and left pupil 2 mm in light. The right pupil was poorly reactive to light, and there was a relative afferent pupillary defect in the left eye that manifested with a swinging flashlight test. The right pupillary reaction to a near fixation target was tonic, exhibiting light near dissociation. The right pupil constricted to 0.125% topical pilocarpine, thereby showing evidence of denervation supersensitivity. Vermiform movements of the right pupil with sectoral tonic iris constrictions were visible with a slit-lamp examination, which were not present in the left pupil. The intraocular pressures were measured by Goldmann applanation tonometry as 8 and 11 mm Hg in the right and left eye, respectively. Anterior segment examination was normal in both eyes. A dilated examination of the fundus showed a normal disc and macula in the right eye, whereas the left eye revealed the presence of pallid disc edema with venous tortuosity (Fig. 2). Visual field testing by Humphrey visual field analyzer 30-2 was normal in the right eye. The left eye visual field showed a superior altitudinal field defect extending to the inferonasal quadrant (Fig. 3A) with a mean deviation of –23.84 dB.Fig. 3A, Humphrey visual field report 30-2 program with Swedish Interactive Threshold Algorithm ( SITA) standard of the left eye showing a superior altitudinal scotoma. B, Humphrey visual field report 30-2 program with SITA standard of the left eye showing slight recovery.View Large Image Figure ViewerDownload (PPT)Fig. 3A, Humphrey visual field report 30-2 program with Swedish Interactive Threshold Algorithm ( SITA) standard of the left eye showing a superior altitudinal scotoma. B, Humphrey visual field report 30-2 program with SITA standard of the left eye showing slight recovery.View Large Image Figure ViewerDownload (PPT) Based on the earlier findings, the patient was diagnosed with tonic pupil in the right eye and AION in the left eye with type IIa Takayasu arteritis. Thereafter she underwent bilateral carotid angioplasty with stenting. Ocular examination repeated after 3 months showed persistence of the tonic pupil and relative afferent pupillary defect with normal disc in the right eye and optic atrophy in the left eye with resolution of the optic disc edema. Fig. 4 Visual field testing showed marginal improvement in the left eye fields with the mean deviation changing from –23.84 to –14.79 dB (Fig. 3B). Takayasu arteritis is a nonspecific aortoarteritis characterized by granulomatous inflammation of the large arteries first described by Takayasu in 1908. The inflammation is pan-arteritic with proliferation of tunica intima, destruction of elastic tunica media, followed by fibrosis of the adventitia. The ischemic features due to vessel thrombosis and occlusion can range from transient ischemic attacks, stroke, mesenteric ischemia, renovascular hypertension, myocarditis to cardiac failure.2Johnston S.L. Lock R.J. Gompels M.M. Takayasu arteritis: a review.J Clin Pathol. 2002; 55: 481-486Crossref PubMed Scopus (485) Google Scholar It is more common in females, and the majority of patients present before the age of 40 years. Children usually have type I Takayasu arteritis with fever, arthralgias, and cardiac involvement simulating rheumatic fever or with features of hypertension because of renal vessel involvement (type IV).3Cakar N. Yalcinkaya F. Duzova A. et al.Takayasu arteritis in children.J Rheumatol. 2008; 35: 913-919PubMed Google Scholar The ocular presentations in Takayasu arteritis may be variable and depend on the branches of the carotid artery involved, duration, and rate of vascular insufficiency and development of collateral blood supply. Pathogenesis of these manifestations is either hypoperfusion after occlusion of the branches of the carotid or hypertension secondary to renal artery involvement.4Peter J. David S. Joseph G. Horo S. Danda D. Hypoperfusive and hypertensive ocular manifestations in Takayasu arteritis.Clin Ophthalmol. 2010; 4: 1173-1176Crossref PubMed Scopus (23) Google Scholar The most common ocular feature of hypoperfusion in the carotid arteries is Takayasu retinopathy,4Peter J. David S. Joseph G. Horo S. Danda D. Hypoperfusive and hypertensive ocular manifestations in Takayasu arteritis.Clin Ophthalmol. 2010; 4: 1173-1176Crossref PubMed Scopus (23) Google Scholar followed by ocular ischemic syndrome leading to neovascular glaucoma, vitreous hemorrhage, and tractional retinal detachment. Hypertensive retinopathy and bilateral papilledema are less common and might occur because of renal artery stenosis.5Chun Y.S. Park S.J. Park I.K. Chung H. Lee J. The clinical and ocular manifestations of Takayasu arteritis.Retina. 2001; 21: 132-140Crossref PubMed Scopus (91) Google Scholar AION in Takayasu arteritis has been reported in few adult cases.6Schmidt M.H. Fox A.J. Nicolle D.A. Bilateral anterior ischemic optic neuropathy as a presentation of Takayasu’s disease.J Neuroophthalmol. 1997; 17: 156-161Crossref PubMed Scopus (28) Google Scholar, 7Nithyanandam S. Mohan A. Sheth U. Anterior ischemic optic neuropathy in a case of Takayasu arteritis.Oman J Ophthalmol. 2010; 3: 94-95Crossref PubMed Google Scholar Ocular features are rare in children, and the occurrence of AION has not yet been reported in children with Takayasu arteritis (search MEDLINE database). Hypoperfusion in the internal carotid vessels results in nonarteritic AION, whereas thrombotic narrowing of the posterior ciliary arteries can cause the arteritic type of AION.8Hayreh S.S. Ischemic optic neuropathy.Prog Retin Eye Res. 2009; 28: 34-62Crossref PubMed Scopus (367) Google Scholar In our case, the acute-onset disc edema with pallor may be an arteritic form of AION that appears to be secondary to the near-total occlusion of the left internal carotid artery. Pallid disc edema is described as chalky white swelling of the optic disc in 68.7% of cases with arteritic AION,9Hayreh S.S. Anterior ischemic optic neuropathy. II. Fundus on ophthalmoscopy and fluorescein angiography.Br J Ophthalmol. 1974; 58: 964-980Crossref PubMed Scopus (151) Google Scholar mostly caused by giant cell arteritis (GCA). However, in the younger age group, presence of pallid disc edema should arouse suspicion of other causes of vasculitis such as Takayasu arteritis because GCA is uncommon at this age. Tonic pupils caused by ischemia have been reported in GCA10Foroozan R. Buono L.M. Savino P.J. Sergott R.C. Tonic pupils from giant cell arteritis.Br J Ophthalmol. 2003; 87: 510-512Crossref PubMed Scopus (20) Google Scholar and resolving internal carotid artery occlusion.11Wilhelm H. Tonic pupil caused by ischemia.Fortschr Ophthalmol. 1989; 86: 380-382PubMed Google Scholar The mechanism by which these entities produce tonic pupils is not well-known. Ischemia of the ciliary ganglion and postganglionic parasympathetic fibres,10Foroozan R. Buono L.M. Savino P.J. Sergott R.C. Tonic pupils from giant cell arteritis.Br J Ophthalmol. 2003; 87: 510-512Crossref PubMed Scopus (20) Google Scholar, 12Prasad S. Baccon J. Galetta S.L. Mydriatic pupil in giant cell arteritis.J Neurol Sci. 2009; 284: 196-197Abstract Full Text Full Text PDF PubMed Scopus (8) Google Scholar ischemia of the iris sphincter itself,12Prasad S. Baccon J. Galetta S.L. Mydriatic pupil in giant cell arteritis.J Neurol Sci. 2009; 284: 196-197Abstract Full Text Full Text PDF PubMed Scopus (8) Google Scholar or damage to the ciliary ganglion mediated by immunologic mechanisms13Coppeto J.R. Greco T. Mydriasis in giant cell arteritis.J Clin Neuro-ophthalmol. 1989; 9: 267-269Crossref PubMed Google Scholar are proposed as the mechanisms in patients with GCA. A literature search (MEDLINE database) did not show any reports of tonic pupil in Takayasu arteritis. The complete occlusion of right internal carotid artery as evidenced by the MRA may have resulted in the reduced orbital blood flow to the ciliary ganglion and postganglionic ciliary nerves resulting in ischemia and a tonic pupil. This case highlights the hitherto unreported features of tonic pupil and AION in a teenager with Takayasu arteritis. We suggest that the presence of features of ocular ischemia in children should raise the suspicion of an underlying age-specific systemic vasculitis like Takayasu disease." @default.
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• W2152992004 title "Tonic pupil, anterior ischemic optic neuropathy in a teenager with Takayasu arteritis" @default.
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