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Show '91993 Raven Pres'. " York Tonic Pupil in Lymphomatoid Granulomatosis Sajjad Haider, F.C.Ophth. Tonic pupil is due to a lesion of orbital parasympathetic neurons in the ciliary ganglion or short ciliary nerves (1). Precise etiology of the condition remains obscure. In the case described here, development of "tonic pupil" was followed by systemic features typical of lymphomatoid granulomatosis, and diagnosis was confirmed by histology of the skin rash. This case underlines the heterogeneity of the causes of tonic pupil. Vascular occlusion can be a possible mechanism in the pathogenesis of tonic pupil. Key Words: Tonic pupil-Lymphomatoid granulomatosis. From the Department of Ophthalmology, West Norwich Hospital. Bowthorpe Road, Norwich NR2 3TU, United Kingdom. Address correspondence and reprint requests to Dr. Sajjad Haider, West Norwich Hospital. Bowthorpe Road, Norwich NR2 3TU, United Kingdom. 38 Lymphomatoid granulomatosis is a postthymic angiocentric T-cell proliferation (2) causing obstruction of arterial lumina. Commonly involved organs are lungs, skin, and central and peripheral nervous system. There are no ocular features characteristic of the condition. Uveitis and eyelid involvement (3), bilateral peripheral retinal vasculitis with posterior uveitis (4), and, more recently, choroidal involvement (5) have been reported. Tonic pupil has never been described in this illness. CASE REPORT A 31-year-old Caucasian male was seen in the ophthalmic department while convalescing from a laparotomy (3 days earlier). He gave a history of blurring of vision in the left eye for 2 weeks. His visual acuity was 20115 in the right eye and 20/30 in the left, improving to 20/20 with a pinhole. The left pupil was dilated to 6 mm, only sluggishly reactive to light and near stimulus. On slit lamp microscopy the reaction to light was found to be sectoral and tonic. On instillation of pilocarpine V8%, the left pupil constricted to 3 mm. The knee reflexes were intact. An orbital computed tomography (CT) scan was reported as normal. A photograph of the patient taken 6 months previously, enlarged to study the size of the pupils, showed the pupils to be equal. This proved the pupil abnormality to be recent. The patient was examined 4 weeks later. All the parameters were the same except the pupil retlex to near, which had improved. This development of light near dissociation at the second consultation, confirmed the diagnosis of tonic pupil and proves it to be as recent as 1 month. He had been admitted on the surgical floor the previous week with sudden-onset umbilical pain accompanied by nausea. He later vomited and pain became generalized. An exploratory laparotomy showed ishaemic small bowel and enlarged mesenteric lymph nodes, which were resected. Histology of resected specimen showed ischaemia TONIC PUPIL IN LYMPHOMATOID GRANULOMATOSIS 39 and mesenteric arterial thrombosis with low-grade vasculitis affecting medium-sized vessels. He later developed a fluctuating pyrexia with exacerbations at night and night sweats, glove stocking parasthesia, and an itchy rash (erythematous indurated plaques) on his extremities. Viral titres, antibody screen, and blood cultures were all negative. The ESR was normal at 15. The only positive laboratory findings were transient opacification on chest x-ray, a raised value of C-reactive protein (16 mg/ L), and neutropenia (white cell count, 2.5), but a bone marrow showed no abnormality. A skin biopsy of the rash showed an angiocentric I-cell lymphoma. The morphology and infiltrative pattern was suggestive of lymphomatoid granulomatosis. DISCUSSION The precise etiology of tonic pupil remains unknown, but viral infections have been suspected to be responsible. Sera from 75 consecutive patients with Adie's tonic pupil, examined for viral and microbial antibodies, however, showed no significant elevation (6). In a recent report, human parvovirus B19 was linked with Adie's pupil in an 18-year-old woman (7). Chickenpox (8) has also been implicated. It is possible that tonic pupil in the latter was due to orbital vasculitis, which is a recognised complication of varicella-zoster infection (9). Another case of tonic pupil was reported in a patient with temporal arteritis (10). In the case described above, development of tonic pupil was followed by systemic features typical of lymphomatoid granulomatosis. This temporal sequence points to lymphomatoid granulomatosis as the cause. It is suggested that vascular occlusion can be a possible pathogenic mechanism for the development of tonic pupil. REFERENCES 1. Lowenfeld IE, Thompson HS. The tonic pupil: a reevaluation. Am I Ophtha/mo/1967;63(1):46-87. 2. Lipford EH Jr, Margolick JB, Longo DL, Fauci AS, Jaffe ES. Angiocentric immunoproliferative lesions: a clinicopathologic spectrum of post thymic T-cell proliferations. Blood 1988;72(5):1674-81. 3. Katzenstein AA, Carrington CB, Liebow AA. Lymphomatoid granulomatosis: a clinicopathological study of 152 cases. 1979; Cancer 43:360-373. 4. Tse DT, Mandlebaum 5, Chuck DA, Nichols PW, Smith RE. Lymphomatoid granulomatosis with ocular involvement. Retina 1985;5:9~7. 5. Pearson AD/. Craft AW, Howe JM. Choroidal involvement in lymphomatoid granulomatosis. Br I Ophtha/mol 1991;75: 688-9. 6. Meek ES, Thompson HS. Serum antibodies in Adie's syndrome. In: Thompson HS, Daroff R, Frisen L, Glaser JS, Saunders MD, eds. Topics in neurophthalmology. Baltimore: Williams & Wilkins. 1979:119-21. 7. Corridan PG, Laws DE, Morrell A/. Murray PI. Tonic pupils and human parvovirus (819) infection. J Clin NeuroophthalmoI1991; 11(2):109-10. 8. Thompson HS. Adie's syndrome: some new observations. Trans Am Ophthalmol Soc 1977;75:587-{j26. 9. Hedges TR. Albert DM. The progression of the ocular abnormalities of herpes zoster: histopathologic observation of nine cases. Ophthalmology 1982;89:165. 10. Davis RH, Daroff RB, Hoyt WF. Tonic pupil after temporal arteritis. [Letter]. LAncet 1968;1:822-823. I Clin NeuTll-t1pittitalmol. Vol. 13. No. 1. 1993 |
