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Tonic pupils in children are rare, and only a handful of reports exist in the literature. We present the case of an orbital neural-glial hamartoma in an infant with a congenital tonic pupil and no proptosis. We have previously described this association in another child. This new case, the 6-year follow-up from the previous case, and a discussion of tonic pupils are presented.
Tonic pupils developed in two patients with malignancies outside the nervous system. Symptoms and signs of more generalized somatic and autonomic nervous system involvement were also present. Although the exact morphologic basis for autonomic dysfunction in patients with paraneoplastic neurologic deterioration is uncertain, recent studies suggest that in some cases an autoimmune mechanism is responsible and may be directed against autonomic ganglion cells.
Tonic pupil is usually an idiopathic condition. In some cases, the cause of the ciliary ganglion lesion leading to tonic pupils is obvious. Rarely ischemia causes a lesion of the ciliary ganglion or the short ciliary nerves due to the good blood supply of the ciliary ganglion. Only two cases of tonic pupils in the course of giant cell arteritis are mentioned in the literature, but tonic pupils are probably much more common with this disease. Five cases are demonstrated here. All had associated ischemic optic neuropathy, and stagnation of the blood flow in the supratrochlear artery could be demonstrated in two cases by Doppler sonography. Tonic pupils may also occur when an oclusion of the internal carotid artery resolves, probably because of transient stasis of the orbital blood flow. In another case, tonic pupils were associated with choroidal ischemia (proved by video fluorescent angiography) of unknown origin. The diagnosis of tonic pupils was made by pharmacological testing for cholinergic hypersensitivity with 0.1% pilocarpine.
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.
Of 60 patients with tonic pupils, 29 had serologic tests for syphilis. Five patients had positive blood serology and confirmatory tests; four had other manifestations of neurosyphilis and positive CSF serology. All seropositive patients had bilateral tonic pupils with light-near dissociation and denervation hypersensitivity. Of the 10 tonic pupils, only 1 was miotic and 1 dilated. Although tonic pupils tend to become small and bilateral, they should be distinguished readily from Argyll Robertson pupils, which react briskly, not tonically, to near stimuli. Patients with bilateral tonic pupils should have serologic tests for syphilis.
BACKGROUND: Tonic pupil (TP) is a common disorder of parasympathetic innervation. In contrast to textbook recommendations, cranial imaging is still being performed in most of the patients with TP. The intention of the present study is to show that cranial imaging is of no benefit. PATIENTS AND METHODS: The medical records of 33 patients with TP were analyzed retrospectively. All patients had undergone a complete ophthalmological, orthoptic, and neurological investigation. Cranial imaging was performed by computed tomography or magnetic resonance imaging. Serology tests were carried out in some of the patients. RESULTS: Diagnostic imaging provided no additional data revealing the underlying cause of TP. CONCLUSIONS: Cranial imaging in isolated tonic pupil is not helpful. Because of therapeutic implications, diagnostic evaluation can be recommended only in patients older than 50 years to exclude giant cell arteritis and syphilis.
Two cases of tonic pupil, hyporeflexia, and segmental hypohidrosis (Ross' syndrome) are reported. The relationship of this syndrome to other autonomic dysfunction is discussed. Those symptoms (emotional instability, loss of sweating, orthostatic hypotensive symptoms, and signs of bilaterality of the tonic pupil) which should alert the clinician to more extensive disease states are noted. It is suggested that these conditions may represent a continuum or spectrum of disorders with a widespread degree of severity and progression.
The tonic pupil has a typical appearance and characteristic reactions to both light and the near reflex. Testing with Mecholyl (methacholine) is not possible since the drug is no longer manufactured. The reactions of a group of 25 patients with tonic pupil were studied using dilute pilocarpine, and compared with the pupil responses of a separate control group. We found that concentrations of 0.2% pilocarpine produced too many false-positive reactions in the control group and that 0.05% pilocarpine produced an insufficient response. The 0.1% concentration seemed suitable for ordinary clinical examinations and is recommended for pharmacologic confirmation of the diagnosis of tonic (Adie's) pupil.
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A tonic pupil may occur in isolation or as part of a systemic disorder. We report a patient who developed tonic pupils and an abduction deficit in the setting of polyarteritis nodosa. The combination of a tonic pupil and an abduction deficit should suggest the possibility of a vasculopathic disorder, because the ciliary ganglion and lateral rectus muscle are both supplied by the lateral muscular artery. Widespread small artery and arteriolar narrowing and occlusion are the hallmarks of polyarteritis nodosa. Unusual ischemic syndromes may occur, such as this rare combination of neuro-ophthalmic signs, by involvement of both the nutrient artery and its collaterals. We are unaware of other reports of neuropathic tonic pupils in association with polyarteritis nodosa.
PURPOSE: To present a case of tonic pupil associated with enhancement in the region of the ciliary ganglion on magnetic resonance imaging in a patient with sarcoidosis. DESIGN: Observational case report. METHODS: A 52-year-old woman with sarcoidosis had a right pupil exhibiting sectoral palsy to light and light-near dissociation. RESULTS: Topical administration of 0.1% pilocarpine demonstrated supersensitivity of the right pupillary sphincter muscle. Magnetic resonance imaging (MRI) with contrast revealed enhancement in the region of the right ciliary ganglion. CONCLUSIONS: Tonic pupil may be a manifestation of sarcoidosis, and in such cases, MRI may show enhancement at the level of the ciliary ganglion. Sarcoidosis should be included in the differential diagnosis of tonic pupil.
PURPOSE/METHODS: Tonic pupils in early childhood are rare. We studied an otherwise healthy 31/2-month-old girl who had a right pupil that was poorly reactive to light, without other signs of oculomotor nerve palsy. RESULTS/CONCLUSIONS: Constriction of the right pupil after instillation of 0.125% pilocarpine eyedrops confirmed denervation hypersensitivity, consistent with a tonic pupil. There was no strabismus or proptosis. A magnetic resonance imaging scan demonstrated a right orbital mass, interposed between the lateral and inferior recti muscles. Biopsy was consistent with a benign, glial-neural hamartoma. Thus, in this young patient, a tonic pupil was associated with a benign orbital mass.
BACKGROUND: To describe 4 patients with bilateral tonic pupils in neurosyphilis. CASES: Four young men had pupillary abnormalities, which involved irregular shapes, large sizes, tonic reactions, and vermiform movements of both pupils with light-near dissociation. Cholinergic supersensitivity defined bilateral tonic pupils. OBSERVATIONS: Neurosyphilis was diagnosed in these patients on the basis of ocular and laboratory findings. CONCLUSIONS: Bilateral tonic pupils may be important as initial findings in neurosyphilis. We conclude that patients with bilateral tonic pupils should undergo serologic tests for syphilis.
Cholinergic supersensitivity of the iris sphincter helps to make the diagnosis of "tonic pupil." Because uncertain responses sometimes occur with mecholyl 2.5%, dilute solutions of pilocarpine are often used. There has always been some question as to whether individual variation in corneal permeability was one of the factors contributing to the variability in pupillary constriction to topical cholinergics in these patients. In this study, the degree of intraocular penetration of topical fluorescein was compared with the degree of pilocarpine-induced miosis in six tonic pupils and in seven age-matched normal controls. A noncontact fluorophotometer and a computerized videopupillogram were used. The patients showed a significant cholinergic supersensitivity in their affected tonic pupils and had normal intraocular penetration of fluorescein through the cornea in both eyes. It is concluded that the cholinergic supersensitivity of the iris in tonic pupils is not the result of increased corneal permeability but instead results from an increased sensitivity of the cholinergic receptors in iris sphincter muscles.
Only a few cases of carcinomatous neuropathy with tonic pupils have been reported. In the present paper we described a 53-year-old woman with subacute sensory neuropathy who had presented with bilateral tonic pupils. She noticed numbness over the medial aspect of the left thigh in March, 1988, and then developed dysesthesia over the left forearm and hand, mild weakness of left upper and lower extremities and urinary disturbance. Neurological symptoms were subacutely progressive and she was bed-ridden in May. She was admitted to our hospital in June, 1988. On examination, she had body-weight loss of 6 kg during the last six months and general status was otherwise unremarkable. She had anisocoria; the left pupil was larger in daylight than the right, while smaller in dim light. The left pupil scarcely reacted to light, but promptly constricted to near vision. The right pupil constricted normally to light and near vision. An instillation of 0.0625% pilocarpine solution showed supersensitive response of both pupils. An instillation of 1.25% epinephrine solution demonstrated mild dilation of both pupils. Thus, it was conceivable that she had postganglionic ciliary nerve damage characteristic of tonic pupil as well as the lesion of sympathetic nerve innervating pupillary dilator. She had severe sensory ataxia and pseudoathetosis of the hands. Weakness was mild to moderate in extremities. Almost all deep tendon reflexes were absent. All modalities of sensation, particularly on deep sense, were severely involved with sea-level-type distribution below Th7, and over C2 to C3 regions on the left side.(ABSTRACT TRUNCATED AT 250 WORDS)
PURPOSE: To report the association of tonic pupil, congenital neuroblastoma, Hirschsprung disease, and central hypoventilation syndrome. METHODS: Case reports. RESULTS: Two infants with central hypoventilation syndrome and Hirschsprung disease were evaluated for dilated, nonreactive pupils present since birth. In both cases, pharmacologic testing with dilute pilocarpine confirmed denervation supersensitivity consistent with tonic pupil. The diagnosis of congenital neuroblastoma was subsequently established in both infants. CONCLUSIONS: The close association of these conditions in these two young children supports the concept of a common neural crest abnormality being present. A careful systemic evaluation to rule out congenital neuroblastoma should be performed in any young child who presents with tonic pupil in the setting of central hypoventilation syndrome and Hirschsprung disease.
Lyme disease is a multisystemic disorder caused by Borrelia burgdorferi spirochete, transmitted by ticks. Mainly described in the northern hemisphere and rarely in Brazil. The purpose of this report is to describe a patient with Lyme disease who developed bilateral tonic pupil as the only remaining sign. A 13-year-old female with Lyme disease, presented with bilateral peripheral facial and oculomotor paralysis. After recovery from neurological abnormalities the patient sustained anisocoria, reduced fotomotor reflex, less than normal amplitude of accommodation, tonic pupil constriction for near objects and slow redialation in both eyes. Dilute 0.1% pilocarpine test was positive in both eyes, confirming the suspicion of bilateral tonic pupil. This is the first case report of bilateral tonic pupil caused by Lyme disease.