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Show LETTERS TO THE EDITOR Horner Syndrome and Ipsilateral Abduction Deficit Attributed to Giant Cell Arteritis We recently examined a 77- year- old woman with Homer syndrome and ipsilateral abduction deficit that we attribute to giant cell arteritis ( GCA). This combination of findings has not been previously reported in GCA. She had a history of hypertension, hyperlipidemia, atrial fibrillation, congestive heart failure, and colon carcinoma. Six weeks before presentation, she developed bitemporal headaches. She had stopped eating solids as a result of jaw claudication and had lost weight. She had diffuse myalgia and was unable to sleep well because of bilateral scalp tenderness. She noticed binocular horizontal double vision that was worse on left gaze and had experienced numerous episodes of blurring of vision in both eyes. Her family noted drooping of her left upper lid. Neuro- ophthalmic examination revealed a best-corrected visual acuity of 20/ 40 in both eyes consistent with cataracts. Visual fields and color vision were normal. Left upper lid ptosis of 2 mm was present. In dim light, the right pupil measured 5.5 mm and the left 3.5 mm. In bright light, the right pupil measured 4.5 mm and the left 3 mm. Both pupils reacted briskly to light without afferent defect. Horner syndrome was confirmed by instilling 10% cocaine into both eyes ( Fig. 1). ( Hydroxyamphetamine was not instilled as a result of its unavailability.) Ocular ductions were full except for reduced abduction of the left eye. There was a 12 prism- diopter esotropia in primary position increasing to 20 on left gaze. The fundus examination was normal. Temporal arterial pulsation was absent and the scalp was tender, but there was no thickening or nodularity. Westergren erythrocyte sedimentation rate ( ESR) was 57, C- reactive protein was 4.1, and platelet count was 475,000. MRI of the brain with contrast was normal and magnetic resonance angiography showed an incidental small basilar tip aneurysm. FIG. 1. Left Horner syndrome. After instillation of one drop of a 10% cocaine solution, the right pupil has become dilated and the left pupil has remained small. Left upper lid ptosis is evident. We made a diagnosis of GCA with concurrent Homer syndrome and left lateral rectus paresis. Intravenous methylprednisolone at a dosage of 250 mg per day and 80 mg oral prednisone per day was initiated. She was also started on alendronate, calcium with vitamin D, and ranitidine. Temporal artery biopsy on the left side was consistent with giant cell arteritis ( Fig. 2). Within a week of initiation of treatment, she had resolution of diplopia, headaches, and temporal scalp pain. Ptosis and pupillary abnormality remained unchanged. ESR, C- reactive protein, and platelet counts normalized in a week. Prednisone was tapered slowly over 18 months. Horner syndrome resulting from GCA is very rare. Three cases of GCA and Horner syndrome have been reported in the English literature ( 1- 3). Bell et al ( 1) reported a patient with GCA and Homer syndrome resulting from a brainstem stroke. Askari et al ( 2) reported a patient with internuclear ophthalmoplegia and Horner syndrome resulting from presumed giant cell arteritis. Bromfield and Slakter ( 3) reported a patient with GCA with postganglionic Homer syndrome. To our knowledge, no one has reported the combination of Horner syndrome and ipsilateral abduction deficit in GCA. We were unable to perform the hydroxyamphetamine test to confirm the location of the lesion. The absence of brainstem and spinal cord signs and a normal brain MRI argue against a preganglionic location. Hollenhorst et al ( 4) suggested that paresis of different extraocular muscles at different times is the result of ischemia of the muscles or the nerve and not a brainstem lesion. Barricks et al ( 5) found extraocular muscle ischemia at autopsy in a patient with FIG. 2. Temporal artery biopsy shows inflammatory cells in the vessel wall. Inset shows giant cells ( arrows). J Neuro- Ophthalmol, Vol. 26, No. 3, 2006 231 J Neuro- Ophthalmol, Vol. 26, No. 3, 2006 Letters to the Editor bilateral ophthalmoplegia resulting from GCA. Sibony and Lessell ( 6) reported aberrant regeneration in a patient with pupil- sparing third cranial nerve palsy resulting from GCA and suggested that ophthalmoplegia is neurogenic rather than myogenic. Meadows ( 7) suggested ischemia of vasa nervorum, whereas Martin ( 8) suggested ischemia of the extraocular muscles as the likely cause of diplopia in GCA. In our patient, the combination of Horner syndrome and an ipsilateral abduction deficit suggests a cavernous sinus lesion. Within the cavernous sinus, the postganglionic sympathetic fibers travel with the sixth cranial nerve lateral to the internal carotid artery. Bromfield and Slakter ( 3) proposed that granulomatous inflammation of the internal carotid artery might directly involve the sympathetic fibers. We believe that the sixth cranial nerve in our patient could have been similarly affected. Guruswami Arunagiri, MD Shanmugam Santhi, MD Thomas Harrington, MD Departments of Ophthalmology and Rheumatology Geisinger Medical Center Danville, PA REFERENCES 1. Bell TA, Gibson RA, Tullo AB. A case of giant cell arteritis and Horner's syndrome. Scott Med J 1980; 25: 302. 2. Askari A, Jolobe OM, Shepherd DI. Internuclear ophthalmoplegia and Horner's syndrome due to presumed giant cell arteritis. J R Soc Med 1993; 86: 362. 3. Bromfield EB, Slakter JS. Horner's syndrome in temporal arteritis. Arch Neurol 1988; 45: 604. 4. Hollenhorst RW, Brown JR, Wagener HP, et al. Neurologic aspects of temporal arteritis. Neurology 1960; 10: 490- 8. 5. Barricks ME, Traviesa DB, Glaser JS, et al. Ophthalmoplegia in cranial arteritis. Brain 1977; 100: 209- 21. 6. Sibony PA, Lessell S. Transient oculomotor synkinesis in temporal arteritis. Arch Neurol 1984; 4: 87- 8. 7. Meadows SP Temporal or giant cell arteritis- ophthalmic aspects. In: Smith JL, ed. Neuro- Ophthalmology Symposium of the University of Miami and Bascom Palmer Eye Institute, vol IV St. Louis: Mosby; 1968: 148- 57. 8. Martin EA. Double vision and temporal arteritis. J Ir Med Assoc 1970; 63: 191- 3. Mydriasis and Accommodative Failure From Exposure to Topical Glyco pyrrol ate Used in Hyperhidrosis We report a case of mydriatic pupils and accommodative failure caused by exposure to glycopyrrolate cream 0.5% ( Robinul, Antigen Pharmaceuticals, Goldshield PLC, Croydon, UK) used in treatment of axillary hyperhidrosis. The connection was not realized until much later because no one thought to ask about the use of this agent. We are unaware of previous reports associating this agent with these findings. A 19- year- old nursing student consulted her primary care physician for a 1- day history of blurred vision and heaviness in the left eye. There was no associated headache or other pertinent neurologic history. She had migraine and asthma and was using salbutamol inhalers. She denied the use of recreational drugs or any exposure to pharmacologic agents as a possible contamination source. Ophthalmologic examination was normal except that the left pupil was dilated and did not constrict to light or a near target. Slit lamp examination was unremarkable; no sectoral paralysis or vermiform movement was noted. The pupil did not constrict after instillation of 0.125% pilocarpine. Neurologic examination was normal. The mydriasis was attributed to migraine. On two return visits, she was asymptomatic and the pupils were normal. However, she returned a fourth time with recurrence of symptoms. On that occasion, she had a fixed, fully dilated left pupil and a mid- dilated right pupil demonstrating some constriction to light. Neither pupil constricted after instillation of 0.125% or 4% pilocarpine. On further questioning, we discovered that she had axillary hyperhidrosis and was regularly applying 0.5% glycopyrrolate cream before applying makeup. We reasoned that the pupil abnormality was the result of periocular contamination with the glycopyrrolate cream. After she stopped applying the cream, her pupils returned to normal within 1 week and she has been symptom- free ever since. Primary hyperhidrosis is a disorder of excessive sweating. Treatment options range from antiperspirants or anticholinergics to iontophoresis, botulinum toxin injection, and thoracic sympathectomy in severe cases ( 1,2). The antimuscarinic properties of glycopyrrolate reduce sweating. The mydriatic effect of 0.5% topical glycopyrrolate drops has been tested in animal eyes in which the pupil dilated within 5 minutes of application, reaching near-maximal levels by 15 minutes. These effects were faster, stronger, and more persistent than those of 1% atropine and lasted 1 week after initial application ( 3). The mydriatic effects of glycopyrrolate have also been demonstrated with its use as an anticholinergic agent in general anesthesia ( 4,5). However, to our knowledge, this is the first case demonstrating these effects with topical glycopyrrolate in humans. Physicians and patients should be aware of the potential side effects of mydriasis and accommodative failure with the use of topical glycopyrrolate and of the 232 © 2006 Lippincott Williams & Wilkins Letters to the Editor J Neuro- Ophthalmol, Vol. 26, No. 3, 2006 importance of careful hygiene after its application so as not to contaminate the periocular skin or ocular surface. Shahrnaz Izadi, MBChB, BSc Hons Anshoo Choudhary, MRCS ( Ed), MS William Newman, MB FRCS ( Glasg) FRCOphth St. Paul's Eye Unit Royal Liverpool University Hospital Liverpool, United Kingdom william. newman@ nhs. net REFERENCES 1. Eisenach JH, Atkinson JL, Fealey RD. Hyperhidrosis: evolving therapies for a well- established phenomenon. Mayo Clin Proc 2005; 80: 657- 66. 2. Collin J, Whatling P. Treating hyperhidrosis. Surgery and botulinum toxin are treatments of choice in severe cases. BMJ 2000; 320: 1221- 2. 3. Varssano D, Rothman S, Haas K, et al. The mydriatic effect of topical glycopyrrolate. Graefes Arch Clin Exp Ophthalmol 1996; 234: 205- 7. 4. Greenan J, Prasad I Comparison of the ocular effects of atropine or glycopyrrolate with two IV induction agents. Br J Anaesth 1985; 57: 180- 3. 5. Schwartz H, Apt L. Mydriatic effect of anticholinergic drugs used during reversal of nondepolarizing muscle relaxants. Am J Ophthalmol 1979; 88: 609- 12. Mechanism of Bitemporal Hemianopia The article entitled " A Mechanical Theory to Account for Bitemporal Hemianopia From Chiasmal Compression" that appeared in the March 2005 issue of the Journal ( 1) has stimulated discussion among us. The " structural collapse theory" does not explain why neural transmission in the distorted nerve fiber may remain disrupted for many years yet be reversed in a matter of hours or days as seen in patients after decompression. We suggest that the model may better explain the mechanism of injury in patients with traumatic chiasmal syndrome in which the bitemporal hemianopia is permanent. Various theories exist to account for the mechanism of injury in traumatic chiasmal syndrome ( 2- 5), but none completely explains the relative sparing of uncrossed fibers. It has been suggested that a severe frontal head injury may cause separation of the skull in the midline with resultant sagittal tearing of the chiasm ( 2); however, not all injuries causing traumatic chiasmal syndrome are frontal. Rand ( 6) stated that the optic chiasmal nasal fibers are more prone to damage as they are " weaker." Perhaps a head injury produces intense, if brief, compression leading to selective disruption of crossing fibers, which lack the frictional support of contact with surrounding fibers as described in your model. Shveta Bansal, MRCS, Ed Nishant Kumar, MRCOphth Royal Liverpool University Hospital Liverpool, United Kingdom shveta@ dr. com Graham Kyle, FRCOphth Walton Hospital Liverpool, United Kingdom REFERENCES 1. Mcllwaine GG, Carrim ZI, Lueck CJ, et al. A mechanical theory to account for bitemporal hemianopia from chiasmal compression. JNeuroophthalmol 2005; 25: 40- 3. 2. Hassan A, Crompton JL, Sandhu A. Traumatic chiasmal syndrome: a series of 19 patients. Clin Exp Ophthalmol 2002; 30: 273- 80. 3. Heinz GW, Nunery WR, Grossman CB. Traumatic chiasmal syndrome associated with midline basilar skull fractures. Am J Ophthalmol 1994; 117: 90- 6. 4. Savino PJ, Glaser JS, Schatz NJ. Traumatic chiasmal syndrome. Neurology 1980; 30: 963- 70. 5. Tang RA, Kramer LA, Schiffman J, et al. Chiasmal trauma: clinical and imaging considerations. Surv Ophthalmol 1994; 38: 381- 3. 6. Rand CW. Chiasmal injury complicating fracture of the skull. Bull Los Angeles Neurol Soc 1937; 2: 91^. Authors' Reply: Our article is not trying to make claims of the material performance of nerve fibers. We were simply asserting that as a result of the concentrated loading experienced in the crossover nerve fibers, the tubes could " nip" and disrupt signals. FIG. 1. A comparison of the different deforming actions of compression applied to crossing ( perpendicular) and non-crossing ( parallel) fibers. D, diameter of axon and length of nerve segment; p, proportion of circumference flattened by pressure ( Reprinted from reference 1). 233 J Neuro- Ophthalmol, Vol. 26, No. 3, 2006 Letters to the Editor B FIG. 2. The degree of flattening has progressed to the point at which the area of contact between crossing and non-crossing cylinders is equal ( see Figure 3). " Elastic buckling" is a state in which the material remains within its elastic behavioral zone and, when unloaded, returns to its undamaged state. A good example would be a Bunsen burner tube. We do not know the material properties of the nerve fiber well enough to be definitive. However, if it were elastic, we may expect retransmission of signals once the loading is removed provided the ionic link can be reestablished. Furthermore, it cannot be presumed that nerve fiber malfunction occurs after the point of irreversible distortion/ collapse. Indeed, the clinical recovery after chiasmal decompression would suggest that the point of nerve fiber malfunction occurs before irreversible distortion/ collapse. With increasing compression, the proportion of the circumference flattened will increase. The area of flattening will change from that in Figure 1 to that in Figure 2. The formula 1/ TT X p ( Fig. 1) dictates that if p = 1/ TT, then the pressure difference between crossing and non- crossing cylinders is equal ( Figs. 2 and 3). We proposed that it seems unlikely that the nerve fibers could adopt the configuration shown in Figures 2 and 3 without further collapse or malfunctioning. It seems quite possible that a nerve fiber that has adopted the shape in Figure 3 could spontaneously and relatively rapidly return to its normal shape. Therefore, we see no reason why a physically distorted cylinder such as a nerve fiber should not return to its previous shape and achieve normal function within hours/ days of cessation of a compressive force. In addition, there may also be an element of biologic repair. p = 0 p = 1/ 71 FIG. 3. Deformation of two cylinders ( representing stylized axons) compressed against each other. Before compression starts ( A), neither cylinder is deformed. As the cylinders are compressed against each other ( B), there is flattening of the interface ( shown in blue), which occupies a fraction, p, of the circumference. When the length of the interface reaches the diameter of the circle, p = 1/ TT ( Reprinted from reference 1). Gawn G. Mcllwaine, FRCS, FRCOphth Zia I. Carrim, MB, ChB Christian J. Lueck, PhD, FRCP ( UK), FRCP ( Ed), FRACP T. Malcolm Chrisp, PhD Western General Hospital Edinburgh, Scotland Southern General Hospital Glasgow, Scotland The Canberra Hospital and Australian National University Canberra, Australia Heriot- Watt University Edinburgh, Scotland Gawn. McIlwaine@ mater. n- i. nhs. uk REFERENCE 1. Mcllwaine GG, Carrim ZI, Lueck CJ, et al. A mechanical theory to account for bitemporal hemianopia from chiasmal compression. JNeuroophthalmol 2005; 25: 40- 3. 234 © 2006 Lippincott Williams & Wilkins |
