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Short communication

Adie’s pupil following Le Fort I maxillary osteotomy: case report Anna Sayan a,∗ , Abeysinghe H.M.K. Abeysinghe b , Ilanko Ilankovan a a b

Poole Hospital NHS Foundation Trust University of Peradeniya

Accepted 22 May 2014

Abstract Complications after Le Fort I osteotomy are rare. We report, to our knowledge, the first case of bilateral Adie’s pupil after Le Fort I osteotomy. Crown Copyright © 2014 Published by Elsevier Ltd. on behalf of The British Association of Oral and Maxillofacial Surgeons. All rights reserved.

Keywords: Le Fort I osteotomy; Adie’s pupil

Introduction Le Fort I osteotomy is a standard operation to correct maxillary disproportions, and complications are rare. Orbital complications previously described involve fractures that extended to the orbital apex, and injury to the III and VI cranial nerves.1–2 To our knowledge this is the first report of a case of bilateral Adie’s pupil after Le Fort I osteotomy.

Case report A 30-year-old white woman was listed for Le Fort I advancement osteotomy to correct maxillary hypoplasia. Preoperative results of biochemical and haematological investigations were all within normal ranges, including the clotting profile. Standard perioperative preparations were made at the start of the procedure including the placement of protective eye shields. Bone was cut using drills, a saw, and ∗

Corresponding author. fax: +01202 448410. E-mail addresses: [email protected], [email protected] (A. Sayan).

fine osteotomes. The pterygoid disjunction was achieved with a Tessier osteotome. The maxilla was down-fractured, advanced, and stabilised with 4 x 1.5 mm titanium plates. Subsequently, the mucosa was repaired in the usual manner. When routine postoperative checks were made at the end of the operation, both eye shields were removed and the pupils were asymmetrical: the left was larger than the right and there was no reaction to light stimulation. About 30 minutes after operation further ocular examination found that the left pupil measured 9 mm and the right 6 mm. The patient complained of blurred vision but had no pain or motility disorder. There was no direct or consensual light reflex in the left eye and a sluggish response in the right. An urgent computed tomogram showed no injury to the orbital apex. An ophthalmological opinion was sought and examination confirmed the measurements of the pupils. Visual acuity was 6/4 unaided in both eyes and there was no motility disorder. Examination of the lids, conjunctiva, cornea, lens, and iris, were normal. There was no response to light stimulation. The anterior chamber was quiet and deep. Intraocular pressure was 13 for the left eye and 12 for the right. Both retinas appeared to be normal. Drops of 0.1% pilocarpine

http://dx.doi.org/10.1016/j.bjoms.2014.05.012 0266-4356/Crown Copyright © 2014 Published by Elsevier Ltd. on behalf of The British Association of Oral and Maxillofacial Surgeons. All rights reserved.

Please cite this article in press as: Sayan A, et al. Adie’s pupil following Le Fort I maxillary osteotomy: case report. Br J Oral Maxillofac Surg (2014), http://dx.doi.org/10.1016/j.bjoms.2014.05.012

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were instilled in both eyes and both pupils responded; there was greater constriction in the left than in the right. A diagnosis of bilateral Adie’s pupil was made and the patient recovered fully within 48 hours.

Discussion Adie’s pupil is a neurological condition of unknown origin that has an unusual presentation known as anisocoria (inequality in the size of the pupils). It is thought to result from damage to the innervation of the ciliary muscle or a because of a problem in the ciliary ganglion, and commonly affects women aged between 20 and 40 years. Presentation is bilateral in 20% of cases. When deep tendon reflexes of the legs are also affected, and accompanying symptoms include localised areas of skin that do not sweat, hypertension, and unsteady heart rhythms, the condition is called Adie syndrome.3 Severe orbital trauma can cause Adie’s pupil. Systemic diseases such as sarcoidosis, viral infection, ischaemia, syphilis, and tumour involvement can also cause pupillary abnormality,4 but they were absent in our case. The only relevant clinical factor was the planned hypotensive anaesthesia during Le Fort I maxillary osteotomy. A reduction in blood pressure can potentially reduce the blood supply to peripheral structures such as the ciliary ganglion, and this may have caused the pupillary manifestation. The case described by Sirikumara and Sugar failed to identify this as a possible explanation.5 The diagnostic test is a rapid response to the instillation of 0.1% pilocarpine eye drops, which in our case

was positive. Although the response in the right eye was not as pronounced as the left, the ophthalmological opinion was that it was a bilateral problem, and the left eye presented more features than the right. The incidence of Adie’s pupil is rare in the general population (4.7/100 000), and the age range of those affected is between 20 and 40 years,3 which is the age at which patients would normally have orthognathic surgery. It is not possible to recommend preoperative ophthalmic assessment or to inform patients about the possibility of this type of occurrence during the process of obtaining informed consent, but the knowledge is of paramount importance to clinicians so that a diagnosis can be made without undue concerns. Lastly, in our case, bilateral presentation supports the hypothesis that selective hypotension may have been the cause.

References 1. Carr RJ, Gilbert P. Isolated partial third nerve palsy following Le Fort I maxillary osteotomy in a patient with cleft lip and palate. Br J Oral Maxillofac Surg 1986;24:206–11. 2. Watts PG. Unilateral abducent nerve palsy: a rare complication following a Le Fort I maxillary osteotomy. Br J Oral Maxillofac Surg 1984;22: 212–5. 3. Thompson HS. Adie’s syndrome: some new observations. Trans Am Ophthalmol Soc 1977;75:587–626. 4. Bell RA, Thompson HS. Ciliary muscle dysfunction in Adie’s syndrome. Arch Ophthalmol 1978;96:638–42. 5. Sirikumara M, Sugar AW. Adie’s pupil following Le Fort I maxillary osteotomy. A complication or coincidence? Br J Oral Maxillofac Surg 1990;28:306–8.

Please cite this article in press as: Sayan A, et al. Adie’s pupil following Le Fort I maxillary osteotomy: case report. Br J Oral Maxillofac Surg (2014), http://dx.doi.org/10.1016/j.bjoms.2014.05.012

Adie's pupil following Le Fort I maxillary osteotomy: case report.

Complications after Le Fort I osteotomy are rare. We report, to our knowledge, the first case of bilateral Adie's pupil after Le Fort I osteotomy...
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