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Acquired Segmental Iris Dilator Muscle Synkinesis CASE REPORTS AND SMALL CASE SERIES within a few seconds. The iris ap- bers. The swallowing disorder, the Acquired Segmental Iris peared to tighten radially at the 7:30- flacidity of the soft palate, and the Dilator Muscle Synkinesis and 1:30-o’clock positions. When we remaining deviation of the ex- Due to Deglutition examined the patient at 10 years of tended tongue indicate involve- age we confirmed these findings. A ment of hypoglossal and glossopha- Congenital ocular “misdirection dys- right-sided ptosis, facial vasodys- ryngeal fibers. We assume that the kinesis” typically involves multiple regulation (flushed left side of the origin of the misinnervation was va- cranial nerves. Acquired peripheral face, pale right side of the face after gal, because the pupillary distor- misdirection usually occurs in the exercise), and anhidrosis (from fore- tion could be elicited only by drink- same nerve; less frequently, more head to larynx region; iodine starch ing, when permanent, significant than one nerve are involved. We de- reaction) were present, but not iris esophageal peristalsis is required; but scribe an unusual synkinesis of the heterochromia. The right pupil di- not by lower swallowing frequency iris dilator muscle due to degluti- lated poorly on instillation of 4% co- (eating) and low volume load (sa- tion, presumably caused by post- caine hydrochloride and 5% phole- liva) at the same swallowing fre- traumatic aberrant outgrowth of va- drine formate (equivalent to 1% quency as when the child was drink- gal nerve fibers to the cervical hydroxyamphetamine hydrobro- ing. That is, the pupillary distortion sympathetic chain. mide; Figure 1). was elicited mainly by the volun- tary sequence of the swallowing act, Report of a Case. We describe an Comment. The findings indicated a which requires activity of the hypo- unusual synkinesis in a 10-year- lesion of the postganglionic (third glossal and glossopharyngeal nerves. old boy. On the sixth day of life a order) sympathetic neuron, either Descending vagal fibers in the neuroblastoma was removed from primary or by transsynaptic degen- neck are adjacent to the fibers of the the right side of his neck. A right- eration. Some axons of the postgan- ascending sympathetic chain sided Horner syndrome and pare- glionic neuron must have been in- (Figure 2). If these fibers are in- sis of the recurrent laryngeal nerve tact, because the synkinesis requires jured at the same time, aberrant va- occurred postoperatively, as well as adrenergic innervation of the iris di- gal sprouts can grow into the cervi- a flacidity of the right soft palate, a lator muscle. There is a close topo- cal sympathetic path.2 Since the deviation of the tongue to the right graphic relationship among the sym- vagal sprouts are cholinergic, some side, and an atony of the esophagus pathetic superior cervical ganglion, of them may be lost owing to phar- and the stomach that regressed the inferior ganglion of the vagus macological incompatibility. But, if within 2 weeks. When the child was nerve, the hypoglossal nerve, and they reach sympathetic ganglion 2 years old, the parents recognized the glossopharyngeal nerve.1 The cells in the superior cervical gan- that his right pupil became dis- esophagus and stomach atony and glion, they may make a permanent— torted when he was drinking and af- the recurrent laryngeal nerve pare- albeit inappropriate—cholinergic terward regained its round shape sis indicate an injury to vagal fi- connection. If these sprouting fi- Figure 1. Right-sided Horner syndrome, without swallowing (A), with pupillary distortion due to swallowing (B), 30 minutes after instilling 5% pholedrine formate in both eyes (an identical anisocoria was present after instilling 4% cocaine hydrochloride in both eyes) (C), and 15 minutes after instilling 5% phenylephrine hydrochloride in both eyes (D). Pupillary diameter expressed in millimeters for the right eye/left eye is as follows: (A) 4.5/5.2, (B) 7.334.1/5.2, (C) 4.5/7.4, and (D) 7.4/7.4. ARCH OPHTHALMOL / VOL 116, FEB 1998 248 ©1998 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 10/02/2021 Figure 2. Topographic relationship of the 7th, 9th, 10th, and 12th cranial nerves, the inferior ganglion of the 10th nerve (IGV), and the superior cervical ganglion (SCG) in the retropharyngeal space.3 IC and EC indicate Figure 1. Left sympathizing eye, 1 year after onset of sympathetic ophthalmia. A yellowish macular lesion internal and external carotid arteries, with focal intraretinal hemorrhage is shown with a surrounding ring of hyperpigmentation and circinate respectively. hard exudates, clinically consistent with choroidal neovascularization. bers are to survive, they must still be connected to their original vagal ganglion cells. Brigitte I. Boehme, MD Michael H. Graef, MD Giessen, Germany Reprints: Michael H. Graef, MD, Uni- versity of Giessen, Department of Stra- bismology and Neuroophthalmol- ogy, Friedrichstrasse 18, D-35385 Giessen, Germany. 1. Miller NR. Walsh and Hoyth’s Clinical Neuro- ophthalmology. Baltimore, Md: Williams & Wilkins; 1985;2:425. 2. Loewenfeld IE. The Pupil: Anatomy, Physiology, and Clinical Applications. Detroit, Mich: Wayne State University Press; 1993:520-627. 3. Platzer W. Taschenatlas der Anatomie. Stuttgart, Figure 2. Left eye, 3 months after commencing cyclosporine (cyclosporin A) therapy, showing resolution Germany: Georg Thieme Verlag; 1979;1:343. of macular edema and hemorrhage. lustrating resolution of CNV in a ity of finger counting OD and 20/30 child with active refractory sympa- OS while receiving a maintenance Cyclosporine-Induced thetic ophthalmia after starting cy- dose of 1 mg/kg on alternate days. Resolution of Choroidal closporine therapy. Funduscopy of the left eye revealed Neovascularization mild vitritis and a yellowish macu- Associated With Report of a Case. A 3-year-old boy lar lesion, with a surrounding hyper- Sympathetic Ophthalmia had a limbal rupture involving uveal pigmented ring and circinate hard prolapse of his right eye after he fell exudates, which was clinically con- Choroidal neovascularization (CNV) on his feeder cup. Primary repair was sistent with CNV (Figure 1). De- is a sight-threatening complication performed and postoperative visual spite systemic prednisone therapy, of sympathetic ophthalmia, a clas- acuity was 20/80 OD and 20/20 OS. his visual acuity worsened to 20/200 sic example of endogenous poste- When sympathetic ophthalmia de- OS 10 months later. Systemic cy- rior uveitis.1,2 Cyclosporine (cyclo- veloped 4 months later, his visual closporine therapy was added at 5 sporin A) has been shown to be acuity deteriorated to finger count- mg/kg per day. Three months later, effective in the treatment of sight- ing in both eyes. Oral prednisone visual acuity improved to 20/100 OD threatening disease associated with therapy at 1 mg/kg per day was and 20/60 OS with resolution of left active intraocular inflammation in started. His uveitis improved slowly; macular edema and hemorrhage adults.3 We present a case report il- 1 year later he achieved a visual acu- (Figure 2). ARCH OPHTHALMOL / VOL 116, FEB 1998 249 ©1998 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 10/02/2021 Report of a Case. A 24-year-old woman had been diagnosed with abetalipoproteinemia at age 2 years. The diagnosis was based on periph- eral blood acanthocytosis, low se- rum cholesterol levels, absence of se- rum lipoprotein, and characteristic lipid deposits in mucosal cells on small bowel biopsy. She was treated with vitamin E injection and oral vi- tamins A, D, and K. Results of ini- tial ocular examinations were within normal limits. At age 18 years, the patient vol- untarily discontinued treatment with vitamin supplementation; after- ward she reported worsening of night vision and progressive field changes. She was evaluated at the Figure 3. Left eye, after 25 months of cyclosporine (cyclosporin A) therapy, with complete resolution of Ocular Genetics Clinic of The Hos- macular edema and ring of hyperpigmentation, indicating resorption of subretinal blood and complete regression of choroidal neovascularization. A residual atrophic macular scar remains. pital for Sick Children, Toronto, Ontario, at age 24 years. Best- corrected vision was 20/20 OU. Re- Visual improvement continued, due 1. Chew EY, Crawford J. Sympathetic ophthalmia fraction was −0.75+0.503165° OD to vitritis remission and complete and choroidal neovascularization. Arch Ophthal- and −1.00+1.003175° OS. Ophthal- mol. 1988;106:1507-1508. CNV regression (Figure 3). 2. Carney MD, Tessler HH, Peyman GA, Gold- moscopy showed bilateral and sym- Cyclosporine therapy was with- berg MF, Williams DP. Sympathetic ophthal- metric helicoid peripapillary changes mia and subretinal neovascularization. Ann Oph- (Figure 1), equatorial retinal pig- drawn 25 months after commence- thalmol. 1990;22:184-186. ment. Then the patient’s visual 3. Nussenblatt RB, Palestine AG, Chan CC. Cyclo- ment epithelium (RPE) mottling, acuity was 20/100 OD and 20/30 sporin A therapy in the treatment of intraocu- and attenuation of blood vessels lar inflammatory disease resistant to systemic cor- but absence of true angioid streaks. OS despite a residual atrophic left ticosteroids and cytotoxic agents. Am J macular scar. Ophthalmol. 1983;96:275-282 There was no sign of inflammation. 4. Pivetti-Pezzi P, Accorinti M, Abdulaziz MA, Fluorescein angiography showed La Cava M, Torella M, Riso D. Behc¸et’s disease Comment. Sympathetic ophthal- in children. Jpn J Ophthalmol. 1995;39:309- a large peripapillary defect with mia–induced CNV has been re- 314. no evidence of leakage or angioid 1,2 5. Tugal-Tutkun I, Havrlikova K, Power WJ, Fos- streaks (Figure 2). A Goldmann ported twice, to our knowledge, ter CS. Changing patterns in uveitis of child- both cases in children. Sympa- hood. Ophthalmology. 1996;103:375-383. visual field examination revealed thetic ophthalmia–induced CNV bilateral constriction of the periph- regression may have been sponta- eral field and enlargement of the neous,1 but is not described when blind spot.
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