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atrophy, and fibrosis. The reference lists of relevant articles were also reviewed. References 1. Ugalde I, Christiansen SP, McLoon LK. Botulinum toxin treatment of extraocular muscles in rabbits results in increased myofiber remodeling. Invest Ophthalmol Vis Sci 2005;46:4114-20. 2. Spencer RF, McNeer KW. Botulinum toxin paralysis of adult monkey extraocular muscle. Structural alterations in orbital, singly innervated muscle fibers. Arch Ophthalmol 1987;105:1703-11. 3. Ohtsuki H, Hasebe S, Okano M, Furuse T. Morphological changes in the orbital surface layer muscle of the rabbit eye produced by botulinum toxin. Ophthalmologica 1998;212:53-60. 4. Borodic GE, Ferrante R. Effects of repeated botulinum toxin injections on orbicularis oculi muscle. J Clin Neuroophthalmol 1992;12:121-7. 5. Porter JD, Strebeck S, Capra NF. Botulinum-induced changes in monkey eyelid muscle. Comparison with changes seen in extraocular muscle. Arch Ophthalmol 1991;109:396-404. 6. Mohan M, Tow S, Fleck BW, Lee JP. Permanent extraocular muscle damage following botulinum toxin injection. Br J Ophthalmol 1999; 83:1309-10. 7. McKelvie P, Friling R, Davey K, Kowal L. Changes as the result of ageing in extraocular muscles: a post-mortem study. Aust N Z J Ophthalmol 1999;27:420-25. 8. Scott AB. Change of eye muscle sarcomeres according to eye position. J Pediatr Ophthalmol Strabismus 1994;31:85-8. 9. Olabisi R, Chamberlain CS, Petr S, Steiner S, Consigny D, Best TM, et al. The effects of botulinum toxin A on muscle histology during distraction osteogenesis. J Orthop Res 2009;27(3):310-17.
Ophthalmomyiasis interna masquerading as orbital cellulitis Simon S. M. Fung, MA, FRCOphth,a Stephanie J. West, BM, FRCOphth,a,b and Anthony T. Moore, MA, FRCOphtha,c Ophthalmomyiasis interna posterior is a rare condition caused by the invasion of the ocular posterior segment by the larval form of certain flies in the order Diptera. The main clinical features are reduced visual acuity, intense posterior uveitis, and neuroretinitis. Treatment of this condition is predominantly surgical, including argon laser photocoagulation for subretinal larva and pars plana vitrectomy for intravitreal infection. We report a case of ophthalmomyiasis interna posterior in a 12-year-old girl that masqueraded as orbital cellulitis and was successfully treated with Nd:YAG laser photodisruption.
Author affiliations: aMoorfields Eye Hospital NHS Foundation Trust, London, England; b University Hospital Southampton NHS Foundation Trust, Southampton, England; c UCSF Benioff Children’s Hospital, UCSF Medical Center (Mission Bay), San Francisco, California Submitted February 23, 2016. Revision accepted July 15, 2016. Published online October 31, 2016. Correspondence: Simon S. M. Fung, MA, FRCOphth, Moorfields Eye Hospital NHS Foundation Trust, 162 City Road, London, England, EC1V 2PD (email: Simon.Fung@ Moorfields.nhs.uk). J AAPOS 2016;20:546-548. Copyright Ó 2016, American Association for Pediatric Ophthalmology and Strabismus. Published by Elsevier Inc. All rights reserved. 1091-8531/$36.00 http://dx.doi.org/10.1016/j.jaapos.2016.07.228
FIG 1. Fundus image of the left eye at presentation, showing optic nerve head swelling, retinal hemorrhages, and marked exudative retinal detachment, which caused the central defocus.
Case Report A 12-year-old girl presented for emergent care at Moorfields Eye Hospital, London, with a 3-day history of reduced vision, diplopia, and swollen upper lid of her left eye. She had recently returned from the Serengeti, Tanzania, where her left eyelid had been bitten by an insect. A local ophthalmologist had diagnosed preseptal cellulitis. She had no previous medical history and was not known to have any allergies. On examination, corrected distance visual acuity was 0.00 logMAR in the right eye and 0.40 logMAR in the left eye. No relative afferent pupillary defect was detected, and optic nerve function was preserved. There was diffuse palpebral edema without erythema of the left upper lid. The left globe was proptosed by 3 mm, and she had a left exotropia and hypotropia in primary position, with limited abduction and elevation. Intraocular pressure in the left eye was mildly raised (26 mm Hg), with a quiet anterior chamber. Dilated fundus examination showed left optic nerve head swelling, retinal hemorrhages, and marked exudative retinal detachment involving the fovea without vitreous inflammation (Figure 1). The child was admitted to Great Ormond Street Hospital, London, with a presumptive diagnosis of infective orbital cellulitis and secondary neuroretinitis. She was started on intravenous ceftriaxone, flucloxacillin and metronidazole, oral prednisolone (20 mg twice daily), and topical dexamethasone 0.1% drops four times daily. Subsequent computed tomography and magnetic resonance imaging scans showed optic nerve sheath enhancement but no retrobulbar lesions nor venous congestions. Blood tests, including white cell count, inflammatory markers, autoimmune serology, and blood cultures were all negative. Despite improvement of palpebral swelling and proptosis with initial treatment, her left corrected distance
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FIG 2. A, Left inferiortemporal fundus photograph, day 6, showing a curvilinear hypopigmented track (arrows) and a white elevated lesion (arrowhead). B, the same area shown on B-mode ultrasound, showing intraretinal thickening (arrowhead). C, Optical coherence tomography, day 7, revealed a larva penetrating the retina and entering the vitreous cavity (arrow).
visual acuity deteriorated to counting fingers by day 6. Review of the left fundus showed persisting neuroretinitis and a curvilinear hypopigmented track inferotemporally (Figure 2A). A highly echogenic intraretinal mass was detected along the track on B-mode ultrasound (Figure 2B). The suspicion of ophthalmomyiasis was raised, and a white larva was visualized in the area on day 7 with movement confirmed on serial photographs. Optical coherence tomography located the larva at the vitreoretinal interface (Figure 2C). A single dose (6 g) of ivermectin was administered, but the larva was unaffected when the child was reexamined the next day. The patient was reviewed by the vitreoretinal service, and it was decided to treat with Nd:YAG laser photodisruption. The laser was focused at the larval head and 3 6– 8 mJ laser shots were fired. The larval head disintegrated on the third shot, and the organism ceased to move. On the next day, the patient’s visual acuity and level of intraocular inflammation remained stable. Systemic and topical corticosteroids were tapered over the next 6 weeks and vi-
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sual acuity and fundal appearance gradually improved over the course of 3 months. The patient was discharged at 8 months posttreatment with a corrected distance visual acuity of 0.06 logMAR in the left eye (eFigure 1).
Discussion Ophthalmomyiasis is a rare spectrum of disorders caused by larval infestation in the ocular and periocular region. Larval infestation of the adnexa and the orbit causes ophthalmomyiasis externa. Intraocular larval infestation is known as ophthalmomyiasis interna. This is further divided into ophthalmomyiasis interna anterior and ophthalmomyiasis interna posterior (OIP) depending on the intraocular segment that is affected. Children appear to be more susceptible than adults, and the diagnosis of OIP should be considered particularly in the presence of subretinal tracks.1 OIP is a rare cause of uveitis, with the commonest clinical presentations being visual loss, ocular pain, red eye,
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and floaters.2 Georgalas and colleagues3 reported a case of OIP in a 27-year-old presenting with visual loss, ocular pain, and limitation of eye movements, who was initially diagnosed with orbital cellulitis. To our knowledge, ours is the first report of pediatric OIP masquerading as a presumably reactive orbital cellulitis. There are features distinguishing an infective and a reactive orbital cellulitis, such as presence of pyrexia, erythema, tenderness, and systemic clinical signs in the former. Currently, no specific blood investigation can distinguish OIP from other diagnoses. Treatment of OIP is varied as it depends on the viability and the location of the larva and the degree of associated inflammation. Medical treatment is limited: there is 1 case report of successful single-dose ivermectin treatment of subretinal OIP in a 11-year-old boy4; however, in our case, this was not helpful. Surgical treatment includes argon laser photocoagulation and pars plana vitrectomy (PPV), which have been shown to be effective treatments for subretinal and intravitreal larvae, respectively.5,6 Our case is unique in that the larva was at the vitreoretinal interface in an anterior–posterior orientation. If argon laser photocoagulation had been used, it would have required a large number of laser shots precisely aimed at the head in order to produce sufficient thermal damage to destroy the larva. Misdirected argon laser could penetrate deep into the retinal pigment epithelium, risking possible choroidal neovascular membrane in the future. PPV in our case would have been technically challenging and may have led to complications such as retinal detachment and subretinal hemorrhage, possibly jeopardizing the long-term visual outcome. Nd:YAG laser is noninvasive and has been extensively used for iridotomy and capsulotomy as well as vitreolysis to relieve uveal effusions and subhyaloid vitreous hemorrhages.7,8 It has also been used in 1 adult case of intravitreal larva with good result.3 We considered Nd:YAG laser a safe mode of treatment in our patient; selective larval destruction was achieved without any complications. It is important to note that high-dose systemic corticosteroid with a slow taper postoperatively was given for the macular edema as well as to prevent the development of posterior uveitis.
Literature Search MEDLINE was searched using the following terms and combinations, with no other exclusion criteria: ophthalmomyiasis, ophthalmomyiasis interna, posterior; ophthalmomyiasis AND interna AND posterior AND (orbital cellulitis [MeSH Terms] OR orbital AND cellulitis OR orbital cellulitis).
References 1. Gass JD, Lewis RA. Subretinal tracks in ophthalmomyiasis. Arch Ophthalmol 1976;94:1500-505.
Volume 20 Number 6 / December 2016 2. Lagace-Wiens PR, Dookeran R, Skinner S, Leicht R, Colwell DD, Galloway TD. Human ophthalmomyiasis interna caused by Hypoderma tarandi, Northern Canada. Emerg Infect Dis 2008;14:64-6. 3. Georgalas I, Ladas I, Maselos S, Lymperopoulos K, Markomichelakis N. Intraocular safari: ophthalmomyiasis interna. Clin Exp Ophthalmol 2011;39:84-5. 4. Taba KE, Vanchiere JA, Kavanauge AS, Lusk JD, Smith MB. Successful treatment of ophthalmomyiasis interna posterior with ivermectin. Retin Cases Brief Rep 2012;6:91-4. 5. Phelan MJ, Johnson MW. Acute posterior ophthalmomyiasis interna treated with photocoagulation. Am J Ophthalmol 1995;119:106-7. 6. Slusher MM, Holland WD, Weaver RG, Tyler ME. Ophthalmomyiasis interna posterior. Subretinal tracks and intraocular larvae. Arch Ophthalmol 1979;97:885-7. 7. Debrouwere V, Stalmans P, Van Calster J, Spileers W, Zeyen T, Stalmans I. Outcomes of different management options for malignant glaucoma: a retrospective study. Graefes Arch Clin Exp Ophthalmol 2012;250:131-41. 8. Ulbig MW, Mangouritsas G, Rothbacker HH, Hamilton AM, McHugh JD. Long-term results after drainage of premacular subhyaloid hemorrhage into the vitreous with a pulsed Nd:YAG laser. Arch Ophthalmol 1998;116:1465-9.
Bilateral retinoblastoma with one eye manifesting only posterior chamber infiltration and no retinal involvement Diego Ossandon, MD,a,b Alan Kastner, MD,c Mario Zanolli, MD,a Federica Solanes, MD,c Ver onica P erez, MD,d Gabriela Repetto, MD,e Felipe Benavides, BSc,e and Carol L. Shields, MDf We report the case of a 23-month-old girl with bilateral retinoblastoma that demonstrated absence of retinal lesions in one eye but had an isolated white tumor in the posterior chamber. Genetic testing confirmed a novel and de novo RB1 germline mutation in the proband that was not carried by her parents. After intravenous chemotherapy and brachytherapy to the eye with apparently disease-free retina, anatomic and functional preservation of the eye was achieved. The patient has been in remission for 18 months.
Author affiliations: aOphthalmology Department, Clınica Alemana de Santiago, Facultad de Medicina Universidad del Desarrollo, Santiago, Chile; bOphthalmology Department, Hospital San Juan de Dios, Santiago, Chile; cOphthalmology Department, Pontificia Universidad Catolica de Chile, Santiago, Chile; dMinisterio de Salud, Gobierno de Chile, Programa Infantil Nacional de Drogas Antineoplasicas (PINDA); eGenetic and Genomic Centre, Clınica Alemana de Santiago, Facultad de Medicina, Universidad del Desarrollo, Santiago, Chile; fOcular Oncology Service, Wills Eye Hospital, Thomas Jefferson University, Philadelphia, Pennsylvania Submitted March 11, 2016. Revision accepted July 25, 2016. Published online November 1, 2016. Correspondence: Alan Kastner, MD, Pontificia Universidad Catolica de Chile, Departamento de Oftalmologıa, Santiago, Chile (email:
[email protected]). J AAPOS 2016;20:548-550. Copyright Ó 2016, American Association for Pediatric Ophthalmology and Strabismus. Published by Elsevier Inc. All rights reserved. 1091-8531/$36.00 http://dx.doi.org/10.1016/j.jaapos.2016.07.230
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eFig 1. Following completion of intravenous chemotherapy and brachytherapy, the diffuse anterior retinoblastoma is completely gone, leaving no remnant.
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eFig 1. Six months after surgery, there was resolution of ptosis, lower scleral show, and the widening of the palpebral fissure.
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