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Pathogenic Implications of Subretinal Gas Migration Through Pits Andatypical Colobomas of the Optic Nerve

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Pathogenic Implications of Subretinal Gas Migration Through Pits Andatypical Colobomas of the Optic Nerve CLINICAL SCIENCES Pathogenic Implications of Subretinal Gas Migration Through Pits and Atypical Colobomas of the Optic Nerve T. Mark Johnson, MD, FRCSC; Mark W. Johnson, MD Objective: To describe subretinal migration of gas and cases. Subretinal migration of gas or silicone oil was seen silicone oil in a series of patients with congenital cavi- intraoperatively in one case and first appeared between tary optic disc anomalies and to further clarify the patho- 1 and 17 days postoperatively in the remaining cases. genesis of the associated maculopathy. Theoretical calculations suggest that the pressure differ- ential required for migration of gas through a small de- Methods: Medical records of 4 female patients, aged 8 fect in the roof of a cavitary disc lesion is within the range to 34 years, who developed subretinal gas migration af- of expected fluctuations in cerebrospinal fluid pressure. ter vitreous surgery for macular detachment associated with cavitary optic disc anomalies were reviewed. A theo- Conclusions: These observations provide clinical con- retical model was used to calculate the pressure differ- firmation of a defect in tissue overlying cavitary optic disc ential required to induce subretinal gas migration through anomalies and imply interconnections between the vit- an optic pit. reous cavity, subarachnoid space, and subretinal space. We theorize that intermittent pressure gradients result- Results: The 4 patients had bilateral atypical optic nerve ing from normal variations in intracranial pressure play colobomas or a unilateral large optic pit. A definite de- a critical role in the pathogenesis of retinopathy associ- fect in the tissue overlying the disc excavation could be ated with cavitary disc anomalies. seen in one eye, and intraoperative drainage of subreti- nal fluid through the disc anomaly was possible in all Arch Ophthalmol. 2004;122:1793-1800 ONGENITAL CAVITARY havior of intraocular gas and silicone oil. anomalies of the optic We believe that our clinical observa- nerve that may be associ- tions, coupled with recent optical coher- ated with serous detach- ence tomographic findings and consider- ments of the macula in- ation of cerebrospinal fluid (CSF) Cclude optic disc pit, optic nerve coloboma dynamics, provide important new in- (typical and atypical), and morning glory sights into the pathogenesis of the macu- disc anomaly.1-4 Frank macular detach- lopathy complicating optic pits and re- ment appears to be preceded by the accu- lated disc anomalies. mulation of intraretinal fluid emanating from the disc anomaly and constituting an unusual form of retinoschisis-like separa- METHODS tion.5 Subsequently there is breakthrough of fluid into the subretinal space leading to We retrospectively identified 4 patients who detachment of the macula and occasion- developed subretinal gas migration after vit- ally larger areas of the retina. The origin of reous surgery for macular detachment associ- Author Affiliations: Kellogg the fluid and precise pathogenesis of macu- ated with cavitary optic disc anomalies. The pa- Eye Center, Department of lar detachment associated with cavitary op- tients were derived from the practices of 4 retina Ophthalmology and Visual specialists at 3 centers. One patient (case 1) was tic disc anomalies remain unclear. 6 Sciences, University of We present 4 cases of retinal detach- described in a previous report. The medical Michigan School of Medicine, ment associated with excavated optic disc records and available fundus photographs were Ann Arbor. Dr T. M. Johnson is reviewed. Although institutional review board now with the National Retina anomalies in which vitreous surgery was oversight was not required for this chart re- Institute and George complicated by subretinal migration of gas view, each patient gave written informed con- Washington University, Chevy and silicone oil. This rare and unex- sent before undergoing surgical intervention. Chase, Md. pected event cannot readily be explained Using the physical principles governing the be- Financial Disclosure: None. by the principles known to govern the be- havior of intraocular gas, we calculated the (REPRINTED) ARCH OPHTHALMOL / VOL 122, DEC 2004 WWW.ARCHOPHTHALMOL.COM 1793 ©2004 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/26/2021 A B Figure 1. Case 1 fundus photographs. A, Right eye, showing anomalous optic disc with associated retinal detachment and outer lamellar foveal break. B, The left optic disc is also anomalous, but without an associated maculopathy. in the temporal neuroretinal rim. The left disc was anoma- lous, with a large cup and nasalization of disc vessels but no evidence of associated maculopathy (Figure 1). B-scan ultrasonography of the right eye showed no evidence of pos- terior vitreous detachment. Orbital ultrasound and com- puted tomographic scans were normal bilaterally. The patient underwent pars plana vitrectomy with re- moval of the attached posterior hyaloid, subretinal fluid drainage through a small retinotomy, fluid-gas exchange with 20% sulfur hexafluoride, and 10 days of postopera- tive face-down positioning. Two months postoperatively, a moderate posterior subcapsular cataract was evident, along with a small macular hole and shallow subretinal fluid in the macula extending nasally to the optic disc. Contact lens Figure 2. Case 1, 7 days after fluid-gas exchange. Fundus photograph of the examination demonstrated a defect in the tissue overlying right eye shows gas bubbles under the retina and trapped within the disc the temporal aspect of the disc cavitation. cavity beneath a neural tissue layer. A small hole (arrow) in this tissue could When the subretinal fluid persisted 2 months later, be seen on biomicroscopy. krypton red laser burns were placed in 3 rows in the tem- poral juxtapapillary area. The patient then underwent theoretical pressure differential required for gas migration into phacoemulsification with placement of an intraocular lens an optic pit and compared this with information derived from followed by repeat vitrectomy with fluid-gas exchange a model of CSF pressure dynamics. and postoperative prone positioning. Seven days post- operatively, several gas bubbles were noted in the sub- REPORT OF CASES retinal space between the optic disc and central macula (Figure 2). There was also gas trapped under neural tis- CASE 1 sue overlying the deep optic disc cavitation. The gas re- solved during the subsequent 3 weeks. A 24-year-old woman was examined because of decreased Two months later, the patient noted an abrupt de- and darkened vision in the central visual field of the right cline in vision in the right eye. Examination showed ex- eye. The ocular history was significant for mild myopia. The tensive detachment of the macular region and fluid com- maternal family history was notable for glaucoma. munication with the small hole in the neural tissue over The visual acuity measured 6/200 OD and 20/20 OS. The the optic disc. A 50% fluid-gas exchange using 20% per- anterior segment was normal in each eye. Examination of fluoropropane was performed. After 7 days of face- the right fundus demonstrated retinal detachment involv- down positioning, the macula was flat and supplemen- ing the macula and superotemporal midperiphery and ex- tal krypton laser was applied to the temporal aspect of tending to the temporal border of the optic nerve the optic disc. After 10 additional days of face-down po- (Figure 1). A stellate outer foveal defect was present, with sitioning, the patient noted an abrupt decline in vision a tiny full-thickness defect at the center of the fovea. The and was found to have recurrent detachment of the pos- retina between the optic nerve and the fovea had an ap- terior retina. Numerous small subretinal gas bubbles were pearance suggesting retinal thickening or schisis. Exami- located in the superior aspect of the detachment nation of the optic disc demonstrated nasalization of the (Figure 3). An additional cluster of bubbles appeared vessels with a deep, large, horizontally oval cup and a notch to be located within the schisis cavity in the papillomacu- (REPRINTED) ARCH OPHTHALMOL / VOL 122, DEC 2004 WWW.ARCHOPHTHALMOL.COM 1794 ©2004 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 09/26/2021 Figure 4. Fundus photograph of the right eye of case 1, 2 weeks after the Figure 3. Fundus photograph of the right eye of case 1, 17 days after the final vitrectomy procedure, shows retinal reattachment and extensive laser second fluid-gas exchange. Numerous subretinal gas bubbles have appeared scarring around the optic disc. in the superior aspect of the detachment. A smaller cluster of bubbles appears to be located within the schisis-like cavity in the papillomacular bundle. lar bundle area. No intraocular pressure measurement greater than 25 mm Hg was recorded at any postopera- tive examination. Two months later, a total and highly bullous retinal de- tachment developed, obscuring a view of the optic disc and macula. No peripheral retinal breaks were found. The pa- tient underwent repeat vitrectomy. During fluid-air ex- change, subretinal fluid was drained through a small macu- lar hole and over the optic disc. Moderately heavy laser photocoagulation was applied around the entire optic nerve, and lighter burns were placed in the papillomacular bundle and at the edge of the macular hole. Two weeks postop- Figure 5. Photograph of the left optic disc of case 2 shows deep pitlike eratively, the visual acuity had improved to 20/100 and excavation in a slightly enlarged optic nerve head. the retina was completely flat (Figure 4). During the sub- sequent 10 years, the visual acuity remained stable and the Repeat vitrectomy with lensectomy, fluid-gas exchange, retina remained attached in the right eye. and scleral buckle was performed. No retinal breaks could be found. Recurrent retinal detachment inferiorly was CASE 2 noted 2 weeks postoperatively and treated with repeat vitrectomy followed by injection of silicone oil. An 8-year-old girl was diagnosed as having an optic pit Ten days postoperatively, the patient was found to have in her left eye on routine ophthalmologic examination.
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