Clinical Ophthalmology Dovepress open access to scientific and medical research Open Access Full Text Article PERSPECTIVES Surgical Options for Optic Disc Pit Maculopathy: Perspectives and Controversies This article was published in the following Dove Press journal: Clinical Ophthalmology 1–3 Abstract: Optic disc pit (ODP) is a rare congenital optic nerve head abnormality, which can be Salvador Pastor-Idoate Claudia García- complicated by intraretinal and subretinal fluid at the macula (ODP-maculopathy) with progres- Arumí Fusté 4,5 sive visual loss. The source of this fluid remains unclear and the most dominant hypotheses have Luis García-Onrubia 1 pointed to vitreous cavity or cerebrospinal fluid. Although spontaneous resolution has been reported, the majority of untreated cases of ODP-maculopathy result in final visual acuity less Sergio Copete 6 than 20/200 or worse. A wide array of interventions, either individually or in combination with José García-Arumí 3,5,7,8 adjuvant treatments, have been tried with varying degrees of success. Recently, different surgical 1 IOBA Eye Institute, University of procedures to fill the ODP by self-sealing materials in combination with pars plana vitrectomy Valladolid, Valladolid, Spain; 2Clinical University Hospital of Valladolid, have been reported as an effective adjuvant treatment. However, given the relative rarity of this Valladolid, Spain; 3Red Temática de condition, the majority of reports describe a small retrospective case series, making it difficult to Investigación Cooperativa en Salud compare among different treatments options and create a consensus regarding the optimal (RETICS), Oftared, Instituto de Salud Carlos III, Madrid, Spain; 4Vall d´Hebron treatment for ODP-maculopathy. In this situation, a mini-review about surgical treatment University Hospital, Barcelona, Spain; modalities and their results can be a useful approach to identify the most effective surgical 5 Ophthalmology Research, Vall d´Hebron option in the management of ODP-maculopathy. Research Institute (VHIR), Barcelona, Spain; 6Complex University Hospital, Keywords: optic disc pit, optic disc pit maculopathy, optic disc pit maculopathy treatments, Albacete, Spain; 7Ocular Microsurgery optic nerve stuffing, post-operative outcomes, pars plana vitrectomy Institute (IMO), Barcelona, Spain; 8 Department of Ophthalmology, Autonomous University of Barcelona, Barcelona, Spain Introduction Optic disc pit (ODP) is considered to be part of the spectrum of rare congenital cavitary abnormalities of the optic disc, occurring in the majority of the cases on the temporal side of the optic disc.1 It is usually unilateral, sporadic in occurrence and can occur equally in men and women with an estimated prevalence of 1 out of 10,000 people.2 In the absence of other sequelae, many of the patients may remain asymptomatic and it may be found incidentally. Sometimes, the symptoms are generally limited to arcuate scotomas or enlargement of the blind spot in visual field tests.2–4 However, when ODP is complicated by maculopathy (ODP-M), it may cause significant visual deterioration. ODP-M is the term used to describe the existence of intrar- etinal and subretinal fluid at the macula as well as retinal pigment changes. It can occur in up to 25–75% of all cases.2–5 Regarding the pathophysiology of ODP-M and the origin of fluid, the exact mechanism remains unclear. It has been proposed Correspondence: José García-Arumí that either vitreous or cerebrospinal fluid (CSF) may be responsible for macular Vall d´Hebron University Hospital, Passeig detachment in eyes with OPD.6–8 De La Vall D’hebron, 119-129, Barcelona 08035, Spain When ODP-M is present, visual acuity (VA) is usually affected to 20/70 or Tel +34 934 893 000 worse,2,5,9–13 but in the untreated and long-standing cases, which can be associated Fax +34 932 746 185 Email josgarci@vhebron.net with cystoid changes, lamellar or full-thickness macular holes, and retinal pigment submit your manuscript | www.dovepress.com Clinical Ophthalmology 2020:14 1601–1608 1601 DovePress © 2020 Pastor-Idoate et al. This work is published and licensed by Dove Medical Press Limited. The full terms of this license are available at https://www.dovepress.com/ http://doi.org/10.2147/OPTH.S250921 terms.php and incorporate the Creative Commons Attribution – Non Commercial (unported, v3.0) License (http://creativecommons.org/licenses/by-nc/3.0/). By accessing the work you hereby accept the Terms. Non-commercial uses of the work are permitted without any further permission from Dove Medical Press Limited, provided the work is properly attributed. For permission for commercial use of this work, please see paragraphs 4.2 and 5 of our Terms (https://www.dovepress.com/terms.php). Pastor-Idoate et al Dovepress epithelium (RPE) atrophy, the visual impairment is usually series of PPV and adjuncts treatments for ODP-M is given irreversible, leading to a reduced VA to 20/200 or worse in in Table 1. the affected eye.2,5,9-13 Therefore, interventions are valu- A wide array of adjuvant treatments, including injec- able, as they may prevent significant visual loss in these tion of gas,20,21,24,25 air17 or silicone tamponade,26 juxta- patients. However, despite the improved insight into this papillary laser,20,24,27 retinal fenestration,28 glial tissue unusual pathology, currently, there is no consensus regard- removal29 and macular internal limiting membrane (ILM) ing its optimal treatment. Maybe because the nature and peeling,25,27,30 in combination with PPV have been tried the origin of the fluid causing the macular detachment are with varying degrees of success. still controversial with no known triggers.2,5,9-13 The pur- The majority of these studies reported high anatomical pose of this mini-review is to discuss the possible surgical success rates (50% to 95%) and similar functional results, treatments options and the postoperative outcomes data of with VA improvement in >50% of cases in routine this challenging entity. cases.9–13,17,20,24–27,31,32 However, these adjuvant inter- ventions are not exempt from risks and complications whilst successfully long-term anatomical results have Conservative Management been achieved with just PPV alone.19–22 In the management of ODP-M, especially in pediatric In cases where a vitreous substitute is necessary, the cases, a spontaneous resolution with an improvement of use of gas would be a reasonable approach to prevent the visual acuity is possible.14,15 Therefore, a more conserva- migration into the brain or subretinal space.26,33–36 Thus, tive approach with 3–6 months of follow-up before any avoiding the use of perfluorocarbon liquid or silicone oil surgical procedure could be appropriate. However, it must because of their lower surface tension and easier migra- be kept in mind that the number of pediatric cases is small, tion. Also, migration of gas is much less problematic and relapses are frequent, and the prognosis remains poor in easy to manage than silicone oil or heavy liquid migration. approximately 25% to 75% of the patients.2–5 Temporal endolaser photocoagulation has been proposed Since patients with ODP may remain asymptomatic as a treatment for ODP-M,20,24,27 with the reasoning that with normal visual acuity until 2nd to 4th decades of laser scars will act as a “permanent” barrier to prevent fluid life2–5 an individualized approach should be considered from leaking into the macula. However, this method of in each case assessing the visual symptoms with the clin- treatment can cause significant visual field defects because ical and imaging findings, but currently, in the presence of of the damage over the maculopapular bundle.24,30,37 progression of fluid accumulation and/or visual loss, Furthermore, it has been reported that laser treatment does observation is deemed unjustified.5,13 not aid in anatomical success.19,27,38 Peeling the ILM in these cases may result in a high incidence of macular hole creation, Surgical Treatment Options as well as anatomical changes of the macula including dim- Currently, there is no optimal accepted treatment for ODP-M pling or damage to the inner retina.39 In addition, there are because the pathogenesis of the disease is not yet fully under- numerous surgical series showing excellent results without stood. However, since vitreous traction, specifically the ILM peeling.23–25,40,41 Thus, this maneuver seems to be vitreopapillary traction,2,5,16–18 has been suggested to play questionable and probably not essential in the treatment of an important role in the pathogenesis of ODP-M, pars plana most cases of ODP-M. Creation of an inner retinal vitrectomy (PPV) with the induction of posterior vitreous fenestration28 or glial tissue removal,29 procedures that detachment (PVD) remains the most widely accepted treat- involve risk of optic nerve injury, fall short of providing ment of choice for this entity.9–13,19–22 Tight vitreous attach- adequate evidence to be recommended for ODP-M, as they ment over the optic nerve has been reported in previous did not show an increment in the surgical success rate. studies preoperatively by OCT and intraoperatively by Therefore, if the vitreous cavity is the source of the iOCT in ODP-M patients.16,17,23 Therefore, inducing fluid in ODP-M, then the role of PPV + induction of PVD a PVD by PPV can relieve the traction exerted by the vitreous would be the most reasonable and effective treatment, as on either the macula or the ODP, facilitating the absorption of the adjuvant procedures may not confer any tangible added subretinal fluid. Most of the published literature on ODP-M benefit over PPV alone.19–22 is focused on PPV techniques which have evolved along with However, this vitreous traction hypothesis has some the breakthroughs.2,5,9-13 A summary of some published limitations as it cannot explain the recurrent cases of submit your manuscript | www.dovepress.com Clinical Ophthalmology 2020:14 1602 DovePress Dovepress Pastor-Idoate et al Table 1 Main Recent Series Describing Treatment for Optic Disc Pits with Maculopathy Study [Ref.] Cases, n Main Surgery Main Results Effect of the Adjuvant Treatment with/ Without Adjuvant Techniques Avci et al 201725 51 eyes 20 G, 23 G, 25 58.8% showed CR within 6 months, while 25.5% No significant effect of EL, ILM-P, C3F8 or SF6 on G, TA, EL, ILM-P, resolved at month 12 and thereafter CR with no the final success rate C3F8, SF6 further treatments: 86.3% Bottoni et al 201821 11 eyes 20 G, 25 G, TA, The macular detachment resolved within a mean of No significant effect of EL, ocriplasmin or SF6 on EL, ocriplasmin, 14 months after surgery CR with no further the final success rate SF6 treatments: 80% Rayat et al 201530 32 eyes 20 G, 23 G, 25 The median time to reattachment was 416 days CR No significant effect of EL, ILM-P, C3F8 or SF6 on G, TA, EL, ILM-P, with no further treatments: 80.3% the final success rate C3F8, SF6 Abouammoh et al 46 eyes 20 G, 23 G, 25 The mean time to resolution was 12.1±10.6 months No significant effect of EL, ILM-P, C3F8 or SF6 on 201627 G, TA, EL, ILM-P, for all eyes CR with no further treatments: 72% the final success rate C3F8, SF6 Teke and Citirik 2015 20 17 eyes 23 G, TA, EL, The mean time to reattachment was {20.9}3.2 No significant effect of EL or C3F8 on the final C3F8 months in the adjuvant group and 25.5±4.0 months success rate in the group with PPV alone CR with no further treatments: ?% 19 Hirakata et al 2012 8 eyes 20 G, 25 G, TA Up to about 1 year for retinal detachment resolution Gas tamponade does not seem to be crucial Lack CR with no further treatments: 88% of a control group Nadal et al 2015 49 19 eyes 20 G, 23 G, TA, Retinal fluid reabsorption within a median of 3.5 APC and C3F8 are highly effective alternative APC, C3F8 months CR with no further treatments: 100% techniques, but a control group was lacking Avci et al 201324 13 eyes 23 G, TA, EL, Fluid reabsorption by month 12 (84.6%) CR with no ILM may not be required for anatomical success C3F8 further treatments: 92% Lack of a control group Gregory-Roberts et al 9 eyes ? G, forceps for CR with no further treatments: 89% Higher rate of CR when glial tissue was removed, 201323 glial tissue, EL, but a control group was lacking gas? Kiang and Johnson 11 eyes ? G, previous The average time to macular fluid resolution was 8.5 2 eyes required additional treatment Secondary 201731 laser treatment, months CR with no further treatments: 100% approach if vitrectomy alone fails Absence of EL, gas? a control group Pastor-Idoate 9 eyes 23 G, 25 G, TA, The mean duration of preoperative symptoms was Residual postoperative ILM fragments (78%) Laser et al 201916 EL, I–ILM, C3F8 422 days Retinal fluid reabsorption in 56% at 6 and gas do not seem to be crucial Secondary months approach in chronic/recurrent cases Lack of a control group 32 Ghosh et al 2008 7 eyes ? G, EL, air, SO, The duration of their symptoms was between 2 4 eyes required additional treatment Lack of SF6, C3F8, weeks in a child to 5 months in an elderly man. CR a control group with no further treatments: 100% Ooto et al 201428 18 eyes 23 G, 25 G, The macular detachment decreased within a mean of Spontaneous closure in the early postoperative fenestration 6.1 months CR with no further treatments: 94% period, suggesting no significant effect Lack of a control group Theodossiadis and 23 eyes SB The macular reattachment was noticed between 6 Intraoperative B scan required Satisfactory long- Theodossiadis 2001*56 and 12 months CR with no further treatments: 85% term results Lack of a control group Theodossiadis, 12 eyes SB The mean followed up was 12.8±1.5 years after the No cataract induction during the follow-up period. Chatziralli, and surgical intervention with successful anatomical and Lack of a control group Theodossiadis 2015*42 functional results (Continued) submit your manuscript | www.dovepress.com Clinical Ophthalmology 2020:14 1603 DovePress Pastor-Idoate et al Dovepress Table 1 (Continued). Study [Ref.] Cases, n Main Surgery Main Results Effect of the Adjuvant Treatment with/ Without Adjuvant Techniques 52 Lei et al 2015* 9 eyes C3F8, LT The period required for complete reattachment was Repeat treatment was required in 5 eyes with 6–18 months Complete retinal reattachment was complete treatment in 3 eyes Lack of a control achieved in seven out of nine eyes group Akiyama et al 2014* 53 8 eyes SF6 The period required for reattachment after final gas Secondary approach if intravitreal gas injection treatment was 12 months The mean number of gas alone fails Absence of a control group injections was 1.8 CR with no further treatments: 50% Notes: Presented are only studies that included more than 7 patients with optic disc pits with maculopathy. *Treatment without vitrectomy; ?, no data available. Abbreviations: APC, autologous platelet concentrate; C3F8, perfluoropropane; CR, complete resolution; EL, endolaser; LT, laser photocoagulation; G, gauge; I–ILM, inverted internal limiting membrane; ILM-P, internal limiting membrane peeling; PPV, pars plana vitrectomy; SF6, sulphur hexafluoride; SB, scleral buckling; SO, silicone oil; TA, triamcinolone acetonide assisted. macular detachment after PPV + induction of PVD or even autologous fibrin,49 homologous scleral tissue50 or even sili- after the removal of tight vitreous adhesion at the ODP.28 cone lacrimal plug,51 is based on the recent hypothesis pos- Neither it can explain the ODP-M cases developed in tulated by Jain and Johnson,7 in which different pressure patients with total PVD. ranges between the eye and intracranial pressure can lead to But if the source of the fluid is the CSF, then further migration of fluid from the vitreous cavity or CSF to intrar- explanation regarding the efficacy of PPV is needed.42 etinal or subretinal space due to a defect either in the lamina Maybe in those cases, the key point for obtaining cribrosa or the juxtapapillary area.7 Therefore, from a successful surgery could be the emergence of recent adju- a theoretical point of view, a procedure to create a “broad vant treatments that are principally focused on sealing the pit. plug” along the “involved” disc margin could prevent the The justification for sealing the ODP with different materials translaminar difference of pressure or block the outflow of such as ILM flap (Figure 1),14,41–46 fibrin glues47,48 or fluids to the retinal layers or subretinal space. Figure 1 (A) Preoperative OCT image showing the anatomic features of an ODP and the associated maculopathy. The translaminar defect which can lead to migration of fluid (arrow) from the vitreous cavity or CSF to intraretinal or subretinal space. (B and C) Intraoperative vertical and horizontal OCT scans through the optic disc showing the ILM flap “sealing” the ODP (white arrows). Abbreviations: OCT, optical coherence tomography; ODP, optic disc pit; CSF, cerebrospinal fluid; ILM, internal limiting membrane. submit your manuscript | www.dovepress.com Clinical Ophthalmology 2020:14 1604 DovePress Dovepress Pastor-Idoate et al However, even though reports describe successful anato- results, the use of ILM or scleral flap may be beneficial mical and functional results that have been maintained long only in selected cases of optic pit maculopathy. However, term in some cases, the restricted number of cases and low the evidence remains low and more studies are warranted. level of evidence limit the use of these methods.9–14,41–51 One of the main concerns that should be addressed in Our experience using the ILM flap technique in nine the future is the potential damage to the optic nerve and consecutive patients suggested a moderate rate of anatomical neurosensory retina caused by surgical trauma and/or the success, as 44% of our patients remained with unattached cytotoxicity of introducing different sealing materials into macula even 6 months after the surgery.14 However, pub- the ODP, as the majority of results are presented without lished studies44,45 using the same technique have reported visual field tests or macular sensitivity tests.14 quick improvements and promising functional outcomes. Such differences might be related to the reduced function Other Techniques of the retinal pigment epithelium-Bruch’s membrane com- Pneumatic tamponade with or without laser photocoagulation plex, as well as by degeneration within the retinal layers in is another therapeutic option for ODP-M.52 Theoretically, this chronic and/or refractory macular detachment patients.30,50 dual treatment could ensure the induction of PVD, alleviating In some of these patients, we have observed a relapse. the vitreous tractions and facilitating the reattachment of the However, in those refractory cases, we have considered pre- macula, and the formation of a barrage to seal the passage ferable to apply for another novel technique initially described between the ODP and the macula.53 However, this technique by Travassos et al50 in which autologous scleral graft is used has only reported retinal reattachment in approximately 50% for sealing the pit with good initial outcomes (Figure 2). of cases. Additionally, significant visual field defects and no The concept of these techniques would consist of cover- improvement in VA have been reported after peripapillary ing the optic disc, including the ODP, creating a barrier to the barrier with photocoagulation.2,5,7 fluid passage from the pit to the macular region. Although Macular buckling (MB) is another alternative for ODP- both techniques have demonstrated favorable long-term M and it has been reported a success rate of about 85% Figure 2 (A) Preoperative horizontal OCT scan through the optic disc and fovea, showing an abundance of subretinal macular fluid, extending towards the ODP as well as subretinal hyperreflective deposits suggesting a long-standing case. (B) Intraoperative fundus picture showing the autologous scleral plugging into the ODP. (C) Postoperative follow-up image showing foveal re-attachment with no evidence of subretinal fluid but with residual local alterations at the Interdigitation and ellipsoid zones. Abbreviations: OCT, optical coherence tomography; ODP, optic disc pit. submit your manuscript | www.dovepress.com Clinical Ophthalmology 2020:14 1605 DovePress Pastor-Idoate et al Dovepress with long-term visual improvement and low rates of com- Nowadays, the gold standard for the treatment of ODP-M plications or recurrences.42,54–56 The macular buckle is still the PPV and PVD induction.9–13,19–22 Adjuvant proce- pushes the macular surface toward the vitreous cavity, dures in combination with PPVare several and varied but they preventing the entrance of fluid and alleviating vitreous would probably be pertinent when the primary surgery fails. tractions. It is worthy to note that MB seems to provide favorable results independently of the origin of the fluid in Disclosure ODP-M. However, MB is considered a difficult and The authors report no conflicts of interest in this work. demanding technique and, for this reason, is not widely applied nowadays.9–13 References 1. 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Am J Ophthalmol. 2001;132(2):184–190. doi:10.1016/ 93d93 S0002-9394(01)00997-7 Clinical Ophthalmology Dovepress Publish your work in this journal Clinical Ophthalmology is an international, peer-reviewed journal cover- Central and CAS, and is the official journal of The Society of ing all subspecialties within ophthalmology. Key topics include: Clinical Ophthalmology (SCO). The manuscript management system Optometry; Visual science; Pharmacology and drug therapy in eye dis- is completely online and includes a very quick and fair peer-review eases; Basic Sciences; Primary and Secondary eye care; Patient Safety system, which is all easy to use. Visit http://www.dovepress.com/ and Quality of Care Improvements. This journal is indexed on PubMed testimonials.php to read real quotes from published authors. Submit your manuscript here: https://www.dovepress.com/clinical-ophthalmology-journal submit your manuscript | www.dovepress.com Clinical Ophthalmology 2020:14 1608 DovePress