Spontaneous repositioning of a dislocated autologous scleral plug after vitrectomy with autologous scleral plug for optic disc pit maculopathy: A case report

Abstract

Purpose

Presented herein is a case of optic disc pit maculopathy (ODP-M) treated with vitrectomy and autologous scleral plug implantation, complicated by scleral plug dislocation. The dislocation was subsequently corrected by spontaneous repositioning without surgical intervention.

Observations

A 58-year-old female was diagnosed with ODP-M and underwent vitrectomy with internal limiting membrane peeling and autologous scleral plug implantation. Postoperatively, the scleral plug successfully sealed the ODP. There was an improvement in visual acuity and a decrease in subretinal fluid (SRF) and cystoid macular edema (CME). Seven weeks after surgery, dislocation of the scleral plug was observed, which was associated with frequent upside-down positioning using an inverting table. The patient was instructed to maintain a supine position, which resulted in spontaneous repositioning of the scleral plug. The scleral plug remained stable over 10 months postoperatively, with continued resolution of CME and SRF. Visual acuity improved to 20/25.

Conclusions and importance

Upside-down positioning may lead to scleral plug dislocation following autologous scleral plug implantation for ODP-M and should be avoided. However, in cases of dislocation, maintaining a supine position may facilitate spontaneous repositioning, potentially eliminating the need for additional surgery. This case underscores the importance of patient education regarding postoperative positioning and demonstrates the potential effectiveness of positional management for scleral plug dislocation.

1. Introduction

Optic disc pit (ODP) is a congenital anomaly characterized by a small, oval excavation in the optic nerve head, with an estimated prevalence of 1 in 11,000.¹ Macular changes including accumulation of serous retinal fluid (SRF) and the development of cystoid macular edema (CME), collectively termed as optic disc pit maculopathy (ODP-M), occurs in 25 %–75 % of ODP cases.^2,3

Although many patients with ODP-M maintain good long-term visual acuity and resolution of SRF without surgical intervention,⁴ treatment is required when SRF and CME worsen, leading to vision deterioration. Treatment options include juxtapapillary laser photocoagulation,^5,6 vitrectomy with peeling of the internal limiting membrane (ILM),7, 8, 9 and autologous scleral plug implantation.9, 10, 11, 12

Herein, we report a case of a patient with ODP-M treated with vitrectomy and autologous scleral plug implantation, which reduced SRF and CME. However, scleral plug dislocation occurred, but spontaneous repositioning was achieved without surgical intervention.

2. Case report

A 58-year-old female patient presented with best-corrected visual acuity (BCVA) of 20/100 in her right eye. Examination of the fundus revealed an ODP cavity located at the inferotemporal outer border of the optic nerve. Optical coherence tomography (OCT) demonstrated CME and SRF. Initial management included juxtapapillary laser photocoagulation, which worsened SRF and CME (Fig. 1). Therefore, the patient underwent pars plana vitrectomy, ILM peeling, and autologous scleral plug implantation. The surgical technique was as follows. Core vitrectomy was performed under general anesthesia. After staining the vitreous with triamcinolone acetate, the posterior vitreous detachment induction was achieved. The ILM was stained with 0.005 % indocyanine green dye and peeled circumferentially, including the area of the CME. The tissues of the superior conjunctiva and tenon were incised, exposing the sclera. A 1 × 1 mm half-thickness scleral flap was then created. The scleral flap was trimmed to an approximately 1 × 0.5 mm scleral plug, which was placed into the vitreous cavity and positioned into the ODP using retinal forceps. Fluid-air exchange was performed, followed by 14 % perfluoropropane (C₃F₈) gas tamponade. The patient was instructed to maintain a prone position for three days. One month postoperatively, the BCVA improved to 20/50, and the scleral plug successfully sealed the ODP. OCT showed a reduction in SRF and CME (Fig. 2). Three weeks after the last visit, the patient presented with floaters and decreased vision. Examination of the fundus revealed a dislocated scleral plug in the vitreous, along with a progression of SRF and CME (Fig. 3). The patient reported frequent use of an inverting table for upside-down positioning and noted the onset of floaters after assuming this position. The patient was instructed to maintain a supine position for one week. After one week, the scleral plug was spontaneously repositioned, successfully sealing the ODP (Fig. 4). At 10 months postoperatively, the scleral plug remained stable, effectively sealing the ODP (Fig. 5). OCT showed continued reduction of SRF and CME, and the BCVA improved to 20/25.

Fig. 1.

Photograph of the fundus and optical coherence tomography (OCT) findings at baseline. (A) Examination of the fundus showed an optic disc pit located at the inferotemporal outer border of the optic nerve. (B) OCT image demonstrates subretinal fluid (SRF) and cystoid macular edema (CME), (C) consistent with communication between the perineural and intraretinal space.

Fig. 2.

Photograph of the fundus and optical coherence tomography (OCT) findings at 1 month postoperatively. (A) Examination of the fundus showed an autologous scleral plug sealing the optic disc pit. (B) OCT image illustrates a significant reduction in subretinal fluid (SRF) and cystoid macular edema (CME).

Fig. 3.

Wide-field imaging of the fundus and optical coherence tomography (OCT) findings at 7 weeks postoperatively. (A) Examination of the fundus revealed a dislocated scleral plug within the vitreous cavity (white arrow). (B) OCT image demonstrates recurrence of subretinal fluid (SRF) and cystoid macular edema (CME).

Fig. 4.

Photograph of the fundus and optical coherence tomography (OCT) findings 1 week after adopting a supine position. (A) Examination of the fundus showed successful repositioning of the scleral plug into the optic disc pit. (B) OCT image confirms reduction in the subretinal fluid (SRF) and cystoid macular edema (CME), and (C) the scleral plug was located at the optic disc pit.

Fig. 5.

Photograph of the fundus and optical coherence tomography (OCT) findings at 10 months postoperatively. (A) Examination of the fundus showed stabilization of the scleral plug within the optic disc pit. (B) OCT image demonstrates continued reduction of subretinal fluid (SRF) and cystoid macular edema (CME).

3. Discussion

The exact etiology of ODP-M remains uncertain, but several mechanisms have been proposed. Ohno-Matsui et al.¹³ suggest that the ODP directly connects the subarachnoid and subretinal spaces, allowing cerebrospinal fluid to flow into the subretinal space, resulting in SRF and CME. Theodossiadis et al.¹⁴ proposed that vitreous traction on the ODP and macula generates negative pressure, enabling vitreous fluid to pass through the pit into the subretinal space.

Based on these proposed mechanisms, vitrectomy with ILM peeling has been performed to eliminate vitreous traction, with reported success rates of 70 %–85.7 %.7, 8, 9 Additionally, autologous scleral plug implantation has been employed to block the connection between the subarachnoid and subretinal spaces, achieving success rates of 83.3 %–100 %.9, 10, 11, 12

Although autologous scleral plugs are considered effective and generally achieve stable closure of the optic disc pit, there remains a risk of dislocation. In a previous study involving 53 eyes, no cases of plug dislocation were reported,¹⁵ suggesting that such events are exceedingly rare. Nevertheless, clinicians should be aware of the potential for instability, particularly in cases of poor postoperative compliance or unfavorable positioning.

Although the inverted ILM flap technique has shown favorable outcomes in ODP-M, it was not feasible in our case due to intraoperative separation of the ILM during ILM peeling around the optic disc. Therefore, an autologous scleral plug was used as an alternative, which has demonstrated comparable anatomical success in previous reports.9, 10, 11, 12

In the present case, the scleral plug effectively sealed the ODP postoperatively, resulting in a reduction of SRF and CME. However, 7 weeks postoperatively, the scleral plug was dislocated. Notably, the patient frequently engaged in exercises using an inverting table for upside-down positioning. Since ODP is usually located on the inferotemporal border of the optic nerve head,¹⁶ upside-down positioning may have contributed to the dislocation of the scleral plug from the ODP. This highlights the importance of patient education regarding post-operative positioning and careful monitoring during follow-up.

Another notable finding is that the dislocated scleral plug spontaneously repositioned into the ODP when the patient maintained a supine position, without any surgical intervention. Since the ODP is recessed within the optic disc, it is positioned at the lowest point when in the supine position. This anatomical configuration may facilitate the spontaneous repositioning of the scleral plug into the ODP.

A limitation of this report is the absence of visual field testing, which would have provided additional information regarding possible visual field defects related to the optic disc pit and the scleral plug, although the patient did not report any visual field disturbances.

4. Conclusions

In case of ODP-M treated with vitrectomy and autologous scleral plug implantation, upside-down positioning may lead to scleral plug dislocation and should be avoided. However, if dislocation occurs, spontaneous repositioning of the scleral plug may be achieved in a supine position and should be considered before surgical intervention.

CRediT authorship contribution statement

Yong Koo Kang: Writing – review & editing, Writing – original draft. Seonyong Jeong: Writing – review & editing. Dong Ho Park: Writing – review & editing. Jae Rock Do: Writing – review & editing, Conceptualization.

Patient consent

The participant signed a written consent form.

Authorship

All authors attest that they meet the current ICMJE criteria for Authorship.

Ethics approval

The study protocol was approved by the Ethics Committee of the Kyungpook National University and was conducted in accordance with the ethical principles of the Declaration of Helsinki.

Data availability statement

The original contributions presented in the study are included in the article/Supplementary Material. The datasets analyzed during this study are available from the corresponding author on reasonable request.

Funding

DHP is financially supported by the Basic Science Research Program of the National Research Foundation of Korea (NRF), funded by the Ministry of Science and ICT (MSIT) (RS-2024-00334982); the Information Technology Research Center (ITRC) support program funded by MSIT and supervised by the Institute of Information and Communications Technology Planning & Evaluation (IITP) (IITP-2025-2020-0-01808); the Korea Health Technology R&D Project through the Korea Health Industry Development Institute (KHIDI), funded by the MOHW (HR22C1832, RS-2024-00437643); and the BK21 FOUR KNU Convergence Educational Program of Biomedical Sciences for Creative Future Talents.

Declaration of competing interest

The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

Glossary

BCVA

best-corrected visual acuity

CME

cystoid macular edema

ILM

internal limiting membrane

ODP

optic disc pit

ODP-M

optic disc pit maculopathy

OCT

optical coherence tomography

SRF

subretinal fluid