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 Table of Contents  
Year : 2022  |  Volume : 2  |  Issue : 1  |  Page : 149-151

Lamellar hole-associated epiretinal proliferation closing an ectopic macular hole

Chaithanya Eye Hospital and Research Institute, Trivandrum - 695 004, Kerala, India

Date of Submission21-Nov-2020
Date of Acceptance05-Jul-2021
Date of Web Publication07-Jan-2022

Correspondence Address:
Dr. Asmita Indurkar
Chaithanya Eye Hospital and Research Institute, Trivandrum - 695 004, Kerala
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ijo.IJO_3468_20

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The aim of this study was to describe a case where lamellar hole–associated epiretinal proliferation developed into an ectopic macular hole and eventually led to the closure of the defect. This is a retrospective case review of a 61-year-old patient with documented spectral-domain optical coherence tomography (Spectralis, Heidelberg, Germany) follow-up of 3 years, which shows the presence of epiretinal proliferation closing an iatrogenic ectopic lamellar hole developed after a failed macular hole surgery.

Keywords: LHEP, ectopic hole, macular hole, Müller cell

How to cite this article:
Soman M, Indurkar A, Mohan A, Nair U. Lamellar hole-associated epiretinal proliferation closing an ectopic macular hole. Indian J Ophthalmol Case Rep 2022;2:149-51

How to cite this URL:
Soman M, Indurkar A, Mohan A, Nair U. Lamellar hole-associated epiretinal proliferation closing an ectopic macular hole. Indian J Ophthalmol Case Rep [serial online] 2022 [cited 2022 Jan 28];2:149-51. Available from: https://www.ijoreports.in/text.asp?2022/2/1/149/334943

Epiretinal proliferations with medium reflectivity existing in contiguity with the middle retinal layers and conforming to the adjacent retinal anatomy in association with lamellar holes seen on spectral-domain optical coherence tomography (SD-OCT) are termed as lamellar hole–associated epiretinal proliferation (LHEP).[1],[2] They are distinct from the epiretinal membranes in appearance[1] and have immunocytochemical and ultrastructural characteristics.[3] They have been surgically removed along with the internal limiting membrane (ILM), with considerable visual improvement as reported by some,[4] whereas others have reported unsatisfactory outcomes.[5] There are some theories that support their origin from the middle retinal layers, specifically the Müller cells,[4] and some that support their origin from the vitreous.[2]

Although there are several reports that describe LHEP, we could find only one mention[1] of extrafoveal or ectopic holes that develop LHEP. There was, however, no description of their follow-up and prognosis. We hereby present a case with an iatrogenic lamellar hole secondary to ILM peeling that developed LHEP and its follow-up.

  Case Report Top

A 61-year-old female presented with unsatisfactory vision in the right eye after she was treated surgically 2 months back elsewhere for a retinal problem. Her previous reports confirmed that a pars plana vitrectomy with ILM peeling was performed and that the macular hole did not close postoperatively. Her corrected distance visual acuity was 20/63 and 20/32 in the right and left eyes, respectively. She was pseudophakic in both eyes and had normal intraocular pressure. The right eye had a persistent full-thickness macular hole (FTMH) [Figure 1] with a cellophane-like glistening retinal surface around it temporally. Around one-disc diameter temporal to the macular hole, another lesion was seen that appeared like a lamellar macular hole (LMH). The left eye fundus was normal. SD-OCT confirmed the presence of a FTMH and a paracentral LMH [Figure 2]. Cystic spaces were seen surrounding the primary macular hole and the LMH. Additionally, we observed the presence of a medium reflective epiretinal lesion on both sides of the lamellar hole originating from the middle layers (LHEP). This tissue was, however, not associated with the open full-thickness hole. We did not have access to the previous SD-OCT reports, and based on the current visit, we diagnosed a failed macular hole surgery with an iatrogenic LMH (ectopic macular hole) with LHEP.
Figure 1: Multicolor fundus photo of the right eye documents the presence of the full-thickness macular hole and the paracentral iatrogenic lamellar hole (arrowhead) with altered surface glistening (arrow)

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Figure 2: Serial optical coherence tomography images demonstrating persistent central full-thickness macular hole (FTMH) and adjacent lamellar hole (arrow) with epiretinal proliferation (LHEP) progressively filling the hole (arrowhead). The left images are taken through the center of the FTMH and the right images through the upper border of the FTMH. Note the clear epiretinal proliferation seen as a distinctly reflective tissue covering the lamellar defect and lying over the retinal surface on both sides (arrow) in the enlarged picture

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The patient was not keen on any further surgical intervention and preferred to follow-up. Over the next 3 years, her visual acuity remained stable, and the medium reflective LHEP increased in substance to completely fill up the lamellar hole. The primary full-thickness open macular hole remained unaltered, and the vision remained at 6/18 in this eye.

  Discussion Top

To the best of our knowledge, this is the first report to document the progression of LHEP in an iatrogenic lamellar hole leading to spontaneous closure of the hole.

Steven et al.[6] and Rubinstein et al.[7] reported secondary paracentral retinal holes following ILM peeling for idiopathic FTMH. We also do believe that during surgery, an inadvertent iatrogenic LMH could have been caused due to ILM peeling trauma. It is, however, interesting that the LHEP occurred only around the LMH and not around the FTMH. It has been speculated that ILM delamination may cause a retinal weakening by Müller cell damage. This may be followed by a structural breakdown and consequently hole formation.[6] Histopathological analysis has shown that LHEP consists primarily of fibroblasts and hyalocytes.[8] These epiretinal proliferations are believed to have their origin from Müller cells and react positively with antiglial fibrillary acidic protein and antiglutamine synthetase, a Müller cell–specific antibody.[1],[4]

It may be subsequently hypothesized that traumatic holes may trigger a more pronounced proliferation of the Müller cells within the substance of the hole. In instances where there is an iatrogenic inner layer defect of the retina, Müller cell damage and proliferation could be triggered and LHEP could follow. Pang et al.[9] have reported that eyes with LMH and LHEP were associated with poorer visual acuity, larger LMH diameters, thinner retina, and higher ellipsoid disruption, suggesting a process involving more severe retinal tissue loss and injury.

Long-term follow-up of LHEP has revealed an increased area of LHEP that correlated with the enlargement of hole diameter and ellipsoid zone defects.[10] Hence, while ours is not the first report to document a long-term follow-up of LHEP, it documents the filling up of the lamellar hole, which is previously unreported. It can also be observed that there were no changes in the ellipsoid zone underlying the LMH and LHEP. A few noteworthy points from the literature are cytohistological reports that LHEP is likely of Müller cell origin, OCT evidence of the tissue originating from the middle layers within a retinal defect, and the observation that LHEP can occur in situations without a lamellar hole.[1],[4],[6] In view of these reports, we feel that the term LHEP does not best describe this pathology; a better term could be Müller cell proliferation. This best represents the tissue of origin and does not limit the pathology to lamellar holes. Additionally, it justifies the early location of this abnormal tissue within retinal defects even before it reaches the epiretinal location. The patient was not keen on surgical management; otherwise, it would have been interesting to study the LHEP histopathologically. Although we tend to believe that LHEP associated with idiopathic LMH may be different from those associated with traumatic/iatrogenic LMH, we would like to exercise caution in the surgical removal of these proliferations as poor outcomes have been described.

  Conclusion Top

In conclusion, we report a well-documented case of LHEP associated with an iatrogenic paracentral lamellar hole, which progressed and filled up the hole over a 3-year period all the while uninvolving the primary FTMH.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

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Conflicts of interest

There are no conflicts of interest.

  References Top

Pang CE, Spaide RF, Freund KB. Epiretinal proliferation seen in association with lamellar macular holes: A distinct clinical entity. Retina 2014;34:1513-23.  Back to cited text no. 1
Compera D, Entchev E, Haritoglou C, Mayer WJ, Hagenau F, Ziada J, et al. Correlative microscopy of lamellar hole-associated epiretinal proliferation. J Ophthalmol 2015;2015. doi: 10.1155/2015/450212.  Back to cited text no. 2
Compera D, Entchev E, Haritoglou C, Scheler R, Mayer WJ, Wolf A, et al. Lamellar hole-associated epiretinal proliferation in comparison to epiretinal membranes of macular pseudoholes. Am J Ophthalmol 2015;160:373-84.e1  Back to cited text no. 3
Pang CE, Maberley DA, Freund KB, White VA, Rasmussen S, To E, et al. Lamellar hole-associated epiretinal proliferation: A clinicopathologic correlation. Retina 2016;36:1408-12.  Back to cited text no. 4
Ko J, Kim GA, Lee SC, Lee J, Koh HJ, Kim SS, et al. Surgical outcomes of lamellar macular holes with and without lamellar hole-associated epiretinal proliferation. Acta Ophthalmol 2017;95:e221-6.  Back to cited text no. 5
Steven P, Laqua H, Wong D, Hoerauf H. Secondary paracentral retinal holes following internal limiting membrane removal. Br J Ophthalmol 2006;90:293-5.  Back to cited text no. 6
Rubinstein A, Bates R, Benjamin L, Shaikh A. Iatrogenic eccentric full thickness macular holes following vitrectomy with ILM peeling for idiopathic macular holes. Eye (Lond) 2005;19:1333-5.  Back to cited text no. 7
dell'Omo R, Virgili G, Rizzo S, De Turris S, Coclite G, Giorgio D, et al. Role of lamellar hole-associated epiretinal proliferation in lamellar macular holes. Am J Ophthalmol 2017;175:16-29.  Back to cited text no. 8
Pang CE, Spaide RF, Freund KB. Comparing functional and morphologic characteristics of lamellar macular holes with and without lamellar hole associated epiretinal proliferation. Retina 2015;35:720-6.  Back to cited text no. 9
Compera D, Schumann RG, Cereda MG, Acquistapace A, Lita V, Priglinger SG, et al. Progression of lamellar hole-associated epiretinal proliferation and retinal changes during long-term follow-up. Br J Ophthalmol 2018;102:84-90.  Back to cited text no. 10


  [Figure 1], [Figure 2]


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