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Middle East African Journal of Ophthalmology Middle East African Journal of Ophthalmology
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CASE REPORT
Year : 2017  |  Volume : 24  |  Issue : 4  |  Page : 216-218  

Localized trichiasis causing focal full-thickness corneal edema, endothelial cell loss, and corneal scarring requiring penetrating keratoplasty


Department of Ophthalmology and Visual Science, Yale School of Medicine, New Haven, CT, USA

Date of Web Publication12-Jan-2018

Correspondence Address:
Tiffany Sara Liu
Department of Ophthalmology and Visual Science, Yale School of Medicine, 40 Temple Street, Suite 3B, New Haven, CT 06510
USA
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/meajo.MEAJO_304_16

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   Abstract 

The purpose of the study was to report a case of focal trichiasis causing full-thickness corneal edema, scarring, and endothelial cell loss requiring penetrating keratoplasty (PK). A 66-year-old male was referred for trichiasis of the right upper eyelid corresponding to an area of full-thickness corneal edema. No keratic precipitates or guttata was noted. Specular microscopy showed diffuse endothelial cell loss. He was treated with topical steroids and acyclovir with epilation of lashes. Anterior chamber paracentesis was negative for varicella-zoster virus, cytomegalovirus, and herpes simplex virus. The patient developed diffuse stromal scarring with a decrease in vision and ultimately underwent PK with preceding eyelid repair. To the best of our knowledge, we present the first reported case of chronic trichiasis causing full-thickness corneal edema, scarring, and endothelial cell loss requiring PK.

Keywords: Cornea, penetrating keratoplasty, trichiasis


How to cite this article:
Liu TS, Meskin S. Localized trichiasis causing focal full-thickness corneal edema, endothelial cell loss, and corneal scarring requiring penetrating keratoplasty. Middle East Afr J Ophthalmol 2017;24:216-8

How to cite this URL:
Liu TS, Meskin S. Localized trichiasis causing focal full-thickness corneal edema, endothelial cell loss, and corneal scarring requiring penetrating keratoplasty. Middle East Afr J Ophthalmol [serial online] 2017 [cited 2019 Dec 6];24:216-8. Available from: http://www.meajo.org/text.asp?2017/24/4/216/223111




   Introduction Top


Corneal transplants are the most common organ transplantation procedure in the United States.[1] The indications for full-thickness penetrating keratoplasty (PK) are declining as new minimally invasive corneal transplantation techniques seek to replace targeted layers of the cornea.[2] However, full-thickness stromal scars from trauma, infection, or chronic edema secondary to endothelial dysfunction still require PK. PK made up 7.2% of total corneal transplants done in 2014.[2]

Superficial keratopathy has been shown to affect the morphology and cell count of the endothelium but has never been reported as a cause for permanent scarring and edema with need for PK.[3] Herein, we describe the first case of trichiasis as a cause of focal full-thickness corneal edema, corneal scarring, and endothelial cell loss eventually requiring PK.


   Case Report Top


A 66-year-old male patient with a history of right upper and lower eyelid laxity repair 4 years ago was referred to a cornea specialist for blurry vision and foreign body sensation for 1 year. He had a medical history of hypercholesterolemia, gout, and back pain, for which he was on atorvastatin, colchicine, and hydrocodone, respectively. He had been started on sodium chloride 5% one drop every 4 h in the right eye by the referring ophthalmologist. He had no history of ocular trauma and no history of any other eye surgery aside from the eyelid repair. Examination of his right eye revealed visual acuity of 20/50, intraocular pressure of 14, and trichiasis of the upper eyelashes with cornea touch. This area corresponded to a 4 mm × 4 mm well-circumscribed focal area of full-thickness corneal edema with epithelial microbullae and 3 + Descemet's folds [Figure 1]a. There were otherwise no keratic precipitates (KPs), cell or flare in the anterior chamber, or guttata in either eye. Specular microscopy of the right eye showed diffuse endothelial cell loss without guttata. On examination of palpebral conjunctiva of both eyes, there was no evidence of prior scarring or concretions. The cornea of the left eye showed no punctate epithelial erosions or any other evidence of ocular surface disease.
Figure 1: (a) Slit-lamp photograph of the right eye in a patient with a well-circumscribed area of focal full-thickness corneal edema. (b) Slit-lamp photograph demonstrating increased diffuse edema and stromal scarring. (c) Slit-lamp photograph demonstrating trichiasis with cornea touch

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Initially, the patient refused any additional eyelid or corneal surgery. He was managed conservatively with epilation/trimming of trichiatic eyelashes on repeat visits, sodium chloride 5% drops, artificial tears, and tobramycin/dexamethasone ointment. For coverage of possible infectious endotheliitis (despite no KPs and a quiet anterior chamber), he was started on oral acyclovir and topical steroids. The patient was very resistant to any surgical intervention and was initially stable with conservation management. However, after 2 years, his visual acuity declined to 20/100, and the previously focal edema became more diffuse with stromal scarring [Figure 1]b and [Figure 1]c. The patient agreed to full-thickness PK with preceding right upper and lower eyelid repair with lash rotation.

Due to the unusual clinical course, an aqueous tap was done before PK to rule out potential viral etiology. Polymerase chain reaction (PCR) analysis of aqueous was negative for varicella-zoster virus (VZV), cytomegalovirus (CMV), and herpes simplex virus (HSV). Surgical pathology of the patient's cornea showed severe endothelial hypocellularity, secondary prominent stromal edema with Descemet's membrane folds, and secondary chronic bullous keratopathy with degenerative fibrous pannus. The cornea was negative for herpes viral DNA (VZV, HSV, and CMV).


   Discussion Top


To the best of our knowledge, we present the first reported case of focal trichiasis causing progressive corneal edema, scarring, and endothelial cell loss requiring PK. While superficial keratopathy such as from trichiasis has been shown to cause stromal edema leading to reversible changes in endothelial cell morphology and a decrease in cell count, it has never been reported to cause such dramatic permanent findings requiring PK.[3] Brooks et al. looked at the effect of different types of superficial keratopathy including keratoconjunctivitis sicca, epithelial basement membrane dystrophy, exposure keratopathy, trichiasis, and contact lens wear on the endothelium. None of the cases were found to have irreversible changes in endothelial morphology or cell count.[4]

In this unusual case, perhaps the persistent and focal microtrauma to the ocular surface from trichiasis led to diffuse endothelial cell loss and scarring. Despite foreign body sensation and irritation, our patient took approximately 1 year to present for more specialized care. Then, for the next 2 years, he deferred more definitive treatment, while his cornea suffered additional microtrauma from recurrent trichiasis.

In cases of isolated epithelial keratopathy causing endothelial changes, the exact mechanism is unknown. In the previously published case series by Brooks et al., inflammatory factors were considered unlikely given the absence of inflammation on examination.[5] Anoxia or interference with osmosis has been considered a possible mechanism given it is known to cause corneal changes in contact lens wearers and could contribute to cases of mechanical irritation as well.[5]

Other potential causes of our patient's corneal findings were excluded. There was no history of trauma or intraocular surgery, no evidence of endothelial dystrophy in either eye, and no iris or angle defects suggestive of an iridocorneal endothelial syndrome. There was no anterior chamber reaction, KP, or elevation of intraocular pressure to suggest an endotheliitis or inflammatory etiology. PCR of the aqueous was negative for the most common viral etiologies. Finally, the surgical histopathology did not identify any other reason for the corneal findings. In our case, we hypothesize that the persistent microtrauma of trichiasis led to a break in Bowman's membrane, which caused full-thickness stromal edema and scarring as a reaction to injury. The amount of injury and edema likely overwhelmed the local endothelial cells, leading to diffuse endothelial cell involvement and ultimately loss.


   Conclusion Top


This is the first case to demonstrate the mechanical effect of trichiasis severe and persistent enough to elicit endothelial decompensation. This highlights the need to proactively treat trichiasis before possible irreversible corneal damage occurs. Finally, further study into the exact underlying mechanism of corneal damage is needed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Bentley TS. U.S. organ and tissue transplant cost estimates and discussion. Milliman Res Rep 2014. Available from: http://www.milliman.com/uploadedFiles/insight/Research/health-rr/1938HDP_20141230.pdf.  Back to cited text no. 1
    
2.
Brooks AM, Grant G, Gillies WE. Corneal endothelial changes in superficial epithelial keratopathy. Aust N Z J Ophthalmol 1986;14:171-5.  Back to cited text no. 2
[PUBMED]    
3.
Brooks AM, Grant G, Gillies WE. Reversible corneal endothelial cell changes in diseases of the anterior segment. Aust N Z J Ophthalmol 1987;15:283-9.  Back to cited text no. 3
[PUBMED]    
4.
Brooks AM, Grant G, Gillies WE. The influence of superficial epithelial keratopathy on the corneal endothelium. Ophthalmology 1989;96:704-8.  Back to cited text no. 4
[PUBMED]    
5.
Park CY, Lee JK, Gore PK, Lim CY, Chuck RS. Keratoplasty in the United States: A 10-year review from 2005 through 2014. Ophthalmology 2015;122:2432-42.  Back to cited text no. 5
[PUBMED]    


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