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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 1  |  Issue : 4  |  Page : 637-639

Combined deep anterior lamellar keratoplasty and sural nerve transplant in a child with corneal anesthesia and maculo-leucomatous opacity


1 Cornea Consultant, Sankara Eye Hospital, Bengaluru, Karnataka, India
2 Oculoplasty Consultant, Sankara Eye Hospital, Bengaluru, Karnataka, India
3 Oculoplasty Consultant, Apollo & Fortis, Navi Mumbai, Maharashtra, India
4 Cornea Department, Sankara Eye Hospital, Bengaluru, Karnataka, India

Date of Submission18-Dec-2020
Date of Acceptance20-Mar-2021
Date of Web Publication09-Oct-2021

Correspondence Address:
Dr. Devika Singh
Department of Cornea and Refractive Services, Sankara Eye Hospital, Varthur Road, Kundalahalli, Bangalore - 560 037, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_3452_20

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  Abstract 


Sural nerve transplantation is an established procedure for neurotization in patients with corneal anaesthesia. In a child presenting with corneal anesthesia and maculo-leucomatous opacity, a combined deep anterior lamellar keratoplasty (DALK) and sural nerve transplant procedure was done. Under general anesthesia, a sural nerve graft was obtained from the child's own foot and co-apted with contra-lateral supra-orbital nerve. Simultaneously, lamellar dissection of host corneal bed, followed by the apposition of the graft was done. At 1 year follow up, visual acuity had improved, the graft was clear,avascular and the corneal epithelium was healthy.The corneal sensations were appreciated in all quadrants.The child was started on amblyopia therapy. Combined keratoplasty with neurotization surgery is a feasible alternative to staged surgical procedure. It helps achieve faster visual and anatomical recovery.

Keywords: Corneal anesthesia, deep anterior lamellar keratoplasty, sural nerve transplant


How to cite this article:
Joshi P, Rambhatla S, Moreker SR, Singh D. Combined deep anterior lamellar keratoplasty and sural nerve transplant in a child with corneal anesthesia and maculo-leucomatous opacity. Indian J Ophthalmol Case Rep 2021;1:637-9

How to cite this URL:
Joshi P, Rambhatla S, Moreker SR, Singh D. Combined deep anterior lamellar keratoplasty and sural nerve transplant in a child with corneal anesthesia and maculo-leucomatous opacity. Indian J Ophthalmol Case Rep [serial online] 2021 [cited 2021 Oct 18];1:637-9. Available from: https://www.ijoreports.in/text.asp?2021/1/4/637/327670



Corneal anesthesia is an extremely rare condition carrying diagnostic and therapeutic challenges. The cornea may be affected in isolation or with other ocular and systemic anomalies.[1] Corneal neurotization is a method for surgical restoration of nerve supply in a previously anesthetic cornea.


  Case Report Top


A 9-year-old boy presented with complaints of diminution of vision and a history of recurrent redness in the left eye from 3 months of age. His parents gave a history of fingernail trauma at 3 months of age, following which he developed central corneal whitening. He was being treated locally with intermittent topical antibiotics and lubricating eye drops for a nonhealing ulcer. On ocular examination, his corrected distance visual acuity (CDVA) was 20/20 and finger counting in the right and left eye, respectively. The right eye was normal. The left eye anterior segment examination revealed a 6 × 3 mm maculo-leucomatous corneal opacity (MLCO) with two clock hours of deep vascularization in the temporal quadrant [Figure 1]. Mild punctate corneal fluorescein staining was seen. The anterior chamber was quiet, with no conjunctival congestion, and the lens was clear. Corneal sensations were absent. Fundus was normal bilaterally. Anterior segment optical coherence tomography (AS-OCT) in left eye showed an ectatic cornea with deep stromal hyperreflectivity measuring 316 microns in depth sparing the descemet membrane. Complete pediatric systemic evaluation was normal.
Figure 1: Preoperative image of left eye showing 6 × 3 mm maculo-leucomatous corneal opacity involving the visual axis with two clock hours of deep corneal vascularization

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A presumptive diagnosis of left eye postviral keratitis MLCO with corneal anesthesia was made. Management options such as recombinant nerve growth factor and scleral contact lenses were infeasible due to the nonaffordability. After a detailed discussion with his parents, the patient was planned for left eye deep anterior lamellar keratoplasty (DALK) with a sural nerve transplant.

Under general anesthesia, a multifaceted team of ophthalmologists worked concurrently toward nerve graft procurement, keratoplasty, and nerve coaptation. Four inches of the sural nerve was harvested from the left leg. Simultaneously, after manual lamellar dissection of the host corneal bed, a 7.5-mm donor corneal button was grafted and secured with 16 interrupted sutures. Via a subbrow incision, a blunt dissection was carried out bilaterally down to the medial orbital rim. Side-to-end coaptation of right supraorbital nerve with the proximal end of sural nerve graft was done. Sural nerve graft was passed submusculocutaneous to the left subbrow area, and the distal end was dissected into five fascicles that were secured within three perilimbal scleral tunnels. The distal ends were then tunneled up to the limbus to provide a source of axons for corneal reinnervation [Figure 2]. Standard postkeratoplasty topical regimen was prescribed.
Figure 2: Postoperative Day 1 image showing the left subbrow incision (red arrow) for coaptation of right supraorbital nerve and sural nerve graft (a). Nerve fascicles seen secured concentrically around the limbus (white arrows) (a). One year postsurgery image showing a clear graft with well apposed margins, subconjunctival nerve fascicle is also seen (white arrow) (b). One year postsurgery AS-OCT scan through the center of the cornea showing the lamellar graft (c) and through inferonasal limbus showing a hyporeflective area corresponding to the nerve fascicle (d)

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The corneal lamellar button was sent for a histopathological examination, confirming the absence of nerve tissue in the specimen [Figure 3].
Figure 3: Histopathological image of the dissected corneal button. Specimen showing corneal tissue lined by stratified squamous epithelium. Stroma shows scattered blood vessels and sparse lymphoid cells. No nerve bundle or plexus was identified in any section

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Postoperatively, on 3 months follow-up, the patient was able to perceive light touch sensation with a cotton wisp. He localized the sensation to the other side brow. Amblyopia therapy was initiated, and appropriate glasses were prescribed. Subsequently, visual acuity at 6 months follow-up improved to 3/60. No neurotization and corneal graft–related complications from the surgery were noted. Mild sensory deficit was observed at the donor site on the left foot, which improved at 6 months. At 1 year follow-up, visual acuity was stable, the graft was noted to be clear, and no vascularization was noticed in any quadrant [Figure 2]. The patient was advised to continue medications and amblyopia therapy.


  Discussion Top


The spectrum of corneal lesions in children with corneal anesthesia includes neurotrophic ulcers, impending perforation, and secondary infectious keratitis.[1] Treatment of a nonhealing ulcer consists of topical nerve growth factors and intensive ocular surface lubrication with preservative-free artificial tears.[2] Surgical options, such as tarsorrhaphy, amniotic membrane transplantation, and conjunctival flaps, are used in advanced stages, but these are temporary measures.[2] Surgical advances such as corneal neurotization surgery have shown promise for a permanent solution.

Animal studies have shown that decreased corneal innervation reduces the corneolimbal progenitor/stem cell number and function, which can possibly lead to nonhealing epithelial surfaces.[3] Postsurgical neurotization, the corneal nerves secrete substances, such as Substance Pand calcitonin gene-related peptide, that aid in corneal epithelial proliferation and wound healing.[3],[4] Conversely, the corneal epithelial cells secrete growth factors and neurotrophins such as nerve growth factor and ciliary neurotrophic factor that promote corneal nerve survival.[4],[5]

Corneal neurotization was introduced in 2009 by Terzis et al.[5] Many neurotization techniques are in development and different nerves (sural nerve, great auricular nerve) have been explored for interposition between the branches of frontal nerve and the cornea.[5] Current literature states that corneal neurotization can help objectively improve corneal sensation and maintain corneal epithelial health.[6] Most esthesiometry and confocal microscopy studies predict that corneal nerves regeneration happens 6 months after the procedure.[7]

The outcome measures in children are different from adults as esthesiometry is unachievable, and in vivo confocal microscopy is challenging; thus we used surrogate measures.[8] Impending perforation in patients with corneal anesthesia needs a tectonic graft, but corneal transplantation alone as a primary surgical option has poor outcomes because of a lack of corneal innervation and trophic factors. Although there is limited literature, our experience suggests that nerve transfers using sural nerve grafts combined with corneal transplantation in such patients is a well-tolerated procedure. We chose to do DALK instead of penetrating keratoplasty since the opacity was superficial, and evidence suggests that corneal reinnervation is earlier in DALK compared with penetrating keratoplasty.[9],[10]


  Conclusion Top


A case documenting a single-stage sural nerve transplant with DALK in pediatric acquired corneal anesthesia has not yet been documented in the literature. In children, a combined procedure promotes speedy corneal epithelial healing, permits early amblyopia therapy, and thereby promotes a rapid visual recovery. It also reduces the need for repeated general anesthesia in children.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Ramappa M, Chaurasia S, Chakrabarti S, Kaur I. Congenital corneal anesthesia. JAAPOS 2014;18:427-32.  Back to cited text no. 1
    
2.
Alder J, Mertsch S, Menzel-Severing J, Geerling G. Current and experimental treatment approaches for neurotrophic keratopathy. Ophthalmologe 2019;116:127-37.  Back to cited text no. 2
    
3.
Sacchetti M, Lambiase A. Neurotrophic factors and corneal nerve regeneration. Neural Regen Res 2017;12:1220-4.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Yang AY, Chow J, Liu J. Corneal innervation and sensation: The eye and beyond. Yale J Biol Med 2018;91:13-21.  Back to cited text no. 4
    
5.
Terzis JK, Dryer MM, Bodner BI. Corneal neurotization: a novel solution to neurotrophic keratopathy. Plast Reconstr Surg 2009;123:112–20.   Back to cited text no. 5
    
6.
Leyngold IM, Yen MT, Tian J, Leyngold MM, Vora GK, Weller C. Minimally invasive corneal neurotization with acellular nerve allograft: Surgical technique and clinical outcomes. Ophthalmic Plast Reconstr Surg 2019;35:133-40.  Back to cited text no. 6
    
7.
Koaik M, Baig K. Corneal neurotisation. Current Opin Ophthalmol 2019;30:292-8.  Back to cited text no. 7
    
8.
Sepehripour S, Lloyd MS, Nishikawa H, Richard B, Parulekar M. Surrogate outcome measures for corneal neurotization in infants and children. J Craniofac Surg 2017;28:1167-70.  Back to cited text no. 8
    
9.
Keane M, Coster D, Ziaei M, Williams K. Deep anterior lamellar keratoplasty versus penetrating keratoplasty for treating keratoconus. Cochrane Database Syst Rev. 2014 Jul 22;(7):CD009700.  Back to cited text no. 9
    
10.
Lin X, Xu B, Sun Y, Zhong J, Huang W, Yuan J. Comparison of deep anterior lamellar keratoplasty and penetrating keratoplasty with respect to postoperative corneal sensitivity and tear film function. Graefes Arch Clin Exp Ophthalmol 2014;252:1779-87.  Back to cited text no. 10
    


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  [Figure 1], [Figure 2], [Figure 3]



 

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