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A New Approach to a Difficult Problem Research Original Investigation Restoration of Corneal Sensation With Regional Nerve Transfers and Nerve Grafts A New Approach to a Difficult Problem Uri Elbaz, MD; Robert Bains, MBChB, FRCS(Plast); Ronald M. Zuker, MD, FRCSC; Gregory H. Borschel, MD; Asim Ali, MD, FRCSC CME Quiz at IMPORTANCE Corneal anesthesia is recalcitrant to conventional treatment and can lead to jamanetworkcme.com and permanent visual loss. CME Questions 1388 OBJECTIVE To assess the outcomes of a novel sensory reconstructive technique for the treatment of corneal anesthesia. DESIGN, SETTING, AND PARTICIPANTS This prospective study evaluating a new technique was conducted at a tertiary referral center. Four eyes in 3 patients with corneal anesthesia underwent nerve transfers with nerve grafting to restore corneal sensation. Corneal sensory reconstruction was performed using a segment of the medial cutaneous branch of the sural nerve. Two patients with unilateral trigeminal nerve anesthesia—one following basal skull fracture and another following large posterior fossa tumor resection—underwent corneal sensory reconstruction using the contralateral supratrochlear nerve as the donor sensory nerve. One patient with a history of cerebellar hypoplasia and bilateral congenital corneal anesthesia underwent bilateral corneal sensory reconstruction using the respective ipsilateral supratrochlear nerves as the sensory donor nerves. Corneal anesthesia was evaluated preoperatively and postoperatively in the center of the cornea and in 4 corneal quadrants using a Cochet-Bonnet esthesiometer (Luneau). Complications of the procedure were also documented. MAIN OUTCOMES AND MEASURES Esthesiometry scores. RESULTS All eyes had prior complications of corneal anesthesia and had no measurable corneal sensation in the affected eye(s) preoperatively. Two patients—one with cerebellar hypoplasia and the other with posterior fossa tumor resection—had markedly improved corneal sensation 6 months postsurgery (3 eyes; mean [SD] central esthesiometry, 55 [5] mm). A third patient with a history of basal skull fracture underwent unilateral corneal neurotization and recovered 15-mm esthesiometry score centrally after 7.5 months of follow-up. None of the operated on eyes have developed corneal anesthesia–related complications since reconstruction. CONCLUSIONS AND RELEVANCE Corneal sensory reconstruction provides corneal sensation in previously anesthetic corneas. This can be achieved with minimal morbidity using sural nerve grafts, which surgeons commonly use to reconstruct nerve gaps elsewhere. This Author Affiliations: Department of multidisciplinary approach restores an ocular defense mechanism and may enable Ophthalmology and Vision Sciences, subsequent corneal transplant in these patients. Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada (Elbaz, Ali); Division of Plastic and Reconstructive Surgery, Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada (Bains, Zuker, Borschel). Corresponding Author: Asim Ali, MD, FRCSC, Department of Ophthalmology and Vision Sciences, Hospital for Sick Children, University of Toronto, 555 University Ave, JAMA Ophthalmol. 2014;132(11):1289-1295. doi:10.1001/jamaophthalmol.2014.2316 Toronto, ON M5G 1X8, Canada Published online July 10, 2014. ([email protected]). 1289 Copyright 2014 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 10/01/2021 Research Original Investigation Restoration of Corneal Sensation he insensate cornea represents a difficult problem that has defied definitive management. Corneal sensation Methods T is critical in maintaining epithelial integrity1 and lim- bal stem cell function.2 Reduced corneal sensation renders This study was approved by the Research Ethics Board of the the corneal surface prone to injury and decreased reflex tear- Hospital for Sick Children, Toronto, Ontario, Canada, and ing. In addition, the poor healing3 secondary to corneal sen- written informed consent was obtained from patients and/or sory denervation favors the formation of nonhealing epithe- parents/guardians. We included in the study 4 eyes of 3 con- lial defects that ulcerate and perforate if not appropriately secutive patients who had undergone corneal neurotization treated. surgery using a nerve graft. A surgical recommendation was Corneal anesthesia can be categorized into acquired or rendered after mapping patients’ sensation over the face to congenital causes. Its acquired form arises from infectious, ensure sensation in the territory of the donor supratrochlear inflammatory, traumatic, neoplastic, and iatrogenic causes, nerve. The primary outcome measure was the degree of cor- all of which impair the function of the ophthalmic division of neal sensation following surgery. Corneal anesthesia was the trigeminal nerve and lead to neurotrophic keratopathy. evaluated preoperatively and postsurgical intervention cen- The congenital form of this condition may be either isolated trally and in 4 quadrants using a Cochet-Bonnet esthesiom- or syndromic.4-6 Furthermore, corneal anesthesia can be eter (Luneau).16 Corneal sensation of 60 mm was considered either confined to the ophthalmic nerve or alternatively asso- normal. ciated with other ocular conditions, such as strabismus, lag- Two patients underwent unilateral corneal neurotization ophthalmos, or paralytic ectropion, depending on the etiol- and 1 patient underwent bilateral corneal neurotization. All sur- ogy and the extent of involvement of other cranial nerves. gical procedures were performed under general anesthesia. Children may experience photophobia and decreased vision Harvesting of the median cutaneous branch of the sural nerve without pain or distress. Signs may vary from mild conjuncti- and dissection of the donor supratrochlear nerve were car- val injection to sight-threatening conditions, such as corneal ried out simultaneously. The sural nerve was harvested proxi- ulceration or perforation, especially if the facial nerve is mal to distal using a nerve harvesting device (Figure 1A).17 The affected. Unfortunately, few treatment options preserve nerve harvesting device was placed around the nerve and vision. Initial management seeks to maintain the integrity of passed distally to free the nerve from surrounding tissues. The the ocular surface. In cases where a nonhealing corneal epi- nerve was then divided, yielding approximately 10 to 15 cm thelial defect is present, a tarsorrhaphy is mandatory. Ulti- of nerve graft (Figure 1B). The peroneal component of the nerve mately, in the event of corneal perforation, cyanoacrylate was preserved but could be used through separate incisions, gluing or tectonic corneal transplantation are required. How- if required. ever, because of the lack of sensation and the resultant sub- The supratrochlear nerve was found on the surface of the stantial reduction in corneolimbal stem/progenitor cells,2 corrugator supercilii muscle passing cephalad from the supra- grafted corneas do not heal well and therefore graft longevity trochlear notch. This was accessed through a transverse inci- is limited with poor visual prognosis. sion over the medial upper eyelid just inferior to the brow, deep Neurotization involves the transfer of a healthy donor to the origin of the frontalis (Figure 1C and D). An epineural nerve segment into a tissue to reestablish either motor7,8 or window was then created in the side for end-to-side coapta- sensory9,10 innervation. Neurotization is a well-established tion of the sural nerve graft. End-to-side coaptation was our modality of treatment for different clinical conditions9-14 and preferred option to maximally preserve forehead sensation; the technique continues to evolve and gain popularity.7,8 however, in the bilateral case, the supratrochlear nerves were Direct neurotization of the anesthetic cornea was previously found to be small so we divided the nerve distally and per- reported by Terzis et al15 in adults with unilateral facial formed the coaptation end to end. In unilateral cases, the op- paralysis and corneal anesthesia. The technique involved posite supratrochlear nerve was used, necessitating subcuta- the transfer of the contralateral supraorbital and supratroch- neous tunneling of the nerve graft over the nasal bridge lear nerves to the limbal area of the anesthetic cornea. (Figure 1E). Although successful in restoring sensation to the cornea, this The distal nerve graft was reversed and tunneled subcon- approach denervates the contralateral forehead and scalp, junctivally to the perilimbal area of the cornea using a medium- requires a large bicoronal incision with subsequent scarring, sized eyelet Wright needle (Figure 1F). Distally, the epineu- and is not applicable in bilateral palsies wherein all branches rium was removed and the individual fascicles were separated of the ophthalmic division of the trigeminal nerve are (Figure 1G). The fascicles were then placed around the entire affected. limbal circumference to reinnervate all 4 quadrants (Figure 1H The purpose of this study was to report early outcomes shows the separated fascicles prior to tunneling under the con- following a novel corneal neurotization technique in young junctiva; Figure 1I) and secured to the sclera with 10-0 nylon patients. To our knowledge, this is the first report of restora- sutures (Figure 1J). Proximally, coaptation of the graft to the tion of corneal sensation using a sural nerve graft in unilat- cut surface of the supratrochlear nerve was
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