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Journal of EuCornea 2 (2019) 6–9

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JournalofEuCornea

journal homepage: www.elsevier.com/locate/xjec

Cornealepithelialrestorationafterpenetratingkeratoplastyinrepeated
T
failedcultivatedlimbalstemcellgrafts
JoséphineBehaegela,b,c,⁎ ,CarinaKoppen VeerleVanGerwena ,Marie-JoséTassignon
CarinaKoppena,b,VeerleVanGerwen Marie-JoséTassignona,b,
SorchaNíDhubhghailla,b
a FacultyofMedicineandHealthSciences:DeptofOphthalmology,VisualOpticsandVisualRehabilitation,UniversityofAntwerp,CampusDrieEiken–GebouwT;T4–

Oogheelkunde,Universiteitsplein1,2610Antwerpen,Belgium
b DepartmentofOphthalmology,AntwerpUniversityHospital,Wilrijkstraat10,2650Edegem,Belgium
c DepartmentofOphthalmology,UniversityHospitalBrussels,Laarbeeklaan101,1090Jette,Belgium

ARTICLEINFO ABSTRACT

Keywords:
Limbalstemcelldeficiency
Cultivatedlimbalstemcelltransplantation
Cornealepithelialhomeostasis
Purpose: To report a case of corneal epithelial restoration after penetrating keratoplasty following multiple
complicationsinrepeatedfailedcultivatedstemcellgrafts.
Methods: Thisisacasereportofa62-yearoldpatientwhowastreatedforunilateralseverelimbalstemcell
deficiencyafterachemicalburnoftherighteye.
Results: Two attempts to reconstruct the ocular surface using autologous cultivated limbal stem cell grafts
(CLET)failed,andwerecomplicatedbyperforationsrequiringtwoemergencypenetratingkeratoplasties.While
the first donor cornea revascularised quickly, the corneal epithelium of the second remained transparent,
avascularandstableuntiloneyearpostoperatively.Invivoconfocalmicroscopicexaminationconfirmedpre-
senceofahealthycornealepithelialphenotype.
Conclusions: Occurrenceofseriousadverseeventsarerelatedtoaninadequateepithelialhomeostasisfollowing
CLET.Themechanismsofactionoflimbalstemcelltransplantationarestillpoorlyunderstoodandthecorneal
epithelialintegritymaybemaintainedwithoutlimbalinput.

1. Introduction significant stromal disease, however, visual improvement is not ex-

Limbalstemcelldeficiency(LSCD)isarareclinicalentityresulting
fromalossordeficiencyofthestemcellsinthecorneallimbusthatare
essentialforcornealepithelialregeneration.Whenthestemcellsare
lost,thecornealepithelialhomeostasisfailsandthecellsarereplaced
byaconjunctiva-likelayer,resultinginlossoftransparency,vascular-
isation and corneal blindness [1]
[1]. Corneal homeostasis is dependent
upontheself-renewingpropertiesofthecornealepithelium.Thepre-
vailinghypothesisisthatthesepropertiesrelyonthelimbalepithelial
stem cells located at level of the limbal palisades of Vogt [1,2]
[1,2]. Al-
thoughthetherapeuticoptionsforLSCDareimproving,themanage-
mentofthesecasesremainschallenging.
Cultivatedlimbalstemcelltransplantation(CLET)issurgicalcell-
basedtherapythataimstorestorethelimbalfunctionsandimprovethe
cornealmicroenvironment.Whileitsexactmechanismofactionisstill
debated, the technique has proven to be successful in restoring an
avascularcornealepitheliuminselectedcases [3] [3].Inthepresenceof
pectedtooccurafterCLETandthesepatientsultimatelyrequireape-
netrating keratoplasty (PK) to improve vision. Performing a kerato-
plastywithoutstemcellrepairwillfailduetoimproperhealingand
revascularisation [4]
[4], and most centres recommend performing a PK
onlyaftersuccessfulepithelialrepair.
InthisreportwedescribeaseverecaseofLSCDwhosetreatments
were complicated by adverse events where two consecutive CLETs
failed.Unexpectedly,anemergencyPKwithamnionmembranetrans-
plantation successfully restored the corneal epithelium. This case il-
lustratesononehandthechallengesofsevereLSCD,andontheother
handquestionsthemechanismofactionoflimbalstemcellsandCLET
therapy.
2. Casepresentation
A 62-year old male patient presented for a second opinion, with
severe limbal stem cell deficiency in the right eye one year after a

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⁎ Correspondingauthorat:DepartmentofOphthalmology,AntwerpUniversityHospital,Wilrijkstraat10,2650Edegem,Belgium.

E-mailaddresses: josephine.behaegel@uzbrussel.be (J.Behaegel), carina.koppen@uza.be (C.Koppen), Veerle.vangerwen@uza.be (V.VanGerwen),

marie-jose.tassignon@uza.be (M.-J.Tassignon), sorcha.nidhubhghaill@uza.be (S.NíDhubhghaill).

https://doi.org/10.1016/j.xjec.2019.03.001
Received24October2018;Receivedinrevisedform25February2019;Accepted3March2019
Available online 05 March 2019