r e v b r a s r e u m a t o l . 2015;55(4):387–389
w w w . r e u m a t o l o g i a . c o m . b r
REVISTA
BRASILEIRA
DE
REUMATOLOGIA
Case
report
Peripheral
corneal
melting
syndrome
in
psoriatic
arthritis
treated
with
adalimumab
夽
Juan
Pablo
Restrepo
a,∗,
Luis
Fernando
Medina
b,
María
del
Pilar
Molina
caUniversidadedoQuindío,Armenia,Colombia bUniversidadedoVale,Cali,Colombia cColsalud,Armenia,Colombia
a
r
t
i
c
l
e
i
n
f
o
Articlehistory:
Received5June2013
Accepted21October2013
Availableonline26November2014
Keywords:
Cornealperforation
Psoriaticarthritis
Adalimumab
a
b
s
t
r
a
c
t
Peripheralcornealmeltingsyndromeisarareimmuneconditioncharacterizedbymarginal
corneal thinningand sometimesperforation.Itis associatedwithrheumaticand
non-rheumaticdiseases.Fewcasesofperipheralcornealmeltinghavebeenreportedinpatients
withpsoriasis.Thepathogenesisisnotfullyunderstoodbutmetalloproteinasesmayplay
apathogenicrole.Anti-TNFtherapyhasshowntodecreaseskinandserum
metallopro-teinaseslevelsinpsoriasis.Wereporta61-year-oldmanwithperipheralcornealmelting
syndromeassociatedwithpsoriaticarthritiswhoreceivedadalimumabtocontrolskinand
ocularinflammation.Toourknowledge,thisisthefirstcasereportofperipheralcorneal
meltingsyndromeinpsoriaticarthritistreatedwithadalimumabshowingresolutionofskin
lesionsandcompletehealingofcornealperforationinthreemonths.
©2014ElsevierEditoraLtda.Allrightsreserved.
Síndrome
da
ceratomalácia
(
Corneal
Melting
)
periférica
na
artrite
psoriásica
tratada
com
adalimumabe
Palavras-chave:
Perfurac¸ãodacórnea
Artritepsoriásica
Adalimumabe
r
e
s
u
m
o
Asíndromedocornealmeltingperiféricaéumararacondic¸ãoimunecaracterizadapor
afi-namentodamargemdacórneae,àsvezes,perfurac¸ão.Estáassociadaadoenc¸asreumáticas
enãoreumáticas.Poucoscasosdesíndromedocornealmeltingperiféricaforam
relata-dosempacientescompsoríase.Apatogênesenãofoicompletamenteentendida,masas
metaloproteinasespodemterpapelpatogênico.AterapiaAnti-TNFdiminuiuosníveisde
metaloproteinasesnapeleenosangueempsoríase.Reportamosocasodeumhomemde
61anoscomsíndromedocornealmeltingperiféricaassociadaàartritepsoriásicaque
rece-beuadalimumabeparacontrolarainflamac¸ãonapeleenoolho.Peloquesabemos,esteéo
primeirocasodesíndromedocornealmeltingperiféricaemartritepsoriáticatratadocom
夽
ThecasewasoriginatedinServicioOccidentaldeSalud,Rheumatologysection,Cali,Colombia.
∗ Correspondingauthor.
E-mail:[email protected](J.P.Restrepo).
http://dx.doi.org/10.1016/j.rbre.2013.10.006
388
rev bras reumatol.2015;55(4):387–389adalimumabemostrandoevoluc¸ãonaslesõescutâneasecuratotaldaperfurac¸ãodacórnea
emtrêsmeses.
©2014ElsevierEditoraLtda.Todososdireitosreservados.
Introduction
Peripheralcornealmeltingsyndrome(PCMS)isarareimmune
condition characterized by marginal corneal thinning and
sometimes perforation. It is associated with rheumatic
and non-rheumatic diseases.The list includesrheumatoid
arthritis,Sjogren’ssyndrome,systemiclupuserythematosus,
Wegener’sgranulomatosis, microscopicpolyangiitis, topical
non-steroidalanti-inflammatorydrugs,pregnancyand
psori-asis.Ocularmanifestationsofpsoriasisarerareandextensive.
Psoriasiscanaffectthelids,conjunctiva,corneaandanterior
uvealtract.ThepathogenesisofPCMSisnotfullyunderstood,
butmetalloproteinasesmayplayapathogenicrole.Anti-TNF
therapyhasshowntodecreaseskinandserum
metallopro-teinaseslevelsinpsoriasisandpsoriaticarthritis.Wereport
a61-year-oldmanwithperipheralcornealmeltingsyndrome
associatedwithpsoriasiswhoreceivedadalimumabto
con-trolskinandocularinflammation.Sinceweknowthisisthe
first casereportofPCMS inpsoriatic arthritistreated with
adalimumab.
Case
report
A61-year-oldmanwasreferredtoourrheumatologyservicein
October2010withafour-monthhistoryofleftblurredvision,
photophobiaand painful red eye. Hehad a 6-year chronic
plaquepsoriasiswitharthritisanduveitis.
An ophtalmologic examination revealed a left inferior
paracentralperforation.Schirmer’stestwas5mm/5minand
slit-lampexaminationdidnotshowcellsintheanterior
cham-ber. Due tothe severity ofthe casehewas managed with
cyanoacrylateglue (Fig. 1) andtopicalatropine,prednisone
plusphenylephrine,carboxymethylcellulose,ciprofloxacine.
Fig.1–Leftinferiorparacentralperforationoccludedwith cyanoacrylateglue.
Rheumatologyevaluationshowedswan-neckfingerswith
ulnardeviationandirreversiblelimitationinrangeofmotion
withoutinflammatorysigns.Thephysicalexaminationalso
revealed extense plaques ofpsoriasis involving the trunk,
upperandlowerlimbs.
Bloodcount,glucose,liverfunctiontests,CRPwerenormal,
creatininewas1.44mg/dL(<1mg/dL)withbloodureanitrogen:
8.3mg/dL,anderythrocytesedimentationratewas32mm/h.
Rheumatoidfactor,anti-CCP,antinuclearantibodies,anti-Ro,
anti-La,anti-SM,anti-RNP,HLA-B27werenegative.Thehands
radiographyshowedthetypicalpencil-in-cupdeformity.
Two weeksafterPCMS occurredwestarted adalimumab
80mgfollowedby40mgeveryotherweekadministered
sub-cutaneously with resolution of skin lesions and complete
healingofcornealperforationin3months(Fig.2).
Discussion
Corneal melting syndrome is a rare disease consisting of
corneal thinning with ulceration that sometimes leads to
perforation.1Itismostcommonlyassociatedwith
rheuma-toid arthritis, followed by Wegener’s granulomatosis and
microscopicpolyangiitis respectively.2 Othercausesinclude
Sjogren’ssyndrome,3systemiclupuserythematosus,4topical
non-steroidal anti-inflammatory drugs,5 pregnancy,6
pol-yarteritisnodosaandpsoriasis.7
Psoriasis affectsnearlyeverylayeroftheeyein
approx-imately 10% ofthe cases.Ophalmologicfeatures consistof
cornealopacities,conjunctivitis,superficialkeratitis,chronic
iridocyclitis, uveitisanddry eyes.8PCMS beginswith
ulcer-ation and melting of the cornea; without a prompt and
appropriatedtreatmentthepatientcouldexperiencea
perfo-rationandfinallythelossofvisionandintegrityoftheglobe.
Thediseasemaybeclassifiedbyanatomicallocation:central
rev bras reumatol.2015;55(4):387–389
389
Table1–SummaryoffourcasesofPCMSassociatedwithpsoriasis.1,8
Case Gender Age Location Perforationarthritis Psoriasistype
1 Female 62 Peripheral + Pustular
2 Male 70 Peripheral − Plaques
3 Female 83 Peripheral − Plaques
4 Male 61 Peripheral + Plaques
andperipheralor bypathogenic mechanism: inflammatory andnon-inflammatory.Fourcasesincludingourshavebeen reported(Table1).
ThepathogenesisofPCMS isstillunclear.PCMS can be
initiated by local factors including ophthalmic vasculitis,
ocularsurfacediseaselike cornealinfection and sicca
syn-drome. Increased levels ofmatrix metalloproteinases 1, 2,
3, 9 in melted corneal specimens have been found.9 The
productionofmetalloproteinasescandegradatebasal
mem-brane componentsof thecornea leading toulcerationand
finally perforation. It is rational the treatment with
adali-mumabsinceanti-TNFtherapyhasdemonstratedtodecrease
theexpressionofmetalloproteinases inskinand serum of
patientswithpsoriasisandpsoriaticarthritis.10,11Local
treat-ment of PCMS consists in the use of cyanoacrylate glue
whichcaneffectivelysealsmallcorneal(<3mmdiameter)and
uncomplicatedperforationsandthemeandurationforglue
insituwas45–72days.12
Finally, we believe that despite PCMS in psoriasis and
psoriaticarthritis israre, the role ofmetalloproteinases in
cornealdestructionsuggeststhatthiscomplicationduringthe
courseofpsoriaticarthritiscouldbeacausalassociation.More
researchisneededtodeterminewhethertheuseofanti-TNF
therapycouldbeusefulinacceleratingcornealhealingPCMS
inpsoriaticarthritis.
Conflicts
of
interest
Theauthorsdeclarenoconflictsofinterest.
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