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Herpetic endotheliitis and trabeculitis with delayed corneal involvement
Endotelitis y trabeculitis herpética con afectación corneal tardía
I. Carrillo-Arroyo, E. Gutiérrez-Díaz, E. Mencía-Gutiérrez
Corresponding author
, P. Gómez-Pérez, M. Montero-Rodríguez
Servicio de Oftalmología, Hospital 12 de Octubre, Universidad Complutense, Madrid, Spain
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hyperthyroidism&#44; ischemic cardiopathy and kidney transplant due to chronic renal insufficiency secondary to chronic pyelonephritis&#46; The systemic and medical treatment included prednisone 5<span class="elsevierStyleHsp" style=""></span>mg&#47;24<span class="elsevierStyleHsp" style=""></span>h &#40;Prednisona Alonga<span class="elsevierStyleSup">&#174;</span>&#44; Sanofi Aventis&#41;&#44; tachrolimus &#40;Prograf<span class="elsevierStyleSup">&#174;</span>&#44; MSD&#41; and mycophenolate mofetil &#40;Cellcept<span class="elsevierStyleSup">&#174;</span>&#44; Roche&#41;&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">The initial exploration gave a VA of 1 in the right eye &#40;RE&#41; and 0&#46;1 in LE&#44; normal biomicroscopy in RE with intense conjunctival injection in LE and lackluster corner with central predominance with slight edema and negative fluorescein staining&#44; without Tyndall&#44; synechiae&#44; keratic precipitates or secretion&#46; IOP was of 18<span class="elsevierStyleHsp" style=""></span>mmHg in RE and 46<span class="elsevierStyleHsp" style=""></span>mmHg in LE&#46; The ocular fundus could be seen with difficulty due to the corneal edema&#44; but the papilla and the macular were normal with the retina applied without peripheral lesions&#46; The gonioscopy showed open angle without synechiae or neovessels&#46; During the exploration&#44; we observed that the patient was drowsy and had difficulty breathing&#44; in addition to exhibiting low height&#44; moderate obesity and a short neck&#46;</p><p id="par0020" class="elsevierStylePara elsevierViewall">The initial diagnosis was ocular hypertension in LE&#44; and treatment was established with a fixed combination of 0&#46;2&#37; brimonidine and 0&#46;5&#37; timolole maleate &#40;Combigan<span class="elsevierStyleSup">&#174;</span>&#44; Allergan&#41;&#46; The patient was referred to the glaucoma and neurology services where he was subsequently diagnosed with severe apnea-hypopnea syndrome&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">One week later&#44; the VA remained at 0&#46;1 with persisting conjunctival hyperemia&#44; slight corneal edema&#44; slight superficial keratitis punctata&#44; with IOP at 40<span class="elsevierStyleHsp" style=""></span>mmHg &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; Pachymetry was 512<span class="elsevierStyleHsp" style=""></span>&#956;m in RE and 574<span class="elsevierStyleHsp" style=""></span>&#956;m in LE &#40;this increase was explained by the corneal edema&#41;&#46; A differential diagnosis was proposed with a carotid-cavernous fistula or an orbitary apex syndrome and a computerized axial tomography &#40;CAT&#41; was requested&#46; The treatment was changed to brinzolamide &#40;Azopt<span class="elsevierStyleSup">&#174;</span>&#44; Alcon Cusi&#41;&#44; tafluprost &#40;Saflutan<span class="elsevierStyleSup">&#174;</span>&#44; MSD&#41; and sodium diclofenac &#40;Diclofenaco-lepori<span class="elsevierStyleSup">&#174;</span>&#44; Angelini Farmac&#233;utica&#41;&#46; Two days later&#44; the IOP remained at 30<span class="elsevierStyleHsp" style=""></span>mmHg and therefore acetazolamide was added &#40;Edemox<span class="elsevierStyleSup">&#174;</span>&#44; Chiesi-Spain&#41; and oral prednisone 60<span class="elsevierStyleHsp" style=""></span>mg &#40;Prednisona Alonga<span class="elsevierStyleSup">&#174;</span>&#44; Sanofi Aventis&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">The hyperemia diminished progressively &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#44; and the corticoids dosage was gradually reduced although the IOP remained above 26<span class="elsevierStyleHsp" style=""></span>mmHg&#46; The CAT discarded any orbitary and&#47;or cranial disease&#46; Five weeks later a peripheral ulcer emerged with pseudodendritic appearance which gave positive fluorescein staining with infiltration of edges&#46; A corneal sample was taken for culture and detection of proteins C reactive to virus herpes &#40;<a class="elsevierStyleCrossRefs" href="#fig0015">Figs&#46; 3 and 4</a>&#41;&#44; initiating treatment with topical aciclovir &#40;Zovirax<span class="elsevierStyleSup">&#174;</span>&#44; Glaxo Smith Kline&#41; and oral aciclovir &#40;Virex<span class="elsevierStyleSup">&#174;</span>&#44; Biogen&#41;&#46; The microbiological analysis was positive for HSV&#46; With said treatment&#44; the corneal lesion closed leaving a small peripheral leukoma&#44; the IOP returned to normal values and the visual acuity reached 0&#46;8&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><elsevierMultimedia ident="fig0020"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Discussion</span><p id="par0035" class="elsevierStylePara elsevierViewall">The definitive diagnosis of our case was endotheliitis and trabeculitis with secondary ocular hypertension and epithelial involvement in the form of late corneal ulcer&#46; The absence of clear signs caused a delay in the diagnosis which became obvious when the corneal alteration emerged&#44; with microbiological confirmation and the favorable response to the treatment&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Ohashi et al&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> proposed the hypotheses that&#44; in each reactivation&#44; the HSV latent in the trigeminal ganglion could traverse said nerve and reach the trabecular mesh from where copies of the virus would be secreted to the other ocular structures&#46; These hypotheses are supported by the onset of the corneal edema in the periphery&#44; the presence of HSV DNA in the aqueous humor of patients with endotheliitis and immunoreactivity for HSV described in the <span class="elsevierStyleItalic">trabeculum</span> of a patient intervened with trabeculectomy due to a secondary glaucoma of this type&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> It has also been postulated that the cicatrization of the trabecular mesh would be associated to a certain degree of necrosis produced directly by the virus similar to the iris atrophy or the necrotizing stromal keratitis&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">In our case&#44; the absence of keratic precipitates&#44; ocular pain and typical epithelial or endothelial injuries made the diagnosis difficult&#44; although the multiple pathologies should have suggested an evident possibility&#44; i&#46;e&#46;&#44; a recurring and opportunistic infection in an immunodepressed patient&#46; Erroneously we considered that the corneal edema was explained by the increased IOP although the sequence was the contrary&#58; an endotheliitis that caused corneal edema accompanied by trabeculitis which increased ocular pressure&#46; With the diagnostic suspicion of herpetic endotheliitis and trabeculitis&#44; treatment with antivirals and corticoids must be established without delay because the response is generally quick and prevents possible cicatricial lesions&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle">Conflict of interest</span><p id="par0050" class="elsevierStylePara elsevierViewall">The authors declare no conflict of interest&#46;</p></span></span>"
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        "resumen" => "<span class="elsevierStyleSectionTitle">Case report</span><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">A 62-year-old man with previous renal transplant and immunosuppressive treatment presented with decreased visual acuity &#40;20&#47;100&#41; in his left eye&#44; corneal edema and intraocular pressure of 46<span class="elsevierStyleHsp" style=""></span>mmHg&#46; One month later an inferior marginal dendritic keratitis appeared&#46; Corneal scraping and real-time polymerase chain reaction showed herpes simplex virus &#40;HSV&#41;&#46;</p> <span class="elsevierStyleSectionTitle">Discussion</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">The autoimmune corneal endotheliopathy or acute idiopathic corneal endotheliitis is characterized by corneal stromal edema and keratic precipitates&#46; HSV might be secreted from the trabeculum&#44; innervated by the trigeminal nerve&#46; This hypothesis is supported by the clinical observation that the corneal stromal edema usually starts from the periphery&#46;</p>"
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        "resumen" => "<span class="elsevierStyleSectionTitle">Caso cl&#237;nico</span><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Var&#243;n de 62 a&#241;os de edad con trasplante renal y tratamiento inmunosupresor que acude por disminuci&#243;n de la visi&#243;n &#40;20&#47;100&#41; en ojo izquierdo&#44; edema corneal y presi&#243;n intraocular de 46<span class="elsevierStyleHsp" style=""></span>mmHg&#46; Un mes despu&#233;s aparece queratitis dendr&#237;tica marginal inferior&#46; El raspado corneal y la prote&#237;na C reactiva demuestran la presencia de un virus del herpes simple &#40;HVS&#41;&#46;</p> <span class="elsevierStyleSectionTitle">Discusi&#243;n</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">La epiteliopat&#237;a corneal autoinmunitaria o endotelitis idiop&#225;tica corneal se caracteriza por edema corneal y precipitados quer&#225;ticos&#46; Se cree que el HVS podr&#237;a ser secretado desde el <span class="elsevierStyleItalic">trabeculum</span>&#44; inervado por el nervio trig&#233;mino&#44; apoyado cl&#237;nicamente por la progresi&#243;n del edema estromal desde la periferia&#46;</p>"
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          "en" => "<p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">One week later the reduction of the hyperemia is apparent&#46;</p>"
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          "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">At week 5 the conjunctival hyperemia persisted and a corneal dendritic injury appeared&#46;</p>"
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