Persistent and Severe Mpox Keratitis Despite Systemic and Topical Treatment.

PURPOSE: The purpose of this study was to report a case of peripheral ulcerative keratitis in a patient diagnosed with corneal polymerase chain reaction (PCR) and a positive mpox culture. METHODS: This is a case report. RESULTS: An immunocompetent 54-year-old man was diagnosed with conjunctivitis in his left eye 15 days after being diagnosed with mucocutaneous monkeypox. He received treatment with dexamethasone 0.1% and tobramycin 0.3% eye drops for 2 weeks. Two weeks after discontinuing this treatment, he developed peripheral ulcerative keratitis and a paracentral epithelial defect. Mpox keratitis was diagnosed by corneal culture and PCR. Corneal inflammation persisted for more than 6 months, manifested as corneal epithelial defect, limbitis, endotheliitis, neurotrophic changes, and trabeculitis. This persistence was observed alongside positive corneal PCR results, despite undergoing 2 courses of trifluorothymidine, 2 courses of oral tecovirimat, and intravenous cidofovir. An amniotic membrane transplantation was then performed. CONCLUSIONS: Persistent corneal pain and replication are possible with the mpox virus, even in immunocompetent patients. Having received treatment with topical corticosteroids before antiviral treatment for the pox virus may have contributed to the severity and persistence of the clinical condition. Cycle threshold PCR values can be used to support the diagnosis and monitor treatment effectiveness.

Long-Term Effects of Adipose-Derived Stem Cells for the Treatment of Bilateral Limbal Stem Cell Deficiency.

PURPOSE: To determine the safety and feasibility of human autologous adipose tissue-derived adult mesenchymal stem cells (ASCs) for ocular surface regeneration in patients with bilateral limbal stem-cell deficiency (LSCD). METHODS: A phase IIa clinical trial was designed (https://Clinicaltrials.gov, NCT01808378) with 8 patients, 3 of whom had aniridia, 2 meibomian glands diseases, 2 multiple surgeries and 1 chronic chemical injury. The therapeutic protocol was as follows: 6-mm of central corneal epithelium was removed, 400,000 ASCs were injected into each limboconjunctival quadrant, 400,000 ASCs were suspended over the cornea for 20 min, and finally the cornea was covered with an amniotic membrane patch. RESULTS: No adverse events were detected after a mean of 86,5 months of follow-up. One year after surgery, 6 of the 8 transplants were scored as successful, five patients had improved uncorrected visual acuity (mean of 12 letters), two patients presented epithelial defects (also present at baseline) and the mean percentage of corneal neovascularization was of 28.75% (36.98%, at baseline). Re-examination 24 months after treatment disclosed preserved efficacy in 4 patients. At the last visit (after a mean of 86,5 months of follow up) epithelial defects were absent in all patients although improvement in all of the variables was only maintained in patient 3 (meibomian glands agenesia). CONCLUSION: ASCs are a feasible and conservative therapy for treating bilateral LSCD. The therapeutic effect differs between etiologies and diminishes over time.

A Pilot Study of a Panel of Ocular Inflammation Biomarkers in Patients with Primary Sjögren's Syndrome.

Ocular diseases have a strong impact on individuals, the effects of which extend from milder visual impairment to blindness. Due to this and to their prevalence, these conditions constitute important health, social and economic challenges. Thus, improvements in their early detection and diagnosis will help dampen the impact of these conditions, both on patients and on healthcare systems alike. In this sense, identifying tear biomarkers could establish better non-invasive approaches to diagnose these diseases and to monitor responses to therapy. With this in mind, we developed a solid phase capture assay, based on antibody microarrays, to quantify S100A6, MMP-9 and CST4 in human tear samples, and we used these arrays to study tear samples from healthy controls and patients with Sjögren's Syndrome, at times concomitant with rheumatoid arthritis. Our results point out that the detection of S100A6 in tear samples seems to be positively correlated to rheumatoid arthritis, consistent with the systemic nature of this autoinflammatory pathology. Thus, we provide evidence that antibody microarrays may potentially help diagnose certain pathologies, possibly paving the way for significant improvements in the future care of these patients.

Confirmation of PRDX3 c.568G>C as the Genetic Basis of Punctiform and Polychromatic Pre-Descemet Corneal Dystrophy.

PURPOSE: The aim of this study was to report the results of screening peroxiredoxin 3 (PRDX3) and PDZ domain-containing protein 8 (PDZD8) in a previously unreported pedigree with punctiform and polychromatic pre-Descemet corneal dystrophy (PPPCD) to confirm that the PRDX3 mutation c.568G>C is the genetic basis of PPPCD. METHODS: Ophthalmologic examination of the proband and her affected father was performed with slit lamp biomicroscopy. Saliva was collected from the proband as a source of DNA, after which screening for PRDX3 and PDZD8 was performed. RESULTS: Slit lamp examination of the proband revealed polychromatic deposits diffusely distributed at the pre-Descemet level in both corneas and anterior subcapsular in the crystalline lens of both eyes. The proband's father also demonstrated diffuse pre-Descemetic polychromatic deposits in both eyes but no lenticular deposits. Screening of PRDX3 in the proband demonstrated the c.568G>C (p.Asp190His) variant previously associated with PPPCD and failed to identify any variants in PDZD8. CONCLUSIONS: We report the initial confirmation of PRDX3 as the genetic basis of PPPCD in a previously unreported pedigree and expand the phenotype of PPPCD to include polychromatic lenticular deposits.

Posterior Polymorphous Corneal Dystrophy in a Patient with a Novel ZEB1 Gene Mutation.

Posterior polymorphous corneal dystrophy (PPCD), a rare, bilateral, autosomal-dominant, inherited corneal dystrophy, affects the Descemet membrane and corneal endothelium. We describe an unusual presentation of PPCD associated with a previously unknown genetic alteration in the ZEB1 gene. The proband is a 64-year-old woman diagnosed with keratoconus referred for a corneal endothelium study who presented endothelial lesions in both eyes suggestive of PPCD, corectopia and iridocorneal endothelial synechiae in the right eye and intrastromal segments in the left eye. The endothelial count was 825 in the right eye and 1361 in the left eye, with typical PPCD lesions visible under specular and confocal microscopy. In the next generation sequencing genetic analysis, a heterozygous c.1A > C (p.Met1Leu) mutation was found in the ZEB1 gene (TCF8). The PPCD3 subtype is associated with corneal ectasia, and both can appear due to a pathogenic mutation in the ZEB1 gene (OMIM #189909). However, our patient had a previously unreported mutation in the ZEB1 gene, which mediates the transition between cell lines and provides a pathogenic explanation for the epithelialisation of the corneal endothelium, a characteristic of PPCD.

Cancer-Associated Retinopathy and Treatment with Intravenous Immunoglobulin Therapy. A Seldom Used Approach?

Purpose: To report a case of cancer-associated retinophaty (CAR) treated with intravenous immunoglobulin (IGIV) and review the use of IGIV in the treatment of CARMethods: Case report: A 68-year-old woman, former smoker, presented with bilateral subacute decreased visual acuity with 1 month of evolution, without other symptoms. Clinical examination revealed retinal atrophy and a mild vitritis component. Treatment with corticosteroid and IGIV was initiated empirically with the stabilization of visual loss. Anti-recoverin antibodies tested positive and a small cell lung carcinoma was diagnosed. In a review of the literature, we found that only 12 cases of patients treated with intravenous immunoglobulins have been reported.Conclusions: the early use of IVIG could contribute to an improvement and/or stabilization of visual symptoms in this patient group due to its rapid effect and lower profile of adverse effects when administered with chemotherapy.

Punctiform and Polychromatic Pre-Descemet Corneal Dystrophy: Clinical Evaluation and Identification of the Genetic Basis.

PURPOSE: This study reports the clinical features and genetic bases of 3 previously unreported families with punctiform and polychromatic pre-Descemet corneal dystrophy (PPPCD). DESIGN: Observational case series. METHODS: Full ophthalmic assessment was performed for members of 3 unreported families with PPPCD. Structural and biomechanical alterations of the cornea were screened. Whole exome sequencing (WES) was performed in the first family. Novel or rare variants that segregated with the affected status were screened in the other 2 families using Sanger sequencing. Identified variants that segregated with the affected status in all families were characterized by using in silico prediction tools and/or in vitro splice assays. Additionally, 2 previously reported PPPCD families were screened for variants identified in the 3 unreported PPPCD families. RESULTS: PPPCD was diagnosed in 12 of the 21 examined members of the 3 unreported families. The only refractive, topographic, or biomechanical abnormality associated with PPPCD was a significantly increased corneal stiffness. WES and Sanger sequencing identified 2 variants that segregated with the affected status in all 3 families: a rare intronic PDZD8 c.872+10A>T variant and a novel missense PRDX3 c.568G>C (p.Asp190His) variant. The same PRDX3 variant was identified in the previously reported PPPCD family expressing the common PPPCD phenotype and was predicted by in silico prediction tools to be damaging to protein function. CONCLUSIONS: PPPCD is associated with an alteration of corneal biomechanics and a novel missense variant in PRDX3. Screening of additional families will determine whether all families demonstrate a PRDX3 variant or whether locus heterogeneity may exist for PPPCD.

Correction to: Heredity and in vivo confocal microscopy of punctiform and polychromatic pre-Descemet dystrophy.

In the original publication, presentation of the following author names are incorrect in the HTML version.

Ocular surface changes in recurrent pterygium cases post-operatively treated with 5-fluorouracil subconjunctival injections.

INTRODUCTION:: To investigate the ocular surface changes occurring in eyes with recurrent pterygium post-operatively treated with 5-fluorouracil intralesional injections. METHODS:: Retrospective observational study of recurrent pterygium cases treated with weekly intralesional injections of 0.1 mL (5 mg) of 5-fluorouracil (10 injections). Impression cytology samples taken from the lesion, the healthy conjunctivae (inferior, superior, and contralateral to injury), and the cornea before and after treatment were analyzed. Clinical ocular characteristics (including Schirmer's test and break-up time) were evaluated during treatment. RESULTS:: A total of 15 eyes were treated, with the mean follow-up of 27 ± 8.7 months (mean ± standard deviation). Prior to treatment initiation, the ocular surface citology over the pterygium was found to be abnormal. No epithelial cells (27%) and a lower goblet cell density (73%) compared to the healthy conjunctivae (p < 0.01) were found. Squamous metaplasia was observed to some degree in the cornea (100%), pterygium (81%) and healthy conjunctivae (73%). Following treatment, pterygium composition had changed: epithelial cell number (100%) and goblet cell density (47%) had increased (p < 0.05). Goblet cell density was also increased in healthy conjunctivae (67%; p < 0.05). The degree of squamous metaplasia decreased in the cornea (67%), pterygium (45%), and healthy conjunctivae (60%; p < 0.05). No adverse effects were reported, recurrence progression was arrested, and conjunctival redness and dry-eye severity level were decreased in all cases (p < 0.01). DISCUSSION:: The cytology of ocular surface in recurrent pterygium is abnormal. After weekly intralesional 5-fluorouracil injections, it tends to normalize. The 5-fluorouracil compound is a safe and effective treatment to prevent pterygium recurrence.

Clinical and microbiological profile of infectious keratitis in an area of Madrid, Spain / Perfil clínico y microbiológico de la queratitis infecciosa en un área de Madrid, España

INTRODUCTION: To study antibiotic susceptibility in bacterial keratitis (BK), its profile over 10 years and its influence on ophthalmological practice. METHODS: Retrospective review of BK with positive corneal scraping over a 10-year period. Risk factors for keratitis, visual acuity (VA), empirical topical treatment, corneal infection characteristics and outcomes were analyzed for BK due to Staphylococcus aureus, Staphylococcus epidermidis, Streptococcus pneumoniae, Pseudomonas aeruginosa and Propionibacterium acnes. RESULTS: 389 positive corneal scrapings were collected. All Gram-positive bacteria were susceptible to vancomycin. P. aeruginosa demonstrated >90% sensitivity to the most-commonly-used topical antibiotics. Susceptibility to methicillin was 90.2% for S. aureus and 66.3% for S. epidermidis. The results of 215 patients were available. 1.9% required enucleation and 2.8% required surgical treatments. Final VA improved after treatment in keratitis due to S. aureus (p = 0.026) and S. epidermidis (p = 0.005). There was a correlation between S. aureus resistance to methicillin (p = 0.002) and levofloxacin (p = 0.043) and enucleation (20% and 10%, respectively) compared with a 0% rate of enucleation in S. aureus-susceptible keratitis. CONCLUSIONS: BK due to S. pneumoniae is very aggressive irrespective of antibiotic sensitivity. S. aureus was frequently isolated in patients with systemic diseases. It causes severe keratitis and remains moderately resistant to methicillin and levofloxacin. For this reason, keeping vancomycin in empirical regimens is believed to be necessary

INTRODUCCIÓN: Estudiar la susceptibilidad antibiótica en queratitis bacteriana (QB), el perfil temporal a lo largo de 10 años y su influencia en la clínica ocular. MÉTODOS: Revisión retrospectiva durante un periodo de 10 años de QB con raspado corneal positivo. Se analizaron los factores de riesgo de queratitis, la agudeza visual (AV), el tratamiento empírico tópico, las características de la infección corneal y el resultado clínico para QB por Staphylococcus aureus, Staphylococcus epidermidis, Streptococcus pneumoniae, Pseudomonas aeruginosa y Propionibacterium acnes. RESULTADOS: Se recogieron 389 raspados corneales positivos. Todas las bacterias grampositivas fueron susceptibles a la vancomicina. P. aeruginosa presentó sensibilidad mayor del 90% a los antibióticos tópicos más comúnmente utilizados. La susceptibilidad a la meticilina fue del 90,2% para S. aureus y del 66,3% para S. epidermidis. Los resultados clínicos estaban disponibles para 215 pacientes. El 1,9% requirieron enucleación y el 2,8% tratamientos quirúrgicos. La AV final mejoró después del tratamiento en queratitis por S. aureus (p = 0,026) y por S. epidermidis (p = 0,005). Hubo correlación entre la resistencia de S. aureus a la meticilina (p = 0,002) y levofloxacino (p = 0,043) y enucleación (20 y 10%, respectivamente) en comparación con una tasa de enucleación del 0% en S. aureus susceptible. CONCLUSIONES: Las QB por S. pneumoniae son muy agresivas independientemente de la sensibilidad antibiótica. S. aureus se aisló con frecuencia en pacientes con enfermedades sistémicas, causa queratitis severa y permanece moderadamente resistente a la meticilina y a levofloxacino; debido a esto, consideramos necesario mantener la vancomicina en la pauta empírica

Heredity and in vivo confocal microscopy of punctiform and polychromatic pre-Descemet dystrophy.

PURPOSE: To describe and analyze the biomicroscopic features and in vivo confocal microscopy of the crystalline form of pre-Descemet corneal dystrophy (PDCD). METHODS: We examined two non-related families using biomicroscopy, in vivo confocal microscopy, and a genetic study using a gene panel test, looking for mutations in the PIKFYVE gene. RESULTS: A slit-lamp examination of the first family revealed polychromatic crystalline punctiform opacities distributed all over the stroma in 8 of 11 family members in three generations with an autosomal dominant inheritance. The second family showed in three of four members in two generations the same opacities located in the pre-Descemet region. It was also a hint for autosomal dominant inheritance. The in vivo confocal microscopy identified numerous rounded and hyperreflective stromal particles measuring 10-15 µm in diameter, with the highest density in the posterior stroma and with normal keratocytes. No systemic disease was diagnosed. No variants or mutations were identified in PIKFYVE gene. CONCLUSIONS: Polychromatic deposits in patients with Punctiform and Polychromatic Pre-Descemet corneal dystrophy can be located not only in the deep stroma but also in the anterior and middle stroma. Our presentation reveals the possibility of considering this characteristic corneal disorder as a corneal dystrophy of its own and not as a subtype of pre-Descemet corneal dystrophy.

The analysis of human conjunctival epithelium proteome in ocular surface diseases using impression cytology and 2D-DIGE.

Conjunctival impression cytology samples from patients with meibomian gland dysfunction (MGD), dry eye (DE), and healthy subjects (CT) were collected for determination of the degree of squamous metaplasia (SM) by PAS-hematoxylin staining and for comparative proteomic analyses by 2D-DIGE. The protein spots with discriminant expression were identified by MALDI-TOF/TOF mass spectrometry. Three independent statistical studies were conducted: i). Analysis of differential protein expression between study groups: We observed increased expression of proteins S100A4, S100A8, retinal dehydrogenase-1, peroxiredoxin-1, annexin-A1, annexin-A2, α-enolase, and glutathione S-transferase-P in DE, whereas the highest expression of peroxiredoxin-6, actin cytoplasmic-1, peroxiredoxin-2, and heat shock protein HSP-90-α was observed in MGD; ii). Correlation between changes in the proteome profile and the grade of SM: The expression of 5 different cytokeratins (KRT1, KRT4, KRT8, KRT10, and KRT13) correlated with the degree of SM; iii). Proteome profile differences between pathological and CT groups: An overall proteome analysis revealed upregulation of 9 proteins in the pathological groups (Annexin-A1, α-enolase, Annexin-A2, S100A8, cytokeratin-1, Peroxiredoxin-2 and Leukocyte elastase inhibitor) and downregulation of 2 proteins (Galectin-3 and Lipocalin-1). In conclusion, a sensitive proteomic approach to study conjunctival tissue collected from minimally invasive impression cytology was implemented. Differential proteomics analyses showed that in comparison with the MGD, the DE patients presented higher overexpression of proteins related to antimicrobial defense, tissue-damage response, and regulation of body fluid secretions. Changes in MGD proteome were associated with oxidative stress and anti-apoptotic processes. We found a correlation between the grade of SM and expression of proteins associated with cytoskeleton and keratinization. The studied pathological groups shared elements related to the defense and inflammatory responses. Dot blot assays of proteins ANXA1, S100A8, and S100A4 validated the proteomic results obtained from 2D-DIGE experiments and confirmed the correlation between the expression of these proteins and the clinical parameters.

Clinical and microbiological profile of infectious keratitis in an area of Madrid, Spain.

INTRODUCTION: To study antibiotic susceptibility in bacterial keratitis (BK), its profile over 10 years and its influence on ophthalmological practice. METHODS: Retrospective review of BK with positive corneal scraping over a 10-year period. Risk factors for keratitis, visual acuity (VA), empirical topical treatment, corneal infection characteristics and outcomes were analyzed for BK due to Staphylococcus aureus, Staphylococcus epidermidis, Streptococcus pneumoniae, Pseudomonas aeruginosa and Propionibacterium acnes. RESULTS: 389 positive corneal scrapings were collected. All Gram-positive bacteria were susceptible to vancomycin. P. aeruginosa demonstrated >90% sensitivity to the most-commonly-used topical antibiotics. Susceptibility to methicillin was 90.2% for S. aureus and 66.3% for S. epidermidis. The results of 215 patients were available. 1.9% required enucleation and 2.8% required surgical treatments. Final VA improved after treatment in keratitis due to S. aureus (p=0.026) and S. epidermidis (p=0.005). There was a correlation between S. aureus resistance to methicillin (p=0.002) and levofloxacin (p=0.043) and enucleation (20% and 10%, respectively) compared with a 0% rate of enucleation in S. aureus-susceptible keratitis. CONCLUSIONS: BK due to S. pneumoniae is very aggressive irrespective of antibiotic sensitivity. S. aureus was frequently isolated in patients with systemic diseases. It causes severe keratitis and remains moderately resistant to methicillin and levofloxacin. For this reason, keeping vancomycin in empirical regimens is believed to be necessary.

Intraocular minocycline for the treatment of ocular pythiosis.

PURPOSE: A case of ocular pythiosis successfully treated with surgery and intraocular and oral minocycline is reported. SUMMARY: A 30-year-old man who wore corrective contact lenses traveled to Brazil and Colombia where he swam in salt and fresh waters while wearing contact lenses. He sought treatment at an emergency department after 2 weeks of suffering with a painful corneal ulcer, redness, and loss of vision in his right eye that had been treated at other centers with ophthalmic moxifloxacin for 10 days and with fortified topical antibiotics (amikacin and vancomycin) for 2 days. Examination using a slit lamp revealed a deep central corneal ulcer with surrounding white infiltrate, endothelial plaque, and hypopyon. Due to infection severity, the patient was admitted and received empirical antibiotic therapy and i.v. and topical antifungals. During the first corneal transplantation, the patient's original infection relapsed and was treated with voriconazole and liposomal amphotericin B intraocular injections. A subsequent infection developed, and a second keratoplasty was performed. One month after hospital admission, the patient was diagnosed with ocular pythiosis and therapy with oral minocycline was initiated. After severe infection relapse in the anterior chamber, the patient underwent a third penetrating keratoplasty, where minocycline intraocular injection was administered. After this intervention, complete infection control was achieved, and the patient was discharged 45 days after admission with oral minocycline and 1% cyclosporine and 0.3% ofloxacin eye drops. CONCLUSION: A patient with ocular pythiosis was successfully treated with penetrating keratoplasty and 2 months of treatment with intracameral and oral minocycline.

Orbital and conjunctival metastasis from lobular breast carcinoma.

This article examines the clinical presentation of ocular metastasis from an infiltrating lobular breast carcinoma. We examined a conjunctival biopsy from a 69-year-old woman who developed unilateral conjunctival inflammation together with a neurotrophic corneal ulcer and proptosis. Infiltrating lobular breast carcinoma (ILBC) was diagnosed using routine histology and immunohistochemistry. She had a past history of a hormone receptor-positive infiltrating ILBC 11 years ago with cutaneous and diffuse osteoblastic metastases, and she was kept under treatment with lezotrol. Treatment was initiated with systemic corticosteroids but an annular conjunctival perilimbal infiltration was found to have spread, which did not respond either to local radiotherapy (total dose 60 Gy, 2 Gy per day). A new extensive corneal epithelial defect recurred, and because it had not responded to matrix therapy agent (RGTA, Cacicol®) eye drops, autologous serum eye drops and a therapeutic contact lens, a permanent total tarsorrhaphy was performed. Progression of the diffuse bone metastases was detected and the treatment with lezotrol was replaced by fulvestrant.Infiltrating lobular breast carcinoma is a rare cause of conjunctival metastasis. This aggressive malignancy did not respond to external beam radiotherapy.

Herpes Simplex Keratitis in Rheumatoid Arthritis Patients.

PURPOSE: To describe a series of 5 patients with herpes simplex virus keratitis (HSK) and rheumatoid arthritis (RA) under immunosuppressive treatment. METHODS: Retrospective study. Detailed data were obtained regarding symptoms and signs at the initial evaluation, treatment, microbiological diagnostic tests, evolution, and outcomes. RESULTS: Five patients with HSK and RA were identified. Bilateral involvement occurred in 2 patients (40%). Epithelial keratitis was diagnosed in 5 eyes. Three eyes showed severe melting with eye perforation. Gram-positive bacterial co-infections were common in the group with stromal keratitis. We did not find differences in the evolution of the disease based on anti-rheumatoid treatment. CONCLUSIONS: The characteristics of HSK in patients with RA differed from HSK in immunocompetent patients. The stromal keratitis cases were very aggressive and difficult to manage, with perforation and gram-positive bacterial co-infection as frequently associated conditions. Prophylactic therapy at standard doses was unsuccessful to avoid recurrences.

The effects of methylphenidate on refraction and anterior segment parameters in children with attention deficit hyperactivity disorder.

BACKGROUND: Methylphenidate hydrochloride is used as first-line treatment for attention deficit hyperactivity disorder (ADHD). However, there is concern that this treatment may be associated with increased risk of angle closure glaucoma and disturbance of ocular refraction. The aim of this study was to investigate the effects of methylphenidate treatment on refraction, intraocular pressure, and the anterior chamber in children with ADHD. METHODS: In this prospective pilot study, children diagnosed with ADHD were examined before the start of methylphenidate treatment and again 3 and 9 months after the start of treatment. Ocular examination before and after cycloplegia was performed at each visit, including Pentacam imaging of the anterior chamber. RESULTS: A total of 14 patients (mean age, 11 years) were recruited. The mean visual acuity, sphere, spherical equivalent refraction, intraocular pressure, and cup:disk ratio did not change significantly after the start of treatment. The anterior chamber depth after cycloplegia decreased significantly between baseline and 9 months, from 3.26 ± 0.22 mm to 3.24 ± 0.23 mm in the right eye (P = 0.037) and from 3.28 ± 0.22 mm to 3.25 ± 0.24 mm in the left eye (P = 0.001). CONCLUSIONS: Methylphenidate does not seem to affect refraction in most children with ADHD. After 9 months of treatment, however, there was a reduction in the anterior chamber depth, which has been described as a powerful predictor of angle closure glaucoma. Further investigation of the potential ocular side effects of methylphenidate treatment is warranted.

Recurrent ocular surface inflammation associated with human herpesvirus 6 infection.

OBJECTIVES: To report a case of atypical herpes keratitis and bilateral conjunctivitis associated with human herpesvirus 6 (HHV-6). METHODS: An immunocompetent 34-year-old man was referred for herpetic epithelial keratitis in his left eye, which was non-responsive to topical acyclovir. Biomicroscopy revealed a central dendritic ulcer with a white stromal infiltrate beneath the ulcer. RESULTS: The corneal scraping multiplex polymerase chain reaction (CLART ENTHERPEX, Genomica, Spain) was positive for HHV-6 and negative for herpes simplex virus, varicella zoster virus, cytomegalovirus, and Epstein-Barr virus. An improvement of the keratitis and visual acuity was achieved with topical fluorometholone and systemic valacyclovir. One year later, the patient complained of redness of the eyes. A slit-lamp examination disclosed bilateral follicular conjunctivitis, and HHV-6 DNA was once again detected in a conjunctival scraping of both eyes. CONCLUSIONS: Human herpesvirus 6 may be another causative agent for corneal ulcers and conjunctivitis in isolation. Stromal necrosis is a rare manifestation of herpetic dendritic keratitis. In these cases, we should consider the presence of HHV-6 in the differential diagnosis, even in immunocompetent patients.