Conjuntivitis recurrente de causa no aclarada. ¿Hipertiroidismo? / Recurrent conjunctivitis of unknown origin. Hyperthyroidism?

El hipertiroidismo en la infancia es muy infrecuente; en la mayoría de los casos es de origen autoinmune (enfermedad de Graves). La tríada clásica se compone de bocio, oftalmopatía y dermatopatía. Su presentación clínica en la edad pediátrica es muy variable y de comienzo habitualmente insidioso, lo que en muchas ocasiones conlleva una demora en el diagnóstico y tratamiento. Presentamos el caso de una paciente de 12 años que consulta por una conjuntivitis crónica bilateral refractaria a tratamientos tópicos habituales, que finalmente fue derivada al Servicio de Endocrinología Pediátrica ante la sospecha de oftalmopatía de Graves (AU)

Hyperthyroidism in childhood is a very rare condition, and in most cases of autoimmune etiology (Graves’ disease). The classic triad consists of goiter, ophthalmopathy and dermopathy. Clinical presentation in children is highly variable and often with an insidious onset, which on many occasions may cause a delay on its diagnosis and treatment. A case is reported here where a 12-year-old patient, who showed a refractory bilateral chronic conjunctivitis refractary to topical usual treatments, was finally referred to the Pediatric Endocrinology Department due to the likelihood of her suffering from Graves' ophthalmopathy (AU)

Seasonal Local Allergic Rhinitis in Areas With High Concentrations of Grass Pollen.

BACKGROUND: Local allergic rhinitis (LAR) is a phenotype of allergic rhinitis characterized by the presence of a localized immune response in the nasal mucosa of patients with negative skin prick test (SPT) results and undetectable serum specific IgE (sIgE). It unknown whether LAR is limited to areas with low or moderate aeroallergen exposure. OBJECTIVE: To explore the presence of LAR and the clinical and immunological characteristics of this entity in geographic areas with high grass pollen loads. METHODS: A cross-sectional observational study was carried out in 2 hospitals in central Spain (Madrid and Ciudad Real). Sixty-one patients with seasonal rhinitis and negative SPT results and undetectable serum sIgE were evaluated using a clinical questionnaire, determination of serum total IgE, and a nasal allergen provocation test (NAPT) with Phleum species. The response to NAPT was monitored using assessment of nasal symptoms, acoustic rhinometry, and determination of sIgE, tryptase, and eosinophil cationic protein in the nasal cavity. RESULTS: Seasonal LAR was detected in 37 patients (61%) using the techniques described above. Eleven percent of patients with LAR were adolescents or children, and 14% reported onset of rhinitis in childhood. Most patients reported persistent-moderate seasonal nasal symptoms, and 41% reported worsening of the disease during the last 2 years. Conjunctivitis was the most common comorbidity, affecting 95% of cases. CONCLUSIONS: LAR to grass pollen is relevant in patients with seasonal symptoms indicative of allergic rhinitis but with a negative skin test result who live in areas with high allergenic pollen loads. This entity should be included the differential diagnosis of rhinitis.

Conjuntivitis aguda causada por Metapneumovirus humano / Acute conjunctivitis caused by Human Metapneumovirus

Se presenta el caso de un varón prematuro y con cardiopatía congénita de 4 meses de edad que presentaba unas bronquiolitis y conjuntivitis causadas por CI Metapneumovirus humano. El virus se detectó tanto en el aspirado nasofaríngeo como en la secreción conjuntival. Debido a la rareza de esta entidad, se revisa la literatura correspondiente a esta entidad (AU)

A premature male with congenital heart disease and 4 months of age had a bronchiolitis and conjunctivitis caused by human metapneumovirus. The virus was detected in both the nasopharyrngeal aspirate as coniunctival secretion. Because of the rarity of this entity we reviewed scientific literature (AU)

Linfoma folicular primario de la conjuntiva simulando conjuntivitis crónica / Primary conjunctival follicular lymphoma mimicking chronic conjunctivitis

CASO CLÍNICO: Paciente varón de 43 años con conjuntivitis folicular crónica resistente a tratamiento local, y serologías para bacterias negativas. Se realizó biopsia incisional que fue compatible con hiperplasia reactiva linfoide. Un año después, una nueva biopsia mostró un linfoma folicular, sin afectación sistémica, que fue tratado con radioterapia local. DISCUSIÓN: Ante una conjuntivitis folicular crónica resistente a tratamiento convencional es esencial realizar una biopsia incisional para el diagnóstico histopatológico, que puede abarcar desde la inflamación crónica y la hiperplasia reactiva linfoide al linfoma. El linfoma folicular es raro entre los linfomas de conjuntiva y la estadificación es indispensable para un correcto abordaje terapéutico

CLINICAL CASE: The case is presented of a 43 year-old male patient with chronic follicular conjunctivitis, negative bacterial serology, and refractory to local treatment. The incisional biopsy performed showed to be consistent with reactive lymphoid hyperplasia. A year later, a new incisional biopsy showed follicular lymphoma, with no systemic involvement, and he was treated with local radiotherapy. DISCUSSION: When a chronic follicular conjunctivitis is refractory to treatment, it is essential to perform an incisional biopsy to establish the histopathological diagnosis that can range from chronic inflammation, reactive lymphoid hyperplasia to lymphoma. Follicular lymphoma is rare among conjunctival lymphomas, and the staging is indispensable for the correct therapeutic approach

Long-Term Loss of Response in Proton Pump Inhibitor-Responsive Esophageal Eosinophilia Is Uncommon and Influenced by CYP2C19 Genotype and Rhinoconjunctivitis.

OBJECTIVES: Proton pump inhibitor-responsive esophageal eosinophilia (PPI-REE) is diagnosed in at least one-third of patients with suspected eosinophilic esophagitis (EoE). We aimed to evaluate the durability and factors influencing long-term efficacy of PPI therapy. METHODS: Retrospective multicenter cohort study of patients with PPI-REE who had at least 12 months of follow-up. PPI therapy was tapered to the lowest dose, which maintained clinical remission. Primary outcomes were the proportion of patients with loss of histological response (<15 eos/HPF) and predictors of loss of response. CYP2C19 polymorphisms were determined from blood samples in a subset of patients. RESULTS: Seventy-five PPI-REE patients were included (mean follow-up 26 months (12-85)), of whom fifty-five (73%) had sustained histological remission on low-dose PPI therapy. Loss of response was significantly higher in those patients with a CYP2C19 rapid metabolizer genotype (36% vs. 6%, P = 0.01) and with rhinoconjunctivitis (40% vs. 13%, P = 0.007). On the multivariate analysis, a CYP2C19 rapid metabolizer genotype (odds ratio (OR) 12.5; 95% confidence interval (CI): 1.3-115.9) and rhinoconjunctivitis (OR 8.6; 95% CI: 1.5-48.7) were independent predictors of loss of response. Among relapsing patients, eosinophilia was limited to the distal esophagus in 14/20 (70%). Nine of ten relapsers, with distal eosinophilia, all showing a CYP2C19 rapid metabolizer genotype, regained histological remission after PPI dose intensification. CONCLUSIONS: Most PPI-REE patients remain in long-term remission on low-dose PPI therapy. CYP2C19 rapid metabolizer genotypes and rhinoconjunctivitis were independent predictors of loss of response to PPI, but patients frequently responded to PPI dose escalation.

Enfermedad de Kawasaki / Malaltia de Kawasaki / Kawasaki´s disease

La malaltia de Kawasaki (MK) és una vasculitis sistèmica aguda d'etiologia desconeguda. El diagnòstic es basa en criteris clínics que inclouen febre, exantema, conjuntivitis, canvis en les extremitats, eritema de la mucosa oral i llavis, i adenopaties cervicals. No obstant això, aquests criteris tenen una sensibilitat i una especificitat baixes i, per tant, altres característiques clíniques i de laboratori poden ser útils per establir el diagnòstic, sobretot en els casos d'MK atípica o incompleta. El pronòstic depèn de l'extensió de l'afectació cardíaca; els aneurismes coronaris, que es de-sen volupen en el 20-25% dels pacients no tractats, poden provocar infart de miocardi o mort sobtada en l'edat adulta. El tractament amb altes dosis d'immunoglobulina intrave-nosa és eficaç per reduir el risc d'aneurismes coronaris en la majoria dels casos i és el tractament d'elecció. En aquesta revisió analitzem la clínica, l'epidemiologia i el tractament d'aquesta malaltia típica de l'edat pediàtrica

La enfermedad de Kawasaki (EK) es una vasculitis sistémica aguda de etiología desconocida. El diagnóstico se basa en criterios clínicos que incluyen fiebre, exantema, conjuntivitis, cambios en las extremidades, eritema de la mucosa oral y labios, y adenopatías cervicales. Sin embargo, estos criterios tienen una sensibilidad y una especificidad bajas y, por tanto, otras características clínicas y de laboratorio pueden ser útiles para establecer el diagnóstico, sobre todo en los casos de MK atípica o incompleta. El pronóstico depende de la extensión de la afectación cardiaca; los aneurismas coronarios, que se desarrollan en el 20-25% de los pacientes no tratados, pueden provocar infarto de miocardio o muerte súbita en la edad adulta. El tratamiento con altas dosis de inmunoglobulina intravenosa es eficaz para reducir el riesgo de aneurismas coronarios en la mayoría de los casos y es el tratamiento de elección. En esta revisión analizamos la clínica, la epidemiología y el tratamiento de esta enfermedad típica de la edad pediátrica (AU)

Kawasaki disease (MK) is an acute systemic vasculitis of unknown etiology. The diagnosis is based on clinical criteria that includes fever, rash, conjunctivitis, changes in the limbs, erythema of the oral mucosa and lips, and cervical lymphadenopathy. However, these criteria have a low sensitivity and specificity and, therefore, other clinical and laboratory features may be helpful in establishing the diagnosis, especially in cases of atypical or incomplete MK. The prognosis depends on the extent of heart involvement; coronary aneurysms, which develop in 20-25% of untreated patients can cause a heart attack or sudden death in adulthood. Treatment with high doses of intravenous immunoglobulin is effective to reduce the risk of coronary aneurysms in most cases and is the treatment of choice. In this review we analyze the symptoms, epidemiology and treatment of this disease, typical of paediatric patients (AU)

Pediatric allergic rhinitis.

Allergic rhinitis is a common pediatric problem with significant comorbidities and potential complications. This article is an overview of the epidemiology, pathophysiology, and current therapeutic strategies. Allergic rhinitis management in a specific child is age dependent and influenced by the severity and frequency of the symptoms and the presence of any concurrent conditions. Current strategies permit symptomatic control and improved quality of life for most patients.

300IR 5-Grass pollen sublingual tablet offers relief from nasal symptoms in patients with allergic rhinitis.

BACKGROUND: 300IR 5-grass pollen sublingual immunotherapy tablets have been approved for the treatment of allergic rhinitis (AR) with or without conjunctivitis in adults and children >5 years with grass pollen allergy. This study was designed to review data on nasal symptoms with 300IR 5-grass pollen sublingual tablets in adults and children. METHODS: We reviewed data from four double-blind, placebo-controlled, randomized clinical trials. Two groups of patients who received a daily dose of either placebo or 300IR 5-grass pollen sublingual tablets starting 4 months before the expected start of the pollen season and continuing through the season were compared (analysis of covariance) for scores of sneezing, rhinorrhea, nasal pruritus, nasal congestion, total nasal symptom score (TNSS), and adult Rhinoconjunctivitis Quality of Life Questionnaire (RQLQ) scores. RESULTS: Data for 266 children (one pediatric trial) and 1036 adults (three trials) were analyzed. Compared with the placebo groups, mean TNSS in the 300IR groups was lower by 22% in children and 19-36% in adults. Among the four nasal symptoms, the lowest scores relative to placebo were for nasal congestion in children (31%) and adults (43%). Mean adult RQLQ scores were 21-31% lower in the 300IR group than in the placebo group. CONCLUSION: Allergen immunotherapy with 300IR 5-grass pollen sublingual tablets was consistently associated with AR symptom relief in adults and children and provided a clinically meaningful improvement in quality of life.

The contribution of sublingual immunotherapy to the achievement of control in birch-related mild persistent asthma: A real-life randomised trial

Background: Asthma control represents the main goal of asthma management and different strategies aim to avoid the long term downsides of inhaled corticosteroids. We investigated in real-life conditions the contribution of sublingual immunotherapy in achieving the control of birch-related mild persistent asthma compared to two usual step-up therapeutic options. Methods: A three-year open randomised study included 84 asthmatics, uncontrolled during the previous birch pollen season, despite a treatment with budesonide 400 μg/day. Patients randomly received budesonide 800 μg/day, budesonide 1600 μg/day, budesonide 400 μg/day plus montelukast 10 μg/day and budesonide 400 μg/day plus carbamylated allergoid of betulaceae pre-coseasonally. Asthma Control test, combined allergy symptoms and medications score, albuterol consumption, lung function, nasal eosinophils and nasal steroids usage were assessed as changes from the first to last pollen season. Result: Seventy-six patients concluded the study. All options, except budesonide 800 μg/day, produced an improvement of mean monthly Asthma Control test (p < 0.05). Patients undergoing low-dose budesonide plus immunotherapy achieved, after three years, an appreciable control (ACT mean score 24). A significant improvement was seen in all groups for allergy symptoms plus medications and bronchial reactivity. Albuterol consumption and lung function improved in all but the first group. Only budesonide plus immunotherapy reduced nasal eosinophils and nasal steroids usage. Two mild self-resolving adverse events were reported. Conclusions: For patients with respiratory allergy due to birch pollen and mild persistent asthma, sublingual immunotherapy added to low-dose inhaled corticosteroids appears effective in maintaining long-term seasonal asthma control, representing a safe opportunity to reduce the cumulative amount of delivered corticosteroids (AU)

A Case of Acute Pulmonary Embolism Associated with Dysplasminogenemia

The incidence of pulmonary embolism (PE) rises markedly with age, and only a few cases have been reported in younger adults. Thrombophilia has been reported as one of the predisposing factors for PE in younger adults. Here we report an extraordinary case of PE complicated with dysplasminogenemia, a rare genetic disorder resulting in hypercoagulability, in a young male. An 18-yr-old male visited an emergency room in the United States complaining chest discomfort. He was diagnosed as PE with deep vein thrombosis without apparent risk factors. Anticoagulation therapy with warfarin had been initiated and discontinued after 6 months of treatment. After returning to Korea he was tested for thrombophilia which revealed decreased activity of plasminogen and subsequent analysis of PLG gene showed heterozygous Ala620Thr mutation. He was diagnosed with PE complicated with dysplasminogenemia. Life-long anticoagulation therapy was initiated. He is currently under follow-up without clinical events for 2 yr.

[Efficacy of lid hygiene in treatment of patients with blepharoconjunctival form of dry eye syndrome].

A complex of measures for lid hygiene in ocular surface disease is developed. These measures together with tear substitution and antibacterial treatment were revealed to result in elimination of blepharoconjunctivitis and dry eye signs, recovery of tear film and long-time remission, that significantly improved patient's life quality. When using this complex of measures meibomian glands are not damaged and their anatomy and functions are completely preserved. Lid hygiene affects all glands, there is no pain and no local anesthesia needed, normal tear film is formed

Esofagitis eosinofílica en el diagnóstico de pirosis / Eosinophilic oesophagitis in the diagnosis of heartburn

La esofagitis eosinofílica es una entidad poco frecuente, más común en niños, caracterizada por la infiltración de la mucosa esofágica por múltiples leucocitos eosinófilos. Su clínica más frecuente es disfagia, impactación y pirosis. La apariencia endoscópica varía de una mucosa casi normal a la aparición de anillos o incluso estenosis esofágica. El diagnóstico definitivo es anatomopatológico y el tratamiento actual se basa en el uso de corticoides tópicos. Se presenta, a continuación, el caso de un joven de 17 años con clínica de disfagia y pirosis, diagnosticado de dicha enfermedad tras la realización de endoscopia, con buena evolución clínica tras el tratamiento corticoideo (AU)

Eosinophilic oesophagitis is a rare entity, more common in children, and characterised by the infiltration of the oesophageal mucosa by multiple eosinophilic leukocytes. Its most common clinical symptoms are dysphagia, food impaction and heartburn. The endoscopic appearance varies from an almost normal mucosa to the appearance of rings or oesophageal stricture. The final diagnosis is by histopathology and the current treatment is based on the use of topical corticosteroids. The case is presented of a 17 year-old with symptoms of dysphagia and heartburn. The disease was diagnosed after endoscopy, with a good clinical outcome after corticosteroid treatment (AU)

Lichen planus and cicatricial conjunctivitis: disease course and response to therapy of 11 patients.

BACKGROUND: Lichen planus is an autoimmune, inflammatory dermatosis of unknown cause that affects the skin and mucous membranes. OBJECTIVE: The aim of this study was to report the clinical features and response to therapy in a series of patients with ocular lichen planus. METHODS: A retrospective chart review was performed to identify patients with ocular lichen planus. Information about clinical presentation, treatment, and therapeutic response was extracted from the medical records. RESULTS: Eleven patients with ocular lichen planus were identified. The diagnosis was confirmed histologically for 10 patients. Nine patients were women. The average time from onset of ocular symptoms to diagnosis was 4.1 years. Eight patients had mucous membrane involvement at other sites. Disease was well controlled in eight patients. CONCLUSION: Lichen planus should be considered in the differential diagnosis of cicatricial conjunctivitis, especially when severe lichen planus is noted at other sites.

Systemic monoclonal antibody therapy (daclizumab) in the treatment of cicatrizing conjunctivitis in stevens-johnson syndrome, refractory to conventional therapy.

PURPOSE: At least one-year follow-up of a case series of young Stevens-Johnson syndrome (SJS) patients with cicatrizing ocular surface disease and recurrent inflammation (SJS-RI) treated with systemic humanized monoclonal antibody (daclizumab). METHODS: Five patients (median age 16 yr; range 8-34 yr) with SJS, with recurrent inflammation refractory to conventional immunotherapy, were enrolled in a prospective non-randomized case series study. Inclusion criteria were patients with SJS and ocular cicatrizing inflammatory disease with severe visual impairment, using topical or systemic anti-inflammatory and/or immunomodulatory drugs without clinical improvement resulting in persistent inflammation (SJS-RI). Treatment with Daclizumab 1 mg/Kg (intravenous) was scheduled in three cycles. First cycle with concomitant immunotherapy: a total of 5 doses, with 14 days interval between them (total of this cycle: 10 weeks). Second cycle: interval was increased to 3 weeks; the patients received 2 doses (the second cycle had a total of 6 weeks). Third cycle: maintenance phase with 4 weeks interval between each application, until at least 12 months of the total follow up. After the first cycle (5th dose), the patients were kept with preservative-free lubricants and systemic doxycycline. RESULTS: Control of ocular inflammation was observed at a median of 8 weeks (range 6-10 weeks) in all patients, with relapses in two patients at 20-36 weeks. Relapses were controlled with topical steroids at a median of 10 days, and within 2 weeks the steroids were tapered for both patients. CONCLUSION: In this small case series, daclizumab demonstrated to play a beneficial role in the control of the inflammatory process of the recurrent inflammation in SJS, refractory to conventional immunomodulatory therapy.

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