Tolerance to sterilised cow's milk in patients with eosinophilic oesophagitis triggered by milk.

BACKGROUND: Cow's milk protein is the main food trigger for eosinophilic oesophagitis (EoE) in children and adults and should be continuously avoided once identified as such. AIMS: To evaluate tolerance of sterilised cow's milk (boiled instead of UHT processing) with regard to maintenance of EoE remission, health-related quality of life (HRQoL), nutritional intake and allergic sensitisation in patients of all ages with milk-triggered EoE METHODS: We prospectively recruited patients in whom cow's milk was demonstrated to trigger EoE after an empirical food elimination diet-based study. They were given 200 ml of sterilised cow's milk twice daily for 8 weeks. Endoscopic assessment, peak eosinophil counts, oesophageal-related symptoms, HRQoL, blood eosinophils, eosinophil cationic protein (ECP), skin prick test and serum total and specific immunoglobulin E (IgE) to major milk proteins were monitored before and after sterilised milk intake. RESULTS: Eighteen patients (13 male) in EoE remission underwent a sterilised milk challenge. Twelve maintained EoE remission (<15 eos/hpf) while EoE recurred in the remainder. Endoscopic appearances deteriorated in non-tolerant patients. HRQoL scored well at baseline and was maintained among patients tolerant to sterilised milk, but deteriorated in reactive ones. No significant changes in blood eosinophil count, ECP, tryptase or total and milk-specific IgE serum levels were observed from baseline. However, cow's milk-specific IgE increased slightly in non-tolerant patients. Clinical and histological remission were maintained in patients who regularly consumed sterilised milk for 1 year. CONCLUSION: Sterilised milk did not trigger EoE in two-thirds of patients with documented milk-induced EoE, in either the short or long term.

CHAOS: síndrome de obstrucción congénita de las vías aéreas superiores / CHAOS: congenital high airway obstruction syndrome

Resumen El síndrome de obstrucción congénita de vías áreas superiores (CHAOS) es una condición que se caracteriza por la existencia de una obstrucción en las vías áreas altas en el feto, la cual puede ser parcial o completa. Comúnmente es una situación incompatible con la vida, por lo que su diagnóstico prenatal es importante considerando el pronóstico y los diferentes manejos prenatales y posnatales que existen. Presentamos un caso de CHAOS diagnosticado en la semana 21, con una breve revisión de la literatura sobre su diagnóstico, pronóstico y alternativas terapéuticas.

Abstract Congenital high airway obstruction syndrome (CHAOS) is a condition characterized by the existence of an obstruction of the fetal upper airways, which may be partial or complete. It is commonly incompatible with life, so its prenatal diagnosis is important due to the prognosis and the recently described pre and postnatal management options. We present a case of CHAOS in a pregnancy of 21 weeks with a brief review of the current literature about its diagnosis, prognosis and therapeutic alternatives.

Guidelines on eosinophilic esophagitis: evidence-based statements and recommendations for diagnosis and management in children and adults.

INTRODUCTION: Eosinophilic esophagitis (EoE) is one of the most prevalent esophageal diseases and the leading cause of dysphagia and food impaction in children and young adults. This underlines the importance of optimizing diagnosys and treatment of the condition, especially after the increasing amount of knowledge on EoE recently published. Therefore, the UEG, EAACI ESPGHAN, and EUREOS deemed it necessary to update the current guidelines regarding conceptual and epidemiological aspects, diagnosis, and treatment of EoE. METHODS: General methodology according to the Appraisal of Guidelines for Research and Evaluation (AGREE) II and the Grading of Recommendations Assessment, Development, and Evaluation (GRADE) system was used in order to comply with current standards of evidence assessment in formulation of recommendations. An extensive literature search was conducted up to August 2015 and periodically updated. The working group consisted of gastroenterologists, allergists, pediatricians, otolaryngologists, pathologists, and epidemiologists. Systematic evidence-based reviews were performed based upon relevant clinical questions with respect to patient-important outcomes. RESULTS: The guidelines include updated concept of EoE, evaluated information on disease epidemiology, risk factors, associated conditions, and natural history of EoE in children and adults. Diagnostic conditions and criteria, the yield of diagnostic and disease monitoring procedures, and evidence-based statements and recommendation on the utility of the several treatment options for patients EoE are provided. Recommendations on how to choose and implement treatment and long-term management are provided based on expert opinion and best clinical practice. CONCLUSION: Evidence-based recommendations for EoE diagnosis, treatment modalities, and patients' follow up are proposed in the guideline.

Association between atopic manifestations and eosinophilic esophagitis: A systematic review and meta-analysis.

BACKGROUND: Eosinophilic esophagitis (EoE) has repeatedly been associated with atopic manifestations, which are reported more frequently in these patients than in the general population. OBJECTIVE: To systematically assess the evidence and strength of the associations between EoE and atopy. METHODS: We performed a systematic search of the MEDLINE, EMBASE, and SCOPUS databases for case-control studies comparing the frequency of atopic diatheses among patients with EoE and control subjects representing the general population without EoE. Using random-effects meta-analyses, we calculated summary estimates, including 95% confidence intervals (CIs), for bronchial asthma, atopic rhinitis, and eczema. Publication bias risks were assessed by means of funnel plot analysis and specific statistical tests. RESULTS: Of the 2,954 references identified, data were collected from 21 studies, including a total of 53,542 patients with EoE and 54,759 controls. The criteria for defining a diagnosis of atopy in patients with EoE or controls was not structurally considered in most of the studies. Overall, allergic rhinitis was significantly more common among patients with EoE compared with control subjects (odds ratio [OR], 5.09; 95% CI, 2.91-8.90; I2 = 86.7%) as were bronchial asthma (OR, 3.01; 95% CI, 1.96-4.62; I2 = 84.5%) and eczema (OR, 2.85; 95% CI, 1.87-4.34; I2 = 57.1%). Food allergies and other atopic conditions were also assessed. No significant publication bias was found for studies dealing with allergic rhinitis and eczema in EoE. CONCLUSION: Despite pointing to a significant association between atopy and EoE, most of the studies provided no normalized diagnostic criteria for atopy. Further research should provide clear and standardized definitions of such conditions. TRIAL REGISTRATION: www.crd.york.ac.uk/PROSPERO Trial Identifier: CRD42016036161.

Efficacy of dietary interventions for inducing histologic remission in patients with eosinophilic esophagitis: a systematic review and meta-analysis.

BACKGROUND & AIMS: Various dietary interventions have been used to treat patients with eosinophilic esophagitis (EoE), yielding varied results. This systematic review assesses the efficacy of different dietary therapies in inducing disease remission. METHODS: We performed a systematic search of the MEDLINE, EMBASE, and SCOPUS databases for studies investigating the efficacy of dietary interventions (reducing infiltration by immune cells <15 eosinophils/high-power field in esophageal biopsies) for pediatric and adult patients with EoE. Summary estimates, including 95% confidence intervals (CI), were calculated for exclusive feeding with amino acid-based elemental formulas, allergy test result-directed food elimination diets, and 6-food elimination diets (SFED). A fixed- or random-effects model was used depending on heterogeneity (I(2)); publication bias risks were assessed by means of funnel plot analysis. RESULTS: The search yielded 581 references; of these, 33 were included in the quantitative summary. We analyzed data on a total of 1317 patients with EoE (1128 children and 189 adults) who received different dietary treatments. Elemental diets were effective for 90.8% of cases (95% CI, 84.7%-95.5%; I(2) = 52.3%), SFED for 72.1% (95% CI, 65.8%-78.1%; I(2) = 0), and allergy test result-directed food elimination for 45.5% of cases (95% CI, 35.4%-55.7%; I(2) = 75.1%). Additional strategies (elimination of cow's milk, gluten-free diets, and 4-food elimination diet) were also evaluated. Adults vs children had no significant differences in remission after dietary interventions (67.2% vs 63.3%). CONCLUSIONS: Dietary interventions are effective in producing histologic remission in patients with EoE. Elemental diets and SFEDs were the most effective, achieving <15 eosinophils/high-power field in 90.8% and 72.1% of patients, respectively.

Empiric 6-food elimination diet induced and maintained prolonged remission in patients with adult eosinophilic esophagitis: a prospective study on the food cause of the disease.

BACKGROUND: Although empiric exclusion from the diet of the 6 food groups most likely to trigger allergies achieves eosinophilic esophagitis (EoE) remission in children, data on its prolonged efficacy and effects on adults are lacking. OBJECTIVE: We sought to evaluate the efficacy of a 6-food elimination diet in inducing and maintaining prolonged remission in patients with adult EoE. METHODS: Sixty-seven consecutive patients with adult EoE were prospectively recruited and treated exclusively with a diet avoiding cereals, milk, eggs, fish/seafood, legumes/peanuts, and soy for 6 weeks. Subsequent challenge was undertaken by sequentially reintroducing all excluded single foods, followed by endoscopy and biopsies, which were developed every 6 weeks in case of response (eosinophil peak count reduction to <15/high-power field [hpf]). A food was considered a trigger for EoE and removed from the diet if pathologic eosinophilic infiltration (≥15 eosinophils/hpf) reappeared. Food-specific serum IgE measurements and skin prick tests were performed before initiating the diet. RESULTS: Forty-nine (73.1%) patients exhibited significantly reduced eosinophil peak counts (<15 eosinophils/hpf) before sequential single-food reintroduction. A single offending food antigen was identified in 35.71% of patients, 2 food triggers were identified in 30.95%, and 3 or more food triggers were identified in 33.3%. Cow's milk was the most common food antigen (61.9%), followed by wheat (28.6%), eggs (26.2%), and legumes (23.8%). Prior allergy tests showed no concordance with food-reintroduction challenge results. All patients who continued to avoid the offending foods maintained histopathologic and clinical EoE remission for up to 3 years. CONCLUSIONS: An empiric 6-food elimination diet effectively induced remission of active adult EoE, which was maintained for up to 3 years with individually tailored, limited exclusion diets.

Successful food elimination therapy in adult eosinophilic esophagitis: not all patients are the same.

Eosinophilic esophagitis (EoE) is a chronic, immune/antigen-mediated, esophageal disease characterized by esophageal dysfunction and eosinophilic inflammation, manifested mainly as dysphagia and frequent food impaction. EoE is recognized into the spectrum of food allergy, but food sensitization studies used not to be efficient to identify the triggering food, because of what patients are frequently treated with topic steroids or even endoscopic esophageal dilation. Herein, we describe 3 adult patients-all suffering from EoE, but with different sensitization patterns-who were treated successfully with elimination diets. Allergy tests indicated no food sensitization for patient 1, but challenge with milk and wheat were positive. Food IgE-mediated allergies were found in patients 2 and 3; inflammation was resolved with food elimination. Lack of food allergy sensitization does not exclude the possibility of food allergies as a cause of EoE; elimination diets must therefore be considered as an effective diagnostic and treatment tool.

Montelukast was inefficient in maintaining steroid-induced remission in adult eosinophilic esophagitis.

BACKGROUND AND AIMS: Leukotriene D4 is produced by and functions as a chemotactic factor for eosinophils. Eosinophilic esophagitis (EoE) is characterized by esophageal eosinophilic infiltration, determining structural changes and dismotility symptoms. Montelukast, a selective leukotriene D4 receptor antagonist, has gained increasing consideration as a therapeutic agent for EoE. However, limited available information has shown that montelukast is not effective in reducing eosinophilic infiltration. Our paper aims at evaluating whether montelukast could be consider as a steroid-sparing therapy by assessing its efficacy in maintaining both clinical and histopathological remission achieved after topical corticosteroids in adult EoE patients. METHODS: Eleven consecutively diagnosed adult EoE patients were prospectively studied. Esophageal biopsies were obtained before and after a 6-month treatment with fluticasone propionate 400 µg/twice a day. Immediately after that, montelukast 10 mg/day was instituted. A new endoscopy was foreseen after a new 3-month period, or as soon as the patients presented esophageal symptoms. Symptoms were assessed by using a questionnaire before and after fluticasone propionate treatment and after montelukast therapy. RESULTS: Eosinophils density into the esophageal epithelium and lamina propria was significantly reduced after a 6-month treatment with topical steroids (P = 0.003) and increased to levels similar to baseline level into the first 3 months after treatment with montelukast. Baseline symptom scores significantly decreased after treatment with topical steroids (P = 0.003) and increased again after montelukast therapy, but baseline levels improved. CONCLUSIONS: Montelukast was not efficient in maintaining the histopathological or clinical response achieved by topical steroids in adult EoE patients.

Control del asma y de la calidad de vida en pacientes asmáticos alérgicos graves con tratamiento anti-IgE (omalizumab) / Analysis of asthma control and quality of life in severe allergic asthmatics under treatment with anti-IgE, omalizumab

Fundamento y objetivos: Analizar la eficacia del omalizumab en la evolución del asma alérgica grave mediante los cuestionarios de control del asma (ACQ) y de calidad de vida en asma (AQLQ), así como el FEV1 y la apreciación global del médico. Pacientes y método: Se estudió a 14 pacientes con una evolución promedio del asma de 20,7 años con un test cutáneo positivo o un anticuerpo IgE específico a un aeroalérgeno habitual, todos ellos tratados con altas dosis de corticoides inhalados, y 8 tratados además con corticoides orales. Se suministraron 150–600mg de omalizumab por paciente, con periodicidad quincenal o mensual. Se evaluó a cada paciente asmático mensualmente y, al cabo de 4 meses, se precisó si era respondedor o no para decidir la continuidad del omalizumab. Resultados: La apreciación global del médico fue positiva en 11 de 14 pacientes. Se apreció un marcado beneficio a los 2 meses en el ACQ y el AQLQ (p<0,05) y a los 3 y los 4 meses sólo en el AQLQ y el AQLQ-síntomas, respectivamente (p<0,05). El FEV1 mejoró un promedio del 9,4% en 14 pacientes a los 4 meses (p=0,24). Los corticoides orales se redujeron en 4 de 8 pacientes y en otro paciente se suspendió definitivamente. En 2 de 14 pacientes se produjeron reacciones adversas que obligaron a suspender el tratamiento a pesar de apreciarse mejoría en los AQLQ y ACQ. Conclusiones: En nuestra experiencia, el omalizumab resulta eficaz en la mejora de la calidad de vida en más de 3 de cada 4 pacientes afectados de asma grave alérgica según la apreciación global del médico, y se observan, además, mejorías significativas en los ACQ, AQLQ y AQLQ-síntomas a los 2, a los 3 y a los 4 meses de seguimiento, sin apreciarse cambios significativos del FEV1, probablemente por cambios crónicos irreversibles (AU)

Background and objectives:To evaluate anti-IgE (ie, omalizumab) efficacy on severe allergic asthma in order to achieve the efficacy of this treatment on severe allergic asthma progress by means of asthma control questionnaire (ACQ), asthma quality of life questionnaire (AQLQ) as well as pulmonary function (FEV1) and physician overall assessment. Patients and methods: Fourteen patients were studied who suffered from severe allergic asthma for several years; average 20.7yr. A positive skin prick test or specific IgE antibodies to a common aeroallergen were observed in all patients who were under treatment with high doses of inhaled corticosteroids (eight of them also with oral corticosteroids). Omalizumab 150–600mg were administrated once or twice monthly and each patient was monthly evaluated until sixteen weeks, deciding at this time which patient was or not responder in order to follow up or not omalizumab treatment. Results: Physician overall assessment improved in 11 out of every 14 patients. A marked improvement was observed at two months in ACQ and AQLQ (p<0.05) and at 3 and 4 moths only in AQLQ and AQLQ-symptoms respectively (p<0.05). The FEV1 improved by 9.4% average (p=0.24). Oral corticosteroids were reduced in 4 of 8 patients and in another one there was a definitive suspension. Two patients suffered adverse reactions which suspension of treatment despite the fact that they presented improvement in questionnaires of quality of life and control of asthma. Conclusions: Omalizumab showed a marked global efficacy in more than 3 out of 4 patients suffering severe allergic asthma as revealed by physician overall assessment. Moreover significant improvements were observed in ACQ; AQLQ; AQLQ-symptoms at 2, 3 and 4 months of treatment, without significant changes in FEV1 probably due to chronic irreversible changes in airways (AU)

[Analysis of asthma control and quality of life in severe allergic asthmatics under treatment with anti-IgE, omalizumab]. / Control del asma y de la calidad de vida en pacientes asmáticos alérgicos graves con tratamiento anti-IgE (omalizumab).

BACKGROUND AND OBJECTIVES: To evaluate anti-IgE (ie, omalizumab) efficacy on severe allergic asthma in order to achieve the efficacy of this treatment on severe allergic asthma progress by means of asthma control questionnaire (ACQ), asthma quality of life questionnaire (AQLQ) as well as pulmonary function (FEV1) and physician overall assessment. PATIENTS AND METHODS: Fourteen patients were studied who suffered from severe allergic asthma for several years; average 20.7yr. A positive skin prick test or specific IgE antibodies to a common aeroallergen were observed in all patients who were under treatment with high doses of inhaled corticosteroids (eight of them also with oral corticosteroids). Omalizumab 150-600 mg were administrated once or twice monthly and each patient was monthly evaluated until sixteen weeks, deciding at this time which patient was or not responder in order to follow up or not omalizumab treatment. RESULTS: Physician overall assessment improved in 11 out of every 14 patients. A marked improvement was observed at two months in ACQ and AQLQ (p<0.05) and at 3 and 4 moths only in AQLQ and AQLQ-symptoms respectively (p<0.05). The FEV1 improved by 9.4% average (p=0.24). Oral corticosteroids were reduced in 4 of 8 patients and in another one there was a definitive suspension. Two patients suffered adverse reactions which suspension of treatment despite the fact that they presented improvement in questionnaires of quality of life and control of asthma. CONCLUSIONS: Omalizumab showed a marked global efficacy in more than 3 out of 4 patients suffering severe allergic asthma as revealed by physician overall assessment. Moreover significant improvements were observed in ACQ; AQLQ; AQLQ-symptoms at 2, 3 and 4 months of treatment, without significant changes in FEV1 probably due to chronic irreversible changes in airways.