Anti IgE antibody as treatment of allergic bronchopulmonary aspergillosis in a patient with cystic fibrosis.

Allergic bronchopulmonary aspergillosis (ABPA) occurs in 7-9% of patients with cystic fibrosis (CF) and causes a worsening of lung function and respiratory symptoms. Standard treatment of ABPA consists of oral steroids; however, higher corticosteroid therapy associated to antifungal agent (itraconazole) long-term treatments are often required to reduce respiratory exacerbations and to prevent progressive lung damage. Here we describe the case of a girl with CF who experienced clinical and functional improvement over 12-months treatment with omalizumab. At birth, our patient was diagnosed with mild-to-moderate CF and from childhood she underwent annual cycles of antibiotic and corticosteroid therapies. At 12 years, she presented with a worsening respiratory condition, asthma symptoms and reduced lung function (FEV1 of 78%). Blood tests showed an increased concentration of plasma total IgE and positive specific IgE antibodies to Aspergillus fumigatus; allergic skin tests were also positive for A. fumigatus. The patient started steroid therapy but had impaired glucose tolerance due to long-term steroid use. Subcutaneous omalizumab 300 mg every two weeks was initiated and after 14 weeks she had improved respiratory symptoms (FEV1 99%) and a marked reduction in the use of systemic antibiotic and corticosteroid therapies. No side effects were reported. Our case shows that therapy with omalizumab for a prolonged period can resolve symptoms of asthma.

Vernal keratoconjunctivitis: atopy and autoimmunity.

BACKGROUND: Vernal Keratoconjunctivitis (VKC) is a rare chronic ocular inflammatory disease and it mainly affects boys in the first decade of life. Although it is a self-limiting disease, patients may present many phases characterized by an exacerbation of inflammatory symptoms with a consequent decline of the quality of life. PURPOSE: define the clinical and immunological profile of patients affected by VKC and investigate their familiar history of autoimmune disorders and their autoimmunity pattern. PATIENTS AND METHODS: 28 children were enrolled (20 males, 71%) aged between 4 and 14 years of life affected by VKC. Family history of allergic and immunological diseases was collected for each patient. In particular, it was asked whether some components of their families were affected by Hashimoto's thyroiditis, type I diabetes, psoriasis or rheumatoid arthritis and Systemic Lupus Erythematosus (SLE). All VKC children underwent a serological evaluation of anti-nuclear antibodies (ANA). RESULTS: A family history of immunological disorders was found in 46% of patients, 28% of Hashimoto's thyroiditis, 14% of type I diabetes, 14% of psoriasis, and 1 of Systemic Lupus Erythematosus. Furthermore, 35% of patients was ANA positive and they corresponded to patients with a higher ocular score and with the most important clinical symptoms. CONCLUSIONS: the detection of ANA positivity and of a familiar history of autoimmune disorders in a high percentage of children with VKC may help us to better understand the association of this ocular inflammatory disease with systemic autoimmune disorders and atopic condition.

Habitual snoring in children with previous allergic sensitization.

Previous studies have reported a high prevalence of allergy in children with Habitual Snoring (HS), but the relationship between allergy in the early years of life and the subsequent development of this Sleep Disordered Breathing (SDB) is yet to be elucidated. The purpose of the present study was to determine the role of early, under 36 months of age, allergic sensitization to food (with or without sensitization to airborne allergens) in determining the development of HS 8-10 years after. One hundred and forty-eight children (10-14 years, mean age 12 years) with a history of food allergy were selected. Under the age of 36 months, atopic status was assessed by skin prick test for a panel of airborne and food allergens. Questionnaires filled in by parents were used to collect information on children's snoring and associated symptoms. HS was defined as snoring three or more times per week. At 1-3 years of age 54 children were positive to food allergens alone, and 94 were positive also to airborne allergens. After 8-10 years of life, when patients were aged between 10 and 14 years, habitual snoring was reported in 37 children. Furthermore, among the 54 children under three years of age sensitized only to food, 8 became HS while of the 94 children sensitized to both food and inhalants allergens 29 developed HS. The difference between those two groups was statistically significant (p=0.04). We reported a significant risk of developing HS in children with early allergic sensitization. Specifically this risk was higher when food allergy was associated with inhalant allergy. The onset of upper airway inflammation due to allergic triggers in subjects under three years of age may be related to the subsequent development of SDB after 8-10 years.

Serum levels of IL-17 in patients with vernal keratoconjunctivitis: a preliminary report.

BACKGROUND AND AIM: Vernal Keratoconjunctivitis (VKC) is a chronic and often severe form of bilateral tarsal and/or bulbar conjunctivitis. The purpose of the present study is to measure the Interleukin-17 (IL-17) serum levels in children with VKC evaluating the role of the systemic inflammation in patients affected by VKC. PATIENTS AND METHODS: Fifteen patients were enrolled with VKC aged between 6 and 10 years of life. Serum were obtained from the peripheral blood samples collected from all the children included in the study to evaluate serum level of IL-17. RESULTS: Serum levels of IL-17 were significantly higher in patients with VKC than in healthy controls (10.3 ± 9.36 pg/ml vs. 3.3 ± 6.20 pg/ml respectively; p < 0.04). CONCLUSIONS: The presence of a significantly higher level of IL-17 in patients with VKC suggests a possible role of this cytokine in the pathogenesis of VKC. Further studies on larger samples of patients are warranted to confirm These findings in order to identify new possible therapeutic targets.

Keratoconjunctivitis by confocal microscopy after topical cyclosporine.

AIM: To describe the observations obtained by confocal microscopy concerning corneal stromal structure in the process of adenoviral epidemic keratoconjunctivitis, from the onset of the disease up to 20 weeks of follow-up, after topical cyclosporine. METHODS: A 16-years-old boy has developed subepithelial infiltrates and an epidemic keratoconjunctivitis from adenovirus in both eyes. Clinical case was analyzed by confocal microscopy (40x mode, Nidek ConfoScan 4) before and after administration of topical cyclosporine. RESULTS: Two weeks after the onset of the symptoms, corneal stroma showed the presence of cells with highly reflective area. These cells, comparable to keratocytes, had a typical fusiform shape and assumed a particular rosette disposition, never highlighted in the literature. Hyperreflective areas disappeared in both eyes after administration of topical cyclosporine 1% for 30 days. This outcome was also confirmed after five months. CONCLUSION: The confocal microscopy performed on the surface of the stroma before and after treatment with cyclosporine suggests the presence of a localized immune activation in subepithelial layer.

Food allergy and the development of asthma symptoms.

Allergies are multifactorial diseases the onset of which depends also on genetic and environmental factors in early life. Thus, environmental factors can affect the immune response and modify lung development, thereby leading to asthma. The role of the factors used to date to predict asthma development is modest, and clinical criteria should always be considered in association with familiarity for atopy. The aim of this study is to evaluate the risk of asthma in a population with positive skin prick test (SPT) (which is a reliable marker of atopy) to food allergens, regardless of clinical manifestations in the early years of life. The cohort of children enrolled in our study who had a positive SPT to food in the first three years of life had a prevalence of asthma after 7-14 years, double that of the general pediatric population. This prevalence increased significantly in patients with SPT positivity for food and inhalant allergens. We identified a correlation between the sensitization profile in children under the age of 36 months and the development of asthma during a period of 7-14 years. This study confirms that early sensitization is an important risk factor for the development of asthma, particularly in association with sensitization to inhalants, and that the persistence of food sensitization in school-age children and adolescents is associated to more severe asthma.

The role of rhinomanometry after nasal decongestant test in the assessment of adenoid hypertrophy in children.

INTRODUCTION: Nasal respiratory obstruction is a very common otolaryngologic problem, often caused by adenoid hypertrophy (AH). Nasal fiberoptic endoscopy (NFE) represents the gold standard method to diagnose AH. Rhinomanometry represents a valid diagnostic support. OBJECTIVE: The aim of our study was to analyze the diagnostic value of rhinomanometry after nasal decongestant (ND) test for the evaluation of adenoid hypertrophy in children. MATERIALS AND METHODS: Seventy-one of 97 collaborative children, aged 6-12 years, affected by upper airways obstructive symptoms and diagnosed as 'chronic oral breathers' by a standardized questionnaire were included in the study. The first evaluation included a complete physical examination, anterior rhinoscopy and anterior active rhinomanometry. Patients with a positive rhinomanometry underwent a second rhinomanometry after the administration of the nasal decongestant (ND) xylometazoline. All children were evaluated using nasal fiberoptic endoscopy (NFE). RESULTS: At rhinomanometry a normal nasal airflow was found in 19 (26.8%) of children while nasal obstruction was underlined in 52 (73.2%). These patients were tested also with rhinomanometry after ND which confirmed the presence of nasal obstruction in 29 (55.7%) of patients. All patients included in the study underwent a NFE: 34 (47.8%) of them presented severe AH with an occlusion >75% of the choanal opening (grade ≥ 3) and 37 (52.2%) presented no or a mild form of AH (grade < 3). When compared to NFE, rhinomanometry test after ND had 82.7% sensitivity and 82.6% specificity. Positive predictive value and negative predictive value were 85.7% and 79.2%, respectively. Two receiver operating characteristic (ROC) curves were derived using data related to rhinomanometry vs NFE, and to rhinomanometry after ND vs NFE. CONCLUSIONS: Rhinomanometry after ND, compared to rhinomanometry, is more specific and useful to evaluate nasal obstruction due to AH in children, and it may be helpful to avoid unnecessary surgical procedures in children with temporary nasal obstruction.

An infant with diaphragmatic eventration and respiratory distress.

The authors report a rare case of partial diaphragmatic eventration in a 4-month-old infant with recurrent wheezing and low serum IgA values. Because of persistent respiratory symptoms after therapy with inhaled short-acting beta2 agonists and inhaled nebulized corticosteroids, surgery was undertaken to correct the defect. Despite surgery, the clinical symptoms did not improve. Consequently, gatroesophagel reflux was considered and the diagnosis was confirmed with pH-metry, after which the infant was started on a protonic pump inhibitor therapy (PPI), achieving clinical improvement. Our experience suggests that in infants with congenital diaphragmatic eventration who present with respiratory distress gastro-oesophageal reflux should be suspected, and PPI therapy should be started before planning surgery.