Early sustained unresponsiveness after short-course egg oral immunotherapy: a randomized controlled study in egg-allergic children.

BACKGROUND: No studies have evaluated the potential of egg oral immunotherapy (egg-OIT) to induce sustained unresponsiveness after discontinuing therapy following short-term treatments. OBJECTIVE: We assessed the efficacy of short-course egg-OIT to induce sustained unresponsiveness. METHODS: Sixty-one egg-allergic children, 5 to 17 years old, with positive double-blind placebo-controlled food challenge (DBPCFC) to dehydrated egg white (EW) were randomized to receive egg-OIT (OITG) for 3 months (maintenance dose one undercooked egg every 48 hours) or to continue egg avoidance diet (control group, CG) for 4 months. Children who completed egg-OIT avoided egg for 1 month. At 4 months, both groups underwent a DBPCFC. OITG participants who passed this challenge were instructed to add egg to their diet ad libitum. Immune markers were studied at different time points. RESULTS: Ninety-three percent (28/30) of OITG children were desensitized in a median of 32.5 days (IQR, 14 days). At 4 months, 1/31 (3%) in CG passed DBPCFC and 11/30 (37%) of OITG (95% CI, 14 to 51%; P = 0.003), all of them were consuming egg at 36 months. A decrease in EW, OVA and OVM skin test results and OVA-specific IgE (sIgE) levels was observed on OITG at 4 months (P = 0.001). EW-, OVA- and OVM-sIgE levels prior to the start of egg avoidance diet were lower in OITG children who passed DBPCFC at 4 months than in those who did not pass it. EW- and OVM-sIgE showed the best diagnostic performance in predicting DBPCFC result at 4 months. Levels above optimal EW-sIgE cut-off of 7.1 kU/L indicated 90% probability of failing DBPCFC. CONCLUSION: This is the first demonstration of sustained unresponsiveness with a three-month egg-OIT protocol. Almost all treated subjects were desensitized and 37% achieved sustained unresponsiveness. EW-sIgE levels at the end of treatment predicted sustained unresponsiveness. This protocol shows a new approach to OIT for egg-allergic children.

Renal amyloidosis in a patient with X-linked agammaglobulinemia (Bruton's disease) and bronchiectasis.

PURPOSE: We present a patient with Bruton's disease and bronchiectasis who developed renal AA amyloidosis. CASE REPORT: A 38 year-old man was diagnosed with X-linked agammaglobulinemia (Bruton's disease) when he was 3 years old, and he has been treated with parenteral immunoglobulin since then. Eighteen years later, he was diagnosed with central pulmonary bronchiectasis by computerized tomography (CT). In 2008, he gradually developed anemia, edema of lower limbs, and loss of weight. METHOD AND RESULTS: Laboratory studies revealed deterioration of renal function, normocytic normochromic anemia and nephrotic range proteinuria. Hepatitis B and C and HIV serology were negative. Ultrasound and CT of abdomen were normal. A renal biopsy revealed deposits with positive PAS and Congo red staining in glomeruli, interstitium, and vessel's walls. Immunohistochemistry showed positive staining of the A amyloid. Direct immunofluorescence was positive with thioflavin and showed focal and glomerular mesangial IgG deposits, suggesting renal AA amyloidosis. For 2 years the patient conducted pharmacological treatment and follow-up for the Nephrology department with poor prognosis and progression of renal function impairment. In January 2011 he began dialysis treatment with improvement, and he is currently on the waiting list for renal transplantation. CONCLUSION: We present a patient with Bruton's disease and bronchiectasis who developed renal AA amyloidosis a finding rarely reported.

Type IV reaction due to phenylephrine administered nasally with cross-reactivity with ethylephrine.

Type IV hypersensitivity eye reactions have been described after the administration of the sympathomimetic agent phenylephrine. We report the case of an atopic woman who developed nasal congestion and discharge, dysphagia, and dyspnea 1 hour after the administration of Stopcold pills and Disneumon Pernasal nasal spray for otitis. The same symptoms reappeared after the accidental administration of Rinobanedif ointment in the nasal mucosa. Skin patch tests were performed with a standard True Test panel, preservatives, Disneumon Pernasal, pseudoephedrine, eyedrops (tropicamide, cyclopentolate, and phenylephrine), and other sympathomimetic agents. The patient also underwent oral, ocular, and nasal controlled challenges with the same drugs. Finally, patch tests were performed in 11 controls with phenylephrine and ethylephrine. Our patient had a positive outcome in patch testing with nickel sulphate, fragrance mix, phenylephrine, and ethylephrine. To our knowledge, this is the first report of a type IV reaction to nasally administered phenylephrine with cross-reactivity with ethylephrine detected by patch testing.