Infliximab for treatment of granulomatous disease in patients with common variable immunodeficiency.

PURPOSE: Eight to 22 % of common variable immunodeficiency (CVID) patients exhibit granulomas of the lungs, spleen, liver, and/or skin. Granulomas can be the most medically significant day-to-day problem for CVID patients. Currently, there are limited options for treatment of granulomas associated with CVID. METHODS: We treated five patients with CVID who exhibited significant clinical symptoms secondary to granulomas with infliximab. The patients were selected and treated based solely on clinical need and were not otherwise controlled or blinded to the therapy. After obtaining baseline studies (labs, spirometry, radiology) and excluding infection, they were treated with infliximab 5 mg/kg at week 0, 2, 6 and every 4 weeks thereafter. RESULTS: Post treatment improvements were seen in all 5 patients with significant clinical responses observed for both visceral and cutaneous granulomata. Four of the five patients were maintained on infliximab for 5 to 18 months (mean 9.4 months) without adverse reaction or increased susceptibility to infection. One patient completed 6 months of therapy with improvement of respiratory parameters but discontinued infliximab due to joint stiffness and rash that she attributed to the medication. CONCLUSION: In our series, infliximab (5 mg/kg monthly) was an effective treatment for cutaneous and visceral granulomas in patients with CVID.

Recurrent perioperative anaphylaxis in a 54-year-old man.

Reports suggest that perioperative anaphylaxis in patients undergoing general anesthesia range from 1 in 5000 to 1 in 20,000 with mortality rates as high as 9%. Because of the variety of medications that are used for general anesthesia and the rapid succession in which they are administered, it is often difficult to determine the etiology of a severe allergic episode in this setting. Antibiotics and anesthetics are notorious for precipitating allergic reactions and are often implicated. Other perioperative exposures and patient risk factors must also be considered. In this article, we describe the case of a patient who exhibited recurrent anaphylaxis episodes while trying to undergo a vital cardiac surgery.

Oral food challenge and food allergy quality of life in caregivers of children with food allergy.

BACKGROUND: Food allergy is associated with diminished patient and caregiver quality of life (QoL). Although oral food challenge (OFC) improves QoL of individuals with food allergy, its effects on caregiver QoL are unknown. OBJECTIVES: To determine if differences in caregiver QoL exist based on their child undergoing OFC. METHODS: Caregivers of individuals with food allergy who underwent OFCs between 2001 and 2012 at the University of Michigan Food Allergy Center completed the Food Allergy Quality of Life-Parental Burden index and a questionnaire that assessed details of the most-severe reaction by an individual with food allergy. Results were compared with 305 caregivers of individuals with food allergy who were unchallenged. All questionnaire data regarding the characteristics of the reactions of individuals with food allergy were verified through chart review. RESULTS: A total of 115 caregivers of individuals with food allergy who were undergoing OFC completed the QoL assessment. Caregivers of individuals with food allergy who were undergoing OFC had a significantly lower (better) QoL score than controls who were not challenged (1.5 vs 1.88; P = .02). Furthermore, within the challenged cohort, there was no significant difference in QoL score between those with a passing OFC (eg, non-reactive) and a failing OFC (eg, reactive) (1.42 vs 1.34; P = .83). In an adjusted linear regression model, the QoL score was significantly better among caregivers of individuals with food allergy who were undergoing OFC and with an income >$50,000 but significantly worsened for caregivers with multiple individuals with food allergy or if the individual with food allergy had atopic dermatitis. CONCLUSION: The caregiver QoL score is better with individuals with food allergy who underwent OFC versus controls who were unchallenged but not significantly different based on OFC outcome. QoL is, in addition, moderated by income, the presence of atopic dermatitis, and having multiple individuals with food allergy. OFC is associated with better caregiver QoL, irrespective of challenge outcome.