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An Unusual Case of Successful Desensitization in a Patient with an Atypical Presentation of Salicylate Hypersensitivity / Intolerance

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An Unusual Case of Successful Desensitization in a Patient with an Atypical Presentation of Salicylate Hypersensitivity / Intolerance An Unusual Case of Successful Desensitization in a Patient with an Atypical Presentation of Salicylate Hypersensitivity / Intolerance Abdullah E Laher Division of Emergency Medicine & Department of Critical Care, University of the Witwatersrand The Asthma and Allergy Clinic, Rosebank, Johannesburg Case • 48 year old female • 2 year history of chronic headaches, migraines and fatigue on exposure to sodium salicylate containing foods and beverages • 6 previous hospital admissions with “myoclonic jerk like” movements of her upper body that was not associated with loss of consciousness, after consuming NSAID’s or meal containing high amounts of sodium salicylate • No associated type 1 hypersensitivity / anaphylaxis symptoms – rhinitis, wheeze, urticaria, swollen turbinates, nasal polyps, hypotension, facial oedema, etc. Cont. • Flow-Cellular Antigen Stimulation Test (CAST) for sodium salicylate was positive – stimulation index-3.9 [normal <2], – percentage basophils-6.4% [normal <5%] • Symptoms had improved upon initiation of a low salicylate diet • Due to her concern of the restrictiveness and unsustainability with this diet, she requested to undergo a trial of an aspirin desensitization procedure Desensitization procedure • Desensitization was carried out over 2 days in ICU – Scripps Clinic protocol – 3 doses of aspirin on day 1 • 20mg at 8am, 40mg at 11am and 90mg at 2pm – 3 three doses on day 2 • 150mg at 8am, 250mg at 11am and 325mg at 2pm – Final dose (600mg at 8am) on the third morning Cont. • Post ingestion of the 2nd (40mg), 4th (150mg) and 5th (250mg) doses she developed the severe “myoclonic jerk like” movements that had lasted approximately 20 minutes each Used with permission Before discharge • She however tolerated the final 600mg dose of aspirin on the third morning without any adverse effects • She had also tolerated 1g of turmeric powder (high salicylate content) without developing any of her previous symptoms • Discharged with lifelong high dose maintenance aspirin therapy (600mg bd × 1 month, then 600mg mane + 300mg nocte × 1 month , then 300mg bd – lifelong) At 1 month follow-up • Complete tolerance of salicylate containing foods • Significant subjective improvement in quality of life • No longer complained of headaches /migraines or “myoclonic jerk like” movements What are salicylates • Salicylates are natural or synthetic compounds • Anticancer, anti-aging and anti-oxidant properties • Natural salicylates are mainly found in plants and serve to protect them against environmental diseases and insects • Present in various fruits, vegetables, teas, alcoholic beverages, herbs, spices, cosmetics, fragrances and medications Shirakawa, Elife, 2016 / http://www.webmd.com/allergies/salicylate-allergy Salicylate sensitivity / intolerance: • Symptoms may include Gell and Coombs type 1 hypersensitivity features: – Rhinitis, wheezing and urticaria • But also non-specific / “non-immune type” symptoms: – H+N – headaches, migraines, tinnitus, hyperacusis, hearing loss – CVS – palpitations, arrhythmias – GIT – irritable bowel symptoms (nausea, vomiting, reflux, bloating, flatulence, diarrhoea, constipation) – Rheum – joint pains, arthritis – Behavioural and psychiatric – irritability, restlessness, inattention, ADHD, enuresis, depression, anxiety attacks, panic attacks https://fedup.com.au/factsheets/additive-and-natural-chemical-factsheets/salicylates Cont. • Mostly diagnosed in patients with Aspirin Exacerbated Respiratory Disease (AERD) presenting with Samter’s triad: – recurrent nasal polyps, asthma and aspirin sensitivity • Reportedly present in approximately 20% of adults with asthma • True incidence of salicylate intolerance is not known as diagnosis is seldom considered in patients presenting with headache, gastrointestinal tract symptoms and other “non-immune” type symptoms Jenkins, BMJ, 2004 / http://www.thedailyheadache.com/2012/12/december-so-far-lots-of-migraines-a-new-elimination-diet-2.html Mechanism • +ve CAST result in our patient – implies a non-IgE immune mediated mechanism – But symptoms of headaches, migraine, fatigue and “myoclonic jerk like” movements suggest a non- immune mechanism • Possible that both immune as well as non- immune mechanisms were present in our patient • Immune dysregulation vs overactive/ underactive/ intolerance/ hypersensitivity ......... Salicylate desensitization • Literature is clear with regards benefit of aspirin desensitization in the setting of AERD (Aspirin Exacerbated Respiratory Disease) / Samter’s triad • We have not come across reports of successful desensitization following “non-immune” type symptoms that manifested in our patient Case reports of salicylate intolerance related “non-immune” type symptoms are mostly self-reported – web-based food and diet intolerance forums https://fedup.com.au/factsheets/additive-and-natural-chemical-factsheets/salicylates http://www.thedailyheadache.com/2012/12/december-so-far-lots-of-migraines-a-new-elimination-diet-2.html The successful resolution of “non- immune” type symptoms in our patient: • Mechanism may be attributed to an increase in the tolerance threshold to salicylates • Supported by the fact that food and other intolerances have been shown to be dose dependant – some individuals may safely tolerate lower concentrations of salicylate without manifesting clinical symptoms • Opens a new avenue for the potential therapy of patients manifesting troublesome “non-immune” type symptoms d/t various intolerances https://my.clevelandclinic.org/health/articles/problem-foods-is-it-an-allergy-or-intolerance Current uncertainties and areas of potential research 1. Will salicylate desensitization for “non-immune” type symptoms be associated with the same success as that in AERD? 2. How long would patients have to continue high dose aspirin therapy to maintain tolerance? Lifelong? 3. If so, what is the optimal daily maintenance dose? 4. Do the risks associated with chronic aspirin use (renal, GIT) outweigh the benefits of desensitization and resolution of troublesome symptoms? ???.
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