Recurrent Anaphylactic Reactions: An Uncommon Debut of Lymphocytic Hypophysitis

Abstract

We report on a 24-year-old male, with exercise-induced asthma and intermittent abdominal pain since puberty, who suffered from recurrent anaphylactic reactions. He also complained of occasional headaches. After extensive studies he was eventually diagnosed with idiopathic anaphylaxis, once the following diagnoses had been excluded: allergic origin [foods (including ω5-gliadin), latex and drugs], hydatidosis, carcinoid syndrome, systemic mastocytosis, autonomic epilepsy, hereditary angioedema, pheocromocitoma, Meckel diverticle, medullar thyroid carcinoma, leukemia, hyper-IgE and hypereosinophilic syndromes. Given the frequency and severity of the attacks, we started off-label treatment with omalizumab, initially well tolerated. Some days after the second dose the patient started to develop recurrent urticaria. Because of these new symptoms, blood work was repeated, and elevated TSH, decreased T4, positive antithyroid antibodies and decreased cortisol levels with normal ACTH were found. The antiadrenal autoantibodies were negative. The MRI showed a slight thickening of the infundibulum, without pituitary adenoma. Suspecting an autoimmune hypophysitis, we looked for antipituitary antibodies; the result was positive. A clinical picture of recurrent anaphylactic reactions, the result of complicated adrenal crises in an asthmatic patient, was a manifestation of lymphocytic hypophysitis, a rare chronic inflammatory disease of autoimmune etiology. One year after replacement therapy had been started, the patient remained asymptomatic.

<p>Management Strategies Of Idiopathic Anaphylaxis In The Emergency Room: Current Perspectives</p>

“…Several case reports have identified rare causes of anaphylaxis in patients who were initially diagnosed with IA. Such causes of anaphylaxis include pigeon tick, A. Reflexus ,25 lymphocytic hypophysitis in the context of complicated adrenal crises in an asthmatic patient,26 and wheat-dependent exercise-induced anaphylaxis 27. In fact, in a study done by Heaps et al, 2014,28 wheat protein, omega-5-gliadin, and shrimp were the main causes of anaphylaxis in the study’s cohort of patients who were initially diagnosed with IA.…”

“…Doses of omalizumab ranged from 225 to 375 mg administered at a frequency of every two, four, or eight weeks. The majority of these studies reported resolution of IA episodes that was maintained over 12 months after stopping omalizumab treatment 12–14,21,26,35–38. Two case reports included in this review have described relapse in two patients after a short period of treatment (three and four months of treatment) and an adverse event (anaphylaxis) in another patient undergoing omalizumab treatment for frequent IA episodes 15,39…”

A Case of Nonsmall-Cell Lung Cancer with Anaphylaxis after 41 Courses of Pembrolizumab along with Adrenal Insufficiency as an Immune-Related Adverse Event

“…Low GC levels due to adrenal insufficiency may have led to the dysregulation of these parameters, promoting an overshoot of the allergic reaction and making the patient more susceptible to anaphylaxis. For example, adrenal insufficiency triggered by lymphocytic hypophysitis was reported to induce repeated anaphylaxis [ 11 ].…”