Patrizia Bonadonna1, Roberta Zanotti2, Mauro Pagani3, Massimiliano Bonifacio2, Luigi Scaffidi2, Elisa Olivieri1, Maurizio Franchini4, Federico Reccardini5, Maria Teresa Costantino6, Chiara Roncallo6, Marina Mauro7, Elisa Boni8, Fabio Lodi Rizzini9, Maria Beatrice Bilò10, Anna Rosaria Marcarelli11, Giovanni Passalacqua12. 1. Allergy Unit, Azienda Ospedaliera Universitaria Integrata of Verona, Verona, Italy; Multidisciplinary Mastocytosis Outpatient Clinic, Azienda Ospedaliera Universitaria Integrata of Verona, Verona, Italy. 2. Multidisciplinary Mastocytosis Outpatient Clinic, Azienda Ospedaliera Universitaria Integrata of Verona, Verona, Italy; Department of Medicine, Section of Hematology, Azienda Ospedaliera Universitaria Integrata of Verona, Verona, Italy. 3. Medicine Department, ASST Mantova, Mantova, Italy. 4. Allergy Service, Jesolo, Venice, Italy. 5. Pneumology Unit, Azienda Ospedaliero-Universitaria S Maria della Misericordia di Udine, Udine, Italy. 6. Allergy Unit, Department of Medicine, ASST Mantova, Mantova, Italy. 7. Allergy Unit, S. Anna Hospital, Como, Italy. 8. Allergy Unit, ASL Alessandria, Alessandria, Italy. 9. Allergy Unit, Spedali Civili di Brescia, Brescia, Italy. 10. Allergy Unit, Department of Internal Medicine, University Hospital of Ancona, Ancona, Italy. 11. Pneumology Unit, ASST Cremona, Cremona, Italy. 12. Allergy and Respiratory Diseases, IRCCS San Martino IST, University of Genoa, Genoa, Italy. Electronic address: passalacqua@unige.it.
Abstract
BACKGROUND: Up to 75% of patients with severe anaphylactic reactions after Hymenoptera sting are at risk of further severe reactions if re-stung. Venom immunotherapy (VIT) is highly effective in protecting individuals with ascertained Hymenoptera venom allergy (HVA) and previous severe reactions. After a 3- to 5-year VIT course, most patients remain protected after VIT discontinuation. Otherwise, a lifelong treatment should be considered in high-risk patients (eg, in mastocytosis). Several case reports evidenced that patients with mastocytosis and HVA, although protected during VIT, can re-experience severe and sometimes fatal reactions after VIT discontinuation. OBJECTIVE: To evaluate whether patients who lost protection after VIT discontinuation may suffer from clonal mast cell disorders. METHODS: The survey describes the characteristics of patients who received a full course of VIT for previous severe reactions and who experienced another severe reaction at re-sting after VIT discontinuation. Those with a Red Española de Mastocitosis score of 2 or more or a serum basal tryptase level of more than 25 ng/mL underwent a hematological workup (bone marrow biopsy, KIT mutation, expression of aberrant CD25) and/or skin biopsy. RESULTS: Nineteen patients (mean age, 56.3 years; 89.5% males) were evaluated. All of them had received at least 4 years of VIT and were protected. After VIT discontinuation they were re-stung and developed, in all but 1 case, severe anaphylactic reactions (12 with loss of consciousness, in the absence of urticaria/angioedema). Eighteen patients (94.7%) had a clonal mast cell disorder, 8 of them with normal tryptase. CONCLUSIONS: Looking at this selected population, we suggest that mastocytosis should be considered in patients developing severe reactions at re-sting after VIT discontinuation and, as a speculation, patients with mastocytosis and HVA should be VIT-treated lifelong.
BACKGROUND: Up to 75% of patients with severe anaphylactic reactions after Hymenoptera sting are at risk of further severe reactions if re-stung. Venom immunotherapy (VIT) is highly effective in protecting individuals with ascertained Hymenoptera venom allergy (HVA) and previous severe reactions. After a 3- to 5-year VIT course, most patients remain protected after VIT discontinuation. Otherwise, a lifelong treatment should be considered in high-risk patients (eg, in mastocytosis). Several case reports evidenced that patients with mastocytosis and HVA, although protected during VIT, can re-experience severe and sometimes fatal reactions after VIT discontinuation. OBJECTIVE: To evaluate whether patients who lost protection after VIT discontinuation may suffer from clonal mast cell disorders. METHODS: The survey describes the characteristics of patients who received a full course of VIT for previous severe reactions and who experienced another severe reaction at re-sting after VIT discontinuation. Those with a Red Española de Mastocitosis score of 2 or more or a serum basal tryptase level of more than 25 ng/mL underwent a hematological workup (bone marrow biopsy, KIT mutation, expression of aberrant CD25) and/or skin biopsy. RESULTS: Nineteen patients (mean age, 56.3 years; 89.5% males) were evaluated. All of them had received at least 4 years of VIT and were protected. After VIT discontinuation they were re-stung and developed, in all but 1 case, severe anaphylactic reactions (12 with loss of consciousness, in the absence of urticaria/angioedema). Eighteen patients (94.7%) had a clonal mast cell disorder, 8 of them with normal tryptase. CONCLUSIONS: Looking at this selected population, we suggest that mastocytosis should be considered in patients developing severe reactions at re-sting after VIT discontinuation and, as a speculation, patients with mastocytosis and HVA should be VIT-treated lifelong.
Authors: Peter Valent; Cem Akin; Patrizia Bonadonna; Karin Hartmann; Knut Brockow; Marek Niedoszytko; Boguslaw Nedoszytko; Frank Siebenhaar; Wolfgang R Sperr; Joanna N G Oude Elberink; Joseph H Butterfield; Ivan Alvarez-Twose; Karl Sotlar; Andreas Reiter; Hanneke C Kluin-Nelemans; Olivier Hermine; Jason Gotlib; Sigurd Broesby-Olsen; Alberto Orfao; Hans-Peter Horny; Massimo Triggiani; Michel Arock; Lawrence B Schwartz; Dean D Metcalfe Journal: J Allergy Clin Immunol Pract Date: 2019-02-05
Authors: Jason Gotlib; Tracy I George; Melody C Carter; K Frank Austen; Bruce Bochner; Daniel F Dwyer; Jonathan J Lyons; Matthew J Hamilton; Joseph Butterfield; Patrizia Bonadonna; Catherine Weiler; Stephen J Galli; Lawrence B Schwartz; Hanneke Oude Elberink; Anne Maitland; Theoharis Theoharides; Celalettin Ustun; Hans-Peter Horny; Alberto Orfao; Michael Deininger; Deepti Radia; Mohamad Jawhar; Hanneke Kluin-Nelemans; Dean D Metcalfe; Michel Arock; Wolfgang R Sperr; Peter Valent; Mariana Castells; Cem Akin Journal: J Allergy Clin Immunol Date: 2021-03-11 Impact factor: 14.290
Authors: Peter Valent; Karin Hartmann; Patrizia Bonadonna; Marek Niedoszytko; Massimo Triggiani; Michel Arock; Knut Brockow Journal: Int Arch Allergy Immunol Date: 2022-05-23 Impact factor: 3.767
Authors: Merel C Onnes; Abdulrazzaq Alheraky; Martijn C Nawijn; Tim E Sluijter; André B Mulder; Suzanne Arends; Hanneke N G Oude Elberink Journal: Clin Transl Allergy Date: 2022-09-07 Impact factor: 5.657