Hoigné's syndrome, an uncommon mimicker of anaphylaxis

Consolascio, Danilo; Bronz, Gabriel; Lardelli, Pietro F.; Milani, Gregorio P.; Lava, Sebastiano A. G.; Terziroli Beretta-Piccoli, Benedetta; Bianchetti, Mario G.; Bergmann, Marcel M.; Rizzi, Mattia

doi:10.1016/j.jaut.2023.103164

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Terziroli_2024_Elsevier_Journ autoimmun_Hoigné’s syndrome, an uncommon mimicker

Journal article

Hoigné's syndrome, an uncommon mimicker of anaphylaxis : systematic literature review

Consolascio, Danilo Faculty of Biomedical Sciences, Università della Svizzera italiana, Switzerland
Bronz, Gabriel ORCID Pediatric Institute of Southern Switzerland, Ente Ospedaliero Cantonale, Bellinzona, Switzerland
Lardelli, Pietro F. Faculty of Biomedical Sciences, Università della Svizzera italiana, Switzerland
Milani, Gregorio P. Pediatric Unit, Fondazione IRCCS Ca’ Granda Ospedale Maggiore Policlinico, Milan, Italy - Department of Clinical Sciences and Community Health, Università degli Studi di Milano, Italy
Lava, Sebastiano A. G. ORCID Pediatric Cardiology Unit, Department of Pediatrics, Centre Hospitalier Universitaire Vaudois and University of Lausanne, Switzerland - Clinical Pharmacology and Therapeutics Group, University College London, United Kingdom
Terziroli Beretta-Piccoli, Benedetta ORCID Epatocentro Ticino, Lugano, Switzerland - Faculty of Life Sciences & Medicine, King’s College London, King’s College Hospital, United Kingdom - Faculty of Biomedical Sciences, Università della Svizzera italiana, Switzerland
Bianchetti, Mario G. Faculty of Biomedical Sciences, Università della Svizzera italiana, Switzerland
Bergmann, Marcel M. Faculty of Biomedical Sciences, Università della Svizzera italiana, Switzerland - Pediatric Allergy Unit, Department of Pediatrics, Gynecology and Obstetrics, University Hospitals of Geneva, Switzerland
Rizzi, Mattia ORCID Pediatric Institute of Southern Switzerland, Ente Ospedaliero Cantonale, Bellinzona, Switzerland - Faculty of Biomedical Sciences, Università della Svizzera italiana, Switzerland - Pediatric Oncology/Hematology Unit, Department of Women-Mother-Child, Centre Hospitalier Universitaire Vaudois and University of Lausanne, Switzerland

2024

Published in:

Journal of Autoimmunity. - 2024, vol. 143, p. 103164

English The term Hoigné’s syndrome denotes a mimicker of anaphylaxis, which occurs immediately after the parenteral administration of a drug and is likely caused by non-thrombotic pulmonary and systemic drug micro-embolization. It has so far been documented uniquely in case reports and small case series. Because this condition has never been systematically evaluated, we performed a structured literature review (pre-registered as CRD42023392962). The search was carried out in Excerpta Medica, National Library of Medicine, and Google Scholar. Cases with features consistent with anaphylaxis, urticaria, angioedema, asthma, syncope, anxiety, or panic attack triggered by needle phobia, and local anesthetic systemic toxicity were excluded. For the final analysis, we retained reports published between 1951 and 2021, which presented 247 patients with Hoigné’s syndrome: 37 children and 211 adults with a male: female ratio of 2.1 : 1.0. The patients presented within 1 min after parenteral administration of a drug (intramuscular penicillin in 90 % of the cases) with chest discomfort, shortness of breath, fear of death, psychomotor agitation, and auditory or visual hallucinations and impairment. Recovery occurred within 30 min. The diagnosis of Hoigné’s syndrome was also established in five patients 66–91 years of age with pre-existing cardiovascular or pulmonary diseases, who suddenly died after the administration of penicillin despite not exhibiting the aforementioned symptoms. It was therefore speculated that pulmonary drug micro-embolization induced a lethal cardiovascular compromise in these individuals. Histologic investigations supporting this hypothesis were performed in only one case. The diagnosis of Hoigné’s pulmonary drug micro-embolization was established also in five patients with pre-existing cardiovascular or pulmonary diseases, who suddenly died after the administration of penicillin despite not exhibiting the afore mentioned symptoms. Histologic investigations supporting this hypothesis were performed in only one case. In conclusion, Hoigné's syndrome is an uncommon non-immune-mediated reaction. This report seeks to promote broader awareness and knowledge regarding this alarming mimicker of anaphylaxis. Diagnosis relies solely on clinical evaluation.

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