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1.
J Autoimmun ; 143: 103164, 2024 Feb.
Article in English | MEDLINE | ID: mdl-38194789

ABSTRACT

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.


Subject(s)
Anaphylaxis , Humans , Anaphylaxis/diagnosis , Anaphylaxis/etiology , Anaphylaxis/therapy , Diagnosis, Differential , Male , Female , Aged , Adult , Aged, 80 and over , Syndrome , Child , Penicillins/adverse effects , Drug Hypersensitivity/diagnosis , Drug Hypersensitivity/therapy
2.
Children (Basel) ; 9(2)2022 Feb 15.
Article in English | MEDLINE | ID: mdl-35204985

ABSTRACT

Acute hemorrhagic edema of young children, a benign skin-limited vasculitis, predominantly affects children 2 years of age or younger. The prevalence and clinical features of the Köbner and Pastia signs have never been systematically investigated in this vasculitis. To address this issue, we analyzed the data contained in the Acute Hemorrhagic Edema Bibliographic Database, which incorporates all reports on hemorrhagic edema published after 1969. A total of 339 cases (236 males and 103 females; 11 (8-18) months of age; median and interquartile range) were documented with at least 1 photograph and therefore included in this analysis. The Köbner sign occurred in 24 cases (14 males and 10 females; 11 (7-17) months of age), the Pastia sign in 51 cases (39 males and 12 females; 11 (8-15) months of age), and both Köbner and Pastia signs in 8 cases (7 males and 1 female; 11 (7-17) months of age). The lower legs, thighs, waistline, and groin were the regions that were most commonly affected with the Köbner sign, while the ankle, feet, cubital fossa, and elbow were most affected with the Pastia sign. The Köbner and Pastia signs are clinically relevant; they occur in about every fourth child affected with hemorrhagic edema and do not influence the disease progression.

3.
Mult Scler Relat Disord ; 57: 103365, 2022 Jan.
Article in English | MEDLINE | ID: mdl-35158471

ABSTRACT

Injection-site reactions to glatiramer are common and include erythema, pruritus, pain, or induration. Additionally, the present systematic review of the literature documents 20 cases of Nicolau syndrome following glatiramer, a rare but potentially severe skin reaction. Abdomen and thighs are the most frequently affected areas (80% of reported cases), and permanent skin damage has been observed in 30% of cases. Recurrences are rare (<10%).


Subject(s)
Nicolau Syndrome , Glatiramer Acetate/adverse effects , Humans , Injections, Intramuscular , Pain , Skin
4.
Clin Rev Allergy Immunol ; 63(3): 490-498, 2022 Dec.
Article in English | MEDLINE | ID: mdl-35553000

ABSTRACT

Finkelstein-Seidlmayer vasculitis, also referred to as acute hemorrhagic edema of young children, is a rare small-vessel leukocytoclastic vasculitis. This condition is skin-limited, mainly affects infants up to 2 years of age and spontaneously remits. It has been suggested that an infection or a vaccine precede (by ≤ 14 days) this vasculitis. To better understand the interplay between infections or vaccines and Finkelstein-Seidlmayer vasculitis, we utilized the data contained in the Acute Hemorrhagic Edema BIbliographic Database AHEBID. The database, initiated in 2019, is being regularly updated, encompasses the entire original literature on Finkelstein-Seidlmayer vasculitis published after the original description and is attainable on request. The possible existence of an infectious or a vaccine precursor was addressed in 447 cases. Most cases were preceded by an infection (N = 384; 86%), by a vaccination (N = 20; 4.4%), or both an infection and a vaccination (N = 17; 3.8%). No precursor was reported in the remaining cases (N = 26; 5.8%). Two distinct infections preceded the onset of the vasculitis in 11 of the 381 cases with infection-associated Finkelstein-Seidlmayer vasculitis. The following infectious precursors were reported: upper respiratory tract infection (N = 292); acute gastroenteritis (N = 40); a benign febrile infection (N = 36); lower respiratory tract infection (N = 22); further infections (N = 8). The temporal relationship between the infectious precursor and the onset of the skin eruption was detailed in 336 cases: 54 cases developed before resolution and 282 after resolution of the infection. In conclusion, most cases of Finkelstein-Seidlmayer vasculitis are preceded by an infection. In a minority of cases, this skin vasculitis develops before resolution of the infection. In most cases, however, this vasculitis develops after resolution of the infection. More rarely, this vasculitis is preceded by a vaccination.


Subject(s)
Exanthema , Vaccines , Vasculitis, Leukocytoclastic, Cutaneous , Child , Infant , Humans , Child, Preschool , Skin , Edema/complications , Hemorrhage
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