Anaïs Dartevel1, Benjamin Chaigne2, Laurence Moachon3, Florian Grenier4, Nicolas Dupin5, Loïc Guillevin2, Laurence Bouillet6, Luc Mouthon7. 1. Service de Médecine Interne, hôpital Cochin, Centre de Référence pour les maladies systémiques autoimmunes rares d'Ile de France, DHU Authors (Autoimmune and Hormonal Diseases), Université Paris Descartes, Sorbonne Paris Cité, Assistance Publique-Hôpitaux de Paris (AP-HP), Paris, France; Service de Médecine Interne, Centre national de référence des angiœdème (CREAK), Universités des Alpes-Grenoble 1, 38041 Grenoble, France. 2. Service de Médecine Interne, hôpital Cochin, Centre de Référence pour les maladies systémiques autoimmunes rares d'Ile de France, DHU Authors (Autoimmune and Hormonal Diseases), Université Paris Descartes, Sorbonne Paris Cité, Assistance Publique-Hôpitaux de Paris (AP-HP), Paris, France. 3. Service de Pharmacologie, Hôpital Cochin, AP-HP, and Université Paris Descartes, Sorbonne Paris Cité, Paris, France. 4. Service de médecine légale, Universités des Alpes-Grenoble 1, 38041 Grenoble, France. 5. Service de Dermatologie, Hôpital Cochin, AP-HP, and Université Paris Descartes, Sorbonne Paris Cité, Paris, France. 6. Service de Médecine Interne, Centre national de référence des angiœdème (CREAK), Universités des Alpes-Grenoble 1, 38041 Grenoble, France. 7. Service de Médecine Interne, hôpital Cochin, Centre de Référence pour les maladies systémiques autoimmunes rares d'Ile de France, DHU Authors (Autoimmune and Hormonal Diseases), Université Paris Descartes, Sorbonne Paris Cité, Assistance Publique-Hôpitaux de Paris (AP-HP), Paris, France. Electronic address: luc.mouthon@aphp.fr.
Abstract
OBJECTIVE: To characterize levamisole-induced vasculopathy. METHODS: We performed a systematic review searching MEDLINE for articles published from 1972 to 2016. RESULTS: We retrieved 357 references and abstracts and selected 111 articles. Levamisole-induced vasculopathy was reported in 192 patients, with a female predominance (n = 122, 63.5%). Median [interquartile range] age was 44 [38-50]. Skin was the most frequently involved organ (n = 182, 94.8%). Cutaneous lesions were mostly on the face (n = 136, 70.8%), especially the ears. Purpura (n = 131, 68.2%) was the most reported cutaneous lesion. Organ involvement included acute renal failure (n = 24, 12.5%), and pulmonary involvement (n = 20, 10.4%). Anti-neutrophil cytoplasmic antibodies (ANCAs) were found in 167/178 patients (93.8%), with both anti-myeloperoxydase and anti-proteinase 3 specificity reported in 51/118 patients (43.2%). Anti-phospholipid (APL) antibodies were found in 93/137 patients (67.9%). Leukopenia was detected in 69/138 patients (50%). Skin biopsies identified vasculitis and thrombotic vasculopathy in 73/148 (49.3%) and 62/148 (41.9%) patients, respectively. The outcome was favourable in 116/134 patients (86.6%), but relapses were reported in 33 (28.4%), mainly on levamisole re-exposure. CONCLUSION: Levamisole-induced vasculopathy is characterized by a female predominance, skin involvement, ANCA and/or APL antibody positivity, leukopenia, vasculitis or vascular thrombotic histological lesions, and despite possible systemic involvement, a favourable outcome with levamisole interruption.
OBJECTIVE: To characterize levamisole-induced vasculopathy. METHODS: We performed a systematic review searching MEDLINE for articles published from 1972 to 2016. RESULTS: We retrieved 357 references and abstracts and selected 111 articles. Levamisole-induced vasculopathy was reported in 192 patients, with a female predominance (n = 122, 63.5%). Median [interquartile range] age was 44 [38-50]. Skin was the most frequently involved organ (n = 182, 94.8%). Cutaneous lesions were mostly on the face (n = 136, 70.8%), especially the ears. Purpura (n = 131, 68.2%) was the most reported cutaneous lesion. Organ involvement included acute renal failure (n = 24, 12.5%), and pulmonary involvement (n = 20, 10.4%). Anti-neutrophil cytoplasmic antibodies (ANCAs) were found in 167/178 patients (93.8%), with both anti-myeloperoxydase and anti-proteinase 3 specificity reported in 51/118 patients (43.2%). Anti-phospholipid (APL) antibodies were found in 93/137 patients (67.9%). Leukopenia was detected in 69/138 patients (50%). Skin biopsies identified vasculitis and thrombotic vasculopathy in 73/148 (49.3%) and 62/148 (41.9%) patients, respectively. The outcome was favourable in 116/134 patients (86.6%), but relapses were reported in 33 (28.4%), mainly on levamisole re-exposure. CONCLUSION:Levamisole-induced vasculopathy is characterized by a female predominance, skin involvement, ANCA and/or APL antibody positivity, leukopenia, vasculitis or vascular thrombotic histological lesions, and despite possible systemic involvement, a favourable outcome with levamisole interruption.
Authors: Florian Conrad; Sarah Hirsiger; Sebastian Winklhofer; Markus R Baumgartner; Philipp Stämpfli; Erich Seifritz; Susanne Wegener; Boris B Quednow Journal: J Psychiatry Neurosci Date: 2021-04-12 Impact factor: 6.186
Authors: Alberto Barbotti; Alina Cristiana Gheorghiu; Tanja Fusi-Schmidhauser; Lorenzo Grazioli-Gauthier Journal: Eur J Case Rep Intern Med Date: 2022-07-25