학술논문
The role of ANCA in the management of cocaine-induced midline destructive lesions or ENT pseudo-granulomatosis with polyangiitis: a London multicentre case series.
Document Type
Academic Journal
Author
Pendolino AL; Department of ENT, Royal National ENT & Eastman Dental Hospitals, London, UK.; Ear Institute, UCL, London, UK.; Benshetrit G; Department of ENT, Imperial College Hospitals, London, UK.; Navaratnam AV; Department of ENT, Royal National ENT & Eastman Dental Hospitals, London, UK.; To C; Department of Allergy, Royal Brompton Hospital, London, UK.; Department of Immunology and Allergy, Princess Alexandra Hospital, Brisbane, Australia.; Bandino F; Department of ENT, Royal National ENT & Eastman Dental Hospitals, London, UK.; Scarpa B; Department of Statistical Sciences and Department of Mathematics Tullio Levi-Civita, University of Padova, Padova, Italy.; Kwame I; Department of ENT, Royal National ENT & Eastman Dental Hospitals, London, UK.; Ludwig DR; Rheumatology Department, University College London Hospitals, London, UK.; McAdoo S; Department of ENT, Imperial College Hospitals, London, UK.; Kuchai R; Department of ENT, Imperial College Hospitals, London, UK.; Gane S; Department of ENT, Royal National ENT & Eastman Dental Hospitals, London, UK.; Saleh H; Department of ENT, Imperial College Hospitals, London, UK.; Pusey CD; Department of ENT, Imperial College Hospitals, London, UK.; Renal Department, Imperial College Hospitals, London, UK.; Randhawa PS; Department of ENT, Royal National ENT & Eastman Dental Hospitals, London, UK.; Andrews PJ; Department of ENT, Royal National ENT & Eastman Dental Hospitals, London, UK.; Ear Institute, UCL, London, UK.
Source
Publisher: Wiley-Blackwell Country of Publication: United States NLM ID: 8607378 Publication Model: Print-Electronic Cited Medium: Internet ISSN: 1531-4995 (Electronic) Linking ISSN: 0023852X NLM ISO Abbreviation: Laryngoscope Subsets: MEDLINE
Subject
Language
English
Abstract
Objective: In this multicentric study involving three London hospitals, we compared ANCA-positive and ANCA-negative cocaine-induced midline destructive lesions (CIMDL) patients to assess how presence of antineutrophil cytoplasmic antibodies (ANCA) may correlate with disease severity. Our secondary aims are to better classify etiology centered around ANCA positivity and, consequently, better disease management.
Methods: A retrospective review was performed to identify patients with CIMDL seen between January 2019 and December 2022. Population data including age, sex, presentation, endoscopic findings, duration of cocaine use and active use of cocaine, type of treatment, laboratory (including ANCA serology), radiological, and histological findings were collected.
Results: Forty CIMDL patients (25 male, median age of 42 years) were identified. The majority of them (72.5%) presented with either a septal perforation, a saddle nose deformity (22.5%), and/or a palatal fistula (20.0%). ANCA was positive in 71.1% of cases (66.7% p-ANCA). No statistically significant differences in the general characteristics, type of treatment, laboratory results, radiological or histological findings were observed when comparing ANCA-positive and ANCA-negative CIMDL patients or when comparing p-ANCA and c-ANCA patients. Similarly, no statistically significant difference was obtained when comparing the pattern of distribution of lesions between the two groups.
Conclusions: A large percentage of CIMDL patients showed positive ANCA test (71.1%) and in the majority of the cases a p-ANCA pattern specifically targeting PR3 (p-ANCA, PR3 + MPO-). However, ANCA positivity or presence of a specific ANCA pattern was not associated with more severe presentation or more aggressive disease. Given its similarities to granulomatosis with polyangiitis (GPA), we recommend the use of the term "cocaine-induced ENT pseudo-GPA" instead of CIMDL.
Level of Evidence: IV Laryngoscope, 134:2609-2616, 2024.
(© 2023 The Authors. The Laryngoscope published by Wiley Periodicals LLC on behalf of The American Laryngological, Rhinological and Otological Society, Inc.)
Methods: A retrospective review was performed to identify patients with CIMDL seen between January 2019 and December 2022. Population data including age, sex, presentation, endoscopic findings, duration of cocaine use and active use of cocaine, type of treatment, laboratory (including ANCA serology), radiological, and histological findings were collected.
Results: Forty CIMDL patients (25 male, median age of 42 years) were identified. The majority of them (72.5%) presented with either a septal perforation, a saddle nose deformity (22.5%), and/or a palatal fistula (20.0%). ANCA was positive in 71.1% of cases (66.7% p-ANCA). No statistically significant differences in the general characteristics, type of treatment, laboratory results, radiological or histological findings were observed when comparing ANCA-positive and ANCA-negative CIMDL patients or when comparing p-ANCA and c-ANCA patients. Similarly, no statistically significant difference was obtained when comparing the pattern of distribution of lesions between the two groups.
Conclusions: A large percentage of CIMDL patients showed positive ANCA test (71.1%) and in the majority of the cases a p-ANCA pattern specifically targeting PR3 (p-ANCA, PR3 + MPO-). However, ANCA positivity or presence of a specific ANCA pattern was not associated with more severe presentation or more aggressive disease. Given its similarities to granulomatosis with polyangiitis (GPA), we recommend the use of the term "cocaine-induced ENT pseudo-GPA" instead of CIMDL.
Level of Evidence: IV Laryngoscope, 134:2609-2616, 2024.
(© 2023 The Authors. The Laryngoscope published by Wiley Periodicals LLC on behalf of The American Laryngological, Rhinological and Otological Society, Inc.)