Refractory Otitis Media: An Unusual Presentation of Childhood Granulomatosis With Polyangiitis

被引:2
作者
Uppal, Preena [1 ,2 ]
Taitz, Jonny [1 ,2 ]
Wainstein, Brynn [2 ,3 ]
Soma, Marlene [2 ,4 ]
Belessis, Yvonne [2 ,5 ]
Gray, Paul [2 ,3 ]
机构
[1] Sydney Childrens Hosp Network, Dept Gen Pediat, Randwick, NSW, Australia
[2] Univ New S Wales, Sch Womens & Childrens Hlth, Sydney, NSW 2052, Australia
[3] Sydney Childrens Hosp Network, Dept Immunol, Randwick, NSW, Australia
[4] Sydney Childrens Hosp Network, Dept Ear Nose & Throat Surg, Randwick, NSW, Australia
[5] Sydney Childrens Hosp Network, Dept Resp Med, Randwick, NSW, Australia
关键词
Wegener's granulomatosis; chronic otitis media; hearing loss; atypical pneumonia; childhood vasculitis; ANCA-ASSOCIATED VASCULITIS; HENOCH-SCHONLEIN PURPURA; WEGENERS-GRANULOMATOSIS; CHILDREN; YOUNG;
D O I
10.1002/ppul.22746
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
Childhood granulomatosis with polyangiitis (cGPA), previously known as Wegener's granulomatosis, is a rare, potentially fatal necrotizing vasculitis, the symptoms of which overlap with infection. We present a 16-year-old girl who, following 6 months of treatment for persistent middle ear effusion with progressive sensorineural hearing loss, developed rapidly progressing pneumonia, with pleural effusion, and multiple cavitatory lung lesions. Investigations demonstrated high titer c-ANCA and nasal septal biopsy confirmed the diagnosis of cGPA. This case highlights the difficulty in diagnosing cGPA and the potentially life-threatening consequences of failing to do so. Pediatr Pulmonol. 2014; 49:E21-E24. (c) 2013 Wiley Periodicals, Inc.
引用
收藏
页码:E21 / E24
页数:4
相关论文
共 11 条
[1]   Clinical features and outcome of pediatric Wegener's granulomatosis [J].
Akikusa, J. D. ;
Schneider, R. ;
Harvey, E. A. ;
Hebert, D. ;
Thorner, P. S. ;
Laxer, R. M. ;
Silverman, E. D. .
ARTHRITIS & RHEUMATISM-ARTHRITIS CARE & RESEARCH, 2007, 57 (05) :837-844
[2]   Long- term outcome of paediatric patients with ANCA vasculitis [J].
Arulkumaran, Nishkantha ;
Jawad, Susan ;
Smith, Stuart W. ;
Harper, Lorraine ;
Brogan, Paul ;
Pusey, Charles D. ;
Salama, Alan D. .
PEDIATRIC RHEUMATOLOGY, 2011, 9
[3]   Classification, Presentation, and Initial Treatment of Wegener's Granulomatosis in Childhood [J].
Cabral, David A. ;
Uribe, America G. ;
Benseler, Susanne ;
O'Neil, Kathleen M. ;
Hashkes, Philip J. ;
Higgins, Gloria ;
Zeft, Andrew S. ;
Lovell, Daniel J. ;
Kingsbury, Daniel J. ;
Stevens, Anne ;
McCurdy, Deborah ;
Chira, Peter ;
Abramson, Leslie ;
Arkachaisri, Thaschawee ;
Campillo, Sarah ;
Eberhard, Anne ;
Hersh, Aimee O. ;
Huber, Adam M. ;
Kim, Susan ;
Klein-Gitelman, Marisa ;
Levy, Deborah M. ;
Li, Suzanne C. ;
Mason, Thomas ;
DeWitt, Esi Morgan ;
Muscal, Eyal ;
Nassi, Lorien ;
Reiff, Andreas ;
Schikler, Kenneth ;
Singer, Nora G. ;
Wahezi, Dawn ;
Woodward, Amy .
ARTHRITIS AND RHEUMATISM, 2009, 60 (11) :3413-3424
[4]   Biologic therapy in primary systemic vasculitis of the young [J].
Eleftheriou, Despina ;
Melo, Marianna ;
Marks, Stephen D. ;
Tullus, Kjell ;
Sills, John ;
Cleary, Gavin ;
Dolezalova, Pavla ;
Ozen, Seza ;
Pilkington, Clarissa ;
Woo, Pat ;
Klein, Nigel ;
Dillon, Michael J. ;
Brogan, Paul A. .
RHEUMATOLOGY, 2009, 48 (08) :978-986
[5]   Airway manifestations in childhood granulomatosis with polyangiitis (Wegener's) [J].
Fowler, Nicole M. ;
Beach, Jocelyn M. ;
Krakovitz, Paul ;
Spalding, Steven J. .
ARTHRITIS CARE & RESEARCH, 2012, 64 (03) :434-440
[6]   Incidence of Henoch-Schonlein purpura, Kawasaki disease, and rare vasculitides in children of different ethnic origins [J].
Gardner-Medwin, JMM ;
Dolezalova, P ;
Cummins, C ;
Southwood, TR .
LANCET, 2002, 360 (9341) :1197-1202
[7]   Incidence of Wegener's Granulomatosis in Children [J].
Grisaru, Silviu ;
Yuen, Gilbert W. H. ;
Miettunen, Paivi M. ;
Hamiwka, Lorraine A. .
JOURNAL OF RHEUMATOLOGY, 2010, 37 (02) :440-442
[8]   EULAR/PRINTO/PRES criteria for Henoch-Schonlein purpura, childhood polyarteritis nodosa, childhood Wegener granulomatosis and childhood Takayasu arteritis: Ankara 2008. Part I: Overall methodology and clinical characterisation [J].
Ruperto, Nicolino ;
Ozen, Seza ;
Pistorio, Angela ;
Dolezalova, Pavla ;
Brogan, Paul ;
Cabral, David A. ;
Cuttica, Ruben ;
Khubchandani, Raju ;
Lovell, Daniel J. ;
O'Neil, Kathleen M. ;
Quartier, Pierre ;
Ravelli, Angelo ;
Iusan, Silvia M. ;
Filocamo, Giovanni ;
Magalhaes, Claudia Saad ;
Unsal, Erbil ;
Oliveira, Sheila ;
Bracaglia, Claudia ;
Bagga, Arvind ;
Stanevicha, Valda ;
Manzoni, Silvia Magni ;
Pratsidou, Polyxeni ;
Lepore, Loredana ;
Espada, Graciela ;
Paut, Isabelle Kone ;
Zulian, Francesco ;
Barone, Patrizia ;
Bircan, Zelal ;
del Rocio Maldonado, Maria ;
Russo, Ricardo ;
Vilca, Iris ;
Tullus, Kjell ;
Cimaz, Rolando ;
Horneff, Gerd ;
Anton, Jordi ;
Garay, Stella ;
Nielsen, Susan ;
Barbano, Giancarlo ;
Martini, Alberto .
ANNALS OF THE RHEUMATIC DISEASES, 2010, 69 (05) :790-797
[9]   Wegener granulomatosis in children and young adults - A case study of ten patients [J].
Stegmayr, BG ;
Gothefors, L ;
Malmer, B ;
Wiefel, DEM ;
Nilsson, K ;
Sundelin, B .
PEDIATRIC NEPHROLOGY, 2000, 14 (03) :208-213
[10]   Pathogenesis of ANCA-associated vasculitis: recent insights from animal models [J].
van Timmeren, Mirjan M. ;
Heeringa, Peter .
CURRENT OPINION IN RHEUMATOLOGY, 2012, 24 (01) :8-14