Literature DB >> 28619741

Saddle-nose and bilateral cauliflower ear deformities with pyoderma gangrenosum-like ulcers, cavitary pulmonary lesions, digital gangrene and pulselessness in a young female.

Sweta Subhadarshani1, Vishal Gupta1, Anurag Chahal2, Kaushal K Verma1.   

Abstract

We report a young female who presented with saddle-nose and bilateral cauliflower ear deformities along with pyoderma gangrenosum-like ulcers, digital gangrene and pulselessness. Subsequently, she was found to have bilateral conductive hearing loss, a corneal opacity, mild aortic regurgitation and radiological evidence of cavitary changes in lungs and aortoarteritis. Our patient had a constellation of symptoms which posed a diagnostic challenge. Finally, a diagnosis of relapsing polychondritis with several unusual features was made. Overlap with Takayasu's arteritis and granulomatosis with polyangitis, which has been reported rarely in the literature, cannot be excluded. © BMJ Publishing Group Ltd (unless otherwise stated in the text of the article) 2017. All rights reserved. No commercial use is permitted unless otherwise expressly granted.

Entities:  

Keywords:  dermatology; ear, nose and throat/otolaryngology; rheumatology; vasculitis

Mesh:

Year:  2017        PMID: 28619741      PMCID: PMC5535111          DOI: 10.1136/bcr-2017-220434

Source DB:  PubMed          Journal:  BMJ Case Rep        ISSN: 1757-790X


  14 in total

1.  Antinuclear antibodies in relapsing polychondritis.

Authors:  J C Piette; R El-Rassi; Z Amoura
Journal:  Ann Rheum Dis       Date:  1999-10       Impact factor: 19.103

2.  Wegener's granulomatosis presenting as pyoderma gangrenosum.

Authors:  Hajnal Irén Szõcs; Katalin Torma; Edina Petrovicz; Judit Hársing; György Fekete; Sarolta Kárpáti; Attila Horváth
Journal:  Int J Dermatol       Date:  2003-11       Impact factor: 2.736

3.  The American College of Rheumatology 1990 criteria for the classification of Wegener's granulomatosis.

Authors:  R Y Leavitt; A S Fauci; D A Bloch; B A Michel; G G Hunder; W P Arend; L H Calabrese; J F Fries; J T Lie; R W Lightfoot
Journal:  Arthritis Rheum       Date:  1990-08

4.  Relapsing polychondritis or ANCA-negative Wegener's granulomatosis?

Authors:  Weekitt Kittisupamongkol; Wanla Kulwichit
Journal:  Med J Aust       Date:  2007-10-15       Impact factor: 7.738

Review 5.  Relapsing polychondritis: a review.

Authors:  Aman Sharma; Karthik Gnanapandithan; Kusum Sharma; Susmita Sharma
Journal:  Clin Rheumatol       Date:  2013-07-26       Impact factor: 2.980

6.  The American College of Rheumatology 1990 criteria for the classification of Takayasu arteritis.

Authors:  W P Arend; B A Michel; D A Bloch; G G Hunder; L H Calabrese; S M Edworthy; A S Fauci; R Y Leavitt; J T Lie; R W Lightfoot
Journal:  Arthritis Rheum       Date:  1990-08

Review 7.  Ocular and systemic findings in relapsing polychondritis.

Authors:  B L Isaak; T J Liesegang; C J Michet
Journal:  Ophthalmology       Date:  1986-05       Impact factor: 12.079

Review 8.  Active aortitis in relapsing polychondritis.

Authors:  A G Selim; L G Fulford; R H Mohiaddin; M N Sheppard
Journal:  J Clin Pathol       Date:  2001-11       Impact factor: 3.411

9.  Wegener's granulomatosis and relapsing polychondritis: a case report.

Authors:  P Small; M Black; M Davidman; M L de Champlain; M A Kapusta; H Kreisman
Journal:  J Rheumatol       Date:  1980 Nov-Dec       Impact factor: 4.666

10.  Pyoderma gangrenosum associated with Takayasu's arteritis responding to cyclosporin.

Authors:  L A Fearfield; J R Ross; A M Farrell; C Costello; C B Bunker; R C Staughton
Journal:  Br J Dermatol       Date:  1999-08       Impact factor: 9.302

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