Laryngotracheal Involvement as the Initial Manifestation of Relapsing Polychondritis

Chang, Shu–Jen; Lu, Chunliang; Chung, Yu-Mei; Lee, Shinn-Shing; Chou, Chung‐Tei; Huang, Der-Chen

doi:10.1016/s1726-4901(09)70151-0

Cited by 24 publications

(12 citation statements)

References 9 publications

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“…Involvement of the tracheobronchial tree has been reported to be present in approximately half of all patients in western series [1,18]. The incidence of laryngeal, tracheal or bronchial involvement was 12% in our patients contrasting with the higher rates reported in other populations.…”

Section: Discussioncontrasting

confidence: 70%

Relapsing polychondritis: clinical presentations, disease activity and outcomes

Sharma

Law

Bambery

et al. 2014

Orphanet J Rare Dis

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BackgroundRelapsing polychondritis is a rare disease characterised by inflammation of cartilaginous and proteoglycan rich structures. As there are only a few published single centre case series from all across the world, we describe our experience with 26 patients at a tertiary centre in north India.MethodsA retrospective study with all patients meeting Damiani and Levine’s modification of McAdam’s diagnostic criteria. Clinical details, investigations, disease activity assessment [(Relapsing Polychondritis Disease Activity Index (RPDAI)], treatment and outcomes were recorded.ResultsTen men and sixteen women (median age 45 years) met the diagnostic criteria. Auricular chondritis (96%), arthritis (54%), hearing impairment (42%), ocular (42%), dermal (26%), cardiovascular (11%) and laryngotracheal involvement (11%) characterized the clinical presentations. The median RPDAI was 31 (range 9-66). Two patients died during observation. Overall survival was 92.3% (median survival 13.5 years).ConclusionsApart from reduced laryngotracheal involvement, RP in India was clinically similar to recorded patterns elsewhere.

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Section: Discussioncontrasting

confidence: 70%

Relapsing polychondritis: clinical presentations, disease activity and outcomes

Sharma

Law

Bambery

et al. 2014

Orphanet J Rare Dis

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Section: Discussioncontrasting

confidence: 73%

Relapsing polychondritis: Clinical presentations, disease activity and outcomes

Sharma

Law

Bambery

et al. 2014

Indian Journal of Rheumatology

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Background: Relapsing polychondritis is a rare disease characterised by inflammation of cartilaginous and proteoglycan rich structures. As there are only a few published single centre case series from all across the world, we describe our experience with 26 patients at a tertiary centre in north India. Methods: A retrospective study with all patients meeting Damiani and Levine's modification of McAdam's diagnostic criteria. Clinical details, investigations, disease activity assessment [(Relapsing Polychondritis Disease Activity Index (RPDAI)], treatment and outcomes were recorded. Results: Ten men and sixteen women (median age 45 years) met the diagnostic criteria. Auricular chondritis (96%), arthritis (54%), hearing impairment (42%), ocular (42%), dermal (26%), cardiovascular (11%) and laryngotracheal involvement (11%) characterized the clinical presentations. The median RPDAI was 31 (range 9-66). Two patients died during observation. Overall survival was 92.3% (median survival 13.5 years).

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“…Other chondritis and bilateral scleritis developed 3 months after tracheostomy. Chang et al described the same features leading to tracheostomy but the patient's upper airway was completely collapsed [22]. In fact, tracheostomy was once the most likely surgical procedure in RP.…”

Section: Discussionmentioning

confidence: 95%

Stenosis of the Subglottic Trachea Revealing Relapsing Polychondritis of a Young Adult

Euch¹,

Mahfoudhi²,

Gorsane³

et al. 2015

IJCM

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Relapsing polychondritis is a rare inflammatory disease involving essentially cartilaginous structures. Other systemic manifestations can be encountered as the eye and ear disturbance. Aortic aneurysms affect few cases. If affection of airway cartilages occurs, prognosis may be worsened by eventual stenotic lesions. We report a 22-year-old woman with Relapsing polychondritis. When she was referred to our hospital 4 weeks after the onset of respiratory symptoms, she was having severe breathing difficulty. Immediate tracheostomy followed by steroid therapy improved her respiratory condition, although the treatment was complicated because of her diabetes. While airway involvement of Relapsing polychondritis can be life threatening, it is curable with steroid therapy. Clinicians should keep in mind that airway obstruction could be caused by this disease.

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Laryngotracheal Involvement as the Initial Manifestation of Relapsing Polychondritis

Cited by 24 publications

References 9 publications

Relapsing polychondritis: clinical presentations, disease activity and outcomes

Relapsing polychondritis: clinical presentations, disease activity and outcomes

Relapsing polychondritis: Clinical presentations, disease activity and outcomes

Stenosis of the Subglottic Trachea Revealing Relapsing Polychondritis of a Young Adult

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