Morbidity and mortality associated with subglottic laryngotracheal stenosis in granulomatosis with polyangiitis (Wegener's granulomatosis): a single-centre experience in the United Kingdom

Jordan, Natasha; Verma, Hema; Siddiqui, Ata; Morrison, G. A. J.; D’Cruz, David

doi:10.1017/s0022215114001650

Cited by 11 publications

(22 citation statements)

References 20 publications

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“…Subglottic stenosis management requires a multidisciplinary approach, with both rheumatological and otolaryngological expertise involved 25 , 26 . One of the largest cohorts of granulomatosis with polyangiitis patients was presented by Gluth et al 14 .…”

Section: Discussionmentioning

confidence: 99%

The efficacy of submucosal corticosteroid injection and dilatation in subglottic stenosis of different aetiology

et al. 2016

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Objective:To determine the long-term efficacy of submucosal corticosteroid injection plus dilatation for subglottic stenosis as a single modality treatment in granulomatosis with polyangiitis and relapsing polychondritis, as compared with idiopathic subglottic stenosis and traumatic subglottic stenosis.Method:Patients who underwent dilatation for autoimmune causes were identified. Corticosteroid injection into the submucosa of a stenotic segment was followed by serial dilatation. Definitive improvement was defined as good airway patency for more than 24 months with no further procedures needed. Clinical, demographic and procedural data were recorded.Results:Patients (n = 45) were divided into three subglottic stenosis groups: traumatic (n = 24), idiopathic (n = 9) and autoimmune (n = 12). Patients were treated with dilatations, with a median follow-up time of 76 months. Six patients were tracheostomy-dependent. There were no statistical differences in the number of final improvements between autoimmune, idiopathic and traumatic groups, with values of 75, 56 and 71 per cent, respectively. There was no statistical difference between granulomatosis with polyangiitis plus relapsing polychondritis and idiopathic subglottic stenosis in terms of decannulation rates.Conclusion:Granulomatosis with polyangiitis and relapsing polychondritis patients have better improvement rates than patients with other subglottic stenosis types.

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Section: Discussionmentioning

confidence: 99%

The efficacy of submucosal corticosteroid injection and dilatation in subglottic stenosis of different aetiology

et al. 2016

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“…In the presence of subglottic or tracheal involvement, surgical intervention is often needed to maintain a patent airway [101–103]. …”

Section: Treatmentmentioning

confidence: 99%

Lung involvement in childhood onset granulomatosis with polyangiitis

et al. 2017

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Granulomatosis with polyangiitis is an ANCA-associated systemic vasculitis with a low incidence in the pediatric population. Lung involvement is a common manifestation in children affected by granulomatosis with polyangiitis, both at disease’s onset and during flares. Its severity is variable, ranging from asymptomatic pulmonary lesions to dramatic life-threatening clinical presentations such as diffuse alveolar haemorrhage. Several radiologic findings have been described, but the most frequent abnormalities detected are nodular lesions and fixed infiltrates. Interstitial involvement, pleural disease and pulmonary embolism are less common. Histology may show necrotizing or granulomatous vasculitis of small arteries and veins of the lung, but since typical features may be patchy, the site for lung biopsy should be carefully chosen with the help of imaging techniques such as computed tomography. Bronchoalveolar lavage is helpful to confirm the diagnosis of alveolar haemorrhage. Pulmonary function tests are frequently altered, showing a reduction in the diffusion capacity for carbon monoxide, which can be associated with obstructive abnormalities related to airway stenosis. Nodular lung lesions tend to regress with immunosuppressive therapy, but lung disease may also require second line treatments such as plasmapheresis. In cases of massive diffuse alveolar haemorrhage, ventilator support is crucial in the management of the patient.

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“…An interdisciplinary therapeutic approach (pneumology, rheumatology, nephrology etc.) is needed to achieve the best long-term outcome for the patient and to guide a lifelong follow-up [ 15 ].…”

Section: Discussionmentioning

confidence: 99%

“Exercise induced asthma” is not always asthma

Kofler

Daikeler

Prince

et al. 2018

Respiratory Medicine Case Reports

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A 25 year old woman was referred to our center for further evaluation of an exercise-induced dyspnea. Moreover, the patient suffered from hoarseness and recurrent sinusitis and otitis.After initially finding nothing suspicious, a spiro-ergometry was performed. Interestingly, we saw a relevant limitation of the inspiratory flow-volume curve under maximal exercise load. Further evaluation (in particular the bronchoscopy and the resulting biopsies) led us to the final diagnosis of a granulomatosis with polyangiitis.After 4 weeks of an established therapy regime with prednisone and rituximab the prior detected subglottic stenosis and the inspiratory flow-volume curve limitation could no longer detected.We describe a rare differential diagnosis of an exercise-induced asthma and we underline the importance of a multimodal therapy concept. We highlight the critical nature of the flow-volume curve in spiro-ergometry under maximal exercise load. We recommend frequent follow-up control visits to monitor the subglottic stenosis.

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Morbidity and mortality associated with subglottic laryngotracheal stenosis in granulomatosis with polyangiitis (Wegener's granulomatosis): a single-centre experience in the United Kingdom

Cited by 11 publications

References 20 publications

The efficacy of submucosal corticosteroid injection and dilatation in subglottic stenosis of different aetiology

The efficacy of submucosal corticosteroid injection and dilatation in subglottic stenosis of different aetiology

Lung involvement in childhood onset granulomatosis with polyangiitis

“Exercise induced asthma” is not always asthma

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