Neuro-laryngeal involvement in Churg-Strauss syndrome

Mazzantini, M; Fattori, Bruno; Matteucci, Francesco; Gaeta, P.; Ursino, Francesco

doi:10.1007/s004050050064

Cited by 19 publications

(15 citation statements)

References 8 publications

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“…Interestingly, she is one of the few known cases of CSS primarily presenting with laryngeal symptoms and with laryngeal pathology. In a previous case reported by Mazzantini et al the patient's first clinical manifestation was a persistent dysphonia 6. A videolaryn gostroboscopic examination revealed paresis of the right vocal fold with a reduction in adduction together with incomplete glottal closure.…”

Section: Discussionmentioning

confidence: 70%

A laryngeal presentation of Churg--Strauss syndrome in childhood

Alammar

Yasin

Al‐Muhsen

et al. 2009

Annals of Saudi Medicine

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A 10- year-old female, known to have bronchial asthma, presented with an unusual laryngeal lesion, eventually diagnosed as Churg-Strauss syndrome (CSS). She was referred to our hospital with history of recurrent stridor. On endoscopyhe, the larynx showed signs similar to recurrent respiratory papillomatosis (RRP). CSS is a systemic disorder and is now defined as one of the ANCA (antineutrophil cytoplasmic antibodies)-associated vasculitides. CSS is a systemic disease that may involve unusual sites like the larynx. Such an unusual presenatation of CSS should be kept in mind, especially in patients with history of asthma.

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Section: Discussionmentioning

confidence: 70%

A laryngeal presentation of Churg--Strauss syndrome in childhood

Alammar

Yasin

Al‐Muhsen

et al. 2009

Annals of Saudi Medicine

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“…The disease did not evolve into systemic form 7 . Vocal cord paralysis has also been reported in polyarteritis nodosa, Behcet's disease, rheumatoid arthritis and eosinophilic granulomatosis with polyangiitis, although never as an initial isolated manifestation 8,9 . In these cases, as in ours, glucocorticoid administration improved vocal cord function.…”

Section: Discussionmentioning

confidence: 98%

Severe microscopic polyangiitis with unilateral vocal cord paralysis as initial manifestation

2017

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Case Description: A 16 year-old female who presented with initial ear, nose and throat manifestations who later progressed to severe renal disease, requiring hemodialysis after 11 months of unique laryngeal involvement. Clinical Findings: Unilateral vocal cord paralysis without other symptoms or signs, but with positive perinuclear anti-neutrophil cytoplasmic antibodies (ANCA) and anti-myeloperoxidase autoantibodies, followed an unfavorable course months later with rapidly progressive glomerulonephritis. Renal biopsy confirmed an ANCA-associated vasculitis. She was diagnosed with microscopic polyangiitis. Treatment and Outcome: High-dose glucocorticoids, intravenous cyclophosphamide, plasma exchange and finally, hemodialysis and renal transplantation. Clinical Relevance: In contrast to granulomatosis with polyangiitis (Wegener), ear, nose and throat manifestations in microscopic polyangiitis are uncommon, while involvement of the lungs and kidneys are usual. We present a case with an isolated rare involvement, which progressed to severe disease. This atypical case warns about laryngeal symptoms as initial manifestation of an anti-myeloperoxidase positive systemic vasculitides, and emphasizes the relevance of close observation when unexplained isolated conditions with accompanying evidence of autoimmunity, in this case high levels of specific autoantibodies, are present.

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“…In contrast to the frequent involvement of peripheral nerves, cranial neuropathy is quite rare in CSS and only a few cases of optic, oculomotor, and facial neuropathy have been reported [ 4 , 5 , 6 ]. Moreover, lower cranial nerve palsy in CSS is extremely rare [ 7 , 8 ]. In contrast to past reports, our case had more debilitating dysphagia that resulted in recurrent aspiration pneumonia and tube feeding.…”

Section: Discussionmentioning

confidence: 99%

Churg-Strauss Syndrome as an Unusual Cause of Dysphagia: Case Report

Park

Moon

et al. 2015

Ann Rehabil Med

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Systemic vasculitis is a rare disease, and the diagnosis is very difficult when patient shows atypical symptoms. We experienced an unusual case of dysphagia caused by Churg-Strauss syndrome with lower cranial nerve involvement. A 74-year-old man, with a past history of sinusitis, asthma, and hearing deficiency, was admitted to our department for evaluation of dysphagia. He also complained of recurrent bleeding of nasal cavities and esophagus. Brain magnetic resonance imaging did not show definite abnormality, and electrophysiologic findings were suggestive of mononeuritis multiplex. Dysphagia had not improved after conventional therapy. Biopsy of the nasal cavity showed extravascular eosinophilic infiltration. All these findings suggested a rare form of Churg-Strauss syndrome involving multiple lower cranial nerves. Dysphagia improved after steroid therapy.

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Neuro-laryngeal involvement in Churg-Strauss syndrome

Cited by 19 publications

References 8 publications

A laryngeal presentation of Churg--Strauss syndrome in childhood

A laryngeal presentation of Churg--Strauss syndrome in childhood

Severe microscopic polyangiitis with unilateral vocal cord paralysis as initial manifestation

Churg-Strauss Syndrome as an Unusual Cause of Dysphagia: Case Report

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