Reversible Cochlear Function With ANCA-Associated Vasculitis Initially Diagnosed by Otologic Symptoms

Yoshida, Naohiro; Hara, Mariko; Hasegawa, Masayo; Matsuzawa, Shingo; Shinnabe, Akihiro; Kanazawa, Hiromi; Iino, Yukiko

doi:10.1097/mao.0000000000000175

Cited by 24 publications

(29 citation statements)

References 30 publications

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“…21 Hearing loss has been reported in the clinical course of AAV, but only recently as an early-stage symptom. 10,11,[23][24][25][26][27][28][29][30][31][32][33] Table 1 shows the 41 OMAAV cases reported in the English literature that presented at an early phase or were initially diagnosed by otologic symptoms. Of these 41 cases, 12 were male and 29 were female, with ages ranging from 20 to 77 years, and 20 were PR3-ANCA positive and 21 were MPO-ANCA positive.…”

Section: Diagnostic Information Regarding Systemic and Localized Aavmentioning

confidence: 99%

“…Of these 41 cases, 12 were male and 29 were female, with ages ranging from 20 to 77 years, and 20 were PR3-ANCA positive and 21 were MPO-ANCA positive. In addition to these cases, the OMAAV working group in Japan reported 90 cases, including 8 cases reported previously, 33 that were preliminarily analyzed for the clinical features of OMAAV. 34 A total of 123 cases (35 male and 88 female, with ages ranging from 20 to 85 years) showed the clinical features of OMAAV.…”

Section: Diagnostic Information Regarding Systemic and Localized Aavmentioning

confidence: 99%

“…34 A total of 123 cases (35 male and 88 female, with ages ranging from 20 to 85 years) showed the clinical features of OMAAV. 10,11,[23][24][25][26][27][28][29][30][31][32][33][34] The diagnosis of GPA can be difficult and in most cases can only be provided by histological examination. Of the 123 cases, 16% were GPA positive by histological examination, without any systemic symptoms, and 87% were ANCA positive.…”

Section: Diagnostic Information Regarding Systemic and Localized Aavmentioning

confidence: 99%

“…or MPO-ANCA positive; 4) occasionally clinical complications such as facial palsy or hypertrophic pachymeningitis; 5) tympanic membrane showing a dull appearance similar to OME and vessel dilatation of tympanic membrane 'OME type', otitis media with granulation 'OMG type', or normal appearance only with sensorineural hearing loss; 6) effectiveness of corticosteroid and immunosuppressive therapy using cyclophosphamide or methotrexate. 10,11,20,[23][24][25][26][27][28][29][30][31][32][33][34] In most cases of gradual hearing loss due to effusion and granulation in the middle ear, treated as an intractable otitis media, sudden progressive hearing loss over less than 2 months followed. 10,11,20,[23][24][25][26][27][28][29][30][31][32][33][34] Antibiotics were not effective against the ear infection, and often bacterial examination of the otorrhea was negative, with only normal bacterial flora being detected in the middle ear.…”

Section: The Clinical Features Of Omaavmentioning

confidence: 99%

“…Several case reports have shown that ear condition progressed after myringotomy or mastoidectomy. 33,34,38 As the activation of neutrophils is necessary for generating ANCA, invasive surgical procedures or infection have the possibility of accelerating inflammation. After starting immunosuppressive therapy, ANCA titers rapidly returned to the normal range and hearing levels improved in 67% of ears.…”

Section: The Clinical Features Of Omaavmentioning

confidence: 99%

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Pathogenesis and Diagnosis of Otitis Media with ANCA-Associated Vasculitis

Yoshida

Iino

2014

Allergology International

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Antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV) is histologically characterized by systemic necrotizing vasculitis and is clinically classified into two phases, systemic or localized. Recently, otological symptoms such as otitis media and hearing loss, not previously often associated with AAV, have been reported in AAV cases. In these cases we propose a diagnosis of otitis media with AAV (OMAAV). The ANCA titer is important for the diagnosis of OMAAV, and in most cases rapid progressive hearing loss is observed as localized AAV. Peripheral facial nerve palsy or hypertrophic pachymeningitis are coupled with 25% of cases and 18% of cases respectively. Proteinase 3-ANCA (PR3-ANCA) positive otitis media causes granulomatous formation or middle ear effusion in the middle ear, on the other hand myeloperoxidase-ANCA (MPO-ANCA) positive otitis media predominantly presents as otitis media with effusion. The early diagnosed case and the sensorineural hearing loss not progressed deaf could be recovered by the immunosuppressive therapy. Delayed diagnosis of AAV occasionally leads to progression to the irreversible phase; therefore, diagnosis at the early-localized stage is important for treating AAV. In this review, we discuss the current understanding of this newly proposed concept of OMAAV.

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Section: Diagnostic Information Regarding Systemic and Localized Aavmentioning

confidence: 99%

Section: Diagnostic Information Regarding Systemic and Localized Aavmentioning

confidence: 99%

Section: Diagnostic Information Regarding Systemic and Localized Aavmentioning

confidence: 99%

Section: The Clinical Features Of Omaavmentioning

confidence: 99%

Section: The Clinical Features Of Omaavmentioning

confidence: 99%

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Pathogenesis and Diagnosis of Otitis Media with ANCA-Associated Vasculitis

Yoshida

Iino

2014

Allergology International

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Quantitative assessment of vestibular otopathology in granulomatosis with polyangitis: A temporal bone study

Nogaki

Keskin

Azuma

et al. 2018

Laryngoscope Investig Oto

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ObjectiveTo investigate the temporal bone histopathology of vasculitis, especially in the vestibular organs, in granulomatosis with polyangitis (GPA).MethodsUsing light and differential interference contrast microscopy, we examined 12 human temporal bones from six deceased GPA patients and 12 histopathologically normal human temporal bones from six deceased age‐matched patients.ResultsIn the GPA group, three patients had undergone tympanostomy tube placement. Two of them had suffered mixed hearing loss; one, sensorineural hearing loss; and one, conductive hearing loss. Of the 12 specimens in the GPA group, the granulation tissue invaded the round window niche in seven; cochlear hair cells were not preserved in five. Hemosiderin was deposited in the stria vascularis in eight specimens, in the ampulla or semicircular duct in 10, and in the vestibule in three. The spiral ligament showed severe loss of cellularity in two specimens. In the GPA group, type I vestibular hair cell density was significantly decreased; however, type II vestibular hair cell density did not significantly differ between the GPA group and the control group.ConclusionOur histopathologic findings in human temporal bone specimens of GPA patients delineated changes in the tympanic membrane, middle ear cavity, round window membrane, organ of Corti, stria vascularis, spiral ligament, ampulla, semicircular duct, and vestibule. Type I vestibular hair cell density significantly decreased in the GPA group, as compared with the control group.Level of EvidenceN/A

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Facial nerve paresis in the course of masked mastoiditis as a revelator of GPA

Marszał

Bartochowska

et al. 2021

Eur Arch Otorhinolaryngol

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Purpose The aim of this study was to present a series of 6 patients with facial nerve palsy and masked mastoiditis which constituted as revelators of localized granulomatosis with polyangiitis (GPA) and to evaluate the utility of the ACR/EULAR 2017 provisional classification criteria for GPA in such cases. Methods Study group included 58 patients with GPA. Cases with facial nerve palsy and masked mastoiditis were thoroughly analyzed. Results The mean age of patients was 37 years. All manifested unilateral facial nerve palsy and hearing loss, while only 2 reported aural complaints suggesting inflammatory cause of the disease. All cases were qualified for surgical intervention. Intraoperative findings were similar: granulation tissue in tympanic cavity and/or pneumatic spaces of the mastoid process. Only 50% of histopathological results suggested vasculitis. In all cases, elevated levels of antineutrophil cytoplasmic antibodies (ANCA) against peroxidase 3 (PR3-ANCA) were determined. Two patients presented rapid progression of the disease and died within 1 week and 2 months, respectively. Four other patients manifested gradual improvement of hearing and facial nerve function after treatment. Conclusion GPA should be included into differential diagnosis in all cases of persistent facial nerve palsy especially when otological symptoms coexist. Even localized GPA could be very aggressive, revelating generalized form of the disease. Rapid systemic treatment of GPA can protect hearing and facial nerve from permanent severe dysfunction. The ACR/EULAR 2017 provisional classification criteria for GPA seem to be valuable tool in diagnosing ENT patients with localized otological form of the disease.

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Reversible Cochlear Function With ANCA-Associated Vasculitis Initially Diagnosed by Otologic Symptoms

Cited by 24 publications

References 30 publications

Pathogenesis and Diagnosis of Otitis Media with ANCA-Associated Vasculitis

Pathogenesis and Diagnosis of Otitis Media with ANCA-Associated Vasculitis

Quantitative assessment of vestibular otopathology in granulomatosis with polyangitis: A temporal bone study

Facial nerve paresis in the course of masked mastoiditis as a revelator of GPA

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