Two clinical cases of granulomatosis with polyangiitis with isolated otitis media and mastoiditis

Kukushev, Georgi; Kalinova, Desislava; Sheytanov, Ivan; Rashkov, Rasho

doi:10.5114/reum.2017.71643

Cited by 9 publications

(7 citation statements)

References 8 publications

Supporting

Mentioning

Contrasting

Unclassified

Order By: Relevance

“…[ 18 ] DI in 7/9, 7/9 hypogonadism, 5/9 TSH deficiency, 4/9 hyperprolactemia, 2/9 GH deficiency, 1/9 ACTH deficiency Pituitary disease diagnosed after GPA in 8/9 patients at median of 58.5 months, concomitant in one case Bando et al. [ 51 ] DI, GH deficiency Chronic sinusitis, COM, auditory disturbance when 34 years of age. With 38 years of age, nasal stiffness, fatigue, appetite loss, saddle-nose deformity.…”

Section: Discussionmentioning

confidence: 99%

“…Among the reviewed articles, only one report matched our patient’s complaints of therapy-resistant otitis media, facial nerve paralysis, sensorineural hearing loss, and DI [ 42 ]. Although GPA can occur as a very limited localized disease, e.g., as antibiotic-resistant otitis media with or without mastoiditis [ 51 – 53 ], we want to highlight that GPA can also occur with a combination of involved localized areas, as described herein. The current case, as well as the cases reviewed, highlight the complexity of this disease.…”

Section: Discussionmentioning

confidence: 99%

See 1 more Smart Citation

Granulomatosis with polyangiitis in a patient with polydipsia, facial nerve paralysis, and severe otologic complaints: a case report and review of the literature

et al. 2022

View full text Add to dashboard Cite

Background Granulomatosis with polyangiitis, formerly known as Wegener granulomatosis, is a necrotizing vasculitis with granulomatous inflammation that belongs to the class of antineutrophil cytoplasmic antibodies-positive diseases. It occurs in a localized and a systemic form and may present with a variety of symptoms. Involvement of the upper respiratory tract is very common, while neurologic, endocrinological, and nephrological dysfunction may occur. Case presentation We describe the case of a 29-year-old Central European male patient presenting with severe bilateral sensorineural hearing loss, otorrhea, and one-sided facial nerve paralysis. The patient was unsuccessfully treated with i.v. antibiotics at another hospital in Berlin, and tympanic tubes were inserted. After presentation to our emergency room, he was hospitalized and further diagnostics started. Increased fluid intake and 12 kg weight gain over the last months were reported. The patient was diagnosed with granulomatosis with polyangiitis and diabetes insipidus. The patient’s condition improved after treatment with rituximab. Discussion A comprehensive PubMed search of all articles with granulomatosis with polyangiitis and diabetes insipidus was conducted to assess which combination of symptoms occurs simultaneously and whether other parts of the pituitary are commonly involved. The 39 selected articles, describing 61 patients, showed that ear–nose–throat involvement occurred most commonly, in 71% of cases. Of patients, 59% had involvement of the anterior pituitary gland, while true panhypopituitarism occurred in 13% of cases. Only one case report featured the same set of symptoms as described herein. Conclusion Granulomatosis with polyangiitis is a highly variable disease, commonly involving the upper airways, but that may present with symptoms solely related to the pituitary gland. Clinicians should have a low threshold to investigate for granulomatosis with polyangiitis in patients with therapy-resistant otorrhea. Patients may present with a complex set of symptoms, and integrating different specialists when additional symptoms occur may lead to faster diagnosis.

show abstract

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Granulomatosis with polyangiitis in a patient with polydipsia, facial nerve paralysis, and severe otologic complaints: a case report and review of the literature

et al. 2022

View full text Add to dashboard Cite

show abstract

“…Subepithelial tissues can appear fibrotic, or as edematous granulation tissue, and acute and chronic lymphoplasmacytic inflammation is present. Giant cells and granuloma formation have been described but are not required features [45]. 'Blue necrosis' (geographic tissue necrosis) or collagenous necrosis is often seen (Fig.…”

Section: Histopathologic Findingsmentioning

confidence: 99%

Idiopathic, Infectious and Reactive Lesions of the Ear and Temporal Bone

Magliocca

Vivas

Griffith

2018

Head and Neck Pathol

View full text Add to dashboard Cite

A number of infectious, inflammatory and idiopathic lesions develop within otologic tissues that may share similar clinical and/or microscopic features. This review first provides a working classification for otitis externa, and then otitis media and includes two recently described entities, eosinophilic otitis media and otitis media with ANCA-associated vasculitis. Next, the microscopic findings of a spectrum of otopathologic conditions are described, including post-inflammatory conditions such as tympanosclerosis and aural polyps, an overview of animate aural foreign body as well as iatrogenic aural foreign body reactions. Finally, a review of fungal disease affecting the ear with a brief synopsis of Candida auris, a recently described and virulent organism, is presented.

show abstract

“…In the literature of the last 20 years, we have not found the descriptions of GPA cases manifesting as facial palsy and masked mastoiditis. There are several articles presenting patients with unilateral or bilateral paresis of the VIIth nerve, in most cases presenting as a sole initial symptom or as a component or complication of acute of chronic otitis media [26][27][28]. A wide and interesting review of such cases is presented by Iannella et al [4].…”

Section: Discussionmentioning

confidence: 99%

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

Marszał

Bartochowska

et al. 2021

Eur Arch Otorhinolaryngol

View full text Add to dashboard Cite

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.

show abstract

Two clinical cases of granulomatosis with polyangiitis with isolated otitis media and mastoiditis

Cited by 9 publications

References 8 publications

Granulomatosis with polyangiitis in a patient with polydipsia, facial nerve paralysis, and severe otologic complaints: a case report and review of the literature

Granulomatosis with polyangiitis in a patient with polydipsia, facial nerve paralysis, and severe otologic complaints: a case report and review of the literature

Idiopathic, Infectious and Reactive Lesions of the Ear and Temporal Bone

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

Contact Info

Product

Resources

About