Right ethmoid eosinophilic angiocentric fibrosis with orbital extension

Gorostis, S.; Bacha, Mohamed Mongi; Gravier, S.; Raguin, T.

doi:10.1016/j.anorl.2017.02.012

Cited by 13 publications

(10 citation statements)

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“…Seven cases 7,[12][13][14]18,19 (including one from the present paper) did not meet the initial entry criterion of characteristic organ involvement. Of the remaining seven cases, a further five cases 2,3,7,11,20 were noted to lack sufficient data, including serum IgG4 levels and/or IHC investigations for IgG4. One case 16 did not meet the classification criteria despite furnishing complete data.…”

Section: Discussionmentioning

confidence: 99%

“…1 Less commonly, involvement of the orbit, lung, and skin has also been noted. [2][3][4] Since the original description of this histological entity by Holmes and Panje 5 in 1983, various aetiologies have been proposed; these include a past medical history of atopy, previous injury to the lesional site (either traumatic or surgically related) and an association with granuloma faciale. 6 A landmark study by Deshpande et al 7 in 2011 hypothesised that EAF lies on the spectrum of immunoglobulin-G4 related disease (IgG4-RD), thereby changing the way in which many subsequent EAF cases have been approached.…”

Section: Introductionmentioning

confidence: 99%

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Eosinophilic angiocentric fibrosis and immunoglobulin 4‐related disease revisited

et al. 2022

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Eosinophilic angiocentric fibrosis (EAF) has been postulated to lie on the spectrum of IgG4‐related disease (IgG4‐RD) in a study reported in 2011. However, the aetiology of EAF remains controversial, despite increasing literature reporting IgG4‐RD targeted investigations in those studies. This review aims to formally evaluate the relationship between EAF and IgG4‐RD using the latest 2019 American College of Rheumatology/European League Against Rheumatism (ACR/EULAR) classification scheme for IgG4‐RD. A literature search was performed on the PubMed database for all studies describing patients diagnosed with EAF. Additionally, two EAF cases were retrospectively identified from our institution and presented. The demographics, clinical presentation, histological and serological data of each patient were analysed. A total of 20 cases were evaluated, including 18 patients from 14 articles in the English literature and two cases from our institution. Six of 20 (30%) patients with EAF met the criteria for IgG4‐RD with the 2019 ACR/EULAR classification scheme. This represents the first attempt at formally evaluating the relationship between EAF and IgG4‐RD using a validated set of classification criteria. Using the currently available data, a small proportion of patients with EAF met the criteria for IgG4‐RD. However, various limitations of this study suggest that further efforts at disease characterization of EAF may be required.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Eosinophilic angiocentric fibrosis and immunoglobulin 4‐related disease revisited

et al. 2022

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“…Upper Arms and Chest (n = 1) [13] IgG4 serum levels (normal, 8 to 140 mg/dL) Low (n = 1) [6] Normal (n = 3) [5,10,11] Elevated (n = 3) [8,14,24] Not available (n = 8) [2,9,[12][13][14] In the present case, in which the immunohistochemical tests for IgG4 + plasma cells were negative, it was crucial to rely on the conventional EAF histopathological features in order to avoid misdiagnoses and mistreatments. The evaluation of the presence of IgG4 + plasma cells is helpful to determine whether the lesion is responsive to glucocorticoid treatment and whether surgery can be avoided, since IgG4-RDs are extremely responsive to medical treatment, but it should not rule out EAF diagnosis when negative.…”

Section: Subglottis (N = 1) [12]mentioning

confidence: 99%

Eosinophilic Angiocentric Fibrosis of the Nasal Cavities: A Report of an Uncommon Lesion with Emphasis on the Etiology and Differential Diagnosis

Farina

Broggi

Federico³

et al. 2022

Medicina

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Background and Objectives: Eosinophilic angiocentric fibrosis (EAF) is an indolent but sometimes locally destructive lesion with a predilection for the sinonasal tract. Although it was first described in 1983, its etiology remains unknown. Some authors initially attributed EAF to trauma, hypersensitivity, and/or surgical manipulation, while it has been recently suggested to include EAF within the spectrum of IgG4-related systemic diseases. Materials and Methods: We report an uncommon case of idiopathic EAF in a 76-year-old male who developed two bilateral tumefactive masses in the nasal cavities. Results: As the histological examination showed a subepithelial proliferation of fibroblasts along with sclero-hyaline fibrosis around small-sized vessels (an “onion skin-like” pattern) and an eosinophils-rich inflammatory infiltrate, a diagnosis of EAF was rendered. The differential diagnosis included granuloma faciale, Wegener’s granulomatosis, and Churg–Strauss syndrome. Conclusions: Pathologists should be aware of the possibility that this lesion can be part of the wide spectrum of IgG4-related systemic diseases by performing IgG4 investigations to assess adherence to IgG4-related systemic disease criteria.

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Section: Diskussionunclassified

“…Es handelt sich um einen gleichzeitigen Befall der paranasalen Sinus und der Orbita. Es existieren wenige Fallberichte, die eine ähnliche, jedoch nicht identische Befundkonstellation beschreiben [ 14 , 15 ]. Inwieweit ältere Fallberichte zur eosinophilen angiozentrischen Fibrose den IgG4-assoziierten Erkrankungen der Nase und der Orbita zugeordnet werden müssten, ist im einzelnen Fall unklar.…”

Section: Diskussionunclassified

IgG4-assoziierte Orbitopathie als wichtige Differenzialdiagnose eines fortgeschrittenen Silent-Sinus-Syndroms

Jurkov

Klauschen

Bertelmann

et al. 2020

HNO

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Background Immunoglobulin (Ig)G4-related disease is classified as an immune-mediated disease. The etiology of this condition has not been explained to date. Manifestations of the disease are diverse, and simultaneous involvement of multiple organs is not unusual. Case report We report the case of a patient referred to us after multiple unsuccessful paranasal sinus operations who presented with enophthalmos and a resultant migratory keratitis with a suspected diagnosis of silent sinus syndrome. Preservation of the orbit was no longer feasible. After five years without a definitive diagnosis, we ascertained that this was a case of IgG4-related disease. Discussion IgG4-related disease represents an important element in the differential diagnosis of chronic advanced diseases of the orbit and paranasal sinuses. The diagnosis should be considered in the case of unclear disease presentations. Typical histological findings include a storiform pattern of fibrosis, vasculopathy, and tissue infiltration by IgG4 plasma cells.

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Right ethmoid eosinophilic angiocentric fibrosis with orbital extension

Abstract: The combination of surgery and rituximab achieved lasting pain relief with no recurrence of exophthalmos.

Cited by 13 publications

References 10 publications

Eosinophilic angiocentric fibrosis and immunoglobulin 4‐related disease revisited

Eosinophilic angiocentric fibrosis and immunoglobulin 4‐related disease revisited

Eosinophilic Angiocentric Fibrosis of the Nasal Cavities: A Report of an Uncommon Lesion with Emphasis on the Etiology and Differential Diagnosis

IgG4-assoziierte Orbitopathie als wichtige Differenzialdiagnose eines fortgeschrittenen Silent-Sinus-Syndroms

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