Head and neck immunoglobulin G4 related disease: systematic review

2018 ◽  
Vol 132 (12) ◽  
pp. 1046-1050 ◽  
Author(s):  
G Tirelli ◽  
N Gardenal ◽  
A Gatto ◽  
E Quatela ◽  
G C Del Piero

AbstractBackgroundImmunoglobulin G4 related disease is a recently described systemic syndrome. The head and neck region is the second most common site for presentation after the pancreas.MethodsPubMed and the Cochrane Library were searched from 1995 to July 2017 for all the studies on immunoglobulin G4 related disease diagnosed in the head and neck compartment. Patient-specific data were extracted and basic statistical analysis was performed.ResultsNinety-one patients were identified. Treatment was specified in 76 patients. Twenty patients received surgical treatment, eight of them in association with medical therapy. Fifty-six patients received medical treatment. The disease recurred in 25 per cent of patients treated with surgical treatment alone, in 3.6 per cent of patients treated with medical treatment alone and in 12.5 per cent of patients treated with both. All medical treatment protocols contained high-dose corticosteroids.ConclusionEarly and correct diagnosis can avoid unnecessary surgical treatment, and glucocorticoid therapy can improve the long-term prognosis.

2015 ◽  
Vol 129 (3) ◽  
pp. 226-231 ◽  
Author(s):  
E L Culver ◽  
A Hunt ◽  
E Crewe ◽  
K A Shah ◽  
P Martinez-Devesa

AbstractBackground:ENT surgeons may be the first specialists to encounter and diagnose patients with salivary gland disease. A new entity involving the salivary glands has recently been described of which ENT surgeons need to be aware: immunoglobulin G4 related chronic sclerosing sialadenitis.Method:A literature search of Medline, Embase and Cochrane Library databases was performed, using the search terms ‘IgG4’, ‘hyperIgG4 syndrome’ and ‘IgG4 related chronic sclerosing sialadenitis’.Results:Knowledge concerning immunoglobulin G4 related chronic sclerosing sialadenitis is rapidly increasing. This new entity is part of a fibro-inflammatory corticosteroid-responsive systemic disease (immunoglobulin G4 related disease) and has been described in almost every organ. Biopsy of the submandibular gland can be diagnostic. However, the diagnosis can easily be overlooked if: clinical suspicion is not high, one is unaware of the classical morphology and/or immunoglobulin G4 staining is not performed. This paper presents a summary of the current understanding of the disease and its management.Conclusion:ENT surgeons should be aware of this new disease entity. Patients with systemic disease should be managed under a multidisciplinary team, with input from clinicians who have an interest in such diseases (such as gastroenterologists and rheumatologists), and input from histopathologists and radiologists.


2018 ◽  
Vol 142 (12) ◽  
pp. 1560-1563
Author(s):  
Janice Ahn ◽  
Melina Flanagan

Eosinophilic angiocentric fibrosis is a rare indolent lesion of the head and neck region that has characteristic histologic findings of onionskin fibrosis and prominent eosinophils. Its pathogenesis has been poorly understood and has been most commonly attributed to hypersensitivity or previous trauma. Recently, the lesion has been included in the spectrum of immunoglobulin G4 (IgG4)–related disease. However, few of the existing cases of eosinophilic angiocentric fibrosis have been evaluated for IgG4+ and IgG+ plasma cells. Therefore, we provide an update on the clinical and histologic features of eosinophilic angiocentric fibrosis to increase awareness of the entity and encourage its further characterization as an IgG4-related disease.


Pancreas ◽  
2019 ◽  
Vol 48 (6) ◽  
pp. 799-804 ◽  
Author(s):  
Sonmoon Mohapatra ◽  
Ayush Sharma ◽  
Suresh T. Chari

2014 ◽  
Vol 128 (S2) ◽  
pp. S10-S15 ◽  
Author(s):  
W K Wong ◽  
R Campbell ◽  
R Douglas

AbstractBackground:Immunoglobulin G4 related disease is an inflammatory condition characterised by the presence of fibrotic lesions infiltrated by immunoglobulin G4 positive plasma cells. It can arise from almost any region of the body and it is being increasingly recognised in the head and neck. Regardless of the site of involvement, the histopathological resemblance is remarkable. Dense lymphoplasmacytic infiltration, overabundance of immunoglobulin G4 bearing plasma cells and presence of storiform fibrosis are typical findings.Case reports:This paper presents two cases of immunoglobulin G4 related disease in which there was involvement of the orbit, the infraorbital nerve and the infratemporal fossa. Diagnosis was established in both cases by biopsying radiologically abnormal tissue in the infratemporal fossa.Conclusion:An awareness of this condition is required to establish the diagnosis and initiate appropriate therapy. Glucocorticoids are the mainstay of initial treatment. The effectiveness of B-lymphocyte depletion with rituximab has also been reported. Correct diagnosis may spare patients from unnecessarily radical surgery.


2018 ◽  
Vol 32 (7) ◽  
pp. 824-829
Author(s):  
Hiroaki Kuwahara ◽  
Issei Kawakita ◽  
Ryuta Ueda ◽  
Mayuko Akimoto ◽  
Mitsuru Yanai ◽  
...  

Author(s):  
Fang Bu ◽  
Selene C. Koo

Context.— Immunoglobulin G4 (IgG4)–related disease is rare but well characterized in adults; however, the clinical and histologic manifestations in children may differ. Objective.— To review the clinical and histologic features of IgG4-rich head and neck lesions in a pediatric population. Design.— Retrospective search for cases with IgG4 immunohistochemical staining performed at our institution from 2011 to 2019. Review of clinical courses, serology profiles, histologic patterns, and immunohistochemical staining patterns. Results.— Four pediatric IgG4-rich lesions were identified and showed distinct histologic patterns from adult IgG4-related disease, including absence of pathognomonic findings associated with the latter. One case showed intralesional immunoglobulin light-chain restriction. Clinical review showed serum IgG4 elevation in 2 of 4 cases, presence of additional autoantibody positivity, and a generally benign/treatment-responsive clinical course. Conclusions.— Pediatric IgG4-related disease shows distinct clinical, serologic, and histologic features from its adult counterpart. Pediatric IgG4-related disease involving the orbit has unique clinical characteristics, including frequently normal serum IgG4 levels and female predominance. Awareness and evaluation for these features may improve diagnosis and treatment.


2017 ◽  
Vol 103 (6) ◽  
pp. 516-524 ◽  
Author(s):  
Luca Tagliaferri ◽  
Francesco Bussu ◽  
Bruno Fionda ◽  
Francesco Catucci ◽  
Mario Rigante ◽  
...  

Aim The aim of our study was to evaluate the outcomes of patients reirradiated with high-dose-rate (HDR) intensity-modulated brachytherapy (IMBT) for recurrent head and neck cancer and to perform a systematic review of the literature. Materials and Methods Patients treated with prior radiation doses >65 Gy were considered. After resection of macroscopic disease, catheters were fixed to the tumor bed. The total dose was 30 Gy in 12 fractions of 2.5 Gy twice daily for 5 days a week. A systematic literature search was conducted through several electronic databases including Medline/PubMed, Scopus, Embase and the Cochrane library. Results Seventeen patients were included; median overall survival was 19 months with a median local control interval of 15 months. Median follow-up was 36 months. Seven papers were considered for the review. Conclusions IMBT could play an important role in the retreatment of recurrent head and neck cancer.


2007 ◽  
Vol 25 (24) ◽  
pp. 3759-3765 ◽  
Author(s):  
Charlotte L. Zuur ◽  
Yvonne J. Simis ◽  
Pauline E. Lansdaal ◽  
Augustinus A. Hart ◽  
Jan H. Schornagel ◽  
...  

Purpose Cisplatin concomitantly administered with radiotherapy is increasingly used in locally advanced head and neck squamous cell carcinoma. We aimed to compare the incidence of hearing loss between patients treated with intra-arterial high-dose cisplatin chemoradiation with sodium thiosulfate (CRT-IA) and intravenous high-dose cisplatin chemoradiation without sodium thiosulfate (CRT-IV). Patients and Methods We conducted a prospective analysis of hearing thresholds at low and (ultra-) high frequencies obtained before, during, and after treatment in 158 patients. Patients were randomly assigned for either CRT-IA (150 mg/m2, four courses) with sodium thiosulfate cisplatin neutralization or CRT-IV (100 mg/m2, three courses) without rescue. All patients received concomitant radiation therapy (RT; 70 Gy). Results CRT-IA resulted in approximately 10% less hearing loss at frequencies vital for speech perception, compared with CRT-IV (P < .001). In CRT-IA, fewer ears qualified for hearing aids (36% v 49%; P = .03). However, in both treatment arms, the incidence expressed in National Cancer Institute Common Terminology Criteria of Adverse Events (version 3) did not deviate (P > .14). Age, cumulative cisplatin dose, cumulative RT dose, and the considered frequency area determine the degree of hearing loss (P < .001). Cisplatin induced increasing hearing loss of 24% to 60% with increasing frequencies. RT induced hearing loss at speech frequencies of 9% to 12%. Conclusion Depending on the criteria used to assess hearing loss due to treatment, differences in ototoxicity between CRT-IA and CRT-IV were found in favor of CRT-IA. It is desirable to specify hearing loss criteria toward frequencies vital for speech perception, and to refine grading scales to reveal subtle and clinically relevant dissimilarities in ototoxicity between different treatment protocols.


2021 ◽  
Vol 12 ◽  
Author(s):  
Wei-Chieh Huang ◽  
Ying-Ying Chen ◽  
Yen-Hung Lin ◽  
Jeff S. Chueh

BackgroundSuperior outcomes after surgical treatment over medical treatment for primary aldosteronism (PA) has been reported in small-scale clinical studies, but no solid conclusion has been drawn as results of large randomized trials are lacking.MethodsWe performed a search of PubMed, MEDLINE, Embase and Cochrane Library for randomized or observational studies that investigated cardiovascular outcomes in patients with PA undergoing medical versus surgical treatment. Meta-analyses of both composite and individual outcomes were conducted. Risks of bias of the included studies were assessed with Risk Of Bias In Non-randomized Studies of Interventions (ROBINS-I) checklist. Trial sequential analysis (TSA) was performed to control the risk of random errors and assess whether the results in our meta-analysis were conclusive.ResultsA total of 12 studies, including a total of 6148 PA patients, were included in the meta-analysis. The results of meta-analyses demonstrated lower incidence of composite cardiovascular outcomes among PA patients who underwent surgical treatment over medical treatment (odds ratio (OR): 0.49). Surgical treatment also led to less incidence of persistence of hypertension (OR of non-cure hypertension: 0.31). Fewer major cardiovascular events and mortality events were observed (OR: 0.60) after surgical treatment. TSA result showed that the required information size was 2151 and the cumulative Z curve crossed the futility boundary and reached the required information size.ConclusionSuperior performance of surgical treatment over medical treatment is confirmed with meta-analyses in terms of lower incidences of composite cardiovascular outcomes and non-cure of hypertension. Hence, adrenalectomy could now be concluded as the treatment of choice for lateralized PA.


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