Pediatric Laryngeal Expression and Surgical Treatment of IgG4-Related Disease

Jordan, Victoria A.; Hernandez, Loren P. Herrera; Cofer, Shelagh A.; Roby, Brianne Barnett

doi:10.1001/jamaoto.2018.2258

Cited by 6 publications

(3 citation statements)

References 6 publications

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“…It was the pharmacological treatment that finally gave resolution of symptoms. The non-surgical approach included a wide range of medications: glucocorticoids, immunomodulatory drugs and cytostatic drugs; however, each of them is recommended in systhemic IgG4-RD [ 7 , 8 , 9 , 10 , 11 , 12 , 13 , 14 , 15 , 20 ].…”

Section: Resultsmentioning

confidence: 99%

“…Injury of the semi-rigid structure of larynx and healing process leads to formation of concentric scars and thereafter progressive narrowing and airway compromise [ 6 ]. In the literature, we found only 13 cases of the disease localized in the region [ 7 , 8 , 9 , 10 , 11 , 12 , 13 , 14 , 15 ]. Patients usually complained of shortness of breath, dysphagia, dysphonia or hoarseness and, for that reason, were referred to ENT doctors.…”

Section: Introductionmentioning

confidence: 99%

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Supraglottic Localization of IgG4-Related Disease—Rare and Challenging Equity

Barańska

Makowska

Wągrowska-Danilewicz

et al. 2022

JPM

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Supraglottic stenosis is a rare symptom, particularly in fibroinflammatory multifocal diseases, such as IgG4-related disease (IgG4-RD). There is still an inconsistency in the diagnosis of less-common locations of IgG4-RD, which causes a delay in the diagnosis and treatment. Our paper aims to analyze different aspects of IgG4-RD presenting as supraglottic stenosis, including the possible overlap with ANCA-associated vasculitis. We compare the usefulness of the recently revised ACR/EULAR and Comprehensive criteria and discuss treatment options. The review was performed according to PRISMA guidelines using the MEDLINE Pubmed and Scopus databases. The analysis includes nine papers describing supraglottic laryngeal stenosis in 13 patients. Furthermore, we present a case of a woman with ongoing supraglottic stenosis presenting with cough, temporary dyspnea and stridor as the symptoms of localized IgG4-RD. At the time of writing, the patient remains in remission while receiving treatment with cyclophosphamide and methylprednisolone. The symptoms of supraglottic localization of IgG4-RD may be severe; however, at that point, clinicians should suspect autoimmune etiology and attempt to modulate the autoimmune response instead of performing dilatation surgery—the effects of which may not result in extended intervals between interventions. The ACR/EULAR criteria show great specificity; however, when IgG4-RD is presumed, the specific treatment should be implemented.

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Section: Resultsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Supraglottic Localization of IgG4-Related Disease—Rare and Challenging Equity

Barańska

Makowska

Wągrowska-Danilewicz

et al. 2022

JPM

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“…One recent review article identified only 25 cases of childhood presentation up to 2016—none of them affecting the larynx or trachea [ 24 ]. Moreover, to the best of our knowledge, there is only one previous report of laryngeal involvement in childhood [ 26 ]. Therefore, we think that the current case may improve the understanding and increase familiarity of this generally unknown and multifaceted disease among pediatric practitioners.…”

Section: Discussionmentioning

confidence: 99%

A Case of Tracheal Stenosis as an Isolated Form of Immunoproliferative Hyper-IgG4 Disease in a 17-Year-Old Girl

et al. 2021

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Immunoglobulin G4-related disease (IgG4-RD) is a lymphoproliferative disease which is described almost exclusively in adults. There are only a few pediatric patients who have been observed with this disorder. Here, we describe a rare case of IgG4-RD in a 17-year-old girl with a single manifestation—tracheal stenosis without previous intubation or other inciting event. She had mixed dyspnea and noisy and weakened breathing. Immunoproliferative hyper-IgG4 disease was diagnosed, based on elevated serum IgG4 and histological findings. Until now we have chosen to treat the girl only with corticosteroids with a good response so far. The general condition as well as the respiratory function are regularly monitored. The tracheal involvement of IgG4-RD is uncommon. Nonetheless, it is a manifestation that should be included in the differential diagnosis of tracheal stenosis.

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