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Immunoglobulin G4-related diseases in the head and neck: a systematic review.

Mulholland GB, Jeffery CC, Satija P, Côté DW - J Otolaryngol Head Neck Surg (2015)

Bottom Line: IgG4-RD is a challenging non-surgical disease that has multiple manifestations in the head and neck.It must be distinguished from various mimics including malignancy, systemic diseases, and infectious.Otolaryngology-Head and Neck surgeons should be aware of this condition and its management.

View Article: PubMed Central - PubMed

Affiliation: Division of Otolaryngology-Head and Neck Surgery, 1E4 Walter MacKenzie Centre, University of Alberta, 8440 112 Street, Edmonton, AB, T6G 2B7, Canada. graemem@ualberta.ca.

ABSTRACT

Background: Immunoglobulin G4 related disease (IgG4-RD) is a poorly understood chronic inflammatory disorder affecting the middle-aged and elderly that can present to the otolaryngologist. We aim to summarize the current literature regarding the manifestations and management of IgG4-RD in the head and neck.

Methods: Pubmed and EMBASE were searched using the term relevant search algorithm utilizing keywords such as: IgG4 related disease, head and neck, orbit, salivary glands, sialadenitis, Kuttner, angiocentric eosinophilic fibrosis, submandibular, lacrimal, thyroid, dacryoadenitis, nasal, sinus, and Mikulicz's. Reference lists were searched for identification of relevant studies. Case reports, original research and review articles published in English from 1964 to 2014 whose major topic was IgG4-RD affecting the head and neck were included. Data regarding patient demographics, presentation, histopathology, management and treatment outcomes of IgG4-RD were extracted. Level of evidence was also assessed and data were pooled where possible. Three independent reviewers screened eligible studies; extracted relevant data and discrepancies were resolved by consensus, where applicable. Descriptive and comparative statistics were performed.

Results: Fourty-three articles met our inclusion criteria. IgG4-RD most often presents as a mass lesion in the head and neck region. Common diagnostic features include: (1) elevated serum IgG4 level, (2) marked infiltration of exocrine glands by IgG4-positive plasma cells with fibrosis, and (3) marked improvement with corticosteroid therapy and additional immunosuppressive therapy in corticosteroid refractory cases. Early diagnosis and involvement of rheumatology is important in management.

Conclusions: IgG4-RD is a challenging non-surgical disease that has multiple manifestations in the head and neck. It must be distinguished from various mimics including malignancy, systemic diseases, and infectious. Otolaryngology-Head and Neck surgeons should be aware of this condition and its management.

No MeSH data available.


Related in: MedlinePlus

Flow chart of studies obtained through literature search, eligible and excluded (Uploaded separately as per submission instructions)
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Fig1: Flow chart of studies obtained through literature search, eligible and excluded (Uploaded separately as per submission instructions)

Mentions: We initially identified 1592 articles through a combination of literature search and citation review. After reviewing abstracts, 247 articles of interest were identified. Three authors independently reviewed the articles and 43 articles met inclusion criteria (Fig. 1). Of the 43 articles, 21 case reports and 5 large case series (greater than 15 patients) were included (Table 1). A large proportion of articles were from Japan (41.9 %), contributing 86.6 % of the total individual cases (Table 1).Fig. 1


Immunoglobulin G4-related diseases in the head and neck: a systematic review.

Mulholland GB, Jeffery CC, Satija P, Côté DW - J Otolaryngol Head Neck Surg (2015)

Flow chart of studies obtained through literature search, eligible and excluded (Uploaded separately as per submission instructions)
© Copyright Policy - open-access
Related In: Results  -  Collection

License 1 - License 2
Show All Figures
getmorefigures.php?uid=PMC4482182&req=5

Fig1: Flow chart of studies obtained through literature search, eligible and excluded (Uploaded separately as per submission instructions)
Mentions: We initially identified 1592 articles through a combination of literature search and citation review. After reviewing abstracts, 247 articles of interest were identified. Three authors independently reviewed the articles and 43 articles met inclusion criteria (Fig. 1). Of the 43 articles, 21 case reports and 5 large case series (greater than 15 patients) were included (Table 1). A large proportion of articles were from Japan (41.9 %), contributing 86.6 % of the total individual cases (Table 1).Fig. 1

Bottom Line: IgG4-RD is a challenging non-surgical disease that has multiple manifestations in the head and neck.It must be distinguished from various mimics including malignancy, systemic diseases, and infectious.Otolaryngology-Head and Neck surgeons should be aware of this condition and its management.

View Article: PubMed Central - PubMed

Affiliation: Division of Otolaryngology-Head and Neck Surgery, 1E4 Walter MacKenzie Centre, University of Alberta, 8440 112 Street, Edmonton, AB, T6G 2B7, Canada. graemem@ualberta.ca.

ABSTRACT

Background: Immunoglobulin G4 related disease (IgG4-RD) is a poorly understood chronic inflammatory disorder affecting the middle-aged and elderly that can present to the otolaryngologist. We aim to summarize the current literature regarding the manifestations and management of IgG4-RD in the head and neck.

Methods: Pubmed and EMBASE were searched using the term relevant search algorithm utilizing keywords such as: IgG4 related disease, head and neck, orbit, salivary glands, sialadenitis, Kuttner, angiocentric eosinophilic fibrosis, submandibular, lacrimal, thyroid, dacryoadenitis, nasal, sinus, and Mikulicz's. Reference lists were searched for identification of relevant studies. Case reports, original research and review articles published in English from 1964 to 2014 whose major topic was IgG4-RD affecting the head and neck were included. Data regarding patient demographics, presentation, histopathology, management and treatment outcomes of IgG4-RD were extracted. Level of evidence was also assessed and data were pooled where possible. Three independent reviewers screened eligible studies; extracted relevant data and discrepancies were resolved by consensus, where applicable. Descriptive and comparative statistics were performed.

Results: Fourty-three articles met our inclusion criteria. IgG4-RD most often presents as a mass lesion in the head and neck region. Common diagnostic features include: (1) elevated serum IgG4 level, (2) marked infiltration of exocrine glands by IgG4-positive plasma cells with fibrosis, and (3) marked improvement with corticosteroid therapy and additional immunosuppressive therapy in corticosteroid refractory cases. Early diagnosis and involvement of rheumatology is important in management.

Conclusions: IgG4-RD is a challenging non-surgical disease that has multiple manifestations in the head and neck. It must be distinguished from various mimics including malignancy, systemic diseases, and infectious. Otolaryngology-Head and Neck surgeons should be aware of this condition and its management.

No MeSH data available.


Related in: MedlinePlus