Inflammatory pseudotumour of the alveolar process of the maxilla as clinical manifestation of IgG4-related disease: a case report and literature review. Issue 6 (June 2020)

• Record Type: Journal Article
• Authors: Kouwenberg, W.L.; Dieleman, F.J.; Willems, S.M.; Rosenberg, A.J.W.P.
• Is Part Of: International journal of oral & maxillofacial surgery. Volume 49:Issue 6(2020:Jun.)
• Page Start: 722
• Page End: 725
• Publication Date: 2020-06
• Subjects: IgG4 -- IgG4-related disease -- pseudotumour -- maxilla -- sinonasal; Mouth -- Surgery -- Periodicals; Maxilla -- Surgery -- Periodicals; Dentistry -- Periodicals; Dentistry, Operative; Oral Surgical Procedures; Surgery, Oral; Dentistry; Maxilla -- Surgery; Mouth -- Surgery; Electronic journals; Periodicals; 617.52059
• DOI: 10.1016/j.ijom.2019.11.008
• Languages: English
• ISSNs: 0901-5027

Abstract

Abstract: IgG4-related disease (IgG4-RD) is an uncommon immune-mediated condition considered to be a systemic disease, described in multiple organ systems. IgG4-RD that involves the maxillary and sinonasal region is rare. This report presents a very rare presentation of IgG4-RD in the maxillary alveolar process. The patient presented with left-sided facial pain, headache, and mobility and loss of teeth. The first biopsy and resection specimen reports were inconclusive and showed a non-specific chronic inflammatory process. After the third resection, the diagnosis was finally established through findings that satisfied the 2012 consensus criteria for IgG4-RD. Consequently high doses of oral corticosteroids and azathioprine were given, tapered over a total period of 36 months. Weaning is still in progress, but no recurrence was observed after 34 months. A review of the English-language literature was performed, which identified seven cases of IgG4-RD with maxillary and sinonasal involvement. Cases were excluded from the review if there was any doubt that they met the consensus statement on the pathology.