Advertisement
Oral and maxillofacial pathology Online only article| Volume 115, ISSUE 2, e48-e51, February 2013

IgG4-related sclerosing disease clinically mimicking oral squamous cell carcinoma

Published:August 17, 2012DOI:https://doi.org/10.1016/j.oooo.2012.04.011
      IgG4-related sclerosing disease is a distinct clinicopathologic entity known to involve the maxillofacial region, particularly the salivary, lacrimal, and pituitary glands. We report a case with lesions involving the tongue and palatine tonsil with associated skin lesions. A 45-year-old female patient presented with a history of soreness, dysphagia, and an asymptomatic rash involving the upper trunk. The initial clinical diagnosis of her oral lesions was squamous cell carcinoma. The diagnosis of an IgG4-related lesion was confirmed by histologic examination of the oral and skin lesions as well as confirmation of raised serum IgG4 levels. Tapering systemic corticosteroid therapy resulted in complete resolution of the lesions. This is the first report of IgG4-related sclerosing disease presenting as concurrent oral and skin lesions, with the oral lesion clinically resembling oral squamous cell carcinoma. Such lesions present a diagnostic challenge, but the outcome is very favorable.
      To read this article in full you will need to make a payment

      Purchase one-time access:

      Academic & Personal: 24 hour online accessCorporate R&D Professionals: 24 hour online access
      One-time access price info
      • For academic or personal research use, select 'Academic and Personal'
      • For corporate R&D use, select 'Corporate R&D Professionals'

      References

        • Solomon L.W.
        • Wein R.O.
        • Rosenwald I.
        • Laver N.
        Plasma cell mucositis of the oral cavity: report of a case and review of the literature.
        Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2008; 106: 853-860
        • Neild G.H.
        • Rodriguez-Justo M.
        • Wall C.
        • Connolly J.O.
        Hyper-IgG4 disease: report and characterisation of a new disease.
        BMC Med. 2006; 6: 4-23
        • Cheuk W.
        • Chan J.K.
        IgG4-related sclerosing disease: a critical appraisal of an evolving clinicopathologic entity.
        Adv Anat Pathol. 2010; 17: 303-332
        • Sarles H.
        • Sarles J.C.
        • Muratore R.
        • Guien C.
        Chronic inflammatory sclerosis of the pancreas: an autoimmune pancreatic disease?.
        Am J Dig Dis. 1961; 6: 688-698
        • Yoshida K.
        • Toki F.
        • Takeuchi T.
        • Watanabe S.
        • Shiratori K.
        • Hayashi N.
        Chronic pancreatitis caused by an autoimmune abnormality.
        Dig Dis Sci. 1995; 40: 1561-1568
        • Deshpande V.
        • Mino-Kenudson M.
        • Brugge W.
        • Lauwers G.Y.
        Autoimmune pancreatitis: more than just a pancreatic disease?.
        Arch Pathol Lab Med. 2005; 129: 1148-1154
        • Hamano H.
        • Kawa S.
        • Horiuchi A.
        • Unno H.
        • Furuya N.
        • Akamatsu T.
        • et al.
        High serum IgG4 concentrations in patients with sclerosing pancreatitis.
        N Engl J Med. 2001; 344: 732-738
        • Geyer J.T.
        • Ferry J.A.
        • Harris N.L.
        • Stone J.H.
        • Zukerberg L.R.
        • Lauwers G.Y.
        • et al.
        Chronic sclerosing sialadenitis (Kuttner tumor) is an IgG4-associated disease.
        Am J Surg Pathol. 2010; 34: 202-210
        • Witkiewicz A.K.
        • Kennedy E.P.
        • Kennyon L.
        • Yeo C.J.
        • Hruban R.H.
        Synchronous autoimmune pancreatitis and infiltrating ductal adenocarcinoma: a case report and review of the literature.
        Hum Pathol. 2008; 39: 1548-1551
        • Kamisawa T.
        • Chen P.Y.
        • Tu Y.
        • Nakajima H.
        • Egawa N.
        • Tsuruta K.
        • et al.
        Pancreatic cancer with a high serum IgG4 concentration.
        World J Gastroenterol. 2006; 12: 6225-6228
        • Inoue H.
        • Miyatani H.
        • Sawada Y.
        • Yoshida Y.
        A case of pancreas cancer with autoimmune pancreatitis.
        Pancreas. 2006; 33: 208-209
        • Pace C.
        • Ward S.
        A rare case of IgG4-related sclerosing disease of the maxillary sinus associated with bone destruction.
        J Oral Maxillofac Surg. 2010; 68: 2591-2593
        • Ishida M.
        • Hotta M.
        • Kushima R.
        • Shibayama M.
        • Shimizu T.
        • Okabe H.
        Multiple IgG4-related sclerosing lesions in the maxillary sinus, parotid gland and nasal septum.
        Pathol Int. 2009; 59: 670-675
        • Gill J.
        • Angelo N.
        • Yeong M.L.
        • McIvor N.
        Salivary duct carcinoma arising in IgG4-related autoimmune disease of the parotid gland.
        Hum Pathol. 2009; 40: 881-886
        • Suresh L.
        • Kumar V.
        Significance of IgG4 in the diagnosis of mucous membrane pemphigoid.
        Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2007; 104: 359-362
        • Brooks W.S.
        • Lee Y.Y.
        • Abell E.
        • Deng J.S.
        Comparison of IgG subclasses and complement binding activity of autoantibodies from patients with bullous pemphigoid and pemphigus.
        J Clin Lab Anal. 1989; 3: 307-311
        • Allen J.
        • Shears E.
        • Powell J.
        • Wojnarowska F.
        Assessment of skin basement membrane zone antibodies in the urine of patients with acquired subepidermal immunobullous diseases.
        Br J Dermatol. 2001; 144: 540-545
        • Bernard P.
        • Prost C.
        • Aucouturier P.
        • Durepaire N.
        • Denis F.
        • Bonnetblanc J.M.
        The subclass distribution of IgG autoantibodies in cicatricial pemphigoid and epidermolysis bullosa acquisita.
        J Invest Dermatol. 1991; 97: 259-263
        • Patton T.
        • Plunkett R.W.
        • Beutner E.H.
        • Deng J.S.
        • Jukic D.M.
        IgG4 as the predominant IgG subclass in pemphigoides gestationis.
        J Cutan Pathol. 2006; 33: 299-302
        • Modre B.
        • Allen J.
        • Wojnarowska F.
        Does class switching contribute to remission in bullous pemphigoid?.
        Acta Derm Venereol. 1999; 79: 127-131