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Published in: Arthritis Research & Therapy 1/2015

Open Access 01-12-2015 | Research article

Effectiveness of imaging modalities for screening IgG4-related dacryoadenitis and sialadenitis (Mikulicz’s disease) and for differentiating it from Sjögren’s syndrome (SS), with an emphasis on sonography

Authors: Mayumi Shimizu, Kazutoshi Okamura, Yoshitaka Kise, Yohei Takeshita, Hiroko Furuhashi, Warangkana Weerawanich, Masafumi Moriyama, Yukiko Ohyama, Sachiko Furukawa, Seiji Nakamura, Kazunori Yoshiura

Published in: Arthritis Research & Therapy | Issue 1/2015

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Abstract

Introduction

The aim of this study was to clarify the effectiveness of various imaging modalities and characteristic imaging features in the screening of IgG4-related dacryoadenitis and sialadenitis (IgG4-DS), and to show the differences in the imaging features between IgG4-DS and Sjögren’s syndrome (SS).

Methods

Thirty-nine patients with IgG4-DS, 51 with SS and 36 with normal salivary glands were enrolled. Images of the parotid and submandibular glands obtained using sonography, 2-[18F]-fluoro-2-deoxy-D-glucose positron emission tomography/computed tomography (FDG-PET/CT), computed tomography (CT) and magnetic resonance imaging (MRI) were retrospectively analyzed. Six oral and maxillofacial radiologists randomly reviewed the arranged image sets under blinded conditions. Each observer scored the confidence rating regarding the presence of the characteristic imaging findings using a 5-grade rating system. After scoring various findings, diagnosis was made as normal, IgG4-DS or SS, considering all findings for each case.

Results

On sonography, multiple hypoechoic areas and hyperechoic lines and/or spots in the parotid glands and obscuration of submandibular gland configuration were detected mainly in patients with SS (median scores 4, 4 and 3, respectively). Reticular and nodal patterns were observed primarily in patients with IgG4-DS (median score 5). FDG-PET/CT revealed a tendency for abnormal 18F-FDG accumulation and swelling of both the parotid and submandibular glands in patients with IgG4-DS, particularly in the submandibular glands. On MRI, SS had a high score regarding the findings of a salt-and-pepper appearance and/or multiple cystic areas in the parotid glands (median score 4.5). Sonography showed the highest values among the four imaging modalities for sensitivity, specificity and accuracy. There were significant differences between sonography and CT (p = 0.0001) and between sonography and FDG-PET/CT (p = 0.0058) concerning accuracy.

Conclusions

Changes in the submandibular glands affected by IgG4-DS could be easily detected using sonography (characteristic bilateral nodal/reticular change) and FDG-PET/CT (abnormal 18F-FDG accumulation). Even inexperienced observers could detect these findings. In addition, sonography could also differentiate SS. Consequently, we recommend sonography as a modality for the screening of IgG4-DS, because it is easy to use, involves no radiation exposure and is an effective imaging modality.
Literature
1.
go back to reference Tsubota K, Fujita H, Tsuzaka K, Takeuchi T. Mikulicz’s disease and Sjögren’s syndrome. Invest Ophthalmol Vis Sci. 2000;41:1666–73.PubMed Tsubota K, Fujita H, Tsuzaka K, Takeuchi T. Mikulicz’s disease and Sjögren’s syndrome. Invest Ophthalmol Vis Sci. 2000;41:1666–73.PubMed
2.
go back to reference Yamamoto M, Harada S, Ohara M, Suzuki C, Naishiro Y, Yamamoto H, et al. Clinical and pathological differences between Mikulicz’s disease and Sjögren’s syndrome. Rheumatology. 2005;44:227–34.CrossRefPubMed Yamamoto M, Harada S, Ohara M, Suzuki C, Naishiro Y, Yamamoto H, et al. Clinical and pathological differences between Mikulicz’s disease and Sjögren’s syndrome. Rheumatology. 2005;44:227–34.CrossRefPubMed
3.
go back to reference Yamamoto M, Takahashi H, Sugai S, Imai K. Clinical and pathological characteristics of Mikulicz’s disease (IgG4-related plasmacytic exocrinopathy). Autoimmunity Rev. 2005;4:195–200.CrossRef Yamamoto M, Takahashi H, Sugai S, Imai K. Clinical and pathological characteristics of Mikulicz’s disease (IgG4-related plasmacytic exocrinopathy). Autoimmunity Rev. 2005;4:195–200.CrossRef
4.
go back to reference Wallace ZS, Deshpande V, Mattoo H, Mahajan VS, Kulikova M, Pillai S, et al. IgG4-related disease: clinical and laboratory features in 125 patients. Arthritis Rheumatol. 2015; doi: 10.1002/art.39205. Epub 2015 May 18. Wallace ZS, Deshpande V, Mattoo H, Mahajan VS, Kulikova M, Pillai S, et al. IgG4-related disease: clinical and laboratory features in 125 patients. Arthritis Rheumatol. 2015; doi: 10.​1002/​art.​39205. Epub 2015 May 18.
5.
go back to reference Ishii S, Shishido F, Miyajima M, Sakuma K, Shigihara T, Kikuchi K. Whole-body gallium-67 scintigraphic findings in IgG4-related disease. Clin Nucl Med. 2011;36:542–5.CrossRefPubMed Ishii S, Shishido F, Miyajima M, Sakuma K, Shigihara T, Kikuchi K. Whole-body gallium-67 scintigraphic findings in IgG4-related disease. Clin Nucl Med. 2011;36:542–5.CrossRefPubMed
6.
go back to reference Ishii S, Miyajima M, Sakuma K, Kikuchi K, Shishido F. Comparison between sarcoidosis and IgG4-related disease by whole-body 67Ga scintigraphy. Nucl Med Commun. 2013;34:13–8.CrossRefPubMed Ishii S, Miyajima M, Sakuma K, Kikuchi K, Shishido F. Comparison between sarcoidosis and IgG4-related disease by whole-body 67Ga scintigraphy. Nucl Med Commun. 2013;34:13–8.CrossRefPubMed
7.
go back to reference Ebbo M, Grados A, Guedj E, Gobert D, Colavolpe C, Zaidan M, et al. Usefulness of 2-[18F]-fluoro-2-deoxy-d-glucose-positron emission tomography/computed tomography for staging and evaluation of treatment response in IgG4-related disease: a retrospective multicenter study. Arthritis Care Res (Hoboken). 2014;66:86–96.CrossRef Ebbo M, Grados A, Guedj E, Gobert D, Colavolpe C, Zaidan M, et al. Usefulness of 2-[18F]-fluoro-2-deoxy-d-glucose-positron emission tomography/computed tomography for staging and evaluation of treatment response in IgG4-related disease: a retrospective multicenter study. Arthritis Care Res (Hoboken). 2014;66:86–96.CrossRef
8.
go back to reference Zhang J, Chen H, Ma Y, Xiao Y, Niu N, Lin W, et al. Characterizing IgG4-related disease with 18F-FDG PET/CT: a prospective cohort study. Eur J Nucl Med Mol Imaging. 2014;41:1624–34.PubMedCentralCrossRefPubMed Zhang J, Chen H, Ma Y, Xiao Y, Niu N, Lin W, et al. Characterizing IgG4-related disease with 18F-FDG PET/CT: a prospective cohort study. Eur J Nucl Med Mol Imaging. 2014;41:1624–34.PubMedCentralCrossRefPubMed
9.
go back to reference Shimizu M, Moriyama M, Okamura K, Kawazu T, Chikui T, Goto TK, et al. Sonographic diagnosis for Mikulicz disease. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2009;108:105–13.CrossRefPubMed Shimizu M, Moriyama M, Okamura K, Kawazu T, Chikui T, Goto TK, et al. Sonographic diagnosis for Mikulicz disease. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2009;108:105–13.CrossRefPubMed
10.
go back to reference Asai S, Okami K, Nakamura N, Shiraishi S, Sugimoto R, Anar D, et al. Localized or diffuse lesions of the submandibular glands in immunoglobulin g4-related disease in association with differential organ involvement. J Ultrasound Med. 2013;32:731–6.CrossRefPubMed Asai S, Okami K, Nakamura N, Shiraishi S, Sugimoto R, Anar D, et al. Localized or diffuse lesions of the submandibular glands in immunoglobulin g4-related disease in association with differential organ involvement. J Ultrasound Med. 2013;32:731–6.CrossRefPubMed
11.
go back to reference Takagi Y, Nakamura H, Origuchi T, Miyashita T, Kawakami A, Sumi M, et al. IgG4-related Mikulicz's disease: ultrasonography of the salivary and lacrimal glands for monitoring the efficacy of corticosteroid therapy. Clin Exp Rheumatol. 2013;31:773–5.PubMed Takagi Y, Nakamura H, Origuchi T, Miyashita T, Kawakami A, Sumi M, et al. IgG4-related Mikulicz's disease: ultrasonography of the salivary and lacrimal glands for monitoring the efficacy of corticosteroid therapy. Clin Exp Rheumatol. 2013;31:773–5.PubMed
12.
go back to reference Katsura M, Mori H, Kunimatsu A, Sasaki H, Abe O, Machida T, et al. Radiological features of IgG4-related disease in the head, neck, and brain. Neuroradiology. 2012;54:873–82.CrossRefPubMed Katsura M, Mori H, Kunimatsu A, Sasaki H, Abe O, Machida T, et al. Radiological features of IgG4-related disease in the head, neck, and brain. Neuroradiology. 2012;54:873–82.CrossRefPubMed
13.
go back to reference Fujita A, Sakai O, Chapman MN, Sugimoto H. IgG4-related disease of the head and neck: CT and MR imaging manifestations. Radiographics. 2012;32:1945–58.CrossRefPubMed Fujita A, Sakai O, Chapman MN, Sugimoto H. IgG4-related disease of the head and neck: CT and MR imaging manifestations. Radiographics. 2012;32:1945–58.CrossRefPubMed
14.
go back to reference Okazaki K, Kawa S, Kamisawa T, Shimosegawa T, Tanaka M. Research Committee for Intractable Pancreatic Disease and Japan Pancreas Society. Japanese consensus guidelines for management of autoimmune pancreatitis: I. Concept and diagnosis of autoimmune pancreatitis. J Gastroenterol. 2010;45:249–65.CrossRefPubMed Okazaki K, Kawa S, Kamisawa T, Shimosegawa T, Tanaka M. Research Committee for Intractable Pancreatic Disease and Japan Pancreas Society. Japanese consensus guidelines for management of autoimmune pancreatitis: I. Concept and diagnosis of autoimmune pancreatitis. J Gastroenterol. 2010;45:249–65.CrossRefPubMed
15.
go back to reference Yamamoto M, Takahashi H, Ohara M, Suzuki C, Naishiro Y, Yamamoto H, et al. A new conceptualization for Mikulicz’s disease as an IgG4-related plasmacytic disease. Mod Rheumatol. 2006;16:335–40.PubMedCentralCrossRefPubMed Yamamoto M, Takahashi H, Ohara M, Suzuki C, Naishiro Y, Yamamoto H, et al. A new conceptualization for Mikulicz’s disease as an IgG4-related plasmacytic disease. Mod Rheumatol. 2006;16:335–40.PubMedCentralCrossRefPubMed
16.
go back to reference Fujibayashi T, Sugai S, Miyasaka N, Hayashi Y, Tsubota K. Revised Japanese criteria for Sjögren's syndrome (1999): availability and validity. Mod Rheumatol. 2004;14:425–34.CrossRefPubMed Fujibayashi T, Sugai S, Miyasaka N, Hayashi Y, Tsubota K. Revised Japanese criteria for Sjögren's syndrome (1999): availability and validity. Mod Rheumatol. 2004;14:425–34.CrossRefPubMed
17.
go back to reference Vitali C, Bombardieri S, Jonsson R, Moutsopoulos HM, Alexander EL, Carsons SE, et al. Classification criteria for Sjogren's syndrome: a revised version of the European criteria proposed by the American–European Consensus Group. Ann Rheum Dis. 2002;61:554–8.PubMedCentralCrossRefPubMed Vitali C, Bombardieri S, Jonsson R, Moutsopoulos HM, Alexander EL, Carsons SE, et al. Classification criteria for Sjogren's syndrome: a revised version of the European criteria proposed by the American–European Consensus Group. Ann Rheum Dis. 2002;61:554–8.PubMedCentralCrossRefPubMed
18.
go back to reference Takagi Y, Kimura Y, Nakamura H, Sasaki M, Eguchi K, Nakamura T. Salivary gland ultrasonography: can it be an alternative to sialography as an imaging modality for Sjogren's syndrome? Ann Rheum Dis. 2010;69:1321–4.CrossRefPubMed Takagi Y, Kimura Y, Nakamura H, Sasaki M, Eguchi K, Nakamura T. Salivary gland ultrasonography: can it be an alternative to sialography as an imaging modality for Sjogren's syndrome? Ann Rheum Dis. 2010;69:1321–4.CrossRefPubMed
19.
go back to reference Shimizu M, Okamura K, Yoshiura K, Ohyama Y, Nakamura S, Kinukawa N. Sonographic diagnostic criteria for screening Sjögren's syndrome. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2006;102:85–93.CrossRefPubMed Shimizu M, Okamura K, Yoshiura K, Ohyama Y, Nakamura S, Kinukawa N. Sonographic diagnostic criteria for screening Sjögren's syndrome. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2006;102:85–93.CrossRefPubMed
20.
go back to reference Sun Z, Zhang Z, Fu K, Zhao Y, Liu D, Ma X. Diagnostic accuracy of parotid CT for identifying Sjögren's syndrome. Eur J Radiol. 2012;81:2702–9.CrossRefPubMed Sun Z, Zhang Z, Fu K, Zhao Y, Liu D, Ma X. Diagnostic accuracy of parotid CT for identifying Sjögren's syndrome. Eur J Radiol. 2012;81:2702–9.CrossRefPubMed
21.
go back to reference Takashima S, Takeuchi N, Morimoto S, Tomiyama N, Ikezoe J, Shogen K, et al. MR imaging of Sjögren syndrome: correlation with sialography and pathology. J Comput Assist Tomogr. 1991;15:393–400.CrossRefPubMed Takashima S, Takeuchi N, Morimoto S, Tomiyama N, Ikezoe J, Shogen K, et al. MR imaging of Sjögren syndrome: correlation with sialography and pathology. J Comput Assist Tomogr. 1991;15:393–400.CrossRefPubMed
22.
go back to reference Shimizu M, Okamura K, Yoshiura K, Ohyama Y, Nakamura S. Sonographic diagnosis of Sjögren syndrome: evaluation of parotid gland vascularity as a diagnostic tool. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2008;106:587–94.CrossRefPubMed Shimizu M, Okamura K, Yoshiura K, Ohyama Y, Nakamura S. Sonographic diagnosis of Sjögren syndrome: evaluation of parotid gland vascularity as a diagnostic tool. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2008;106:587–94.CrossRefPubMed
23.
go back to reference Cohen C, Mekinian A, Uzunhan Y, Fauchais AL, Dhote R, Pop G, et al. 18F-fluorodeoxyglucose positron emission tomography/computer tomography as an objective tool for assessing disease activity in Sjögren's syndrome. Autoimmun Rev. 2013;12:1109–14.CrossRefPubMed Cohen C, Mekinian A, Uzunhan Y, Fauchais AL, Dhote R, Pop G, et al. 18F-fluorodeoxyglucose positron emission tomography/computer tomography as an objective tool for assessing disease activity in Sjögren's syndrome. Autoimmun Rev. 2013;12:1109–14.CrossRefPubMed
24.
go back to reference Nakamoto Y, Tatsumi M, Hammoud D, Cohade C, Osman MM, Wahl RL. Normal FDG distribution patterns in the head and neck: PET/CT evaluation. Radiology. 2005;234:879–85.CrossRefPubMed Nakamoto Y, Tatsumi M, Hammoud D, Cohade C, Osman MM, Wahl RL. Normal FDG distribution patterns in the head and neck: PET/CT evaluation. Radiology. 2005;234:879–85.CrossRefPubMed
25.
go back to reference Ohta M, Moriyama M, Goto Y, Kawano S, Tanaka A, Maehara T, et al. A case of marginal zone B cell lymphoma mimicking IgG4-related dacryoadenitis and sialoadenitis. World J Surg Oncol. 2015;13:459. doi:10.1186/s12957-015-0459-z. Epub 2015 Feb 21.CrossRef Ohta M, Moriyama M, Goto Y, Kawano S, Tanaka A, Maehara T, et al. A case of marginal zone B cell lymphoma mimicking IgG4-related dacryoadenitis and sialoadenitis. World J Surg Oncol. 2015;13:459. doi:10.​1186/​s12957-015-0459-z. Epub 2015 Feb 21.CrossRef
Metadata
Title
Effectiveness of imaging modalities for screening IgG4-related dacryoadenitis and sialadenitis (Mikulicz’s disease) and for differentiating it from Sjögren’s syndrome (SS), with an emphasis on sonography
Authors
Mayumi Shimizu
Kazutoshi Okamura
Yoshitaka Kise
Yohei Takeshita
Hiroko Furuhashi
Warangkana Weerawanich
Masafumi Moriyama
Yukiko Ohyama
Sachiko Furukawa
Seiji Nakamura
Kazunori Yoshiura
Publication date
01-12-2015
Publisher
BioMed Central
Published in
Arthritis Research & Therapy / Issue 1/2015
Electronic ISSN: 1478-6362
DOI
https://doi.org/10.1186/s13075-015-0751-x

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