gg4-related inflammatory pseudotumour of the trigeminal nerve: imaging findings and clinical features
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1 gg4-related inflammatory pseudotumour of the trigeminal nerve: imaging findings and clinical features Poster No.: C-2603 Congress: ECR 2013 Type: Scientific Exhibit Authors: Y. Kawamura, Y. Kikuchi, I. Yamazaki, S. Nakaji, N. Hirata; Chiba/ JP Keywords: Neuroradiology brain, Head and neck, MR, CT, PET, Contrast agent-intravenous, Education, Inflammation DOI: /ecr2013/C-2603 Any information contained in this pdf file is automatically generated from digital material submitted to EPOS by third parties in the form of scientific presentations. References to any names, marks, products, or services of third parties or hypertext links to thirdparty sites or information are provided solely as a convenience to you and do not in any way constitute or imply ECR's endorsement, sponsorship or recommendation of the third party, information, product or service. ECR is not responsible for the content of these pages and does not make any representations regarding the content or accuracy of material in this file. As per copyright regulations, any unauthorised use of the material or parts thereof as well as commercial reproduction or multiple distribution by any traditional or electronically based reproduction/publication method ist strictly prohibited. You agree to defend, indemnify, and hold ECR harmless from and against any and all claims, damages, costs, and expenses, including attorneys' fees, arising from or related to your use of these pages. Please note: Links to movies, ppt slideshows and any other multimedia files are not available in the pdf version of presentations. Page 1 of 8
2 Purpose Since the first report on patients with elevated serum IgG4 in sclerosing pancreatitis in 2001, IgG4-related multiple organ lymphoproloferative syndrome (IgG4+MOLPS) has been encountered and diagnosed including autoimmune pancreatitis (AIP), retroperitoneal fibromatosis, sclerosing cholangitis, and Mikulicz's disease. It is important to understanf this disease entity to provide proper treatment. To discuss the involvement of IgG4+MOLPS, CT and MRI images of the head, neck, and brain showing soft-tissue masses by IgG4+MOLPS are reviewed and the radiological features and clinical characteristics of IgG4-related disease are evaluated. Methods and Materials Five patients with autoimmune disorders were evaluated for head and neck lesions by MDCT (Toshiba, Aquilion 64) and MRI (SIEMENS, Avanto 1.5T). Clinical diagnoses were all confirmed pathologically except one (Patient 5) Patient 1: 61 year-old woman with retroperitoneal fibromatosis, complained right orbital discomfort Patient 2: 67 year-old man with retroperitoneal fibromatosis revealed bilateral and painless swelling of the parotid and submandibular glands Patient 3: 62 year-old woman with AIP and bilateral salivary glands swelling. Patient 4: 64 year-old man with retroperitoneal fibromatosis, and suspicious sclerosing cholangitis evaluated for left nasal to orbital mass. Patient 5: 73 year-old man with retroperitoneal fibromatosis, complained chronic headache. Results #Summary of imaging features of patients# In Patient 1, MRI demonstrated homogeneously enhancing soft-tissue mass involving the skull base (Fig. 1 on page 3) along with the second and the third divisions of right trigeminal nerve. PET-CT showed FDG uptake; SUV=5.99 to 6.46 (Fig. 2 on page 4). Page 2 of 8
3 In Patient 2 and 3, CT and MRI showed symmetric enlargement of bilateral salivary glands with homogeneous enhancement. In Patient 4, hypointensity lesion in left nasal cavity extending into left orbit on T2 weighted images showing homogeneous enhancement after Gd admonistration (Fig. 3 on page 5). PET-CT showed marked FDG accumulation (SUV=11.33, Fig. 4). In Patient 5, MRI demonstrated the dural thickening and enhancement in the skull base. #Patient 1: Fig. 1 on page 3 and Fig. 2#: 61yo#woman Chief Complaint: discomfort of right eye Present Illness: From two weeks prior, The twitching sensation of right eyelid started followed by discomfort of right temporoparietal area and the feeling of right eye pulled outside for about two minutes, 5-12 times/day. Past History: 19y ago: diabetes mellitus# 11y ago: bil. dacryoadenitis and submandibular sialoadenitis on steroid therapy# 5y ago, left bleary eyes and diplopia; suspected retrobulbar neuritis. Social History: smoke (-)#alcohol (-)# allergy (-) #Patient 4: Fig. 3 and Fig. 4#: 64yo#man Chief Complaint: left ophthalmalgia Present Illness: The patient developed ophthalmalgia and left nasal bleeding. Chronic sinusitis was diagnosed and was treated by local doctor with no improvement. He was transfered to our department for further evaluation. Past History: 10y ago: diabetes mellitus# 4y ago: submandibular lymphadenopathy, resected and cured. Images for this section: Page 3 of 8
4 Fig. 1: MRI shows soft-tissue mass involving the skull base along with the second and the third divisions of right trigeminal nerve. Page 4 of 8
5 Fig. 2: PET-CT reveals moderate FDG uptake; SUV of 5.99to Page 5 of 8
6 Fig. 3: T2WI demonstrates hypointensity lesion in left nasal cavity extending into left orbit (upper right). The lesion is seen low intensity on T1WI (lower left) and shows homogeneous enhancement after Gd admonistration(lower right). Page 6 of 8
7 Conclusion Diagnostic criteria for IgG4+MOLPS (1) serum IgG4 concentration >135 mg/dl (2) >40% of IgG+ plasma cells being IgG4+ and >10 cells/high powered field of biopsy sample. Although the comprehensive diagnostic criteria are not sufficiently sensitive for the diagnosis of type 1 IgG4-related autoimmune pancreatitis (IgG4-related AIP), they are adequately sensitive for IgG4-related Mikulicz's disease (MD) and kidney disease (KD). In addition, the comprehensive diagnostic criteria, combined with organ-specific diagnostic criteria, have increased the sensitivity of diagnosis to 100% for IgG4-related MD, KD, and AIP. The unique IgG4-related lymphproliferative disease may be demonstrated as soft-tissue mass extending along meninges and the cranial nerves such as the trigeminal nerve. As described in our cases, the clinical symptoms are not so severe even MRI and CT show remarkable dilatation of involved nerves. In majority of cases, the steroid therapy is effective. Careful interpretation of MRI is helpful to suspect the uncommon IgG4-related disease and to lead the IgG4 measurement as well as to reduce the risk of the invasive biopsy planning. References Hamano H. et. al. High Serum IgG4 Concentrations in Patients with Sclerosing Pancreatitis. N Engl J Med. 344: , 2001 #mehara H. et. al. Comprehensive Diagnostic Criteria for IgG4-related disease (IgG4-RD). Mod. Rheumatol. 22: 21-30, 2012 Katsura M. et. al. IgG4-related inflammatory pseudotumor of the trigeminal nerve: a nother component of IgG4-related sclerosing disease? AJNR. 32: E150-2, 2011 Stone JH,. et. al. Mechanisms of Disease IgG4-Related Disease. N Engl J Med. 366: , 2012 Page 7 of 8
8 Personal Information Page 8 of 8
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