Clinicopathologic similarities between Mikulicz disease and Küttner tumor

Published on Nov 1, 2010in American Journal of Otolaryngology1.267
· DOI :10.1016/J.AMJOTO.2009.08.002
Kenichi Takano23
Estimated H-index: 23
(Sapporo Medical University),
Motohisa Yamamoto35
Estimated H-index: 35
(Sapporo Medical University)
+ 3 AuthorsTetsuo Himi35
Estimated H-index: 35
(Sapporo Medical University)
Abstract Purpose Recent studies have revealed that Mikulicz disease (MD) differs from Sjogren syndrome and is an immunoglobulin G 4 (IgG 4 )–related systemic disease. Kuttner tumor (KT) is also reported to be an IgG 4 -related disease. In this study, we examined the clinicopathologic and serologic findings in MD (39 patients) and KT (6 patients) and attempted to discern the similarities between MD and KT. Materials and methods We diagnosed 39 patients with MD and 6 patients with KT. We analyzed the clinicopathologic and serologic findings (IgG subclasses) in 39 patients with MD and 6 patients with KT. Submandibular and labial salivary gland specimens obtained from patients with MD and KT were stained with anti-IgG 4 antibodies. Results The mean IgG 4 concentration (±SD) was 931.1 ± 796.2 mg/dL in patients with MD and 756.2 ± 449.2 mg/dL in patients with KT. Abundant infiltration of IgG 4 -positive plasmacytes into the salivary glands was observed in both patients with MD and patients with KT. Conclusion The serologic and histopathologic findings in MD and KT are very similar, and these 2 conditions may be IgG 4 -related systemic diseases.
📖 Papers frequently viewed together
2,772 Citations
390 Citations
443 Citations
#1Samuel L. Murphy (Children's Hospital of Philadelphia)H-Index: 12
#2Hojun Li (UPenn: University of Pennsylvania)H-Index: 9
Last. Katherine A. High (Children's Hospital of Philadelphia)H-Index: 80
view all 7 authors...
Humoral immune responses occur following exposure to Adeno-associated virus (AAV) or AAV vectors. Many studies characterized antibody responses to AAV, but human IgG subclass responses to AAV have not been previously described. In this study, IgG subclass responses were examined in serum samples of normal human subjects exposed to wild-type AAV, subjects injected intramuscularly with AAV vectors and subjects injected intravascularly with AAV vectors. A diversity of IgG subclass responses to AAV ...
71 CitationsSource
#1Takako SaekiH-Index: 29
#2Akihiko Saito (Niigata University)H-Index: 28
Last. Fumitake Gejyo (Niigata University)H-Index: 59
view all 8 authors...
We report three elderly men with high serum IgG4 concentrations and multiple lymphoplasmacytic inflammation of the salivary glands, lymph nodes, pancreas, and renal interstitium. The infiltrating plasma cells had strong immunoreactivity for IgG4, even in patients without pancreatic lesions. These cases show that IgG4 is associated not only with autoimmune pancreatitis, but also with other systemic lymphoplasmacytic disease.
135 CitationsSource
#1Motohisa Yamamoto (Sapporo Medical University)H-Index: 35
#2Hiroki Takahashi (Sapporo Medical University)H-Index: 51
Last. Kohzoh Imai (Sapporo Medical University)H-Index: 87
view all 8 authors...
in Sjogren’s syndrome with a combination of prednisone and cyclophosphamide. Arch Neurol 2001;58:815–19. 5. Hermisson M, Klein R, Schmidt F, Weller M, Kuler W. Myelopathy in primary Sjogren’s syndrome: diagnostic and therapeutic aspects. Acta Neurol Scand 2002;105:450–3. 6. Hawley RJ, Hendricks WT. Treatment of Sjogren syndrome myelopathy with azathioprine and steroids. Arch Neurol 2002;59:875. 7. Vitali C, Bombardieri S, Moutsopoulos HM, Coll J, Gerli R, Hatron PY, et al. Assessment of the Euro...
98 CitationsSource
#1Satoshi Kitagawa (Kanazawa University)H-Index: 6
#2Yoh ZenH-Index: 69
Last. Yasuni NakanumaH-Index: 87
view all 16 authors...
Chronic sclerosing sialadenitis (CSS) is a cryptogenic tumor-like condition of the salivary gland(s). While immunemediated processes are suspected in its pathogenesis, and CSS is occasionally reported to be associated with sclerosing pancreatitis, an IgG4-related disease, the exact immunopathologic processes of CSS remain speculative. In this study, we examined the clinicopathologic findings of CSS (12 cases) in comparison with sialolithiasis (8 cases) and Sjogren's syndrome (13 cases), and trie...
443 CitationsSource
#1Motohisa Yamamoto (Sapporo Medical University)H-Index: 35
#2Hiroki Takahashi (Sapporo Medical University)H-Index: 51
Last. Kohzoh Imai (Sapporo Medical University)H-Index: 87
view all 4 authors...
Abstract Mikulicz's disease (MD) has been considered part of primary Sjogren's syndrome (SS) since Morgan's report in 1953. MD represents a unique condition involving enlargement of the lacrimal and salivary glands, as is also seen in SS; however, MD is characterized by few autoimmune reaction and its good responsiveness to glucocorticoid. Recent reports have shown that the frequency of apoptosis in glands of MD patients is lower when compared with SS. This phenomenon reflects the histologically...
200 CitationsSource
#1Toshiro Fukui (Kyoto University)H-Index: 21
#2Kazuichi Okazaki (Kansai Medical University)H-Index: 68
Last. Tsutomu Chiba (Kyoto University)H-Index: 81
view all 12 authors...
We report a very rare case of autoimmune pancreatitis (AIP) associated with sclerosing cholangitis, retroperitoneal fibrosis and Sjogren’s syndrome. The patient had an enlarged pancreas, and autoantibodies were detected in the serum. Serum IgG and IgG4 concentrations were also elevated. Endoscopic retrograde cholangiopancreatography revealed an irregular narrowing of the main pancreatic duct from the head to the body and sclerotic change in the intrapancreatic common bile duct, which later exten...
83 CitationsSource
#1Yoh Zen (Kanazawa University)H-Index: 69
#2Kenichi HaradaH-Index: 52
Last. Yasuni NakanumaH-Index: 87
view all 15 authors...
AbstractSclerosing cholangitis (SC) is a heterogeneous disease entity. Different etiologies such as choledocholithiasis, biliary tumor, or pericholangitis can manifest as SC. Hepatic inflammatory pseudotumor (IP) is rarely associated with SC (sclerosing cholangitis associated with hepatic inflammato
667 CitationsSource
#1C. DuvicH-Index: 9
#2Jérôme DesrameH-Index: 1
Last. G. NedelecH-Index: 8
view all 4 authors...
67 CitationsSource
#1Maryam Ayatollahi (Shiraz University of Medical Sciences)H-Index: 13
#2S Joubeh (Shiraz University of Medical Sciences)H-Index: 1
Last. Abbas Ghaderi (Shiraz University of Medical Sciences)H-Index: 34
view all 5 authors...
27 CitationsSource
#1Brian S. Dooreck (UM: University of Miami)H-Index: 1
#2Paul KatzH-Index: 1
Last. Jamie S. BarkinH-Index: 36
view all 3 authors...
23 CitationsSource
Cited By54
#2Katarzyna Błochowiak (Poznan University of Medical Sciences)H-Index: 3
#1Norberto Mancera (USF: University of South Florida)H-Index: 3
#2Jasmina Bajric (USF: University of South Florida)H-Index: 1
Last. Curtis E. Margo (USF: University of South Florida)H-Index: 30
view all 3 authors...
ABSTRACTWe describe an otherwise healthy 24-year-old woman with reactive lymphoid hyperplasia of one lacrimal gland. She was found to have modest elevation of serum IgG4 and high density of IgG4+ p...
#1Mizuki Sakamoto (Kyushu University)H-Index: 21
#2Masafumi Moriyama (Kyushu University)H-Index: 23
Last. Seiji Nakamura (Kyushu University)H-Index: 42
view all 16 authors...
AbstractObjectives: In this study, we investigated the diagnostic utility of submandibular gland (SMG) sonography and labial salivary gland (LSG) biopsy as a less invasive procedure for diagnosing ...
3 CitationsSource
#1Kai-Min Fang (PRC: China Medical University (PRC))H-Index: 1
#2Ming-Hsun Wen (Memorial Hospital of South Bend)H-Index: 1
Last. Li-Jen Liao (YZU: Yuan Ze University)H-Index: 8
view all 6 authors...
Specifications about the size and stiffness of healthy salivary glands with ultrasound (US) are not available for Asian people. Using a Toshiba Apolio 500 US platform, we determined the size (including anterior-posterior median length, median paramandibular depth dimension, and cranio-caudal height) and hardness of 100 healthy submandibular and parotid glands in volunteers without a history of disease affecting the salivary glands or post-radiation, and compared the dimensions to those of 36 par...
1 CitationsSource
#1Nima Hafezi-Nejad (Johns Hopkins University)H-Index: 12
#1Nima Hafezi-Nejad (Johns Hopkins University)H-Index: 39
Last. Atif Zaheer (Johns Hopkins University)H-Index: 20
view all 5 authors...
: Autoimmune pancreatitis (AIP) is characterized by autoimmune inflammatory destruction of the pancreatic tissue. Imaging plays an essential role in the diagnosis. AIP type 1 is the pancreatic manifestation of immunoglobulin G4 (IgG4)-related disease and is associated with IgG4-positive plasma cell infiltration and fibrosis of multiple organ systems. Type 2 is a related disease with pancreatic inflammation with or without concurrent inflammatory bowel disease. The authors demonstrate the imaging...
7 CitationsSource
#1Kenichi Takano (Sapporo Medical University)H-Index: 23
#2Ryoto Yajima (Sapporo Medical University)H-Index: 4
Last. Tetsuo Himi (Sapporo Medical University)H-Index: 35
view all 8 authors...
OBJECTIVES/HYPOTHESIS: The aim of this study was to evaluate the utility of 18 F-fluorodeoxyglucose (FDG) positron emission tomography/computed tomography (PET/CT) for accurately diagnosing immunoglobulin G4-related sclerosing sialadenitis (IgG4-SS). STUDY DESIGN: Retrospective cohort study. METHODS: We reviewed the records of 64 patients with IgG4-SS (35 male and 29 female patients) and 10 patients with clinically suspected IgG4-SS. Pathological diagnoses of patients clinically suspected with I...
4 CitationsSource
#1Andrea Gallo (Sapienza University of Rome)H-Index: 25
Last. M. De VincentiisH-Index: 17
view all 7 authors...
: Le patologie autoimmuni delle ghiandole salivari maggiori comprendono la sindrome di Sjogren e l’insieme di condizioni morbose raccolte sotto il nome di patologie IgG4-correlate. Tanto la sindrome di Sjogren quanto le patologie IgG4-correlate sono caratterizzate da una reazione autoimmune mediata dai linfociti T-Helper il cui bersaglio e rappresentato dai dotti delle ghiandole esocrine nella sindrome di Sjogren e dal parenchima ghiandolare nelle malattie IgG4-correlate. Queste ultime, di intro...
7 CitationsSource
#1Kenichi Takano (Sapporo Medical University)H-Index: 23
#2Motohisa Yamamoto (Sapporo Medical University)H-Index: 35
Last. Tetsuo Himi (Sapporo Medical University)H-Index: 35
view all 4 authors...
IgG4-related disease (IgG4-RD) is a chronic inflammatory disorder, characterized by elevated serum IgG4 levels as well as abundant infiltration of IgG4-positive plasmacytes and fibrosis in various organs, including the head and neck region. In particular, the salivary glands, orbit, and thyroid are common sites of disease involvement. IgG4-RD is diagnosed based on various clinical, serological, and histopathological findings, none of which are pathognomonic. Hence, various differential diagnoses...
30 CitationsSource
#1Satomi Omotehara (Hokkaido University)H-Index: 4
#2Mutsumi Nishida (Hokkaido University)H-Index: 10
Last. Chikara Shimizu (Hokkaido University)H-Index: 17
view all 10 authors...
Purpose We evaluated the sonographic findings of immunoglobulin G4-related sclerosing sialadenitis (IgG4-SS).
4 CitationsSource