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Hideaki Hamano

Publications and source records attributed to Hideaki Hamano.

At least 19 recordsLinked to original sources
PubMed2006

IgG4-associated multifocal systemic fibrosis complicating sclerosing sialadenitis, hypophysitis, and retroperitoneal fibrosis, but lacking pancreatic involvement.

A 71-year-old man was admitted with malaise, mild fever, anorexia, body weight loss, lower back pain, thirst, and polydipsia. He showed bilateral swelling of the submandibular glands. Examinations showed panhypopituitarism and a high serum IgG4 concentration. Fluorodeoxyglucose positron emission tomography (FDG-PET) revealed uptake in the pituitary gland, bilateral submandibular gland, bilateral hilar and mediastinal lymph nodes, and a mass consistent with retroperitoneal fibrosis, but not in the pancreas. Biopsy specimens from the submandibular gland and retroperitoneal mass indicated sialadenitis and retroperitoneal fibrosis respectively, and showed severe fibrosis and inflammation with marked lymphoplasmacytic infiltration and IgG4-positive plasma cell infiltration. Hormone replacement therapy with hydrocortisone resulted in marked clinical improvement. Systemic involvement found in this patient possibly corresponded to the new concept of IgG4-associated multifocal systemic fibrosis.

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PubMed2006

Two critical genes (HLA-DRB1 and ABCF1)in the HLA region are associated with the susceptibility to autoimmune pancreatitis.

We have previously reported that autoimmune pancreatitis (AIP) is a bioclinical entity characterized by high serum immunoglobulin G4 concentrations and association with the HLA-DRB1*0405-DQB1*0401 haplotype. However, the precise identity of gene(s) within this haplotype directly responsible for AIP pathogenesis is yet to be established. To dissect the genetic contribution of the incriminated haplotype, we have now performed an association analysis within the human leukocyte antigen (HLA) region using various types of polymorphic markers. Genomic DNAs from 43 AIP patients and 213 unrelated Japanese controls were used in this analysis. In each DNA sample, we established the genotype of 25 microsatellite markers distributed throughout the HLA region, that of single nucleotide polymorphism within the 5'-flanking regions of the TNFA and IkBLI (also known as NFKBIL1) as well as HLA class I and II genes. The HLA-linked susceptibility regions for AIP were localized to two segments: HLA-DRB1 (*0405; OR = 3.20, P = 0.00063, Pc = 0.0016) -DQB1 (*0401; OR = 3.29, P = 0.00046, Pc = 0.0069) in the HLA class II and C3-2-11 microsatellite (allele 219; OR = 2.96, P = 0.0076, Pc = 0.099) in the HLA class I regions. Upon stratification analysis in search for a synergistic effect given the extensive linkage disequilibrium within the major histocompatibility complex, it was established that each segment contributed to disease pathogenesis. The two critical HLA regions for susceptibility to AIP are limited to the HLA-DRB1*0405-DQB1*0401 in the class II and the ABCF1 proximal to C3-2-11, telomeric of HLA-E, in the class I regions.

ATP-Binding Cassette Transporters↗
PubMed2006

Autoimmune pancreatitis and complement activation system.

OBJECTIVES: Autoimmune pancreatitis is characterized by increased serum level of IgG4, but its pathogenesis has not been fully elucidated. Because this disease is occasionally associated with decreased levels of complements, we sought to clarify which complement activation system was operating in its active state. METHODS: We measured serum levels of complements, mannose-binding lectin, and circulating immune complex in patients with autoimmune pancreatitis, patients with chronic pancreatitis, and healthy controls. RESULTS: We found high serum circulating immune complex values, which decreased significantly after corticosteroid therapy. In patients with autoimmune pancreatitis, elevated levels of circulating immune complex, as determined by C1q assay, were significantly associated with increased serum levels of IgG1 and decreased levels of C4, as well as with a tendency toward decreased levels of C3. There were no significant differences in the serum levels of mannose-binding lectin or in the frequency of a mutant allele of mannose-binding lectin between patients with autoimmune pancreatitis and those with chronic calcifying pancreatitis. Furthermore, corticosteroid therapy had no effect on the level of mannose-binding lectin. CONCLUSIONS: Autoimmune pancreatitis exhibits a high serum circulating immune complex values in its active state, which links to a complement activation system with a classic pathway rather than the mannose-binding lectin pathway or alternative pathways.

Adult↗
PubMed2005

Seronegative Sjögren syndrome with asymptomatic autoimmune sclerosing pancreatitis.

We report two elderly patients with seronegative Sjögren syndrome who showed benign swelling of the pancreas on computed tomography. Immunostaining of the biopsied lip tissue or serum examination confirmed an increase in production of IgG4, leading to a diagnosis of autoimmune sclerosing pancreatitis (ASP) as a cause of the asymptomatic swelling of the pancreas. Sicca symptoms and ASP spontaneously improved in one patient, and the other responded well to oral prednisolone. Seronegative Sjögren syndrome and ASP can concurrently occur as a clinical manifestation of the IgG4-related systemic disorder, particularly in elderly subjects, and, in such a case, corticosteroid may be a potent therapeutic option.

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PubMed2005

Corticosteroid-responsive pancreatic cyst found in autoimmune pancreatitis.

There have been only a few reports of autoimmune pancreatitis complicated with pancreatic cyst and such cysts are rare, probably due to the absence of severe tissue necrosis and/or lack of stasis of the pancreatic juice in this condition. However, during a follow-up of 48 patients with this disease, we found 3 patients with pancreatic cysts, and this enabled us to evaluate their clinicopathological findings. Between September 1994 and July 2003, we treated and followed 48 patients with autoimmune pancreatitis, and found 3 patients with pancreatic cyst formation that was responsive to corticosteroid therapy. All of the patients with cysts had high serum IgG4 concentrations. After corticosteroid therapy, rapid resolution of the pancreatic cysts was observed. Immunostaining with goat polyclonal antibody for each IgG subclass showed severe infiltration of IgG4-positive plasma cells in the cyst wall in one patient. The high serum IgG4 concentration and favorable response to corticosteroid therapy suggests that a highly active state of the inflammatory process is closely associated with cyst formation, and that a corticosteroid-responsive pancreatic cyst is a characteristic feature of autoimmune pancreatitis.

Antibodies, Antinuclear↗
PubMed2005

High prevalence of hypothyroidism in patients with autoimmune pancreatitis.

Autoimmune pancreatitis is a unique form of chronic pancreatitis and has been correlated with various extrapancreatic lesions. To search for a correlation between autoimmune pancreatitis and thyroid lesions, we measured thyroid functions in 41 patients with autoimmune pancreatitis and in 41 patients with chronic calcifying pancreatitis and investigated the correlation between HLA antigens and hypothyroidism. We found a significant difference in the prevalence of antithyroglobulin antibody and hypothyroidism between patients with autoimmune pancreatitis and those with chronic pancreatitis (34.1 vs. 7.3%, P = 0.005, and 26.8 vs. 0%, P = 0.0005, respectively). Patients with hypothyroidism had a significantly higher frequency of antithyroglobulin antibody (63.6%) than those without hypothyroidism but showed no differences in other findings, including serum IgG4 concentration. We could find no significant association between any HLA antigens and the hypothyroid state of autoimmune pancreatitis. One quarter of the patients with autoimmune pancreatitis have hypothyroidism that may be independent of the active state of the pancreatic lesion or systemic fibrosing disorder, and thus patients suspected of having autoimmune pancreatitis should be evaluated for possible hypothyroidism.

Adult↗
PubMed2005

Immunoglobulin G4-related lymphoplasmacytic sclerosing cholangitis that mimics infiltrating hilar cholangiocarcinoma: part of a spectrum of autoimmune pancreatitis?

BACKGROUND: Autoimmune pancreatitis has been designated as sclerosing pancreatocholangitis, because this disease shows a high prevalence of bile-duct lesions. We present herein the clinical characteristics of unusual cases that show dominant bile-duct lesions and mimicking infiltrating hilar cholangiocarcinomas. METHODS: Clinical and pathologic findings of 3 patients with immunoglobulin (Ig) G4 related sclerosing cholangitis who had no apparent pancreatic lesions comparable with autoimmune pancreatitis were analyzed. OBSERVATIONS: All patients were middle-aged or elderly individuals with slightly elevated serum IgG4 concentrations and showed long-segment narrowing of the bile-duct system, mimicking infiltrating hilar cholangiocarcinoma without significant pancreatic change. The first patient was treated with a corticosteroid, resulting in amelioration of the narrowing of the bile duct. The second patient underwent surgery based on a diagnosis of cholangiocarcinoma. In the third patient, the bile-duct stricture reversed spontaneously 1 month after the drainage procedure. Pathologic findings of the bile ducts for all patients disclosed significant lymphoplasmacytic infiltration, including abundant IgG4-bearing plasma cells. CONCLUSIONS: The use of IgG4 immunostaining in biopsy specimens of the bile duct may identify the presence of corticosteroid-responsive lymphoplasmacytic sclerosing cholangitis.

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PubMed2005

Distinct clinicopathological entity 'autoimmune pancreatitis-associated sclerosing cholangitis'.

Autoimmune pancreatitis (AIP) is a recently proposed disease entity, in which an elevated serum IgG4 is characteristic. This disease is sometimes associated with other inflammatory diseases: Sjögren's disease, or sclerosing cholangitis. The aim of the present paper was to examine the difference in pathophysiology between AIP-associated sclerosing cholangitis (AIP-SC) and primary sclerosing cholangitis (PSC). The clinicopathological findings and the immunohistochemical expressions of IgG subclasses (IgG1, IgG2, IgG3, and IgG4) were evaluated for the two aforementioned diseases (six patients with each disease). Radiologically, the extrahepatic bile duct was involved in AIP-SC, whereas both extrahepatic and intrahepatic bile ducts were involved in PSC. Clinically, bile duct lesions in the former responded well to steroid therapy. Histologically, various degrees of mononuclear cell infiltration and fibrosis around bile ducts and portal tracts were found in all patients. Immunohistochemically, the IgG4-positive plasma cell/mononuclear cell ratio was significantly higher in AIP-SC than in PSC (P < 0.05). The IgG4-positive plasma cell/mononuclear cell ratio is a useful index to help distinguish AIP-SC from PSC. A mechanism similar to that involved in AIP may be involved in AIP-SC. The latter is a distinct clinicopathological entity that should be distinguished from PSC because it responds well to steroid therapy.

Adult↗
PubMed2004

Autoimmune pancreatitis is closely associated with gastric ulcer presenting with abundant IgG4-bearing plasma cell infiltration.

BACKGROUND: Autoimmune pancreatitis is characterized by high serum IgG4 concentrations and lymphoplasmacytic infiltration. Because of the diversity of extrapancreatic involvement in this disease, the present study sought to identify other associated GI-tract lesions. METHODS: EGD findings were compared between a group of 23 patients with autoimmune pancreatitis undergoing ERCP for obstructive jaundice and 230 age- and gender-matched control patients. To clarify the histopathologic differences found between these two groups, the histopathologic findings (Updated Sydney System) and the immunohistochemistry of each IgG subclass were compared between 8 patients with autoimmune pancreatitis and gastric ulcer, and 23 control patients with gastric ulcer from which biopsy specimens had been obtained. RESULTS: Gastric ulcer was found significantly more frequently in patients with autoimmune pancreatitis compared with control patients (34.8% vs. 13.5%; p=0.007). There was no significant difference between the groups with respect to the frequency of other GI lesions. Four of 8 gastric ulcers in patients with autoimmune pancreatitis were linear, with the long axis perpendicular to the incisura on the lesser curvature of the stomach. The activity score for the gastric lesions was significantly lower in patients with autoimmune pancreatitis compared with control patients (mean score 0.38 vs. 1.08; p=0.012). There were no significant differences in histopathologic findings with respect to inflammation, atrophy, metaplasia, or Helicobacter pylori scores between the two groups. IgG4-bearing plasma cells were significantly more abundant in gastric lesions in patients with autoimmune pancreatitis compared with those in control patients (mean score 1.75 vs. 0.39; p=0.0008). CONCLUSIONS: Autoimmune pancreatitis is closely associated with gastric ulcer with abundant IgG4-bearing plasma cell infiltration.

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PubMed2004

Recurrent attacks of autoimmune pancreatitis result in pancreatic stone formation.

OBJECTIVES: Autoimmune pancreatitis has been characterized by irregular narrowing of the main pancreatic duct and sonolucent swelling of the parenchyma, both of which are due to lymphoplasmacytic inflammation at the active stage of the disease, and by the absence of pancreatic stone formation. The aim of the present study was to confirm or deny whether or not this disease is progressive with recurrent attacks, resulting in pancreatic stone formation like ordinary chronic pancreatitis. METHODS: Forty-two patients, 36 of whom were treated with prednisolone, were followed up for periods longer than 12 months (median follow-up period: 54.5 months, range: 13-111 months) by regular interview and examination of their medical records for laboratory tests and image tests. RESULTS: Eleven patients (26.2%) who were treated with prednisolone showed recurrent attacks during median follow-up periods of 22 months. Eight patients (19%) showed the formation of pancreatic stones during the follow-up periods. Because 6 of 11 patients (54.5%) who suffered relapse showed pancreatic stone formation, it is significantly associated with relapse in comparison with nonrelapse (p= 0.0019). CONCLUSIONS: Contrary to previous reports, we observed both relapse and pancreatic stone formation in some patients with autoimmune pancreatitis, which suggests that autoimmune pancreatitis has the potential to be a progressive disease with pancreatic stones.

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PubMed2003

Hilar and pancreatic gallium-67 accumulation is characteristic feature of autoimmune pancreatitis.

INTRODUCTION AND AIMS: Autoimmune pancreatitis is characterized by severe lymphocytic inflammation, suggesting that gallium-67 scintigraphy provides a useful tool for detecting characteristic lesions of this disease, because gallium-67 concentrates in lymphoid cells. We tried to determine whether gallium-67 accumulates in the characteristic lesions. METHODOLOGY: We performed gallium-67 scintigraphy in 24 patients with autoimmune pancreatitis before and after 4 weeks of corticosteroid therapy and determined the factors associated with positive images. RESULTS: Sixteen patients (67%) had marked gallium-67 accumulation in the pancreas before corticosteroid therapy and negative images after 4 weeks of therapy, and they had significantly higher serum IgG4 values than did those without gallium-67 accumulation (median, 758 mg/dL versus 329 mg/dL; p = 0.011). Marked hilar gallium-67 accumulation was found in 16 patients (67%) and was also associated with significantly higher serum IgG4 values than did those without it (median, 758 versus 239 mg/dL; p = 0.0044). Among 16 patients with positive hilar images, 12 had positive pancreatic uptake and 5 had both pancreatic and salivary gland uptakes. CONCLUSIONS: Hilar and pancreatic accumulation of gallium-67 is a characteristic feature of autoimmune pancreatitis during the active stage of the disease, when IgG4 serum levels are high.

Adrenal Cortex Hormones↗
PubMed2002

Hydronephrosis associated with retroperitoneal fibrosis and sclerosing pancreatitis.

Sclerosing pancreatitis is associated with raised concentrations of IgG4. We treated 22 patients with sclerosing pancreatitis, and identified and followed-up three with concomitant hydronephrosis caused by ureteral mass, later diagnosed as retroperitoneal fibrosis. We histologically examined the ureteral and pancreatic lesions of these patients and noted abundant infiltration of IgG4-bearing plasma cells in both tissues. Treatment with corticosteroids lowered serum concentrations of IgG4. IgG4 might also have a pathological role in a systemic fibrosing process that includes pancreatic and retroperitoneal lesions.

Aged↗
PubMed2002

HLA DRB10405-DQB10401 haplotype is associated with autoimmune pancreatitis in the Japanese population.

BACKGROUND & AIMS: Autoimmune pancreatitis is a distinctive disease entity characterized by high serum immunoglobulin G4 concentrations. Because of the close association between some autoimmune diseases and particular alleles of major histocompatibility complex genes, we investigated the association between HLA alleles and autoimmune pancreatitis. METHODS: HLA-A, -B, -C, -DR, and -DQ gene typing and HLA-DRB1, -DQB1, and -DPB1 allele typing were performed by the polymerase chain reaction sequence-specific primers method and the restriction fragment length polymorphism method, respectively, in 40 patients with autoimmune pancreatitis, 43 patients with chronic calcifying pancreatitis, and 201 healthy subjects. RESULTS: In patients with autoimmune pancreatitis compared with healthy subjects, we found a significant increase in DR4 (73% vs. 44%, corrected P = 0.01) and DRB1*0405 (58% vs. 21%, corrected P = 0.000026) and DQ4 (58% vs. 26%, corrected P = 0.001) and DQB1*0401 (58% vs. 21%, corrected P = 0.000017). The DRB1*0405-DQB1*0401 haplotype in autoimmune pancreatitis showed no significant association with any HLA class I antigens, in contrast to the B54-DRB1*0405-DQB1*0401 haplotype reported in autoimmune hepatitis. The frequencies of DRB1*0405 and DQB1*0401 were significantly high in patients with autoimmune pancreatitis compared with chronic calcifying pancreatitis. CONCLUSIONS: It is probable that DRB1*0405-DQB1*0401 haplotype is associated with autoimmune pancreatitis in the Japanese population.

Adult↗
PubMed2002

ERCP features in 27 patients with autoimmune pancreatitis.

BACKGROUND: Autoimmune pancreatitis has been proposed as a new clinical entity. However, the ERCP features of this entity have not been well described. METHODS: Clinical and radiographic features in 27 patients with a diagnosis of autoimmune pancreatitis were evaluated. RESULTS: All 27 patients were at first suspected to have pancreatic cancer. The patients were predominantly elderly men and presented with jaundice or mild symptoms and pancreatic enlargement, but no attack of acute pancreatitis. A cholestatic biochemical profile and serum IgG elevation were usually present. A diffusely or segmentally irregular and narrow main pancreatic duct and a positive response to corticosteroid therapy were characteristic. During observation without treatment, serial pancreatography in 2 patients demonstrated progression of diffuse, irregular narrowing of the main pancreatic duct over periods, respectively, of 2 and 6 months. Both the irregular narrowing of the main pancreatic duct and distal bile duct strictures improved in various degrees in response to corticosteroid therapy. CONCLUSIONS: Segmental or diffuse irregular narrowing of the main pancreatic duct are ERCP features of autoimmune pancreatitis.

Adult↗
PubMed2002

Usefulness of endoscopic observation of the main duodenal papilla in the diagnosis of sclerosing pancreatitis.

BACKGROUND: There are no descriptions of the appearance of the main duodenal papilla in sclerosing pancreatitis. The intent of the present study was to clarify the characteristics of the main duodenal papilla in patients with sclerosing pancreatitis. METHODS: Macroscopic findings at ERCP with reference to the main duodenal papilla of 17 patients with sclerosing pancreatitis were compared with those of 24 patients with normal ERCP findings, 11 with chronic pancreatitis, 13 with primary sclerosing cholangitis, 21 with pancreatic cancer, and 18 with bile duct cancer. Endoscopic photographs of the papilla were reviewed retrospectively by 3 observers blinded to the underlying pancreaticobiliary pathology. Degree of swelling was scored in all patients. Biopsy specimens from swollen papillae were assessed histopathologically in 3 patients with sclerosing pancreatitis. RESULTS: Severe swelling of the main duodenal papilla was observed in 7 (41%) of 17 patients with sclerosing pancreatitis. The total score for the degree of swelling in patients with sclerosing pancreatitis was significantly higher than that for patients with a normal ERCP, chronic pancreatitis, primary sclerosing cholangitis, pancreatic cancer, and bile duct cancer (p < 0.01). T-lymphocyte infiltration of the papilla was evident in the biopsies from 3 patients with sclerosing pancreatitis. CONCLUSIONS: A swollen main duodenal papilla was a characteristic finding in patients with sclerosing pancreatitis. T-lymphocyte infiltration is present in the swollen main duodenal papilla. These features may be useful in the diagnosis of sclerosing pancreatitis.

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