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1.
Case Rep Endocrinol ; 2012: 280319, 2012.
Article in English | MEDLINE | ID: mdl-22934199

ABSTRACT

Thyroglobulin gene mutation is a rare cause of congenital hypothyroidism, but thyroglobulin gene mutations are thought to be associated with thyroid cancer development. A 21-year-old Japanese man treated with levothyroxine for congenital hypothyroidism had an enlarged thyroid gland with undetectable serum thyroglobulin despite elevated serum TSH level. The patient was diagnosed with thyroglobulin gene mutation, with compound heterozygosity for Gly304Cys missense mutation and Arg432X nonsense mutation. Ultrasonography showed a hypovascular large tumor in the left lobe that appeared as a cold nodule on thyroid scintigraphy. He underwent total thyroidectomy, but pathological study did not reveal findings of thyroid carcinoma, but rather a hyperplastic nodule with hemorrhage. Strong cytoplasmic thyroglobulin immunostaining was observed, but sodium iodide symporter immunostaining was hardly detected in the hyperplastic nodule. The clinical characteristics of patients with thyroglobulin gene mutations are diverse, and some patients are diagnosed by chance on examination of goiter in adults. The presence of thyroid tumors that appear as cold nodules on thyroid scintigraphy should consider the potential for thyroid carcinoma, if the patient has relatively low serum thyroglobulin concentration in relation to the degree of TSH without thyroglobulin autoantibody.

2.
Neuropathology ; 29(1): 85-90, 2009 Feb.
Article in English | MEDLINE | ID: mdl-18498285

ABSTRACT

Chordoid glioma, which generally occurs in adults, is a rare CNS tumor arising in the anterior part of the third ventricle. We report two cases of chordoid glioma of the third ventricle in a 42-year-old woman and a 51-year-old man, respectively. Both tumors showed essentially the same histological and immunohistochemical features; the tumors were composed of cords and nests of epithelioid, GFAP-immunoreactive cells in a mucinous stroma with lymphoplasmacytic infiltrates at the tumor periphery. Ultrastructural examination in one case revealed that the tumor cells were characterized by the presence of hemidesmosomes and associated focal basal lamina formation, intermediate junctions, microvilli and cilia, and intercellular microrosettes with microvilli. Of interest was that small blood vessels with fenestrated endothelial cells were present in the stroma. In the brain, the presence of fenestrated endothelial cells is a feature of the circumventricular organs (except the subcommissural organ), among which the organum vasculosum of the lamina terminalis is located in the anterior part of the third ventricular floor that is lined by specialized ependymal cells known as tanycytes. These findings further strengthen the hypothesis that chordoid glioma may represent a peculiar clinicopathological subtype of ependymoma (chordoid ependymoma) originating from the lamina terminalis area.


Subject(s)
Cerebral Ventricle Neoplasms/pathology , Glioma/pathology , Third Ventricle , Adult , Basement Membrane/pathology , Blood Vessels/pathology , Cerebral Ventricle Neoplasms/blood supply , Cerebral Ventricle Neoplasms/chemistry , Cerebral Ventricle Neoplasms/ultrastructure , Cilia/pathology , Endothelial Cells/pathology , Ependymoma/pathology , Epithelioid Cells/pathology , Female , Glial Fibrillary Acidic Protein/analysis , Glioma/blood supply , Glioma/chemistry , Glioma/ultrastructure , Hemidesmosomes/pathology , Humans , Immunohistochemistry , Magnetic Resonance Imaging , Male , Microscopy, Electron , Microvilli/pathology , Middle Aged , Tomography, X-Ray Computed
3.
Intern Med ; 47(23): 2087-9, 2008.
Article in English | MEDLINE | ID: mdl-19043267

ABSTRACT

A patient presented with Sjögren's syndrome associated with pulmonary multiple cystic lesions and a pulmonary arteriovenous fistulae. A histological examination of the lungs during the autopsy revealed the stenosis of the bronchiole lumens with hyperplasia of goblet cells, proliferation of smooth muscles in the inner wall of the bronchioli and retention of mucus in the airway lumens. These small airway changes were accompanied with chronic inflammatory changes of the airways in Sjögren's syndrome and led to the formation of cystic lesions via a ball-valve mechanism. Arteriovenous fistulae were situated around the cystic lesions. There may have been a correlation between the formation of the fistulae and cysts, but no mechanism was indicated in the histological findings. This report reveals that chronic inflammatory changes of the airways in Sjögren's syndrome are sufficient to cause the formation of cystic lesions.


Subject(s)
Arteriovenous Fistula/diagnosis , Cysts/diagnosis , Lung/abnormalities , Sjogren's Syndrome/diagnosis , Arteriovenous Fistula/complications , Cysts/complications , Fatal Outcome , Female , Humans , Lung/pathology , Middle Aged , Sjogren's Syndrome/complications
4.
Arerugi ; 57(8): 1061-6, 2008 Aug.
Article in Japanese | MEDLINE | ID: mdl-18781111

ABSTRACT

A 58-year-old Japanese female consulted our staff with multiple localized ground-glass opacities in chest CT. She underwent video assisted thoracoscopic surgery for diagnosis. Histopathologic finding from surgery specimen in one of ground-glass opacities revealed bronchioloalveolar carcinoma. Six months later, we performed second video assisted thoracoscopic surgery, and histopathologic finding of all other ground-glass opacities revealed pulmonary alveolar proteinosis. Serum anti GM-CSF antibody elevated, and she was diagnosed as having idiopathic pulmonary alveolar proteinosis. A case of idiopathic pulmonary alveolar proteinosis presenting multiple localized ground-glass opacities is rare. And, differentiating ground-glass opacities of pulmonary alveolar proteinosis and bronchioloalveolar carcinoma by chest CT is difficult.


Subject(s)
Pulmonary Alveolar Proteinosis/diagnostic imaging , Female , Humans , Lung/diagnostic imaging , Middle Aged , Tomography, X-Ray Computed
5.
Intern Med ; 47(13): 1237-40, 2008.
Article in English | MEDLINE | ID: mdl-18591847

ABSTRACT

A 54-year-old man with Graves' disease had been treated with thiamazole (5 mg/day). His thyroid hormone level was increased after exodontia in February 2006. Although his prescribed dose of thiamazole was increased after exodontia on the fourth day, he developed thyroid crisis on exodontia 52 nd day. Laboratory findings also showed renal dysfunction (from Cr 1.0 mg/dL in July 2005 to Cr 1.8 mg/dL on exodontia 37th day). His thyroid hormone level was normalized after subtotal thyroidectomy; however, serum Cr level was still high. He was diagnosed with interstitial nephritis as a result of renal biopsy, and he was treated with prednisolone 30 mg/day. This present case developed thyroid crisis even though the quantity of thiamazole was increased after exodontia. It seems that interstitial nephritis, as well as exodontia, is an aggravation factor of thyroid function. After a poor response to anti-thyroid drugs, it is necessary to prevent thyroid crisis by determining the aggravating factor and to then provide appropriate treatment.


Subject(s)
Graves Disease/complications , Nephritis, Interstitial/complications , Thyroid Crisis/etiology , Anti-Inflammatory Agents/therapeutic use , Humans , Male , Middle Aged , Nephritis, Interstitial/drug therapy , Nephritis, Interstitial/pathology , Oral Surgical Procedures/adverse effects , Prednisolone/therapeutic use , Thyroid Crisis/pathology
7.
Hepatol Res ; 37(11): 923-31, 2007 Nov.
Article in English | MEDLINE | ID: mdl-17610500

ABSTRACT

AIM: Serum antinuclear antibodies (ANA) are occasionally noted in patients with non-alcoholic steatohepatitis (NASH). We examined the significance of ANA in NASH. METHODS: We compared clinicopathological features in patients with ANA-positive NASH (n = 35) and ANA-negative NASH (n = 36). Inflammatory cell profiles and the distribution of oxidative stress markers were also examined immunohistochemically. RESULTS: ANA-positive NASH was significantly associated with female gender (P = 0.005), high degree of portal inflammation (P = 0.039), interface activity (P = 0.036) and hepatocellular ballooning (P = 0.0008). In addition, ANA of high titer (320-fold or more) was significantly associated with the histological grade and stage of NASH (P = 0.02). The degree of steatosis wais rather mild in the high-titer ANA group(P = 0.01). The analysis of inflammatory cell profiles revealed that CD3-positive T cells were predominant and plasma cells were rather few in the portal area and hepatic lobules in both ANA-positive and ANA-negative groups. There was no difference in the distribution of oxidative stress markers between ANA-positive and ANA-negative groups. CONCLUSION: These findings suggest that the presence of ANA may be related to the progression of NASH and that a different type of autoimmune mechanism may be involved in the pathogenesis of NASH with ANA, compared to the pathogenesis of autoimmune hepatitis.

8.
Endocr J ; 54(4): 543-51, 2007 Aug.
Article in English | MEDLINE | ID: mdl-17575368

ABSTRACT

A right adrenal tumor was incidentally discovered on abdominal computed tomography performed on a 53-year-old Japanese man, who had been hospitalized with diabetic ketoacidosis. Normal values were obtained for adrenal hormones in the morning after an overnight fast and urinary cortisol excretion after treatment of diabetic ketoacidosis with insulin. However, overnight dexamethasone administration with 1 mg or 8 mg did not completely suppress serum cortisol levels. There were no remarkable physical findings related to Cushing's syndrome. The patient was diagnosed as having preclinical Cushing's syndrome (PCS). Histological examination of the adrenalectomy specimen demonstrated adrenal black adenoma. Blood glucose levels subsequently improved after adrenalectomy, and the patient never developed adrenal insufficiency after hydrocortisone withdrawal. The patient was treated with diet therapy alone, and maintained good glycemic control. However, the patient still showed a diabetic pattern in an oral glucose tolerance test. It seems that the existence of PCS in addition to the underlying type 2 diabetes mellitus contributed to aggravation of blood glucose levels. Although there are many aspects of the natural course of PCS that have not been thoroughly elucidated, it is necessary to remain aware that a PCS patient with abnormal glucose metabolism may develop diabetic ketoacidosis by environmental agents.


Subject(s)
Adenoma/complications , Adrenal Gland Neoplasms/complications , Cushing Syndrome/etiology , Diabetes Mellitus, Type 2/etiology , Diabetic Ketoacidosis/etiology , Adenoma/pathology , Adenoma/surgery , Adrenal Gland Neoplasms/pathology , Adrenal Gland Neoplasms/surgery , Adrenalectomy , Blood Glucose , Cushing Syndrome/pathology , Diabetes Mellitus, Type 2/pathology , Diabetic Ketoacidosis/pathology , Humans , Magnetic Resonance Imaging , Male , Middle Aged , Tomography, X-Ray Computed
9.
Intern Med ; 45(22): 1291-5, 2006.
Article in English | MEDLINE | ID: mdl-17170503

ABSTRACT

We report a case with immunoglobulin A (IgA) nephropathy, showing IgA deposition which disappeared after peripheral blood stem cell transplantation (PBSCT) for acute lymphocytic leukemia (ALL). In 1996, a 28-year-old man was diagnosed with IgA nephropathy by renal biopsy. Steroid therapy improved proteinuria from 3 g/day to 1 g/day. In 2003, he received PBSCT following the initial therapy for ALL. After complete remission, urinary protein and hematuria remained at between (-) and (+/-). In 2004, the second renal biopsy specimen revealed no deposit of IgA or C3. These findings suggested that immune reconstruction with PBSCT following immunosuppression therapy was of benefit to IgA nephropathy.


Subject(s)
Glomerulonephritis, IGA/complications , Glomerulonephritis, IGA/pathology , Immunosuppression Therapy , Peripheral Blood Stem Cell Transplantation , Precursor Cell Lymphoblastic Leukemia-Lymphoma/surgery , Adult , Humans , Kidney/pathology , Male , Remission Induction
11.
Clin Rheumatol ; 25(4): 562-3, 2006 Jul.
Article in English | MEDLINE | ID: mdl-16292471

ABSTRACT

We report the case of a 77-year-old woman with biopsy-confirmed polyarteritis nodosa (PAN) associated with cryofibrinogenemia presenting with polyarthralgia and digital gangrene induced by cold exposure. The clinical manifestations and parameters measured by laboratory markers including cryofibrinogen responded well to corticosteroid therapy. To our knowledge, the case of the combination of PAN and cryofibrinogenemia has not been reported. Our case indicates that cryofibrinogenemia might be associated with PAN. The PAN patients with cold-induced symptoms should be screened for cryofibrinogen.


Subject(s)
Cryoglobulinemia/complications , Cryoglobulins/analysis , Fibrinogens, Abnormal/analysis , Polyarteritis Nodosa/complications , Sural Nerve/pathology , Aged , Cryoglobulinemia/etiology , Cryoglobulinemia/physiopathology , Female , Glucocorticoids/therapeutic use , Humans , Japan , Polyarteritis Nodosa/drug therapy , Polyarteritis Nodosa/physiopathology
12.
Arerugi ; 53(7): 696-9, 2004 Jul.
Article in Japanese | MEDLINE | ID: mdl-15492495

ABSTRACT

A 52-year-old woman who has worked at factory to check pack Lyophyllum karst for six years consulted another doctor for dry cough as common cold. After a month, low grade fever, dyspnea on effort, and productive cough brought her to our hospital. Her chest radiographs showed multiple small nodular shadows and ground glass opacities in entire lung fields. Histopathological examination of transbronchial lung biopsy specimen revealed granuloma and thickening of alveolar septa with lymphocytes infiltration. She was admitted to our hospital for hypersensitivity pneumonitis and prescribed predonisorone 30 mg/day. Her symptoms and the finding of chest radiograph improved in two weeks. The precipitation antibodies to Lyophyllum karst were positive and the symptoms exacerbated by going to work. Those finding suggests that hypersensitivity pneumonitis in this case. After quitting her job, she has had no relapse by tapering steroids.


Subject(s)
Alveolitis, Extrinsic Allergic/immunology , Basidiomycota/immunology , Occupational Diseases/immunology , Female , Humans , Middle Aged
14.
Am J Surg Pathol ; 28(9): 1193-203, 2004 Sep.
Article in English | MEDLINE | ID: mdl-15316319

ABSTRACT

Sclerosing 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 inflammatory pseudotumor; SC-hepatic IP), but sclerosing pancreatitis (SP) is not infrequently associated with bile duct lesions (sclerosing pancreatitis-associated sclerosing cholangitis; SP-SC). In this study, we compared the histologic changes of hepatic hilar and extrahepatic bile duct lesions of SC (7 cases), SC-hepatic IP (5 cases), SP-SC (5 cases), and typical primary sclerosing cholangitis (PSC) (5 cases). Histologically, all SP-SC cases showed extensive and dense fibrosis with marked lymphoplasmacytic infiltration, many eosinophils, and obliterative phlebitis. Four cases of SC showed bile duct lesions similar to those of SP-SC, whereas other three cases of SC showed milder lymphoplasmacytic infiltration, scant eosinophilic cell infiltration, and no obliterative phlebitis. All SC-hepatic IP cases showed bile duct lesions identical to those of SP-SC. Immunohistochemically, many IgG4-positive plasma cells were found in the bile duct lesions of all SP-SC cases, 4 SC cases with marked lymphoplasmacytic infiltration, and all SC-hepatic IP cases. By contrast, IgG4-positive plasma cells were scarce or hardly found in the remaining 3 SC cases and all PSC cases. In conclusion, 4 SC cases and all SC-hepatic IP cases showed bile duct lesions identical to those of SP-SC, suggesting that these three conditions may be a single disease entity. Their pathogenesis may be similar or closely related to that of SP, and in that respect they may represent an IgG4-related biliary disease. They may respond to steroid therapy as SP does.


Subject(s)
Cholangitis, Sclerosing/complications , Cholangitis, Sclerosing/pathology , Pancreatitis/pathology , Adult , Aged , Cholangitis, Sclerosing/blood , Female , Granuloma, Plasma Cell/complications , Humans , Immunoglobulin G/analysis , Immunohistochemistry , Liver Diseases/complications , Male , Middle Aged , Pancreatitis/blood , Pancreatitis/complications , Plasma Cells/chemistry , Retrospective Studies
15.
Circ J ; 68(4): 308-14, 2004 Apr.
Article in English | MEDLINE | ID: mdl-15056826

ABSTRACT

BACKGROUND: Unstable plaque and coronary arterial thrombi sometimes induce a no-reflow phenomenon after intervention whereby there is sufficient reperfusion. The greater susceptibility of the right coronary artery to development of large thrombi makes successful reperfusion more difficult, therefore the characteristics of the pathological images of coronary arterial thrombi according to the infarct-related coronary artery were investigated. METHODS AND RESULTS: Coronary arterial thrombi were extracted from 77 patients with acute myocardial infarction (AMI) using a thrombectomy catheter. The 36 patients had a thrombus containing atherosclerotic cells. Platelets, fibrin, and neutrophils were seen in all cases. The mean ratios of structural components of thrombi were 51.0 +/- 29.5% (mean +/- SD) of the platelet component, 19.9 +/- 25.7% of the erythrocyte component and 11.9 +/- 22.5% of atherosclerosis component. Erythrocyte-rich thrombi and mixed thrombi mainly composed of erythrocytes were seen in 14 of the 30 cases involving the right coronary artery, 6 of the 35 cases in the left anterior descending artery, 2 of the 11 cases of the left circumflex artery, and in the 1 case of saphenous vein bypass graft. There was significantly more erythrocyte component in the thrombi from the right coronary artery (28.7 +/- 30.1%) than in those from the left coronary artery (12.1 +/- 18.4%). CONCLUSION: Coronary artery thrombi in AMI are composed principally of platelets. Atherosclerotic cells were identified within thrombi from some patients. In the right coronary artery there were many more thrombi that were rich in erythrocytes than in thrombi from the left coronary artery.


Subject(s)
Atherectomy, Coronary , Coronary Thrombosis/pathology , Coronary Vessels/pathology , Aged , Angioplasty, Balloon, Coronary , Blood Platelets , Coronary Angiography , Coronary Thrombosis/diagnostic imaging , Coronary Thrombosis/surgery , Coronary Vessels/surgery , Erythrocytes , Female , Humans , Japan , Male , Middle Aged , Neutrophils
16.
Surg Today ; 33(12): 909-12, 2003.
Article in English | MEDLINE | ID: mdl-14669081

ABSTRACT

We report two cases of breast cancer with endocrine differentiation. Case 1 was a 56-year-old woman with a 2-cm tumor in the upper outer quadrant of the right breast and right axillary lymphadenopathy. Excisional biopsy suggested carcinoma and we performed breast-conserving surgery with lymph node dissection. Histologic examination revealed breast cancer with endocrine differentiation resembling small cell carcinoma of the lung, with one nodal metastasis. Case 2 was a 71-year-old woman with a 2.5-cm tumor in the upper outer quadrant of the right breast. Aspiration cytology suggested carcinoma and we performed mastectomy with lymph node dissection. Histologic examination revealed a carcinoid tumor, as one of the breast cancers with endocrine differentiation, but no nodal metastasis. The two patients are now disease-free 26 and 12 months after surgery, respectively.


Subject(s)
Breast Neoplasms/physiopathology , Carcinoma, Small Cell/physiopathology , Carcinoma/physiopathology , Endocrine System/pathology , Aged , Breast Neoplasms/pathology , Breast Neoplasms/surgery , Carcinoma/pathology , Carcinoma/surgery , Carcinoma, Small Cell/pathology , Carcinoma, Small Cell/surgery , Female , Humans , Middle Aged
18.
Hepatogastroenterology ; 50(49): 27-30, 2003.
Article in English | MEDLINE | ID: mdl-12629984

ABSTRACT

BACKGROUND/AIMS: We analyzed integrin alpha v and c-erbB2 expression and DNA ploidy in primary tumors and pulmonary metastases from colorectal carcinoma. METHODOLOGY: Fifty primary tumors and 21 lung metastases were examined for immunohistochemical detection of integrin alpha v, c-erbB2 and DNA ploidy. RESULTS: In integrin alpha v-positive tumors, integrin alpha v was strongly recognized in the cytoplasm of colorectal carcinoma. In total cases, in a multivariate model using logistic stepwise regression analysis, venous invasion was significantly and independently related to lung metastasis in patients who underwent curative resection. Integrin alpha v, c-erbB2 and DNA ploidy in primary tumors were not predictable for lung metastasis. On the other hand, in the 21 patients with lung metastases, integrin alpha v overexpression was identified in 52.4% (11/21) of primary tumors and in 85.7% (18/21) of lung metastases, respectively. The positive ratio was significantly higher in lung metastases than in primary tumors (p < 0.05). There was no significant difference between primary tumors and lung metastases in DNA ploidy or c-erbB2 expression. CONCLUSIONS: Integrin alpha v might be one of the important signals for formation of lung metastases from colorectal cancer as a late event.


Subject(s)
Adenocarcinoma/genetics , Adenocarcinoma/secondary , Colorectal Neoplasms/genetics , Colorectal Neoplasms/pathology , Gene Expression/genetics , Genes, erbB-2/genetics , Integrin alphaV/genetics , Lung Neoplasms/genetics , Lung Neoplasms/secondary , Ploidies , Adenocarcinoma/surgery , Adult , Aged , Colorectal Neoplasms/surgery , Female , Humans , Lung Neoplasms/surgery , Male , Middle Aged , Neoplasm Staging
19.
J Hepatobiliary Pancreat Surg ; 9(4): 522-6, 2002.
Article in English | MEDLINE | ID: mdl-12483278

ABSTRACT

A 68-year-old man complaining of jaundice was admitted to our hospital in October 1996. Radiological imaging studies, including dynamic computed tomography, endoscopic retrograde cholangiography, and angiography, were highly suggestive of pancreatic head cancer, and laparotomy was performed on October 25, 1996. On gross examination, the pancreas appeared firm, as in chronic pancreatitis, with a mass lesion in the pancreatic head measuring 35 x 35 x 25 mm. A pylorus-preserving pancreatoduodenectomy was carried out. Histological findings were characterized by uniform fibrosis with diffuse lymphoplasmacytic infiltration and lymph follicles in the thickened wall of the bile duct and in and around the pancreas, with acinar atrophy. The histological diagnosis was lymphoplasmacytic sclerosing pancreatocholangitis. Approximately 5 years postoperatively, the patient was alive and well without adjunctive corticosteroid therapy. This positive result suggests that pancreatoduodenectomy may be effective for lymphoplasmacytic sclerosing pancreatocholangitis that is localized in the pancreatic head.


Subject(s)
Cholangitis, Sclerosing/surgery , Pancreaticoduodenectomy , Pancreatitis/surgery , Aged , Cholangiopancreatography, Endoscopic Retrograde , Cholangitis, Sclerosing/pathology , Dilatation, Pathologic , Fibrosis , Humans , Male , Pancreatic Ducts/pathology , Pancreatitis/pathology
20.
Surg Today ; 32(10): 887-90, 2002.
Article in English | MEDLINE | ID: mdl-12376786

ABSTRACT

Malignant fibrous histiocytoma (MFH) is the most common type of soft tissue sarcoma, but it rarely develops as a primary tumor in the breast. Furthermore, no case of the myxoid variant of MFH in the breast has ever been documented. We report the case of a 52-year-old woman with a breast tumor that was immunohistochemically confirmed to be myxoid MFH. She underwent a radical mastectomy and is currently well with no evidence of local recurrence or metastatic spread after 3 years of follow-up.


Subject(s)
Breast Neoplasms/pathology , Histiocytoma, Benign Fibrous/pathology , Breast Neoplasms/metabolism , Breast Neoplasms/surgery , Female , Histiocytoma, Benign Fibrous/metabolism , Histiocytoma, Benign Fibrous/surgery , Humans , Immunohistochemistry , Mastectomy, Radical , Middle Aged
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