1/24. Minimal-change nephrotic syndrome and acute renal failure in a patient with aged onset insulin-dependent diabetes mellitus and autoimmune thyroiditis.A 61-year-old woman with a 2-year history of insulin-dependent diabetes mellitus (IDDM) developed nephrotic syndrome. Renal biopsy showed minimal-change nephrotic syndrome (MCNS), and no evidence of diabetic glomerulosclerosis. Although steroid therapy was initiated, plasma urea and creatinine rose and hemodialysis was required. After 4 weeks, she responded to steroids and her renal function returned to normal. MCNS, which is not associated with diabetic glomerulosclerosis, has rarely been seen in IDDM patients with nephrotic syndrome. Her human leukocyte antigen typing was A24, BW52, BW61, DR2 and DR9. This typing has been reported to be associated with both IDDM and renal disease.- - - - - - - - - - ranking = 1keywords = antigen (Clic here for more details about this article) |
2/24. Hashimoto's disease during interferon-alpha therapy in a patient with pre-treatment negative anti-thyroid autoantibodies and with the specific genetic susceptibility to the thyroid disease.OBJECTIVES: The authors described a case of Hashimoto's disease during interferon-alpha (IFN-alpha) treatment for chronic viral C hepatitis in a patient with the specific genetic susceptibility associated with the thyroid disease. RESULTS: A 60-year-old woman with chronic active viral C hepatitis (HCV genotype = 3a) started IFN-alpha therapy in November '96. Before treatment thyroid function tests were normal and anti-thyroid (anti-thyroglobulin and anti-thyroid peroxidase) Abs were negative. During IFN therapy, serum aminotransferases fell within the normal range and viremia (serum HCV-rna) became negative after one year. After 20 months, the patient presented clinical features of primary hypothyroidism. Anti-thyroid Abs were found positive. Hormonal, ultrasonographic, radioiodine scanning and fine needle aspiration findings were consistent with the diagnosis of Hashimoto's thyroiditis. The tissutal typing of the patient showed the presence of Human Leukocyte Antigen (HLA) DRB1*11 gene (corresponding to DR5 antigen). IFN-alpha therapy was suspended and a treatment with l-T4 started. Chronic viral infection relapsed after the suspension of the IFN-alpha therapy. CONCLUSIONS: This case report showed that the clinical appearance of Hashimoto's disease after IFN-alpha therapy for chronic C hepatitis in our patient was associated with a specific genetic predisposition (DR5) for this pathology. Further studies are necessary to evaluate whether the study of hla antigens may be a very useful tool to detect the patients with a predisposition to develop autoimmune thyroiditis, in order to make a early diagnosis of thyroid disorders during the IFN-alpha treatment.- - - - - - - - - - ranking = 2keywords = antigen (Clic here for more details about this article) |
3/24. Macroamylasemia attributable to gluten-related amylase autoantibodies: a case report.BACKGROUND: Macroamylasemia (MA) is a benign condition caused by circulating macroamylase complexes of pancreatic or salivary amylase bound to plasma proteins, which cannot be cleared by the renal glomeruli. In most cases, the macromolecular amylase represents a complex of normal amylase and either immunoglobulin a or G and may be a specific antigen-antibody complex. celiac disease (CD) is a permanent intolerance to ingested gluten that results in immunologically mediated inflammatory damage of the small intestinal mucosa. Several recent population-based serologic surveys have shown CD to be a common disorder, possibly affecting 1 in 200 to 250 individuals in most countries studied, including the united states, where overt CD is rare, indicating a high proportion of subclinical disease. The diagnosis of CD currently rests on the histological demonstration of the characteristic lesion in the small intestine and the subsequent clinical response to the introduction of a gluten-free diet. MA associated with CD has been described in adult patients, and in a few cases, MA decreased or resolved after a strict gluten-free diet. A few single cases of MA have been described in childhood, but no association with CD has been reported so far. We report a girl with CD, autoimmune thyroiditis, and MA, in whom CD-related antibodies to amylase and to exocrine pancreas tissue resolved with a gluten-free diet. CASE REPORT: An 11-year-old girl was referred for chronic abdominal pain and growth retardation associated with persistent hyperamylasemia and suspected chronic pancreatitis. We confirmed elevated serum amylase, normal serum lipase, and very low 24-hour urine amylase and amylase clearance/creatinine clearance ratio, consistent with MA. serologic tests for CD were positive, and the diagnosis was confirmed by small bowel biopsy showing subtotal villous atrophy. thyroid function tests showed a pronounced hypothyroidism, associated with high titers of thyroid microsomal and thyroglobulin antibodies. Screening for other autoantibodies-including antinuclear, islet cell, glutamic acid decarboxylase, protein tyrosine phosphatase islet antigen 512, adrenal gland, and cytoplasmic neutrophil granulocyte antibodies-was negative. A diagnosis of CD, MA, and hypothyroidism attributable to autoimmune thyroiditis was made. A gluten-free diet and oral replacement with L-thyroxine was started with clinical improvement. serum amylase and amylase clearance/creatinine clearance ratio normalized, consistent with resolution of MA. STUDY DESIGN AND methods: The patient's serum samples were obtained at the time of CD diagnosis and at 3 and 12 months after instituting a gluten-free diet. serum samples from 10 consecutive untreated celiac children were disease controls, and 39 participants with no gastrointestinal symptoms and no family history of CD served as healthy controls. The origin of MA as determined by complexes of amylase with circulating immunoglobulins was tested by the measurement of amylase on supernatants after precipitation of immune complexes with either protein A sepharose or polyethylene glycol. The precipitation of >60% of amylase activity was consistent with the presence of MA. immunoglobulin g (IgG) and immunoglobulin a (IgA) circulating autoantibodies to amylase were measured using recently developed enzyme-linked immunosorbent assay (ELISA), using porcine amylase as antigen. Results were expressed as arbitrary units (AUs). Statistical analysis was performed by Student's t test for unpaired data. IgA and IgG antibodies to exocrine pancreas tissue were detected by indirect immunofluorescence on human pancreas cryosections. RESULTS: serum immunoprecipitation with either protein A sepharose or polyethylene glycol reduced amylase activity from 1698 to 89 U/L (94.8%) and to 75 U/L (95.6%), with only marginal reduction in control serum samples. The ELISA for autoantibodies to amylase detected high values, both IgA (3531 AU) and IgG (1855 AU), in the serum sample from the patient at CD diagnosis. IgA autoantibodies (mean /- standard deviation) were 3.4 /- 2.5 AU in healthy controls, and 2.1 /- 1.2 AU in celiac controls; IgG autoantibodies were 10 /- 4.8 AU in healthy controls and 8.5 /- 3.2 AU, respectively. autoantibodies to exocrine pancreas tissue were documented in patient sera at the time of CD diagnosis, both IgA and IgG, but not in control groups. Preincubation of patient's serum with excess of alpha-amylase specifically inhibited antibody binding to coated amylase in the ELISA, and partially inhibited immunoreactivity to exocrine pancreas. autoantibodies to alpha-amylase and to exocrine pancreas declined in CD patients after institution of a gluten-free diet. CONCLUSIONS: Few cases of MA have been described in children, and in all amylase determination was part of the clinical investigation for abdominal pain or trauma. (ABSTRACT TRUNCATED)- - - - - - - - - - ranking = 3keywords = antigen (Clic here for more details about this article) |
4/24. association of the HLA A2-B46-DR9 haplotype with autoimmune thyroid dysfunction after bone marrow transplantation in Chinese patients.Autoimmune thyroid disease (AITD) may occur in patients after bone marrow transplantation (BMT). At a median follow-up of 4 years, among 194 allografts and 28 autografts, four patients (three allografts, one autograft) developed AITD. All carried the human leucocyte antigen (HLA) A2-B46-DR9 haplotype, strongly associated with AITD in the Chinese population. No significant thyroid disorder was detected in 190 patients without this haplotype. The frequency of AITD in BMT patients with the HLA A2-B46-DR9 haplotype was 12.5%, with a relative risk of 7.8 times that of non-carriers (P < 0.001). The risk of AITD should be recognized in recipients with high-risk HLA haplotypes, and regular screening might be warranted.- - - - - - - - - - ranking = 1keywords = antigen (Clic here for more details about this article) |
5/24. PBC-AIH overlap syndrome with concomitant ITP and Hashimoto's disease with positivity for anti-centromere antibody.We report a case of primary biliary cirrhosis (PBC)-autoimmune hepatitis (AIH) overlap syndrome with concurrent idiopathic thrombocytopenic purpura (ITP) and Hashimoto's disease with positivity for anticentromere antibody. The patient was a 64-year-old woman with symptoms of jaundice and general fatigue. About 30 years earlier, she had been diagnosed as having ITP and had undergone splenectomy. As part of her present history, she had exhibited liver dysfunction in 1995, during the follow-up of Hashimoto's disease, and a liver biopsy led to the diagnosis of PBC. In March 2000, she was admitted to hospital because of general fatigue and jaundice. blood tests revealed: total protein (TP), 6.6 g/dl; gamma-globulin (glb), 35.9%; total bilirubin (T-bil), 9.41 mg/dl; direct bilirubin (D-bil), 7.52 mg/dl; aspartate aminotransferase (AST), 957 U/l; alanine aminotransferase (ALT), 651 U/l; alkaline phosphatase (ALP), 595 U/l; gamma-guanosine triphosphate (GTP), 129 U/l; IgG, 2620 mg/dl; IgM, 223 mg/dl; hepatitis b surface antigen (HBsAg), negative; anti-hepatitis c virus (HCV), negative; antinuclear antibody, positive; antimitchondrial antibody (AMA), negative (by the immunofluorescence [IF] method); and anti-pyruvate dehydrogenase complex (PDC)-E2 antibody, positive (by Western blotting). Anticentromere antibody (ACA), which is an alternative diagnostic marker for PBC, was detected in this patient. prednisolone was administered after admission and liver function test results improved markedly. The liver biopsy in 1995 had revealed infiltration of lymphocytes and plasma cells in the portal areas with fibrous expansion and periportal necrosis. Destructive cholangitis was observed, as well as scattered epitheloid cell granulomas in some portal areas. liver biopsy after the steroid treatment revealed alleviated necrotic inflammatory responses of hepatocytes, while the destructive cholangitis persisted. This is a very rare case of PBC-AIH overlap syndrome accompanied by ITP and Hashimoto's disease which provides a possible insight into the mechanisms and interplay of autoimmune diseases.- - - - - - - - - - ranking = 1keywords = antigen (Clic here for more details about this article) |
6/24. Postparathyroidectomy transient thyrotoxicosis.Three patients are described who had spontaneously resolving transient thyrotoxicosis after resection of a parathyroid adenoma without thyroidectomy or an apparent thyroid abnormality before or during surgery. All had documented thyrotoxicosis that developed within 2 weeks after surgery, which was clinically symptomatic in two of three patients. The thyrotoxicosis was associated with subnormal radioactive iodine thyroid uptake when performed in the two symptomatic patients and was consistent with a postsurgical inflammatory etiology secondary to thyroid gland trauma during parathyroidectomy. In all patients, the clinical and biochemical evidence of thyrotoxicosis resolved within 2 months. Antithyroglobulin and antimicrosomal antibodies were not detected in the two patients who had a complete recovery 3 months after surgery. However, in the patient who had autoimmune thyroiditis, hyperthyroidism due to Graves' disease subsequently developed 19 months after parathyroidectomy and was associated with increasing titers of antithyroglobulin and antimicrosomal thyroidal autoantibodies. From these observations, we conclude that 1) spontaneously resolving transient thyrotoxicosis of varying severity may occur in some patients after parathyroidectomy, which could be secondary to intraoperative thyroid gland manipulation, and 2) while the occurrence of subsequent Graves' hyperthyroidism in a patient with underlying autoimmune thyroiditis may have been a coincidence, this observation also raises the possibility that thyroidal autoantigen released during parathyroidectomy may trigger the reactivation of autoimmune thyroid disease in a predisposed subject.- - - - - - - - - - ranking = 1keywords = antigen (Clic here for more details about this article) |
7/24. Symptomatic C-cell hyperplasia associated with chronic lymphocytic thyroiditis.A 58-year-old euthyroid man with episodic flushing and a 2-year history of progressive wheezing was found to have a hypoechoic lesion in one lobe of his thyroid and hypercalcitoninemia in response to pentagastrin stimulation. Thyroidectomy revealed bilateral C-cell hyperplasia unexpectedly associated with chronic lymphocytic thyroiditis. The C-cells exhibited positive immunohistochemical staining for calcitonin and polyclonal carcinoembryonic antigen (CEA). Postoperatively, the wheezing and flushing subsided and the serum calcitonin level was not elevated with pentagastrin stimulation. The substance or substances responsible for the wheezing and flushing were not specifically identified. Nine other specimens of chronic lymphocytic thyroiditis were examined for C-cell hyperplasia and two had small hyperplastic foci, but of a lesser degree than the index case. These patients did not exhibit wheezing and flushing. The development of C-cell hyperplasia in chronic lymphocytic thyroiditis is uncommon and the mechanism for its occurrence is unexplained. This patient appears to be the first reported case of symptomatic C-cell hyperplasia associated with chronic lymphocytic thyroiditis. The substance or substances responsible for the clinical symptoms remain to be identified.- - - - - - - - - - ranking = 1keywords = antigen (Clic here for more details about this article) |
8/24. Mucoepidermoid carcinoma of the thyroid gland.An extremely rare case of mucoepidermoid carcinoma of the thyroid in a 56-year-old woman is presented. The patient clinically having Hashimoto's thyroiditis was noted a nodule in her neck. The tumor was sited in the midportion of the left lobe of the thyroid, and histologically it showed both squamous features and mucin production. The squamous cells were arranged in solid sheets with horny pearls and the mucous cells tended to line dilated duct-like elements. Ultrastructurally, the epidermoid cells had aggregates of tonofilaments and well-developed desmosomal attachments, and the mucous cells contained numerous mucin granules in their cytoplasm. Immunohistochemical studies revealed that cytokeratin antibodies showed positivity for both the lining cells and squamous cells, whereas carcinoembryonic antigen positivity was found in the lining cells and intraluminal material. The tumor cells were negative for thyroglobulin, calcitonin, vimentin, chromogranin, and neuron-specific enolase. These unusual histologic and immunohistochemical features are suggestive of a tumor related to the so-called "solid cell nest" of the thyroid.- - - - - - - - - - ranking = 1keywords = antigen (Clic here for more details about this article) |
9/24. Autoimmune thyroiditis following interleukin-2 and LAK cell therapy for metastatic renal cell carcinoma: correlation with tumor regression.A 63-year-old woman receiving recombinant interleukin-2 (rIL-2) lymphokine activated killer cells for metastatic renal cell carcinoma developed autoimmune thyroiditis with clinical hypothyroidism and high titer anti-thyroglobulin and anti-microsomal antibodies. The onset of thyroid dysfunction was associated with tumor regression and resulted in complete response at the end of the treatment. Cytologic and cytofluorimetric studies on thyroid tissue showed two distinct populations, mainly consisting of small lymphocytes and large thyrocytes, and the latter expressed MHC class II antigens. After completion of rIL-2 treatment, hypothyroidism gradually decreased until resolution; complete tumor remission lasted 18 months. Mechanisms underlying the association between autoimmune thyroiditis and cancer regression are discussed.- - - - - - - - - - ranking = 1keywords = antigen (Clic here for more details about this article) |
10/24. Thyroid hormone autoantibodies (THAA) in three sisters: a four-year follow up.We have found three sisters who had thyroid hormone autoantibodies (THAA) in serum. Two of them were hypothyroid due to Hashimoto's thyroiditis. They were treated with synthetic thyroxine (T4) (Case 1) or T4 and triiodothyronine (T3) (Case 2). The other sister (Case 3) was euthyroid throughout the investigation period. Changes in the titers of THAA, antithyroglobulin (Tg) antibodies as well as in the concentrations of thyrotropin (TSH), and total and free thyroid hormones (T3, T4, FT3, and FT4) in sera from each patient were examined for four years. A significant positive correlation was observed between titers of THAA and anti-Tg antibodies in both Case 1 (anti-T3 and anti-T4) and 2 (anti-T4). On the other hand, a significant negative correlation between titers of anti-T3 and anti-Tg antibodies was observed in Case 3. These results suggest that antigen of THAA might be Tg in Case 1 and 2 and mechanism(s) of the production of THAA of Case 1 might be different from that of Case 1 and 2 in terms of their antigen or immune recognition of Tg.- - - - - - - - - - ranking = 2keywords = antigen (Clic here for more details about this article) |
| | Next -> |