A Case of Nontuberculous Mycobacteria Infection That Was Misdiagnosed as an Invasion of Wegener's Granulomatosis / 대한류마티스학회지

Wegener's granulomatosis is a rare disease that pathologically causes necrotizing granulomatous vasculitis in the arterioles and venules and it can invade the whole body. In addition, it is difficult to distinguish between a nontuberculous mycobacteria infection that shows manifestations of granuloma and Wegener's granulomatosis. There has been no reported on a patient who had the 2 abovementioned two diseases at the same time. A 69 year old male patient had Wegener's granulomatosis that had invaded the prostate. He also had a scrotal swelling and back pain. He had manifestations of granulomatous infection on the scrotum and spine biopsies. However, there was no clinical evidence of Wegener's granulomatosis. As a result, we examined him for other diseases that can cause a granuloma. Consequently, he was also diagnosed as suffering with a nontuberculous mycobacteria infection. We report here on this case and we review the relevant medical literature.

A Case of Licorice Induced Hypokalemic Paralysis with Rhabdomyolysis / 대한내분비학회지

Prolonged ingestion of licorice can cause hypermineralocorticoidism, with sodium retention, potassium loss and hypertension. Nevertheless, its initial presentation with a very severe degree of hypokalemic paralysis and rhabdomyolysis are exceedingly rare. We describe a patient who experienced hypokalemic paralysis and rhabdomyolysis after licorice ingestion. The patient's initial blood pressure was 160/80mmHg. The major biochemical abnormalities included; hypokalemia(K+ 1.3mEq/L), metabolic alkalosis, with a pH of 7.64, and urine myoglobin > 3000ng/mL. The plasma rennin activity and aldosterone level were suppressed. The 24 hour urine cortisol concentration was normal. The patients, over a 1 month period, had ingested 500g of licorice boiled in water. After quitting the licorice, the hypokalemia and muscle paralysis gradually improved and blood pressure returned to normal

A Case of Hyperthyroid Myopathy Due to Graves' Disease

A 49-year-old woman was admitted with a three-month history of myalgia and progressive proximal and distal muscle weaknesses. Physical examination showed diffuse enlargement of the thyroid gland and mild muscle atrophy. Serum creatine kinase was slightly increased and electromyography showed a myopathic pattern. Muscle biopsy showed nonspecific myopathic changes. Serum thyroid-stimulating hormone was very low, whereas thyroxine (T4) was greatly increased as well as anti-thyroglobulin, anti-microsome, and TSH-receptor antibody. The patient's symptoms were improved during the treatment with propilthiouracil.

A Case of Graves' Disease with Spuriously Elevsted TSH due to Interference of Heterophilin Actibodies / 대한내분비학회지

Thyroid-stimulating hormone (TSH) is the most sensitive marker reflecting thyroid function. TraditionaUy, TSH concentration was measured by the method of RadioImmunoAssay (RIA) with the detection limits around 1 to 2 mIU/L, which was unable to differentiate hyperthyroid status. Since 1980s, owing to the sensitive assay for TSH, immunoradiometric assay (IRMA), it has been possible to detect low concentration of TSH by 0.001 mlU/L. TSH is composed of two glycopeptide subunits, a-subunit and B-subunit. Monoclonal antibodies, directed against two different sites of the TSH peptides, are used in IRMA. One antibody is directed toward the specific B-subunit of TSH molecule and is used to extract it from serum, a second antibody labelled with a radioactive material is then attached to the separated TSH to form "sandwhich" molecule that can be measured. Generally, mouse monoclonal antibodies are used as capture and detection antibodies. Infrequently, when there is heterophilic antibody, i.e. human anti-mouse antibody (HAMA), TSH can be measured as spuriously elevated, since HAMA may form a link between the signal and capture molecules. We report a case of inappropriately elevated TSH concentration due to heterophilic antibody, later diagnosed as Graves disease. A 41-year-old woman visited our clinic with the chief complaints of hand tremor, hyperphagia, weight loss for 3 months. Two years earlier, she underwent total colectomy due to colon cancer and had treat on multiple chemotherapies. The results of thyroid function test shows that TSH was 0.77 mIU/L, free T was 7.1 ng/dL (0.8~1.9), free T was 11.3 pg/mL (0.2~5.5). Thyroid specific auto- antibody results were anti-Tg-Ab 21.3 m/mL(0 100), anti-TPO-Ab 87.9m/mL(0100), TBIAb 7.8% (-15/15). Thyroid scan showed that radioactiveiodine uptake was increased and thyroid gland wasenlarged diffusely. Because TSH level was elevated, further evaluations were performed to differentiate with TSH producing pituitary tumor and pituitary resistance to thyroid hormone. Sellar MRI was normal, TRH stimulation test showed flat response. Since spurious elevation of TSH is possible at the presence of hetrophilic antibody, we rechecked TSH concentration after adding mouse monoclonal antibody to the patients serum with result of TSH less than 0.05 mIU/L. She was able to be diagnosed as Graves disease, and started with methimazole. Three months later, thyroid function test showed that TSH was 10.5 mIU/L, free T4 was 1.0 ng/dL, free T3 was 4.0 pg/mL. TSH level after removal the effect of heterophilic antibody with mouse monoclonal antibody was 0.71 mIU/L. Neutropenia was developed 5 months after methimazole therapy, to stop antithyroid medication. With the plan of radioactive iodine therapy if she relapses, she is being followed with periodic thyroid function test. We report a case of Graves disease with spuriously elevated TSH due to the effect of heterophilic antibodies.

Effect of Radioactive Iodine Therapy in Patients with Scan-Negative, Thyroglobulin-Positive Thyroid Cancer / 대한내분비학회지

BACKGROUND: After total thyroidectomy the presence of detectable serum thyroglobulin(Tg) concentration is an index of residual or metastatic thyroid tissue and is usually well correlated with positive I whole body scan. However, it is not rare to find a patient with detectable serum Tg levels but without any uptake on I whole-body scan. At present it is not certain how to manage such patients. We performed whole body scan after administration of therapeutic dose of 131I to evaluate the usefulness of radioactive iodine therapy in the above setting. METHODS: Fifteen patients (4 males and 11 females, ranging in age from 17 to 74 years) were studied. They had been previously treated with total thyroidectomy for papillary thyroid cancer followed by therapy with 131I for ablation of their thyroid residue. Tg levels were determined by immunoradiometric assay method. 131I (100-200 mCi) therapy was administered and whole body scan was performed. 99mTc MIBI scans were taken in 9 patients. Follow up data of Tg were available in 12 patients at time interval of 6 12 months from the first study and treatment. RESULTS: Tg(on) levels of these patients were in a range of 2.2210 ng/mL (mean 36.1 +/- 59.1 ng/mL) and Tg(off) levels were 17.3 1,592 ng/mL (mean 197.3 +/- 400.3 ng/mL). After radioiodide therapy, Tg(on) levels were in 1.48.5 ng/mL (mean 11.0 +/- 13.5 ng/mL), Tg (off) were 11.9 478.0 ng/mL (mean 159.3 +/- 159.8 ng/mL). The Tg (on) levels were decreased significantly after RAI therapy, but Tg (off) levels had no significant difference, In 8 of the 15 patients (53.3%), posttreatment whole body scan showed definite positive uptakes which were not evident in pretreatment diagnostic scan. There were local recurrence in 3 cases, regional lymph node metastasis in 4 cases, and lung in I case. Diffuse hepatic uptake was definitely seen in 7 cases. The MIBI scan showed abnorml uptakes in 4 of 9 cases. CONCLUSION: The therapeutic usefulness of 100 to 200 mCi of 131I treatment in patients with 131I scan-negative and Tg-positive was unclear. And the MIBI scan was only partially effective. Further studies with other diagnostic and therapeutic approachs are required to evaluate the exact lesions and to improve prognosis.

Allelotyping and Comparative Genomin Hybridization Studies in Papillary Thyroid Carcinomas and Follicular Adenomas / 대한내분비학회지

BACKGROUND: In our previous study, the prevalence of the known causes of thyroid tumorigenesis was relatively rare in Korean population, suggesting genetic and environmental differences exist. Screening of genetic alteration in papillary thyroid carcinoma(PTC) and follicular adenoma(FA) in whole genomic scale was needed prior to search on individual genes of possible causes. METHODS: Ten cases of PTC without ret/PTC-I, -2, -3 rearrangement and 5 cases of follicular adenoma were included in the study of microsatellite marker allelotyping. Sixty two microsatellite markers available, were chosen to cover the known sites of loss of heterozygosity(LOH) involved in thyroid tumors, tumor suppressor genes and terminal portion of each chromosomes. PCR was performed on tumor DNA and leukocytes DNA from each patient with MDE gel electrophoresis to detect LOH. Same specitnens as above, 3 case of normal thyroid tissues and NPA, ARO cell lines were included in the study of comparative genomic hybridization(CGH). Tumor and control DNAs were hybridized to metaphase chromosome with differential stainings with fluorescein and rhoda-mine-dUTP. Obtained results were analyzed by multicolor fluorescence computer assisted image analyzer. RESULTS: In allelotyping, LOH were detected in 5 cases of PTC, 2 cases on D10S1435, 1 case each on D2S1780, DSS1099, D11S1986, D16S539, 1 case of PTC revealed LOH on DSS1099, D11S1986. In FA, LOH were detected in 3 cases on D1S534, D1S226, Dl 1S907, D22S683, DXS9807. In CGH, Xp addition was noticed in 1 case of PTC, 12q and 10p addition was noticed in 1 case each, 16q deletion and 17q addition in 1 case of FA. CONCLUSION: No hot spot of LOH was noticed in microsatellite marker allelotyping, neither of common chromosomal change in CGH study suggesting unbalanced translocation or gene amplification more than 5-10 Mb may be involved in the genetic alteration of PTC and FA.

Percutaneous Ethanol Injection in Benigh Thyroid Nodules / 대한내분비학회지

BACKGROUND: Percutaneous ethanol injection(PEI) performed in guidance of ultrasonography has been used in cases of thyroid cyst and autonomous functiong thyroid nodule(AFTN). We performed this study to determine the feasibility of PEI on the various type of benign cold nodules(solid, pure cyst, complex cyst) and AFTN. METHOD: Ninety patients(age 41+12 years; 83 women and 7 men) with hot and cold nodule were included in this study. All cases were subjected to FNAB, and sono-guided in cases of complex cyst, at least twice with results of colloid nodule. T4 suppression treatment was done for cold solid nodules for at least 6 months and cases which had partial response(50% or more volume reduction but no further volume decrease on T4 suppression) were included in this study. After PEI, we classified AFTN into three different response groups; complete response (normali- zation of TFT and thyroid scan finding), partial response(normalized freeT4 but suppressed TSH or persistently suppressed scan), or failure group. Each subtypes(solid, pure cyst, complex cyst) of cold nodules were also classified into three groups in accordance with volume reduction; complete response(above 90% of volume reduction), partial response(50-90%), and failure(below 50% or increase in size) group. RESULTS: Overall responses rate of PEI for benign thyroid nodule were complete; 70(78%), partial; 18(20%), and failure; 2(2%). In 27 cold solid nodules, complete response was observed in 23(85%) and partial response in 4(15%). In 11 pure cysts, complete response was observed in 7(64%), partial response in 3(27%), and 1 case(9%) of failure was lost after 1 trial of PEI. In 45 cases of complex cyst, complete response was observed in 36(80%), partial response in 8 (18%), and 1 case(2%) of failure was subjected to operation with pathology report of Hiirthle cell adenoma. In 7 cases of AFTN, complete response was observed in 4(57%) and partial response in 3(43%). We observed complications during PEI therapy such as transient neck pain(n=10), transient unilateral vocal cord palsy(n=l), and intracavitary hemorrhage(n=2), transient hypotension during ethanol injection(n 1). CONCLUSION: Our data suggest that efficacy of PEI(57%) in AFTN is inferior to conventional therapies like surgery and radioiodine, but still can be an alternative therapeutic modality in selected cases. In cold nodules, especially of solid type and complex cyst, PEI may have feasibility as a therapeutic modality in restricted cases. Further studies of prolonged follow-up for the possibility of neglecting occult malignancy are warranted.

The Expression of the Bcl-2 Family Proteins in Thyroid Neoplasms / 대한내분비학회지

BACKGROUND: Proteins of the Bcl-2 family are intracellular membrane-associated proteins that regulate programmed cell death either positively or negatively by as yet unknown mechanism. Bcl-2 family proteins have an antiapoptotic function, such as the Bcl-2, the long form of Bcl-x and Mcl-l, or a proapoptotic function, like the short form of Bcl-x and Bax. To investigate the potential role of Bcl-2 family proteins in thyroid tumorigenesis, the authors examined the pattern of expression of the Bel-2 family proteins in various thyroid neoplasms. METHODS: Bcl-2 family proteins, including Bcl-2, Bcl-x, Mcl-1 and Bax proteins were immunohistochemically stained in 57 cases of various thyroid neoplasms using formalin-fixed and paraffin embedded tissues; 18 cases of papillary carcinoma, 6 cases of medullary carcinoma, 4 cases of anaplastic carcinoma, 10 cases of follicular adenoma, 9 cases of adenomatous goiter, and 10 autopsy cases of fetal thyroid galnd. The intensity and frequency of the immunostaining were evaluated with the program of Image-Pro Plus Version 3.0 for image analysis. RESULT: Consistent expression of Bcl-2, Mcl-1, and Bax proteins were present in the surrounding normal thyroid tissue, however the expression of Bcl-x protein was not observed. Compare to the expression patterns of adenomatous goiter, and fetal and surrounding normal thyroid tissues, papillary and anaplastic carcinomas showed the decreased Bcl-2 and increased Bcl-x protein expressions(p (0.05). Medullary carcinoma revealed the increased Bcl-x protein expression only(p 0.05). CONCLUSION: These data suggest that combined patterns of decreased Bcl-2 and increased Bcl-x protein expressions may eontribute to the carcinogenesis of thyroid cancers originated from thyroid follicular cells, and an increased expression of Bcl-x protein may be related to the pathogenesis of medullary carcinoma from parafollicular C cells.

A case of giant hemangioma of the liver presenting with fever of unknown origin

A 37 year-old-woman was admitted to the hospital because of 15 days' duration of continuous fever. Routine studies for detection of fever foci were negative. Imaging studies revealed giant hemangioma of the liver with central thrombosis. The fever persisted for a period of 4 weeks, and subsided after conservative management. We report a case of hepatic hemangioma presenting with fever of unknown origin. The condition is very rare, but should be regarded as one of the causes of fever of unknown origin.