We report here on a rare case of adrenal paragonimiasis that presented with an adrenal incidentaloma. A 52-year-old male presented with fatigue and weight loss. The laboratory findings revealed eosinophilia (8.5%) and an increased eosinophil count (910/microL). The computed tomography scan showed 6.5 x 5 cm sized multilocular cystic mass in the right adrenal gland, which was non-functioning, and cystic lesions with variable sizes were also noted in the abdominal cavity. On the surgical field, whitish patches were spread out in the peritoneum, the omentum, the dome of the liver and the diaphragm. The right adrenal gland was replaced by a cystic mass filled with mucopurulent creamy materials. The pathologic findings revealed numerous eggs of Paragonimus spp. Also, the ELISA was positive for IgG paragonimus antibody. The adrenal gland can become infected by various microbial pathogens, including parasites, although it is relatively uncommon. However, in the case of a cystic adrenal mass with accompanying eosinophilia in an endemic area, clinicians should consider the possibility of parasitic infection.
Abdominal Cavity ; Adrenal Gland Neoplasms ; Adrenal Glands ; Diaphragm ; Eggs ; Enzyme-Linked Immunosorbent Assay ; Eosinophilia ; Eosinophils ; Fatigue ; Humans ; Immunoglobulin G ; Liver ; Male ; Middle Aged ; Omentum ; Ovum ; Paragonimiasis ; Paragonimus ; Parasites ; Peritoneum ; Weight Loss

Intra-operative parathyroid hormone (IOPTH) assay is a useful tool to confirm complete excision of all hyper-functioning parathyroid gland tissue. In this report, we describe a case with successful localization of distant metastasis in a patient with parathyroid carcinoma using the IOPTH assay. A 53-year-old man presented to our clinic with a serum calcium level of 11.8 mg/dL and an intact PTH level of 233.3 pg/mL. He had been treated for parathyroid carcinoma eleven years ago. Two suspected metastatic lesions were detected on the chest computed tomography. Due to the vastly different surgical field necessary to excise each lesion, we preferentially removed only one lesion and we monitored the other remaining suspected lesion without resection via IOPTH assay. Six months later, the patient's serum calcium and intact PTH levels remained within their normal ranges. To the best of our knowledge, this is the first case to effectively utilize IOPTH assay for the management of metastatic parathyroid carcinoma.
Calcium ; Humans ; Middle Aged ; Neoplasm Metastasis ; Parathyroid Glands ; Parathyroid Hormone ; Parathyroid Neoplasms ; Reference Values ; Thorax

Von Hippel-Lindau (VHL) disease is an autosomal dominant disorder that results from a germline mutation of the VHL gene. The affected individuals might develop several benign or malignant tumors such as central nervous system or retinal haemangioblastomas, endolymphatic sac tumors, renal cell carcinomas, pheochromocytomas or pancreatic cysts and neuroendocrine tumors. We report here on a case of a 21 year old female with von Hippel-Lindau disease and she presented with only pancreatic neuroendocrine tumor and no evidence of haemangioblastomas or other visceral complications. Further, direct sequencing of the VHL gene reveals a novel germline frameshift mutation of codon 198 from the deletion of nucleotide 592 (cytosine), leading to truncation of the VHL protein.
Carcinoma, Renal Cell ; Central Nervous System ; Codon ; Endolymphatic Sac ; Female ; Frameshift Mutation ; Germ-Line Mutation ; Humans ; Neuroendocrine Tumors ; Pancreas ; Pancreatic Cyst ; Pheochromocytoma ; Retinaldehyde ; von Hippel-Lindau Disease

We report here on a case of genetically confirmed type Ia glycogen storage disease (GSD) that was diagnosed in the military hospital. A twenty-year old soldier was admitted to the hospital with abdominal fullness. He had a past medical history of hepatomegaly that was firstly recognized at six months after birth, and he had been followed-up at an outpatient clinic with the presumptive diagnosis of type III GSD. He also had a history of growth hormone therapy because of growth retardation. However, he arbitrarily refused medical observation from 14 years of age. On the physical examination, the height of the patient was 163.1 cm and significant hepatomegaly was observed. Significantly abnormal liver-associated paramters were observed on the laboratory findings and multiple hepatic adenomas were observed on the CT exam and MRI scan. To determine the proper treatment, we tried to confirm the exact type of GSD in the patient. By mutational analysis, we found the c.648G>T homozygote splicing mutation in the G6PC gene and the patient was confirmed as having the type Ia GSD.
Adenoma ; Ambulatory Care Facilities ; Chromosome Disorders ; Glycogen ; Glycogen Storage Disease ; Growth Hormone ; Hepatomegaly ; Homozygote ; Hospitals, Military ; Humans ; Magnetic Resonance Imaging ; Military Personnel ; Parturition ; Physical Examination

Although pulmonary metastasis of thyroid cancer is not uncommon, it mostly occurs as multiple discrete nodules on the lung parenchyma. Because thyroid cancer presenting with an isolated large lung mass is extremely rare and the diagnosis is frequently based on small pieces of tissue obtained by a fine needle, the wrong diagnosis such as lung cancer is prone to be made. A 60-year-old man was admitted for evaluation of a lung mass detected on chest radiography. Cytological examination of the bronchial washing specimens suggested adenocarcinoma. Surgery for early lung cancer was performed considering that no other abnormalities were found during the work-up that included 18-fludeoxyglucose positron emission tomography computer tomography (18FDG-PET/CT). Unexpectedly, the diagnosis of papillary thyroid cancer with lung metastasis was made, which prompted us to evaluate the thyroid gland and then remove the primary cancer by subsequent operation. Although it is uncommon, physician should be aware of this possibility, which could help to avoid the wrong diagnosis. Here we report on a typical case of solitary pulmonary metastasis of thyroid cancer and we summarize the previously reported cases with a review of the relevant literature.
Adenocarcinoma ; Humans ; Lung ; Lung Neoplasms ; Middle Aged ; Needles ; Neoplasm Metastasis ; Positron-Emission Tomography ; Thorax ; Thyroid Gland ; Thyroid Neoplasms

Pituitary abscess is a rare pathology, but it is a potentially life-threatening condition. Therefore, timely intervention, including antibiotics and an operation, can prevent the morbidity and mortality in such cases. A 31-year-old woman, who was 16 months after her second delivery, presented with intermittent headache for 3 months. Amenorrhea, polyuria and polydipsia were noticed and the endocrinological hormone studies were compatible with panhypopituitarism and diabetes insipidus. Pituitary MRI demonstrated a 2.3 cm sized cystic mass with an upper small nodular lesion. Her symptoms such as headache and fever were repeatedly improved whenever corticosteroid was administered, which led us to suspect the diagnosis of an inflammatory condition like lymphocytic hypophysitis. During the hormone replacement therapy, her cystic pituitary mass had grown and her symptoms progressively worsened for another two months. The patient underwent trans-sphenoidal exploration and she turned out to have a pituitary abscess. At the 3-month follow-up, amenorrhea was noticed and her residual function of the pituitary was tested by a combined pituitary stimulation test. The results were compatible with panhypopituitarism. She received levothyroxine 100 microg, prednisolone 5 mg and desmopressin spray and she is being observed at the out-patient clinic. The authors experienced a patient with primary pituitary abscess that was confirmed pathologically and we report on its clinical course with a literature review.
Abscess ; Adrenal Cortex Hormones ; Adult ; Amenorrhea ; Anti-Bacterial Agents ; Deamino Arginine Vasopressin ; Diabetes Insipidus ; Female ; Fever ; Follow-Up Studies ; Headache ; Hormone Replacement Therapy ; Humans ; Hypopituitarism ; Outpatients ; Polydipsia ; Polyuria ; Prednisolone ; Thyroxine

Pituitary stalk interruption and ectopic neurohypophysis seen on magnetic resonance Imaging (MRI) are often associated with either isolated growth hormone (GH) deficiency or combined anterior pituitary hormone deficiency, but their pathogenesis is not clear and the clinical data regarding these anatomical defect is limited. We experienced a 23-year-old male with the absence of secondary sexual characteristics and this was accompanied with pituitary stalk dysgenesis and ectopic neurohypophysis. He received growth hormone for a year when he was 12 years old due to his short stature. Sella MRI showed no visible pituitary stalk with minimal high signal change, suggesting ectopic neurohypophysis. The combined pituitary stimulation test revealed blunted responses of growth hormone, follicle stimulating hormone and luteinizing hormone. For the hypogonadotropic hypogonadism, the patient was given testosterone intramuscularly and he gradually developed secondary sexual characteristics. We concluded that the hypogonadism and growth hormone deficiency in this patient was caused by hypopituitarism due to pituitary stalk dysgenesis and ecopic nuerohypophysis.
Follicle Stimulating Hormone ; Growth Hormone ; Humans ; Hypogonadism ; Hypopituitarism ; Luteinizing Hormone ; Magnetic Resonance Imaging ; Male ; Pituitary Gland ; Pituitary Gland, Posterior ; Testosterone ; Young Adult

BACKGROUND: The objective of this study is to demonstrate the relationship between the volume of a parathyroid adenoma and the preoperative biochemical parameters in patients undergoing surgery for primary hyperparathyroidism. METHODS: The medical records of 68 patients who underwent a parathyroidectomy for a single parathyroid adenoma were retrospectively reviewed. The volume of the adenoma was estimated using its measured size and a mathematical formula. The correlation between the volume of the parathyroid adenoma and the preoperative laboratory data was assessed. RESULTS: There were no correlations between the estimated volume of the adenoma and the serum calcium, alkaline phosphatase and parathyroid hormone levels. However, the volume of the adenoma was associated with the preoperative level of serum phosphorus. After excluding 5 adenomas with cystic degeneration, a positive correlation was noted between the adenoma volume and the preoperative levels of alkaline phosphatase and parathyroid hormone. CONCLUSION: The preoperative serum levels of calcium, alkaline phosphatase and parathyroid hormone are of limited use to predict the volume of the parathyroid adenoma in patients with a single parathyroid adenoma. We suggest that the absence of a correlation between the volume of the adenoma and the biochemical parameters can be attributed to the cystic degeneration of the adenomas.
Adenoma ; Alkaline Phosphatase ; Calcium ; Humans ; Hyperparathyroidism ; Hyperparathyroidism, Primary ; Medical Records ; Parathyroid Hormone ; Parathyroid Neoplasms ; Parathyroidectomy ; Phosphorus ; Retrospective Studies

BACKGROUND: Various staging systems for thyroid cancer that focus on cancer specific death have been suggested, but this approach had a limitation due to the relatively long clinical course and very low rate of cancer death. This study was performed to evaluate the staging systems and to determine the most predictive staging system for predicting recurrence. METHODS: The patients who underwent first total or near total thyroidectomy due to papillary thyroid cancer (PTC) at Asan Medical Center between January 1995 and December 2001 were the subjects of this study. The commonly used 8 staging systems were applied to these subjects. Disease free survival (DFS) and the relative importance of each staging system were determined by the Kaplan-Meier method, the Cox-proportional hazards model and the proportion of variation in the survival time explained (PVE). RESULTS: A total of 952 patients (M = 117, F = 835) were enrolled and their mean age was 45 years. During a median of 10 years of follow-up, 146 (15.3%) of 952 patients had recurred tumor. The independent prognostic factors were male gender, tumor size, extrathyroidal invasion and cervical lymph node metastasis. Risk stratification according to the American thyroid association (ATA) guideline was the most predictive staging system for recurrence of PTC (PVE 88.6%). The staging systems from EORTC (PVE 79.5%), and MACIS (PVE 68.4%) had significant values for predicting recurrence of PTC. The stage of NTCTCS could not predict recurrence (PVE 4.5%, P = 0.11). CONCLUSION: Risk stratification according to the ATA was most predictive staging system for predicting recurrence of PTC. The MACIS and EORTC staging systems have good value for predicting recurrence of PTC.
Carcinoma ; Carcinoma, Papillary ; Disease-Free Survival ; Factor IX ; Follow-Up Studies ; Humans ; Lymph Nodes ; Male ; Neoplasm Metastasis ; Proportional Hazards Models ; Recurrence ; Thyroid Gland ; Thyroid Neoplasms ; Thyroidectomy

BACKGROUND: Cardiovascular disease is the leading cause of death in patients with type 2 diabetes. Clinically, evaluating cardiovascular autonomic neuropathy (CAN) is important to predict cardiovascular mortality because it is correlated with cardiovascular death. The pulse wave velocity (PWV) correlates well with arterial distensibility and stiffness. It is also a useful approach for evaluating the severity of systemic atherosclerosis in adults. So, we evaluated that the relationship between cardiac autonomic neuropathy and the brachial-ankle pulse wave velocity (baPWV) in patients with type 2 diabetes. METHODS: We retrospectively analyzed 465 patients (209 men and 256 women) with type 2 diabetes. We checked the clinical characteristics and the laboratory tests and we assessed the diabetic complications. Standard tests for CAN were performed by DiCAN (Medicore, Seoul, Korea): 1) heart rate variability during deep breathing (the E/I ratio), 2) a Valsalva maneuver, 3) 30:15 ratio of R-R interval the blood pressure response to standing, and 5) the blood pressure response to handgrip. The CAN score was determined according to the results of the test as following: 0 = normal, 0.5 = borderline, 1 = abnormal. We also measured the baPWV by using a VP 1000 (Colin, Japan) and all the analyses were performed with the SPSS version 14.0. P values < 0.05 were considered significant. RESULTS: The CAN score is associated with the maximal baPWV, age, systolic blood pressure, microalbuminuria, the duration of diabetes, angiotensin II receptor blocker treatment, calcium channel blocker treatment, beta-blocker treatment and nephropathy. After adjusting for age, the baPWV is a independent predictor of the risk for CAN (beta = 0.108, P = 0.021). CONCLUSION: The CAN is associated with the baPWV in patient with type 2 diabetes.
Adult ; Atherosclerosis ; Blood Pressure ; Calcium Channels ; Cardiovascular Diseases ; Cause of Death ; Diabetes Complications ; Heart Rate ; Humans ; Male ; Pulse Wave Analysis ; Receptors, Angiotensin ; Respiration ; Retrospective Studies ; Valsalva Maneuver