Background Recent investigations suggest that activation of coagulation and fibrinolysis occurs in patients with ulcerative colitis (UC). However, the role of the hypercoagulable state in UC has not been determined. On the other hand, there are no reports dealing with coagulation in ischemic colitis (IC), in which acute bowel inflammation and reversible vascular occlusion affect the colon. Thus, our aim was to evaluate the hyper states of coagulation and fibrinolysis in UC by comparing activations of coagulation and fibrinolysis in patients with active UC and in those with IC. Methods Twenty-four patients with active UC and 12 patients with IC were studied, with 18 patients with inactive UC serving as controls. We investigated the activation of the coagulation system, including platelet counts, activated partial thromboplastin time (APTT), thrombin time (TT), prothrombin time (PT), serum concentrations of von Willebrand factor (vWF), activated factors XII, XI, X, IX, VIII, VII, V, II, fibrinogen, prothrombin fragments 1+2 (F1+2), thrombin-antithrombin complexes (TAT), protein S, protein C, plasminogen, α-2 plasminogen inhibitor (α-2PI) and D-dimer (D-D). Results Median serum vWF concentrations, F1+2, TAT, fibrinogen, activated factor XI, IX, VIII and V were significantly elevated in patients with active UC and IC compared to those in patients with inactive UC. There was no significant difference between active UC and IC patients in the mean values of any of the factors that were measured. Conclusion The results of the present study indicate that the coagulation-fibrinolysis system is activated in patients with active bowel inflammation such as active UC and IC, and that the hyper states of coagulation and fibrinolysis in patients with active UC are secondary to bowel inflammation.
Objective Metabolic syndrome (MetS) is a cluster of metabolic abnormalities that includes hyperglycemia, dyslipidemia, hypertension and increased waist circumference. Individuals with this syndrome are at increased risk for development of cardiovascular disease. Since an insulin-resistance state has a critical role in the development of MetS, there is growing concern about insulin-sensitizing effects of antihypertensives, including angiotensin II receptor blockers (ARBs). Telmisartan has been reported to have an effect on the activity of peroxysome proliferator-activated receptorγ, a well-known target for insulin-sensitizing antidiabetic drugs. The aim of this study was to determine the effects of administration of two different ARBs at low doses (telmisartan at 20 mg/day and valsartan at 40 mg/day) on insulin sensitivity. Methods Patients with MetS meeting the Japanese criteria were treated with telmisartan or valsartan for 4 weeks in combination with lifestyle modification. Results A significant reduction in blood pressure was found after 4 weeks of both treatments. The homeostasis model assessment of insulin resistance (HOMA-R) was significantly reduced by telmisartan compared to the baseline value (3.11±2.06 vs 2.56±1.48, p=0.031) but was not significantly changed by valsartan. A statistically significant correlation was found between HOMA-R at baseline and changes in HOMA-R after 4 weeks of treatment only in telmisartan-treated subjects. Body mass index, glycosylated hemoglobin and lipid profile were not changed by either treatment. Conclusion Our data revealed that treatment with telmisartan even at a low dose improves insulin sensitivity in hypertensive patients with MetS. This ameliorating effect of telmisartan on glucose metabolism clinically deserves to be considered.
Objectives The aim of this study was to investigate the impact of statin on systemic inflammation, left ventricular systolic and diastolic function and prognosis in low risk ischemic heart disease (IHD) patients. Methods A total of 430 consecutive IHD patients without congestive heart failure were enrolled. One hundred and thirtytwo patients (31%) were treated with statin (statin group) and 298 patients (69%) were not (no statin group). Echocardiographic indices, high sensitivity CRP, and prognosis were compared. Results Ejection fraction (EF) was significantly higher in the statin group (p<0.01). The ratio of the early transmitral flow velocity to early diastolic velocity of the mitral annulus (E/E') was significantly lower in the statin group than in the no statin group (p<0.01). Although LDL-cholesterol level did not differ, high sensitivity CRP level was significantly lower in the statin group (0.3±0.5 vs. 1.1±2.3 mg/dl, p=0.005). Cardiac event-(cardiac death and congestive heart failure)free survival rate was significantly higher in the statin group than in no statin group (Log-rank p<0.0001). By multivariate logistic regression analysis, E/E'> 15 (p=0.002), EF < 50% (p=0.003), lack of statin use (p=0.009), left atrial dimension (p=0.02), use of diuretics (p=0.03) and lack of beta-blockers (p=0.04) were independent predictors of cardiac events. In 248 patients matched by propensity scores, statin remained associated with better event-free survival (Log-rank p=0.006). Conclusion Statin may improve left ventricular function and thus improve the prognosis in low risk patients with IHD.
Objects Various morbid conditions constituting the metabolic syndrome could be also caused by excessive ethanol consumption. Thus, it is conceivable that excessive ethanol consumption may affect the diagnosis of the metabolic syndrome using its current diagnostic criteria. Here, we investigated this. Materials and Methods A cross-sectional study involving 2,130 Japanese man subjects aged 20 to 65 was performed. Results The rate of subjects judged to have metabolic syndrome using its diagnostic criteria for Japanese was 15.7%. However, the prevalence was significantly higher in excessive drinkers who consume more than 20 grams of ethanol per day (n=473, 22.0%) than average drinkers (n=1,657, 13.9%, χ2=18.0, p<0.0001). The rate of subjects who satisfied each component of the criteria of the metabolic syndrome, namely that of an excessive waist circumference, hypertension, dislipidemia, or hyperglycemia was significantly higher in the former than in the latter, respectively. When subjects with a waist circumferences of 85 cm or more were selectively studied, the prevalence of the metabolic syndrome was still higher in excessive drinkers (39.2%) than in average drinkers (32.4%, χ2=4.0, p=0.049), whereas the waist circumference was not significantly different between the two groups. Conclusion Excessive ethanol consumption is associated with an increased prevalence of the metabolic syndrome following the current clinical diagnostic criteria. Excessive ethanol consumption could simply be a factor worsening the metabolic syndrome. However, we must be aware of another possibility that excessive ethanol consumption increases the number of subjects regarded as the metabolic syndrome via mechanisms differing from visceral fat accumulation.
Objective We investigated the relationship between serum uric acid (SUA) and body fat area, serum lipid level, insulin resistance, and metabolic syndrome in Japanese men. Method We studied 508 Japanese man industrial workers who underwent an annual medical examination and agreed to participate in the CT scanning examination. Body fat area was measured at the umbilical level. Metabolic syndrome was defined by the presence of visceral fat Accumulation (≥100 cm2) accompanied by two or more disorders; dislipidemia, hypertension, and hyperglycemia. Results SUA was positively correlated with visceral fat area, subcutaneous fat area, serum total cholesterol level, serum triglyceride level, the Homeostasis Model Assessment index, and was negatively correlated with the high-density lipoprotein cholesterol level. In multiple regression analysis, the most influential factor for SUA was visceral fat area (p=0.0027), followed by the serum triglyceride level (p=0.0245). We clarified a higher SUA in the metabolic syndrome group as compared with the non-metabolic syndrome group: 6.67±1.14 mg, 6.09±1.14 mg, respectively (p<0.0001). The median SUA was elevated with increasing metabolic syndrome factors (p<0.0001). Conclusion The present study indicated that SUA is related to visceral fat accumulation. Patients with metabolic syndrome revealed a higher SUA.
Objective To clarify the associations of periodontal damage and tooth loss with atherogenic factors among diabetic patients. Methods We examined the correlations of age, sex, smoking, oral hygiene score, blood pressure, body mass index, and blood chemical data with the mean depth of periodontal pockets or the number of remaining teeth. Patients One hundred outpatients with type 2 diabetes aged 29 to 77 years. Results The mean depth of periodontal pockets was significantly associated with smoking, oral hygiene score, and HbA1c; the Spearman correlation coefficients (r) were 0.220, 0.417, and 0.260, respectively. Age, oral hygiene score, and HbA1c were inversely correlated with the number of remaining teeth (r=-0.306, -0.287, and -0.275, respectively). Serum total cholesterol was significantly correlated with the mean depth of pockets after adjustment for smoking, oral hygiene score, and HbA1c (r=0.211; P=0.044), while serum HDL cholesterol tended to be negatively associated with depth (r=-0.202; P=0.055). Serum HDL cholesterol was also associated with an increased number of teeth, which remained significant after adjustment for age, oral hygiene score, and HbA1c (r=0.202; P=0.048). The estimated glomerular filtration rate was significantly and positively correlated with the number of teeth in the univariate analysis, although consideration of the potential confounding factors somewhat weakened the association (r=0.186; P=0.069). Conclusions We may expect better management of oral health in diabetic patients with control of dyslipidemia in addition to blood glucose.
Objective Recently, a new concept of IgG4-related systemic disease including autoimmune pancreatitis, characterized by a high serum IgG4 level and tissue infiltration by IgG4-positive plasma cells, has been proposed. Our aim was to investigate the renal involvement in this condition. Patients and Methods We investigated the results of laboratory and imaging studies of the kidneys in 7 patients with IgG4-related systemic disease, and examined the renal histology in four of them. All patients showed elevated serum IgG4 levels, and 4 had autoimmune pancreatitis. The other three patients showed involvement of various extrapancreatic organs (lymphadenopathy, sialadenitis or renal insufficiency), and abundant IgG4-positive plasma cell infiltration was confirmed in their affected tissues. Results Six of the 7 patients showed some renal abnormalities. In one patient, hydronephrosis was observed accompanied by retroperitoneal fibrosis. Another patient showed multiple low-density areas in both kidneys by computed tomography, and gallium citrate scintigraphy showed gallium-67 accumulation in both kidneys, although renal function was normal. Four patients had tubulointerstitial nephritis. In two of them, the tubulointerstitial nephritis was diffuse. In one patient, marked diffuse but patchily distributed lymphoplasmacytic infiltration of the renal interstitium was observed. In another patient, computed tomography showed a tumor-like low-density mass; open biopsy of the mass showed aggregates of lymphocytes and plasma cells in the renal interstitium. Conclusion Renal parenchymal lesions in IgG4-related systemic disease are due to dense lymphoplasmacytic infiltration of the renal interstitium, and the lesions vary from diffuse tubulointerstitial nephritis to tumor-like masses according to the distribution patterns of the infiltrating cells.
Objective The Asian variant of intravascular lymphoma (AIVL) is a rare non-Hodgkin's lymphoma, characterized by hemophagocytic syndrome and the absence of neurological abnormality or skin lesions, which are typical features of classical IVL. The purpose of this study was to describe the characteristics and outcomes of AIVL patients. Patients and Methods We conducted a retrospective chart review of AIVL patients who were admitted to our institution between January 1999 and December 2006. Results Eight patients met the criteria. All patients presented with typical clinical features, including fever, hepatosplenomegaly, and cytopenia. The median time from onset to diagnosis was 4 weeks (range 2-12). Pathological diagnoses were made by bone marrow biopsy in three patients, transjugular liver biopsy in two patients, splenectomy in two patients, and percutaneous liver biopsy in one patient. All patients were treated with CHOP-like chemotherapy and rituximab was administered in five patients. Six patients achieved complete response, one achieved partial response, and two patients had central nervous system (CNS) relapse. Among the five patients treated with rituximab, four are still alive, with a median follow-up of 28 months. Conclusion A CHOP-like regimen with rituximab may be effective for AIVL cases, but, as in classical IVL, CNS prophylaxis should be considered.
Objective Sarcoidosis is considered to be provocated by highly activated Th1 lymphocytes. Because interleukin 12 (IL-12) is one of the most important cytokines for promoting Th1 reaction, we tried to detect IL-12 and its ligand IL-12 receptor in sarcoidosis patients. Patients and methods We measured the serum concentration of IL -12 p40 and IL-12 p70 by ELISA method with serum obtained from 60 sarcoidosis patients, and compared the serum concentration of IL-12 p40 with other clinical markers of disease activity. Next, we examined mRNA production of IL-12 p35, IL-12 p40, IL-12Rβ1, and IL-12β2 of sarcoid lymph nodes with semi-quantitative RT-PCR method. Results First, we showed that circulating IL-12 p40 was highly increased in sarcoidosis patients and was related to various other clinical markers. In particular, it was correlated with the number of involved organs which means systemic disease expansion. Second, we showed that the mRNA expression of IL-12 p40 and IL-12 receptor β2 subunit was increased in sarcoid lymph nodes. Conclution Our data suggest that increased circulating IL-12 p40 is an important systemic marker for disease activity, and it reflects the increased interaction between Il-12 and its ligand IL-12R in sarcoid lesions of involved organs.
Objective The objective of this prospective study was to investigate the status of acute respiratory tract infections caused by Haemophilus influenzae and Streptococcus pneumoniae in tsunami disaster evacuation camps. Methods Nasopharyngeal swabs (NP) of 324 internally displaced persons (IDP) in 3 different tsunami disaster evacuation camps of Sri Lanka were collected between March 18th and 20th, 2005, and analyzed for MIC, β-lactamase production, serotypes, PCR and pulsed-field gel electrophoresis (PFGE). Results Many IDP had respiratory symptoms and the prevalence of cough and/or sputum was 84%, 70.5% and 64.7% in the three camps. Twenty-one H. influenzae from 20 IDP and 25 S. pneumoniae from 22 IDP were isolated from the NP. All H. influenzae isolates were nontypeable, and 5 were β-lactamase producing. Seventeen pneumococci were susceptible, 5 showed intermediate resistance and 3 were fully resistant to penicillin G. Molecular analysis showed the 21 H. influenzae strains had 13 PFGE patterns and 25 pneumococci had 16 PFGE patterns. All 4 different PFGE patterns of H. influenzae strains were detected in a few IDP in camps 1 and 3, and 5 different PFGE patterns of serotype 3, 22A, 9A, 10A and 11A pneumococci were detected in a few IDP in camps 1 and 3. Conclusion Our data indicate acute respiratory tract infections caused by various types of H. influenzae and S. pneumoniae appear to have been prevalent, some of which were potentially transmitted from person to person in tsunami disaster evacuation camps.
Objectives To investigate autonomy preferences and the factors to promote active patient participation in a primary care setting in Japan. Patients Ninety-two hypertensive outpatients who consecutively visited a Japanese hospital between January and May of 2005 in Tokyo, Japan. Methods This cross-sectional study was conducted by using a self-administered questionnaire. The main outcome measures were patient preferences for autonomy (i.e., decision-making and information-seeking preferences), measured by the Autonomy Preference Index (API). The variables studied were patient sociodemographic characteristics, physician characteristics based on patient preference (i.e., ability to communicate, extent of clinical experience, qualifications, educational background, gender, and age), and the Multidimensional Health Locus of Control. Results On the API scale from 0 to 100, the patients had an intermediate desire for decision-making (median: 51) and a greater desire for information (median: 95). A multivariate regression model indicated that decision-making preference increased when patients were woman and decreased as physician age increased, and information-seeking preference was positively associated with good communication skills, more extensive clinical experience, physicians of middle age, and patient beliefs that they were responsible for their own health, and was negatively associated with a preference for man physicians. Conclusions Physicians may need to understand that patient autonomy preferences pertain to physician age and gender, physician communication ability and extent of clinical experience, and patient beliefs about self-responsibility toward health, and could use the information to promote reliable patient-physician relationships.
Recently, it has been reported that autoimmune pancreatitis (AIP) can be complicated with various extrapancreatic lesions. Here, we report a very rare case of pancreatic and hepatic inflammatory pseudotumor (IPT) with the infiltration of IgG4-positive plasmacytes. The patient showed pancreatic and hepatic masses with elevated levels of serum IgG4. Endoscopic retrograde cholangiopancreatography revealed narrowing of the intrapancreatic bile duct. Fluorine-18fluorodeoxyglucose positron emission tomography suggested pancreatic cancer with hepatic metastasis. Histopathologic findings showed fibrosis and infiltration of IgG4-positive plasmacytes, suggesting IPT. The present case suggests a possible common mechanism in the development of AIP and IPT of the liver.
Two young man patients with refractory post-bulbar duodenal ulcer (post-bulbar ulcer) were encountered. They had a single punched-out ulcer in the absence of an underlying disease. Patient 1 was Helicobacter pylori (Hp)-positive, and did not respond to Hp eradication therapy. The ulcer scarred after the long-term administration of a proton pump inhibitor (PPI), but recurred after a reduction in the dose. Patient 2 was Hp-negative. His ulcer did not scar even after long-term PPI administration, but it formed a fistula into the gallbladder, and the fistula was surgically closed. In both patients, laboratory and imaging studies excluded Zollinger-Ellison syndrome, but suggested a hyperacidic tendency. Unlike duodenal bulb ulcer (bulbar ulcer), the post-bulbar ulcer in Patient 1 did not heal with Hp eradication therapy, suggesting that post-bulbar ulcer differs etiologically from bulbar ulcer. We speculate that the possible causes of the refractoriness to treatment in both patients were ulcer penetration, callosity formation, and insufficient inhibition of gastric acid secretion due to the impaired passage of PPI into the deep portion of the duodenum as a result of luminal narrowing.
We report a case of tako-tsubo cardiomyopathy associated with brain metastasis of seminoma. This disease is characterized by transient cardiac wall motion abnormalities, electrocardiographic changes and minimal myocardial enzymatic release. During the hospital days, acute congestive heart failure suddenly appeared. The electrocardiogram showed a ST segment elevation in V1-3 and a giant negative T wave in I, aVL, aVF and V3-6. The echocardiogram revealed left ventricular dysfunction with severe hypokinesis to akinesis of anterior and apical wall regions, and hyperkinesis of the basal wall despite the lack of cardiac enzymatic abnormalities. With proper treatment, the patient's overall condition, wall motion and electrocardiographic abnormalities greatly improved.
This case report describes a 78-year-old man with recurrent angina attacks due to coronary spasm. He was treated with maximum daily doses of antianginal and antioxidative medications, including isosorbide mononitrate (40 mg), diltiazem (200 mg), and tocopherol nicotinate (300 mg). Despite the use of these medications, rest angina occurred 2 or 3 times during sleep. Although his symptoms disappeared promptly with the use of sublingual glycerine trinitrate (GTN), an angiotensin II receptor blocker, valsartan (80 mg), was added on a daily basis with the intent of improving endothelial function and controlling his angina. After beginning 80 mg/day of valsartan, the number of the anginal attacks decreased by about 66%. The anginal attacks totally disappeared after the dose of valsartan was increased to 160 mg/day. To confirm the effect of valsartan on his angina, valsartan was stopped temporarily with his consent. His anginal attacks increased to the same frequency that was observed before valsartan; therefore, valsartan therapy was resumed. The data indicate that the addition of valsartan to maximum antianginal medications may be effective in helping to control angina attacks at rest due to coronary spasm.
We present a young Thai man who developed acute flaccid paralysis after receiving pulse methylprednisolone for chronic inflammatory demyelinating polyneuropathy. Hypokalemia from intracellular shift was confirmed by calculation of transtubular potassium gradient (TTKG). His muscle strength and serum potassium fully recovered with a small amount of potassium replacement. Graves' disease was subsequently diagnosed and treated with radioactive iodine. We suggest that acute paralysis after the use of steroids should raise a suspicion of thyrotoxic periodic paralysis (TPP). The potential mechanisms of steroid-induced TPP are discussed.
A 61-year-old woman was admitted to our hospital because of acute kidney injury. She complained of general fatigue, appetite loss, and a high fever. Nodular lesions were observed on chest X-rays and there were >100 erythrocytes per high power field in her urinary sediment. A renal biopsy revealed necrotizing granulomatous glomerulonephritis, and her serum proteinase 3-antineutrophil cytoplasmic anitibody (PR3-ANCA) titer was elevated (55 EU). Based on these findings we made a diagnosis of Wegener's granulomatosis (WG). Hemodialysis was started immediately after admission. Steroid therapy was administered and her symptoms were relieved, but her renal function did not improve. On the 50th hospital day her condition suddenly became complicated by hemoperitoneum and massive intestinal bleeding, and the descending, transverse, ascending colon and part of the ileum were surgically resected. The cytomegalovirus (CMV) antigen titer was elevated, and histologic examination of the bowel specimen showed positive staining for CMV in the ulcer lesion, suggesting that CMV infection had caused the bowel hemorrhage. After treatment with ganciclovir, the bleeding was resolved and the CMV antigens became negative. We considered that this patient was further complicated by thrombotic thrombocytopenic purpura (TTP) because of thrombocytopenia, hemolytic anemia and neurologic symptoms. She was treated by plasma exchange. We report here a case of WG complicated by acute intestinal ulcer due to CMV infection and by TTP.
A 47-year-old woman with a severe cough and high-grade fever demonstrated proteinuria of 3.2 g/day. Chest radiograph and CT scan revealed scattered small nodules and ground-glass opacities with interlobular septal thickening in both lungs. The serum levels of surfactant A, surfactant D, and KL-6 were increased to 190 ng/ml (normal: 0-43.8), 360 ng/ml (normal:0-110), and 4850 U/ml (normal: 0-500), respectively. Video-assisted thoracoscopic lung biopsy revealed eosinophilic amorphous material within alveoli and thickened alveolar septa, which is compatible with pulmonary alveolar proteinosis. Kidney biopsy exhibited membranous nephropathy (Stage I-II) accompanied by granular IgG deposition along the glomerular basement membrane. Although the patient refused treatment with granulocyte macrophage colony stimulating factor (GM-CSF) for pulmonary alveolar proteinosis, her proteinuria and the pulmonary lesion gradually diminished and disappeared after one year.
The effect of polymixin B-immobilized fiber column (PMX) hemoperfusion treatment for acute exacerbation of interstitial pneumonia (IP) has been reported. Here, we report 2 cases of acute exacerbation of IP successfully treated with PMX hemoperfusion. One is a 55-year-old woman who was diagnosed as microscopic polyangiitis (MPA) with IP. The other is a 58-year-old man, diagnosed as having idiopathic pulmonary fibrosis. Both cases were treated with PMX hemoperfusion and other therapies. One died on day 44 and the other is still alive. The PMX hemoperfusion treatment decreased the serum levels of several cytokines and activated neutrophil percentage in bronchoalveolar lavage fluid.
A 22-year-old woman was admitted with symptoms of dyspnea and fever with pulmonary infiltrates noted on her chest X-ray study. She developed these symptoms in the workplace; her job included the removal of body hair using a diode-laser with 1,1,1,2-tetrafluoroethane (HFC134a, an alternative to chlorofluorocarbon) as a coolant. A chest X-ray examination revealed ground-glass opacities in the lower lung fields, and a chest computed tomographic study showed diffuse centrilobular opacities. An examination of the bronchoalveolar lavage fluid revealed increased lymphocytes with a slight increase in the number of eosinophils. An examination of the transbronchial biopsy specimens revealed eosinophil infiltration. A peripheral blood eosinophilia was also seen. The patient's symptoms, chest X-ray findings, and arterial blood gas analysis all returned to normal within a week. A challenge test of 1,1,1,2-tetrafluoroethane (HFC134a) inhalation was performed, which resulted in an elevation of body temperature, the development of a cough, and laboratory data indicating increased inflammation. We then determined the patient's diagnosis to be extrinsic allergic alveolits with eosinophil infiltration, caused by HFC134a.
Dual surface immunoglobulin light-chain expression in B-cell malignant neoplasm is a rare event, and has been predominantly reported in chronic lymphocytic leukemia. Herein, we report a case of aggressive B-cell lymphoma with κ/λ-dual surface immunoglobulin light-chain expression of a 69-year-old woman. The lymphoma cells were positive for CD5, CD19, CD20, HLA-DR, Igκ and Igλ. Southern blot analysis confirmed rearranged bands for both light chains with a monoclonal heavy chain rearrangement. She was treated with a combination of rituximab and CHOP regimen, but died of the progressive disease. To our knowledge, this is the first case of aggressive B-cell lymphoma showing dual κ/λ expression; the possible mechanisms of abnormal light chain expression are discussed.
A 78-year-old man with Parkinson's disease, paroxysmal atrial fibrillation, and congestive heart failure was admitted to our hospital due to global aphasia and right-sided hemiparesis. A cardioembolic stroke from a left ventricular thrombus was diagnosed; several days later, anticoagulants were started. On the seventh day, the patient suddenly developed severe acidosis and kidney and liver dysfunction. He died the following afternoon. Autopsy revealed an isolated celiac artery embolism from the left ventricular thrombus. This is the first reported case of isolated celiac artery embolism occurring after acute ischemic stroke.
We report a patient with dissection of the bilateral intracranial vertebral artery (VA) that did not present any symptoms other than occipital headache, which was probably associated with sleeping overnight in a car seat with unsteady head position. Although cerebral angiography revealed extensive dissection of the bilateral VA after branching of the posterior inferior cerebral artery, retrograde flow to the basilar artery (BA) via the right posterior communicating artery contributed to preserved posterior circulation. These findings indicate that even in patients without neurological deficits, the involvement of BA cannot be excluded and that accurate evaluation using radiological techniques should be considered.
A 69-year-old man complained of knee pain, subsequent polyarthralgia, and pains of the muscles of the pelvic girdle and thighs. At the same time, erythema of the face and hands appeared. Biopsy of the skin and muscle revealed non-caseating granuloma of epithelioid cells. The level of serum angiotensin-converting enzyme was normal, but that of lysozyme was elevated. Chest X-ray and CT did not show bilateral hilar lymphadenopathy (BHL) but revealed infiltrative ground glass appearance-like shadows of both lungs, and a Ga scintigram disclosed accumulation in the right hilar region, but not in the muscles. These complaints were quickly ameliorated by the administration of prednisolone. The present patient represented a rare case of acute musculoskeletal system involvement in sarcoidosis not typical of Löfgren's syndrome.