An Unusual Case of Sarcoidosis with a Prolonged Fever and Progressive Arthropathy.

A 70-year-old man had developed a high fever and arthralgia in his right elbow 6 months prior. Loxoprofen improved the symptoms temporarily, but arthropathy developed in other joints. Long-term recurrent arthropathy and the fever caused activity reduction and progressive debilitation. We performed fluorine-18 fluorodeoxyglucose-positron emission tomography and detected a positive accumulation in multiple joints and lymph nodes. A lymph node biopsy revealed epithelioid cell granulomas, which, along with elevated angiotensin-converting enzyme levels, led to the diagnosis of sarcoid arthropathy. After prednisolone administration, the fever and arthralgia resolved, and his activities of daily living improved. Clinicians should be aware of this type of sarcoid arthropathy.

Lifestyle Factors Associated With Erosive Esophagitis in Japanese Individuals With and Without Obesity Undergoing Health Checkups.

BACKGROUND: Several lifestyle factors have been reported to be associated with erosive esophagitis (EE). Many studies examined the associated factors for EE independent of obesity. However, lifestyle factors associated with EE might differ depending on sex or the presence of obesity. This study thus aimed to investigate the lifestyle risk factors for EE stratified by obesity and sex. METHODS: A cross-sectional survey was conducted among Japanese people who underwent an annual health checkup at the Oita Health Checkup Center between 2019 and 2021. Multivariate analysis was performed to determine the factors associated with EE in participants stratified by sex and the presence of obesity. RESULTS: EE was diagnosed in 676 of 5240 participants (12.9%) who were analyzed, and occurred most often in males (601 cases). The percentage of obesity based on body mass index was significantly higher among EE than non-EE participants (50.3% and 26.5%, P < .001). In obese males, eating just before bedtime (OR = 1.34, 95% CI 1.01-1.78) and hiatal hernia (OR = 3.42, 95% CI 2.57-4.54) were significantly associated with EE in multivariate analysis. In non-obese males, current smoking (OR = 1.48, 95% CI 1.07-2.04), alcohol consumption of 40 to 60 g/day (OR = 1.60, 95% CI 1.11-2.30), and hiatal hernia (OR = 3.63, 95% CI 2.79-4.73) were significantly associated with EE. CONCLUSIONS: We found that factors associated with EE were different between obese and non-obese males. In obese males, avoiding going to bed within 2 h after dinner might contribute to preventing the occurrence of EE. In non-obese males, prevention of obesity, quitting smoking, and avoiding heavy alcohol consumption might be important for preventing the development of EE.

A Case of Tumor-Induced Osteomalacia Detected by Venous Sampling.

Tumor-induced osteomalacia (TIO) can cause osteomalacia due to excessive production of fibroblast growth factor 23 (FGF23) by the tumor. Since TIO is a very rare disease, it is often misdiagnosed as intervertebral disc herniation, spondyloarthritis, or osteoporosis. We report a 65-year-old man who developed generalized arthralgia and difficulty walking two years ago and was diagnosed with multiple fractures throughout his body. He was initially diagnosed with osteoporosis and was treated with calcitriol. However, he was referred to our hospital since his symptoms did not improve. We diagnosed tumor-induced osteomalacia based on low serum phosphorus, high bone-type alkaline phosphatase, high FGF23 levels, and the presence of two tumors. The responsible tumor was identified using FGF23 levels in venous sampling. As the location of the tumor made surgical resection difficult, we selected treatment with burosumab, a human monoclonal antibody against FGF23, leading to improvement in the hypophosphatemia and pain, such that he was able to walk with a cane. In cases of osteoporosis with hypophosphatemia, general physicians should keep TIO in mind, and attempt to identify the responsible tumor lesion.

Positive Rate and Utility of Blood Culture among Nursing and Healthcare-associated Pneumonia Inpatients: A Cross-sectional Study.

Objective Although blood cultures to identify the presence of bacteremia are recommended for nursing- and healthcare-associated pneumonia (NHCAP), the incidence of true bacteremia and the relationship between true bacteremia and the outcome remain unclear. Physicians can therefore sometimes be confused regarding whether or not blood cultures should be obtained for NHCAP patients. This study assessed the incidence of true bacteremia and the relationship between true bacteremia and the outcome of NHCAP in a Japanese hospital setting. Methods We retrospectively analyzed NHCAP patients hospitalized between April 2016 and March 2021. The primary outcome was the incidence of true bacteremia in blood cultures. The incidence of true bacteremia was also examined according to quick Sequential Organ Failure Assessment (qSOFA) and A-DROP scores. In addition, we compared the incidence of true bacteremia between survivors and non-survivors. Results In total, 205 patients were included in this study. Blood cultures were obtained from 150 of the 205 patients (73.2%). Positive blood cultures were detected in 26 patients (17.3%), of which only 8 cases (5.3%; 95% confidence interval, 2.3-10.2%) were considered true bacteremia. Trend analyses for the incidence of true bacteremia according to qSOFA and A-DROP scores did not show any statistically significant results (p=0.49 for qSOFA; p=0.14 for A-DROP). The proportion of true bacteremia cases did not differ significantly between survivors and non-survivors. Conclusions The incidence of true bacteremia among NHCAP patients was very low. A strategy for determining indications for obtaining blood cultures from NHCAP patients needs to be established.

Disseminated nontuberculous mycobacterial infection in a patient with idiopathic CD4 lymphocytopenia and IFN-γ neutralizing antibodies: a case report.

BACKGROUND: Disseminated nontuberculous mycobacterial (NTM) infection usually occurs in immunodeficient patients, such as those with human immunodeficiency virus infection and idiopathic CD4 lymphopenia. However, disseminated NTM diseases have also been reported in immunocompetent patients. Autoantibodies to interferon-gamma (IFN-γ) are known to be involved in disseminated NTM disease, although anti-IFN-γ antibodies are mainly seen in immunocompetent patients rather than those with immunodeficiency. Here, we report a rare case of disseminated NTM patient with idiopathic CD4 lymphopenia and anti-IFN-γ antibodies. CASE PRESENTATION: A 64-year-old Asian male presented with fever, back pain, anorexia and weight loss. Physical examination revealed subcutaneous masses in the forehead, sternoclavicular joint, and right inguinal region. Computed tomography showed multiple osteosclerotic changes with soft structures and osteolytic changes. Both blood and sputum cultures were positive for Mycobacterium intracellulare, confirming the presence of disseminated NTM infection. Histopathological evaluation of the subcutaneous mass in the right inguinal region showed numerous granulomas consisting of epithelioid cells with Langhans-type giant cells. He was diagnosed with idiopathic CD4 lymphocytopenia. Interestingly, he also had anti-IFN-γ autoantibodies with suppression of IFN-γ-dependent signal transducer and activator of transcription 1 (STAT1) phosphorylation. Two-drug combination therapy with clarithromycin and ethambutol was started for the NTM infection, which resulted in a favorable disease course. CONCLUSIONS: In patients with disseminated NTM infection, idiopathic CD4 lymphocytopenia and anti-IFN-γ autoantibody-positive immunodeficiency can be coexisted. It is necessary to clarify the pathogenesis and clinical course of CD4 lymphocytopenic conditions and IFN-γ neutralizing antibody-positive in the disseminated NTM disease.

Cognitive Function and Olfactory Impairment in Community-Dwelling Older Adults Attending a Salon.

BACKGROUND: Early detection, treatment, and care are important for the prevention of dementia. Though olfactory impairment has been reported to be associated with mild cognitive impairment (MCI), there are few reports involving community-dwelling older adults with no difficulty in daily life in Japan. OBJECTIVE: This study aimed to identify the frequency of MCI in community-dwelling elderly people attending community salons in a city with a high aging population, and to determine the usefulness of a pocket olfactory discrimination test. METHODS: We recruited 268 independent older adults attending community salons. The Japanese version of the MCI Screen was used to evaluate cognitive function, defining MCI as <49.8 out of 100 scores of the memory performance index (MPI). The Japanese version of the University of Pennsylvania Smell Identification Test (UPSIT-J) was used for olfactory discrimination and the number of correct answers out of 4 questions was noted. RESULTS: Of the salon participants, 138 (51.5%) were classified as having MCI. As the number of correct answers on the olfactory test decreased, the percentage of participants with MCI increased. The MPI score of the low UPSIT score group was significantly lower than that of the high UPSIT score group. CONCLUSIONS: This study revealed that approximately half of the elderly local residents living without daily problems were suspected of having MCI. The UPSIT-J-4 is a simple olfactory identification test, and can be used at any time and any place. It is useful for screening cognitive function via olfactory identification in a salon-like setting.

Esophageal achalasia: An unusual reason for lung abscess.

Our case serves as a reminder that clinicians should pay attention to the presence of esophageal diseases in patients with lung abscesses. The esophagus should be evaluated during pulmonary CT imaging.

Bacteremia caused by Enterobacter asburiae misidentified biochemically as Cronobacter sakazakii and accurately identified by matrix-assisted laser desorption/ionization time-of-flight mass spectrometry: a case report.

BACKGROUND: Biochemical analyses of causative bacteria do not always result in clear identification, and new technologies aimed at improving diagnostic accuracy continue to be developed. Matrix-assisted laser desorption/ionization time-of-flight mass spectrometry is a rapid and accurate technique for bacterial identification. Misidentification of Cronobacter sakazakii is related to clinical and industrial problems. Here, we encountered a case of rare bacteremia in which the causative organism Enterobacter asburiae was biochemically misidentified as C. sakazakii before being correctly identified by matrix-assisted laser desorption/ionization time-of-flight mass spectrometry. CASE PRESENTATION: An 87-year-old Asian man with no diabetes or active disease developed bacteremia and was admitted to our hospital. While the route of infection could not be determined despite various examinations, the clinical course was good following antibiotic therapy. Biochemical analyses identified the causative organism as C. sakazakii, but colonies on the blood agar medium showed a grayish coloration, differing from the yellowish coloration of typical Cronobacter colonies. Matrix-assisted laser desorption/ionization time-of-flight mass spectrometry was therefore performed, identifying the bacterium as E. asburiae on three independent analyses. This result was confirmed by multilocus sequence analysis using five housekeeping genes. CONCLUSIONS: Matrix-assisted laser desorption/ionization time-of-flight mass spectrometry may reduce misidentification of bacteria as C. sakazakii and improve the reporting rate of E. asburiae. This technique should be considered when biochemical bacterial misidentification is suspected.

Plasma Exchange Did Not Reduce Viral Load in a Recovered Case of Severe Fever with Thrombocytopenia Syndrome.

Severe fever with thrombocytopenia syndrome (SFTS) is an emerging tick-borne infectious disease caused by the SFTS virus (SFTSV). There is no specific treatment for SFTS, although several reports have indicated that plasma exchange (PE) can be an effective therapy for severe SFTS. However, whether or not PE can reduce the viral load is unclear. We herein report a woman with SFTS who had her SFTSV viral load measured just before and after PE. While the patient recovered, there was no significant difference in the SFTSV viral load after PE. Our results confirmed that PE itself does not reduce the SFTSV viral load.

A case of community-onset Acinetobacter pneumonia in a healthy person.

A 56-year-old woman with fever, sore throat and productive cough was referred to our hospital, where mild community-acquired pneumonia was diagnosed. Sputum smears revealed Gram-negative coccobacilli. Treatment with ceftriaxone was initiated, but symptoms continued without progression to respiratory failure or bacteremia. As sputum cultures identified Acinetobacter baumannii, antibiotics were changed to levofloxacin, resulting in complete remission. A. baumannii is a very rare cause of community-acquired pneumonia in Japan. However, in cases of pneumonia where Gram-negative coccobacilli are identified and prove resistant to initial treatment, the possibility of A. baumannii pneumonia should be kept in mind even for healthy subjects with low severity score.

Inhaled Corticosteroid-Containing Regimens Reduce Hospitalizations and Healthcare Costs among Elderly Asthmatics: Real-World Validation Using the National Health Insurance Claims Database.

The high rates of mortality and hospitalization among elderly asthmatics, as well as their increasing healthcare costs have become an important public health issue. It would be worthwhile to assess whether inhaled corticosteroid (ICS) can resolve these problems. To explore ICS prescription rates for elderly asthmatics and the factors influencing them and to investigate their association with hospitalization and healthcare costs, we analyzed data from the National Health Insurance Claims Database for the same time frame (December 1 to February 28) across three different periods (2011-2012; 2014-2015; and 2017-2018), from which we identified 6,619, 5,619, and 6,880 elderly individuals, respectively. The prescription rates of ICS increased (52.8%, 65.5% and 68.8%, in the first, second and third survey period, respectively) and inversely the hospital admission rates declined (3.7%, 3.2% and 2.5%, in the first, second and third survey period, respectively). The total healthcare costs per month were significantly lower for patients who received ICS-containing regimens than for those who did not. A multivariate analysis revealed that increasing age, rural residence, receiving a prescription from a clinic, hospital admission, and prescription of asthma medications other than ICS were associated with non-prescription of ICS, whereas cross-boundary treatment increased the ICS-prescription rate. Our study suggests that increases in the prescription rate of ICS are associated with reduced hospital admission rates and lower medical costs in the real-world. ICS prescription rates in rural areas and at clinics, which remain low, need to be increased.

Diffuse Alveolar Hemorrhage Caused by Warfarin after Rifampicin Discontinuation.

An 83-year-old man under warfarin therapy presented for assessment of prolonged prothrombin time and cough. High-resolution computed tomography findings of the chest showed diffuse alveolar hemorrhage. His international normalized ratio (INR) was 11.89. He had been treated with rifampicin for a persistent infection, but this had been discontinued about two months before admission. Rifampicin suppresses the anticoagulant activity of warfarin, which can lead to a need for increased doses of warfarin to achieve and maintain a therapeutic INR. More frequent INR monitoring is needed even after discontinuing rifampicin.

A case of maoto-induced interstitial pneumonia.

A 64-year-old man was prescribed maoto, a prevailing Chinese herbal, for a cold with upper respiratory inflammation. Two days later, he developed a high fever, progressive dyspnea and pulmonary infiltration on chest high-resolution computed tomography (HRCT) including diffuse ground-glass opacity mainly around bronchovascular bundles and partial distribution of subpleural cysts resembling honeycombing. Despite the administration of azithromycin and pazufloxacin, the pulmonary infiltration and hypoxemia has rapidly progressed, so he was referred to our hospital. Although fulminant pneumonia or the acute exacerbation of idiopathic pulmonary fibrosis (IPF) was considered, his respiratory symptoms and pulmonary infiltration immediately improved and oxygen therapy was not needed on the fifth hospital day. Based on the clinical course, laboratory findings and the chest imaging findings, drug induced interstitial lung disease was suspected. The drug-induced lymphocyte test (DLST) as well as a scratch test against maoto demonstrated positive results. This is the first case report of maoto-induced interstitial pneumonia that was diagnosed based on the patient's clinical course, chest imaging findings and laboratory findings.

Significant elevation of the levels of B-cell activating factor (BAFF) in patients with sarcoidosis.

B-cell activating factor (BAFF) plays an important role in the survival and differentiation of B-cells and production of antibodies. Recent studies show that the serum BAFF levels are elevated in patients with sarcoidosis; however, they have not studied the relationship of the finding with the clinical features of the disease. The purpose of the present study is to analyze the BAFF and to elucidate the relationship between BAFF levels and the disease activity or severity of sarcoidosis. Eighty-eight patients with sarcoidosis and 21 healthy volunteers were enrolled in the present study. The BAFF levels were measured by an enzyme-linked immunosorbent assay. To assess the disease severity, we examined the number of affected organs, Schadding stages, respiratory function impairment (RFI), and the scoring system developed by Wasfi et al. The serum BAFF levels in sarcoidosis patients were significantly higher than those in healthy volunteers (median 1553.0 vs 984.6 pg/ml, p < 0.001). There were positive correlations between the serum BAFF level and disease activity markers. In addition, there were positive correlations between the BAFF levels and the disease severity score in both the serum (R = 0.367, p < 0.001) and bronchoalveolar lavage fluid (BALF) (R = 0.376, p < 0.001). This study demonstrated that the BAFF levels in both the serum and BALF were positively correlated with the disease activity markers and disease severity. BAFF may be useful as an indicator of both the disease activity and severity.

Intrascrotal and osseous sarcoidosis mimicking intrascrotal organ cancer and bony metastasis.

We report a case of sarcoidosis with concomitant epididymis, testes, and phalanxes involvement mimicking intrascrotal organ cancer and metastatic bony disease. A 23-year-old man developed blurred vision and hyperemia of the left eye, and was diagnosed as having left iritis. A chest computed tomography scan detected bilateral hilar lymphadenopathy and lung nodular shadows. A transbronchial lung biopsy revealed a non-caseating granuloma and he was diagnosed with sarcoidosis. One year after the onset of his eye symptoms, he noticed enlargement of his right scrotum. Magnetic resonance imaging detected lesions of the right epididymis, bilateral testes, and bilateral phalanxes. A technetium-99m scintigram revealed a high accumulation in the bilateral bones of extremities. These radiological findings mimicked intrascrotal organ cancer and metastatic bony disease. Pathologic evaluation following the right epididymectomy revealed non-caseating granulomas compatible with sarcoidosis. Three and half years after the appearance of intrascrotal and bony lesions, they improved spontaneously. (Sarcoidosis Vasc Diffuse Lung Dis 2017; 34: 373-376).

Familial Mediterranean Fever with Rheumatoid Arthritis Complicated by Pulmonary Paragonimiasis.

A 42-year-old woman presented with an intermittent fever and chest and back pain, and an abnormal chest shadow was detected. She was diagnosed with paragonimiasis caused by Paragonimus westermani. Praziquantel therapy improved the abnormal chest shadow, but did not relieve her symptoms. She was also diagnosed with familial Mediterranean fever (FMF), and colchicine therapy resolved her symptoms. She subsequently developed arthralgia and morning stiffness in her hands. We also diagnosed the patient with rheumatoid arthritis (RA), and corticosteroid and salazosulfapyridine therapy improved her symptoms. The existence of paragonimiasis complicated the diagnosis of FMF. The coexistence of FMF and RA is very rare, but does exist.

Angiopoietin-2 expression in patients with an acute exacerbation of idiopathic interstitial pneumonias.

BACKGROUND AND OBJECTIVE: We hypothesized that increased pulmonary vascular permeability may play a role in the pathogenesis of an acute exacerbation of the idiopathic interstitial pneumonias (AE-IIPs). Angiopoietin-2 (Ang-2) promotes endothelial activation, destabilization, and inflammation. The purpose of this study was to examine whether Ang-2 expression was associated with the pathogenesis of AE-IIPs. METHODS: Twenty-three patients with AE-IIP patients, 18 acute lung injury/acute respiratory distress syndrome (ALI/ARDS) patients, 37 idiopathic pulmonary fibrosis (IPF) patients, and 33 healthy volunteers (HVs) were enrolled. The serum level of Ang-2 was measured by an enzyme-linked immunosorbent assay. RESULTS: The serum levels of Ang-2 were higher in AE-IIPs and ALI/ARDS patients than in IPF patients and HVs; the BALF levels of Ang-2 were also higher than in IPF patients. There was a positive correlation between the serum level of Ang-2 and the CRP in patients with AE-IIP patients, whereas a significant positive correlation was found between the serum Ang-2 level and the CRP or SOFA scores of the ALI/ARDS patients. Although the baseline Ang-2 level was not related to survival, the Ang-2 levels significantly declined in survivors during treatment, while they did not change in non-survivors. CONCLUSIONS: Increased pulmonary vascular permeability and inflammation due to Ang-2 may play a role in the pathogenesis of AE-IIPs.

Subcutaneous sarcoidosis: a clinical analysis of nine patients.

Subcutaneous sarcoidosis is the specific subset of cutaneous sarcoidosis frequently associated with systemic disease. However, the disease activity, severity, and prognosis have not yet been elucidated due to the limited number of reported cases. The purpose of this study was to identify the clinical, laboratory, and prognostic differences between subcutaneous sarcoidosis and other type of cutaneous sarcoidosis. All patients with sarcoidosis diagnosed histopathologically from 2000 to 2012 at our institution were enrolled. The clinical, laboratory, chest X-ray, and pulmonary function test results were retrospectively evaluated in the patients with cutaneous sarcoidosis. In the 130 patients with sarcoidosis, cutaneous sarcoidosis was diagnosed in 37 patients (28.4 %), and 9 (6.9 %) of these patients had subcutaneous sarcoidosis. The serum levels of soluble interleukin-2 receptor (sIL-2R) were significantly elevated in the group of patients with subcutaneous sarcoidosis in comparison to the patients with other types of cutaneous sarcoidosis, whereas there was no significant difference in the severity score between the two groups. Following a 2-year observation period, three patients were in remission, five patients demonstrated stable disease, and one patient had progressive disease. Subcutaneous sarcoidosis may be associated with the disease activity, although it was not found to be associated with the disease severity and it was not a predictive factor for the prognosis. Furthermore, the prevalence of subcutaneous sarcoidosis may be higher than that in previously reported series.