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STUDY OBJECTIVES: To assess the seasonality of restless legs syndrome (RLS) using data from the Korean national health insurance database. METHODS: We retrospectively reviewed a randomly selected sample representing 3% of the national health insurance claims database in South Korea. From this sample, we obtained the monthly numbers of patients with RLS and diagnoses from 2009 to 2016, along with prescriptions for monthly dopamine agonists and clonazepam for patients with RLS from 2009 to 2013. Total dopamine agonist and clonazepam doses were converted to levodopa-equivalent doses, and the monthly cumulative prescription dose was calculated. Cosinor analysis was used to evaluate the seasonal pattern of each variable. RESULTS: This study included 11,466 patients with RLS and their diagnoses and 4,887 prescriptions for dopamine agonists and clonazepam. There were significant seasonal patterns in the numbers of patients with RLS (P < .001) and diagnoses (P < .001), both of which peaked in August. The magnitude of the greatest difference in the number of patients with RLS between August (highest) and February (lowest) was 29.96% (95% confidence interval, 24.03–100.80), and that of the number of RLS diagnoses was 39.56% (95% confidence interval, 31.24–47.89). The cumulative prescription dose of medication showed no significant seasonality. CONCLUSIONS: Our findings suggest that the prevalence of RLS is seasonally affected, with an increase during summer. CITATION: Oh SM, Son K-L, Choi S-J, Lee MH, Yoon SY, Lee YJ. The seasonal pattern of restless legs syndrome in a sample from the Korean health insurance review and assessment service national database. J Clin Sleep Med. 2021;17(5):1051–1056.

BACKGROUND Thymectomy is required for the treatment of thymoma-associated myasthenia gravis (MG). However, MG may develop only after thymectomy, a condition known as post-thymectomy MG. This study aimed to investigate the risk factors for post-thymectomy MG in patients with thymoma. METHODS We retrospectively identified 235 patients with thymoma who underwent thymectomy at a single hospital from January 2008 to December 2017: 44 with preoperatively diagnosed MG were excluded, leaving 191 patients in the final analysis. Univariable survival analyses using Cox proportional hazards regression model and Kaplan-Meier estimate were conducted to identify risk factors for post-thymectomy MG. RESULTS Post-thymectomy MG developed in 4.2% (8/191) of the patients with thymoma between 18 days and 108 mo after surgery. Hazard ratios (HRs) of pre- and postoperative anti-acetylcholine receptor antibody (AChR-Ab) titers were 2.267 (P = .002) and 1.506 (P

123I-Metaiodobenzylguanidine (MIBG) scintigraphy is a widely used functional imaging tool with a high degree of sensitivity and specificity in diagnosis of pheochromocytoma. However, rare cases of false positive reactions have been reported. A 67-year-old male patient was admitted with epigastric pain. Abdominal computed tomography (CT) revealed a heterogeneous left adrenal mass 6 cm in diameter; following hormone testing, 123I-MIBG scintigraphy was performed to determine the presence of pheochromocytoma, which confirmed eccentric uptake by a large left adrenal gland mass. Chest CT and PET-CT confirmed metastatic lymphadenopathy; therefore, endobronchial ultrasound transbronchial needle aspiration was performed. Metastatic carcinoma of unknown origin was suspected from a lymph node biopsy and surgical resection was performed for definitive diagnosis and correction of excess hormonal secretion. A final diagnosis of undifferentiated adrenal malignant tumor was rendered, instead of histologically malignant pheochromocytoma, despite the uptake of MIBG demonstrated by scintigraphy. When the degree of MIBG uptake is low-grade in relationship to a large tumor mass, then a false-positive result should be entertained, particularly in the absence of typical clinical and biochemical features of pheochromocytoma.

A common cause of drug hypersensitivity reactions is iodinated contrast media (ICM). ICM-induced hypersensitivity had been considered to be a non-immunological reaction, but evidence for an immunological mechanism has increased recently. Thus, we evaluated whether HLA-A, -B, and -C alleles were associated with ICM-induced hypersensitivity. In total, 126 patients who underwent contrast-enhanced computed tomography studies through outpatient clinics at a tertiary referral hospital between 2008 and 2012 were assessed. Sixty-one patients experienced ICM-induced hypersensitivity and the remainder, 65, were ICM-tolerant patients (control). ICM-induced hypersensitivity patients showed 51 with immediate, 7 with non-immediate, 3 with both or mixed type. HLA-A, -B, and -C genotyping was performed using a PCR sequence-based typing method. Four kinds of ICM were used: iopromide, iohexol, iobitridol, and iodixanol. The most used ICM among the hypersensitivity patients was iopromide. Significant difference in the frequency of HLA-B*58:01 (odds ratios [OR], 3.90; p = 0.0200, 95% confidence interval [CI], 1.16-13.07) was observed between ICM-induced immediate hypersensitivity and control. There were statistically significant differences in the frequencies of the HLA-B*38:02 (OR, 10.24; p = 0.0145; 95% CI, 1.09-96.14) and HLA-B*58:01 (OR, 3.98; p = 0.0348; 95% CI, 1.03-15.39) between iopromide-induced immediate hypersensitivity and control. The mechanism of ICM-induced hypersensitivity remains unknown, but this study showed associations, although weak, with HLA-B*58:01 alleles for ICM-induced immediate hypersensitivity and HLA-B*38:02 and HLA-B*58:01 for iopromide-induced immediate hypersensitivity as risk predictors. Further studies are needed to validate the associations in larger samples and to identify the functional mechanism behind these results.

Background Minor adverse events of intravenous immunoglobulin (IVIg) include flu-like symptoms, eczematous skin reaction, electrolyte disturbance, and transient leukopenia. On rare occasions, serious complications such as aseptic meningitis, arrhythmia, decrease in blood pressure, and thromboembolic complications (TEC) have been described. The current study aimed to understand the frequency and clinical features of TEC related to IVIg administration in patients with immune-mediated neurological disorders. Methods We conducted a retrospective chart review of hospitalized patients with immune-mediated neuromuscular or neuroimmunological disorders treated with IVIg from January 2018 to March 2020 in a single tertiary hospital. Results During the study period, 61 patients were treated with a total of 364 IVIg infusions over 84 treatment courses. Among them, we identified 3 TEC cases that occurred during or after the completion of IVIg therapy: two patients with myasthenia gravis (F/60 and F/80) and one patient with Guillain-Barre syndrome (F/79) had undergone arterial TEC (two for ischemic stroke and one for pulmonary thromboembolism). The rates of TEC per patient, per treatment course, and per infusion were 4.91% (3/61), 3.57% (3/84), and 0.82% (3/364), respectively. Conclusion The risk of developing TEC upon receiving IVIg infusions is generally low in patients with immune-mediated neurological disorders; however, IVIg-related TEC should be cautiously monitored for in critically ill elderly patients with vascular risk factors, especially those suffering from myasthenic crisis.

Background Antibodies to the aquaporin-4 (AQP4) water channel in neuromyelitis optica spectrum disorder (NMOSD) are reported to trigger the complement cascade, which is implicated in astrocyte damage and subsequent neuronal injury. The PREVENT study demonstrated that the terminal complement inhibitor eculizumab reduces adjudicated relapse risk in patients with anti-AQP4 immunoglobulin G-positive (AQP4+) NMOSD. The objective of this analysis was to evaluate the efficacy of eculizumab in reducing relapse risk and its safety in AQP4+ NMOSD across clinically relevant subgroups in PREVENT. Methods In the randomized, double-blind, time-to-event, phase 3 PREVENT trial, 143 adults received eculizumab (maintenance dose, 1200 mg/2 weeks) or placebo (2:1), with stable-dose concomitant immunosuppressive therapy (IST) permitted (except rituximab and mitoxantrone). Post hoc analyses of relapses and adverse events were performed for prespecified and post hoc subgroups based on concomitant IST and prior rituximab use, demographic and disease characteristics, and autoimmune comorbidity. Results The significant reduction in relapse risk observed for eculizumab versus placebo in the overall PREVENT population was consistently maintained across subgroups based on concomitant IST and previous rituximab use, age, sex, region, race, time since clinical onset of NMOSD, historical annualized relapse rate, baseline Expanded Disability Status Scale score, and history of another autoimmune disorder. The serious infection rate was lower with eculizumab than placebo regardless of rituximab use in the previous year, concomitant IST use, or history of another autoimmune disorder. Conclusion Across a wide range of clinically relevant AQP4+ NMOSD patient subgroups in PREVENT, eculizumab therapy was consistently effective versus placebo in reducing relapse risk, with no apparent increase in serious infection rate. Trial registration NCT01892345 (ClinicalTrials.gov).

The aim of the present study was to investigate the frequency and clinical features of Guillain-Barré syndrome (GBS) with hyperCKemia. We retrospectively identified 139 patients with GBS at 2 teaching hospitals in South Korea. We excluded patients with Miller-Fisher syndrome (n = 19), acute bulbar palsy (n = 3), and those whose serum creatine kinase (CK) levels were not measured (n = 45). Twelve of 72 patients (16.7%) had transient hyperCKemia, defined as serum CK ≥300 IU/L. The frequency of male sex and non-demyelinating electrodiagnostic features were higher in patients with hyperCKemia than those without. Transient hyperCKemia, occasionally seen in patients with GBS may be associated with the non-demyelinating subtype.

The present study aimed to investigate the prognostic importance of the neutrophil-to-lymphocyte ratio (NLR) in patients with amyotrophic lateral sclerosis (ALS). Among 322 patients diagnosed as having definite, probable, or possible ALS at a single tertiary hospital, 194 patients were included in the final analysis. Patients were divided into three groups (T1, T2, and T3) according to the tertile of their NLR. Survival rate was significantly lower in T3 compared to the other groups (log-rank test; T1 vs. T3, p = 0.009; T2 vs. T3, p = 0.008). Median survival duration was 37.0 (24.0–56.0), 32.5 (19.5–51.2), and 22.0 (17.0–38.0) months in T1, T2, and T3, respectively. In a multivariable Cox proportional hazards regression analysis, the hazard ratio of age at onset, bulbar-onset, and NLR (T3/T1) was 1.04 (1.02–1.06, p

There is a lack of research on the effects of physical activity (PA) on the progression of Alzheimer's disease (AD).We investigated whether PA is associated with progression of dementia and mortality in AD.In the present study, 934 patients with mild-to-moderate AD were included. PA was evaluated using a questionnaire written by the caregiver. The outcome measures were the Clinical Dementia Rating-Sum of Boxes (CDR-SB), Seoul-Instrumental Activities of Daily Living (S-IADL), Caregiver-Administered Neuropsychiatric Inventory (CGA-NPI), a global composite score of neuropsychological subtests, and mortality. They were evaluated annually and received a maximum of three follow-up examinations.Between-group differences compared with the no PA group in the change of CDR-SB scores were -0.431 (95% CI = -0.824∼-0.039; p = 0.031) for the moderate PA group (150-750 minutes per week of moderate intensity PA), and -1.148 (-1.656∼-0.639; p 0.001) for the high PA group (750 minutes per week). As PA increased, there was a significant trend to slow the rate of increase in the CDR-SB, S-IADL, and CGA-NPI scores. The patients with ≥150 minutes per week for each of non-recreational and recreational PAs had a lower risk of mortality compared to those with150 minutes per week for each of the PAs (hazard ratio 0.22, 95% CI = 0.05∼0.88; p = 0.033).More PA is associated with slower progression of dementia severity, functional decline, and abnormal behavior, and with a lower risk of mortality in AD.

Objective To investigate the frequency, features, and prognosis of takotsubo cardiomyopathy (TTC) in patients with amyotrophic lateral sclerosis (ALS). Methods We reviewed detailed clinical, laboratory, and cardiovascular data from 64 ALS patients (38 men and 26 women) who underwent echocardiographic evaluation for various reasons at a single referral center between January 2011 and December 2015. Results TTC was diagnosed in 9 ALS patients (4 men and 5 women). Mean age was 61.3 years (range 55–71 years), and median disease duration was 51.5 months (range 18–134 months). All patients were bulbar or cervical onset, and were at advanced stages of ALS when TTC was diagnosed. Acute exacerbation of dyspnea was an invariable presentation, and chest discomfort mimicking acute coronary syndrome was present in 2 patients. Six patients had significant hypotension requiring intravenous fluid challenge and inotropic support. Three patients showed altered mentality, and 2 of them suffered cardiopulmonary arrest. Conclusions TTC should be suspected in ALS patients presenting with acute exacerbation of dyspnea and chest discomfort, particularly at advanced stages of the disease. This study highlights the need for proper evaluation and management of cardiac dysfunction in ALS.

Spinobulbar muscular atrophy (SBMA) is an X-linked recessive disease, presenting motor weakness and wasting of facial, bulbar and limb muscles. Hereditary neuropathy with liability to pressure palsy (HNPP) is autosomal dominant disorder characterized by recurrent neuropathies at common entrapment sites. We report a case of co-existence of SBMA and atypical HNPP with genetic confirmation of CAG expansion in the androgen receptor (AR) gene and deletion of the peripheral myelin protein 22 (PMP22) gene. A 62-year-old man presented with progressive muscle weakness, fasciculations in upper and lower limbs and dysesthesia predominantly in the distal regions. No family members, including his children, experienced similar symptoms. The electrodiagnostic examination was compatible with demyelinating sensorimotor polyneuropathy. Simultaneous hereditary polyneuropathy and motor neuron disease were suspected and relevant genetic testing was confirmed HNPP and SBMA. This case presented with 2 rare genetic neuromuscular disorders and the atypical HNPP phenotype. This case highlight the importance of detailed patient histories, as well as neurological and electrophysiological examinations for diagnosis of atypical and combination of rare genetic disorders.

Background Myelin oligodendrocyte glycoprotein immunoglobulin G1 (MOG-IgG1)-associated disease is suggested as a separate disease entity distinct from multiple sclerosis and neuromyelitis optica spectrum disorder. Nonetheless, the optimal treatment regimen for preventing relapses in MOG-IgG1-associated disease remains unclear. Case presentation We describe the case of a 45-year-old man with MOG-IgG1-positive highly relapsing optic neuritis who had experienced 5 attacks over 21 months and had monocular blindness despite prednisolone and azathioprine therapy. He began treatment with rituximab, which reduced the rate of relapse markedly. Following discontinuation of rituximab, however, the patient experienced two successive optic neuritis attacks 2 and 4 months after B-lymphocyte restoration. Conclusions Highly relapsing MOG-IgG1-associated disease can be prevented with rituximab even when the MOG-IgG1 titers are relatively stationary. Discontinuation of rituximab and restoration of B-lymphocytes may be associated with the rebound of disease activity.

Purpose We evaluated the risk factors for progression to chronic complicated bronchopleural fistula (BPF) after pulmonary resection using follow-up CT. Materials and Methods We retrospectively reviewed 45 cases with BPF that had undergone pulmonary resection during 2010-2018. We compared the clinical and radiological characteristics of those with complicated BPF (n = 24) and those without complicated (sterilized) BPF (n = 21). The clinical and radiological risk factors for progression to chronic complicated BPF were examined by logistic regression analysis. Results The thickness of the pleural cavity wall (p = 0.022), the size of the pleural cavity (p = 0.029), and the size increase of BPF on follow-up (p = 0.012) were significantly different between the two groups. The risk factors for progression to chronic complicated BPF were age > 70 years (odds ratio, 6.43; 95% confidence interval, 1.2–33.7), the thickness of the cavity wall > 5 mm (odds ratio, 52.5; 95% confidence interval, 5.1–545.4), and an increase in the size of the pleural cavity on follow-up CT (odds ratio, 12.5; 95% confidence interval, 2.1–73.5), only in the univariate analysis. Conclusion The risk factors for progression to chronic complicated BPF can be evaluated using follow-up CT.

Rheumatoid meningitis is a rare complication of rheumatoid arthritis that requires a meningeal biopsy for diagnosis. The authors thank Drs. YoonJin Kwak and Kyu Sang Lee of the Department of Pathology at Seoul National University Bundang Hospital for comments and technical assistance in preparing the figures.

ObjectiveThis study aimed to explore the correlations between electrodiagnostic (EDX) features in patients with chronic inflammatory demyelinating polyneuropathy (CIDP) and to investigate whether EDX data-driven clustering can identify a distinct subgroup regarding clinical phenotype and treatment response.MethodsWe reviewed clinical and EDX data of 56 patients with definite CIDP fulfilling the 2010 European Federation of Neurological Societies and Peripheral Nerve Society criteria at two teaching hospitals. A hierarchical agglomerative clustering algorithm with complete linkage was used to partition the patients into subgroups with similar EDX features. A stepwise logistic regression analysis was performed to evaluate predictors of the long-term outcome.ResultsEDX data-driven clustering partitioned the patients into two clusters, identifying a distinct subgroup characterised by coexistence of prominent conduction slowing and markedly reduced distally evoked compound muscle action potential (CMAP) amplitudes. This cluster of patients was significantly over-represented by an atypical subtype (distal acquired demyelinating symmetric polyneuropathy) compared with the other cluster (70% vs 26.1%, p=0.042). Furthermore, patients in this cluster invariably showed favourable long-term treatment outcome (100% vs 63%, p=0.023). In logistic regression analyses, the initial disability (OR 6.1, 95% CI 2.4 to 25.4), F-wave latency (OR 0.93, 95% CI 0.86 to 0.98) and distal CMAP duration (OR 0.96, 95% CI 0.91 to 0.99) were significant predictors of the poor long-term outcome.ConclusionOur results show that EDX data-driven clustering could differentiate a pattern of EDX features with prognostic implication in patients with CIDP. Reduced distally evoked CMAPs may not necessarily predict poor responses to treatment, and active treatment is warranted when prominent slowing of conduction is accompanied in the distal segments.

Background Little is known about the correlation between microbiological yield and radiographic activity, on chest computed tomography (CT), in suspected pulmonary tuberculosis (PTB) cases, despite CT being widely used, clinically. Methods We used multicenter retrospective data, obtained from medical records, focusing on the diagnostic performance for definite PTB. We categorized patients into four groups, by radiographic activity: definitely active, probably active, indeterminate activity, and probably inactive. Results Of the 650 patients included, 316 had culture-confirmed PTB; 190 (29.2%), 323 (49.7%), 70 (10.8%), and 67 (10.3%) were classified into the definitely active, probably active, indeterminate activity, and probably inactive groups, respectively. The corresponding observed culture rates for CT radiographic activity were 61.6%, 60.7%, 4.3% and 0%, respectively. When not only culture rates but TB-PCR and histological results were taken into consideration as definite PTB, it showed 66.6%, 67.2%, 14.3%, and 0% of each CT radiographic activity, respectively. Regarding the diagnostic performance for definite PTB, radiographic activity displayed high sensitivity (97.1%, 95% confidence interval (CI), 94.6-98.5) and negative predictive values (92.7%, 95% CI, 86.6-96.2), considered definitely and probably active PTB. Apart from PTB, other etiologies, according to radiographic activity, were predominantly respiratory infections such as bacterial pneumonia and non-tuberculous mycobacterial infection. Conclusions Radiographic activity showed good diagnostic performance, and can be used easily in clinical practice. However, clinicians should consider other possibilities, because radiologic images do not confirm microbiological PTB.

123I-Metaiodobenzylguanidine (123I-MIBG) scintigraphy is a widely used functional imaging tool with a high degree of sensitivity and specificity in diagnosis of pheochromocytoma. However, rare cases of false positive reactions have been reported. A 67-year-old male patient was admitted with epigastric pain. Abdominal computed tomography (CT) revealed a heterogeneous left adrenal mass 6 cm in diameter; following hormone testing,123I-MIBG scintigraphy was performed to determine the presence of pheochromocytoma, which confirmed eccentric uptake by a large left adrenal gland mass. Chest CT and PET-CT confirmed metastatic lymphadenopathy; therefore, endobronchial ultrasound transbronchial needle aspiration was performed. Metastatic carcinoma of unknown origin was suspected from a lymph node biopsy, and surgical resection was performed for definitive diagnosis and correction of excess hormonal secretion. A final diagnosis of undifferentiated adrenal malignant tumor was rendered, instead of histologically malignant pheochromocytoma, despite the uptake of123I-MIBG demonstrated by scintigraphy.

OBJECTIVE: The aim of this study was to compare the predictive value of the VitalPAC Early Warning Score-lactate (ViEWS-L) score with that of the trauma and injury severity score (TRISS), which is a pre-existing risk scoring system used in trauma patients. METHODS: The patients were blunt trauma victims admitted consecutively to the study hospital between 1 April 2010 and 31 March 2011, who were 15 years or older and had an injury severity score of 9 or higher. The lactate level, the ViEWS and revised trauma score upon arrival at the emergency department, and the injury severity score and TRISS were evaluated. The ViEWS-L score was calculated according to the formula: ViEWS-L=ViEWS+lactate (mmol/l). The ability to predict mortality was assessed by area under the receiver operating characteristic curve (AUC) analysis and calibration analysis. RESULTS: A total of 299 patients were available for analysis, of whom 33 died (11.0%). The median ViEWS-L score was 3.7 (interquartile range:1.8-6.4) and the median TRISS was 96.8 (interquartile range: 93.4-98.6). The ViEWS-L score was better than TRISS at predicting hospital mortality (AUC, 0.838; 95% confidence interval, 0.771-0.906 vs. AUC, 0.734; 95% confidence interval, 0.635-0.833, P=0.031). Calibration of the ViEWS-L score (χ=11.13, P=0.194) was good but that of TRISS was not (χ=16.97, P=0.018). CONCLUSION: The prognostic value of the ViEWS-L score in terms of discrimination was better than that of TRISS in the blunt trauma patients admitted to the emergency department with an injury severity score of 9 or higher, and the ViEWS-L score showed good calibration. Language: en

Miliary tuberculosis (TB) is a rare extrapulmonary form of TB, and there have been only two reports of miliary TB associated with infection with multidrug-resistant (MDR)-TB pathogen in an immunocompetent host. A 32-year-old woman was referred to our hospital because of abnormal findings on chest X-ray. The patient was diagnosed with MDR-TB by a line probe assay and was administered proper antituberculous drugs. After eight weeks, a solid-media drug sensitivity test revealed that the pathogen was resistant to ethambutol and streptomycin in addition to isoniazid and rifampicin. The patient was then treated with effective antituberculous drugs without delay after diagnosis of MDR-TB. To the best of our knowledge, this is the first case of miliary TB caused by MDR-TB pathogen in Korea.

The objective of this study was to prospectively evaluate whether intraarticular anesthesia can reduce pain after MR or CT arthrography of the shoulder.This study included 120 patients who underwent CT or MR arthrography of the shoulder. The patients were randomized into two groups: the study group (n = 60), each administered an intraarticular injection of the contrast agent mixed with a local anesthetic (2% mepivacaine); and the control group (n = 60), each injected with the contrast agent only. Each patient's pain level was assessed at five phases-baseline and immediately, 2 hours, 1 day, and 2 days after injection-by using a visual analog scale or a verbal rating scale. The net pain score at each phase was also calculated. The pain course and net pain score were compared between study and control groups by repeated-measures analysis of variance. After the patients were divided into subgroups according to patient- or procedure-related factors, the efficacy of the intraarticular local anesthetic in each subgroup was evaluated.The pain course showed a quadratic trend and was not significantly different between study and control groups. The net pain score also was not significantly different between the two groups. No subgroup showed a significantly different efficacy of the intraarticular local anesthetic between the two groups.Intraarticular local anesthesia did not reduce arthrography-related pain.

A patient who has multiple lung masses with a history of malignancy in organs other than the lung is more likely to be diagnosed with metastatic rather than primary lung cancer. Rarely, metastatic cancer can coexist with primary. We experienced a case of concurrent diagnosis of primary small cell lung cancer and pulmonary metastasis of uterine malignant mixed Mullerian tumor (MMMT). The patient was a 52-year-old female with femur fracture and multiple lung masses with a history of an operation for uterine MMMT. The small cell lung cancer was diagnosed by bronchoscopic biopsy. The central lung mass decreased after chemotherapy for small cell lung cancer but multiple peripheral masses increased. A percutaneous biopsy for one of peripheral masses revealed metastatic uterine MMMT. We suggest that we have to consider the possible presence of concomitant malignancies of different origins in one organ especially with patients who had a history of malignancy in another organ.

Purpose To evaluate technical success and clinical efficacy of crisscross-configured dual biliary stent implantation in patients with a biliary hilar tumor extending beyond the segmental ducts. Materials and Methods Between January 2002 and December 2006, two metallic stents were placed crossing each other in a hepatic hilum in 42 patients. One stent was placed between one right sectoral duct and the left hepatic duct and the other was placed between another right sectoral duct and the common bile duct. The patients ranged in age from 36 to 83 years (mean, 63.3 y) and included 26 men and 16 women. Technical success, clinical success, complications, and long-term results were analyzed by retrospective review. Results Stent placement was performed through two right accesses ( n = 30), one right and one left access ( n = 3), or two right accesses and one left access ( n = 6). Successful "trisectoral" drainage was obtained with two stents in 41 patients (98%). In one case of initial technical failure, an additional stent was needed to connect the two previously placed stents. Clinical success was obtained in 34 of the remaining 36 patients (92%). No procedure-related major complication was observed. The median primary stent patency time was 187 days and the median patient survival time was 247 days. Conclusions Crisscross-configured dual stent implantation is a feasible, safe, and effective method to maximize hepatic drainage with a minimal number of stents in patients with advanced biliary hilar malignancy.

Objective To evaluate changes in lung function in individuals before and after treatment for pulmonary tuberculosis (PTB) in relation to extent of disease. Design Using a retrospective cohort design, changes in and predictors of lung function were evaluated. Results A total of 41 patients were included in the final analysis. The median decline in annualised forced expiratory volume in 1 sec (FEV1) was 180.0 ml/year (95%CI 118.9-356.1) in advanced PTB and 94.7 ml/year (95%CI 33.4-147.3) in localised PTB (ΔFEV1% predicted/year 9.4%, 95%CI 4.4-14.0 vs. 3.8%, 95%CI 1.8-6.2). The median decline in annualised forced vital capacity (FVC) was 309.6 ml/year (95%CI 137.0-359.0) in advanced PTB and 101.1 ml/year (95%CI 30.3-219.6) in localised PTB (ΔFVC % predicted/year 7.3%, 95%CI 5.3-12.3 vs. 2.9%, 95%CI 0.9-6.5). Conclusions As the sample size of our study was small, the conclusions could be biased. Nevertheless, our findings show that PTB causes a significant decline in lung function even in localised PTB, whereas advanced PTB was associated with excessive or even higher decline. This study suggests that early diagnosis and treatment of PTB is needed to preserve lung function.

Aggressive fibromatosis arising from the retroperitoneum is extremely rare. It may occur in association with previous trauma, abdominal surgery, drugs, Gardner’s syndrome, or familial adenomatous polyposis. We report a case of retroperitoneal fibromatosis presenting as a presacral mass with an infiltrating nature, relatively intense enhancement on enhanced computed tomography scanning, and low-signal intensity on T2-weighted magnetic resonance imaging in a patient with no significant medical or surgical history.

Uterine adenosarcoma (AS) are rare tumors and have more favorable outcomes than the aggressive uterine carcinosarcomas. Uterine adenosarcoma with sarcomatous overgrowth (ASSO) is a variant form of AS and exhibits aggressive growth of tumor and the prognosis is relatively poor compared with typical AS. Usually patterns of metastasis have been known to behave like endometrial carcinoma and spread through the lymphatics. Brain metastasis from uterine AS is extremely rare. Herein, we report a case of successfully surgically removed solitary brain metastasis without any extracranial recurrence from uterine ASSO after 4 years of primary treatment.

Transvenous coil embolization has been successfully applied for the treatment of cavernous sinus dural arteriovenous fistula (CSDAVF). Unfortunately, the technique cannot be applied in cases of poor or absent inferior petrosal sinus or facial venous access route to the fistula. Recently, we experienced a successful embolization using direct superior ophthalmic vein approach in cases of CSDAVF which were no opacification of inferior petrosal sinus or facial vein.

Dear Editor, Multiple symmetric lipomatosis (MSL), also called Madelung's disease, is a rare disease characterized by the growth of nonencapsulated masses of adipose tissue.1 The disease causes not only cosmetic issues but also various clinical problems depending on the location and extent of the lesion,2 with neuromuscular manifestations such as polyneuropathy, myopathy, and ataxia having been reported.3 Herein we report on a case of bilateral brachial plexopathy caused by extensive lipoma associated with MSL. A 55-year-old man presented with progressive bilateral hand weakness. He first noticed a weak grip with his right hand 2 years before the presentation. The symptom progressed slowly, and his left hand also became weak a few months later. The patient often experienced paresthesia in both arms, but did not complain of proximal arm or leg weakness. He had been drinking about 200 mL of alcohol every day for 30 years. A physical examination revealed excessive soft tissues around his neck, shoulder, and forearm (Fig. 1A). His hand grip, finger abduction, wrist flexion/extension, and elbow extension were extremely weak (Medical Research Council scale grades from 1 to 3) on both sides with muscle atrophy in the first dorsal interossei, hypothenar, and pectoralis major muscles. Deep tendon reflexes were absent in both arms. A nerve conduction study revealed no potentials in the ulnar and radial motor nerves or the medial and lateral antebrachial cutaneous nerves of the left arm. Electromyography revealed positive sharp waves and fibrillation potentials in the first dorsal interosseous, abductor pollicis longus, and extensor carpi radialis of the left arm. Long-duration, high-amplitude motor unit action potentials (MUPs) were present in all muscles of the left arm with reduced recruitment of MUPs. Magnetic resonance images showed extensive fat proliferation and infiltration at the face, neck, shoulder, and axilla that resulted in compression of the brachial plexus (Fig. 1B). He was diagnosed with bilateral brachial plexopathy secondary to MSL, and was operated on to relieve the compression of the brachial plexus. After the surgery, the weaknesses in his hands were slightly improved. Fig. 1 A: Excessive fatty tissues around the neck and shoulder. B: Coronal fat-suppressed T2-weighted MRI reveals the brachial plexus (thick arrow) compressed by lipoma (thin arrow). The incidence of MSL is highest in Mediterranean countries, and it is uncommon in Asia (including Korea). A case-series study of Korean MSL patients found that most patients visited hospital because of a mass lesion and some of them were complicated with peripheral neuropathy.4 The etiology of the disease is not fully understood, but most patients included in previous studies have had a history of chronic excessive alcohol consumption.5 While the exact role of alcohol in the pathophysiology remains elusive, defects in adrenergic stimulated lipolysis or hypertrophy of the brown fat cells have been proposed as an alternative explanation for the pathomechanism.6 The differential diagnosis should include other forms of generalized lipomatosis such as Bannayan-Zonana syndrome, Cowden syndrome, Proteus syndrome, and Dercum disease.7 Familial multiple lipomatosis manifests as multiple encapsulated lipomas with autosomal dominant inheritance, but with relative sparing of the head and shoulders.8 Surgical excision is the treatment of choice. Liposuction can be applied alone9 or in combination with lipectomy.10

OBJECTIVE To determine the effects of respiration on the size of lung cysts by comparing inspiratory and expiratory high-resolution CT (HRCT) scans. MATERIALS AND METHODS The authors evaluated the size of cystic lesions, as seen on paired inspiratory and expiratory HRCT scans, in 54 patients with Langerhans cell histiocytosis (n = 3), pulmonary lymphangiomyomatosis (n = 4), confluent centrilobular emphysema (n = 9), paraseptal emphysema and bullae (n = 16), cystic bronchiectasis (n = 13), and honeycombing (n = 9). Using paired inspiratory and expiratory HRCT scans obtained at the corresponding anatomic level, a total of 270 cystic lesions were selected simultaneously on the basis of five lesions per lung disease. Changes in lung cyst size observed during respiration were assessed by two radiologists. In a limited number of cases (n = 11), pathologic specimens were obtained by open lung biopsy or lobectomy. RESULTS All cystic lesions in patients with Langerhans cell histiocytosis, lymphangiomyomatosis, cystic bronchiectasis, honeycombing, and confluent centrilobular emphysema became smaller on expiration, but in two cases of paraseptal emphysema and bullae there was no change. CONCLUSION In cases in which expiratory CT scans indicate that cysts have become smaller, cystic lesions may communicate with the airways. To determine whether, for cysts and cystic lesions, this connection does in fact exist, paired inspiratory and expiratory HRCT scans are necessary.

Occupational lung disease comprises a wide variety of disorders caused by the inhalation or ingestion of dust particles or noxious chemicals. These disorders include pneumoconiosis, asbestos-related pleural and parenchymal disease, chemical pneumonitis, occupational infection, hypersensitivity pneumonitis, and organic dust toxic syndrome. Most of these disorders produce diffuse lung disease. Although many of the disorders can be detected at chest radiography, high-resolution computed tomography (CT) has been shown to be superior to chest radiography in depicting parenchymal, airway, and pleural abnormalities. Some occupational lung diseases have characteristic radiologic features suggesting the correct diagnosis, whereas in others, a combination of clinical features, related occupational history, radiologic findings, and literature supporting an association between the exposure and the disease process is required for diagnosis. With advances in chest radiology, including high-resolution CT, radiologists play a key role in the clinical evaluation of occupational lung diseases and should continue their involvement in the diagnosis and treatment of these diseases.

Turner syndrome is well known to be associated with significant cardiovascular abnormalities. This paper studied the incidence of cardiovascular abnormalities in asymptomatic adolescent patients with Turner syndrome using multidetector computed tomography (MDCT) instead of echocardiography. Twenty subjects diagnosed with Turner syndrome who had no cardiac symptoms were included. Blood pressure and electrocardiography (ECG) was checked. Cardiovascular abnormalities were checked by MDCT. According to the ECG results, 11 had a prolonged QTc interval, 5 had a posterior fascicular block, 3 had a ventricular conduction disorder. MDCT revealed vascular abnormalities in 13 patients (65%). Three patients had an aberrant right subclavian artery, 2 had dilatation of left subclavian artery, and others had an aortic root dilatation, aortic diverticulum, and abnormal left vertebral artery. As for venous abnormalities, 3 patients had partial anomalous pulmonary venous return and 2 had a persistent left superior vena cava. This study found cardiovascular abnormalities in 65% of asymptomatic Turner syndrome patients using MDCT. Even though, there are no cardiac symptoms in Turner syndrome patients, a complete evaluation of the heart with echocardiography or MDCT at transition period to adults must be performed.

Background: This study was performed to evaluate the prevalence and risk factors on the adverse reactions caused by iodinated contrast media (CM) for computed tomography (CT) examination in a university hospital. Methods: Clinical and demographic data among outpatients with CM use were collected at 0000 University Busanpaik Hospital in Busan, Korea between 2008 and 2010. Adverse reaction rate was calculated by the number of adverse reaction among total outpatients with CM use, which was stratified by seasons and sex. The association of risk factors on adverse reaction was investigated using logistic regression model. Results: The total outpatients and events of administered CMs were 27,587 and 48,616, respectively. The administered CMs were iopromide, iohexol, iobitridol, and iodixanol. Adverse reactions occurred in 300 outpatients among the total outpatients (1.1 %). The number of outpatients administered CM more than twice were 8,348. Among them, outpatients who experienced adverse reaction(s) more than once and twice were 124 (1.5 %) and 26 (0.3 %), respectively. Adverse reaction rate was significantly different by sex(p=0.01). The other risk factors were cancer history (OR 2.57, 95 % CI 2.00-3.31) and previous CM administration (OR 1.89, 95 % CI 1.47-2.44). Urticaria was the most frequent symptoms. Conclusion: Total adverse reaction rate was 1.1 % with most common symptom in skin system. Related risk factors were female, cancer history, and previous CM administration. These results were similar to previous studies and will be contribute to clinical practice and future research especially in Koreans.

Bone metastasis of primary colorectal cancer is uncommon. When it occurs, it is usually a late manifestation of disease and is indicative of poor prognosis. We describe a patient with multiple metachronous bone metastases from lower rectal cancer who was successfully treated with multimodal treatment including surgical resections and has shown 32 mo disease-free survival. Surgical resection of metastatic bone lesion(s) from colorectal cancer may be a good treatment option in selected patients.

Purpose: To evaluate the incidence of malignancy-mimicking sclerosed thyroid nodules, from long-term follow-up ultrasonography (US) after an US-guided percutaneous ethanol injection (PEI). Materials and Methods: We examined 86 benign cystic thyroid nodules from 80 patients. The nodules were classified into two groups based on whether an aspiration (Group A, n=26) or non-aspiration (Group B, n=60) of infused ethanol was performed. The final follow-up US over 12 months was performed in all patients. Results: Of the 86 nodules, the cystic portion of 82 (95.3%) cases, from 76 patients, completely disappeared subsequent to the first follow-up US (Group A, n=24, Group B, n=58) (Chi-square test, p>0.05). Moreover, 46 sclerosed thyroid nodules showed two or more of the 'five sonographic criteria' upon a follow-up US (Group A, n=13, 50.0%, Group B, n=32, 53.3%). A higher ratio of the cystic portion of the nodules was associated with a higher incidence of the 'five sonographic criteria' for malignancies detected via a follow-up US (p

Transverse testicular ectopia (TTE) is a rare congenital anomaly in which both testes migrate toward the same hemiscrotum. In most cases, the correct diagnosis is not made preoperatively, but it's made during an inguinal herniotomy or during surgical exploration for an undescended testis because TTE is clinically misdiagnosed as an symptomatic inguinal hernia or as a tumor of the testis on the side to which the ectopic testis has migrated or as an undescended testis on the contralateral side. US and MR imaging can detect the transverse testicular ectopia by its characteristic appearance and so provide useful information about any associated anomalies. We report here on a case of transverse testicular ectopia that was diagnosed by US and MR imaging in a 10-month-old boy, and we review the relevant literature.

Angiosarcoma is a rare primary malignancy occurring anywhere in the body, but most often in the skin, liver or breast. The radiological findings of angiosarcoma of the breast have rarely been reported. We encountered a case in which the condition involved a 32-year-old woman in whom mammography revealed a obscuved- marginated and lobular-shaped high-density lesion, and ultrasonography demonstrated an ovalshaped, microlobular-marginated, inhomogenous hypoechoic mass which metastasized to the whole body after surgery. We report the radiological findings of this case of angiosarcoma of the breast, and review the literature.

Hyperimmunoglobulin E (Job's) syndrome is a rare hereditary immunodeficiency disease characterized by recurrent infection by staphylococcus aureus, extremely high levels of serum immunoglobulin E, recurrent pneumonia, and air cyst. We report the radiologic findings of recurrent pneumonia and air cyst in a nine-yearold child with hyperimmunoglobulin E syndrome, and review the literature.

A localized fibrous tumor of the pleura (LFTP) is a slow growing, rare primary pleural neoplasm. Typically, it takes the form of a well-delineated large mass lesion based on the pleura. Several radiologic descriptions of benign LFTP have been published, but the radiologic findings of malignant LFTP involving invasion of the chest wall have not been reported. We describe a case of malignant LFTP with chest wall invasion, confirmed pathologically, and review the literature

Purpose: To describe the angiographic findings of patients with recurrent hemoptysis after bronchial artery embolization (BAE) according to the point at which relapse occurred. Materials and Methods: From 125 patients who underwent BAE due to hemoptysis between 1996 and 2000, we selected 18 of 23 who underwent additional BAE due to recurrent bleeding after initial BAE . Depending on the point at which relapse occurred, they were divided into two groups (I and II, according to whether additional BAE was performed within two weeks of initial BAE or more than two weeks after this). We retrospectively compared the two groups in terms of angiographic findings, number of embolized arteries, and character of feeding arteries at initial and additional BAE. Results: Nine patients in group I (additional BAE: n=10) and nine in group II (additional BAE: n=13) were admitted for recurrent hemoptysis within two weeks of initial BAE and more than two weeks after this, respectively. In group I(n=29) and II(n=31), angiography demonstrated two direct and 27 indirect, and two direct and 29 indirect signs of hemorrhage, respectively. No statistically significant differences were observed (x=0.005, p=0.945). Among the embolized feeder ressels in group I (n=30) there were 20 bronchial artery and 10 non bronchial systemic collaterals, while for group II(n=35), the corresponding totals were 21 and 14. Again, no statistically significant differences were encountered(x=0.308; p=0.579). In group I, feeders were newly developed in one case(10%), previously embolized in five(50%), and missed in four(40%), while in group two the corresponding figures were none, twelve(92.3%), and one(7.7%). No significant differences were noted, though the incidence of previously embolized feeders in Group II was very high (x=5.383,p=0.068). Conclusion: Among patients in whom hemoptysis after BAE recurred at different times, the angiographic findings and number of embolized arteries were not significantly different, but differences in the nature of the feeder were noted. Patients in whom hemoptysis recurred more than two weeks after BAE showed more recanalization of previously embolized feeders than those in whom there was recurrence within two weeks.

Purpose : To determine the rate of successful biopsy and frequency of post-biopsy pneumothorax and hemorrhage using an 18-gauge cutting needle in CT-guided automated needle biopsies according to lesion size, and the distance between lesion and pleura. Materials and Methods : Ninety-four patients with focal lung lesion who had undergone CT-guided automated needle biopsies using an 18-gauge cutting needle were retrospectively reviewed. We evaluated the relationship between successful biopsy rate and pneumothorax and hemorrhage according to lesion size and distance between lesion and pleura. For the purposes of this study, size and distance were grouped as follows : ≤1 cm ; 1.1 -2cm; 2.1 -3 cm ; >3cm . Results : Biopsy was successful in 78 of 94 patients(83%). When lesions were larger than 2cm in size the diagnostic rate increased (P=0.002), but the distance between lung lesion and pleura was not statistically related to successful biopsy (P>0.005). Where there were post-biopsy complications, the pneumothorax rate was higher in lesions less than 2 cm in size (P=0.041) and in those separated by more than 2 cm from the pleura (P=0.006). Where separation was of this order, the hemorrhage rate was higher (P=0.021), but statistically, this was not affected by lesion size (P>0.05). Conclusion : When 18-gauge cutting needle is used in CT-guided automated needle biopsies of pulmonary lesions, the rate of successful biopsy is affected by lesion size ; the pneumothorax rate is also affected by lesion size, as well as by the distance between lesion and pleura; the hemorrhage rate is influenced only by the dis-tance between lesion and pleura.