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Acute ascending hemorrhagic longitudinally extensive transverse myelitis is a rare inflammatory demyelinating disorder, which invades several vertebral segments and progresses rapidly and manifests severe symptoms. We present a case of acute necrotizing myelitis associated with COVID-19 infection. A 10-year-old female, with no previous medical history and no prior administration of COVID-19 vaccination, contracted COVID-19 in early April 2022. Two weeks later, she suffered from severe posterior neck pain and also presented with motor weakness and numbness in both lower extremities, making it difficult to walk independently and spontaneously void urine. Initial spinal cord MR showed longitudinally segmental extensive T2 hyperintensities. Cerebrospinal fluid (CSF) analysis revealed elevated red blood cell, normal white blood cell, and elevated protein levels and absence of oligoclonal bands. CSF culture and viral polymerase chain reaction were negative. Autoimmune work-up was negative. She was started on intravenous methylprednisolone 1g/day for 5 days and immunoglobulin (Ig) 2 g/kg for 5 days. She was also treated with six courses of therapeutic plasma exchange. Nevertheless, her pain and motor weakness persisted. She eventually developed respiratory failure. Follow-up MR presented a newly noted small hemorrhagic component. She was consequently treated with two additional courses of methylprednisolone and Ig. At 6-months follow-up, neurological examination showed improvement with normal sensory function and motor grade IV function in both upper extremities. We present the case of acute necrotizing myelitis associated with COVID-19 infection. Multiple courses of methylprednisolone and Ig showed mild improvement in motor and sensory function. However, poor prognosis was unavoidable due to rapid progression of the disease.
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Purpose@#To analyze the efficacy and safety of nusinersen in patients with spinal muscular atrophy (SMA) type I with chronic respiratory failure. @*Materials and Methods@#We retrospectively reviewed seven patients diagnosed with SMA type I and chronic respiratory failure who were on permanent ventilation and treated with nusinersen at Gangnam Severance Hospital between January 2018 and July 2023. Patient demographics and clinical characteristics were recorded, and treatment progress was evaluated according to Hammersmith Infant Neurological Examination (HINE-2) and Children’s Hospital of Philadelphia Infant Test of Neuromuscular Disorders (CHOP-INTEND) scores. @*Results@#Patients initially developed hypotonia at a mean age of 3.7 months. Mean age at start of nusinersen was 7.3 years; the mean duration of follow-up after starting nusinersen was 46.2 months. At 6-, 18-, 38-, 58-, and 74-month follow-up, the mean changes in CHOP-INTEND scores were 1.0, 2.9, 1.8, 1.5, and 1.5, respectively, and the proportions of patients who showed disease amelioration were 28.6%, 71.4%, 75.0%, 100%, and 100%, respectively. @*Conclusion@#Nusinersen is safe and effective in patients with SMA type I, even those with chronic respiratory failure and those on permanent ventilation. No significant adverse effects of nusinersen were observed.
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In patients with chronic renal failure, spontaneous tendon rupture may occur due to degenerative changes in the tendon. Transosseous suture and suture anchor are commonly used for repair of quadriceps tendon rupture. But in chronic renal failure patients, the fixation of the repaired tendon is weak due to degenerative changes in the tendon, and decreased bone mineral density results in a relatively high rerupture rate. In this case, spontaneous quadriceps tendon rupture in a patient who has chronic renal failure with dialysis over 10 years was repaired with a newly designed transosseous suture method in order to increase contact of patella base and to reduce the rate of rerupture. Divide ruptured tendon arbitrarily into four layers using the anatomical structure of quadriceps tendon, then pull them out through the three vertical transosseous tunnels and tied them each. This procedure may be considered as another option in the case with high rerupture risk.
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A case of persistent Ralstonia mannitolilytica bacteremia in the neonatal intensive care unit prompted source investigation due to its rarity. After an extensive investigation, a contaminated ultrasonic nebulizer was identified as the source, and the infection was controlled by removing the source. This study emphasizes the importance of further investigations, even in single cases of rare pathogens.
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The aim of this study was to evaluate the differences in setting times based on the methods for dental root canal sealers and calcium silicate cement used in root-end filling. Five kinds of dental root canal sealers and four kinds of calcium silicate cement for root-end filling were selected for the experiments. All materials were mixed according to the manufacturers’ instructions and stored at 37 ℃ with a relative humidity of 95%. Setting time was measured using a 1/4 pound Gillmore needle and a 1 pound Gillmore needle to determine the time until indentation was no longer visible or the time until 2 mm penetration was no longer possible. The determination of indentation was based on the absence of visible impressions on the material surface when Gillmore needle was placed vertically. When comparing indentation time and penetration time using same type of Gillmore needle, only ProRoot MTA using 1 pound Gillmore needle showed significant difference between measuring methods (P0.05). By this study, we could expect to measure a setting time relatively similar to real clinical conditions through indentation method.
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Purpose@#This study investigated changes in the angle of exodeviation at different distances and the characteristics of a group that showed an increased angle following monocular occlusion in intermittent exotropia. @*Methods@#In all, 102 patients diagnosed with basic intermittent exotropia were enrolled. The deviation angle was measured using a mirror at 33 cm, 3 m, 6 m, 12 m, and 6 m. Moreover, the deviation was measured at 33 cm and 3 m after patching the non-dominant eye for 1 hour. Patients with a higher deviation angle at 33 cm after occlusion were categorized as the ‘increased group’, whereas those with lower deviation angles were classified as the ‘stationary group’. @*Results@#There was a male-to-female ratio of 46:56 and an average age at surgery of 9.23 years. No significant differences were observed in deviation measurements taken at 3, 6, 12, and 6 m using a mirror. However, after occlusion, there was a significant increase in deviation at 33 cm (p < 0.01). The average log value of near stereoacuity was 1.96 ± 0.35 in the increased group and 2.16 ± 0.41 in the stationary group. The group with an increased near deviation after occlusion demonstrated a significantly improved near stereoacuity (p = 0.01) and an increased proportion of good results in the near Worth 4 dot test (p = 0.03). @*Conclusions@#Preoperative measurement of maximum deviation after 1 hour of monocular occlusion can help reduce recurrence and insufficient correction, particularly in patients with good fusional ability.
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Neuro-Behcet’s disease (NBD) is defined as a combination of neurologic symptoms and/or signs in a patient with Behcet’s disease. A 38-year-old woman was admitted due to sensory aphasia. She had past medical history of generalized tonic-clonic seizure, retinal vasculitis, oral ulcer and cerebral vasculitis. Brain magnetic resonance imaging showed broad lesion in the left parieto-occipital lobe. Brain biopsy was performed to differentiate between central nervous system lymphoma and NBD. We report a rare case of NBD with extensive involvement of cerebral parenchyma.
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Objective@#: This retrospective study investigated the factors that affect cage obliquity angle despite orthogonal maneuvers performed during oblique lateral interbody fusion (OLIF) and assessed the relationship between cage obliquity angle and radiological outcomes post-surgery. @*Methods@#: Twenty-nine males who underwent L4-L5 OLIF for lumbar degenerative disease between 2019 and 2021 with a followup duration greater than 12 months were analyzed. Radiological parameters were measured including psoas muscle volume, total psoas area index (total psoas muscle area [cm2]/height squared [m2]), distance from the iliac artery to the origin of the psoas muscle (DIAPM), angle between the origin of the psoas muscle and the center of the vertebral disc (APCVD), iliac crest height, disc height, lumbar flexibility (lumbar flexion angle minus extension angle), cage location ratio, cage-induced segmental lumbar lordosis (LL) (postoperative index level segmental LL minus used cage angle), foraminal height changes, fusion grade. @*Results@#: DIAPM, APCVD, iliac crest height, postoperative index level segmental LL, and cage-induced segmental LL were significantly correlated with OLIF cage obliquity angle. However, other radiological parameters did not correlate with cage obliquity. Based on multiple regression analysis, the predictive equation for the OLIF cage obliquity angle was 13.062–0.318×DIAPM+0.325×A PCVD+0.174×iliac crest height. The greater the cage obliquity, the smaller the segmental LL compared to the cage angle used. @*Conclusion@#: At the L4-L5 level, OLIF cage obliquity was affected by DIAPM, APCVD, and iliac crest height, and as the cage obliquity angle increases, LL agnle achievable by the used cage could not be obtained.
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Background@#Paroxysmal nocturnal hemoglobinuria (PNH) is a rare hematologic disorder characterized by uncontrolled terminal complement activation. Eculizumab, a monoclonal antibody C5 inhibitor was introduced in Korea in 2009 and has been the standard treatment option for PNH. @*Methods@#This study assessed the long-term efficacy/safety of eculizumab in PNH using real-world data from the Korean Health Insurance Review and Assessment Service. Eighty patients who initiated eculizumab from 2009–2020 were enrolled. @*Results@#At eculizumab initiation, the median age was 51.5 years, lactate dehydrogenase (LDH) 6.8 × upper limit of normal, and granulocyte clone size 93.0%. All patients had at least one PNH-related complication before eculizumab initiation, including renal failure (n = 36), smooth muscle spasm (n = 24), thromboembolism (n = 20), and pulmonary hypertension (n = 15). The median (range) duration of eculizumab treatment was 52.7 (1.0, 127.3) months (338.6 total treated patient-years). Despite high disease activity in the study population before treatment initiation, overall survival was 96.2% and LDH levels were stabilized in most patients during treatment. PNH-related complications at treatment initiation were resolved in 44.4% of patients with renal failure, 95.8% with smooth muscle spasm, 70.0% with thromboembolism, and 26.7% with pulmonary hypertension. Extravascular hemolysis occurred in 28.8% of patients (n = 23; 0.09 per patient-year) and breakthrough hemolysis in 18.8% (n = 15; 0.06 per patient-year). No treatment discontinuation cases related to eculizumab were observed. @*Conclusion@#These data provided evidence for the long-term efficacy and safety of eculizumab in Korean PNH patients with high disease burdens.
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Background@#Interscalene brachial plexus block (ISB) is a common regional technique to manage acute postoperative pain for arthroscopic rotator cuff tear repair. However, rebound pain may compromise its overall benefit. Our aim was to investigate the primary hypothesis that perineural and intravenous dexamethasone have different effects on rebound pain after resolution of ISB for arthroscopic rotator cuff tear repair. @*Methods@#Patients aged ≥ 20 years scheduled for elective arthroscopic rotator cuff tear repair under general anesthesia with preoperative ISB were included. The participants were randomized to receive dexamethasone either perineurally (perineural group) or intravenously (intravenous group). In the perineural group, patients received ISB with 12 mL of 0.5% ropivacaine containing 5 mg of dexamethasone; simultaneously, 1 mL of 0.9% normal saline was administered intravenously. In the intravenous group, patients received ISB with 12 mL of 0.5% ropivacaine; simultaneously, 1 mL of dexamethasone 5 mg was administered intravenously. The primary outcome was the difference in the pain score (0–10 on numeric rating scale) between before and after ISB resolution. The secondary outcomes were the incidence of rebound pain; onset, duration, and intensity of rebound pain; time to the first analgesic request; and pain-related sleep disturbance. @*Results@#A total of 71 patients were randomized to either perineural group (n = 36) or intravenous group (n = 35). After block resolution, pain scores increased significantly more in the perineural group (mean ± standard deviation, 4.9 ± 2.1) compared to the intravenous group (4.0 ± 1.7, P = 0.043). The duration of ISB was more prolonged in the perineural group (median [interquartile range], 19.9 [17.2–23.1] hours) than the intravenous group (15.1 [13.7–15.9] hours, P < 0.001). The incidence of rebound pain and pain-related sleep disturbance during the first postoperative week was significantly higher in the perineural group than in the intravenous group (rebound pain: 44.4% vs. 20.0%, P = 0.028; sleep disturbance: 55.6% vs. 25.7%, P = 0.011). The duration and intensity of rebound pain were similar between the two groups. @*Conclusion@#Although perineural dexamethasone provided longer postoperative analgesia, intravenous dexamethasone was more beneficial in reducing pain increase after ISB resolution, incidence of rebound pain, and pain-related sleep disturbance.
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Purpose@#This study aimed to assess the feasibility of ultrashort echo time (UTE)-T2* mapping in comparison with T2 mapping for quantitative evaluation of meniscal degeneration. @*Materials and Methods@#This study included 208 menisci of 99 patients (59 women and 40 men, median age 52 years old [16–80 years]) who underwent knee MRI with both standard T2 mapping and UTE-T2* mapping sequences. A radiologist reviewed the images and graded meniscal degeneration according to the morphologic criteria on T2-weighted and proton density-weighted sequences. Manually drawn regions of interest were placed along the outline and hyperintensity subregion within the meniscus, and in the same location on midsagittal images of each T2 and UTE-T2* sequence. Meniscal T2 and T2* values (T2m and T2*m) as well as T2 and T2* values of hyperintensity subregions (T2h, T2*h) were calculated. @*Results@#There was a strong correlation between T2m, T2*m, T2h, and T2*h, and morphological grades (correlation coefficient 0.793–0.943, 95% CI). On morphologic analysis, 50, 52, 50, and 56 menisci were graded as 0, 1, 2, and 3, respectively. T2m, T2*m, T2h, and T2*h were found to be significantly different in all the grades and tended to be higher in the more degraded meniscus (p < 0.001 for both). Mean T2m was 10.78 ± 2.91 ms, 15.81 ± 2.99 ms, 20.26 ± 3.19 ms, and 30.80 ± 7.38 ms and mean T2*m was 7.10 ± 1.12 ms, 9.64 ± 1.27 ms, 12.01 ± 1.58 ms, and 18.98 ± 4.67 ms for grades 0, 1, 2, and 3, respectively. Mean T2h was 20.05 ± 3.67 ms, 24.39 ± 4.73 ms, and 38.92 ± 9.49 ms and mean T2*h was 10.94 ± 1.65 ms, 13.67 ± 2.41 ms, and 22.36 ± 5.20 ms for grades 1, 2, and 3, respectively. @*Conclusion@#UTE-T2* mapping was feasible for quantitative evaluation of meniscal degeneration in patients. With a few improvements UTE-T2* mapping is a potential substitute for the standard T2 mapping, with improved efficacy.
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Purpose@#Prior research has indicated that stroke can influence the symptoms and presentation of neurogenic bladder, with various patterns emerging, including abnormal facial and linguistic characteristics. Language patterns, in particular, can be easily recognized. In this paper, we propose a platform that accurately analyzes the voices of stroke patients with neurogenic bladder, enabling early detection and prevention of the condition. @*Methods@#In this study, we developed an artificial intelligence-based speech analysis diagnostic system to assess the risk of stroke associated with neurogenic bladder disease in elderly individuals. The proposed method involves recording the voice of a stroke patient while they speak a specific sentence, analyzing it to extract unique feature data, and then offering a voice alarm service through a mobile application. The system processes and classifies abnormalities, and issues alarm events based on analyzed voice data. @*Results@#In order to assess the performance of the software, we first obtained the validation accuracy and training accuracy from the training data. Subsequently, we applied the analysis model by inputting both abnormal and normal data and tested the outcomes. The analysis model was evaluated by processing 30 abnormal data points and 30 normal data points in real time. The results demonstrated a high test accuracy of 98.7% for normal data and 99.6% for abnormal data. @*Conclusions@#Patients with neurogenic bladder due to stroke experience long-term consequences, such as physical and cognitive impairments, even when they receive prompt medical attention and treatment. As chronic diseases become increasingly prevalent in our aging society, it is essential to investigate digital treatments for conditions like stroke that lead to significant sequelae. This artificial intelligence-based healthcare convergence medical device aims to provide patients with timely and safe medical care through mobile services, ultimately reducing national social costs.
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Myxoma most commonly occurs in the atria, but is rare in the nasal cavity. A 58-year-old male patient presented with a nasal mass. We used endoscopic endonasal approach for treatment, and the patient was finally diagnosed with nasal ala myxoma. We report here a rare case of a patient with nasal ala myxoma along with a literature review.
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Bilateral asymmetric fracture dislocation of a shoulder joint is a rare subtype of shoulder dislocation and refers to the simultaneous fracture-dislocations of the glenohumeral joints in opposite directions. This paper reports a case of a 43-year-old male patient with a bilateral asymmetric fracture dislocation of the shoulder joints following a hypoglycemic shock who was treated with arthroscopic Bankart repair and the arthroscopic modified McLaughlin procedure. To our knowledge, this case is the first in domestic literature, and therefore the authors report this case with the treatment outcome and a literature review.
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Background/Aims@#Daratumumab has shown an encouraging antitumor effect in patients with multiple myeloma (MM), and was known to alter the immune properties by off-targeting immunosuppressive cells. Here, we aimed to evaluate the change in absolute lymphocyte count (ALC) as a surrogate marker for predicting survival outcomes of patients treated with daratumumab. @*Methods@#Between 2018 and 2021, the medical records of patients with relapsed/refractory MM (RRMM) treated with daratumumab monotherapy at 10 centers in South Korea were reviewed. We collected the ALC data at pre-infusion (D0), day 2 after the first infusion (D2), and prior to the third cycle of daratumumab therapy (D56). @*Results@#Fifty patients who were administered at least two cycles of daratumumab were included. Overall response rate was 54.0% after two cycles of daratumumab treatment. On D2, almost all patients experienced a marked reduction in ALC. However, an increase in ALC on D56 (ALCD56) was observed in patients with non-progressive disease, whereas failure of ALC recovery was noted in those with progressive disease. Patients with ALCD56 > 700/μL (n = 39, 78.0%) had prolonged progression- free survival (PFS) and overall survival (OS) than those with ALCD56 ≤ 700/μL (median PFS: 5.8 months vs. 2.6 months, p = 0.025; median OS: 24.1 months vs. 6.1 months, p = 0.004). In addition, ALCD56 >700/μL was a significant favorable prognostic factor for PFS (hazard ratio [HR], 0.22; p = 0.003) and OS (HR, 0.23; p = 0.012). @*Conclusions@#Increase in ALC during daratumumab treatment was significantly associated with prolonged survival outcomes in patients with RRMM. The ALC value can predict clinical outcomes in patients treated with daratumumab.
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Background@#To report the clinical manifestations of non-arteritic anterior ischemic optic neuropathy (NAION) cases after coronavirus disease 2019 (COVID-19) vaccination in Korea. @*Methods@#This multicenter retrospective study included patients diagnosed with NAION within 42 days of COVID-19 vaccination. We collected data on vaccinations, demographic features, presence of vascular risk factors, ocular findings, and visual outcomes of patients with NAION. @*Results@#The study included 16 eyes of 14 patients (6 men, 8 women) with a mean age of 63.5 ± 9.1 (range, 43–77) years. The most common underlying disease was hypertension, accounting for 28.6% of patients with NAION. Seven patients (50.0%) had no vascular risk factors for NAION. The mean time from vaccination to onset was 13.8 ± 14.2 (range, 1–41) days. All 16 eyes had disc swelling at initial presentation, and 3 of them (18.8%) had peripapillary intraretinal and/or subretinal fluid with severe disc swelling. Peripapillary hemorrhage was found in 50% of the patients, and one (6.3%) patient had peripapillary cotton-wool spots. In eight fellow eyes for which we were able to review the fundus photographs, the horizontal cup/ disc ratio was less than 0.25 in four eyes (50.0%). The mean visual acuity was logMAR 0.6 ± 0.7 at the initial presentation and logMAR 0.7 ± 0.8 at the final visit. @*Conclusion@#Only 64% of patients with NAION after COVID-19 vaccination have known vascular and ocular risk factors relevant to ischemic optic neuropathy. This suggests that COVID-19 vaccination may increase the risk of NAION. However, overall clinical features and visual outcomes of the NAION patients after COVID-19 vaccination were similar to those of typical NAION.
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Intramuscular myxoma is a rare benign myxoid tumor that is difficult to differentiate from other benign soft tissue tumors and sarcoma, and as a result, intramuscular myxoma is commonly misdiagnosed as another type of soft tissue tumor. Accordingly, awareness of the existence of this condition is a fundamental requirement for treatment decision-making. Furthermore, although intramuscular myxoma appears grossly to be well-circumscribed, it can infiltrate adjacent soft tissue microscopically. Tumor resection is the recommended treatment, but appropriate surgical margin sizes remain controversial. To the best of our knowledge, this is the first South Korean report to be issued on the treatment of intramuscular myxoma of the foot.
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Purpose@#Tyrosine kinase inhibitors (TKI) targeting vascular endothelial growth factor receptor (VEGFR) signaling pathways have been used for metastatic clear cell renal cell carcinoma (mCCRCC), but resistance to the drug develops in most patients. We aimed to explore the underlying mechanism of the TKI resistance with regard to programmed death-ligand 1 (PD-L1) and to investigate signaling pathway associated with the resistant mechanism. @*Materials and Methods@#To determine the mechanism of resistance, 10 mCCRCC patients from whom tumor tissues were harvested at both the pretreatment and the TKI-resistant post-treatment period were included as the discovery cohort, and their global gene expression profiles were compared. A TKI-resistant renal cancer cell line was established by long-term treatment with sunitinib. @*Results@#Among differentially expressed genes in the discovery cohort, increased PD-L1 expression in post-treatment tissues was noted in four patients. Pathway analysis showed that PD-L1 expression was positively correlated with the mammalian target of rapamycin (mTOR) signaling pathway. The TKI-resistant renal cancer cells showed increased expression of PD-L1 and mTOR signaling proteins and demonstrated aggressive tumoral behaviour. Treatment with mTOR inhibitors down-regulated PD-L1 expression and suppressed aggressive tumoral behaviour, which was reversed with stimulation of the mTOR pathway. @*Conclusion@#These results showed that PD-L1 expression may be increased in a subset of VEGFR-TKI–resistant mCCRCC patients via the mTOR pathway.
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Purpose@#High-dose chemotherapy followed by autologous stem cell transplantation (ASCT) is the standard management for relapsed or high-risk non-Hodgkin’s lymphoma (NHL). We reported the busulfan, melphalan, and etoposide (BuME) conditioning regimen was effective in patients with relapsed or high-risk NHL. Moreover, the busulfan, cyclophosphamide, and etoposide (BuCE) conditioning regimen has been used widely in ASCT for NHL. Therefore, based on these encouraging results, this randomized phase II multicenter trial compared the outcomes of BuME and BuCE as conditioning therapies for ASCT in patients with NHL. @*Materials and Methods@#Patients were randomly assigned to receive either BuME (n=36) or BuCE (n=39). The BuME regimen was comprised of busulfan (3.2 mg/kg/day, intravenously) administered on days –7, –6, and –5, etoposide (400 mg/m2 intravenously) on days –5 and –4, and melphalan (50 mg/m2/day intravenously) on days –3 and –2. The BuCE regimen was comprised of busulfan (3.2 mg/kg/day intravenously) on days –7, –6, and –5, etoposide (400 mg/m2/day intravenously) on days –5 and –4, and cyclophosphamide (50 mg/kg/day intravenously) on days –3 and –2. The primary endpoint was 2-year progression-free survival (PFS). @*Results@#Seventy-five patients were enrolled. Eleven patients (30.5%) in the BuME group and 13 patients (33.3%) in the BuCE group had disease progression or died. The 2-year PFS rate was 65.4% in the BuME group and 60.6% in the BuCE group (p=0.746). There were no non-relapse mortalities within 100 days after transplantation. @*Conclusion@#There were no significant differences in PFS between the two groups. Therefore, busulfan-based conditioning regimens, BuME and BuCE, may be important treatment substitutes for the BCNU-containing regimens.
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Purpose@#Diffuse large B-cell lymphoma (DLBCL) is the most common hematologic malignancy worldwide. Although substantial improvement has been achieved by the frontline rituximab-based chemoimmunotherapy, up to 40%-50% of patients will eventually have relapsed or refractory disease, whose prognosis is extremely dismal. @*Materials and Methods@#We have carried out two prospective cohort studies that include over 1,500 DLBCL patients treated with rituximab plus CHOP (#NCT01202448 and #NCT02474550). In the current report, we describe the outcomes of refractory DLBCL patients. Patients were defined to have refractory DLBCL if they met one of the followings, not achieving at least partial response after 4 or more cycles of R-CHOP; not achieving at least partial response after 2 or more cycles of salvage therapy; progressive disease within 12 months after autologous stem cell transplantation. @*Results@#Among 1,581 patients, a total of 260 patients met the criteria for the refractory disease after a median time to progression of 9.1 months. The objective response rate of salvage treatment was 26.4%, and the complete response rate was 9.6%. The median overall survival (OS) was 7.5 months (95% confidence interval, 6.4 to 8.6), and the 2-year survival rate was 22.1%±2.8%. The median OS for each refractory category was not significantly different (p=0.529). @*Conclusion@#In line with the previous studies, the outcomes of refractory DLBCL patients were extremely poor, which necessitates novel approaches for this population.