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Purpose: This study aimed to investigate the expression levels of progranulin (PGRN) in the tears of patients with diabetic retinopathy (DR) versus healthy controls. Additionally, we sought to explore the correlation between PGRN levels and the severity of ocular surface complications in patients with diabetes. Methods: In this prospective, single-visit, cross-sectional study, patients with DR (n = 48) and age-matched healthy controls (n = 22) were included and underwent dry eye examinations. Tear fluid was collected, and its components were analyzed using the Luminex assay. The subbasal nerve plexus of all participants was evaluated by in vivo confocal microscopy. Results: Patients with DR exhibited more severe dry eye symptoms, along with a reduction in nerve fiber density, length, and branch density within the subbasal nerve plexus, accompanied by an increase in the number of dendritic cells. Tear PGRN levels were also significantly lower in patients with diabetes than in normal controls, and the levels of some inflammatory factors (TNF-α, IL-6, and MMP-9) were higher in patients with DR. Remarkably, the PGRN level significantly correlated with nerve fiber density (R = 0.48, P < 0.001), nerve fiber length (R = 0.65, P < 0.001), and nerve branch density (R = 0.69, P < 0.001). Conclusions: Tear PGRN levels might reflect morphological changes in the corneal nerve plexus under diabetic conditions, suggesting that PGRN itself is a reliable indicator for predicting the advancement of neurotrophic keratopathy in patients with diabetes. Translational Relevance: PGRN insufficiency on the ocular surface under diabetic conditions was found to be closely associated with nerve impairment, providing a novel perspective to discover the pathogenesis of diabetic complications, which could help in developing innovative therapeutic strategies.
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Biomarcadores , Córnea , Diabetes Mellitus Tipo 2 , Retinopatía Diabética , Progranulinas , Lágrimas , Humanos , Lágrimas/metabolismo , Lágrimas/química , Masculino , Femenino , Progranulinas/metabolismo , Persona de Mediana Edad , Estudios Transversales , Estudios Prospectivos , Biomarcadores/análisis , Biomarcadores/metabolismo , Diabetes Mellitus Tipo 2/complicaciones , Diabetes Mellitus Tipo 2/metabolismo , Córnea/inervación , Córnea/metabolismo , Córnea/patología , Retinopatía Diabética/metabolismo , Retinopatía Diabética/diagnóstico , Retinopatía Diabética/patología , Anciano , Microscopía Confocal , Síndromes de Ojo Seco/metabolismo , Síndromes de Ojo Seco/diagnóstico , Síndromes de Ojo Seco/etiología , Síndromes de Ojo Seco/patología , Fibras Nerviosas/patología , Fibras Nerviosas/metabolismoRESUMEN
Background and Objective: To compare the efficacy of EUS-guided celiac plexus neurolysis (CPN) and celiac plexus irradiation with iodine-125 (125I) seeds with absolute ethanol for relieving pain in patients with advanced pancreatic cancer. Methods: We retrospectively analyzed data of 81 patients with advanced pancreatic cancer who underwent EUS-CPN or EUS-125I implantation between January 2017 and December 2020. Postoperative pain was assessed using visual analog scale (VAS) scores; self-assessments of quality of life and the median survival time were compared between the 2 groups. Results: EUS-CPN and 125I implantation were performed in 43 and 38 patients, respectively. Postoperative VAS scores were significantly lower than the preoperative levels in both groups. One week after the operation, 26 patients (60.5%) in the EUS-CPN group achieved partial pain relief, whereas no patients in the EUS-125I seed group experienced pain relief. However, after 4 weeks postoperatively, VAS scores had decreased, and the rate of partial pain relief was higher for EUS-125I seeds than for EUS-CPN. Self-assessments of quality of life were similar in both groups during the first 1 month after the procedure. Conclusions: Both EUS-CPN and EUS-125I seeds can safely and effectively relieve pain in patients with advanced pancreatic cancer. Although EUS-125I seeds take additional time to show effects, the extent and duration of pain relief are better compared with CPN, and interestingly, the median survival time was different.
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BACKGROUND: Sesterterpenoids are rare species among the terpenoids family. Ophiobolins are sesterterpenes with a 5-8-5 tricyclic skeleton. The oxidized ophiobolins exhibit significant cytotoxic activity and potential medicinal value. There is an urgent need for large amounts of ophiobolins supplication for drug development. The synthetic biology approach has been successfully employed in lots of terpene compound production and inspired us to develop a cell factory for ophiobolin biosynthesis. RESULTS: We developed a systematic metabolic engineering strategy to construct an ophiobolin biosynthesis chassis based on Saccharomyces cerevisiae. The whole-cell biotransformation methods were further combined with metabolic engineering to enhance the expression of key ophiobolin biosynthetic genes and improve the supply of precursors and cofactors. A high yield of 5.1 g/L of ophiobolin F was reached using ethanol and fatty acids as substrates. To accumulate oxidized ophiobolins, we optimized the sources and expression conditions for P450-CPR and alleviated the toxicity of bioactive compounds to cells through PDR engineering. We unexpectedly obtained a novel ophiobolin intermediate with potent cytotoxicity, 5-hydroxy-21-formyl-ophiobolin F, and the known bioactive compound ophiobolin U. Finally, we achieved the ophiobolin U titer of 128.9 mg/L. CONCLUSIONS: We established efficient cell factories based on S. cerevisiae, enabling de novo biosynthesis of the ophiobolin skeleton ophiobolin F and oxidized ophiobolins derivatives. This work has filled the gap in the heterologous biosynthesis of sesterterpenoids in S. cerevisiae and provided valuable solutions for new drug development based on sesterterpenoids.
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Ingeniería Metabólica , Saccharomyces cerevisiae , Sesterterpenos , Sesterterpenos/metabolismo , Saccharomyces cerevisiae/metabolismo , Saccharomyces cerevisiae/genéticaRESUMEN
It is important to improve the production of bioactive secondary products for drug development. The Escherichia coli-Streptomyces shuttle vector pSET152 and its derived vector pIB139 containing a strong constitutive promoter ermEp* are commonly used as integrative vectors in actinomycetes. Four new integrative vectors carrying the strong constitutive promoter kasOp*, hrdBp, SCO5768p, and SP44, respectively, were constructed and proven to be functional in different mangrove-derived Streptomyces host strains by using kanamycin resistance gene neo as a reporter. Some biosynthetic genes of elaiophylins, azalomycin Fs, and armeniaspirols were selected and inserted into these vectors to overexpress in their producers including Streptomyces sp. 219807, Streptomyces sp. 211726, and S. armeniacus DSM 43125, resulting in an approximately 1.1-1.4-fold enhancement of the antibiotic yields.
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Actinobacteria , Streptomyces , Streptomyces/genética , Antibacterianos , Regiones Promotoras Genéticas/genética , Vectores Genéticos , Actinobacteria/genética , PlásmidosRESUMEN
AIM: To evaluate the efficacy and safety of silicone oil (SO) as a corneal lubricant to improve visualization during vitrectomy. METHODS: Patients who underwent vitreoretinal surgery were divided into two groups. Group 1 was operated on with initial SO (Oxane 5700) as a corneal lubricant. Group 2 was operated on with initial lactated ringer's solution (LRS) and then replaced with SO as required. Fundus clarity was scored during the surgery. Fluorescein staining was performed to determine the damage to corneal epithelium. RESULTS: Totally 114 eyes of 114 patients were included. Single SO use maintained a clear cornea and provided excellent visualization of surgical image. In group 1, the fundus clarity was grade 3 in 41/45 eyes and grade 2 in 4/45 eyes. In group 2, corneal edema frequently occurred after initial LRS use. The fundus clarity was grade 3 in 19/69 eyes, 2 in 37/69 eyes and 1 in 13/69 eyes (P<0.05). SO was applied in 29 eyes of initial LRS use with subsequent corneal edema, which eliminated the corneal edema in 26 eyes. Corneal fluorescein staining score in group 1 was 0 in 28 eyes, 1 in 11 eyes and 2 in 6 eyes, and 40, 20 and 9, respectively, in group 2 (all P>0.05). CONCLUSION: The use of SO as a corneal lubricant is effective and safe for preserving and improving corneal clarity and providing clear surgical field during vitrectomy.
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Objective@#To explore the longitudinal correlation between smartphone multitasking and depressive symptoms, so as to provide an evidence based basis for promoting the mental health of college students.@*Methods@#A total of 967 college students were recruited from one university in Taiyuan, Chongqing, and Shenzhen cities, China, by using multi stage randomized cluster sampling from October to December 2021 at baseline, and a follow up survey was conducted in May 2022. Smartphone multitasking behaviors were assessed by means of the Assessment of Smartphone Multitasking for Adolescents (ASMA), and depressive symptoms were assessed using the Patient Health Questionnaire-9 (PHQ-9) among college students. Chi square tests were performed to compare the differences in depressive symptoms between different groups of demographic characteristics, and binary Logistic regression models were employed to analyze the associations between smartphone multitasking and depressive symptoms among college students.@*Results@#The rates of depressive symptoms among college students at baseline and follow up were 35.2% and 42.3%, respectively. Compared to the low level smartphone multitasking index group at baseline, the moderate and high level groups were more likely to experience depressive symptoms at baseline (moderate level group: OR=1.74, 95%CI =1.22-2.50, high level group: OR=2.77, 95%CI =1.94-3.95) and followup (moderate level group: OR=1.41, 95%CI =1.01-1.95, high level group: OR=1.64, 95%CI =1.17-2.29) ( P <0.05). In addition, compared to the persistently low smartphone multitasking index, increased risk of depressive symptoms was associated with maintaining a moderate to high ( OR=2.94, 95%CI =1.83-4.71), and a higher ( OR=2.07, 95%CI =1.31-3.27) or lower smartphone multitasking index ( OR=2.02, 95%CI =1.27-3.19) ( P <0.05). Moreover, higher smartphone multitasking index scores were positively associated with the risk of new-onset depressive symptoms at follow up ( OR=1.87, 95%CI=1.07-3.27, P <0.05).@*Conclusions@#Smartphone multitasking behaviors are find to be associated with an increased risk of depressive symptoms in college students. There is a need to reduce smartphone multitasking in order to decrease depressive symptoms and promote students mental health.
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Background: Early-stage ampullary adenomas have only been reported in a small case series on endoscopic management. Hence, this study aimed to evaluate the long-term outcomes of early ampullary adenoma with endoscopic management and identify the risk factors for acute pancreatitis after endoscopic papillectomy (EP). Methods: In this study, 115 patients who underwent EP at Changhai Hospital (Shanghai, China) between January 2012 and December 2018 were retrospectively analysed. Endoscopy was performed at 1, 3, 6, and 12 months after EP. Data were statistically analysed using the t-test or the Mann-Whitney U test. Results: A total of 107 patients were included in this study and the follow-up period was 75 ± 43 months. The average age of the 107 patients was 54.6 years and the average tumor size was 17 mm. The average age of the patients (53.7 ± 10.7 years vs 55.2 ± 10.5 years, P = 0.482), minimum tumor size (13 vs 19 mm, P = 0.063), and complete resection rate (84.78% vs 85.25%, P = 0.947) did not differ significantly between the stent placement and non-stent placement groups. Post-EP acute pancreatitis rates in the non-stent placement and stent placement groups were 11.48% and 4.35%, respectively. The risk of post-EP acute pancreatitis was significantly associated with the preoperative carcinoembryonic antigen level in univariate analysis, but not in multivariate analysis. The risk of post-EP acute pancreatitis was not significantly associated with the placement of the pancreatic stent in either univariate or multivariate analysis. Moreover, delayed proximal pancreatic duct stenosis was not noted in either group during long-term follow-up. Conclusions: EP is a satisfactory option for treating adenomas of the ampulla of the duodenum.
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INTRODUCTION/AIMS: Some patients with Hirayama disease (HD) may have generalized joint hypermobility (GJH), which may excessively increase cervical range of motion (ROM) and then worsen the HD. The purpose of this study was to identify the frequency of GJH in HD patients and to analyze the effect of GJH on cervical ROM and the severity of HD. METHODS: The Beighton scoring system (≥4) was used to diagnose GJH in 84 HD patients. All patients underwent assessments of cervical-flexion/extension ROM; motor unit number estimation in bilateral abductor pollicis brevis (APB) muscles; handgrip strength; and the disabilities of the arm, shoulder, and hand assessments. RESULTS: Concomitant GJH was identified in 20 (23.8%) HD patients. The HD patients with GJH exhibited greater cervical-flexion (P < .001) and cervical-extension (P = .033) ROM than those without GJH. Both greater single motor unit potential amplitudes (symptomatic side: P = .005; less-symptomatic side: P = .011) and lower motor unit numbers (symptomatic side: P = .008; less-symptomatic side: P = .013) in bilateral APB, along with lower compound muscle action potential amplitudes on the symptomatic-side APB (P = .039), were observed in patients with GJH than those without GJH. There was a mild negative correlation between motor unit number and cervical-flexion ROM in HD patients (symptomatic side: r = -0.239, P = .028; less-symptomatic side: r = -0.242, P = .027). DISCUSSION: The frequency of GJH in HD patients may be higher than in the general population. Importantly, GJH may exacerbate excessive cervical-flexion ROM, thereby worsening motor unit loss in HD patients. A cautious approach should be taken when treating HD due to possible comorbid GJH.
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Background: The phenotype of peripheral neuropathy (PN) associated with glial fibrillary acidic protein-immunoglobulin G (GFAP-IgG) has not been well described. Objectives: The aim of this study was to report the frequency, clinical, and electrophysiological characteristics of PN in GFAP-IgG-positive patients. Design: This study is a single-center retrospective observational study. Data Sources and methods: GFAP-IgG-positive patients with PN were retrospectively identified from the Huashan Hospital Autoimmune Encephalitis Cohort between 2017 and 2021. Eight patients who presented with PN from other published studies were also included in the analysis. The clinical and electrophysiological characteristics of GFAP-IgG-related PN were described. Results: A total of 21 (31%) patients (7 females, 14 males; M age: 42 ± 16 years) from a cohort of 68 GFAP-IgG-positive patients presented with PN. Twenty of 21 patients had symmetrical weakness. Sensory and autonomic symptoms were present in 16 and 15 patients, respectively. Lower extremities were the most frequently involved regions for both motor (20/21) and sensory (15/21) symptoms. Moreover, 13 patients (4 females, 9 males; M age: 43 ± 13 years) had electrodiagnostic study data, and 12 of 13 patients had abnormal findings. Regarding clinical features, motor nerve fibers were predominantly involved (12/13), and symmetric lower extremities (12/13) were the most commonly affected regions. Axonal neuropathy is the typical underlying pathophysiologic process of PN. All 21 patients responded to immunotherapy. However, four patients with tetraplegia had poor outcomes, and PN was the major determinant of their long-term disability. Most cases (6/8) from the literature presented with similar clinical and electrophysiological features to those from our cohort. Conclusion: Peripheral nerves could be involved in autoimmune GFAP astrocytopathy. Predominant motor axonal neuropathy mainly involving the lower extremities is the most common PN phenotype in this disorder. GFAP-IgG-related PN is responsive to immunotherapy. Registration: Chinese Clinical Trial Registry: ChiCTR2000029115 (http://www.chictr.org).
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BACKGROUND: This study aimed to investigate the role of neurofascin186 (NF186) in the pathogenesis of the concurrent focal segmental glomerulosclerosis (FSGS) in CIDP-like autoimmune nodopathy patients. METHODS: We presented a case of CIDP-like autoimmune nodopathy complicated with FSGS. We measured NF186 antibodies by cell-binding assay (CBA) method. We performed immunofluorescence analysis in the renal cryosection samples from a patient with minimal nephropathy with rabbit anti-NF186 antibody or NF186 antibody positive human serum. Then we performed western blotting of recombinant NF186 protein and component of NF186 including Ig and FNIII domains incubating with human serum and corresponding rabbit polyclonal antibody. Cases of CIDP complicated with FSGS were searched form PubMed and reviewed. RESULTS: We reported a 66-year-old Chinese woman with CIDP-like autoimmune nodopathy and concurrent FSGS. Her NF186 antibody was positive. The fluorescent signal for NF186 was detected in the renal tissue sections of the patient with minimal nephropathy. The staining for NF186 matched the podocyte spatially. In western blotting analysis, patients had antibodies in their serum recognizing the NF186 protein and their antibodies recognized the Ig domain of NF186. 3 cases of CIDP-like autoimmune nodopathy with positive NF186 antibody and FSGS have been reported. All these patients were responsive to corticosteroids rather than the intravenous immunoglobulin, in terms of both the neuropathy and renal disease. CONCLUSIONS: NF186 was probably a targeted antigen in the pathogenesis of concurrent FSGS in CIDP-like autoimmune nodopathy with positive NF186 antibody. CIDP-like autoimmune nodopathy with positive NF186 antibody and FSGS is a rare entity, which may be responsive to corticosteroids combined with immunosuppressant.
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Glomeruloesclerosis Focal y Segmentaria , Polirradiculoneuropatía Crónica Inflamatoria Desmielinizante , Humanos , Femenino , Animales , Conejos , Anciano , Glomeruloesclerosis Focal y Segmentaria/complicaciones , Glomeruloesclerosis Focal y Segmentaria/patología , Polirradiculoneuropatía Crónica Inflamatoria Desmielinizante/complicaciones , Anticuerpos , Inmunoglobulinas IntravenosasRESUMEN
AIM: To introduce a simple resistance controlled suprachoroidal space (SCS) injection technique using a disposable 30-gauge needle connected to a 1 mL syringe and evaluate the effectiveness and applicability of this technique in the treatment of macular edema. METHODS: A total of 20 patients with various types of macular edema were subjected to a resistance controlled SCS injection of triamcinolone acetonide (TA) with a disposable 30-gauge needle connected to a 1 mL syringe. This technique allows the easy and smooth injection of the TA only once the tip of the needle reached the potential SCS which was indicated by the lower resistance on the plunger. The main outcome measures were anterior segment spectral-domain optical coherence tomography (AS-OCT) measurements post-operation immediately and central subfield thickness (CST), best-corrected visual acuity (BCVA), and intraocular pressure (IOP) measurements at 3mo post-operation. RESULTS: AS-OCT examination showed the expansion of the SCS near the injection site immediately after SCS injection. At three months of follow-up, as compared to the baseline, the mean CST was significantly decreased from 535.0±157.24 to 319.55±127.30 µm (P<0.001), the mean BCVA was significantly improved from 1.05±0.41 to 0.73±0.41 logMAR (P<0.001), and the mean IOP was not significantly different, from 15.05±2.54 to 15.85±3.60 mm Hg (P=0.185). Any complication related to the injection procedure including cataract, choroidal and retinal hemorrhage, retinal detachment, or endophthalmitis was not observed in this study. CONCLUSION: The simple and minimally invasive technique of SCS injection of TA with a disposable 30-gauge needle connected to a 1 mL syringe is useful and applicable for macular edema.
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Benzylisoquinoline alkaloids (BIAs) are a class of plant secondary metabolites with great pharmacological value. Their biosynthetic pathways have been extensively elucidated in the species from the Ranunculales order, such as poppy and Coptis japonica, in which methylation events play central roles and are directly responsible for BIA chemodiversity. Here, we combined BIA quantitative profiling and transcriptomic analyses to identify novel BIA methyltransferases (MTs) from Liriodendron chinense, a basal angiosperm plant. We identified an N-methyltransferase (LcNMT1) and two O-methyltransferases (LcOMT1 and LcOMT3), and characterized their biochemical functions in vitro. LcNMT1 methylates (S)-coclaurine to produce mono- and dimethylated products. Mutagenesis experiments revealed that a single-residue alteration is sufficient to change its substrate selectivity. LcOMT1 methylates (S)-norcoclaurine at the C6 site and LcOMT3 methylates (S)-coclaurine at the C7 site, respectively. Two key residues of LcOMT3, A115 and T301, are identified as important contributors to its catalytic activity. Compared with Ranunculales-derived NMTs, Magnoliales-derived NMTs were less abundant and had narrower substrate specificity, indicating that NMT expansion has contributed substantially to BIA chemodiversity in angiosperms, particularly in Ranunculales species. In summary, we not only characterized three novel enzymes that could be useful in the biosynthetic production of valuable BIAs but also shed light on the molecular origin of BIAs during angiosperm evolution.
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Alcaloides , Bencilisoquinolinas , Liriodendron , Magnoliopsida , Bencilisoquinolinas/metabolismo , Magnoliopsida/genética , Magnoliopsida/metabolismo , Metiltransferasas/metabolismo , Liriodendron/metabolismo , Alcaloides/metabolismoRESUMEN
Objectives: Spinal muscular atrophy with lower extremity predominance 1 (SMALED1) and Charcot-Marie-Tooth diseasetype 2O (CMT2O) are two kinds of hereditary neuromuscular diseases caused by DYNC1H1 mutations. In this study, we reported two patients with SMALED1 caused by DYNC1H1 mutations. The genotype-phenotype correlations were further analyzed by systematically reviewing previous relevant publications. Materials and Methods: Two patients' with SMALED1 and their parents' clinical data were collected, and detailed clinical examinations were performed. WES was then applied, which was confirmed by Sanger sequencing. PubMed, Web of Science, CNKI, and Wanfang Data were searched, and all publications that met the inclusion criteria were carefully screened. Any individual patient without a detailed description of clinical phenotypes was excluded. Results: The two patients manifested delayed motor milestones and muscle wasting of both lower extremities. The diagnosis was further confirmed as SMALED1. Genetic testing revealed heterozygous DYNC1H1 mutations c.1792C>T and c.790C>G; the latter is a novel dominant mutation. Genotype-phenotype analysis of DYNC1H1 variants and neuromuscular diseases revealed that mutations in the DYN1 region of DYNC1H1 protein were associated with a more severe phenotype, more complicated symptoms, and more CNS involvement than the DHC_N1 region. Conclusion: Our study potentially expanded the knowledge of the phenotypic and genetic spectrum of neuromuscular diseases caused by DYNC1H1 mutations. The genotype-phenotype correlation may reflect the pathogenesis underlying the dyneinopathy caused by DYNC1H1 mutations.
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Choroidal vascular diseases, such as age-related macular degeneration, are the leading cause of vision impairment and are characterized by pathological angiogenesis. Verteporfin-mediated photodynamic therapy is a current strategy that selectively occludes choroidal neovasculature. However, the clinically used large-dose systemic administration increases the risk of systemic adverse events, such as phototoxicity to superficial tissues. In this study, we developed an in situ verteporfin delivery system with a photoswitching synergistic function that disassembles in response to intraocular inflammatory enzymes. Under light-on conditions, verteporfin-mediated photodynamic therapy effectively occurs and this leads to vascular occlusion. Under light-off conditions, non-photoactive verteporfin negatively regulates vascular endothelial growth factor-induced angiogenesis as a yes-associated protein inhibitor. Taken together, our system serves as an intraocular verteporfin reservoir to improve the bioavailability of verteporfin by innovatively exploiting its photochemical and biological functions. This work provides a promising strategy with synergistic antiangiogenic effects for the treatment of choroidal vascular diseases.
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Antibiotic resistance in Helicobacter pylori has been growing worldwide with current treatment regimens. Development of new compounds for treatment of H. pylori infections is urgently required to achieve a successful eradication therapy in the future. Armeniaspirols, a novel class of natural products isolated from Streptomyces armeniacus, have been previously identified as antibacterial agents against Gram-positive pathogens. In this study, we found that armeniaspirol A (ARM1) exhibited potent antibacterial activity against H. pylori, including multidrug-resistant strains, with MIC range values of 4-16 µg ml-1 . The underlying mechanism of action of ARM1 against H. pylori involved the disruption of bacterial cell membranes. Also, ARM1 inhibited biofilm formation, eliminated preformed biofilms and killed biofilm-encased H. pylori in a dose-dependent manner. In a mouse model of multidrug-resistant H. pylori infection, dual therapy with ARM1 and omeprazole showed efficient in vivo killing efficacy comparable to the standard triple therapy, and induced negligible toxicity against normal tissues. Moreover, at acidic pH 2.5, ARM1 exhibited a much more potent anti-H. pylori activity than metronidazole. Thus, these findings demonstrated that ARM1 is a novel potent anti-H. pylori agent, which can be developed as a promising drug lead for treatment of H. pylori infections.
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Infecciones por Helicobacter , Helicobacter pylori , Animales , Antibacterianos/farmacología , Infecciones por Helicobacter/tratamiento farmacológico , Infecciones por Helicobacter/microbiología , Metronidazol/farmacología , Metronidazol/uso terapéutico , Ratones , Pirroles , Compuestos de EspiroAsunto(s)
Lentes Intraoculares , Vitrectomía , Cámara Anterior/cirugía , Drenaje , Humanos , Técnicas de Sutura , SuturasRESUMEN
OBJECTIVE: To investigate the impact of early vs. delayed surgical decompression on peripheral motor axonal dysfunction following acute traumatic central cord syndrome (ATCCS). METHODS: Both axonal excitability testing and motor unit number estimation (MUNE) were performed in 30 ATCCS patients (early- vs. delayed-surgical treatment: 12 vs. 18) before operation and 28 healthy subjects. Axonal excitability testing was repeated 3-5 days and 1-year after operation, and MUNE was re-evaluated 1-year after operation. RESULTS: Preoperatively, an obvious modification in membrane potentials was observed in ATCCS patients that mostly coincided with depolarization-like features, and MUNE further revealed reduced motor units in tested muscles (P < 0.05). Unlike delayed-surgical cases, early-surgical cases showed recoveries of most measurements of axonal excitabilities soon after operation (P < 0.05). Postoperative one-year follow-up demonstrated that greater motor unit numbers in tested muscles were obtained in early-surgical cases than in delayed-surgical cases (P < 0.05). CONCLUSIONS: ATCCS has adverse downstream effects on peripheral nervous system, even in the early stage of ATCCS. Early surgical treatment can ameliorate both excitability abnormalities and motor unit loss in distal motor axons. SIGNIFICANCE: Optimizing axonal excitability in the early phases of ATCCS may alleviate peripheral nerve injury secondary to lesions of upper motor neuron and improve clinical outcomes.
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Potenciales de Acción/fisiología , Axones/fisiología , Síndrome del Cordón Central/fisiopatología , Neuronas Motoras/fisiología , Conducción Nerviosa/fisiología , Adulto , Anciano , Síndrome del Cordón Central/complicaciones , Síndrome del Cordón Central/diagnóstico , Estudios de Cohortes , Femenino , Estudios de Seguimiento , Humanos , Masculino , Persona de Mediana Edad , Estudios Prospectivos , Prevención Secundaria/métodosRESUMEN
OBJECTIVE: To assess the feasibility of motor unit number index (MUNIX) in quantitatively evaluating Hirayama disease (HD) with proximal involvement and to identify the effectiveness of anterior cervical fusion (ACF) in treating atypical HD with proximal involvement. METHODS: This study included 28 atypical HD patients with proximal involvement (proximal-distal vs. distal-proximal groups: 5 vs. 23) and 41 healthy controls. All patients underwent pre- and postoperative 1-year MUNIX tests on abductor pollicis brevis (APB), abductor digiti minimi (ADM), biceps brachii (BB) and deltoid (Del). The disabilities of arm, shoulder and hand (DASH) and Medical Research Council (MRC) scales were also performed in these patients before and one year after operation. RESULTS: Preoperatively, the patients in the distal-proximal group showed reduced compound muscle action potential (CMAP), decreased MUNIX and increased motor unit size index (MUSIX) in bilateral distal muscles and symptomatic-side proximal muscles (Pâ¯<â¯0.05), and similar abnormalities were also observed in ADM, BB and Del on the symptomatic side in the proximal-distal groups (Pâ¯<â¯0.05). Postoperative follow-up analysis identified increased MUNIX in the symptomatic-side proximal muscles with improved motor function in the proximal-distal groups (Pâ¯<â¯0.05), and distal-proximal group patients showed an increase in both CMAP and MUSIX in the symptomatic-side proximal muscles (Pâ¯<â¯0.05). CONCLUSIONS: MUNIX may serve as an available supplementary test to quantitatively evaluate the motor dysfunction and treatment outcome in HD with proximal involvement. ACF procedures can effectively treat these atypical HD patients, especially for those whose symptoms started in proximal muscles.
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Trastornos Motores , Electromiografía , Humanos , Neuronas Motoras , Músculo Esquelético , Atrofias Musculares Espinales de la Infancia , Resultado del TratamientoRESUMEN
BACKGROUND: To introduce a modified deep anterior lamellar dissection technique to improve visibility during surgery for vitreoretinal diseases with coexisting corneal opacity. CASE PRESENTATION: Two patients with retinal detachment and coexisting corneal blood staining or corneal decompensation underwent modified deep anterior lamellar dissections followed by vitrectomy. The modified deep anterior lamellar dissection techniques, unlike the dissection and removal of corneal lamellar in a typical deep anterior lamellar keratoplasty, included the creation and preservation of a deep lamellar corneal flap, the retroillumination to visualize and easily remove the remaining opaque stroma on the Descemet membrane, and the big air bubble technique in the eye with endothelial decompensation. The patient's own cornea flap was sutured back after vitrectomy was done. The modified dissection techniques provided adequate fundus view during vitrectomy while removing as less corneal tissue as possible and decreasing the surgical complications and the requirement of a fresh cornea. Postoperatively, in case 1, the corneal blood staining was gradually absorbed and the vision improved from light perception to counting fingers. In case 2, even though the cornea remained cloudy and the vision was poor, the cornea endothelial decompensation was stable and asymptomatic. Both retinas were attached after silicone oil removal at 6-month follow-up. CONCLUSIONS: This modified and limited deep anterior lamellar corneal dissection procedure appears to be a useful alternative to penetrating keratoplasty, ophthalmic endoscope and temporary keratoprosthesis during the vitrectomy with coexisting corneal opacity.