Abstract Introduction: Autoimmune polyglandular syndrome (APS) is a multiorgan genetic autoimmune disease. APS-3B subtype is autoimmune thyroiditis with pernicious anemia. In this case, we will discuss an elderly female patient diagnosed with APS-3B. Case Presentation: A 69-year-old Caucasian female patient with a past medical history of autoimmune thyroiditis presented to the emergency department with a two-month history of generalized weakness and nausea. Associated symptoms included shortness of breath and diarrhea. Review of systems was otherwise unremarkable. Physical exam was positive for depigmented skin macules over the upper extremities. Lab results showed hemoglobin 8.2 [11.7 - 15.5 g/dL], MCV 121[80 - 100 fL], platelets 144,000 [150 - 450 X10E9/L], WBC 1.9 [4.0 - 11.0 X10E9/L], LDH 1153[100 - 235 U/L], TSH 0.28[0.49 - 4.67 uIU/mL], free T4 1.7 [0.61 - 1.60 ng/dL], direct Coombs test negative. Iron saturation 55%, vitamin B12 level <50 [180 - 914 pg/mL], folate >25[>5.8 ng/mL], total bilirubin 2.3 [0.3 - 1.2 mg/dL], haptoglobin <30 [32 - 228 mg/dL], AST 43 [0 - 41 U/L], reticulocyte 1.4%. Blood smear showed absolute neutropenia with flow cytometry unremarkable. Chest x-ray and urinalysis were negative. Immunofixation showed low IgM 44 [45 - 281 mg/dL], low IgG 619 [635 - 1,741 mg/dL]. Intrinsic factor antibodies (IF-Ab) were positive. Hematology reported that hemolytic anemia is less likely given Coombs test was negative. About 1.5% of Vitamin B12 deficiency present with a hemolytic picture due to ineffective erythropoiesis while Coombs test help to differentiate it from autoimmune hemolytic anemia. Diagnosis of pernicious anemia was made and the patient started on vitamin B12 injections. The combination of pernicious anemia, autoimmune thyroiditis, and vitiligo supported the diagnosis of autoimmune APS-3B. There was a normalization of vitamin B12 level and symptomatic improvement on a one-week follow-up. Discussion: The patient was diagnosed with autoimmune thyroiditis in 2014 with positive anti-TPO antibodies and elevated TSH; she required levothyroxine supplementation since diagnosis. Hypothyroidism causes macrocytic anemia, which may delay pernicious anemia diagnosis. APS-3B is associated with HLA-B8 and/or DR3 and DR5. Many studies reported that autoantibodies can be detected before developing symptoms of organ involvement. Thorough family history provides support for autoantibody testing to detect cases of APS-3B earlier. Active surveillance and early diagnosis will help minimize invasive testing such as bone marrow biopsy, so proper history taking is a key factor to early diagnose these conditions. Conclusion: APS-3B is a rare disorder. Diagnosis is difficult hypothyroidism causes macrocytic anemia. Early detection of APS-3B may help to prevent complications that increase the risk of mortality and morbidity, particularly in the elderly population.
Introduction: Poor scope maneuverability and loop formation are some of the reasons for incomplete colonoscopies and suboptimal polyp resection even amongst expert endoscopists. The rigidizing overtube (RO) has been reported as a useful device used to overcome difficulties during colonoscopies and assisting in the completion of technically challenging procedures. In this study, we conducted a systematic review to assess the clinical success of using the RO to complete difficult colonoscopies and achieve complex polyp removal. Methods: We used multiple electronic databases from August 2019 through May 2023 to identify studies evaluating the use of the novel RO for colonoscopies. We followed the PRISMA guidelines. Quality/risk for bias was assessed using the Newcastle-Ottawa Scale (NOS). Results: Six studies were included; one study was divided into 2 different indications with a total of 165 patients (Figure 1). In the 3 studies that the indication was difficult to complete colonoscopies involving (53 patients), the colonoscopy completion rate was 88.7% (n = 47), the procedure facilitation was reported in 2/3 studies with a 93.1% (n = 27) and 100% (n = 9) respectively. Failure in these studies were reported in 2 patients due to anatomical restrictions and no adverse events were reported. One study compared polyp resection with and without the RO, reporting similar en-bloc resection and curative rates using RO (RO-ESD) when compared with conventional ESD (C-ESD), en-bloc resection success (RO-ESD 92% vs 96% C-ESD) and curative resection (RO-ESD 88% vs 92% C-ESD). In 2 studies that evaluated RO in the removal of complex polyps (62 patients), technical success was achieved 100% for both studies. One of the studies reported clinical success of 90% (n = 52), en-bloc resection of 86.4% and R0 resection of 80%, for both studies procedure facilitation ranged from 98.3-100%. None of these studies reported adverse events (Table 1). Conclusion: Our findings suggest that using RO shows promise in enhancing the clinical success of completing difficult colonoscopies and achieving successful ESD and EMR of colonic lesions by avoiding looping and offering stability. Further randomized studies evaluating the efficacy and cost of the RO are warranted.Figure 1.: PRISMA 2020 flow diagram for new systematic reviews which included searches of databases, registers and other sources. Table 1. - General Characteristics of the Studies N Study AIM Study Type Age, mean (SD) Sex (Male) Overtube Indication Indication of Colonoscopy Change of endoscope Success Procedure Facilitation Wei et al. (2021) 29 Difficult Colonoscopies Case Series 66 (13.6) 18 (62.1%) Incomplete Colonoscopy (n = 12, 41.4%)Enhancing Insertion (n = 6, 20.7%)Stabilization during endoscopic Resection (n = 11, 37.9%) Screening (n = 3, 10.3%)Surveillance (n = 7, 24.1%)Recent large polyp (n = 10, 34.5%)IDA/GI Bleeding (n = 3, 10.3%)Recent Incomplete colonoscopy (n = 1, 3.4%) Pediatric Endoscope (n = 4 , 13.8%) Colonoscopy Completion, (n = 23, 79.3%) 27 (93.1) Park N. et al. (2022) 15 Difficult Colonoscopies Prospective descriptive 63.2 (18) NA Incomplete Colonoscopy (n = 15, 100%) Surveillance (n = 9, 60%)Recent large polyp (n = 3, 20%)Evaluation of Chron's Disease (n = 2, 13.3%)Other (n = 1, 6.7%) NA Colonoscopy Completion, (n = 15, 100%) NA Friedland (2020) 9 Difficult Colonoscopies Case Series NA NA Incomplete Colonoscopy (n = 7, 77.8%)Intraprocedure failed Colonoscopy (n = 2, 22.2%) Incomplete colonoscopy (n = 9, 100%) Pediatric colonoscope (n = 5, 55.5%)Longer enteroscope (n = 2, 22.2%) Colonoscopy Completion (n = 9, 100%) 100% Farha et al. (2022) 50 Polyp Resection (ESD) Retrospective MatchedStudy RO vs No RO 66.7 (12.1) 18 (72%) Polyp Resection using ESD (100%) Polyps (100%) NA En-bolc resection: RO-ESD 92% vs 96% C-ESDCurative Resection: RO-ESD 88% vs 92% NA Jawaid et al. (2023) 58 Challenging Polyp Removal Observational 67.6 (9.9) 28 (50%) 2 polypectomies in the cecum1 ascending colon mucosal resection1 cecal submucosal dissection Polyps (100%) Intervention changed: 6 (13.6%) Technical Success: 100%Clinical Success: 90%En-bolc resection: 86.4%R0 resection: 80% 98.30% Friedland (2020) 4 Challenging Polyp Removal Case Series NA NA Challenging Polyp Removal (100%) Polyps (100%) NA Technical Success: 100% 100%
PURPOSE: Prone positioning (PP) in awake patients has been recently proposed as an adjunctive treatment for spontaneously breathing non-intubated coronavirus disease 2019 (COVID-19) patients requiring oxygen therapy to reduce the risk of intubation.However, the magnitude of the effect of PP on clinical outcomes (e.g., the risk of endotracheal intubation, intensive care unit [ICU] admission, or mortality) in these patients remains uncertain.Therefore, we performed a systematic review and metaanalysis to evaluate the effectiveness of PP to improve the clinical outcomes in non-intubated patients with COVID-19. METHODS:We performed a comprehensive literature search using PubMed, Embase, and Cochrane Library databases from inception through February 24, 2020 for all the studies all studies that all compared PP versus no PP in non-intubated patients with COVID-19.The primary outcome of interest was the rate of endotracheal intubation.The secondary outcomes were inhospital mortality and intensive care unit (ICU) rates.Pooled odds risk (OR) and 95% confidence intervals (CIs) were obtained by the Mantel-Haenszel method within a random-effect model. RESULTS:A total of five studies (two randomized controlled trials and three observational studies), involving 470 non-intubated patients with COVID-19 (185 patients received PP and 285 did not) were included.The mean age was 59.82 years, and males represented 67% of total patients.The follow-up period ranged from 14 to 30 days.The endotracheal intubation rate was similar between PP and control groups (OR 0.75, 95% CI 0.41-1.35,P = 0.33, I2 = 20%).There was no difference in the in-hospital mortality rate between the two groups (OR 0.68, 95% CI 0.16-2.85,P = 0.60, I2 = 60%).Four studies reported the risk of ICU admission and demonstrated no difference between the two groups (OR 0.77, 95% CI 0.30-1.95,P = 0.58, I2 = 37%). CONCLUSIONS:Our meta-analysis demonstrated that prone positioning in non-intubated COVID-19 patients did not reduce the risk of endotracheal intubation.Furthermore, PP failed to reduce in-hospital mortality and ICU admission rates.CLINICAL IMPLICATIONS: Although our meta-analysis showed that prone positioning might not reduce the risks of intubation, in-hospital mortality, or ICU admission rate in spontaneously breathing non-intubated COVID-19 patients, more large-scale trials with a standardized protocol for prone positioning are needed to better evaluate the effectiveness of prone positioning in this select population.
Introduction: Given the high recurrence rate and the risk of fecal incontinence with surgical options, Injection of adipose tissue-derived stem cells (ASC) has been arising as a novel method for treating complex perianal fistulas (CPAF). Therefore, we conducted a meta-analysis to evaluate the efficacy and safety of ASC in the management of CPAF not associated with Crohn’s disease. Methods: We systematically searched Medline and Embase databases through April 20, 2022, for all studies that assessed the efficacy and safety of ASC for the treatment of CPAF not associated with Crohn’s disease. We excluded patients with rectovaginal fistulas and perianal fistulas associated with Crohn’s disease. Our primary outcome was the complete closure. The secondary outcomes included overall nonserious adverse events (NSAE), serious adverse events (SAE), and perianal abscess rate. All meta-analyses were conducted using a random-effect model. The publication bias was assessed by Egger’s test. Results: Ten studies (eight clinical trials and two observational studies) with 271 patients were included in the pooled analysis. Eight studies used autologous stem cells, one used allogeneic stem cells, and one did not report the source of stem cells. The mean age of the patients was 43.7 years. The follow-up period ranged from 3 months to 2 years. The pooled complete closure rate was 59.7% (95% confidence interval (CI): 0.46-0.73, Figure 1A). On subgroup analysis based on country of origin, six studies with 213 patients were conducted in European countries, and four studies with 58 patients were conducted in non-European countries. The complete closure rate was higher in European countries than non-European countries, 64.1% vs. 52.6%. Eight studies reported overall NSAEs with the pooled NSAE rate of 22.5% (95% CI: 0.11-0.34, Figure 1B). Seven studies reported SAEs with the pooled SAE rate of 1.7% (95% CI: 0.001-0.034, Figure 1C). Seven studies reported the perianal abscess rate with a pooled perianal abscess rate of 7.1% (95% CI: 0.016-0.125, Figure 1D). No evidence of publication bias was found (Egger’s test: P=0.36). Conclusion: Our meta-analysis demonstrated that ASC is a promising therapeutic option for CPAF not associated with Crohn’s disease with a clinically adequate efficacy and low rate of adverse events. However, more studies with larger sample sizes are needed to provide a definitive assessment of the effectiveness of ASCs for CPAF not associated with Crohn’s disease.Figure 1
INTRODUCTION: Data on the risk of post-polypectomy bleeding after endoscopic mucosal resection (EMR) in patients with cirrhosis are limited. METHODS: This retrospective cohort study used the US Collaborative Network to assess post-polypectomy bleeding risk after colorectal EMR in patients with cirrhosis compared with controls. Using one-to-one propensity score matching, the primary outcome measured was bleeding within 30 days post-EMR. The bleeding risk was further stratified by cirrhosis severity (compensated and decompensated). RESULTS: After propensity score matching, each cohort included 2,381 patients. Patients with cirrhosis had a higher risk of post-polypectomy bleeding (odds ratio [OR] 1.38, 95% confidence interval [CI] 1.05–1.82, P = 0.02). However, endoscopic reinterventions for hemostasis were similar between cirrhosis and control cohorts (OR 1.20, 95% CI 0.71–2.05, P = 0.50). The post-polypectomy bleeding risk was significantly higher in patients with decompensated cirrhosis (OR 1.65, 95% CI 1.12–2.42, P = 0.01), while those with compensated cirrhosis showed no increased risk of post-polypectomy bleeding (OR 1.15, 95% CI 0.85–1.54, P = 0.37). DISCUSSION: The post-polypectomy bleeding risk was higher in patients with decompensated cirrhosis, while the bleeding risk was not increased in those with compensated disease. Optimizing patients with cirrhosis, especially with decompensated disease, before colorectal EMR and monitoring for post-procedure bleeding remain important. A conservative, supportive approach to managing bleeding in patients with cirrhosis may be as effective as in those without cirrhosis.
Introduction: Insulin therapy (IT) and plasmapheresis are used to treat hypertriglyceridemia-associated pancreatitis (HTAP). However, the optimal treatment modality for lowering the triglyceride level in patients with HTAP remains unclear. Therefore, we evaluated the efficacy and safety of IT and plasmapheresis in managing HTAP. Methods: We performed a comprehensive literature search using PubMed, Embase, and Web of Science databases through May 30, 2022, for all studies that compared IT vs. plasmapheresis in patients with HTAP. The primary outcomes were effectiveness (reduction in triglycerides within 24-hours of admission) and clinical outcomes, including hospital length-of-stay (LOS), mortality, acute renal failure (ARF), hypotension, and need for invasive mechanical ventilation (IMV). The secondary outcome was the overall treatment-related adverse events (AEs). Random-effects meta-analysis was conducted, and risk ratio (RR) and mean difference (MD) or standardized mean difference (SMD) for proportional and continuous variables were computed, respectively. For each outcome, forest plot, 95% confidence interval (CI), P-value (< 0.05 considered statistically significant), and I2 statistic ( > 50% considered as significant heterogeneity) were generated. Results: Six studies (1 randomized controlled trial [RCT] and 5 cohort studies) with 302 patients with HTAP (167 on IT vs. 135 on plasmapheresis) were included. Plasmapheresis was more effective than IT in reduction of triglycerides within 24-hours (SMD -0.57; 95% CI -1.02, -0.13; P=0.01, I2=56.8%, Figure 1A). However, LOS (MD -1.96; 95% CI -4.45, 0.54; P=0.12; I2=0%, Figure 1B), mortality (RR 0.68, 95% CI 0.28-1.64, P=0.39, I2=0%, Figure 1C), ARF (RR 0.44, 95% CI 0.06-3.05, P=0.41, I2=84%), hypotension (RR 0.63, 95% CI 0.16-2.52, P=0.51, I2=79%), and need for IMV (RR 0.52, 95% CI 0.12-12.35, P=0.40, I2=80%) were similar between two groups. The treatment-related AEs were significantly lower in IT than plasmapheresis (RR 0.14, 95% CI 0.04-0.51, P=0.003, I2=0%, Figure 1D). Conclusion: Our meta-analysis demonstrated that despite the greater reduction of triglycerides with plasmapheresis compared to insulin therapy, the clinical outcomes, including LOS, mortality, ARF, hypotension, and need for IMV, were comparable with lower treatment-related adverse events with insulin therapy. Future large-scale RCTs are necessary to validate our findings.Figure 1.: Forest plots comparing between insulin therapy and plasmapheresis regarding: (A) the reduction of triglyceride within 24 hours, (B) length of hospital stay, (C) mortality, and (D) treatment-related adverse events.
Introduction: Peptic ulcer disease (PUD) is the leading cause of upper GI bleeding. PUDs with an adherent clot (Forrest IIb) are associated with a high risk of rebleeding and mortality. However, the optimal management of bleeding PUD with adherent clots, including endoscopic or conservative therapy, remains unclear. The most recent ACG guidelines in 2021 did not clearly endorse endoscopic treatment for bleeding PUDs with adherent clot as opposed to medical therapy. We performed this systematic review and meta-analysis to compare the endoscopic and conservative therapeutic approaches to managing bleeding PUDs with adherent clot. Methods: We systematically searched MEDLINE, EMBASE, and the Web of Science databases through May 15, 2022, to include all randomized controlled trials (RCTs) comparing the endoscopic and conservative therapeutic approaches for bleeding PUDs with adherent clots. The outcomes of interest in our meta-analysis were rebleeding, need for surgery, and mortality. The random-effects model was used to calculate the weighted pooled risk ratio (RR) with the corresponding 95% confidence intervals (CI) of our desired outcome. A P-value < 0.05 was considered statistically significant. Heterogeneity was assessed using the Higgins I2 index (I2 values >50% implied the presence of significant heterogeneity). Results: Eleven RCTs with 839 patients (434 received endoscopic therapy vs. 405 received conservative therapy) were included. Endoscopic therapy group underwent clot removal and treatment of the underlying lesion with thermocoagulation, electrocoagulation, injection of sclerosants such as epinephrine or ethanol, or hemoclipping. Rebleeding occurred in 8.1% of patients in the endoscopic therapy group, compared to 20.7% in the conservative therapy group (RR 0.45, 95% CI 0.26-0.78, P=0.004, I2=34%, Figure A). We observed lower mortality of 3.7% in the endoscopic therapy group compared to 8% in the conservative therapy group (RR 0.49, 95% CI 0.25-0.96, P=0.04, I2=0%, Figure B). In addition, the need for surgery was significantly lower in the endoscopic therapy group (4.3%) compared to the conservative therapy group (9.9%) (RR 0.48, 95% CI 0.24-0.97, P=0.04, I2=0%, Figure C). Conclusion: Our meta-analysis demonstrated that endoscopic therapy is superior to conservative treatment for bleeding PUDs with an adherent clot regarding rebleeding, need for surgery, and mortality. However, large-scale RCTs are needed to validate our findings.Figure 1
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