Recurrent Symptoms Research Articles

Efficacy of oral viscous budesonide to reduce dilation treatment after esophageal atresia repair: a retrospective study

IntroductionAnastomotic stricture is a common complication following esophageal atresia (EA) repair, substantially affecting the patient’s quality of life (QoL). Multiple dilations are often required to maintain the appropriate diameter of the esophagus, leading to ongoing challenges. The aim of this study is to assess the efficacy of oral viscous budesonide (OVB) in prolonging the time between symptom recurrence and subsequent dilation.MethodsWe carried out a retrospective single-center study for pediatric patients (0–18 years) who had undergone recurrent esophageal dilations (≥3) following EA repair and initiated treatment with OVB (1 mg/day <10 years, otherwise 2 mg/day). Efficacy of treatment was determined by assessing a dysphagia symptom score (DSS) ≤1 for at least 3 months. Recurrence time to dysphagia and dilation were analyzed according to Kaplan-Meier method.ResultsOf 29 patients screened, 19 were enrolled: 19/19 were responsive to OVB and 13/19 (68%) didn’t required further dilations. The median time between dilations was significantly prolonged compared to the pre-treatment period [30 months vs 2 months; p<0.01] as well as the time to dysphagia relapse [18 months vs 1 month; p<0.01].ConclusionTopical budesonide has proven to be an effective treatment for recurrent esophageal stricture in repaired EA. Further investigation is required to assess the long-term sustained response of symptoms to topical steroids.

What Happens to Youth Baseball Players Diagnosed With Little League Shoulder and Little League Elbow Syndrome?

The long-term prognosis of baseball and softball players diagnosed with Little League elbow (LLE) and Little League shoulder (LLS) is unknown. Many of these athletes are potentially at risk of developing future shoulder and elbow injuries that may require surgical intervention. This study's purpose is to retrospectively assess 5-year patient-reported outcomes and career progression of a series of youth baseball and softball players diagnosed with LLE and LLS. This institutional review board-approved, single-center, retrospective study evaluated 5-year outcomes of a cohort of youth baseball and softball players diagnosed with LLE and LLS between 2013 and 2017. Demographic and clinical data was recorded including age, gender, primary position, and months played per year. A standardized phone survey was obtained approximately 5 years post-treatment to assess upper extremity function, career progression, and pain recurrence. Sixty-one patients (44 LLE, 17 LLS) met the inclusion criteria and participated in a standardized phone survey. The mean age at the time of diagnosis was 13.2 years and all but one of the athletes were male. On average, about 80% (34/44 LLE, 15/17 LLS) played baseball 9 months or more per year. After diagnosis and nonoperative management, more than 80% (37/44 LLE, 14/17 LLS) were able to return to competition, but up to 40% of players changed positions (9/44 LLE, 7/17 LLS). Five years later, less than half (21/44 LLE, 8/17 LLS) were playing baseball actively. The recurrence rate was >20% (9/42 LLE, 6/17 LLS) with a mean time of recurrence of 8.8 months. Patients who experienced symptom recurrence were less likely to play baseball 5 years later and had lower patient-reported outcomes. A diagnosis of LLE and LLS in early adolescence can be a setback for a young athlete. Fortunately, most athletes will be able to return to competition with ∼50% still competing at 5 years. Very few will progress to future arm surgery, but recurrence rates are relatively high, and these athletes are less likely to participate in their sport 5 years later. Level IV-Retrospective cohort study.

Endoscopic pilonidal sinus treatment (EPSiT) for sacrococcygeal pilonidal sinus disease: a prospective long-term study

Objective: To assess short-term and long-term outcomes of endoscopic pilonidal sinus treatment for pilonidal sinus disease. Method: The prospective study was conducted at Shifa International Hospital, Islamabad, Pakistan, from July 2015 to July 2021, and comprised all pilonidal sinus cases undergoing minimal invasive endoscopic pilonidal sinus treatment who were treated by a single surgical team. The primary outcomes were duration of healing, post-operative morbidities, persistence of discharge and recurrence at 1-7 years. The secondary outcomes were operative time, return to work, cosmetic results and patient satisfaction. The patients were observed for wound healing and discharge on follow-up in the out-patient department at 1, 3, 6 and 24 weeks. They were further followed up every year through telephonic survey for persistence or recurrence of symptoms. Patient satisfaction was assessed using the 36-item Short Form Survey questionnaire filled at admission and then at 6 weeks post-surgery. Data was analysed using SPSS 23. Results: Of the 67 patients, 55(82%) were males and 12(18%) were females. The overall mean age was 25.69±8.305 years. There were 13(19.4%) patients with a history of recurrent disease and previous procedures for pilonidal sinus, while 54(80.6%) had no previous surgery. The median operative time was 35 minutes (interquartile range: 20-45 minutes). Complete wound healing was achieved in 60(89.6%) patients, while recurrence was seen in 7(10.4%). The median time off work was 2.5 days (interquartile range: 1-3 days). Patient satisfaction with the procedure was significantly high (p<0.05). Conclusion: Endoscopic pilonidal sinus treatment appeared to be a good minimally invasive surgical technique for the treatment of pilonidal sinus disease in terms of both short-term and long-term outcomes. Key Words: Pilonidal sinus disease, Minimally invasive surgery, Endoscopic pilonidal sinus treatment, EPSiT, Recurrent pilonidal sinus.

Blinatumomab-induced macrophage activating syndrome (MAS) in adult with B-cell acute lymphoblastic leukemia (B-ALL).

Blinatumomab as a single agent has demonstrated superiority over salvage chemotherapy in patients with relapsed and refractory B-cell acute lymphoblastic leukemia (B-ALL), with manageable safety and efficacy. Though known to have anticipated drug toxicities including cytokine release syndrome (CRS) and neurotoxicity, there is only one prior report of macrophage activating syndrome (MAS) due to blinatumomab. Case Presentation: We report the first case of blinatumomab-induced MAS in an adult. The patient presented with fever, cough, and weakness on the second cycle of blinatumomab. Complete blood count was notable for severe leukopenia, with comprehensive metabolic panel notable for elevated alkaline phosphatase, AST, ALT, LDH, and hyperferritinemia consistent with MAS. The patient was already in MRD-negative remission at presentation with MAS. She responded rapidly to withholding the drug and administration of both tocilizumab and dexamethasone. She was able to restart therapy with blinatumomab dosed at 9 mcg/day with no recurrence of symptoms. Though MAS is not an expected association with blinatumomab, the risk for CRS is. Secondary MAS in this case likely shares a mechanism with other hyperinflammatory conditions. Management includes holding the offending agent, like blinatumomab, and administering tocilizumab and dexamethasone. Future research will be needed to predict which patients are at highest risk to develop MAS after similar T-cell therapies.

Treatmen of atrial fibrillation with cryoballoon pulmonary vein isolation - results of a 5-year follow-up

Abstract Background Atrial fibrillation is the most common arrhythmia in the general population and cryoballoon ablation is gradually becoming a first-line treatment. However, data regarding long-term outcomes after this procedure are still lacking. Aim The goal was to show the results of a long-term follow-up following the cryoballoon pulmonary vein isolation in patients with atrial fibrillation (AF). Methods A total of 126 patients were included in the study of which 77.0% had paroxysmal atrial fibrillation (PAF). Successful treatment outcome was defined as AF recurrence-free survival with stabile sinus rhythm during 5 years after the procedure. A treatment failure was defined as symptomatic AF recurrence with ECG verification (12 lead ECG or AF lasting >30s by Holter EKG). The patients on antiarrhythmic therapy and those who underwent a redo pulmonary vein isolation procedure were also included. Results Following cryoballoon pulmonary vein isolation in a 5-year period 52.4% of patients were in stable sinus rhythm without AF recurrence. With redo pulmonary vein procedures a total of 61.9% of patients were without AF recurrence. Together with antiarrhythmic drugs 57.9% of patients were in sinus rhythm without AF recurrence in a 5-year period. With redo pulmonary vein isolation and antiarrhythmic drugs a total of 73.8% of patients were without AF recurrence. There was a statistically significant difference regarding AF recurrence between patients who underwent redo pulmonary vein isolation and those who did not (p=0.006). In patients with PAF, 62.9% remained without AF recurrence, and 79.4% underwent a redo procedure. In patients with persistent atrial fibrillation (PersAF), 41.4% were without AF recurrence, and 55.1% underwent a redo procedure. The difference between PAF and PersAF was statistically significant (p=0.009). Conclusion Data from our center show good long-term results of cryoballoon pulmonary vein isolation in patients with atrial fibrillation. The procedure is especially successful in patients with PAF.1 The use of redo procedures is justified as it increases the long-term success rate.

Optimized workflow for pulmonary vein isolation using very high power short duration radiofrequency ablation

Abstract Background Point-by-point radiofrequency ablation is the most used invasive treatment of atrial fibrillation (AF). However, single-shot technologies are largely widespread due to their proved safety and efficacy. QDOT MICRO (QDM) is a novel contact force–sensing catheter optimized for temperature-controlled radiofrequency (RF) ablation that allows to maintain target temperature during 90 W ablations for ≤4 seconds. Purpose This study evaluates procedural safety and effectiveness as well as long-term effects of an optimized procedural workflow using QDM catheter to perform wide atrial circumferential ablation in patients with paroxysmal AF. Methods Consecutive patients referred for paroxysmal AF first ablation through pulmonary vein isolation (PVI) were prospectively enrolled. Every procedure was performed in general anesthesia by using CARTO3 Electroanatomical Mapping System BiosenseWebster QDM catheter and Deflectable Sheet VIZIGO as single sheet in left atrium. VHPSD was performed to encircling the posterior wall, while conventional-power temperature-controlled (CPTC) mode was used to encircling the anterior wall. The primary safety endpoint was incidence of primary adverse events within ≤7 days of ablation. The primary effectiveness endpoint was freedom from documented atrial tachyarrhythmia recurrence and acute procedural, repeat ablation, and antiarrhythmic drug failure. Results 120 enrolled participants (61 ± 12 years, 45% female) met eligibility criteria. Median procedural skin-to-skin time and RF application for ablating PVs were 56 and 17 minutes, respectively. Median ablation time was 22 minutes. First-pass isolation (entrance and exit block validation) was obtained in 98 % and inter-lesion distance was 4,8 ± 0,8 mm. In 100% of patients PVI was obtained without adverse events. The Kaplan-Meier estimated 12-month rate for clinical success (freedom from symptomatic recurrence of AF) was 93%, freedom from AF was 89% Conclusions PVI performed with point by point optimized workflow using VHPSD RF catheter in combination with steerable sheet is highly efficient and effective without compromising safety.

An economic evaluation of first-line cryoballoon ablation versus antiarrhythmic drug therapy for the treatment of paroxysmal atrial fibrillation from a Danish healthcare perspective

Abstract Background As demonstrated in three randomized clinical trials (RCTs), initial rhythm control with first-line pulmonary vein isolation (PVI) using cryoballoon ablation decreases atrial arrhythmia recurrence compared to antiarrhythmic drug (AAD) therapy in patients with symptomatic paroxysmal atrial fibrillation (PAF). Purpose To assess the cost-effectiveness of first-line cryoablation versus first-line AADs in a Danish healthcare setting. Methods Data from 703 participants with symptomatic PAF enrolled into Cryo-FIRST, STOP AF First and EARLY-AF RCTs were used to estimate risk equations (rate of ablation, AF recurrence and resolution, AF-related hospital attendance in addition to health-related quality-of-life (HRQoL) utilities). These were then incorporated into a cost-effectiveness model (CEM) . The Danish cost data was derived from published Danish literature, DRG tariffs and pharmaceutical list prices in Denmark. Where parameters could not be derived, inputs were sourced from published literature or expert opinion. The CEM consisted of a decision tree (one-year time horizon) and a Markov model (three-month cycle length) hybrid, with a lifetime (40 years) time horizon. The CEM is from the perspective of the Danish healthcare system. Health benefits were expressed in quality-adjusted life-years (QALYs), and all costs and benefits were discounted at 3%. As there is no official cost-effectiveness threshold in Denmark, the UK threshold of £20,000 (~€23,200) was assumed. Input uncertainty within the CEM was explored using probabilistic sensitivity analysis. The results presented include data from the initial 12-week blanking period for all studies. Results The three-monthly rate of AF symptom recurrence had a reduction, on average, of 46.7% (p<0.001) in those treated with cryoablation. In addition, the cryoablation arm had a 72.8% (p<0.001) reduction in the monthly rate of receiving an ablation following initial treatment. The average cryoablation patient was also associated with a 4.3% (p=0.025) increase in their HRQoL. Finally, there was no difference in the rate of AF symptom resolution in those who failed initial treatment. Base case results per patient are presented in Table 1. Cryoablation was shown to be dominant when compared to AADs (0.159 more QALYs and €2,519 less costly), with a 99.96% probability of being cost-effective at a willingness-to-pay threshold of ~€23,200 per QALY gained. Individuals in both arms of the model were also expected to receive ~1.2 ablations over a lifetime horizon. Nonetheless, there was a 45% relative reduction in time spent in symptomatic AF states for individuals who were initially treated with cryoablation. Scenario analysis results are presented in Table 2. Cryoablation remained cost-effective over AADs in all scenarios conducted. Conclusions Initial rhythm control with cryoballoon ablation is cost-effective (dominant) when compared to AADs in the Danish healthcare system.Table 1:Key results (per patient)Table 2.Deterministic scenario analysis

Recurrence of sleep apnea in children after intracapsular coblation tonsillectomy: A comprehensive exploration of tonsil regrowth

ObjectivesSleep apnea is a prevalent issue in children, associated with significant morbidities such as cardiovascular and neurocognitive disorders. There is increasing interest in intra-capsular tonsillectomy by coblation (ICTC) as a method to address obstructive sleep apnea (OSA) in children. However, the literature remains controversial regarding the most effective intra-capsular tonsillectomy (ICT) technique with the least morbidity. Our current research extends a previous study that established the effectiveness and safety of ICTC, demonstrating rapid post-surgical recovery with minimal analgesic needs. This new investigation specifically focuses on long-term follow-up. Our aim is to assess tonsil regrowth and the risk of recurrence of OSA symptoms at a mean follow-up of 6.1 years post-surgery. By presenting the results of this extended study, our goal is to gain a better understanding of the long-term effectiveness of this surgical intervention in treating OSA in children. Thus, considering the initial benefits, we will also explore potential long-term implications. Materials and methodsThis research follows up on children from our previous study who underwent ICTC, with or without adenoidectomy, for OSA resulting from tonsillar hypertrophy at a tertiary-level university hospital between March 2016 and March 2018. They were followed up for an average of 6.1 years postoperatively. Symptom recurrence is assessed by comparing preoperative OSA-18 questionnaire results with those obtained at the 6.1-year mark. Tonsil regrowth is evaluated by comparing preoperative Brodsky scores with those obtained at 6.1 years. ResultsThe mean total score of OSA-18 significantly decreased from 79.41 (SD = 14.95) before ICTC to 25.47 (SD = 8.92) at 6.1 years postoperatively (p < 0.001, mean difference = 53.94, 95 % CI [50.32, 57.56]). Similarly, the mean Brodsky score dropped from 2.95 (SD = 0.51) before ICTC to 1.04 (SD = 0.24) 6.1 years postoperatively (p < 0.001, mean difference = 1.92, 95 % CI [1.80, 2.04]). The overall regrowth rate was 2.35 % (n = 2), with a revision surgery rate of 1.18 % (n = 1). ConclusionICTC exhibits minimal risk of tonsil regrowth and maintains long-term efficacy in preventing the recurrence of OSA symptoms. Therefore, it justifies broader utilization in addressing OSA symptoms arising from tonsillar hypertrophy in children.

Carpal tunnel syndrome.

Carpal tunnel syndrome (CTS) is the most common nerve entrapment disorder worldwide. The epidemiology and risk factors, including family burden, for developing CTS are multi-factorial. Despite much research, its intricate pathophysiological mechanism(s) are not fully understood. An underlying subclinical neuropathy may indicate an increased susceptibility to developing CTS. Although surgery is often performed for CTS, clear international guidelines to indicate when to perform non-surgical or surgical treatment, based on stage and severity of CTS, remain to be elucidated. Neurophysiological examination, using electrophysiology or ultrasonography, performed in certain circumstances, should correlate with the history and findings in clinical examination of the person with CTS. History and clinical examination are particularly relevant globally owing to lack of other equipment. Various instruments are used to assess CTS and treatment outcomes as well as the effect of the disorder on quality of life. The surgical treatment options of CTS-open or endoscopic-offer an effective solution to mitigate functional impairments and pain. However, there are risks of post-operative persistent or recurrent symptoms, requiring meticulous diagnostic re-evaluation before any additional surgery. Health-care professionals should have increased awareness about CTS and all its implications. Future considerations of CTS include use of linked national registries to understand risk factors, explore possible screening methods, and evaluate diagnosis and treatment with a broader perspective beyond surgery, including psychological well-being.

Endovascular Rescue of Previously Treated Carotid Aneurysm: Higher Radial Force 48-Wire Flow Diverter for Salvage of Collapsed 64-Wire Device With Braid Deformation: 2-Dimensional Operative Video.

Aneurysm treatment presents various therapeutic options. Flow diverters (FD) stents are a revolutionizing endovascular technique for previously untreatable aneurysms.1 FD stents have documented success rates up to 95% and complication rates as low as 2.3%.2 This modality has proven its effectiveness and safety, but it is critical to consider potential complications. Complications include vascular-related problems such as perforation, occlusion, cerebral hyperperfusion, stent thrombosis, restenosis, and embolic events.3 To date, there is no standardized definition for braid collapse and fish-mouthing; however, braid collapse has been reported to occur at a rate of 1% and the latter at a rate of 3%.4 This technical video presents a case of a 51-year-old woman initially treated with a Surpass Evolve flow diverter (Stryker) for a right ophthalmic aneurysm. Secondary to device malapposition and collapse, she developed recurrent ischemic progressive neurologic impairment symptoms. The braid collapse was treated using a Pipeline Embolization device 3.25 × 20 mm (Medtronic) because this device exhibited a larger partial compressive force when compared with the 64-wire device.5 The procedure was uneventful and significantly improved the patient's neurologic symptoms. This technical video illustrates rescue stenting as a salvage intervention for FD collapse cases. The patient gave informed consent for surgery. Institutional review board approval was deemed unnecessary.

#823 Effect of dapsone on the treatment of refractory IgA vasculitis

Abstract Background and Aims IgA vasculitis (IgAV), previously known as Henoch-Schönlein purpura, is a systemic IgA-mediated vasculitis of the small vessels commonly seen in children. The natural history of IgAV is generally self-limiting. However, one-third of patients experience symptom recurrence and a refractory course. We performed this study to examine the use of dapsone (4,4'-sulfonyldianiline or diaminodiphenyl sulfone) in refractory IgAV cases. Method A literature search of PubMed databases retrieved 16 articles published until May 31, 2023. A total of 38 patients with IgAV were recruited. Results A palpable purpura persisted in nearly all the patients with refractory course, followed by joint pain (25 of 38 patients; 69.2%). Hematuria and proteinuria were noted in 11 (28.9%) and 7 (18.4%) of the patients, respectively. Treatment response within 1-2 days was obtained in 12 of 38 patients (31.6%), compared to within ≥3 days in 22 patients (57.9%). Relapse after treatment discontinuation was reported in 21 patients (55.3%) but not in 11 patients (28.9 %). Twelve of the 38 patients (31.6%) reported adverse effects of dapsone, including arthralgia (5.3%), rash (5.3%), dapsone hypersensitivity syndrome (2.6%), methemoglobinemia (21%), and hemolysis (15.8%). These findings indicate that dapsone may have a beneficial effect on refractory IgAV. Conclusion It is observed that in spite of several adverse effects, dapsone treatment exerts a beneficial effect in IgAV patients with prolonged cutaneous manifestation refractory to corticosteroid. Close renal monitoring may be needed to discover renal involvement of IgAV. Multicenter randomized placebo-controlled trials are necessary to determine the standard dosage of dapsone at initial or tapering of treatment in IgAV patients.

Use of Artificial Intelligence in the Prediction of Chiari Malformation Type 1 Recurrence After Posterior Fossa Decompressive Surgery.

Purpose The purpose of this study was to train a deep learning-based method for the prediction of postoperative recurrence of symptoms in Chiari malformation type 1 (CM1) patients undergoing surgery. Studies suggest that certain radiological and clinical features do exist in patients with treatment failure, though these are inconsistent and poorly defined. Methodology This study was a retrospective cohort study of patients who underwent primary surgical intervention for CM1 from January 2010 to May 2020. Only patients who completed pre- and postoperative 12-item short form(SF-12) surveys were included and these were used to classify the recurrence or persistence of symptoms. Forty patients had an improvement in overall symptoms while 17 had recurrence or persistence. After magnetic resonance imaging (MRI) data augmentation, a ResNet50, pre-trained on the ImageNet dataset, was used for feature extraction, and then clustering-constrained attention multiple instance learning (CLAM), a weakly supervised multi-instance learning framework, was trained for prediction of recurrence. Five-fold cross-validation was used for the development of MRI only, clinical features only, and a combined machine learning model. Results This study included 57 patients who underwent CM1 decompression. The recurrence rate was 30%. The combined model incorporating MRI, pre-operative SF-12 physical component scale (PCS), and extent of cerebellar ectopia performed best with an area under the curve (AUC) of 0.71 and an F1 score of 0.74. Conclusion This is the first study to our knowledge to explore the prediction of postoperative recurrence of symptoms in CM1 patients using machine learning methods and represents the first steptowarddeveloping a clinically useful prognostication machine learning model. Further studies utilizing a similar deep learning approach with a larger sample size are needed to improve the performance.

Invasive Streptococcal Infection in Children: An Italian Case Series

Since October 2022, alerts have spread from several countries about the increase in invasive group A streptococcal (iGAS) and scarlet fever cases affecting young children. We aim to analyze the epidemiology of GAS infections in the last 12 years in our hospital and identify the clinical features of invasive cases observed in 2023. We conducted a retrospective study enrolling children and adolescents hospitalized at our pediatric clinic from January to December 2023 for a definitive diagnosis of iGAS infection. Clinical, laboratory, and imaging data were collected and analyzed. Comparing 2016 and 2023, we observed a similar number of GAS infections (65 vs. 60 cases). Five children with iGAS infection were hospitalized between March and April 2023. The median age was five years. At admission, all patients showed tachycardia disproportionate to their body temperature. Vomiting was a recurrent symptom (80%). Laboratory tests mostly showed lymphopenia, hyponatremia, and high inflammatory markers. The number of pediatric iGAS cases significantly increased in 2023. Clinical (pre-school-aged children with high fever, unexplained tachycardia, and vomiting) and laboratory parameters (high procalcitonin levels, hyponatremia, and lymphopenia) could help identify and suspect a potential iGAS infection.

Chronic rhinosinusitis with nasal polyps-extension of dupilumab treatment intervals

In patients with severe, uncontrolled chronic rhinosinusitis with nasal polyps (CRSwNP), dupilumab 300 mg every 2weeks can completely resolve nasal polys, sinus disease, and symptoms. In this case, patients ask for de-escalation. Although trials have demonstrated recurrence after stopping the biologic at 24weeks, reducing the dose of dupilumab to once every 4weeks did not result in deterioration of control. An extension of the treatment intervals would, however, diverge from the approval text, and is currently not recommended. The course of 29patients with severe CRSwNP, type‑2 inflammation-associated comorbidities, and an indication for biologic was retrospectively analyzed. After resolution of CRSwNP and symptoms under biweekly dupilumab 300 mg, the dupilumab interval had been prolonged individually, initially up to 4weeks, thereafter up to 6weeks, if applicable. Control was assessed via quality of life (22-item sinonasal outcome test, SNOT-22), nasal polyp score, and smell identification test (Sniffin' Sticks; Burghart Messtechnik, Holm, Germany). All patients showed an excellent improvement within the first 3months. The dupilumab application interval was extended to 4weeks after 7-31months (median 13months) and to 6weeks (n = 9) after 17-35months (median 23months). No recurrent polyps or symptoms were subsequently observed. In case of maximal regression of polyps and discomfort, extension of dupilumab injection intervals to4 and potentially 6weeks is possible without clinical worsening. Further studies on de-escalation or termination of biologic treatment when CRSwNP control is achieved are essential.

A Unique Case of SCN2A Variant-Associated Catatonia and Response to Electroconvulsive Therapy.

The SCN2A gene encodes a subunit that forms part of voltage-gated sodium channels in the brain. Gain-of-function mutations are associated with epilepsy as well as numerous movement/motor abnormalities. Loss-of-function mutations may also cause epilepsy in addition to a variety of neurodevelopmental anomalies, including autism and intellectual disability. The occurrence of catatonia has also been described in 1 previous report that involved a 4-year-old boy. We describe a 20-year-old intellectually disabled female patient who developed recurrent catatonic symptoms in her teenage years that remitted with electroconvulsive therapy. This is only the second report of catatonia occurring in relation to an SCN2A mutation and the first involving a female. Moreover, this case is unique given our patient's later age of symptom onset and given that her symptoms responded well to electroconvulsive therapy.

LYME AND DENGUE FEVER: FROM BANGLADESH TO ITALY

Abstract Background Dengue fever (Flavivirus) and Lyme disease (Borrelia burgdorferi) are rare tropical infectious diseases transmitted by hematophagous vectors (Aedes mosquitoes and ticks, respectively), and they are extremely uncommon in Italy. In European countries, both represent a public health concern as they manifest as imported diseases. Case Report A 54–year–old man from Bangladesh was admitted with symptoms of fever, dyspnea, and pericarditic chest pain. Laboratory and imaging examinations revealed severe acute respiratory failure secondary to left lower lobe pneumonia and pleuropericardial effusion. Broad–spectrum antibiotic therapy and anti–inflammatory treatment were initiated, suspecting para–pneumonic pleuropericarditis. During hospitalization, there was a gradual improvement with a reduction in inflammatory indices and resolution of lung consolidation. The patient was discharged after 14 days with anti–inflammatory therapy (ibuprofen and colchicine). Approximately 7 days later, the patient returned to the emergency department complaining of a recurrence of symptoms and fatigue. The EKG showed phases of 2:1 AV block, EAS and EPS, complete de novo left bundle branch block (LBBB), alternating with an escape atrial rhythm, not evident in previous EKG. During hospitalization, complete AV block emerged with prolonged asystolic phases, requiring urgent placement of a temporary pacemaker. Concurrently, blood tests revealed anemia and increased levels of inflammatory markers, troponin, creatinine, transaminases, cholestatic indices, LDH, and lipase, indicative of multiple organ failure (MOF). Acute pathologies or active bleeding were ruled out by total body CT with contrast. Blood cultures and serological tests were performed to exclude infections by Trichinella, Mycoplasma, Borrelia, Dengue, and other rare infectious diseases. Due to progressive deterioration, the patient was transferred to a tertiary care hospital. In the following days, laboratory results showed positive IgG and IgM for Dengue (without serological evidence of viral RNA) and positive IgG and IgM antibodies for Borrelia burgdorferi. Conclusion In Italy, the incidence of these diseases is low, and their coexistence in the same host is even rarer (if not anecdotal). However, coinfection cannot be excluded in patients from areas where these diseases are endemic, such as Bangladesh.

Isolated Fallopian Tube Torsion in Children With Hydrosalpinx: Is Conservative Management an Option?

Isolated fallopian tube torsion (IFTT) is very rare gynecological emergency in pediatric population. Our objective is to assess treatment options and discuss outcome of a cohort of IFTT with a focus on the association between IFTT and hydrosalpinx (HSX). A retrospective review was conducted. Pediatric patients with IFTT operated in the same center were included. Seventeen girls (aged: 11-16 years) were managed for acute abdominal pain between 2008 and 2018, with intraoperative diagnosis of IFTT. All patients underwent laparoscopic exploration, with laparoscopically fallopian tube detorsion in all patients. Based on the association of IFTT with HSX after fallopian tube detorsion, patients were divided into 2 groups: group 1 (IFTT without HSX; 12 girls) and group 2 (IFTT with HSX; 5 girls). During the same surgery, complementary surgical procedures were done. In group 1: salpingectomies (4), partial salpingectomies (2) and cystectomies (6) were done. In group 2: salpingectomy (1), salpingotomy (1), and cyst ablation (1). The treatment was called conservative when the tube was preserved.Follow-up was uneventful in group 1. In group 2, for all patients with initial fallopian tube preservation, further surgical procedures were necessary (1-4 surgeries/patient), and, finally, another 3 patients required salpingectomy. Conservative treatment with tube preservation of IFTT without HSX appeared to be beneficial compared to those with HSX, with no recurrence of torsion or symptoms during the follow-up. However, the same conservative treatment was not sufficiently effective for IFTT with HSX and required further procedures due to recurrence of torsion. IV.

Clinical profile and management of a Spanish single-center retrospective cohort of patients with post-chikungunya associated complications

BackgroundThis study aims to describe post-chikungunya complications chronically developed cases in returning travelers from some epidemic/endemic regions, and the variables that are associated with the progression of acute or subacute cases to the chronic phase. MethodsThis single-center retrospective cohort study included chikungunya fever cases treated at La Paz-Carlos III University Hospital in Madrid, Spain, April 2014 to September 2016, when the chikungunya outbreak in Latin America started through the time of its greatest impact. ResultsThe analysis included 119 cases. Of these, 67.2 % were male, with a median age of 41.0 years [IQR 16.0 to 76.0] years. Only 25.6 % of the patients attended a pre-travel advice consultation. Most patients reported arthralgias, which significantly impacted their daily quality of life (86 %). The mean duration of joint symptoms was 129.4 days, with a median of 90 days [IQR 0 to 715]. Factors found to be associated with chronic arthralgia include female sex, country of infection, age at diagnosis, previous diseases, symptoms during the acute phase, pain in previously injured tendons/joints, acute phase severity, and various laboratory markers such as hemoglobin, hematocrit, total serum bilirubin, and creatinine. Progression to chronic arthralgia significantly increased the need for changes in daily activity. Furthermore, 42.6 % of patients with chronic arthralgia reported recurrence of symptoms once they felt they had disappeared. Targeted treatment regimens led to significant improvements in these patients. ConclusionsThe results of this study underscore the need for: (1) comprehensive pre-travel advice; (2) effective management of patients in specialized units, alongside early diagnosis and treatment, to prevent trivialization of these viral infections; and (3) the development of interdisciplinary recommendations to assist physicians in treating patients and enhancing outcomes.

Hedyotis diffusa Willd inhibits inflammation and oxidative stress to protect against chronic prostatitis via the NRF2/ARE signaling pathway.

Chronic Prostatitis/Chronic Pelvic Pain Syndrome (CP/CPPS) is a common and serious disease with unclear pathogenesis and recurrent symptoms. Hedyotis diffusa Willd (HDW) has been recognized for its potential in managing various chronic inflammatory diseases. This research aimed to interrogate the mechanism of HDW in treating CP/CPPS. Complete Freund Adjuvant (CFA) and LPS were utilized to establish the rat and cell models of CP/CPPS. Results showed that HDW decreased levels of inflammation-related factors in CP rat prostate tissue and LPS-elicited RWPE-1 cell injury model. Moreover, HDW administration impaired oxidative stress in the prostate and RWPE-1 cells. In addition, HDW treatment activated the NRF2/ARE signaling in rat prostate tissue and cell models. Interestingly, NRF2/ARE pathway inhibitor ML385 reversed the inhibition effects of cell apoptosis, inflammation, and oxidative stress triggered by HDW. In summary, HDW alleviated inflammation and oxidative stress by activating NRF2/ARE signaling in CP/CPPS rat model and human prostate epithelial cell injury model.

Multiple sarcomatoid carcinomas in the small intestine with perforation: A case report and literature review.

Sarcomatoid carcinoma of the small intestine is an exceedingly rare and aggressive malignancy, often diagnosed at advanced stages with a poor prognosis. This study documents a detailed case of sarcomatoid carcinoma of the small intestine, highlighting the diagnostic challenges and treatment approaches, underscored by a comprehensive review of related literature. Given the rarity of this condition, our report aims to enrich the existing diagnostic and treatment frameworks for this malignancy, emphasizing the necessity for early detection and intervention strategies. By presenting this case in conjunction with a literature review, we seek to shed light on the elusive nature of sarcomatoid carcinoma in the small intestine and propose avenues for improving patient outcomes. Case presentation A 61-year-old male patient initially presented with recurrent abdominal pain and gastrointestinal symptoms. Initial abdominal computed tomography (CT) scans and gastrointestinal endoscopy revealed only inflammatory and hyperplastic changes in the duodenum and jejunum, with a diagnosis of intestinal obstruction. Two years later, due to gastrointestinal perforation, the patient was hospitalized again. CT scans and other examinations revealed small intestinal lesions. Four small intestinal lesions were surgically removed, and pathology and immunohistochemistry confirmed sarcomatoid carcinoma of the small intestine. A short time later, enhanced CT scans revealed metastatic lesions in the hepatic portal and adrenal glands. After surgery, the gastrointestinal function gradually recovered, and the patient was discharged from the hospital on a semiliquid diet. No further treatment such as radiotherapy or chemotherapy was administered postoperatively. Five months after the surgery, the patient died due to brain metastasis. The study outcomes reveal the aggressive nature of sarcomatoid carcinoma of the small intestine, characterized by rapid progression and poor prognosis despite surgical interventions. The patient condition rapidly deteriorated, leading to metastasis and death within 5 months postsurgery. These findings underscore the critical need for early detection and possibly innovative treatment approaches to improve survival rates. This case also highlights the potential for gastrointestinal sarcomatoid carcinoma to metastasize to distant organs, including the brain, suggesting a propensity for hematogenous spread.