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Expert Opinion on Biological Therapy Mar 2020: Anti-Tumor Necrosis Factor-alpha (TNF-α) therapy, primarily infliximab and adalimumab, are now increasingly used to induce and maintain disease remission in the...
: Anti-Tumor Necrosis Factor-alpha (TNF-α) therapy, primarily infliximab and adalimumab, are now increasingly used to induce and maintain disease remission in the pediatric perianal Crohn's disease (CD) population, however, their optimal use has not yet been defined in the pediatric setting.: In accordance with a published protocol (PROSPERO no. CRD42019118838), we systematically and critically evaluated all published evidence on the efficacy and safety of anti-TNF-α in children with perianal CD, in the PubMed, MEDLINE, Embase, Cochrane and clinicalTrials.gov databases until October, 18, 2018. We included in our systematic review 29 articles yielding a total of 565 perianal CD patients aged between 9 months to 18 years.: According to low-quality evidence from small, uncontrolled and heterogeneous descriptive studies, and very few randomized controlled trial, nearly three-fifths children with perianal CD achieved remission with anti-TNF-α treatment and in approximately 40% remission was maintained after 12 months, with practically low discontinuation rate due to serious adverse events. More than half of the patients achieved complete fistula closure. There is still a need for more robust evidence adequately assessing the efficacy and safety of anti-TNF-α therapy in pediatric perianal CD, as well as in comparison with other therapies.
Topics: Adalimumab; Anemia; Clinical Trials as Topic; Crohn Disease; Humans; Infliximab; Outcome Assessment, Health Care; Treatment Outcome; Tumor Necrosis Factor-alpha
PubMed: 31971447
DOI: 10.1080/14712598.2020.1718096 -
Frontiers in Immunology 2022Haploinsufficiency of A20 (HA20) is a newly described rare autoinflammatory disease caused by gene mutations. HA20 has seldom been documented in the Chinese population....
OBJECTIVE
Haploinsufficiency of A20 (HA20) is a newly described rare autoinflammatory disease caused by gene mutations. HA20 has seldom been documented in the Chinese population. Herein, we report eight patients with HA20 from three unrelated families in China.
METHODS
Eight Chinese Han patients were diagnosed with HA20 in our department from 2018 to 2021. Their clinical data and genotypes were carefully documented and studied. The newly identified variants were functionally verified. We also conducted a systematic literature review of HA20, and the clinical characteristics and genotype of HA20 between the Chinese population and other populations were compared.
RESULTS
Eight HA20 patients from three families comprised six adults and two children. There was one man and seven women. The clinical characteristics included recurrent oral ulcers (8/8, 100%), fever (4/8, 50%), perianal ulcer (3/8, 38%), skin lesions (2/8, 25%), arthritis (1/8, 13%), and uveitis (1/8, 13%). Three variants, A547T, c.1906+2T>G, and R271X, were identified. Two novel variants, A547T and c.1906+2T>G, were validated to be pathogenic in our study. In a literature review a total of 126 patients with HA20 reported by 35 articles were included. The clinical phenotype of Chinese HA20 patients was similar to that of patients from other populations except for a lower frequency of genital ulcers (16.7% vs. 54.4%, p < 0.01). Autoantibodies were detectable in approximately one-third of the 126 patients, among which ANA and anti-thyroid antibodies were commonly seen.
CONCLUSION
The rarity and diversity of phenotypes make the diagnosis of HA20 a huge challenge to physicians. HA20 should be considered in child-onset patients with manifestations that resemble Behçet's syndrome, especially those whose family members have similar symptoms. Gene testing is critically helpful for the diagnosis of HA20. Two novel variants, A547T and c.1906+2T>G, were identified in this study.
Topics: Asian People; Cytokines; Female; Haploinsufficiency; Humans; Pedigree; Phenotype
PubMed: 35958611
DOI: 10.3389/fimmu.2022.955079 -
Gastroenterology Jan 2021A better understanding of prognostic factors within the heterogeneous spectrum of pediatric Crohn's disease (CD) should improve patient management and reduce... (Meta-Analysis)
Meta-Analysis
Predicting Outcomes in Pediatric Crohn's Disease for Management Optimization: Systematic Review and Consensus Statements From the Pediatric Inflammatory Bowel Disease-Ahead Program.
BACKGROUND & AIMS
A better understanding of prognostic factors within the heterogeneous spectrum of pediatric Crohn's disease (CD) should improve patient management and reduce complications. We aimed to identify evidence-based predictors of outcomes with the goal of optimizing individual patient management.
METHODS
A survey of 202 experts in pediatric CD identified and prioritized adverse outcomes to be avoided. A systematic review of the literature with meta-analysis, when possible, was performed to identify clinical studies that investigated predictors of these outcomes. Multiple national and international face-to-face meetings were held to draft consensus statements based on the published evidence.
RESULTS
Consensus was reached on 27 statements regarding prognostic factors for surgery, complications, chronically active pediatric CD, and hospitalization. Prognostic factors for surgery included CD diagnosis during adolescence, growth impairment, NOD2/CARD15 polymorphisms, disease behavior, and positive anti-Saccharomyces cerevisiae antibody status. Isolated colonic disease was associated with fewer surgeries. Older age at presentation, small bowel disease, serology (anti-Saccharomyces cerevisiae antibody, antiflagellin, and OmpC), NOD2/CARD15 polymorphisms, perianal disease, and ethnicity were risk factors for penetrating (B3) and/or stenotic disease (B2). Male sex, young age at onset, small bowel disease, more active disease, and diagnostic delay may be associated with growth impairment. Malnutrition and higher disease activity were associated with reduced bone density.
CONCLUSIONS
These evidence-based consensus statements offer insight into predictors of poor outcomes in pediatric CD and are valuable when developing treatment algorithms and planning future studies. Targeted longitudinal studies are needed to further characterize prognostic factors in pediatric CD and to evaluate the impact of treatment algorithms tailored to individual patient risk.
Topics: Adolescent; Child; Child, Preschool; Consensus; Crohn Disease; Female; Humans; Infant; Infant, Newborn; Male; Outcome Assessment, Health Care; Predictive Value of Tests; Prognosis
PubMed: 32979356
DOI: 10.1053/j.gastro.2020.07.065 -
European Journal of Pediatric Surgery :... Jun 2024This systematic review and meta-analysis of nonrandomized studies (NRSs) aimed to evaluate the clinical efficacy and safety of two types of surgical interventions... (Meta-Analysis)
Meta-Analysis Comparative Study
This systematic review and meta-analysis of nonrandomized studies (NRSs) aimed to evaluate the clinical efficacy and safety of two types of surgical interventions (respectively drainage alone and drainage with primary fistula treatment) for perianal abscesses (PAs) in children. Studies from 1992 to July 2022 were searched in 10 electronic databases. All relevant NRSs with available data which compared surgical drainage with or without primary fistula treatment were included. Patients with underlying diseases which led to abscess formation were excluded. The Newcastle-Ottawa Scale was used to assess the risk of bias and quality of the included studies. The outcomes were the healing rate, fistula formation rate, fecal incontinence, and wound healing duration. A total of 16 articles with 1,262 patients were considered suitable for the final meta-analysis. Primary fistula treatment was associated with a significantly higher healing rate when compared with incision and drainage alone (odds ratio [OR]: 5.76, 95% confidence interval [CI]: 4.04-8.22). This aggressive procedure for PA resulted in an 86% reduction in the fistula formation rate (OR: 0.14, 95% CI: 0.06-0.32). Limited data showed patients who underwent primary fistula treatment have a minor effect on postoperative fecal incontinence. Primary fistula treatment demonstrates a better clinical efficacy in promoting the healing rate and decreasing the formation of fistulas in PAs in children. The available evidence for a minor impact on anal function after this intervention is less strong.
Topics: Humans; Drainage; Child; Abscess; Rectal Fistula; Anus Diseases; Fecal Incontinence; Treatment Outcome; Wound Healing
PubMed: 37023788
DOI: 10.1055/a-2070-3613 -
Stem Cell Research & Therapy Feb 2024
PubMed: 38365762
DOI: 10.1186/s13287-024-03664-w -
BJS Open Feb 2020
Systematic review and meta-analysis of endorectal advancement flap and ligation of the intersphincteric fistula tract for cryptoglandular and Crohn's high perianal fistulas.
PubMed: 32011811
DOI: 10.1002/bjs5.50249 -
Biomolecules Jan 2021Inflammatory bowel disease (IBD) is a chronic inflammatory disorder of the gut that can lead to severe gastrointestinal symptoms, malnutrition, and complications such as...
Inflammatory bowel disease (IBD) is a chronic inflammatory disorder of the gut that can lead to severe gastrointestinal symptoms, malnutrition, and complications such as fistulas and cancer. Mesenchymal stem/stromal cells (MSCs) are being investigated as a novel therapy for IBD and have been demonstrated to be safe and effective for perianal fistulizing Crohn's disease (PFCD). This systematic review aims to present the most recent studies on the safety and efficacy of MSC therapy in IBD. A detailed search strategy of clinical trials on MSCs and IBD was performed on PubMed, with 32 studies selected for inclusion in this review. The newest studies on local MSC injection for PFCD continue to support long-term efficacy while maintaining a favorable safety profile. The evidence for systemic MSC infusion in luminal IBD remains mixed due to marked methodological heterogeneity and unclear safety profiles. Although further studies are needed to better establish the role of this novel treatment modality, MSCs are proving to be a very exciting addition to the limited therapies available for IBD.
Topics: Crohn Disease; Humans; Inflammatory Bowel Diseases; Injections; Mesenchymal Stem Cell Transplantation; Mesenchymal Stem Cells
PubMed: 33440772
DOI: 10.3390/biom11010082 -
Chirurgia (Bucharest, Romania : 1990) 2020Lung cancer is the most frequent cancer, accounting for over 2 million new cases per year worldwide. In Romania, the cancers with the highest incidence are lung cancer...
Lung cancer is the most frequent cancer, accounting for over 2 million new cases per year worldwide. In Romania, the cancers with the highest incidence are lung cancer for men and breast cancer for women. Cancer-related deaths follow the same pattern. More than 50% of the patients with lung cancers have distant metastases at the time of diagnosis. Metastases from lung cancer occur mainly in the brain, bones, liver, and adrenals. Anal metastases from primary lung cancer are extremely uncommon. As far as we know there are only 12 cases reported in the literature until now. Case report: Case report and systematic review. We performed a systematic literature search in PubMed using the following MeSH terms: "lung cancer metastasis" AND "anal" OR "anus" OR "perianal". The search was conducted from the beginning of the database onwards. No language exclusion criteria were used. We report the case of a patient with advanced lung adenocarcinoma presenting with a painful, ulcerated and bleeding anal tumor. The anal tumor was excised with primary wound closure in spinal anesthesia. The pathology report and the immunohistochemistry of the specimen revealed metastasis of the lung adenocarcinoma. The review included 12 papers reporting 12 cases. The male/female ratio was 3/1. The average age was 62,16 years. Among these 12 patients, one had a SCLC. From the remaining 11 cases with NSCLC, 4 were adenocarcinomas, 4 squamous cell carcinomas, two anaplastic carcinomas and one mucoepidermoid carcinoma. The anal metastases were synchronous in 4 cases and metachronous in the rest of 8 cases. Since 2006 immuno-histochemistry plays an important role in the diagnosis of these metastases. Our patient presented an anal metastasis of a lung adenocarcinoma that was masquerading as a complicated hemorrhoidal disease. Because anal masses can be easily misdiagnosed, patients with lung cancers and anal or perianal complaints should be evaluated for metastatic disease. Medical teams that are managing such cases must be aware of this rare but possible situation. Immunohistochemistry plays an important role in establishing the diagnosis for the anal or perianal tumours in patients with lung cancer.
Topics: Adenocarcinoma; Anus Neoplasms; Diagnosis, Differential; Hemorrhoids; Humans; Lung Neoplasms
PubMed: 33138907
DOI: 10.21614/chirurgia.115.5.681 -
PloS One 2024Intracutaneous pyonex therapy (IPT), a novel acupuncture technique also known as intradermal thumbtack needle embedding therapy, has been reported to optimize... (Meta-Analysis)
Meta-Analysis
Intracutaneous pyonex therapy (IPT), a novel acupuncture technique also known as intradermal thumbtack needle embedding therapy, has been reported to optimize postoperative pain management following perianal surgery. This meta-analysis aimed to analyze the efficacy of IPT for postoperative pain management following perianal surgery. The Cochrane Library, PubMed, EMBASE, Web of Science, CNKI, SinoMed, Wanfang, and VIP databases were systematically searched for randomized controlled trials (RCTs) on IPT as a treatment for postoperative pain management following perianal surgery from inception until June 15, 2022. The analyzed outcomes from the eleven RCTs included in this meta-analysis were as follows: postoperative visual analogue scale(VAS), analgesic duration, ineffective cases following treatment, and adverse events. Subgroup analyses were conducted according to different time points. Risk-of-bias assessment, publication bias analysis, sensitivity analysis, and trial sequential analysis were performed. Of the 895 patients, 450 and 445 were included in the IPT and control groups, respectively. The IPT group showed a better analgesic effect[standard mean difference (SMD) = -0.77, 95% CI: -1.00 to -0.53, P < 0.00001; P for heterogeneity = 0.009, I2 = 59%] and longer analgesic duration [SMD = 0.56, 95% CI: 0.31 to 0.82, P < 0.0001; P for heterogeneity = 0.6, I2 = 0%], fewer ineffective cases following treatment [risk ratio(RR) = 0.23; 95% CI: 0.13 to 0.39, P < 0.00001; P for heterogeneity = 0.76, I2 = 0%], and lower overall occurrence of postoperative complications [RR = 0.35; 95% CI: 0.17 to 0.70; P = 0.003; P for heterogeneity = 0.85, I2 = 0%] than the control group. Thus, our findings indicated that IPT can provide better pain management following perianal surgery compared to controls. This novel approach complements a reasonable modality for postoperative multimodal analgesia and is worth promoting.
Topics: Humans; Analgesics; Pain Management; Pain, Postoperative; Postoperative Complications
PubMed: 38241273
DOI: 10.1371/journal.pone.0296439 -
International Journal of Molecular... Jul 2020Stromal vascular fraction (SVF) containing adipose stem cells (ASCs) has been used for many years in regenerative plastic surgery for autologous applications, without... (Meta-Analysis)
Meta-Analysis
Systematic Review: Allogenic Use of Stromal Vascular Fraction (SVF) and Decellularized Extracellular Matrices (ECM) as Advanced Therapy Medicinal Products (ATMP) in Tissue Regeneration.
Stromal vascular fraction (SVF) containing adipose stem cells (ASCs) has been used for many years in regenerative plastic surgery for autologous applications, without any focus on their potential allogenic role. Allogenic SVF transplants could be based on the possibility to use decellularized extracellular matrix (ECM) as a scaffold from a donor then re-cellularized by ASCs of the recipient, in order to develop the advanced therapy medicinal products (ATMP) in fully personalized clinical approaches. A systematic review of this field has been realized in accordance with the Preferred Reporting for Items for Systematic Reviews and Meta-Analyses-Protocols (PRISMA-P) guidelines. Multistep research of the PubMed, Embase, MEDLINE, Pre-MEDLINE, PsycINFO, CINAHL, Clinicaltrials.gov, Scopus database, and Cochrane databases has been conducted to identify articles and investigations on human allogenic ASCs transplant for clinical use. Of the 341 articles identified, 313 were initially assessed for eligibility on the basis of the abstract. Of these, only 29 met all the predetermined criteria for inclusion according to the PICOS (patients, intervention, comparator, outcomes, and study design) approach, and 19 have been included in quantitative synthesis (meta-analysis). Ninety-one percent of the studies previously screened (284 papers) were focused on the in vitro results and pre-clinical experiments. The allogenic use regarded the treatment of perianal fistulas, diabetic foot ulcers, knee osteoarthritis, acute respiratory distress syndrome, refractory rheumatoid arthritis, pediatrics disease, fecal incontinence, ischemic heart disease, autoimmune encephalomyelitis, lateral epicondylitis, and soft tissue defects. The information analyzed suggested the safety and efficacy of allogenic ASCs and ECM transplants without major side effects.
Topics: Adipose Tissue; Animals; Extracellular Matrix; Humans; Mesenchymal Stem Cell Transplantation; Mesenchymal Stem Cells; Regenerative Medicine; Tissue Engineering; Tissue Scaffolds; Transplantation, Homologous
PubMed: 32679697
DOI: 10.3390/ijms21144982