Diagnostic performance of serum CA-125 for overall and complicated acute appendicitis: a systematic review and meta-analysis.

This study aimed to analyze the diagnostic performance of serum CA-125 in acute appendicitis (AA). This review was registered in PROSPERO (CRD42023450988). We included prospective and retrospective original clinical studies evaluating the diagnostic performance of serum CA-125 in AA. A search was conducted in PubMed, Web of Science, Scopus, and OVID. Search terms and keywords were: (appendicitis OR appendectomy) AND (CA-125 OR CA125). Two independent reviewers selected the articles and extracted relevant data. Methodological quality was assessed using the QUADAS-2 index. A synthesis of the results, standardization of the metrics, and three random-effect meta-analyses were performed. Five studies with data from 533 participants (including 219 patients with a confirmed diagnosis of AA and 107 controls) were included in this review. The random-effect meta-analysis of serum CA-125 (AA vs controls) included 3 articles (125 AA and 70 controls) and resulted in a non-significant mean difference [95% CI] of - 6.80 [- 20.51, 6.92] U/mL (p = 0.33). The meta-analysis by subgroups that included only male patients resulted in a significant mean difference [95% CI] of 3.48 [0.46, 6.49] U/mL (p = 0.02). Although serum CA-125 does not appear to be a good overall marker for the diagnosis of AA, our subgroup analyses show that this marker could be useful for diagnosing AA in males. It also appears to be a potentially useful tool for discriminating complicated and uncomplicated AA. However, the limited number of included studies precludes drawing generalizable conclusions. Future prospective studies focused on males and in its potential ability to discriminate between complicated and uncomplicated AA are required.Registration. PROSPERO (CRD42023450988).

Thoracic lipoblastoma in a 6-year-old African male: a case report.

Lipoblastoma is a very infrequent tumor, characteristic of early childhood. The thoracic location is infrequent, with isolated reports to date. We present the case of a 6-year-old male patient with a right thoracic tumor of months of evolution that was surgically removed by right anterolateral thoracotomy and in which the diagnosis of classic well-differentiated lipoblastoma was histologically confirmed. The patient evolved favorably and was discharged. He is currently under follow-up and without recurrence 1 year after surgery. This is, to our knowledge, the first thoracic lipoblastoma reported in an African pediatric patient. The importance of knowing the clinical, semiological, and intraoperative characteristics of this tumor becomes even more important, as in our case, in the context of international cooperation, where in many cases, there is no possibility of performing pre-operative imaging studies or subsequent genetic studies.

El lipoblastoma es un tumor muy infrecuente, característico de la primera infancia. La localización torácica es infrecuente, con reportes aislados hasta la fecha. Presentamos el caso de un paciente varón de 6 años con una tumoración torácica derecha de meses de evolución que fue extirpada quirúrgicamente mediante toracotomía anterolateral derecha y en la que se confirmó histológicamente el diagnóstico de lipoblastoma clásico bien diferenciado. El paciente evolucionó favorablemente y fue dado de alta. Actualmente se encuentra en seguimiento y sin recidiva un año después de la cirugía. Este es, hasta donde sabemos, el primer lipoblastoma torácico reportado en un paciente pediátrico africano. La importancia de conocer las características clínicas, semiológicas e intraoperatorias de este tumor cobra aún más importancia, como en nuestro caso, en el contexto de la cooperación internacional, donde en muchos casos no existe la posibilidad de realizar estudios de imagen preoperatorios ni estudios genéticos posteriores.

Pediatric Rectosigmoid Atypical Juvenile Polyps Presenting With Rectal Prolapse and Acute Bleeding: A Case Report and a Comprehensive Literature Review.

Rectosigmoid solitary juvenile polyps are benign lesions, relatively frequent in childhood. The clinical debut of a pediatric polyp with bleeding is relatively frequent, but there are very few reports of rectal prolapse of polyps. We present the case of a 7-year-old female patient with no previous history who presented with rectal prolapse of a polyp with acute bleeding. An urgent endoscopic examination was performed and 2 rectosigmoid polypoid lesions were found and resected. The anatomopathological study showed that these were 2 hamartomatous polyps with mild dysplasia. The patient is asymptomatic and is being followed up. The literature concerning rectal prolapse of polyps in the pediatric population is scarce. In a pediatric patient with a rectal prolapse, this entity should be considered in the differential diagnosis.

Pop-off mechanisms as renoprotective mediators in children with posterior urethral valves: A systematic review and meta-analysis.

BACKGROUND: Pop-off mechanisms are potential pressure-relieving mediators in patients diagnosed with posterior urethral valves (PUV). This systematic review aimed to synthesize the existing evidence regarding the protective effect of pop-off mechanisms on renal function in children with PUV. METHODS: We conducted a systematic review of the literature that involved an extensive search in the main databases of the medical bibliography. Three independent reviewers selected the relevant articles. Methodological quality was rated using the Newcastle Ottawa Scale index. We used random meta-analyses to compare different outcomes (serum creatine, Nadir serum creatinine, and Chronic Kidney Disease) between children with PUV and pop-off mechanisms and those with PUV without pop-off mechanisms. RESULTS: 10 studies with data from 896 participants were included in this review. Seven articles reported serum creatinine values for each group and 3 of them found significant differences between groups. The random-effects meta-analysis for serum creatinine showed significant lower mean (diff = -52.88 µmol/L [95 % CI -73.65 to -32.11]) in the group of children with pop-off mechanisms, and the random-effects meta-analysis for Nadir serum creatinine showed a marginally significantly lower mean in the group of children with pop-off mechanisms (diff = -12.00 µmol/L [95 % CI -24.04 to 0.04]). The random-effect meta-analysis for Chronic Kidney Disease resulted in a significant risk reduction for the group of children with pop-off mechanisms (odds ratio = 0.48 [95 % CI 0.23 to 0.98]). CONCLUSIONS: Children with PUV and pop-off mechanisms show better renal function and lower risk of Chronic Kidney Disease than those with PUV without pop-off mechanisms suggesting these mechanisms may act as renoprotective mediators. The high heterogeneity between studies in the assessment of renal function and long-term outcomes calls for a cautious interpretation of these findings. Future studies that stratify by different types of pop-off mechanisms and use standardized metrics, such as Nadir creatinine, are needed.

Superior Vesical Fissure as an Incomplete Form of Bladder Exstrophy: A Case Report with Clinico-Pathological Correlation and a Comprehensive Literature Review.

Multiple variants of classic bladder exstrophy have been described, all of them infrequent. Superior vesical fissure is a mild variant of this pathology in which genital involvement is scarce or absent. To date, there are only isolated reports of this entity. We report a full-term female patient of Arabian descent with a clinical and radiological diagnosis of superior vesical fissure that was surgically corrected in our center with a favorable evolution. Histological study, supported by immunohistochemical techniques, showed squamous and transitional epithelium and discrete chronic inflammation. Our literature review identified 26 reports of superior vesical fissure (including ours), with high heterogeneity in terms of clinical characterization and associated malformations and with only two histological reports. The clinical evolution of the patients reported in the literature was favorable, with lower morbidity and mortality than in classical forms of bladder exstrophy.

Diagnostic performance of urinary 5-Hydroxyindoleacetic Acid in acute appendicitis: a systematic review and diagnostic test accuracy meta-analysis.

OBJECTIVES: This study aimed to analyze the diagnostic performance of urinary 5-Hydroxyindoleacetic Acid (5-HIAA) in acute appendicitis (AA). METHODS: This review was registered in PROSPERO (CRD42023399541). We included prospective or retrospective original clinical studies evaluating the diagnostic performance of 5-HIAA in AA. A search was conducted in PubMed, Web of Science, Scopus and OVID. Search terms and keywords were: (appendicitis OR acute appendicitis) AND (5-HIAA OR 5-Hydroxyindoleacetic acid OR serotonin metabolite). Two independent reviewers selected the articles and extracted relevant data. Methodological quality was assessed using the QUADAS2 index. A synthesis of the results, standardization of the metrics, and a random-effect meta-analysis were performed. Additionally, a coupled forest plot and a diagnostic test accuracy meta-analysis (DTA) were performed. RESULTS: Twelve studies with data from 1467 participants (724 patients with a confirmed diagnosis of AA and 743 controls) were included in this review. The random-effect meta-analysis of urinary 5-HIAA (AA vs controls) included 7 articles (352 AA and 258 controls) and resulted in a significant mean difference [95% CI] of 23.30 [15.82-30.77] µmol/L (p < 0.001). The DTA meta-analysis of urinary 5-HIAA included 8 articles and resulted in a pooled sensitivity [95% CI] of 68.6 [44.1-85.9]% and a pooled specificity [95% CI] of 82 [54.7-94.5]%. CONCLUSIONS: Although the evidence is heterogeneous and limited, urinary 5-HIAA emerges as a potential non-invasive diagnostic tool for AA. Urinary 5-HIAA does not seem to be a useful biomarker to distinguish between NCAA and CAA. Future prospective studies with a large sample size and a rigorous design are necessary to validate these findings. TRIAL REGISTRATION: PROSPERO (CRD42023399541).

Sea-blue histiocytosis.

A 78-year-old woman with hypertrophic cardiomyopathy underwent a septal myomectomy and valve replacement. In the immediate postoperative period she developed shock of mixed etiology and died. At autopsy, hepatomegaly and splenomegaly were identified, with PAS and Giemsa positive intracellular ceroid granular deposits. Sea-blue histiocytosis is an extremely rare, chronic and benign deposit disease. It is characterized by hepatosplenomegaly, thrombocytopenia and lymphadenopathy. The presence of ceroid substance in granules in PAS and Giemsa stains should establish the diagnosis of suspicion.

Flow cytometric characterization of cecal appendix lymphocyte subpopulations in children: a pilot study.

INTRODUCTION: Scientific literature regarding the characterization of lymphocyte subpopulations of the cecal appendix is sparse, with few precedents limited to immunohistochemical techniques. METHODS: We conducted a prospective pilot study to characterize lymphocyte subpopulations of the cecal appendix in children. Participants were divided into three groups: (1) patients without histological acute appendiceal inflammation, (2) patients with histological uncomplicated acute appendicitis, and (3) patients with histological complicated acute appendicitis (gangrenous, perforated). A fresh sample of the base of the appendix was taken from all patients and a flow cytometric study was performed. Quantitative variables were compared using Kruskal-Wallis test and Mann-Whitney U test. RESULTS: This study included 57 patients divided into Group 1 (n = 5), Group 2 (n = 37), and Group 3 (n = 15). Median values (IQR) of the percentage of B-lymphocytes were 67.8 [66.8-68.1] in group 1, 61.15 [53.74-66.4] in group 2, and 52.1 [33-62.02] in group 3 (p = 0.02). Median values (IQR) of the percentage of NK-lymphocytes were 0.26 [0.2-0.3] in group 1, 0.55 [0.37-0.66] in group 2, and 0.84 [0.35-1.45] in group 3 (p = 0.008). Median values (IQR) of the percentage of T-lymphocytes were 31.9 [31.7-33.1] in group 1, 37.68 [32.15-45.69] in group 2, and 46.9 [37.03-67] in group 3 (p = 0.02). Pair comparisons of groups 2 and 3 also showed significant differences in the percentage of B lymphocytes (p = 0.03) and NK-lymphocytes (p = 0.02). CONCLUSIONS: Significant differences in lymphocyte subpopulations were identified according to the histologic grade of the cecal appendix. More specifically, a lower percentage of B-lymphocytes and a higher percentage of T- and NK-lymphocytes were observed in cases of acute appendicitis. These findings must be confirmed and their etiopathogenic, diagnostic, and prognostic implications elucidated in future studies with larger sample sizes.

Congenital Meatal Urethral Stenosis in a Female Patient: A Case Report.

We present the case of a 6-year-old girl who presented with alterations in the voiding stream. On physical examination, a very small urethral meatus was identified at the expense of a membrane. The renovesical ultrasound showed no alterations. An uroflowmetric study was performed, showing a bladder outlet obstruction pattern. The urethral meatus was calibrated and a ventral meatotomy was performed. The histological study of the resected membrane showed a transitional urethral mucosa with chronic focal inflammation and discrete hyperplasia. The patient evolved favorably, with resolution of the symptoms and no notable complications. This is, to the best of our knowledge, the first reported case with a histological study of a congenital meatal urethral stenosis. In the presence of lower urinary tract obstruction, this entity should be considered in the differential diagnosis. Surgical treatment is curative.

Post-Operative Increase in Serum Interleukin-6 Is Associated With Longer Hospital Stay in Children Operated on for Acute Appendicitis: A Pilot Study.

Background: Pediatric acute appendicitis (PAA) involves a substantial consumption of health and economic resources. The identification of serum biomarkers that may help predict the post-surgical evolution of these patients is a field of great interest. Patients and Methods: This was a prospective, observational substudy within the Biomarkers for the Diagnosis of Appendicitis in Pediatrics (BIDIAP) cohort aimed at evaluating the association between post-surgical increase in serum IL-6 and different outcomes related to the clinical evolution of children operated on for PAA. Sixty-nine children with a confirmed diagnosis of acute appendicitis and both pre-operative and post-operative serum IL-6 were included in the study. Three multivariable-adjusted linear regression models were fitted to analyze the association between an increase of >10% in post-operative serum IL-6 level with the length of stay, the number of post-operative emetic episodes, and the onset of oral feeding. Two multivariable-adjusted logistic regression models were fitted to assess the association of the same exposure with the indication of antibiotherapy at discharge and with positivity in peritoneal fluid culture. Results: Thirteen children showed an increase of >10% in the post-operative serum IL-6 value (group 1) whereas 56 showed only a minor increase, or no change (group 2). After accounting for potential confounders, children in group 1 had a mean of three-day longer hospital stay (difference, 3.33; 95% confidence interval [CI], 0.57-6.09) and higher odds of a positive result in peritoneal fluid culture (odds ratio [OR], 37.43; 95% CI, 1.02-1361.28) than children in group 2. Conclusions: An increase of >10% in post-operative serum IL-6 value could predict longer hospital stay and higher odds of positive peritoneal fluid culture. Future prospective studies are needed to replicate these findings and to broaden the range of biomarkers that could predict the post-operative evolution of children operated on for PAA.

Leucine-Rich Alpha-2-Glycoprotein as a non-invasive biomarker for pediatric acute appendicitis: a systematic review and meta-analysis.

The aim of this study was to analyze the diagnostic performance of Leucine-Rich Alpha-2-Glycoprotein (LRG1) in pediatric acute appendicitis (PAA). We conducted a systematic review of the literature in the main databases of medical bibliography. Two independent reviewers selected the articles and extracted relevant data. Methodological quality was assessed using the QUADAS2 index. A synthesis of the results, standardization of the metrics and 4 random-effect meta-analyses were performed. Eight studies with data from 712 participants (305 patients with confirmed diagnosis of PAA and 407 controls) were included in this review. The random-effect meta-analysis of serum LRG1 (PAA vs control) resulted in a significant mean difference (95% CI) of 46.76 µg/mL (29.26-64.26). The random-effect meta-analysis for unadjusted urinary LRG1 (PAA vs control) resulted in a significant mean difference (95% CI) of 0.61 µg/mL (0.30-0.93). The random-effect meta-analysis (PAA vs control) for urinary LRG1 adjusted for urinary creatinine resulted in a significant mean difference (95% CI) of 0.89 g/mol (0.11-1.66).   Conlusion: Urinary LRG1 emerges as a potential non-invasive biomarker for the diagnosis of PAA. On the other hand, due to the high between-study heterogeneity, the results on serum LRG1 should be interpreted with caution. The only study that analyzed salivary LRG1 showed promising results. Further prospective studies are needed to confirm these findings. What is Known: • Pediatric acute appendicitis continues to be a pathology with a high rate of diagnostic error. • Invasive tests, although useful, are a source of stress for patients and their parents. What is New: • LRG1 emerges as a promising urinary and salivary biomarker for the noninvasive diagnosis of pediatric acute appendicitis.