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Abstract We assess the performance of different break detection methods on three sets of benchmark data sets, each consisting of 120 daily time series of integrated water vapor differences. These differences are generated from the Global Positioning System (GPS) measurements at 120 sites worldwide, and the numerical weather prediction reanalysis (ERA‐Interim) integrated water vapor output, which serves as the reference series here. The benchmark includes homogeneous and inhomogeneous sections with added nonclimatic shifts (breaks) in the latter. Three different variants of the benchmark time series are produced, with increasing complexity, by adding autoregressive noise of the first order to the white noise model and the periodic behavior and consecutively by adding gaps and allowing nonclimatic trends. The purpose of this “complex experiment” is to examine the performance of break detection methods in a more realistic case when the reference series are not homogeneous. We evaluate the performance of break detection methods with skill scores, centered root mean square errors (CRMSE), and trend differences relative to the trends of the homogeneous series. We found that most methods underestimate the number of breaks and have a significant number of false detections. Despite this, the degree of CRMSE reduction is significant (roughly between 40% and 80%) in the easy to moderate experiments, with the ratio of trend bias reduction is even exceeding the 90% of the raw data error. For the complex experiment, the improvement ranges between 15% and 35% with respect to the raw data, both in terms of RMSE and trend estimations.

Summary: Few studies investigated the genetics of relapsed Wilms tumor (WT), suggesting the SIX1 gene, the microRNA processing genes, and the MYCN network as possibly involved in a relevant percentage of relapses. We investigated 28 relapsing WT patients (10 new cases and 18 cases in which the involvement of SIX and miRNAPG had been excluded) with a panel of ∼5000 genes. We identified variants affecting genes involved in DNA damage prevention and repair in 12/28 relapsing patients (42.9%), and affecting genes involved in chromatin modification and regulation in 6/28 relapsing patients (21.4%), widening the spectrum of anomalies detected in relapsed tumors. The disclosure of molecular pathways possibly underlying tumor progression might allow to use molecularly targeted therapies at relapse. Surprisingly, germline anomalies, mostly affecting DNA damage prevention and repair genes, were identified in 13/28 patients (46.4%), raising the issue of performing a genetic testing to all children presenting with a WT.

The Bosniak classification of renal cysts aims to provide a probabilistic risk assessment indicating the likelihood of malignancy from imaging findings. Originally designed to classify adult renal cysts based on computed tomography findings, the Bosniak classification has been extended to pediatric patients, with some adjustments made with the aim of accommodating magnetic resonance imaging (MRI) and ultrasonography (US). Bosniak IV lesions are rare in adolescents, indicating localized renal cell carcinoma and requiring surgical intervention. In contrast, Bosniak III lesions can be treated conservatively, although there is a lack of specific guidelines on their management. We present a case of a 14-year-old boy with a Bosniak III lesion, which was incidentally detected during the US evaluation of a left varicocele. After a 12-month follow-up, MRI revealed progression to a Bosniak IV cyst. Robot-assisted tumor enucleation was performed with selective artery clamping when the patient was 15. Histopathology showed tubulocystic renal cell carcinoma without adverse features. Immunocytochemistry supported a favorable prognosis of this rare tumor (

IntroductionThis study examined the efficacy of eye movement desensitisation and reprocessing (EMDR) therapy compared with standard psychotherapy (SP) in treating post-traumatic stress disorder (PTSD) in paediatric oncology patients and their families in the early stage of cancer treatment. The secondary aim of this study was to assess whether EMDR therapy has a different impact on post-traumatic growth compared to SP.MethodsForty patients were randomly assigned to EMDR or SP groups. The Impact of Event Scale – Revised (IES-R) and the Distress Thermometer (DT) were used to assess PTSD symptoms at pre-treatment (at cancer diagnosis) and in the post-treatment stages (after 8 sessions). The Post-traumatic Growth Inventory-PTGI was administered in the post-treatment stage in order to evaluate positive changes.ResultsBoth EMDR and SP are effective in reducing PTSD, but EMDR was significantly more effective than the SP in reducing scores on the IES-R, especially regarding the intrusive symptom subscale. Also, in the EMDR group there were higher scores of PTGI than in the standard group.ConclusionEMDR thus represents a promising treatment in the paediatric psycho-oncology setting.

Background: Wilms tumor (WT) is the most frequent renal tumor in children. The SIOP-UMBRELLA Guidelines allow for nephron-sparing surgery (NSS) in syndromic patients, as well as in cases of small (

IntroductionChildhood cancer is rare, but it is the most frequent serious event with a high risk of traumatization for children, adolescents, parents and siblings. EMDR is widely studied as clinical intervention that addressed cancer-related stressors among cancer adult population, but to our knowledge, no researches have been conducted among children and adolescent with cancer.MethodsThe purpose of this case study is to describe for the first time the application of the EMDR protocol in a pediatric hospital setting with a 17-years-old Italian adolescent who received a diagnosis of leukemia. He accessed the psychological support service complaining of feelings of anxiety and general discomfort. EMDR protocol started after the diagnosis and ended after the usual eight phases. The Impact of Event Scale—Revised (IES-R) was used to assess stress disorders symptoms as outcome at the baseline (before the First Phase) and at the end of the EMDR protocol (after the Eight Phase).ResultsBy using EMDR protocol the patient reported a decrease of emotional activation after a few EMDR sessions.ConclusionEMDR protocol may be effective for pediatric cancer patients in treating stress disorders symptoms and it can be proposed immediately after diagnosis as a standard care also in pediatric hospital setting.

IntroductionMulibrey nanism (MUL) is a rare disorder caused by TRIM37 gene variants characterized by growth failure, dysmorphic features, congestive heart failure (CHF), and an increased risk of Wilms’ tumor. Although immune system impairment has been documented in MUL, the underlying mechanisms remain poorly understood.MethodsWe present a case of MUL with progressive lymphopenia and review similar cases from the literature.ResultsOur patient presented with prenatal onset growth restriction, characteristic dysmorphic features, and Wilms’ tumor. She developed progressive lymphopenia starting at 10 years of age, leading to the initiation of intravenous immunoglobulin (IVIG) replacement therapy and infection prophylaxis. Genetic analysis detected a likely pathogenic variant on the maternal allele and copy number loss on the paternal allele in TRIM37. Subsequently a cardiac magnetic resonance imaging was conducted revealing signs of pericardial constriction raising concerns for intestinal lymphatic losses. The cessation of IVIG therapy did not coincide with any increase in the rate of infections. The patient exhibited a distinct immunological profile, characterized by hypogammaglobulinemia, impaired antibody responses, and skewed T-cell subsets with an altered CD4+/CD8+ ratio, consistent with previous reports. Normal thymocyte development assessed by artificial thymic organoid platform ruled out an early hematopoietic intrinsic defect of T-cell development.DiscussionThe immunological profile of MUL patients reported so far shares similarities with that described in protein-losing enteropathy secondary to CHF in Fontan circulation and primary intestinal lymphangiectasia. These similarities include hypogammaglobulinemia, significant T-cell deficiency with decreased CD4+ and CD8+ counts, altered CD4+/CD8+ ratios, and significantly modified CD4+ and CD8+ T-cell phenotypes toward effector and terminal differentiated T cells, accompanied by a loss of naïve CD45RA+ T lymphocytes. In MUL, CHF is a cardinal feature, occurring in a significant proportion of patients and influencing prognosis. Signs of CHF or constrictive pericarditis have been evident in the case reported here and in all cases of MUL with documented immune dysfunction reported so far. These observations raise intriguing connections between these conditions. However, further investigation is warranted to in-depth define the immunological defect, providing valuable insights into the pathophysiology and treatment strategies for this condition.

Background: The pandemic of coronavirus disease 2019 (COVID-19) was undoubtedly a stressful experience for everyone. General opinion believed that children with acute or chronic illness could experience additional burden, but this is not confirmed. The aim of this study is to understand how children and adolescents already suffering from acute or chronic illness (e.g., cancer, cystic fibrosis, neuropsychiatric disorders) feel about the COVID-19 pandemic, and if the experience is significantly different between these children and children without illness. Methods: Children and adolescents affected by acute or chronic illness (named the “fragile group”) treated at the Regina Margherita Children Hospital in Italy, were enrolled in the study by filling a questionnaire about their pandemic experiences. Also, a group of children and adolescents without acute or chronic illness (named the “low-risk group”) recruited in the emergency department of the hospital, participated in the study in order to compare experiences. Results: The study group was composed of 166 children and adolescents (Median age = 12 yrs; 78% fragile group, 22% low-risk group). Participants experienced a general state of fear of the virus and of a potential infection for both themselves and their families, while feelings and thoughts that interfere with daily functioning were less frequent. The fragile group seems to be more resilient towards the pandemic situation than low-risk group and some differences on the basis of the type of illness were found in the fragile group. Conclusions: Dedicated psychosocial intervention must be proposed in order to support fragile children and adolescents’ well-being during the pandemic, also on the basis of their clinical and mental history.

Simple Summary: Adrenocortical tumors (ACTs) are rare in children and can be benign (adrenocortical adenoma or ACA) or malignant (adrenocortical carcinoma or ACC). Children with Beckwith–Wiedemann spectrum (BWSp) are at an increased risk of developing ACTs, but there is uncertainty around the prognosis, management, and molecular characteristics associated with these tumors. In this study, we provide a literature review of data from 54 published patients with BWSp-ACT and report on one additional new patient, totaling 55 cases: 19 ACA, 33 ACC, and 3 uncertain malignant potential (umACT). Almost half of the ACC patients were clinically diagnosed with BWSp after ACC onset, suggesting that the BWSp clinical diagnostic score has limited value for early diagnosis in this setting. Two patients with metastatic ACC had a low histopathological Wieneke score, confirming limitations of the current histopathological classification, as previously documented. Ultrasound screening failed to identify ACC before metastasis in two cases, indicating a need for new screening strategies for ACTs in children with BWSp. Notably, in some cases, metastatic ACC exhibited unexpectedly indolent behavior, despite their malignancy. Background/Objectives: Adrenocortical tumors (ACTs), including adrenocortical adenoma (ACA) and carcinoma (ACC), represent 0.3–0.4% of pediatric tumors. Beckwith–Wiedemann spectrum (BWSp) confer an increased risk of ACTs, but prognosis, management, and associated molecular characteristics are unclear. Methods: This paper combines a literature review of 54 published cases of BWSp-ACT with a report of one newly identified patient, totaling 55 cases with a confirmed BWSp clinical and/or molecular diagnosis. Results: Nineteen patients with ACA, 33 with ACC, and 3 with ACT of uncertain malignant potential (umACT) were included. Twenty patients had uniparental disomy of chromosome 11p15.5 (patUPD11), 11imprinting Center 2 Loss-of-methylation (IC2-LoM), and had 2 11p15 locus duplication. Eleven patients were diagnosed during cancer screening procedures, including two metastatic at diagnosis ACC. Conclusions: Almost half of ACC patients reached the minimum score for clinical BWSp diagnosis only after ACC onset, suggesting that the BWSp score has limited value for the early diagnosis in such a setting. Two patients with metastatic ACC had a histopathological Wieneke score ≤2, not correlating with clinical malignancy and confirming limitations of the current histopathological classification, as previously documented. Ultrasound screening failed identifying the ACC before metastasis in two cases, indicating an urgent need to develop new strategies for screening of ACTs in BWSp. Furthermore, some cases of metastatic ACC exhibited unexpectedly indolent behavior despite being malignant. [ABSTRACT FROM AUTHOR]

Intestinal graft-versus-host disease (I-GvHD) represents a life-threatening complication in allogeneic stem cell transplantation (SCT). Unfortunately, non-invasive validated diagnostic tools to diagnose I-GvHD, evaluate treatment response, and guide the duration of immunosuppression are still lacking. We employed standard ultrasound and power Doppler to diagnose and follow up on pediatric intestinal GvHD. We herein report on three patients, prospectively evaluated among 24 pediatric patients referred to our center for allogeneic SCT. These three patients presented abdominal pain and diarrhea within the first 200 days after transplantation. In the reported cases, we performed small- and large-intestine ultrasound (US) at clinical onset of lower-intestinal symptoms and, when intestinal GvHD was confirmed, at GvHD flares, if any, and at follow-up. US constantly (3/3 patients) revealed increased bowel wall thickening (BWT) with different bowel segments’ involvement from patient to patient. Further, a moderate or strong increased Doppler signaling was seen in 2 out of 3 patients, according to clinical GVHD staging (e.g., the more the increase, the more the staging). Standard sonography corroborated GvHD diagnosis in all patients considered and was able to detect GvHD progression or complete normalization of findings, thus simplifying ensuing clinical decisions. Our report highlights the need to design clinical trials for the validation of non-invasive radiologic tools for diagnosis and follow-up of GvHD, especially in pediatric patients.

Children and adolescents affected by brain tumors are at risk for neuropsychological sequelae that need to be evaluated in order to plan adequate rehabilitation programs, and to support their development and recovery. This work aims to describe an innovative prospective observational study protocol for the early evaluation and monitoring over time of neuropsychological outcomes in this pediatric population. Pediatric patients aged 3–17 with a brain tumor diagnosis will be assessed through the use of a battery of Italian standardized neuropsychological tests, with good psychometric properties and age-appropiate, at three different time points of their clinical course: at diagnosis and before surgery (T0), after surgical removal and before the start of potential adjuvant therapies (T1), and at the one-year follow-up after potential adjuvant therapies (T2). This study will allow clinicians to support the neuropsychological development of these children by promoting appropriate and timely rehabilitation and educational programs from the early phases of their clinical course.

Since the 1970s, Italian pediatric oncologists have collaborated through the Italian Association for Pediatric Hematology Oncology (AIEOP) network using a common centralized system for the registration of childhood cancer, known as Model 1.01 (Mod. 1.01). In this study, we report on recruitment trends, extra‐regional migration and changes in outcome over time in the Italian population of children (0–14 years) and adolescents (15–19 years) registered and treated within the national AIEOP network in the period between 1989 and 2017. In almost 30 years, a cohort of 43,564 patients with a neoplasia diagnosis was registered in Mod. 1.01. The analysis of national extra‐regional migration showed that patients tend to migrate from the South to the North and, to a lesser extent, to the Center of the country. During the study period, migration apparently decreased, especially for lymphohematopoietic diseases, whereas it remained substantial for solid tumors. Our data showed a progressive and significant increase in the cumulative survival 5 years after diagnosis since the 1990s, reaching almost 84% for all patients diagnosed in the last decade. Survival rates of Mod. 1.01 patients are similar to those provided by the main national and international reports showing childhood cancer surveillance estimates. The AIEOP Mod 1.01 has proved to be an invaluable tool from both an epidemiological and a health policy point of view, allowing us, in this study, to examine the survival experience of the largest cohort of Italian pediatric cancer patients with a very long follow‐up. [ABSTRACT FROM AUTHOR]

Timely detection of congenital anomalies using ultrasound improves neonatal care. As specialist sonographers are often geographically dispersed, they are sometimes requested to provide a second opinion via tele-expertise. The present study aimed to evaluate the economic impact of asynchronous tele-expertise in obstetric ultrasound care in private medical practice through a comparison with face-to-face consultations. We conducted a cost minimization analysis using decision tree modeling in order to determine whether asynchronous tele-expertise or face-to-face consultation had the lowest cost, under the assumption of equivalent effectiveness in terms of prenatal diagnosis. Costs were measured from the societal perspective. The data for the base case of our modeling came from a retrospective analysis of the clinical practice of an expert who had been conducting asynchronous tele-expertise for 4 years in France. The study included 260 patients for whom 322 requests for expert opinions were made by physicians/midwives from January 2016 to January 2020. The expected average total cost for tele-expertise for a patient was €74.45 (95% CI: €66.36–€82.54) compared to €195.02 (95% CI: €183.90–€206.14) for the conventional face-to-face strategy. Accordingly, using tele-expertise led to a statistically significant reduction of €120.57 in the average total cost per patient. A sensitivity analysis confirmed the robustness of the model produced. The results of the present study underline the efficiency of tele-expertise and highlight related economic benefits. Accordingly, they could inform public health policy on the dissemination of tele-expertise in the field of obstetric ultrasound care. [ABSTRACT FROM AUTHOR]

ObjectiveThe objective of this study was to assess the potential of the use of asynchronous tele-expertise (ASTE) to provide prenatal diagnosis from a medical and economic point of view.PopulationPatients screened by a midwife at a primary center.MethodsA technical and clinical evaluation was conducted retrospectively, and a cost minimization study compared asynchronous tele-expertise to face-to-face consultations that would have been performed without ASTE.Main outcome measuresIn our study we assessed the feasibility of ASTE, what were the origins of the requests for expertise, whether patients need to be moved and the reasons for doing so, and the costs of tele-expertise and conventional consultation.ResultsIn this retrospective analysis 322 advices from 260 patients were interpreted remotely via a platform. The results revealed a 90.68% feasibility of transmitting in a satisfactory and interpretable way ultrasound images and videos via the tele-expertise platform (292/322 files). In our series, asynchronous analysis allowed the required physician to make an accurate diagnosis and identify 74 (28.5%, 95% CI [23% -33.9%]) pregnancies associated with malformations and rule out abnormalities in 186 (71.5%, 95% CI [66.1% -77%]) of the cases. The ASTE was not associated with face-to-face consultations for 72.7% (189/260) of the patients, who without moving, were able to have access to a precise diagnosis by ruling out the presence of anomalies in 163/189 of these patients and confirming them in 26/189 patients. The practice of ASTE would result from a societal point of view, an average saving of 61.8% (€ 120.57) per patient compared to a face-to-face consultation.ConclusionThe use of asynchronous tele-expertise (ASTE) using fetal ultrasound, is feasible and may contribute to increased diagnostic accuracy while generating a significant reduction in costs for society.

Objective The objective of this study was to assess the potential of the use of asynchronous tele-expertise (ASTE) to provide prenatal diagnosis from a medical and economic point of view. Population Patients screened by a midwife at a primary center. Methods A technical and clinical evaluation was conducted retrospectively, and a cost minimization study compared asynchronous tele-expertise to face-to-face consultations that would have been performed without ASTE. Main outcome measures In our study we assessed the feasibility of ASTE, what were the origins of the requests for expertise, whether patients need to be moved and the reasons for doing so, and the costs of tele-expertise and conventional consultation. Results In this retrospective analysis 322 advices from 260 patients were interpreted remotely via a platform. The results revealed a 90.68% feasibility of transmitting in a satisfactory and interpretable way ultrasound images and videos via the tele-expertise platform (292/322 files). In our series, asynchronous analysis allowed the required physician to make an accurate diagnosis and identify 74 (28.5%, 95% CI [23% –33.9%]) pregnancies associated with malformations and rule out abnormalities in 186 (71.5%, 95% CI [66.1% –77%]) of the cases. The ASTE was not associated with face-to-face consultations for 72.7% (189/260) of the patients, who without moving, were able to have access to a precise diagnosis by ruling out the presence of anomalies in 163/189 of these patients and confirming them in 26/189 patients. The practice of ASTE would result from a societal point of view, an average saving of 61.8% (€ 120.57) per patient compared to a face-to-face consultation. Conclusion The use of asynchronous tele-expertise (ASTE) using fetal ultrasound, is feasible and may contribute to increased diagnostic accuracy while generating a significant reduction in costs for society.

The restrictive measures adopted worldwide against SARS-CoV-2 produced a drastic reduction in respiratory pathogens, including RSV, but a dramatic rebound was thereafter reported. In this multicenter retrospective observational study in 15 Pediatric Emergency Departments, all children

Introduction: This study examined the efficacy of eye movement desensitisation and reprocessing (EMDR) therapy compared with standard psychotherapy (SP) in treating post-traumatic stress disorder (PTSD) in paediatric oncology patients and their families in the early stage of cancer treatment. The secondary aim of this study was to assess whether EMDR therapy has a different impact on posttraumatic growth compared to SP. Methods: Forty patients were randomly assigned to EMDR or SP groups. The Impact of Event Scale - Revised (IES-R) and the Distress Thermometer (DT) were used to assess PTSD symptoms at pre-treatment (at cancer diagnosis) and in the posttreatment stages (after 8 sessions). The Post-traumatic Growth Inventory-PTGI was administered in the post-treatment stage in order to evaluate positive changes. Results: Both EMDR and SP are effective in reducing PTSD, but EMDR was significantly more effective than the SP in reducing scores on the IES-R, especially regarding the intrusive symptom subscale. Also, in the EMDR group there were higher scores of PTGI than in the standard group. Conclusion: EMDR thus represents a promising treatment in the paediatric psycho-oncology setting. [ABSTRACT FROM AUTHOR]

Objectives: Pediatric healthcare professionals (HCPs) working in a palliative setting may experience challenges during their clinical practice in addressing the complex end-of-life phase of children and their families. Nurses, especially, have a frontline role in providing assistance, thereby becoming at risk of physical and psychological burden. Pediatric psychologists have an ethical responsibility to help colleagues by proposing self-care interventions that will improve their well-being and, indirectly, the work climate. This study investigated the impact of a complementary therapy, delivered by a pediatric psychologist and a nurse, on physical and psychological variables among nurses at the Paediatric Hospice of the Regina Margherita Children's Hospital in Italy. Methods: Thirty-five nurses participated in 5 weeks of Reiki sessions for an overall total of 175 sessions. The effect of the sessions was analyzed through a paired t -test analysis comparing the values of heart rate, oxygen saturation, and systolic and diastolic pressure collected before and after each session. The same test was conducted comparing the values of the 3 burnout subscales for each of the 35 nurses collected before the beginning of the first session with those collected at the end of the last session 2 months later. Results: Results underlined a positive short-term effect with a significant decrease in heart rate before and after each session (t = 11.5, p <.001) and in systolic pressure (t = 2, p <.05). In addition, a decrease in emotional exhaustion symptoms was found (t = 2.3, p <.05) at the end of the intervention. Significance of results: Reiki could be a valid strategy to complement traditional pediatric psychology clinical practice designed to protect HCPs from emotional and physical demands and to create a more supportive workplace for staff and patients alike. [ABSTRACT FROM AUTHOR]

The Bosniak classification of renal cysts aims to provide a probabilistic risk assessment indicating the likelihood of malignancy from imaging findings. Originally designed to classify adult renal cysts based on computed tomography findings, the Bosniak classification has been extended to pediatric patients, with some adjustments made with the aim of accommodating magnetic resonance imaging (MRI) and ultrasonography (US). Bosniak IV lesions are rare in adolescents, indicating localized renal cell carcinoma and requiring surgical intervention. In contrast, Bosniak III lesions can be treated conservatively, although there is a lack of specific guidelines on their management. We present a case of a 14-year-old boy with a Bosniak III lesion, which was incidentally detected during the US evaluation of a left varicocele. After a 12-month follow-up, MRI revealed progression to a Bosniak IV cyst. Robot-assisted tumor enucleation was performed with selective artery clamping when the patient was 15. Histopathology showed tubulocystic renal cell carcinoma without adverse features. Immunocytochemistry supported a favorable prognosis of this rare tumor (<1% of renal tumor), thus obviating the need for adjuvant treatment. At the 18-month follow-up, no recurrence or distant metastasis were observed. This case highlights the importance of an aggressive treatment in persistent Bosniak III and Bosniak IV renal cysts in children and adolescents and the necessity to offer a nephron-sparing surgery. [ABSTRACT FROM AUTHOR]