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Background: Current guidelines recommend different postpartum approaches for patients started on levothyroxine (LT4) during pregnancy. Objective: We studied the postpartum management of these patients and determined factors associated with long-term hypothyroidism. Methods: A retrospective study was conducted at a tertiary center between 2014 and 2020, with LT4 initiation according to 2014 ETA recommendations. We performed multivariate logistic regression (MVR) and a receiver operating characteristic curve analysis to determine variables associated with long-term hypothyroidism and their optimal cutoffs. Results: LT4 was initiated in 177 pregnant women, and 106/177 (60%) were followed at long-term (at least 6 months post partum) (28.5 (9.0–81.9) months). LT4 could have been stopped in 45% of patients who continued it immediately after delivery. Thirty-six out of 106 (34%) patients were long-term hypothyroid. In them, LT4 was initiated earlier during pregnancy than in euthyroid women (11.7 ± 4.7 vs 13.7 ± 6.5 weeks, P = 0.077), at a higher thyroid-stimulating hormone (TSH) level (4.1 (2.2–10.1) vs 3.5 (0.9–6.9) mU/L, P = 0.005), and reached a higher dose during pregnancy (62.8 ± 22.2 vs 50.7 ± 13.9 μg/day, P = 0.005). In the MVR, only the maximal LT4 dose during pregnancy was associated with long-term hypothyroidism (odds ratio (OR) = 1.03, 95% CI: 1.00–1.05, P = 0.003). The optimal cutoffs for predicting long-term hypothyroidism were an LT4 dose of 68.75 μg/day (87% specificity, 42% sensitivity; P = 0.013) and a TSH level ≥ 3.8 mU/L (68.5% specificity, 77% sensitivity; P = 0.019). Conclusion: One-third of the patients who started on LT4 during pregnancy had long-term hypothyroidism. The TSH level at treatment initiation and the LT4 dose during pregnancy could guide the decision for continuing long-term LT4.

Background: Patients with Cushing syndrome (CS) are at increased risk of venous thromboembolism (VTE). Objective: The aim was to evaluate the current management of new cases of CS with a focus on VTE and thromboprophylaxis. Design and methods: A survey was conducted within those that report in the electronic reporting tool (e-REC) of the European Registries for Rare Endocrine Conditions (EuRRECa) and the involved main thematic groups (MTG’s) of the European Reference Networks for Rare Endocrine Disorders (Endo-ERN) on new patients with CS from January 2021 to July 2022. Results: Of 222 patients (mean age 44 years, 165 females), 141 patients had Cushing disease (64%), 69 adrenal CS (31%), and 12 patients with ectopic CS (5.4%). The mean follow-up period post-CS diagnosis was 15 months (range 3–30). Cortisol-lowering medications were initiated in 38% of patients. One hundred fifty-four patients (69%) received thromboprophylaxis (including patients on chronic anticoagulant treatment), of which low-molecular-weight heparins were used in 96% of cases. VTE was reported in six patients (2.7%), of which one was fatal: two long before CS diagnosis, two between diagnosis and surgery, and two postoperatively. Three patients were using thromboprophylaxis at time of the VTE diagnosis. The incidence rate of VTE in patients after Cushing syndrome diagnosis in our study cohort was 14.6 (95% CI 5.5; 38.6) per 1000 person-years. Conclusion: Thirty percent of patients with CS did not receive preoperative thromboprophylaxis during their active disease stage, and half of the VTE cases even occurred during this stage despite thromboprophylaxis. Prospective trials to establish the optimal thromboprophylaxis strategy in CS patients are highly needed. Significance statement The incidence rate of venous thromboembolism in our study cohort was 14.6 (95% CI 5.5; 38.6) per 1000 person-years. Notably, this survey showed that there is great heterogeneity regarding time of initiation and duration of thromboprophylaxis in expert centers throughout Europe.

Background: Newcomer families with child welfare involvement face complex COVID-19 related challenges that are still less understood within the Canadian context. Objective: This study explored views on the changes in child safety reporting and interventions with newcomer families during the COVID-19 pandemic. Participants: and Setting: Participants were 63 child welfare workers from Children's Aid Societies across Ontario, Canada. Methods: Using cross tabulations with Fisher exact tests, the analysis draws on survey data from the second wave of the pandemic to test for significance of differences in areas of child safety reporting, interventions with newcomer families, and available supports across urban and urban/rural settings. Findings: Parents' emotional instability and substance use (86%), domestic/adult conflict (85.6%), and child emotional harm (66.3%) were the top child safety issues with most increased reporting during the pandemic. Except for domestic/adult conflict, there were no differences in the rating of the six identified areas of child abuse across newcomer/racialized caseloads in different geographical settings. Increasingly, during the COVID-19 pandemic, workers were intervening with newcomer families by ‘connecting them with community agencies or services’ (51.8%), and ‘reaching out to extended family and other community members to support parents’ (44.6%). Mental health supports (68.4%), family access (66.7%), and childcare (65.9%) were the least accessible services. Conclusion: A closer examination of the reported child safety incidents shows that child emotional harm was on the rise and that child sexual and physical abuse were underreported. Unidentified cases of child abuse during the pandemic posed barriers to help-seeking and resulted in limited or no supports for victims.

BackgroundChronic pulmonary embolism (PE) may result in pulmonary hypertension (CTEPH). Automated CT pulmonary angiography (CTPA) interpretation using artificial intelligence (AI) tools has the potential for improving diagnostic accuracy, reducing delays to diagnosis and yielding novel information of clinical value in CTEPH. This systematic review aimed to identify and appraise existing studies presenting AI tools for CTPA in the context of chronic PE and CTEPH.MethodsMEDLINE and EMBASE databases were searched on 11 September 2023. Journal publications presenting AI tools for CTPA in patients with chronic PE or CTEPH were eligible for inclusion. Information about model design, training and testing was extracted. Study quality was assessed using compliance with the Checklist for Artificial Intelligence in Medical Imaging (CLAIM).ResultsFive studies were eligible for inclusion, all of which presented deep learning AI models to evaluate PE. First study evaluated the lung parenchymal changes in chronic PE and two studies used an AI model to classify PE, with none directly assessing the pulmonary arteries. In addition, a separate study developed a CNN tool to distinguish chronic PE using 2D maximum intensity projection reconstructions. While another study assessed a novel automated approach to quantify hypoperfusion to help in the severity assessment of CTEPH. While descriptions of model design and training were reliable, descriptions of the datasets used in training and testing were more inconsistent.ConclusionIn contrast to AI tools for evaluation of acute PE, there has been limited investigation of AI-based approaches to characterising chronic PE and CTEPH on CTPA. Existing studies are limited by inconsistent reporting of the data used to train and test their models. This systematic review highlights an area of potential expansion for the field of AI in medical image interpretation.There is limited knowledge of A systematic review of artificial intelligence tools for chronic pulmonary embolism in CT. This systematic review provides an assessment on research that examined deep learning algorithms in detecting CTEPH on CTPA images, the number of studies assessing the utility of deep learning on CTPA in CTEPH was unclear and should be highlighted.

Background: Thyroperoxidase (TPOAb) and thyroglobulin (TgAb) antibodies are highly prevalent in Graves’ disease (GD), but their significance is con troversial. Methods: We retrospectively analyzed TPOAb and TgAb levels and evolution in 136 patients with newly diagnosed GD between 2000 and 2022, treated with anti-thyroid drugs (ATD) in a block-and-replace (B+R) regimen for at least 12 months and followed up for at least 1 year after ATD discontinuation or until disease relapse. Results: At diagnosis, 98 out of 136 (72%) patients were TPOAb positive and 73 out of 136 (54%) patients were TgAb positive. The presence of TPOAb or TgAb antibodies at diagnosis was generally not related to GD presentation and did not influence the risk of relapse (P = 0.304 and P = 0.348, respectively). There was less TED (thyroid eye disease) in TgAb-positive patients than TgAb-negative patients at diagno sis (11 out of 73 (15.1%) versus 21 out of 63 (33.3%) P = 0.012). In contrast, the presence of TPOAb at diagnosis was not associated with TED (P = 0.354). The absence of TgAb at diagnosis (P = 0.05) and time to euthyroidism (P = 0.009), but not smoking or TRAb levels, were associated with TED in multivariate logistic regression. TPOAb and TgAb levels during treatment and after its discontinuation were not predictive of relapse, except for lower titers of TgAb at 18 months in patients who relapsed (P = 0.034). Conclusion: In GD patients treated with a first course of ATD in a B+R regi men we observed lower titers of TgAb at the end of treatment in patients who relapsed and a significant protection against TED in patients with positi ve TgAb at diagnosis, irrespectively of TPOAb.

Objectives: The incidental diagnosis of nonfunctioning pituitary macroadenomas (NFPMAs) is becoming more prevalent with the spread of modern brain imaging techniques. We sought to uncover new data about their natural history and surgical outcome. Design: This is a retrospective single-center observational study. Methods: Among 210 patients seen for a NFPMA between 2010 and 2019, 70 ( 33%) were discovered incidentally (i-NFPMA). We analyzed outcomes in a total of 65 patients with available follow-up data. Results: Mean age at diagnosis (± s.d.) was 60 ± 14 years and mean maximal diameter was 20.0 ± 7.3 mm. At diagnosis, 29 patients (45%) had pituitary hormone deficits (LH/FSH 41%, TSH 29%, ACTH 15%) and 12% had visual field deficits. 26 patients underwent initial surgery, while 12 had delayed surgery after initial surveillance. In the surveillance group, the risk of tumor growth was estimated at 10%/year. Patients with hormonal deficits at diagnosis experienced earlier growth at 24 months (P < 0.02). Overall, surgical resection of the i-NFPMA led to stable or improved endocrine function in 91% of patients, with only 6% postoperative permanent diabetes insipidus. Moreover, surgery was more effective in preserving intact endocrine function (10/12) than restoring altered endocrine function to normal (6/22, P = 0.03). Conclusion: About one-third of NFPMAs are now discovered incidentally and a significant subset may be responsible for unrecognized endocrine and visual deficits. Under surveillance the risk of further tumor growth is significant (10 %/year) and seems to occur faster in patients already harboring an endocrine deficit. Early surgical removal before onset of endocrine deficits appears to lead to better endocrine outcome.

This qualitative study explores the experiences of child welfare workers during the COVID-19 pandemic through virtual interviews, focusing on the challenges and adaptations in their work and support systems. Participants reported significant difficulties in maintaining a healthy work–life balance, heightened stress, anxiety, and increased workloads due to sick leaves and burnout. This study highlights the dual role of technology as both a stressor and a crucial tool, with rapid integration posing challenges while also enabling continued support for children and families. Despite these challenges, workers demonstrated resilience and creativity, developing innovative solutions to navigate the new landscape. The findings underscore the importance of robust support systems, clear communication, and equitable access to technology. This study suggests integrating lessons learned during the pandemic into future child welfare practices to enhance resilience and adaptability in the face of future crises.

IntroductionCardiac magnetic resonance (CMR) is of diagnostic and prognostic value in a range of cardiopulmonary conditions. Current methods for evaluating CMR studies are laborious and time-consuming, contributing to delays for patients. As the demand for CMR increases, there is a growing need to automate this process. The application of artificial intelligence (AI) to CMR is promising, but the evaluation of these tools in clinical practice has been limited. This study assessed the clinical viability of an automatic tool for measuring cardiac volumes on CMR.MethodsConsecutive patients who underwent CMR for any indication between January 2022 and October 2022 at a single tertiary centre were included prospectively. For each case, short-axis CMR images were segmented by the AI tool and manually to yield volume, mass and ejection fraction measurements for both ventricles. Automated and manual measurements were compared for agreement and the quality of the automated contours was assessed visually by cardiac radiologists.Results462 CMR studies were included. No statistically significant difference was demonstrated between any automated and manual measurements (p > 0.05; independent T-test). Intraclass correlation coefficient and Bland-Altman analysis showed excellent agreement across all metrics (ICC > 0.85). The automated contours were evaluated visually in 251 cases, with agreement or minor disagreement in 229 cases (91.2%) and failed segmentation in only a single case (0.4%). The AI tool was able to provide automated contours in under 90 s.ConclusionsAutomated segmentation of both ventricles on CMR by an automatic tool shows excellent agreement with manual segmentation performed by CMR experts in a retrospective real-world clinical cohort. Implementation of the tool could improve the efficiency of CMR reporting and reduce delays between imaging and diagnosis.

This study investigated circumstances surrounding the impact of COVID-19 on child protection investigations, particularly those affecting newcomer parents in Ontario, Canada. Recognizing that the pandemic inflicted substantial socioeconomic disadvantage on some people, the purpose of the study was to use an intersectional lens to examine challenges and solutions found by child welfare agencies when working with families. Insights for policy and practice are drawn from 11 virtual interviews with child welfare workers and managers in Ontario. Our findings reveal that some newcomer families encountered unique challenges: ineligibility for the available pandemic public assistance; inaccessibility to faith-based supports, which had often been their first key contact for mental wellness in the past; technological inequities; and language barriers. These intersecting conditions impacted newcomer families and led to innovative child protection interventions. Analysis of the interview data shows a gradual shift in Ontario from risk-focused approaches to supportive and preventative child welfare interventions in families. Furthermore, supervisors faced the dilemma of how stringently to enforce ongoing safety policies when some social workers were questioning the benefits of these rules for families with intersecting identities who were experiencing added burdens because of the pandemic.

There has been a paradigm shift in patient care with regards to delivering better oral health, towards a team-delivered, person-focused, risk-related model that is known as minimum intervention oral care (MIOC). Student skills should be developed within the undergraduate dental curricula to ensure that graduating dentists and other oral healthcare team members are able to provide phased personalised care plans alongside behavioural management support to patients/caregivers to prevent and manage oral disease in the long term. The purpose of this study is to establish that using an adjunctive caries risk/susceptibility assessment (CRSA) technology (PreViser) had an impact on the behaviour, perception, and knowledge of dental undergraduate students and their clinical teachers, regarding the benefits of such an oral health assessment in the management of patients. Four cohorts of students who did not have exposure to the caries risk susceptibility software were compared with those who did. This study was conducted using a mixed methods approach using a convergent parallel design consisting of collecting quantitative data through questionnaires presented to four cohorts of Year 4 dental students (n = 150 per cohort) and their clinical teachers (n = 10) and qualitative data from structured interviews with students (n = 5) and teachers (n = 7) with suitable statistical analysis and interpretation.ResultsGenerally, the items that exhibited statistical significance, when reviewed, showed better behaviour, perception, and knowledge towards CRSA in the Group C (BDS4-22T1) cohort in comparison with the Group A (BDS3-20T2) cohort. The Group D (BDS4-22T2) students felt more confident using the PreViser as a CRSA tool. When comparing the Group C and Group D data, we note that the students from the Group C cohort were more likely to carry out a diet analysis for their patients and were less likely to be negatively impacted by time constraints compared with the Group D students. Both cohorts were equally confident in using the PreViser for CRSA. From a qualitative perspective, although competence and confidence appeared high, the students and teachers acknowledged that they would need more support to use it chairside. The main barrier listed to using PreViser rested in the fact that clinical teachers either preferred their own ways of assessing or did not know how to use the tool and therefore did not encourage using it. Those who did use PreViser highlighted that it was straightforward to use and was a systematic approach, enabling communication with the patients as there is ‘evidence’ to back up the clinical recommendations.ConclusionThe cumulative benefit of training and use (even limited) had an impact on the students' knowledge, competence, and confidence regarding CRSA, ultimately facilitating the process of teaching and assisting them in effectively implementing CRSA. The importance of CRSA became more evident immediately following the training. Further research is suggested to understand the factors influencing student behaviour, perception, and knowledge regarding CRSA with the aim to make recommendations on a preferable approach and tool to help streamline CRSA education.

BackgroundSegmentation of cardiac structures is an important step in evaluation of the heart on imaging. There has been growing interest in how artificial intelligence (AI) methods—particularly deep learning (DL)—can be used to automate this process. Existing AI approaches to cardiac segmentation have mostly focused on cardiac MRI. This systematic review aimed to appraise the performance and quality of supervised DL tools for the segmentation of cardiac structures on CT.MethodsEmbase and Medline databases were searched to identify related studies from January 1, 2013 to December 4, 2023. Original research studies published in peer-reviewed journals after January 1, 2013 were eligible for inclusion if they presented supervised DL-based tools for the segmentation of cardiac structures and non-coronary great vessels on CT. The data extracted from eligible studies included information about cardiac structure(s) being segmented, study location, DL architectures and reported performance metrics such as the Dice similarity coefficient (DSC). The quality of the included studies was assessed using the Checklist for Artificial Intelligence in Medical Imaging (CLAIM).Results18 studies published after 2020 were included. The DSC scores median achieved for the most commonly segmented structures were left atrium (0.88, IQR 0.83–0.91), left ventricle (0.91, IQR 0.89–0.94), left ventricle myocardium (0.83, IQR 0.82–0.92), right atrium (0.88, IQR 0.83–0.90), right ventricle (0.91, IQR 0.85–0.92), and pulmonary artery (0.92, IQR 0.87–0.93). Compliance of studies with CLAIM was variable. In particular, only 58% of studies showed compliance with dataset description criteria and most of the studies did not test or validate their models on external data (81%).ConclusionSupervised DL has been applied to the segmentation of various cardiac structures on CT. Most showed similar performance as measured by DSC values. Existing studies have been limited by the size and nature of the training datasets, inconsistent descriptions of ground truth annotations and lack of testing in external data or clinical settings.Systematic Review Registration[www.crd.york.ac.uk/prospero/], PROSPERO [CRD42023431113].

Objective: Primary adrenal insufficiency (PAI) is a rare disease with an inc reasing prevalence, which may be complicated by life-threatening adrena l crisis (AC). Good quality epidemiological data remain scarce. We performed a Belg ian survey to describe the aetiology, clinical characteristics, treatment regimens, comorbidities and frequency of AC in PAI. Methods: A nationwide multicentre study involving 10 major university ho spitals in Belgium collected data from adult patients with known PAI. Results: Two hundred patients were included in this survey. The median a ge at diagnosis was 38 years (IQR 25–48) with a higher female prevalence (F/M s ex ratio = 1.53). The median disease duration was 13 years (IQR 7–25). Autoimmune dis ease was the most common aetiology (62.5%) followed by bilateral adrenalectomy (2 3.5%) and genetic variations (8.5%). The majority (96%) of patients were treated with hydrocortisone at a mean daily dose of 24.5 ± 7.0 mg, whereas 87.5% of patient s also received fludrocortisone. About one-third of patients experienced one or more AC over the follow-up period, giving an incidence of 3.2 crises per 100 pat ient-years. There was no association between the incidence of AC and the maintenance dos e of hydrocortisone. As high as 27.5% of patients were hypertensive, 17.5% had diabe tes and 17.5% had a diagnosis of osteoporosis. Conclusion: This study provides the first information on the management of PAI in large clinical centres in Belgium, showing an increased frequency of postsurgical PAI, a nearly normal prevalence of several comorbidities and an overall good quality of care with a low incidence of adrenal crises, compared with data from other regi stries.

Objectives Early identification of lung cancer on chest radiographs improves patient outcomes. Artificial intelligence (AI) tools may increase diagnostic accuracy and streamline this pathway. This study evaluated the performance of commercially available AI-based software trained to identify cancerous lung nodules on chest radiographs.Design This retrospective study included primary care chest radiographs acquired in a UK centre. The software evaluated each radiograph independently and outputs were compared with two reference standards: (1) the radiologist report and (2) the diagnosis of cancer by multidisciplinary team decision. Failure analysis was performed by interrogating the software marker locations on radiographs.Participants 5722 consecutive chest radiographs were included from 5592 patients (median age 59 years, 53.8% women, 1.6% prevalence of cancer).Results Compared with radiologist reports for nodule detection, the software demonstrated sensitivity 54.5% (95% CI 44.2% to 64.4%), specificity 83.2% (82.2% to 84.1%), positive predictive value (PPV) 5.5% (4.6% to 6.6%) and negative predictive value (NPV) 99.0% (98.8% to 99.2%). Compared with cancer diagnosis, the software demonstrated sensitivity 60.9% (50.1% to 70.9%), specificity 83.3% (82.3% to 84.2%), PPV 5.6% (4.8% to 6.6%) and NPV 99.2% (99.0% to 99.4%). Normal or variant anatomy was misidentified as an abnormality in 69.9% of the 943 false positive cases.Conclusions The software demonstrated considerable underperformance in this real-world patient cohort. Failure analysis suggested a lack of generalisability in the training and testing datasets as a potential factor. The low PPV carries the risk of over-investigation and limits the translation of the software to clinical practice. Our findings highlight the importance of training and testing software in representative datasets, with broader implications for the implementation of AI tools in imaging.

Abstract Background Ectopic ACTH pituitary adenomas (EAPA), located outside the sella turcica and deriving from cellular remnants of Rathke’s pouch are a very rare cause of Cushing’s syndrome (CS). The diagnosis is often difficult and delayed, even after comprehensive work-up. To our knowledge, we report for the first time an ectopic corticotroph tumor of the posterior wall of the sphenoid sinus, leading to false positive results of bilateral inferior petrosal sinus sampling (BIPPS) and which was finally localized by a co-registered11 C Methionine PET/MR imaging. Case presentation A 48-year-old woman was referred for a high clinical suspicion of ACTH-dependent CS. Biological testing comprising low dose dexamethasone suppression and CRH stimulation tests were indicative of pituitary Cushing’s disease, but comprehensive pituitary MRI did not reveal any pituitary adenoma. BIPSS confirmed however a central origin of ACTH secretion (central-to-peripheral ACTH ratio > 100) and revealed a significant right-to-left gradient (6.2), leading to a first right-sided exploratory hypophysectomy, that did not cure the patient. BIPSS images were reviewed and revealed preferential drainage of the left pituitary to the right petrosal sinus, leading us to a left sided exploratory hypophysectomy, which was again unsuccessful. A11 C Methionine PET/MRI was performed and revealed a hypermetabolic lesion adjacent to the posterior wall of the sphenoidal sinus. After surgical resection, this polypoid mass was identified as an ectopic ATCH-secreting pituitary adenoma expressing ACTH and T-Pit and complete remission of hypercortisolism was observed. Conclusions In conclusion, we report a case of ACTH-dependent Cushing’s syndrome, caused by an ectopic corticotroph adenoma located in the sphenoidal sinus, which perfectly mimicked the biological features of a classical pituitary ACTH adenoma on a comprehensive hormonal evaluation including BIPPS, and the features of a benign naso-sinusal polyp at MRI. We report for the first time a key role of11 C Methionine PET co-registered to high resolution MRI for localizing ectopic adenomas, efficiently guiding surgical removal and leading to complete remission of hypercortisolism.

Abstract Background Measurement of peak velocities is important in the evaluation of heart failure. This study compared the performance of automated 4D flow cardiac MRI (CMR) with traditional transthoracic Doppler echocardiography (TTE) for the measurement of mitral inflow peak diastolic velocities. Methods Patients with Doppler echocardiography and 4D flow cardiac magnetic resonance data were included retrospectively. An established automated technique was used to segment the left ventricular transvalvular flow using short-axis cine stack of images. Peak mitral E-wave and peak mitral A-wave velocities were automatically derived using in-plane velocity maps of transvalvular flow. Additionally, we checked the agreement between peak mitral E-wave velocity derived by 4D flow CMR and Doppler echocardiography in patients with sinus rhythm and atrial fibrillation (AF) separately. Results Forty-eight patients were included (median age 69 years, IQR 63 to 76; 46% female). Data were split into three groups according to heart rhythm. The median peak E-wave mitral inflow velocity by automated 4D flow CMR was comparable with Doppler echocardiography in all patients (0.90 ± 0.43 m/s vs 0.94 ± 0.48 m/s, P = 0.132), sinus rhythm-only group (0.88 ± 0.35 m/s vs 0.86 ± 0.38 m/s, P = 0.54) and in AF-only group (1.33 ± 0.56 m/s vs 1.18 ± 0.47 m/s, P = 0.06). Peak A-wave mitral inflow velocity results had no significant difference between Doppler TTE and automated 4D flow CMR (0.81 ± 0.44 m/s vs 0.81 ± 0.53 m/s, P = 0.09) in all patients and sinus rhythm-only groups. Automated 4D flow CMR showed a significant correlation with TTE for measurement of peak E-wave in all patients group (r = 0.73, P

A 52-year-old female presented with recurrent episodes of fasting or post-absorptive hypoglycemia. A 72-h fasting test confirmed endogenous hyperinsulinemia. Conventional imaging was unremarkable. Selective pancreatic arterial calcium stimulation and hepatic venous sampling showed a maximum calcium-stimulated insulin concentration from several pancreatic areas, mainly the proximal splenic artery and the proximal gastroduodenal artery, suggesting the presence of one or more occult insulinoma(s) in the region of the pancreatic body. 68Ga-DOTA-exendin-4 PET/CT showed however generalized increased uptake in the pancreas and a diagnosis of nesidioblastosis was therefore suspected. The patient has been since successfully treated with dietetic measures and diazoxide. Treatment efficacy was confirmed by a flash glucose monitoring system with a follow-up of 7 months.

IntroductionVon Hippel Lindau (VHL) syndrome is caused by an autosomal dominant hereditary or sporadic germline mutation of the VHL gene with more than five hundred pathogenic mutations identified. Pheochromocytomas and rarely paragangliomas occur in 10-50% of patients with VHL syndrome usually around 30 years of age and exceptionally before the age of 10.Case presentationWe diagnosed a 9-year-old girl of normal appearance and severe refractory hypertension, with a norepinephrine-secreting pheochromocytoma related to VHL syndrome due to a known familial germline heterozygous mutation of VHL gene (c.414A>G), also present in three members of her family. At age 13, a pelvic tumor and a left adrenal pheochromocytoma that showed to be multi-metastatic to both lungs were discovered in the patient leading to left adrenalectomy and pelvic tumor resection. In addition to the germline VHL gene mutation, blood analysis using Next Generation Sequencing identified a novel heterozygous germline mutation of the KIF1B gene (c.3331_3332del; p.Asn1111Glnfs*21), which is only present in the girl and not the other family members. The patient is currently under steroid substitution therapy and leads a normal life.DiscussionThis family is notable by the early age of onset of multiple neural crest tumors associated with a high propensity for malignancy and metastatic spread. Most reports in the literature associated the VHL mutation with a later onset in adulthood and a benign course, which contrast with our findings and question the role of this mutation in the phenotype expressed in this kindred. Also, the presence of concomitant mutations in two susceptibility genes for neural crest tumors poses the question of their respective roles in the development of tumors in this family. Our familial case description illustrates the potential for systematic use of targeted Next Generation Sequencing with multi-gene panels in patients with neural crest tumors to confirm the role of known susceptibility genes as well as identifying new ones, but also to contribute to comprehensive databases on gene variants and their phenotypic counterparts in this specific area of medicine.

Complicated Rathke’s cleft cyst (RCC) is a rare occurrence of symptomatic bleeding or growth of a previously asymptomatic (and often undiagnosed) intrasellar cyst derived from remnants of Rathke’s pouch, situated on the midline between the adeno- and neurohypophysis. Symptoms may be identical to those of pituitary apoplexy: acute onset of headache, hypopituitarism, and neurological disturbances. Both syndromes may also exhibit a similar appearance of a large haemorrhagic sellar mass at initial radiological evaluation. We report on two patients who presented with headache and complete hypopituitarism. Based on the initial MRI, they were first diagnosed with pituitary apoplexy but managed conservatively with hormone therapy alone because of the absence of severe visual or neurological threat. Upon follow-up at 4 months, clinical evolution was good in both patients but their pituitary mass had not reduced in size and, after careful radiologic reviewing, was more indicative of a large midline complicated RCC. In conclusion, the diagnosis of complicated RCC is challenging because it can mimic pituitary apoplexy clinically, biologically, and radiologically. Clinicians should distinguish between the two entities using specific radiological signs or evolution of the mass at MRI if the patient does not undergo surgery. To our knowledge, we report conservative management of this rare condition for the first time, though it seems appropriate in the absence of neurological compromise or visual compression. Long-term follow-up is however mandatory.

Objective: In recent years, endoscopic ultrasound‐guided radiofrequency ablation (EUS‐RFA) has emerged as an alternative nonsurgical treatment for pancreatic neuroendocrine tumours. The aim of our study was to assess the long‐term follow‐up of patients treated with EUS‐RFA for a sporadic insulinoma in our centre in terms of efficacy, safety and risk of recurrence. Design, Patients and Measurements: We retrospectively analysed the data of 11 patients with an insulinoma treated by EUS‐RFA in our tertiary centre between June 2018 and April 2022. Clinical and biological, as well as imaging, follow‐up was planned at 3, 6, 12 months and then annually. Results: In our series, there were nine women and two men with a median age of 65 years. All tumours were sporadic, with a mean size of 11 mm. The procedure allowed an immediate and complete symptomatic and biological remission in all patients without notable complications. Complete radiological resolution of the tumour after ablation was observed in seven patients, and persistence of an asymptomatic tumour residue was observed in four patients. During the mean follow‐up period of 26 months, two patients presented a significant but asymptomatic increase of the tumour residue; a second EUS‐RFA session was performed in one patient and the other patient is being closely monitored. Conclusions: EUS‐RFA treatment of benign insulinomas provides a long‐term complete clinical resolution of hypoglycaemia. A long‐term follow‐up is essential if residual tumour persists after initial EUS‐RFA treatment. [ABSTRACT FROM AUTHOR]

Context Current guidelines for distinguishing Cushing's disease (CD) from ectopic ACTH secretion (EAS) are questionable, as they use pituitary magnetic resonance imaging (MRI) as first-line investigation for all patients. CRH testing is no longer available, and they suggest performing inferior petrosal sinus sampling (BIPPS), an invasive and rarely available investigation, in many patients. Objective To establish noninvasive personalized diagnostic strategies based on the probability of EAS estimated from simple baseline parameters. Design Retrospective study. Setting University hospitals. Patients Two hundred forty-seven CD and 36 EAS patients evaluated between 2001 and 2023 in 2 French hospitals. A single-center cohort of 105 Belgian patients served as external validation. Results Twenty-four-hour urinary free cortisol (UFC) had the highest area under the receiver operating characteristic curve for discrimination of CD from EAS (.96 [95% confidence interval (CI),.92-.99] in the primary study and.99 [95% CI,.98-1.00] in the validation cohort). The addition of clinical, imaging, and biochemical parameters did not improve EAS prediction over UFC alone, with only BIPPS showing a modest improvement (C-statistic index.99 [95% CI,.97-1.00]). Three groups were defined based on baseline UFC: < 3 (group 1), 3-10 (group 2), and > 10 × the upper limit of normal (group 3), and they were associated with 0%, 6.1%, and 66.7% prevalence of EAS, respectively. Diagnostic approaches performed in our cohort support the use of pituitary MRI alone in group 1, MRI first followed by neck-to-pelvis computed tomography scan (npCT) when negative in group 2, and npCT first followed by pituitary MRI when negative in group 3. When not combined with the CRH test, the desmopressin test has limited diagnostic value. Conclusion UFC accurately predicts EAS and can serve to define personalized and noninvasive diagnostic algorithms. [ABSTRACT FROM AUTHOR]

Background Pituitary adenomas (PAs)—also now called pituitary neuroendocrine tumours or Pit-NETS—are rare in children and adolescents and exceptional below the age of 10. Most evidence-based high-quality data are derived from larger studies in adult patients. Aims We will review recent knowledge on the epidemiology, clinical features, diagnosis, and treatment modalities of the different types of pituitary adenomas diagnosed in children and adolescents, emphasizing the many reasons why these cases should be discussed within pituitary-specific multidisciplinary teams with experts from both paediatric and adult practice. Conclusions Paediatric PA presents multiple peculiarities that may challenge their adequate management. They are overall proportionally larger and more aggressive than in adults, with potential mass effects including hypopituitarism. Hormonal hypersecretion is frequent, resulting in clinical syndromes affecting normal growth and pubertal development. Prolactinomas represent the most frequent subtype of PA found during childhood, followed by adrenocorticotropin (ACTH) and growth hormone (GH)–secreting adenomas, while clinically non-functioning adenomas are exceptionally diagnosed before the age of 16. The occurrence of a pituitary tumour in a young individual should also prompt genetic testing in each case, searching for either germline mutations in one of the known genes that may drive inherited/familial PA (such as the multiple endocrine neoplasia type 1 or MEN1 gene, or the aryl hydrocarbon receptor interacting protein or AIP gene), or for a mosaic activating mutation of GNAS as found in the McCune-Albright syndrome. [ABSTRACT FROM AUTHOR]

This qualitative study explores the experiences of child welfare workers during the COVID-19 pandemic through virtual interviews, focusing on the challenges and adaptations in their work and support systems. Participants reported significant difficulties in maintaining a healthy work–life balance, heightened stress, anxiety, and increased workloads due to sick leaves and burnout. This study highlights the dual role of technology as both a stressor and a crucial tool, with rapid integration posing challenges while also enabling continued support for children and families. Despite these challenges, workers demonstrated resilience and creativity, developing innovative solutions to navigate the new landscape. The findings underscore the importance of robust support systems, clear communication, and equitable access to technology. This study suggests integrating lessons learned during the pandemic into future child welfare practices to enhance resilience and adaptability in the face of future crises. [ABSTRACT FROM AUTHOR]

Recent years have seen a dramatic increase in studies presenting artificial intelligence (AI) tools for cardiac imaging. Amongst these are AI tools that undertake segmentation of structures on cardiac MRI (CMR), an essential step in obtaining clinically relevant functional information. The quality of reporting of these studies carries significant implications for advancement of the field and the translation of AI tools to clinical practice. We recently undertook a systematic review to evaluate the quality of reporting of studies presenting automated approaches to segmentation in cardiac MRI (Alabed et al. 2022 Quality of reporting in AI cardiac MRI segmentation studies—a systematic review and recommendations for future studies. Frontiers in Cardiovascular Medicine 9:956811). 209 studies were assessed for compliance with the Checklist for AI in Medical Imaging (CLAIM), a framework for reporting. We found variable—and sometimes poor—quality of reporting and identified significant and frequently missing information in publications. Compliance with CLAIM was high for descriptions of models (100%, IQR 80%–100%), but lower than expected for descriptions of study design (71%, IQR 63–86%), datasets used in training and testing (63%, IQR 50%–67%) and model performance (60%, IQR 50%–70%). Here, we present a summary of our key findings, aimed at general readers who may not be experts in AI, and use them as a framework to discuss the factors determining quality of reporting, making recommendations for improving the reporting of research in this field. We aim to assist researchers in presenting their work and readers in their appraisal of evidence. Finally, we emphasise the need for close scrutiny of studies presenting AI tools, even in the face of the excitement surrounding AI in cardiac imaging.

ObjectivesRight ventricle (RV) mass is an imaging biomarker of mean pulmonary artery pressure (MPAP) and pulmonary vascular resistance (PVR). Some methods of RV mass measurement on cardiac MRI (CMR) exclude RV trabeculation. This study assessed the reproducibility of measurement methods and evaluated whether the inclusion of trabeculation in RV mass affects diagnostic accuracy in suspected pulmonary hypertension (PH).Materials and methodsTwo populations were enrolled prospectively. (i) A total of 144 patients with suspected PH who underwent CMR followed by right heart catheterization (RHC). Total RV mass (including trabeculation) and compacted RV mass (excluding trabeculation) were measured on the end-diastolic CMR images using both semi-automated pixel-intensity-based thresholding and manual contouring techniques. (ii) A total of 15 healthy volunteers and 15 patients with known PH. Interobserver agreement and scan-scan reproducibility were evaluated for RV mass measurements using the semi-automated thresholding and manual contouring techniques.ResultsTotal RV mass correlated more strongly with MPAP and PVR (r = 0.59 and 0.63) than compacted RV mass (r = 0.25 and 0.38). Using a diagnostic threshold of MPAP ≥ 25 mmHg, ROC analysis showed better performance for total RV mass (AUC 0.77 and 0.81) compared to compacted RV mass (AUC 0.61 and 0.66) when both parameters were indexed for LV mass. Semi-automated thresholding was twice as fast as manual contouring (p < 0.001).ConclusionUsing a semi-automated thresholding technique, inclusion of trabecular mass and indexing RV mass for LV mass (ventricular mass index), improves the diagnostic accuracy of CMR measurements in suspected PH.