Obstruction and thrombosis of major systemic veins can occur due to indwelling central venous catheters. If obstruction of the innominate vein or superior vena cava occurs, lymphatic drainage can be impaired due to an increase in pressure in the thoracic duct and lymphatics. We describe a case where superior vena cava syndrome, chylopericardium and chylothorax occurred in a 16-year-old girl due to an indwelling central venous catheter. This was successfully treated with removal of the line, anticoagulation and angioplasty of the innominate vein and superior vena cava.
Background Atypical teratoid/rhabdoid tumors (AT/RTs) are rare aggressive central nervous system tumors. The use of radiation therapy (RT) remains controversial, especially for patients younger than three years of age. The purpose of the current investigation is to robustly analyze the impact of RT among pediatric AT/RT patients using the Surveillance, Epidemiology, and End Results (SEER) database. Methods SEER 18 Custom Data registries were queried for AT/RT (ICD‐0‐3 9508/3). A total of 190 pediatric AT/RT patients were identified, of whom 102 underwent surgery + chemotherapy and 88 underwent trimodality therapy. Univariate and multivariable analyses using Kaplan‐Meier and Cox proportional hazards regression modeling were performed. Propensity‐score matched analysis with inverse probability of treatment weighting was performed to account for indication bias. The landmark method was used to account for immortal time bias. Results The majority of patients were <3 years old (75.8%). Patients <3 were more likely to be treated without RT as compared with older patients (62% vs 38%). Doubly robust MVA identified distant disease as a negative prognostic factor (HR 2.1, P = 0.003), whereas trimodality therapy was strongly protective (HR 0.39, P < 0.001). Infants (<1), toddlers (1‐2), and older children (3+) all benefited from trimodality therapy, with largest benefit for infants (HR 0.34, P = 0.02) and toddlers (HR 0.31, P < 0.001). Conclusion The current study provides further evidence that trimodality therapy improves clinical outcomes among patients with AT/RT. This finding was most pronounced for younger patients; therefore, further studies are needed to confirm this finding in this vulnerable population.
ImportanceAcute lymphoblastic leukemia (ALL) is the most common form of pediatric cancer, and a leading cause of death in children. Understanding the causes of pediatric ALL is necessary to enable early detection and prevention; congenital cytomegalovirus (cCMV) has recently been identified as a potential moderate-to-strong factor associated with risk for ALL.ObjectiveTo compare the prevalence of cCMV infection between ALL cases and matched controls.Design, Setting, and ParticipantsIn this population-based case-control study of ALL cases and matched controls, cases consisted of children aged 0 to 14 years between 1987 and 2014 with an ALL diagnosis identified through the Michigan Cancer Surveillance Program and born in Michigan on or after October 1, 1987. Cancer-free controls were identified by the Michigan BioTrust for Health and matched on age, sex, and mother’s race and ethnicity. Data were analyzed from November to May 2022.ExposurescCMV infection measured by quantitative polymerase chain reaction in newborn dried blood spots.Main Outcomes and MeasuresALL diagnosed in children aged 0 to 14 years.ResultsA total of 1189 ALL cases and 4756 matched controls were included in the study. Bloodspots were collected from participants at birth, and 3425 (57.6%) participants were male. cCMV was detected in 6 ALL cases (0.5%) and 21 controls (0.4%). There was no difference in the odds of cCMV infection comparing ALL cases with controls (odds ratio, 1.30; 95% CI, 0.52-3.24). Immunophenotype was available for 536 cases (45.1%) and cytogenetic data for 127 (27%). When stratified by subtype characteristics, hyperdiploid ALL (74 cases) was associated with 6.26 times greater odds of cCMV infection compared with unmatched controls (95% CI, 1.44-27.19).Conclusions and RelevanceIn this case-control study of cCMV and pediatric ALL, cCMV was associated with increased risk of hyperdiploid ALL. These findings encourage continued research.
Background: We have recently controlled intraoperative condylar seating, with adjustable holes such plates as a sliding plate or MOJ plate. However, even when using such plates, postoperative passive condylar seating cannot be done. Objectives: The purpose of the present study was to evaluate the safety and efficacy associated with mandibular advancement by intraoral vertical ramus osteotomy (IVRO) with endoscopicallyassisted intraoral rigid or semi-rigid internal fixation. Methods: The study sample included all patients who had undergone a mandibular advancement by IVRO procedure with endoscopically-assisted intraoral plate fixation from September 2008 to May 2012. A mandibular advancement by IVRO with endoscopically assisted intraoral rigid or semi-rigid internal fixation was used for mandibular advancement. The patients were analysed prospectively, with more than two years of follow-up, and were evaluated in terms of functional results, postoperative complications, and skeletal stability. Findings: A total of 14 patients (bilateral, seven patients with class II; unilateral, seven patients with asymmetry) were included in the present study. The average degree of mandibular advancement was 5.5 ± 1.9 mm (range, 3-9 mm). Both the occlusal relationship and facial appearance in all patients were significantly improved by the surgical-orthodontic treatment, with no major harmful clinical symptoms. In addition, one-screw semi-rigid fixation could control postoperative passive condylar seating. Conclusion: This study showed that mandibular advancement by IVRO with endoscopically assisted, intraoral semi-rigid internal fixation offers a promising treatment alternative for patients with skeletal class II malocclusion or facial asymmetry.
Atypical teratoid/rhabdoid tumor (AT/RT) is a malignant tumor that is commonly associated with biallelic alterations of SMARCB1. Recurrent or refractory AT/RT has not been molecularly characterized as well. We present the case of a child with recurrent AT/RT who underwent clinically integrated molecular profiling (germline DNA and tumor DNA/RNA sequencing). This demonstrated a somatic lesion in CDKN1C alongside hallmark loss of SMARCB1. This data allowed us to explore potential personalized therapies for this patient and expose a molecular driver that may be involved in similar cases.
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