Differentiated Thyroid Cancer in Children and Adolescents: Long Term Outcome and Risk Factors for Persistent Disease.
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ABSTRACT:
SUBMITTER: Sapuppo G
PROVIDER: S-EPMC8345030 | biostudies-literature | 2021 Jul
REPOSITORIES: biostudies-literature
Publications
Differentiated Thyroid Cancer in Children and Adolescents: Long Term Outcome and Risk Factors for Persistent Disease.
Sapuppo Giulia G Hartl Dana D Fresneau Brice B Hadoux Julien J Breuskin Ingrid I Baudin Eric E Rigaud Charlotte C Guerlain Joanne J Al Ghuzlan Abir A Leboulleux Sophie S Schlumberger Martin M Lamartina Livia L
Cancers 20210724 15
<i>Background:</i> Pediatric differentiated thyroid cancer (P-DTC) frequently presents with advanced disease. The study aim was to evaluate the outcome of P-DTC and a modified 2015 American Thyroid Association risk classification (ATA-R). <i>Methods:</i> A retrospective study of consecutive P-DTC patients was performed. The ATA-R for P-DTC was used with a cut-off of ≤ 5 N1a for low-risk. The outcome could be excellent response (ER) (thyroglobulin < 1 ng/mL and no evidence of disease (EoD) at ima ...[more]
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Project description:BackgroundPersistent or recurrent papillary thyroid carcinoma (PTC) occurs in some patients after initial thyroid surgery and often, radioactive iodine treatment. Here, we identify the efficacy, safety, and long-term outcome of our current surgical management paradigm for persistent/recurrent PTC in the central compartment in an interdisciplinary thyroid cancer clinical and research program at a tertiary thyroid cancer referral center.MethodsWe retrospectively analyzed our standardized approach of comprehensive bilateral level VI/VII lymph node dissection (SND [VI, VII]) for cytologically confirmed PTC in the central compartment.ResultsFrom 1994 to 2004, 210 patients, median age 42 (range 12-82) underwent SND (VI, VII). Most patients (106, 51%) had already undergone ≥2 surgical procedures for persistent or recurrent disease, and 31 (15%) had distant metastases at presentation. Postoperatively, 104 (71%) of the 146 patients who were thyroglobulin (Tg) positive had no evidence of disease. Anti-Tg antibodies were present in 38 patients (18%), 17 of whom (53%) did not have anti-Tg antibodies postoperatively. Fourteen patients (7%) were hypoparathyroid at presentation, and 2 more (1%) became permanently hypoparathyroid after surgery. Four patients (2%) experienced recurrent laryngeal nerve paralysis (RLNP) of a previously functioning nerve. Unanticipated RLNP was observed in only one nerve at risk. External beam radiation was given to 33 patients (17%). An additional 17 patients (8%) developed distant metastases during follow-up. At the last follow-up, 130 (66%) of the 196 patients had no detectable Tg; of these, 99 (76%) had no further evidence of disease. A median of 7.25 years after surgery, 167 (90%) of the 185 patients were without evidence of central disease, and 18 (10%) had developed central compartment recurrences within a median interval of 24.3 months. Of those with recurrence, 16 out of 18 patients (89%) underwent a subsequent surgical procedure, thus resulting in an overall 98% central compartment control rate. Kaplan-Meier disease-specific survival at 10 years was 98.9% for patients <45 years old and 77.9% for those ≥45 years old (log-rank p<0.00001). The only predictor of central compartment recurrence was malignancy in a thyroid remnant noted within the central compartment surgical specimen.ConclusionsBilateral comprehensive level VI/VII dissections are safe and effective for long-term control of recurrent/persistent PTC in the central lymphatic compartment.
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Project description:We report here the results of the prospective, non-randomized, historically controlled CWS-2002P study in patients ≤ 21 years with localized RMS developed with the aim to improve the long-term outcome by adapting the burden of therapy to risk profile and to investigate the feasibility and relation to the outcome of maintenance therapy (MT) in the high-risk groups. Patients were allocated into low-risk (LR), standard-risk (SR), high-risk (HR), and very high-risk (VHR) groups. Chemotherapy consisted of vincristine (VCR) and dactinomycin (ACTO-D) for all patients with the addition of ifosfamide (IFO) in the SR, HR, and VHR and doxorubicin (DOX) in the HR and VHR groups. Low-dose cyclophosphamide and vinblastine maintenance therapy (MT) over 6 months was recommended in the HR and VHR groups. A total of 444 patients have been included in this analysis. With a median follow-up of 9·6 years (IQR 7·6-10·9) for patients alive, the 5-year EFS and OS for the whole group was 73% (95% CI 69-77) and 80% (95% CI 76-84), respectively. The 5-year EFS by risk group was 100% in the LR, 79% (95% CI 72-84) in the SR, 69% (95% CI 63-75) in the HR, and 42% (95% CI 23-61) in the VHR (log-rank p = 0.000). The 5-year EFS was 77% (95% CI 70-84) for 155 patients in the HR group who received MT as compared to 63% (95% CI 50-76) for 49 patients who did not (log-rank p = 0.015). Neither the reduction in the IFO dose in the SR nor the increased dose intensity of DOX in HR groups influenced the outcome when compared to the previous CWS and other European studies. MT was feasible, seemed to have an impact on prognosis, and should be studied in a well-controlled prospective trial in this patient population. The weighting of risk factors used for therapy stratification needs to be reevaluated.
| S-EPMC8870315 | biostudies-literature
Project description:This study explored the role of radioiodine therapy (RAI) in low-risk thyroid cancer patients and examined the disease-specific survival (DSS) rates in a large cohort of differentiated thyroid cancer patients (DTC). We obtained patient data from SEER database. Patients who underwent total thyroidectomy were included and categorized into three groups based on histology: classical papillary thyroid carcinoma (C-PTC), follicular type variant carcinoma (FV-PTC), and follicular thyroid cancer (FTC). Patients with distant metastasis, tumor size ≥ 200 mm, chemotherapy, or any type of radiation other than RAI were also excluded. A nomogram was developed and tested for discrimination and calibration. In total, 96,532 thyroid cancer cases were examined, including 59,460 C-PTC, 31,583 FV-PTC, and 5,489 FTC cases. Age > 65 years and male sex were correlated with lower survival rates across the subtypes. In addition, extrathyroidal extension had a worse survival effect in patients with FTC. DSS rates were compared between patients who received RAI and those who did not, with a 3% difference in C-PTC (94% vs. 91%, p < 0.001), 2% in FV-PTC (92% vs. 90%, p < 0.001), and 1% in FTC (89% vs. 88%, p = 0.084) at 15 years. The nomograms for long-term DSS showed high discriminatory abilities with C-indices of 0.815, 0.805, and 0.781 for C-PTC, FV-PTC, and FTC, respectively. The developed nomogram can be used in the treatment plan for patients with DTC. Our study emphasizes the prognostic factors for DTC and highlights the need for personalized treatment plans based on individual risk profiles.
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Project description:As the prevalence of children on renal replacement therapy (RRT) increases world wide and such therapy comprises at least 2% of any national dialysis or transplant programme, it is essential that paediatric nephrologists are able to advise families on the possible outcome for their child on dialysis. Most children start dialysis with the expectation that successful renal transplantation is an achievable goal and will provide the best survival and quality of life. However, some will require long-term dialysis or may return intermittently to dialysis during the course of their chronic kidney disease (CKD). This article reviews the available outcome data for children on chronic dialysis as well as extrapolating data from the larger adult dialysis experience to inform our paediatric practice. The multiple factors that may influence outcome, and, particularly, those that can potentially be modified, are discussed.
| S-EPMC2755764 | biostudies-other
Project description:IntroductionChronic non-bacterial osteomyelitis (CNO) is an autoinflammatory bone-disease of unknown origin. The National Pediatric Rheumatologic Database (NPRD) collects long-term data of children and adolescents with rheumatic diseases including CNO.ObjectiveTo assess characteristics, courses, and outcomes of CNO with onset in childhood and adolescence and to identify outcome predictors.MethodsFrom 2015 to 2021 patients with a confirmed diagnosis of CNO, who were registered in the NPRD during their first year of disease and at least one follow-up visit, were included in this analysis and observed for up to 4 years.ResultsFour hundred patients with recent diagnosis of CNO were enrolled in the NRPD during the study period. After 4 years, patient data documentation was sufficient to be analyzed in 81 patients. A significant decline of clinical and radiological lesions is reported: at inclusion in the registry, the mean number of clinical lesions was 2.0 and 3.0 MRI lesions per patient. A significant decrease of manifestations during 4 years of follow-up (mean clinical lesions 0.5, p < 0.001; mean MRI lesions 0.9 (p < 0.001)) was documented. A significant improvement of physician global disease activity (PGDA), patient-reported overall well-being, and childhood health assessment questionnaire (C-HAQ) was documented. Therapeutically, an increase of disease-modifying anti-rheumatic drugs over the years can be stated, while bisphosphonates rather seem to be considered as a therapeutic DMARD option in the first years of disease. Only 5-7% of the patients had a severe disease course as defined by a PGDA > = 4. Predictors associated with a severe disease course include the site of inflammation (pelvis, lower extremity, clavicle), increased erythrocyte sedimentation rate, and multifocal disease at first documentation. The previously published composite PedCNO disease activity score was analyzed revealing a PedCNO70 in 55% of the patients at 4YFU.ConclusionAn improvement of physician global disease activity (PGDA), patient reported overall well-being and imaging-defined disease activity measures was documented, suggesting that inactivity of CNO disease can be reached. PedCNO score and especially PGDA, MRI-defined lesions and in a number of patients also the C-HAQ seem to be reliable parameters for describing disease activity. The identification of risk factors at the beginning of the disease might influence treatment decision in the future.
| S-EPMC10683360 | biostudies-literature
Project description:PurposeThis multicentric study aimed to investigate the main prognostic factors associated with treatment response at 1 year after radioactive iodine therapy (RAIT) and the last disease status in pediatric patients affected by differentiated thyroid carcinoma (DTC).Materials and methodsIn the period 1990-2020, all consecutive patients ≤ 18 years from six different centers were retrospectively included. Patients were classified as low, intermediate, and high risk for persistence/recurrence. The response to RAIT was evaluated and scored 1 year later according to 2015 ATA guidelines. Moreover, at the last follow-up, the disease status was evaluated and dichotomized as no evidence of disease (NED) or persistent disease.ResultsTwo hundred and eighty-five patients (197 female, 88 male; mean age 14.4 years) were recruited. All, except nine, underwent near-total thyroidectomy followed by RAIT. One-year after first RAIT, 146/276 (53%) patients had excellent response, 37/276 (14%) indeterminate response, and 91/276 (33%) incomplete response. One-year after RAIT, children with excellent response had significantly lower stimulated thyroglobulin (sTg) compared to not excellent group (median sTg 4.4 ng/ml vs 52.5 ng/ml, p < 0.001). ROC curve showed sTg higher than 27.2 ng/ml as the most accurate to predict 1-year treatment response. After a median follow-up of 133 months, NED was present in 241 cases (87%) while persistent disease in 35 (13%). At multivariate analysis, sTg and 1-year treatment response categories were both significantly associated with the last disease status (p value 0.023 and < 0.001).ConclusionsIn pediatric DTC, sTg is significantly associated with 1-year treatment response and final outcome. However, 1-year response is the principal prognostic factor able to predict pediatric DTCs outcome.
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Project description:ImportanceRadiofrequency ablation (RFA) has gained increasing interest as a minimally invasive procedure to treat low-risk papillary thyroid carcinoma (PTC). Considering the indolent nature of this disease, studies in large populations with long follow-up would be invaluable to further substantiate the effectiveness of RFA.ObjectiveTo evaluate the long-term (58.5 months) outcomes of patients with T1N0M0 PTC who underwent RFA and investigate risk factors for local tumor progression (LTP).Design, setting, and participantsThis cohort study included 1613 patients aged 18 years or older with T1N0M0 PTC who underwent ultrasonography-guided RFA between January 2014 and December 2020 at the Chinese People's Liberation Army General Hospital in Beijing, China. Included in the analysis were patients with PTC (confirmed by biopsy) with a maximum diameter of 20 mm or less; no evidence of extrathyroidal extension (capsular disruption or involvement of perithyroidal tissue), lymph node metastasis, or distant metastasis on ultrasonography or computed tomography; and no evidence of an aggressive subtype of PTC on biopsy. Patients with PTC larger than 2 cm, less than 12 months of follow-up, or inadequate follow-up information were excluded. Data were analyzed in April 2023.Main outcomes and measuresLong-term progression rate, disease-free survival, and complete tumor disappearance and their associations with patient and tumor characteristics. Disease-free survival was calculated using Kaplan-Meier analysis. Cox proportional hazards regression analyses were performed to assess risk factors for LTP and complete tumor disappearance.ResultsThe study comprised 1613 patients (mean [SD] age, 43.3 [10.2] years; 1256 women [77.9%]) with 1834 T1N0M0 PTC tumors. During a mean follow-up of 58.5 months (range, 27 to 111 months), LTP was observed in 69 patients (4.3%), including 42 (2.6%) with tumor recurrence and 27 (1.7%) with tumor persistence. Cumulative disease-free survival rates at 1, 3, 5, and 8 years were 98.0%, 96.7%, 96.0%, and 95.7%, respectively. The overall complication rate was 2.0% (32 patients), with 6 (0.4%) major complications. Independent risk factors for LTP included subcapsular tumor location 2 mm or less from the capsule or trachea (hazard ratio [HR], 3.36; 95% CI, 2.02-5.59; P < .001) and multifocal tumors (HR, 2.27; 95% CI, 1.30-3.96; P = .004). Furthermore, 1376 patients (85.3%) showed complete tumor disappearance at follow-up ultrasonographic examination. Factors associated with complete tumor disappearance included age 40 years or less (HR, 0.78; 95% CI, 0.70-0.87; P < .001), stage T1a tumors (HR, 0.37; 95% CI, 0.31-0.45; P < .001), and unifocal tumors (HR, 0.50; 95% CI, 0.42-0.60; P < .001).Conclusions and relevanceIn this cohort study, ultrasonography-guided RFA for T1N0M0 PTC had excellent long-term outcomes. Patients with unifocal T1N0M0 PTC and subcapsular tumor location more than 2 mm from the capsule or trachea may be the best candidates for RFA.
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Project description:BackgroundStudies have reported differing factors associated with poor outcomes in patients with differentiated thyroid cancer (DTC). We aimed to describe our 20 years of experience in the management of thyroid cancer (TC) and identify predictors of treatment outcomes.MethodsWe conducted a retrospective review of medical records of patients with TC seen in the Thyroid Center at King Saud University Medical City (KSUMC) in Riyadh, Saudi Arabia, between the years 2000 and 2020. Demographic and clinical data including pathological characteristics were collected. The American Thyroid Association (ATA) risk stratification was determined for all patients at the postoperative period as well as the response to therapy at the final follow-up visit.ResultsA total of 674 patients (mean age: 47.21 years) with TC, 571 (84.7%) of which were women, were included. There were 404 (60.0%) patients with ATA low risk, 127 (18.8%) with intermediate risk, and 143 (21.2%) with high-risk histology. Overall, 461 patients (68.4%) had an excellent response to treatment, 65 (9.6%) had an indeterminate response, 83 (12.3%) had a biochemical incomplete response, and 65 (9.6%) had a structural incomplete response. Patients who had an excellent response were mostly ATA low risk (n = 318 of 431, 68.1%), whereas 40 of 65 patients (61.5%) of those with ATA high-risk histology had a structural incomplete response to treatment. There were significantly more women who had an excellent response compared with men. Obesity, lymphovascular invasion, and size of the tumor were significant predictors of worse outcomes to therapy.ConclusionTumor size, lymphovascular invasion, and obesity are strong predictors of a worse response to therapy among patients with TC. Patients with obesity should be carefully followed up regardless of their risk stratification in light of the recent compelling evidence associating obesity with thyroid cancer and its higher risk of a worse disease outcome. ATA risk stratification is well correlated with patient long-term outcomes.
| S-EPMC10690932 | biostudies-literature
Project description:A long-term follow-up was made of 12 elbows operated upon between 1971 and 1986, with more than 20 years' follow-up, in nine males and three females, age at the time of surgery between 10 and 19 years . Eight right and four left elbows were involved, and there were three aetiological causes. Seven cases were sequelae of elbow fractures, of which five were supracondylar and two were of the olecranon. There were four cases of juvenile rheumatoid arthritis and one was post-osteomyelitis. The surgical technique involved a modification made by Vainio of MacAusland's technique (wider resection of the osseous ends and total covering of the bloody surfaces) [5, 9]. After extirpating the tissue blocking the joint, we proceeded to remodel the distal humerus in a wide V shape, the proximal end of the ulnar and, if necessary, the radial head. The proximal end of the ulna was sectioned transversely. All surgery was carried out sub-periosteally. Then, an interposition material was placed in one piece and sutured over the distal humerus and cut ends of the ulna and radius. The articular ends were brought together, and the capsule was closed using equidistant stitching, as is the skin. A small compression bandage was applied, and the arm was immobilised with a collar and cuff sling, with the forearm flexed to slightly less than a right angle. In ten cases, the interposition material was fascia lata grafts; in one case, skin graft and in one case, Gelfoam graft. Early rehabilitation began when post-operative pain allowed. Follow-up ranged from 25 to 32 years. Pre-surgical movement ranged between 90 degrees and 120 degrees of flexion and 30 degrees and 90 degrees of extension. Post-operative range varied between 90 degrees and 150 degrees of flexion. The five cases of full pre-operative ankylosis achieved between 90 degrees and 150 degrees of flexion and between 0 degrees and 70 degrees of extension. The total range of motion at the latest follow-up varied from 35 degrees to 150 degrees . Patients who were able to perform flexion of 120 degrees or more were considered to be excellent, those between 90 degrees and 119 degrees were graded good, from 60 degrees to 89 degrees fair and those 59 degrees or less poor. The ability to attain a hand to mouth position requires a mobility of 120 degrees . We obtained excellent results in two patients, good results in three, fair results in four and poor results in three. The fascia lata was used in 83% of cases, obtaining excellent to good results in five patients (41%). Elbow interposition arthroplasty has its indications in children and adolescents where arthrodesis or total joint replacement cannot be performed.
| S-EPMC2269022 | biostudies-other
Project description:BackgroundWe identified differentiated thyroid cancer (DTC) survivors from SEER registries and performed Poisson regression to calculate the relative risks (RRs) of subsequent malignancies (SMs) by different sites associated with radioactive iodine (RAI) treatment, and the attributable risk proportion of RAI for developing different SMs.ResultsWe identified 4628 of 104,026 DTC patients developing a SM after two years of their DTC diagnosis, with a medium follow-up time of 113 months. The adjusted RRs of developing SM associated with RAI varied from 0.98 (0.58-1.65) for neurologic SMs to 1.37 (1.13-1.66) for hematologic SMs. The RRs of developing all cancer combined SMs generally increased with age at DTC diagnosis and decreased with the latency time. We estimated that the attributable risk proportion of RAI treatment is only 0.9% for all cancer combined SMs and 20% for hematologic SMs, which is the highest among all SMs. The tumor features and mortalities in patients treated with and without RAI are generally comparable.ConclusionWith the large population based analyses, we concluded that a low percentage of DTC survivors would develop SMs during their follow-up. Although the adjusted RR of SMs development increased slightly in patients receiving RAI, the attributable risk proportion associated with RAI was low, suggesting the absolute number of SMs induced by RAI in DTC survivors would be low. The attributable risk proportion of RAI treatment is the highest in hematological SMs, but when in consideration of its low incidence among all DTC survivors, the absolute number of hematological SMs was low.
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