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  1 / 337 MEDLINE  
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PMID:29390424
Autor:Yao PS; Chen GR; Shang-Guan HC; Lin QS; Wang XF; Zheng SF; Kang DZ
Dirección:Department of Neurosurgery.
Título:Adult hippocampal ganglioneuroblastoma: Case report and literature review.
Fuente:Medicine (Baltimore); 96(51):e8894, 2017 Dec.
ISSN:1536-5964
País de publicación:United States
Idioma:eng
Resumen:RATIONALE: Intracranial ganglioneuroblastoma represents a rare subtype of primitive neuroectodermal tumor. Here, we report a hippocampal ganglioneuroblastoma and a literature review of cerebral anglioneuroblastoma is carried out. PATIENT CONCERNS: We report a 16-year-old male patient presenting with absence seizure and high-infiltration hippocampal ganglioneuroblastoma. INTERVENTIONS: Magnetic resonance imaging (MRI) indicates a space-occupying lesion with a well-defined margin in the right temporal lobe and hippocampus. However, hyper-signal on flair and diffusion-weighted imaging (DWI) with a low apparent diffusion coefficient (ADC) value is detected, which prompts high tumoral invasiveness. INTERVENTIONS: A total resection of tumor and subsequent chemotherapy combing with radiotherapy is performed. OUTCOMES: For a follow-up period of 60 months, no evidence of recurrence and further seizures are detected. LESSONS: High-infiltration hippocampal ganglioneuroblastoma is a rare event. MRI examination often showed features of low-grade gliomas, while hyper-signal lesion on DWI with a low ADC value can be detected. Complete resection combined with fractionated radiotherapy and chemotherapy was the optimal treatment for cerebral ganglioneuroblastoma.
Tipo de publicación:CASE REPORTS; JOURNAL ARTICLE


  2 / 337 MEDLINE  
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PMID:28549783
Autor:Mody R; Naranjo A; Van Ryn C; Yu AL; London WB; Shulkin BL; Parisi MT; Servaes SE; Diccianni MB; Sondel PM; Bender JG; Maris JM; Park JR; Bagatell R
Dirección:CS Mott Children's Hospital, University of Michigan, Ann Arbor, MI, USA.
Título:Irinotecan-temozolomide with temsirolimus or dinutuximab in children with refractory or relapsed neuroblastoma (COG ANBL1221): an open-label, randomised, phase 2 trial.
Fuente:Lancet Oncol; 18(7):946-957, 2017 Jul.
ISSN:1474-5488
País de publicación:England
Idioma:eng
Resumen:BACKGROUND: Outcomes for children with relapsed and refractory neuroblastoma are dismal. The combination of irinotecan and temozolomide has activity in these patients, and its acceptable toxicity profile makes it an excellent backbone for study of new agents. We aimed to test the addition of temsirolimus or dinutuximab to irinotecan-temozolomide in patients with relapsed or refractory neuroblastoma. METHODS: For this open-label, randomised, phase 2 selection design trial of the Children's Oncology Group (COG; ANBL1221), patients had to have histological verification of neuroblastoma or ganglioneuroblastoma at diagnosis or have tumour cells in bone marrow with increased urinary catecholamine concentrations at diagnosis. Patients of any age were eligible at first designation of relapse or progression, or first designation of refractory disease, provided organ function requirements were met. Patients previously treated for refractory or relapsed disease were ineligible. Computer-based randomisation with sequence generation defined by permuted block randomisation (block size two) was used to randomly assign patients (1:1) to irinotecan and temozolomide plus either temsirolimus or dinutuximab, stratified by disease category, previous exposure to anti-GD2 antibody therapy, and tumour MYCN amplification status. Patients in both groups received oral temozolomide (100 mg/m per dose) and intravenous irinotecan (50 mg/m per dose) on days 1-5 of 21-day cycles. Patients in the temsirolimus group also received intravenous temsirolimus (35 mg/m per dose) on days 1 and 8, whereas those in the dinutuximab group received intravenous dinutuximab (17·5 mg/m per day or 25 mg/m per day) on days 2-5 plus granulocyte macrophage colony-stimulating factor (250 µg/m per dose) subcutaneously on days 6-12. Patients were given up to a maximum of 17 cycles of treatment. The primary endpoint was the proportion of patients achieving an objective (complete or partial) response by central review after six cycles of treatment, analysed by intention to treat. Patients, families, and those administering treatment were aware of group assignment. This study is registered with ClinicalTrials.gov, number NCT01767194, and follow-up of the initial cohort is ongoing. FINDINGS: Between Feb 22, 2013, and March 23, 2015, 36 patients from 27 COG member institutions were enrolled on this groupwide study. One patient was ineligible (alanine aminotransferase concentration was above the required range). Of the remaining 35 patients, 18 were randomly assigned to irinotecan-temozolomide-temsirolimus and 17 to irinotecan-temozolomide-dinutuximab. Median follow-up was 1·26 years (IQR 0·68-1·61) among all eligible participants. Of the 18 patients assigned to irinotecan-temozolomide-temsirolimus, one patient (6%; 95% CI 0·0-16·1) achieved a partial response. Of the 17 patients assigned to irinotecan-temozolomide-dinutuximab, nine (53%; 95% CI 29·2-76·7) had objective responses, including four partial responses and five complete responses. The most common grade 3 or worse adverse events in the temsirolimus group were neutropenia (eight [44%] of 18 patients), anaemia (six [33%]), thrombocytopenia (five [28%]), increased alanine aminotransferase (five [28%]), and hypokalaemia (four [22%]). One of the 17 patients assigned to the dinutuximab group refused treatment after randomisation; the most common grade 3 or worse adverse events in the remaining 16 patients evaluable for safety were pain (seven [44%] of 16), hypokalaemia (six [38%]), neutropenia (four [25%]), thrombocytopenia (four [25%]), anaemia (four [25%]), fever and infection (four [25%]), and hypoxia (four [25%]); one patient had grade 4 hypoxia related to therapy that met protocol-defined criteria for unacceptable toxicity. No deaths attributed to protocol therapy occurred. INTERPRETATION: Irinotecan-temozolomide-dinutuximab met protocol-defined criteria for selection as the combination meriting further study whereas irinotecan-temozolomide-temsirolimus did not. Irinotecan-temozolomide-dinutuximab shows notable anti-tumour activity in patients with relapsed or refractory neuroblastoma. Further evaluation of biomarkers in a larger cohort of patients might identify those most likely to respond to this chemoimmunotherapeutic regimen. FUNDING: National Cancer Institute.
Tipo de publicación:CLINICAL TRIAL, PHASE II; JOURNAL ARTICLE; MULTICENTER STUDY; RANDOMIZED CONTROLLED TRIAL
Nombre de substancia:0 (Antibodies, Monoclonal); 0 (MYCN protein, human); 0 (N-Myc Proto-Oncogene Protein); 624KN6GM2T (temsirolimus); 7673326042 (irinotecan); 7GR28W0FJI (Dacarbazine); 7SQY4ZUD30 (ch14.18 monoclonal antibody); 83869-56-1 (Granulocyte-Macrophage Colony-Stimulating Factor); EC 2.6.1.2 (Alanine Transaminase); W36ZG6FT64 (Sirolimus); XT3Z54Z28A (Camptothecin); YF1K15M17Y (temozolomide)


  3 / 337 MEDLINE  
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PMID:28511265
Autor:Neubauer H; Li M; Müller VR; Pabst T; Beer M
Dirección:Department of Diagnostic and Interventional Radiology, University Hospital Ulm, Germany.
Título:Diagnostic Value of Diffusion-Weighted MRI for Tumor Characterization, Differentiation and Monitoring in Pediatric Patients with Neuroblastic Tumors.
Título:Diagnostischer Stellenwert der diffusionsgewichteten MRT zur Tumorcharakterisierung, Tumordifferenzierung und zur Verlaufskontrolle bei pädiatrischen Patienten mit neuroblastischen Tumoren..
Fuente:Rofo; 189(7):640-650, 2017 Jul.
ISSN:1438-9010
País de publicación:Germany
Idioma:eng
Resumen:We explored the diagnostic value of diffusion-weighted MRI (DWI) for tumor characterization, differentiation and therapy monitoring in pediatric patients with extracranial neuroblastic tumors. All 29 patients (14 girls, median age: 3 years) with neuroblastoma (NB, n = 19), ganglioneuroblastoma (GNB, n = 4) and ganglioneuroma (GN, n = 6) who had had at least one in-house DWI examination since 2005 were identified and retrospectively analyzed. Two independent blinded readers measured ADC values (unit: 10-3 mm /s) and signal intensity ratios (SIRs) of the primary tumor and, if applicable, of the tumor after chemotherapy, metastases and tumor relapse. The pre-treatment ADC was 0.90 ±â€Š0.23 in NB/GNB and 1.70 ±â€Š0.36 in GN without overlap between the two entities for both readers, 0.67 ±â€Š0.14 in metastases and 0.72 ±â€Š0.18 in tumor relapse. With chemotherapy, mean ADC increased to 1.54 ±â€Š0.33 in NB/GNB and to 1.23 ±â€Š0.27 in metastases (p < 0.05). The median SIRs of various tumor lesions vs. liver, vs. muscle tissue and vs. adjacent tissue were significantly higher on DWI (range: 2.4 - 9.9) than on ce-T1w (range: 1.0 - 1.8, all p < 0.05). The coefficient of variation (CV) was ≤ 8.0 % for ADC and ≤ 16.4 % for signal intensity data. Based on mean ADC, DWI distinguishes between NB/GNB and GN with high certainty and provides plausible quantitative data on tumor response to therapy. Lesion conspicuity, as measured by SIR, is superior on DWI, compared to ce-T1w. DWI as a noninvasive, radiation-free and widely available imaging technique should be an integral part of MR imaging for neuroblastic tumors and should undergo prospective evaluation in multicenter studies. · DWI reliably distinguishes neuroblastoma/ganglioneuroblastoma from ganglioneuroma, based on the mean ADC.. · DWI provides plausible quantitative data on tumor response to chemotherapy.. · DWI offers highly superior lesion conspicuity compared to contrast-enhanced T1w imaging.. · DWI should be considered a standard for imaging neuroblastic tumors.. · Neubauer H, Li M, Müller VR et al. Diagnostic Value of Diffusion-Weighted MRI for Tumor Characterization, Differentiation and Monitoring in Pediatric Patients with Neuroblastic Tumors. Fortschr Röntgenstr 2017; 189: 640 - 650.
Tipo de publicación:JOURNAL ARTICLE


  4 / 337 MEDLINE  
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PMID:27988912
Autor:Lin X; Wu D; Chen C; Zheng N
Dirección:Department of Pediatric Surgery, The Second Affiliated Hospital and Yuying Children's Hospital of Wenzhou Medical University, Wenzhou, China. linxk2000@163.com.
Título:Clinical characteristics of adrenal tumors in children: a retrospective review of a 15-year single-center experience.
Fuente:Int Urol Nephrol; 49(3):381-385, 2017 Mar.
ISSN:1573-2584
País de publicación:Netherlands
Idioma:eng
Resumen:OBJECTIVE: Adrenal tumors are rare in children. The aim of this study is to review and analyze clinical data on the diagnosis and management of adrenal tumors in children. METHODS: Between 2001 and 2015, 48 pediatric patients (<14 years old) admitted to our institute with adrenal tumors were reviewed. Clinical features, imaging studies, surgical approaches, as well as pathological diagnoses were recorded. RESULTS: The series comprised 28 males and 20 females. Adrenomedullary tumors were 37, including 24 cases of neuroblastoma, 10 cases of ganglioneuroma, 2 cases of ganglioneuroblastoma, and 1 case of pheochromocytoma. Adrenocortical tumors were 10: 9 cases of cortical adenoma and 1 case of cortical cancer. The other one was hematoma. Fever, pain, and abdominal distention were the main clinical manifestations of adrenomedullary tumors, while Cushing syndrome was the most frequent presenting symptom of adrenocortical tumors. Both computed tomography and magnetic resonance imaging showed a high diagnostic yield. Some patients had an elevated hormone level. Open adrenalectomy was performed in 40 patients (83.3%), while a laparoscopic approach was employed in eight patients (16.7%). RESULTS: Adrenal tumors in children have various types,as well as clinical manifestations. Imaging and laboratory data could be useful for differentiation of malignant from benign tumor. Final diagnosis depends on pathology. Surgical excision of the adrenal tumors is the standard of care.
Tipo de publicación:JOURNAL ARTICLE


  5 / 337 MEDLINE  
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PMID:27984122
Autor:Magro G; Salvatorelli L; Alaggio R; D'Agata V; Nicoletti F; Di Cataldo A; Parenti R
Dirección:Department of Medical and Surgical Sciences and Advanced Technologies, GF Ingrassia, Azienda Ospedaliero-Universitaria "Policlinico-Vittorio Emanuele," Anatomic Pathology Section, School of Medicine, University of Catania, 95123 Catania, Italy. Electronic address: g.magro@unict.it.
Título:Diagnostic utility of cyclin D1 in the diagnosis of small round blue cell tumors in children and adolescents.
Fuente:Hum Pathol; 60:58-65, 2017 Feb.
ISSN:1532-8392
País de publicación:United States
Idioma:eng
Resumen:Small round blue cell tumors (SRBCTs) of children and adolescents are often diagnostically challenging lesions. With the increasing diagnostic approach based on small biopsies, there is the need of specific immunomarkers that can help in the differential diagnosis among the different tumor histotypes to assure the patient a correct diagnosis for proper treatment. Based on our recent studies showing cyclin D1 overexpression in both Ewing sarcoma/primitive peripheral neuroectodermal tumor (EWS/pPNET) and peripheral neuroblastic tumors (neuroblastoma and ganglioneuroblastoma), we immunohistochemically assessed cyclin D1 immunoreactivity in 128 cases of SRBCTs in children and adolescents to establish its potential utility in the differential diagnosis. All cases of EWS/pPNET and the undifferentiated/poorly differentiated neuroblastomatous component of all peripheral neuroblastic tumors exhibited strong and diffuse nuclear staining (>50% of neoplastic cells) for cyclin D1. In contrast, this marker was absent from rhabdomyosarcoma (regardless of subtype) and lymphoblastic lymphoma (either B- or T-cell precursors), whereas it was only focally detected (<5% of neoplastic cells) in some cases of Wilms tumor (blastemal component) and desmoplastic small round cell tumor. Our findings suggest that cyclin D1 can be exploitable as a diagnostic adjunct to conventional markers in confirming the diagnosis of EWS/pPNET or neuroblastoma/ganglioneuroblastoma. Its use in routine practice may also be helpful for those cases of SRBCT with undifferentiated morphology that are difficult to diagnose after application of the conventional markers.
Tipo de publicación:EVALUATION STUDIES; JOURNAL ARTICLE
Nombre de substancia:0 (Biomarkers, Tumor); 0 (CCND1 protein, human); 136601-57-5 (Cyclin D1)


  6 / 337 MEDLINE  
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PMID:27820130
Autor:Cash T; Alazraki A; Qayed M; Katzenstein HM
Dirección:*Aflac Cancer Center, Division of Pediatric Hematology/Oncology, Children's Healthcare of Atlanta †Department of Radiology and Imaging Sciences, Emory University, Atlanta, GA ‡Division of Pediatric Hematology/Oncology, Monroe Carell Jr Children's Hospital, Vanderbilt University, Nashville, TN.
Título:Prolonged Isotretinoin in Ultra High-Risk Neuroblastoma.
Fuente:J Pediatr Hematol Oncol; 39(1):e33-e35, 2017 Jan.
ISSN:1536-3678
País de publicación:United States
Idioma:eng
Resumen:Patients with high-risk neuroblastoma remain a therapeutic challenge with significant numbers of patients failing to respond sufficiently to initial therapy. These patients with poor response to induction are considered as ultra high-risk and are in need of novel treatment strategies. Isotretinoin is part of the standard of care treatment for patients with high-risk disease who undergo high-dose chemotherapy with autologous stem cell rescue although some have questioned the optimal administration schedule. Prolonged use of isotretinoin was well tolerated and may have contributed to long-term survival in a group of patients with ultra high-risk neuroblastoma.
Tipo de publicación:CASE REPORTS; JOURNAL ARTICLE
Nombre de substancia:0 (Antineoplastic Agents); 35MRW7B4AD (3-Iodobenzylguanidine); EH28UP18IF (Isotretinoin)


  7 / 337 MEDLINE  
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PMID:27980262
Autor:Suenaga S; Ichiyanagi O; Ito H; Naito S; Kato T; Nagaoka A; Kato T; Yamakawa M; Obara Y; Tsuchiya N
Dirección:Department of Urology, Yamagata University Faculty of Medicine, Japan.
Título:Expression of Extracellular Signal-regulated Kinase 5 and Ankyrin Repeat Domain 1 in Composite Pheochromocytoma and Ganglioneuroblastoma Detected Incidentally in the Adult Adrenal Gland.
Fuente:Intern Med; 55(24):3611-3621, 2016.
ISSN:1349-7235
País de publicación:Japan
Idioma:eng
Resumen:Composite pheochromocytoma (cPC) is extremely rare, arising in the adrenal medulla as a mixture of PC and other tumors of neural origin. We herein report on a case of adrenal incidentaloma post-operatively diagnosed as cPC with ganglioneuroblastoma (GNBL). The PC component had 7 points on the PASS, a Ki-67 index of 5.1%, a focal absence of sustentacular cells, and no genetic aberrations in succinate dehydrogenase subunit B. The GNBL component exhibited no N-myc amplification. Tumor cells of both components were stained positively for extracellular signal-regulated kinase 5 and ankyrin repeat domain 1. The aberrant activation of growth signaling may play a role in the marginal malignancy of cPC.
Tipo de publicación:CASE REPORTS; JOURNAL ARTICLE
Nombre de substancia:0 (ANKRD1 protein, human); 0 (Muscle Proteins); 0 (Nuclear Proteins); 0 (Repressor Proteins); 0 (SDHD protein, human); EC 1.3.99.1 (Succinate Dehydrogenase); EC 2.7.11.24 (MAPK7 protein, human); EC 2.7.11.24 (Mitogen-Activated Protein Kinase 7)


  8 / 337 MEDLINE  
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PMID:27569100
Autor:Benderli Cihan Y; Aytekin A; Sarigoz T
Dirección:Kayseri Education and Research Hospital, Department of Radiation Oncology, Kayseri, Turkey.
Título:Role of radiotherapy in adult ganglioneuroblastoma and ganglioneuroma.
Fuente:J BUON; 21(3):750, 2016 May-Jun.
ISSN:1107-0625
País de publicación:Greece
Idioma:eng
Tipo de publicación:LETTER


  9 / 337 MEDLINE  
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PMID:27526309
Autor:Sousa NV; Marques de Oliveira LC; Cortez PJ; Valenti VE; Garner DM; Irulegui Rde S; Moreira DA
Dirección:Faculdade de Medicina de Itajubá, Av. Renó Júnior, 368 - São Vicente, Itajubá, MG, Brazil. nathaliavieira100@hotmail.com.
Título:A rare case of Ganglioneuroblastoma Encapsulated in Pheochromocytoma.
Fuente:Acta Medica (Hradec Kralove); 59(2):67-9, 2016.
ISSN:1211-4286
País de publicación:Czech Republic
Idioma:eng
Resumen:Pheochromocytoma and Ganglioneuroblastoma are separate diseases and a rare combination in which the diagnosis can only be confirmed by pathological examination after tumor excision. We reported here a case of ganglioneuroblastoma encapsulated in pheochromocytoma. The patient is a woman, 73 years old, hypertensive, with hypothyroidism, associated for 15 years with hypercholesterolemia and hypertriglyceridemia, which had frequent complaints of low back pain. She underwent magnetic resonance and the findings were consistent with the diagnosis of pheochromocytoma. After surgery, anatomic, pathologic and immunohistochemistry analysis confirmed the diagnosis of pheochromocytoma composed by small ganglioneuroblastoma representation with the identification of small focus of infiltration of the adrenal capsule and adipose tissue by pheochromocytoma. This rare association can instigate the discussion of methods of diagnosis, more effective and more appropriate treatments for each patient.
Tipo de publicación:CASE REPORTS; JOURNAL ARTICLE
Nombre de substancia:0 (Catecholamines); 35MRW7B4AD (3-Iodobenzylguanidine)


  10 / 337 MEDLINE  
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PMID:27506465
Autor:Salet MC; Vogels R; Brons P; Schreuder B; Flucke U
Dirección:Department of Pathology, Radboud University Medical Center, P.O. Box 9101, Nijmegen, 6500 HB, The Netherlands. Myrthe.Salet@radboudumc.nl.
Título:Maturation toward neuronal tissue in a Ewing sarcoma of bone after chemotherapy.
Fuente:Diagn Pathol; 11(1):74, 2016 Aug 09.
ISSN:1746-1596
País de publicación:England
Idioma:eng
Resumen:BACKGROUND: Ewing sarcoma is the second most common bone tumor, occurring mainly in children and young adults. It shows a typical primitive, small round cell morphology and a characteristic fusion oncogene involving EWSR1 and members of the ETS family in most of the cases. Neuronal maturation after chemotherapy is a rare phenomenon and we herein describe such an exceptional case. CASE PRESENTATION: An 8-year old boy was diagnosed with a Ewing sarcoma in the left femur. On biopsy the morphology was typical and there was an EWSR1-FLI1 gene fusion. He underwent neo-adjuvant chemotherapy and resection of the tumor. On microscopic evaluation, part of the tumor showed ganglioneuroblastoma-like differentiation with expression of neuronal markers. The continued presence of EWSR1 rearrangement in both the blue round cell component and the ganglioneuroblastoma-like component was shown by FISH analysis. CONCLUSIONS: In conclusion, this case describes the possibility of a Ewing sarcoma to differentiate into a ganglioneuroblastoma-like lesion after neo-adjuvant chemotherapy treatment; the prognostic value of this phenomenon remains questionable.
Tipo de publicación:CASE REPORTS; JOURNAL ARTICLE
Nombre de substancia:0 (Calmodulin-Binding Proteins); 0 (EWSR1 protein, human); 0 (EWSR1-FLI1 fusion protein, human); 0 (Oncogene Proteins, Fusion); 0 (RNA-Binding Protein EWS); 0 (RNA-Binding Proteins); 5J49Q6B70F (Vincristine); 6PLQ3CP4P3 (Etoposide); 80168379AG (Doxorubicin); UM20QQM95Y (Ifosfamide)



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