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Article ; Online: Hemophagocytic lymphohistiocytosis: presentation and outcome of twenty-one patients at a single institution.

Rodrigues, Jéssica Benigno / Nasr, Bárbara Pinto / Cypriano, Monica Dos Santos

Hematology, transfusion and cell therapy

2021 Volume 44, Issue 4, Page(s) 485–490

Abstract: Introduction: Hemophagocytic lymphohistiocytosis comprises a systemic hyperactivation of macrophages that requires prompt recognition of symptoms and early treatment.: Objective and method: In this context, we described clinical and laboratory ... ...

Abstract	Introduction: Hemophagocytic lymphohistiocytosis comprises a systemic hyperactivation of macrophages that requires prompt recognition of symptoms and early treatment. Objective and method: In this context, we described clinical and laboratory characteristics, therapeutic modality and outcome of 21 patients with HLH treated at a pediatric oncology hospital between January 2000 and February 2019. Results: HLH mainly affected females, fever was the most frequent clinical sign and hyperferritinemia was the most prevalent laboratory abnormality. All patients were admitted to the intensive care unit (ICU) at some point. Fifteen (71.4%) patients presented resolution criteria and eight (53.3%) of them presented reactivation. The mortality rate was 57.1% and the mean time between diagnosis and death was 9.98 months. The 5-year overall survival (OS) was 36.7%. We observed a significant difference in prognosis associated with reactivation of HLH. These patients demonstrated an estimated 5-year OS of 25%, while all patients that did not reactivate were alive until the end of the follow-up. Conclusion: In conclusion, HLH is a rare disease with a high mortality rate, especially in patients with disease reactivation and those with familial- or immunodeficiency-associated forms, which makes early recognition and genetic testing crucial for appropriate management and prompt SCT indication.
Language	English
Publishing date	2021-06-24
Publishing country	Brazil
Document type	Journal Article
ISSN	2531-1387
ISSN (online)	2531-1387
DOI	10.1016/j.htct.2021.05.003
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Article: Surgery in Bilateral Wilms Tumor-A Single-Center Experience.

Souza, Fernanda Kelly Marques de / Fanelli, Mayara Caroline Amorim / Duarte, Alexandre Alberto Barros / Alves, Maria Teresa de Seixas / Lederman, Henrique Manoel / Cypriano, Monica Dos Santos / Abib, Simone de Campos Vieira

Children (Basel, Switzerland)

2023 Volume 10, Issue 11

Abstract: The treatment of bilateral Wilms tumors (BWT) involves curing the cancer, preserving long-term renal function, and maintaining a good quality of life. Established methods for achieving these goals include preoperative chemotherapy and nephron-sparing ... ...

Abstract	The treatment of bilateral Wilms tumors (BWT) involves curing the cancer, preserving long-term renal function, and maintaining a good quality of life. Established methods for achieving these goals include preoperative chemotherapy and nephron-sparing surgery (NSS). This study aimed to evaluate the experience of a single institution in treating patients with BWT. We analyzed cases of BWT treated at the Pediatric Oncology Institute-GRAACC-Federal University of São Paulo over a period of 35 years. Bleeding control was performed with manual compression of the renal parenchyma. Thirty-three patients were included in the study. Thirty cases were synchronous tumors. The mean age at diagnosis was 30.4 months (±22 m) and 66.7% were girls. The median follow-up period was 83 months. Neoadjuvant chemotherapy was the primary approach in most patients (87.9%), with a simultaneous upfront surgical approach performed in 84.8%. Most patients underwent bilateral NSS (70.4%). There were no early complications in this series, but 39.4% had clinical complications. The five-year survival rate was 76%. Therefore, it is clear that the surgical approach to BWT plays a crucial role in achieving good outcomes. However, it is difficult to standardize surgical techniques and technology may have the potential to enhance safety.
Language	English
Publishing date	2023-11-07
Publishing country	Switzerland
Document type	Journal Article
ZDB-ID	2732685-8
ISSN	2227-9067
ISSN	2227-9067
DOI	10.3390/children10111790
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Article ; Online: Surgical Management of Wilms Tumors with Intravenous Extension: A Multicenter Analysis of Clinical Management with Technical Insights.

Pio, Luca / Abib, Simone / Guerin, Florent / Chardot, Christophe / Blanc, Thomas / Sarrai, Nadia / Martelli, Helene / De Souza, Fernanda K M / Fanelli, Mayara C A / Tamisier, Daniel / Guilhen, José Cícero S / Le Bret, Emmanuel / Belli, Emré / Fadel, Elie / Cypriano, Monica D S / Minard, Véronique / Pasqualini, Claudia / Schleiermacher, Gudrun / Lemelle, Lauriane /

Rod, Julien / Irtan, Sabine / Pistorio, Angela / Gauthier, Frederic / Branchereau, Sophie / Sarnacki, Sabine

Annals of surgical oncology

2024

Abstract: Background: About 5% of Wilms tumors present with vascular extension, which sometimes extends to the right atrium. Vascular extension does not affect the prognosis, but impacts the surgical strategy, which is complex and not fully standardized. Our goal ...

Abstract	Background: About 5% of Wilms tumors present with vascular extension, which sometimes extends to the right atrium. Vascular extension does not affect the prognosis, but impacts the surgical strategy, which is complex and not fully standardized. Our goal is to identify elements of successful surgical management of Wilms tumors with vascular extensions. Patients and methods: A retrospective study of pediatric Wilms tumors treated at three sites (January 1999-June 2019) was conducted. The inclusion criterion was the presence of a renal vein and vena cava thrombus at diagnosis. Tumor stage, pre and postoperative treatment, preoperative imaging, operative report, pathology, operative complications, and follow-up data were reviewed. Results: Of the 696 pediatric patients with Wilms tumors, 69 (9.9%) met the inclusion criterion. In total, 24 patients (37.5%) had a right atrial extension and two presented with Budd-Chiari syndrome at diagnosis. Two died at diagnosis owing to pulmonary embolism. All patients received neoadjuvant chemotherapy and thrombus regressed in 35.6% of cases. Overall, 14 patients had persistent intra-atrial thrombus extension (58%) and underwent cardiopulmonary bypass. Most thrombi (72%) were removed intact with nephrectomy. Massive intraoperative bleeding occurred during three procedures. Postoperative renal insufficiency was identified as a risk factor for patient survival (p = 0.01). With a median follow-up of 9 years (range: 0.5-20 years), overall survival was 89% and event-free survival was 78%. Conclusions: Neoadjuvant chemotherapy with proper surgical strategy resulted in a survival rate comparable to that of children with Wilms tumors without intravascular extension. Clinicians should be aware that postoperative renal insufficiency is associated with worse survival outcomes.
Language	English
Publishing date	2024-04-05
Publishing country	United States
Document type	Journal Article
ZDB-ID	1200469-8
ISSN	1534-4681 ; 1068-9265
ISSN (online)	1534-4681
ISSN	1068-9265
DOI	10.1245/s10434-024-15232-w
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Article ; Online: First Transcriptome Analysis of Hepatoblastoma in Brazil: Unraveling the Pivotal Role of Noncoding RNAs and Metabolic Pathways.

Aguiar, Talita Ferreira Marques / Rivas, Maria Prates / de Andrade Silva, Edson Mario / Pires, Sara Ferreira / Dangoni, Gustavo Dib / Macedo, Taiany Curdulino / Defelicibus, Alexandre / Barros, Bruna Durães de Figueiredo / Novak, Estela / Cristofani, Lilian Maria / Odone, Vicente / Cypriano, Monica / de Toledo, Silvia Regina Caminada / da Cunha, Isabela Werneck / da Costa, Cecilia Maria Lima / Carraro, Dirce Maria / Tojal, Israel / de Oliveira Mendes, Tiago Antônio / Krepischi, Ana Cristina Victorino

Biochemical genetics

2024

Abstract: Hepatoblastoma stands as the most prevalent liver cancer in the pediatric population. Characterized by a low mutational burden, chromosomal and epigenetic alterations are key drivers of its tumorigenesis. Transcriptome analysis is a powerful tool for ... ...

Abstract	Hepatoblastoma stands as the most prevalent liver cancer in the pediatric population. Characterized by a low mutational burden, chromosomal and epigenetic alterations are key drivers of its tumorigenesis. Transcriptome analysis is a powerful tool for unraveling the molecular intricacies of hepatoblastoma, shedding light on the effects of genetic and epigenetic changes on gene expression. In this study conducted in Brazilian patients, an in-depth whole transcriptome analysis was performed on 14 primary hepatoblastomas, compared to control liver tissues. The analysis unveiled 1,492 differentially expressed genes (1,031 upregulated and 461 downregulated), including 920 protein-coding genes (62%). Upregulated biological processes were linked to cell differentiation, signaling, morphogenesis, and development, involving known hepatoblastoma-associated genes (DLK1, MEG3, HDAC2, TET1, HMGA2, DKK1, DKK4), alongside with novel findings (GYNG4, CDH3, and TNFRSF19). Downregulated processes predominantly centered around oxidation and metabolism, affecting amines, nicotinamides, and lipids, featuring novel discoveries like the repression of SYT7, TTC36, THRSP, CCND1, GCK and CAMK2B. Two genes, which displayed a concordant pattern of DNA methylation alteration in their promoter regions and dysregulation in the transcriptome, were further validated by RT-qPCR: the upregulated TNFRSF19, a key gene in the embryonic development, and the repressed THRSP, connected to lipid metabolism. Furthermore, based on protein-protein interaction analysis, we identified genes holding central positions in the network, such as HDAC2, CCND1, GCK, and CAMK2B, among others, that emerged as prime candidates warranting functional validation in future studies. Notably, a significant dysregulation of non-coding RNAs (ncRNAs), predominantly upregulated transcripts, was observed, with 42% of the top 50 highly expressed genes being ncRNAs. An integrative miRNA-mRNA analysis revealed crucial biological processes associated with metabolism, oxidation reactions of lipids and carbohydrates, and methylation-dependent chromatin silencing. In particular, four upregulated miRNAs (miR-186, miR-214, miR-377, and miR-494) played a pivotal role in the network, potentially targeting multiple protein-coding transcripts, including CCND1 and CAMK2B. In summary, our transcriptome analysis highlighted disrupted embryonic development as well as metabolic pathways, particularly those involving lipids, emphasizing the emerging role of ncRNAs as epigenetic regulators in hepatoblastomas. These findings provide insights into the complexity of the hepatoblastoma transcriptome and identify potential targets for future therapeutic interventions.
Language	English
Publishing date	2024-04-22
Publishing country	United States
Document type	Journal Article
ZDB-ID	2168-4
ISSN	1573-4927 ; 0006-2928
ISSN (online)	1573-4927
ISSN	0006-2928
DOI	10.1007/s10528-024-10764-y
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Article ; Online: ASO Author Reflections: Surgical Management of Wilms Tumors with Intravenous Extension: A Multicenter Analysis of Clinical Management with Technical Insights.

Pio, Luca / Abib, Simone / Guerin, Florent / Chardot, Christophe / Blanc, Thomas / Sarrai, Nadia / Martelli, Helene / de Souza, Fernanda K M / Fanelli, Mayara C A / Tamisier, Daniel / Guilhen, José Cícero S / Le Bret, Emmanuel / Belli, Emré / Fadel, Elie / Cypriano, Monica D S / Minard, Véronique / Pasqualini, Claudia / Schleiermacher, Gudrun / Lemelle, Lauriane /

Rod, Julien / Irtan, Sabine / Pistorio, Angela / Gauthier, Frederic / Branchereau, Sophie / Sarnacki, Sabine

Annals of surgical oncology

2024

Language	English
Publishing date	2024-04-28
Publishing country	United States
Document type	Journal Article
ZDB-ID	1200469-8
ISSN	1534-4681 ; 1068-9265
ISSN (online)	1534-4681
ISSN	1068-9265
DOI	10.1245/s10434-024-15325-6
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Article: Management of Pediatric Tumors With Vascular Extension.

Fanelli, Mayara Caroline Amorim / Guilhen, José Cícero Stocco / Duarte, Alexandre Alberto Barros / de Souza, Fernanda Kelly Marques / Cypriano, Monica Dos Santos / Caran, Eliana Maria Monteiro / Lederman, Henrique Manoel / de Seixas Alves, Maria Teresa / Abib, Simone de Campos Vieira

Frontiers in pediatrics

2022 Volume 9, Page(s) 753232

Abstract: Background: ...

Abstract	Background:
Language	English
Publishing date	2022-01-04
Publishing country	Switzerland
Document type	Journal Article
ZDB-ID	2711999-3
ISSN	2296-2360
ISSN	2296-2360
DOI	10.3389/fped.2021.753232
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Article ; Online: Kidney-sparing whole abdominal irradiation in Wilms tumor: Potential advantages of VMAT technique.

Chen, Michael Jenwei / Leao, Caio Raposo / Simoes, Roberto Campos Pereira / Belletti, Fernanda Salheb / Figueiredo, Maria Luisa Sucharski / Cypriano, Monica Santos

Pediatric blood & cancer

2020 Volume 67, Issue 5, Page(s) e28223

Abstract: Purpose: To present a preliminary clinical experience and a dosimetric comparison of kidney-sparing volumetric modulated arc therapy (VMAT) with three-dimensional conformal radiotherapy (3D-CRT) for whole abdominal irradiation (WAI), in the setting of ... ...

Abstract	Purpose: To present a preliminary clinical experience and a dosimetric comparison of kidney-sparing volumetric modulated arc therapy (VMAT) with three-dimensional conformal radiotherapy (3D-CRT) for whole abdominal irradiation (WAI), in the setting of Wilms tumor (WT) treatment. Materials and methods: From a total of 20 consecutive WT cases treated with adjuvant irradiation, seven were submitted to WAI with VMAT. Renal function and survival rates were evaluated, and, for comparison purposes, similar VMAT and 3D-CRT treatment plans were performed for WAI patients, and differences were dosimetrically evaluated regarding doses to the remaining kidney and other organs at risk and the planning target volume (PTV). Results: After a median follow-up time of 40.8 months (35.3-52.2), no acute significant intestinal toxicity was observed, and median creatinine clearance was 110.1 and 103.3 mL/min/1.73 m², respectively, before treatment and at last follow-up for WAI patients (P = 0.128). For comparative plans, maximum and median doses were lower for the remaining kidney with VMAT than with 3D-CRT. VMAT was associated with better PTV coverage as compared with 3D-CRT, with superior results for all the evaluated parameters (D95, D2, V100%, V98%, V95%; P = 0.018). Conclusion: The use of VMAT technique is associated with lower radiation doses to the remaining kidney and better coverage to the PTV than 3D-CRT technique for WAI, with preliminary clinical experience showing a favorable toxicity profile. Long-term results from prospective studies might prove the ability of VMAT to spare renal function in the setting of WT treatment.
MeSH term(s)	Abdomen ; Adolescent ; Child ; Child, Preschool ; Disease-Free Survival ; Female ; Follow-Up Studies ; Humans ; Infant ; Kidney ; Kidney Neoplasms/mortality ; Kidney Neoplasms/radiotherapy ; Male ; Organ Sparing Treatments ; Organs at Risk ; Radiotherapy, Intensity-Modulated ; Survival Rate ; Wilms Tumor/mortality ; Wilms Tumor/radiotherapy
Language	English
Publishing date	2020-02-21
Publishing country	United States
Document type	Comparative Study ; Journal Article
ZDB-ID	2131448-2
ISSN	1545-5017 ; 1545-5009
ISSN (online)	1545-5017
ISSN	1545-5009
DOI	10.1002/pbc.28223
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Article ; Online: Preserved executive functioning and low stress symptoms in children treated for acute lymphoblastic leukemia.

Godoy, Priscilla Brandi Gomes / Mello, Claudia Berlim de / Pompéia, Sabine / da Costa, Cecília Maria Lima / Cypriano, Monica Dos Santos / Suchecki, Deborah

Applied neuropsychology. Child

2020 Volume 11, Issue 3, Page(s) 270–279

Abstract: Pediatric cancer treatment can negatively impact cognitive and psychosocial development, although it has been suggested that these adverse effects may be minimized when children have higher resilience and better executive functioning. We aimed to ... ...

Abstract	Pediatric cancer treatment can negatively impact cognitive and psychosocial development, although it has been suggested that these adverse effects may be minimized when children have higher resilience and better executive functioning. We aimed to evaluate the impact of pediatric Acute Lymphoblastic Leukemia (ALL) treatment on executive function, resilience and stress in survivors and to investigate correlations between executive functioning and resilience and between executive functioning and stress. The neuropsychological assessment was performed in 32 ALL survivors aged 7-17 years and 28 age-, sex- and socioeconomic status matched controls. Executive functioning was assessed by inhibitory control, mental flexibility and working memory tasks. Children's self-report scales were used to assess stress symptoms and resilience. Results revealed no executive function impairment nor stress symptom differences between ALL survivors and control group. In the ALL group, executive function and resilience were positively correlated, whereas executive function and stress were negatively correlated. We concluded that ALL treatment was not associated with impairment in executive functioning nor to increased stress symptoms in our sample. ALL survivors with better performance in mental flexibility and inhibition tasks reported fewer stress symptoms and more resilience, indicating a possible relationship between these variables.
MeSH term(s)	Child ; Executive Function/physiology ; Humans ; Memory, Short-Term ; Neuropsychological Tests ; Precursor Cell Lymphoblastic Leukemia-Lymphoma/complications ; Precursor Cell Lymphoblastic Leukemia-Lymphoma/drug therapy ; Survivors/psychology
Language	English
Publishing date	2020-08-13
Publishing country	United States
Document type	Journal Article
ZDB-ID	2673759-0
ISSN	2162-2973 ; 2162-2965
ISSN (online)	2162-2973
ISSN	2162-2965
DOI	10.1080/21622965.2020.1804907
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Article: Diagnostic Errors in Wilms' Tumors: Learning From Our Mistakes.

de Carvalho, Lucas Garschagen / Kobayashi, Thiago / Cypriano, Monica Dos Santos / Caran, Eliana Maria Monteiro / Lederman, Henrique Manoel / Alves, Maria Teresa de Seixas / Abib, Simone de Campos Vieira

Frontiers in pediatrics

2021 Volume 9, Page(s) 757377

Abstract: Aim: ...

Abstract	Aim:
Language	English
Publishing date	2021-10-25
Publishing country	Switzerland
Document type	Journal Article
ZDB-ID	2711999-3
ISSN	2296-2360
ISSN	2296-2360
DOI	10.3389/fped.2021.757377
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Article: Obesity in Survivors of Childhood Cancer: a Review.

Carneiro Teixeira, Julia Ferrari / Maia-Lemos, Priscila Dos Santos / Cypriano, Mônica Dos Santos / Pellegrini Pisani, Luciana

Pediatric endocrinology reviews : PER

2017 Volume 15, Issue 1, Page(s) 33–39

Abstract: Obesity is a late effect of antineoplastic treatment in childhood cancer survivors and this correlates with chronic complications. This review examines the data currently available to health professionals, for increasing awareness and identifying ... ...

Abstract	Obesity is a late effect of antineoplastic treatment in childhood cancer survivors and this correlates with chronic complications. This review examines the data currently available to health professionals, for increasing awareness and identifying strategies to address the treatment and prevention of late effects. The mechanism involved in the pathophysiology of obesity remains unclear. However, damage to the hypothalamus and endocrine disorders (e.g. insulin and leptin resistance) and a positive energetic balance may play a role in increasing obesity rates. A patient's diet during, and after treatment may also influence the weight of survivors. Implementation of an effective educational program by professionals during all stages of treatment enables children to obtain basic knowledge regarding food and nutrition, thereby encouraging them to take responsibility for developing healthy eating behaviors.
Language	English
Publishing date	2017-09
Publishing country	Israel
Document type	Journal Article
ZDB-ID	2434390-0
ISSN	1565-4753
ISSN	1565-4753
DOI	10.17458/per.vol15.2017.tlcp.obesitysurvivors
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