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1.
N Engl J Med ; 384(1): 42-50, 2021 01 07.
Artigo em Inglês | MEDLINE | ID: mdl-33406329

RESUMO

Two cases of pediatric lung cancer (in 23-month-old and 6-year-old boys) resulting from mother-to-infant transmission of uterine cervical tumors were incidentally detected during routine next-generation sequencing of paired samples of tumor and normal tissue. Spontaneous regression of some lesions in the first child and slow growth of the tumor mass in the second child suggested the existence of alloimmune responses against the transmitted tumors. Immune checkpoint inhibitor therapy with nivolumab led to a strong regression of all remaining tumors in the first child. (Funded by the Japan Agency for Medical Research and Development and others; TOP-GEAR UMIN Clinical Trials Registry number, UMIN000011141.).


Assuntos
Adenocarcinoma Mucinoso/etiologia , Carcinoma Neuroendócrino/etiologia , Neoplasias Pulmonares/etiologia , Complicações Neoplásicas na Gravidez , Neoplasias do Colo do Útero , Adenocarcinoma Mucinoso/diagnóstico por imagem , Adenocarcinoma Mucinoso/genética , Adulto , Carcinoma Neuroendócrino/diagnóstico por imagem , Carcinoma Neuroendócrino/genética , Carcinoma de Células Escamosas/patologia , Criança , Evolução Fatal , Feminino , Sequenciamento de Nucleotídeos em Larga Escala , Humanos , Lactente , Pulmão/diagnóstico por imagem , Pulmão/patologia , Neoplasias Pulmonares/diagnóstico por imagem , Neoplasias Pulmonares/genética , Neoplasias Pulmonares/patologia , Masculino , Mães , Gravidez , Vagina , Sequenciamento do Exoma
2.
Pediatr Int ; 66(1): e15754, 2024.
Artigo em Inglês | MEDLINE | ID: mdl-38924208

RESUMO

BACKGROUND: Recently, reports of endoscopic approaches for neuroblastoma, ganglioneuroblastoma, and ganglioneuroma (peripheral neuroblastic tumor; PNTs) have been increasing. This study aimed to clarify the indications for endoscopic surgery for PNTs. METHODS: Pediatric patients who underwent endoscopic surgery for PNTs at our institution were included in this study. Image-defined risk factors (IDRFs) were analyzed using preoperative computed tomography (CT). RESULTS: Twenty-four patients underwent endoscopic surgery for PNTs. The diagnoses included neuroblastoma (n = 11), ganglioneuroma (n = 10), and ganglioneuroblastoma (n = 3). Regarding the tumor site, there were 18 cases of adrenal tumors, five cases of mediastinal tumors, and one case of retroperitoneal tumors. Image-defined risk factors were positive in eight cases (contacted with a renal vessel, n = 6; compression of principal bronchi, n = 2). Complete resection was accomplished in 21 cases (14 of 16 IDRF-negative cases and seven of eight IDRF-positive cases). All patients survived without recurrence during the follow-up period. CONCLUSIONS: The CT findings of contact with renal vessels and compression of principal bronchi do not seem to be indicators of incomplete resection. An endoscopic approach to PNTs in pediatric patients is feasible with a good prognosis if patients are selected strictly.


Assuntos
Ganglioneuroblastoma , Ganglioneuroma , Neuroblastoma , Tomografia Computadorizada por Raios X , Humanos , Masculino , Feminino , Pré-Escolar , Neuroblastoma/cirurgia , Neuroblastoma/diagnóstico , Criança , Lactente , Ganglioneuroma/cirurgia , Ganglioneuroma/diagnóstico , Ganglioneuroblastoma/cirurgia , Ganglioneuroblastoma/diagnóstico , Estudos Retrospectivos , Endoscopia/métodos , Resultado do Tratamento , Adolescente , Seguimentos , Neoplasias das Glândulas Suprarrenais/cirurgia , Neoplasias das Glândulas Suprarrenais/diagnóstico , Neoplasias do Mediastino/cirurgia , Neoplasias do Mediastino/diagnóstico
3.
Surg Today ; 2024 Jul 09.
Artigo em Inglês | MEDLINE | ID: mdl-38980332

RESUMO

The purpose of our narrative review is to summarize the utilization of social media (SoMe) platforms for research communication within the field of surgery. We searched the PubMed database for articles in the last decade that discuss the utilization of SoMe in surgery and then categorized the diverse purposes of SoMe. SoMe proved to be a powerful tool for disseminating articles. Employing strategic methods like visual abstracts enhances article citation rates, the impact factor, h-index, and Altmetric score (an emerging alternative metric that comprehensively and instantly quantifies the social impact of scientific papers). SoMe also proved valuable for surgical education, with online videos shared widely for surgical training. However, it is essential to acknowledge the associated risk of inconsistency in quality. Moreover, SoMe facilitates discussion on specific topics through hashtags or closed groups and is instrumental in recruiting surgeons, with over half of general surgery residency programs in the US efficiently leveraging these platforms to attract the attention of potential candidates. Thus, there is a wealth of evidence supporting the effective use of SoMe for surgeons. In the contemporary era where SoMe is widely utilized, surgeons should be well-versed in this evidence.

4.
Pediatr Surg Int ; 40(1): 229, 2024 Aug 16.
Artigo em Inglês | MEDLINE | ID: mdl-39152284

RESUMO

BACKGROUND/PURPOSE: Living donor liver transplantation (LDLT) is vital for pediatric end-stage liver disease due to organ shortages. The graft-to-recipient weight ratio (GRWR) preoperatively measured predicts the outcomes of LDLT. We typically target between 0.8 and 3.0-4.0%, but the ideal GRWR remains controversial. We compared the outcomes of LDLT according to the GRWR to examine whether the criteria could be expanded while ensuring safety. METHODS: We retrospectively reviewed 99 patients who underwent LDLT in our department by dividing them into three groups according to their GRWR: Group S, with GRWR values lower than the normal range (GRWR < 0.8%); Group M, with GRWR values in the normal range (GRWR ≥ 0.8 to < 3.5%); and Group L, with GRWR values above the normal range (GRWR ≥ 3.5%). RESULTS: In Groups S and L, 46.2 and 44.4% of patients underwent splenectomy and delayed abdominal wall closure, respectively. After these intraoperative adjustments, there were no significant differences between the groups in 5-year patient survival, 5-year graft survival, or the occurrence of post-transplantation thrombosis. CONCLUSION: When the GRWR is beyond the normal threshold, the risk of complications associated with graft size might be reduced by adjustments to provide appropriate portal blood flow and by delayed abdominal wall closure.


Assuntos
Sobrevivência de Enxerto , Transplante de Fígado , Doadores Vivos , Humanos , Transplante de Fígado/métodos , Estudos Retrospectivos , Masculino , Feminino , Tamanho do Órgão , Criança , Pré-Escolar , Lactente , Peso Corporal , Fígado , Doença Hepática Terminal/cirurgia , Adolescente , Complicações Pós-Operatórias/epidemiologia
5.
Pediatr Surg Int ; 40(1): 218, 2024 Aug 08.
Artigo em Inglês | MEDLINE | ID: mdl-39115750

RESUMO

PURPOSE: We aimed to identify factors predicting the need for future liver transplantation (LT) at 18 years of age in patients with biliary atresia (BA). METHODS: BA patients with native liver survival at > 18 years of age were retrospectively reviewed. The clinical characteristics, outcomes, hepatobiliary function, and liver fibrosis markers of native liver survivors (NLS group) were compared with patients who subsequently underwent LT (LT group). RESULTS: The study population included 48 patients (NLS, n = 34; LT, n = 14). The male-to-female ratio, age at Kasai procedure, and type of BA in the two groups did not differ to a statistically significant extent. There was no significant difference in the MELD scores between the groups at 18 years of age. The aspartate aminotransferase-to-platelet ratio index (APRI), albumin-bilirubin (ALBI), and BA liver fibrosis (BALF) scores at 18 years of age were significantly higher in the LT group. The AUCs for APRI, ALBI, and BALF were 0.91, 0.79, and 0.85, respectively. CONCLUSION: Adult BA patients have limited options for LT owing to the lack of donor candidates and the low prevalence of deceased donors. The elucidation of prognostic factors for LT in adulthood is important. APRI was the most useful marker in this study.


Assuntos
Atresia Biliar , Transplante de Fígado , Humanos , Atresia Biliar/cirurgia , Masculino , Feminino , Estudos Retrospectivos , Adolescente , Prognóstico , Adulto Jovem , Adulto , Seguimentos
6.
Pediatr Surg Int ; 40(1): 225, 2024 Aug 14.
Artigo em Inglês | MEDLINE | ID: mdl-39143337

RESUMO

PURPOSE: Megacystis microcolon intestinal hypoperistalsis syndrome (MMIHS) is defined as a congenital visceral myopathy with genetic mutations. However, the etiology and pathophysiology are not fully understood. We aimed to generate a gene leiomodin-1a (lmod1a) modification technique to establish a zebrafish model of MMIHS. METHODS: We targeted lmod1a in zebrafish using CRISPR/Cas9. After confirming the genotype, we measured the expression levels of the target gene and protein associated with MMIHS. A gut transit assay and spatiotemporal mapping were conducted to analyze the intestinal function. RESULTS: Genetic confirmation showed a 5-base-pair deletion in exon 1 of lmod1a, which caused a premature stop codon. We observed significant mRNA downregulation of lmod1a, myh11, myod1, and acta2 and the protein expression of Lmod1 and Acta2 in the mutant group. A functional analysis of the lmod1a mutant zebrafish showed that its intestinal peristalsis was fewer, slower, and shorter in comparison to the wild type. CONCLUSION: This study showed that targeted deletion of lmod1a in zebrafish resulted in depletion of MMIHS-related genes and proteins, resulting in intestinal hypoperistalsis. This model may have the potential to be utilized in future therapeutic approaches, such as drug discovery screening and gene repair therapy for MMIHS.


Assuntos
Sistemas CRISPR-Cas , Colo , Modelos Animais de Doenças , Pseudo-Obstrução Intestinal , Peixe-Zebra , Animais , Peixe-Zebra/genética , Pseudo-Obstrução Intestinal/genética , Colo/anormalidades , Mutação , Bexiga Urinária/anormalidades , Anormalidades Múltiplas/genética , Proteínas Musculares/genética , Proteínas de Peixe-Zebra/genética
7.
Int J Mol Sci ; 25(5)2024 Feb 29.
Artigo em Inglês | MEDLINE | ID: mdl-38474067

RESUMO

Severe obesity in young children prompts for a differential diagnosis that includes syndromic conditions. Rapid-Onset Obesity with Hypothalamic Dysfunction, Hypoventilation, and Autonomic Dysregulation (ROHHAD) syndrome is a potentially fatal disorder characterized by rapid-onset obesity associated with hypoventilation, neural crest tumors, and endocrine and behavioral abnormalities. The etiology of ROHHAD syndrome remains to be established, but recent research has been focusing on autoimmunity. We report on a 2-year-old girl with rapid-onset obesity during the first year of life who progressed to hypoventilation and encephalitis in less than four months since the start of accelerated weight gain. The patient had a high titer of anti-ZSCAN1 antibodies (348; reference range < 40), and the increased values did not decline after acute phase treatment. Other encephalitis-related antibodies, such as the anti-NDMA antibody, were not detected. The rapid progression from obesity onset to central hypoventilation with encephalitis warns about the severe consequences of early-onset ROHHAD syndrome. These data indicate that serial measurements of anti-ZSCAN1 antibodies might be useful for the diagnosis and estimation of disease severity. Further research is needed to determine whether it can predict the clinical course of ROHHAD syndrome and whether there is any difference in antibody production between patients with and without tumors.


Assuntos
Neoplasias das Glândulas Suprarrenais , Doenças do Sistema Nervoso Autônomo , Encefalite , Doenças Hipotalâmicas , Obesidade Infantil , Feminino , Humanos , Pré-Escolar , Hipoventilação/complicações , Hipoventilação/diagnóstico , Obesidade Infantil/complicações , Neoplasias das Glândulas Suprarrenais/complicações , Síndrome , Encefalite/complicações
8.
Pediatr Int ; 65(1): e15666, 2023.
Artigo em Inglês | MEDLINE | ID: mdl-37888751

RESUMO

BACKGROUND: The aim of this study was to clarify the characteristics and outcomes of pediatric patients with solid pseudopapillary neoplasms (SPNs) who underwent pancreatectomy. METHODS: Pediatric patients with SPNs who underwent pancreatectomy at our institution between 1995 and 2020 were included in the study. RESULTS: During the period under review, 12 patients underwent pancreatectomy for SPNs (median age: 10 years; range: 6-15 years). The surgical procedures included pancreatoduodenectomy (n = 2; 16.6%), distal pancreatectomy (n = 3; 25%), and enucleation (n = 7; 58.3%). The most common postoperative complication was postoperative pancreatic fistula (n = 6; 50%). Patients who underwent enucleation tended to have higher postoperative complication rates compared with those who underwent other procedures. All patients were alive without recurrence at the end of the study period. CONCLUSIONS: SPN is associated with a good prognosis, regardless of the surgical procedure. If surgeons select enucleation for pediatric SPNs, they should bear in mind that it is associated with a higher complication rate.


Assuntos
Neoplasias Pancreáticas , Humanos , Criança , Neoplasias Pancreáticas/diagnóstico , Neoplasias Pancreáticas/cirurgia , Prognóstico , Pâncreas , Complicações Pós-Operatórias/epidemiologia , Complicações Pós-Operatórias/etiologia , Resultado do Tratamento , Estudos Retrospectivos
9.
Surg Today ; 2023 Sep 05.
Artigo em Inglês | MEDLINE | ID: mdl-37668735

RESUMO

Hirschsprung disease (HSCR) and its associated disorders (AD-HSCR) often result in severe hypoperistalsis caused by enteric neuropathy, mesenchymopathy, and myopathy. Notably, HSCR involving the small intestine, isolated hypoganglionosis, chronic idiopathic intestinal pseudo-obstruction, and megacystis-microcolon-intestinal hypoperistalsis syndrome carry a poor prognosis. Ultimately, small-bowel transplantation (SBTx) is necessary for refractory cases, but it is highly invasive and outcomes are less than optimal, despite advances in surgical techniques and management. Thus, regenerative therapy has come to light as a potential form of treatment involving regeneration of the enteric nervous system, mesenchyme, and smooth muscle in affected areas. We review the cutting-edge regenerative therapeutic approaches for managing HSCR and AD-HSCR, including the use of enteric nervous system progenitor cells, embryonic stem cells, induced pluripotent stem cells, and mesenchymal stem cells as cell sources, the recipient intestine's microenvironment, and transplantation methods. Perspectives on the future of these treatments are also discussed.

10.
Pediatr Surg Int ; 39(1): 286, 2023 Nov 02.
Artigo em Inglês | MEDLINE | ID: mdl-37919436

RESUMO

BACKGROUND/PURPOSE: Whether Roux-en-Y hepatic jejunectomy (HJ) or duct-to-duct biliary reconstruction (DD) is more useful in pediatric living donor liver transplantation has not yet been fully investigated. Therefore, to assess the feasibility and safety of DD, we compared the surgical outcomes of DD to HJ. METHODS: We divided 45 patients, excluding those with biliary atresia, into the DD group (n = 20) and the HJ group (n = 25), according to the type of biliary reconstruction they received. RESULTS: The 5-year survival rates (DD vs. HJ = 79.7% vs. 83.6%, p = 0.70) and the incidence of biliary complications, including bile leakage and stricture (DD vs. HJ = 1 [5.0%] vs. 1 [4.0%], p = 0.87) were not significantly different between the groups. However, intestinal complications, including bowel perforation or ileus, were significantly common in the HJ group (9/25 [36.0%]) than in the DD group (1/20 [5.0%]; p = 0.01). The three patients in the HJ group with intestinal perforation all suffered perforation at the anastomosed site in the Roux-en-Y procedure. The subgroup analysis showed the non-inferiority of DD to HJ for biliary or intestinal complications in patients weighting < 10 kg. CONCLUSION: With a proper selection of cases, DD should be a safe method for biliary reconstruction in pediatric recipients with little risk of biliary complications equivalent to HJ and a reduced risk of intestinal complications.


Assuntos
Doenças Biliares , Procedimentos Cirúrgicos do Sistema Biliar , Transplante de Fígado , Humanos , Criança , Transplante de Fígado/métodos , Doadores Vivos , Fígado/cirurgia , Anastomose em-Y de Roux/métodos , Doenças Biliares/cirurgia , Ductos Biliares/cirurgia , Procedimentos Cirúrgicos do Sistema Biliar/métodos , Anastomose Cirúrgica , Complicações Pós-Operatórias/etiologia , Estudos Retrospectivos
11.
J Surg Res ; 272: 61-68, 2022 04.
Artigo em Inglês | MEDLINE | ID: mdl-34936913

RESUMO

BACKGROUND AND OBJECTIVES: Mediastinal germ cell tumor (MGCT) is a relatively rare tumor. Complete resection after chemotherapy is a standard treatment against this disease. However, the risk factors of incomplete resection are unclear. Therefore, we analyzed survival rates and risk factors for incomplete resection based on preoperative imaging. METHODS: We retrospectively reviewed the medical records of patients (n = 56) with MGCT operated at National Cancer Center Hospital, and analyzed preoperative computed tomography (CT) data in terms of relationship of the tumor and vessels, and investigated survival rate and risk factors for incomplete resection. RESULTS: A total of 56 patients underwent resection of MGCT. The 5-y progression-free survival (PFS) and overall survival (OS) were 79% and 83%. In multivariate analysis, complete resection was the only significant prognostic factor for better PFS (hazard ratio (HR) = 9.083, P= 0.00021) and OS (HR = 5.519, P= 0.0445). The preoperative CT finding of arteries (including the aorta, right brachiocephalic artery, left common carotid artery, and left subclavian artery) surrounded by the tumor was a predictor of incomplete resection (odds ratio = 10.089, P= 0.049). CONCLUSIONS: Complete resection is essential for improving the survival of MGCT, and the risk stratification using preoperative CT imaging brings important information to achieve the complete resection.


Assuntos
Neoplasias do Mediastino , Neoplasias Embrionárias de Células Germinativas , Humanos , Neoplasias do Mediastino/diagnóstico por imagem , Neoplasias do Mediastino/cirurgia , Neoplasias Embrionárias de Células Germinativas/diagnóstico por imagem , Neoplasias Embrionárias de Células Germinativas/cirurgia , Prognóstico , Estudos Retrospectivos , Taxa de Sobrevida , Tomografia Computadorizada por Raios X/métodos
12.
J Pediatr Hematol Oncol ; 44(7): 393-397, 2022 10 01.
Artigo em Inglês | MEDLINE | ID: mdl-35091523

RESUMO

BACKGROUND: Pneumothorax and tumor-bronchial fistula are rare complications of pulmonary metastasis of osteosarcoma. OBSERVATIONS: We herein report the cases of 3 pediatric and adolescent patients who developed pneumothorax or tumor-bronchial fistula during treatment of pulmonary metastasis of osteosarcoma with chemotherapeutics or antiangiogenic agents. Two patients developed pneumothorax, and the other patient developed tumor-bronchial fistula. All of the patients finally underwent the surgery to treat their complications. CONCLUSIONS: Although it is not a curative surgery, surgery for pneumothorax and tumor-bronchial fistula is acceptable. The operative procedure should be considered on the basis of the predicted prognosis of the patient.


Assuntos
Neoplasias Ósseas , Fístula Brônquica , Neoplasias Pulmonares , Osteossarcoma , Pneumotórax , Adolescente , Inibidores da Angiogênese/uso terapêutico , Neoplasias Ósseas/complicações , Neoplasias Ósseas/tratamento farmacológico , Neoplasias Ósseas/cirurgia , Fístula Brônquica/complicações , Fístula Brônquica/cirurgia , Criança , Humanos , Neoplasias Pulmonares/secundário , Neoplasias Pulmonares/cirurgia , Osteossarcoma/tratamento farmacológico , Pneumotórax/complicações , Pneumotórax/cirurgia
13.
Pediatr Int ; 64(1): e15228, 2022 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-35912459

RESUMO

BACKGROUND: Core-needle biopsy (CNB) is used less frequently for the diagnosis of tumors in pediatric patients. In this report, the utility and safety of CNB for pediatric patients are described. METHODS: The medical records of patients who underwent CNB at the Department of Pediatric Surgery, Kyushu University Hospital from April 2020 to November 2021 were retrospectively reviewed. A 14 G or 16 G BARDMISSION Disposable Needle Instrument was used. For the diagnosis of neuroblastoma, a 14 G needle was selected; for the diagnosis of other tumors a 16 G needle was selected. RESULTS: During the above period 17 CNBs were performed in 17 patients, and the median patient age was 8 years (range, 15 days-19 years). The pathological diagnoses of the tumors were as follows: neuroblastoma, n = 6; lymphoma, n = 3; hepatoblastoma, n = 2; and others, n = 6. The quantity and quality of all tumor samples obtained by CNB was sufficient to make a diagnosis. The postoperative course after CNB was uneventful in most cases, with the exception of one case of hepatoblastoma (pseudoaneurysm). CONCLUSIONS: Core-needle biopsy is useful for pediatric patients. Sufficient tumor specimens were able to be obtained in all cases, irrespective of the type of tumor, and an accurate diagnosis could be made.


Assuntos
Hepatoblastoma , Neoplasias Hepáticas , Neuroblastoma , Humanos , Criança , Recém-Nascido , Estudos Retrospectivos , Biópsia com Agulha de Grande Calibre , Neuroblastoma/diagnóstico , Neuroblastoma/cirurgia
14.
Pediatr Surg Int ; 38(12): 1829-1838, 2022 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-36169671

RESUMO

PURPOSE: This study aims to clarify the relationship between changes in skeletal muscle mass during treatment and prognosis of pediatric malignant solid tumors. METHODS: Patients with pediatric malignant solid tumors who were treated at Kyushu University Hospital from 2007 to 2017 were divided into two groups: the progression-free survival (PFS) group and the relapse/death (R/D) group; the psoas major muscle volume (PMV) was then compared. We also measured the PMV and psoas muscle area (PMA) of pediatric patients with no complications who underwent surgery for acute appendicitis (control) and compared the values with those of patients with malignant tumors. RESULTS: No significant differences were observed in the PMV and PMA between patients with appendicitis and those with malignant tumors. Significant differences were found in the rate of change in PMV between the PFS (1.424) and R/D groups (1.071) (P = 0.0024). When the cut-off value of the rate of change in the PMV was 1.20, patients whose rate of change in PMV was ≥ 1.20 had longer PFS (P = 0.0231) and overall survival (P = 0.0229) than those whose rate of change was < 1.20. CONCLUSION: Pediatric patients with malignant solid tumors and increased skeletal muscle mass during treatment have a good prognosis.


Assuntos
Apendicite , Neoplasias , Sarcopenia , Humanos , Criança , Sarcopenia/complicações , Sarcopenia/patologia , Apendicite/complicações , Estudos Retrospectivos , Prognóstico , Músculos Psoas/diagnóstico por imagem , Músculos Psoas/patologia , Neoplasias/cirurgia , Neoplasias/complicações
15.
Pediatr Surg Int ; 38(12): 1855-1860, 2022 Dec.
Artigo em Inglês | MEDLINE | ID: mdl-36136118

RESUMO

PURPOSE: The number of accessible central veins (CVs) affects the prognosis of patients with intestinal failure (IF). The loss of residual CVs should be avoided. We, therefore, evaluated the efficacy of a new CV catheter-exchange technique using a subcutaneous fibrous sheath (FS) in pediatric IF patients. METHODS: We retrospectively collected the CV catheter (CVC) data of pediatric IF patients managed from January 2009 to December 2019. The data were divided into two groups; Groups 1 (CVCs placed with the FS method) and Group 2 (CVCs placed by the primary or another insertion). The main outcome was the CVC indwelling time. RESULTS: Eighty-five CVCs were analyzed. The FS method was attempted in 47 cases and succeeded in 40 (85%). No significant difference was observed between the groups regarding characteristics. A log-rank test revealed an equivalent CVC indwelling time between the two groups (Group 1: 268 [126-588] days vs. Group 2: 229 [126-387] days, p = 0.256). CONCLUSIONS: The FS method is highly recommended for pediatric IF patients, as its attempt showed a high success rate with an indwelling time equivalent to primary insertion. The FS method leads to the prolonged use of a single CV and thereby contributes to improving the outcomes of pediatric IF patients.


Assuntos
Infecções Relacionadas a Cateter , Cateterismo Venoso Central , Cateteres Venosos Centrais , Insuficiência Intestinal , Criança , Humanos , Cateterismo Venoso Central/métodos , Estudos Retrospectivos
16.
J Surg Res ; 236: 101-105, 2019 04.
Artigo em Inglês | MEDLINE | ID: mdl-30694741

RESUMO

PURPOSE: Sarcopenia is a syndrome that is defined by the loss of skeletal muscle mass, quality, and strength. In adult patients with malignancies, the presence of sarcopenia is known to be correlated with a poor prognosis; however, there have been no reports on the influence of sarcopenia on malignant tumors in pediatric patients. In the present study, we investigated whether or not sarcopenia affects the prognosis of high-risk neuroblastoma. MATERIALS AND METHODS: Thirteen patients with high-risk neuroblastoma who were treated according to the standard protocol at our hospital from 2007 to 2016 were divided into a progression-free survival group (n = 8) and a relapse/death group (n = 5). The rate of change in sarcopenia was calculated by comparing the psoas muscle area (PMA) of the L3-level lumbar spine on computed tomography before and after treatment with the standard protocol. The rate of change in the PMA, Kaup index, and serum albumin level were compared. Furthermore, we determined the cutoff rate of change in the PMA and compared the overall and progression-free survival. RESULTS: The rates of change in the PMA were 1.24 and 0.84 in the progression-free survival and relapse/death groups, respectively (P = 0.0472). There were no significant differences in the rates of change in the Kaup index or the serum albumin level of the two groups. The patients whose rate of change in the PMA was >1.00 showed a prolonged overall (P = 0.0078) and progression-free survival (P = 0.006). CONCLUSIONS: A decrease in the skeletal muscle mass was suggested to be a significant prognostic factor for high-risk neuroblastoma.


Assuntos
Recidiva Local de Neoplasia/diagnóstico , Neuroblastoma/mortalidade , Sarcopenia/diagnóstico , Pré-Escolar , Feminino , Humanos , Lactente , Recém-Nascido , Masculino , Recidiva Local de Neoplasia/epidemiologia , Recidiva Local de Neoplasia/prevenção & controle , Neuroblastoma/complicações , Neuroblastoma/terapia , Prognóstico , Intervalo Livre de Progressão , Músculos Psoas/diagnóstico por imagem , Músculos Psoas/patologia , Estudos Retrospectivos , Fatores de Risco , Sarcopenia/etiologia , Sarcopenia/mortalidade , Análise de Sobrevida , Tomografia Computadorizada por Raios X
17.
Pediatr Surg Int ; 35(5): 551-557, 2019 May.
Artigo em Inglês | MEDLINE | ID: mdl-30778701

RESUMO

BACKGROUND: Technology for detecting liver tumors and identifying the bile ducts using indocyanine green (ICG) has recently been developed. However, the usefulness and limitations of ICG navigation surgery for hepatoblastoma (HB) have not been fully clarified. We herein report our experiences with surgical navigation using ICG for in HB patients. METHODS: In 5 HB patients, 10 ICG navigation surgeries were performed using a 10-mm infrared fluorescence imaging scope after the injection of 0.5 mg/kg ICG intravenously. The surgical and clinical features were collected retrospectively. RESULTS: Navigation surgery using ICG was performed for primary liver tumors in 4 cases, and the timing of ICG injection was 90.5 ± 33.7 h before the operation. All tumors exhibited intense fluorescence from the liver surface. ICG navigation for the primary liver tumor was useful for detecting the residual tumor at the stump and invasion to the diaphragm during surgery. Six lung surgeries using ICG navigation were performed. The timing of ICG injection was 21.8 ± 3.4 h before the operation. The size of the metastatic tumor was 7.4 ± 4.1 mm (1.2-15 mm). Of 11 metastatic tumors detected by computed tomography (CT), 10-including the smallest tumor (1.2 mm)-were able to be detected by ICG from the lung surface. The depth of the 10 ICG-positive tumors from the lung surface was 0.9 ± 1.9 mm (0-6 mm), and the depth of the single ICG-negative tumor was 12 mm. One lesion not detected by CT showed ICG false positivity. CONCLUSION: Navigation surgery using ICG for patients with HB was useful for identifying tumors and confirming complete resection. However, in ICG navigation surgery, we must be aware of the limitations with regard to the tumor size and the depth from the surface.


Assuntos
Hepatoblastoma/diagnóstico por imagem , Hepatoblastoma/cirurgia , Aumento da Imagem/métodos , Verde de Indocianina/farmacocinética , Neoplasias Hepáticas/diagnóstico por imagem , Neoplasias Hepáticas/cirurgia , Adulto , Criança , Corantes/farmacocinética , Feminino , Humanos , Fígado/diagnóstico por imagem , Fígado/cirurgia , Masculino , Imagem Óptica/métodos , Reprodutibilidade dos Testes , Estudos Retrospectivos
18.
Biochem Biophys Res Commun ; 503(3): 1666-1673, 2018 09 10.
Artigo em Inglês | MEDLINE | ID: mdl-30054041

RESUMO

The mechanism underlying the spontaneous regression of neuroblastoma is unclear. Although it was hypothesized that this regression occurs via an immunological mechanism, there is no clinical evidence, and no animal models have been developed to investigate the involvement of immune systems, especially natural antibodies, against neuroblastoma. We performed an immunological analysis of homo- and heterozygous TH-MYCN transgenic mice as a model of aggressive neuroblastoma. Mice with no or small (<5 mm) tumors showed higher antibody titers in plasma than mice with large (>5 mm) tumors. A significant negative correlation was observed between the tumor diameter and the titer of antitumor antibody. This antibody had complement-dependent cytotoxicity but not antibody-dependent cellular cytotoxicity against neuroblastoma cells. Moreover, B-cell depletion had no effect on the tumor incidence in vivo. We revealed that TH-MYCN transgenic mice have a natural antibody against neuroblastoma that correlate with tumor size. However, this antibody does not correlate with the spontaneous regression of neuroblastoma. Thus, the function of the natural antibody is limited.


Assuntos
Anticorpos/imunologia , Produtos Biológicos/imunologia , Proteína Proto-Oncogênica N-Myc/imunologia , Neuroblastoma/imunologia , Animais , Anticorpos/química , Produtos Biológicos/química , Humanos , Camundongos , Camundongos Transgênicos , Proteína Proto-Oncogênica N-Myc/genética , Neuroblastoma/patologia , Células Tumorais Cultivadas
19.
Pathol Res Pract ; 258: 155348, 2024 Jun.
Artigo em Inglês | MEDLINE | ID: mdl-38761648

RESUMO

Hepatoblastoma (HB) is the most common malignant liver tumor in childhood. Although pre-operative cisplatin (CDDP)-based chemotherapy is often used in cases of HB, about 20% of HB patients exhibit resistance to CDDP. Forkhead box protein M1 (FOXM1) and chromo-domain-helicase-DNA-binding protein 4 (CHD4) have been associated with CDDP resistance in various tumors. We here analyzed the immunohistochemical expression of FOXM1 and CHD4 in HB specimens of 33 patients (mean age: 20 months) post-chemotherapy. The differentiation of specimens was assessed using the digital pathology software QuPath®, and then the relation between the FOXM1 or CHD4 expression and the differentiation and various other clinicopathological parameters was investigated. The histological type was epithelial in 19 cases (57.6%) and mixed epithelial and mesenchymal in 14 cases (42.4%). Nine cases had only a fetal component, 1 case had only an embryonal component, 22 cases had both fetal and embryonal components, and 1 case had no viable tumor. Both the FOXM1 and CHD4 immunoexpressions were found significantly more frequently in the embryonal than fetal components (p<0.0001 and p<0.0001, respectively). Regarding chemotherapy efficacy, the alpha-fetoprotein (AFP) level after chemotherapy was correlated with both the imaging shrinkage rate (R=-0.52) and histological residual rate (the percentage of the viable tumors of HB after chemotherapy)(R=0.62). High FOXM1 score was correlated with a high-postoperative AFP value (p<0.01) and a low AFP attenuation rate (p<0.05), but the FOXM1 score was not correlated with the imaging shrinkage rate (p=0.4418) or histological residual rate (p=0.4418). High CHD4 score showed a nonsignificant trend toward correlation with high postoperative AFP value (p=0.0849) and was not significantly correlated with the other parameters. Collectively, our results showed that FOXM1 expression may be useful in evaluating the response to CDDP-based chemotherapeutic regimens. Accurate measurement of FOXM1 expression by our scoring system using QuPath® is important in cases with mixed HB components of various differentiation levels.


Assuntos
Cisplatino , Resistencia a Medicamentos Antineoplásicos , Proteína Forkhead Box M1 , Hepatoblastoma , Neoplasias Hepáticas , Complexo Mi-2 de Remodelação de Nucleossomo e Desacetilase , Humanos , Proteína Forkhead Box M1/metabolismo , Hepatoblastoma/patologia , Hepatoblastoma/tratamento farmacológico , Hepatoblastoma/metabolismo , Masculino , Feminino , Neoplasias Hepáticas/patologia , Neoplasias Hepáticas/tratamento farmacológico , Neoplasias Hepáticas/metabolismo , Lactente , Cisplatino/uso terapêutico , Complexo Mi-2 de Remodelação de Nucleossomo e Desacetilase/metabolismo , Pré-Escolar , Biomarcadores Tumorais/metabolismo , Biomarcadores Tumorais/análise , Antineoplásicos/uso terapêutico , Criança
20.
Am J Surg Pathol ; 48(7): 803-812, 2024 Jul 01.
Artigo em Inglês | MEDLINE | ID: mdl-38726836

RESUMO

Isolated hypoganglionosis (IHG) is histologically characterized by small numbers of myenteric ganglion cells and small myenteric ganglia; however, no numerical diagnostic criteria for IHG have been established. Therefore, this study aimed to develop quantitative pathologic criteria for IHG. We evaluated 160 resected intestinal tissue specimens from 29 pediatric autopsies and 10 IHG cases. These specimens were obtained from the jejunum, ileum, ascending colon, transverse colon, and rectum. Morphologic features of the myenteric ganglion cells and myenteric ganglia were quantified and analyzed in digitized HuC/HuD-immunostained and CD56-immunostained sections, respectively. Quantitative criteria were developed with a scoring system that used parameters with the area under the receiver operating characteristic curve (AUC) values >0.7 and sensitivity and specificity exceeding 70%. The selected parameters were the number of myenteric ganglion cells per cm and the number of myenteric ganglia with an area >2500 µm 2 per cm. The score for each parameter ranged from -1 to 2, and the total score of the scoring system ranged from -2 to 4. With a cutoff value of ≥2 (AUC, 0.98; 95% CI: 0.96-1.00), the scoring system had a sensitivity of 96% (95% CI: 0.82-1.00) and a specificity of 99% (95% CI: 0.95-1.00). We devised a novel pathologic criterion based on the quantification of the number of myenteric ganglion cells and ganglia. Furthermore, this criterion showed high diagnostic accuracy and could lead to a definitive diagnosis of IHG in clinical practice.


Assuntos
Plexo Mientérico , Humanos , Masculino , Feminino , Plexo Mientérico/patologia , Plexo Mientérico/química , Lactente , Pré-Escolar , Criança , Valor Preditivo dos Testes , Imuno-Histoquímica , Recém-Nascido , Autopsia , Biomarcadores/análise , Curva ROC , Adolescente , Contagem de Células , Área Sob a Curva
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