Clinical characteristics, late effects and outcomes in pineoblastomas in children: a single center experience

Görgün, Ömer; Koç, Başak; Kebudi, Rejin; Wolff, Johannes; Kebudi, Abut; Darendeliler, Emin

doi:10.24953/turkjped.2021.06.002

Cited by 4 publications

(5 citation statements)

References 30 publications

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“…The prognosis of pinealoblastomas remains severe with an high rate of mortality as observed in our series of 18 pediatric cases treated in the last twenty years and specially in very young children con rming that age inferior to three years old represent a bad prognostic factor also in case of complete removal [20,21].…”

Section: Discussionsupporting

confidence: 56%

“…A better strati cation of patients should allow tailored treatments, especially for young children, and could represent the evolution of therapeutical strategies in a next future as preconized in literature. [17] The PPT -ID are more frequent in adolescents and young adults patients in age of 33 years old and except for grade II with a total removal they need a complementary treatment to improve the survival [6,21].…”

Section: Discussionmentioning

confidence: 99%

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Results of the treatment of pineal tumors in children: the Lyon experience

Szathmari

Beuriat²,

Vasiljevic³

et al. 2022

Childs Nerv Syst

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Pineal tumors are rare and their incidence is of 1% among all pediatric tumors of the central nervous system. The survival depends on the histology, the extension of the surgical removal, the e cacy of the complementary treatment (chemotherapy and cranio-spinal irradiation) in relation with the age of the children.We report our experience in Lyon concerning 151 patients treated from 1997 to 2021. All patients were recorded in the French Register of Pineal tumors which is centralized in Lyon since 2010.The analysis shows that benign tumors have a good survival rate and a total surgical removal is manadatory. Concerning true pineal tumors, pinealoblastomas have a bad prognostic especially in children aged inferior to three years old. The new pathological classi cation allows a better strati cation with different groups identi ed and also the identi cation of a DICER 1 syndrome in family of patients with a pinealoblastoma that need medical investigations. Results of Germ Cell Tumors are more favorable with a global survival rate of 87 % and a rate of survival for pure germinomas of almost 97%.Results for gliomas of the pineal gland, that are tectal plate gliomas, are good for pilocytic astrocytomas while for other gliomas the prognosis is related to the grade of malignancy and the e cacy of complementary treatment. Papillary tumors need a complete removal for the best chance of survival.Finally, AT/TR still have a bad prognosis.Our results show that many progresses have to be accomplished for pineal region tumors and that these pathologies need a multidisciplinary approach to choose the best treatment to improve the survival rate and the patients' quality of life.Out 891 tumors 316 patients (33%) are children: 165 of the 316 patients (54%) have been treated in different French centers and 151(46%) have been treated in Lyon and are considered for this report.Results of the French register were already published in a previous paper. [6] Materials And Methods All pediatric patients that have been treated from January 1997 to December 2020 in Lyon in the Department of Pediatric Neurosurgery have been considered. All these patients are recorded in the French Register of Pineal Tumors. 151 patients were eligible for the study with an age between one years to eighteen years old. The sex ratio was of 1.2 males /Girl.The symptomatology was characterized in 54% by signs of intracranial hypertension with headaches and vomiting. Ophthalmological troubles were present in 21% of patients, endocrinological problems were associated in 5% of cases, while in others 18% of patients the clinical manifestations were characterized by more insidious manifestations as asthenia, tiredness and gradual lowering of scholar performances.All patients were studied with a cerebro-spinal MRI with and without Gadolinium with angio-MRI sequences to study arterial feeders and the venous structures. The CT-scan was realized in some patients performed in peripheral hospitals. We never realized angiography by the Seldinger technique for the surgical p...

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Section: Discussionsupporting

confidence: 56%

Section: Discussionmentioning

confidence: 99%

Results of the treatment of pineal tumors in children: the Lyon experience

Szathmari

Beuriat²,

Vasiljevic³

et al. 2022

Childs Nerv Syst

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“…Of the 2701 articles retrieved in the search, 44 studies with IPD on 298 patients with pineoblastoma were included in the systematic review ( Table 1 ) [ 5 , 6 , 8 , 9 , 10 , 11 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 , 34 , 35 , 36 , 37 , 38 , 39 , 40 , 41 , 42 , 43 , 44 , 45 , 46 , 47 , 48 , 49 , 50 , 51 , 52 , 53 , 54 , 55 , 56 , 57 , 58 , 59 , 60 , 61 ]. Figure 1 presents the PRISMA flowchart for this systematic review [ 18 ].…”

Section: Resultsmentioning

confidence: 99%

Post-Surgical Prognosis of Patients with Pineoblastoma: A Systematic Review and Individual Patient Data Analysis with Trends over Time

Nandoliya,

Sadagopan,

Thirunavu

et al. 2023

Cancers

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Most of the literature on pineoblastoma consists of case reports and single-institution series. The goal of this systematic review and individual patient data (IPD) analysis was to summarize the existing literature, identify factors associated with overall survival (OS), and provide a contemporary update on prognosis for patients with pineoblastoma. Forty-four studies were identified with 298 patients having IPD. Kaplan–Meier analyses were used to report survival outcomes based on age, tumor metastases, extent of resection (EOR), adjuvant therapy, and publication year. Cox regression was performed to identify independent predictors of time to mortality. Multivariable recursive partitioning analysis was used to identify the most important subgroups associated with mortality. Patients were classified based on publication year before and after the last systematic review on this topic (pre-2012 and 2012 onwards) and compared using univariate and multivariable analyses. This study demonstrates that EOR less-than-gross total resection, metastatic presentation, adjuvant chemotherapy without radiation, and tumor presentation in children less than three years old are associated with poorer prognosis. Since 2012, the 5-year actuarial OS has improved from 32.8% to 56.1%, which remained significant even after accounting for EOR, age, and adjuvant therapy. Pineoblastoma remains a severe rare disease, but survival outcomes are improving.

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“…Pineal parenchymal tumors of intermediate differentiation are often considered as a part of spectrum of grade II and III pineal parenchymal tumors [28,29]. Although there is yet to be a grading criterion for differentiating PPTIDs.The WHO classification of CNS tumors considers that an approach to classification/stratification adopted by Jouvet et al based mainly on two criteria, mitoses index and immunoresponse to neurofilament protein., with less immunoresponse in higher grade tumors [5]. PPTIDs show moderate cellularity, mildmoderate atypical nuclei, low-moderate mitosis, occasional Homer Wright rosettes, lack of small, primitive appearance, and necrosis.…”

Section: Papillary Tumorsmentioning

confidence: 97%

“…There exist a few histopathological subtypes of primary pineal body tumors with varying level of differentiations, thus requiring a multi-disciplinary approach in the management [3,4]. There is evidence of varying response to therapy depending on multiple factors with fairly good prognosis for some of the subtypes [4,5].…”

Section: Introductionmentioning

confidence: 99%

Pineal Body Tumor: An Overview of the Pathophysiology

Glory

Daniel

Favour

et al. 2022

AJMAH

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The pineal gland is a small endocrine gland in the brain that regulates the circadian rhythm in humans. It is responsible for melatonin production, also produced by parenchymal and glial cells. Pineal region tumors account for 3–11% of pediatric brain tumors, and 1% of adult brain tumors according to World health Organization (WHO). These tumors arise from the germ cells, pineal cells, and adjacent structures. It is fundamental for medical knowledge (clinical and laboratory) to differentiate and identify the various types of pineal gland tumors and thus facilitate accurate diagnosis with crafted therapeutic management of the pathology accompanying its incidence. There exist different histological subtypes of pineal body tumors and various management options like surgery, chemotherapy and radiotherapy. The review article is a student’s project on integrated learning, aiming at understanding the pathophysiology of the rare pineal body tumor.

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Clinical characteristics, late effects and outcomes in pineoblastomas in children: a single center experience

Cited by 4 publications

References 30 publications

Results of the treatment of pineal tumors in children: the Lyon experience

Results of the treatment of pineal tumors in children: the Lyon experience

Post-Surgical Prognosis of Patients with Pineoblastoma: A Systematic Review and Individual Patient Data Analysis with Trends over Time

Pineal Body Tumor: An Overview of the Pathophysiology

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