Imaging response assessment for CNS germ cell tumours: consensus recommendations from the European Society for Paediatric Oncology Brain Tumour Group and North American Children's Oncology Group

Morana, Giovanni; Shaw, Dennis; MacDonald, Shannon M.; Alapetite, Claire; Ajithkumar, Thankamma; Bhatia, Aashim; Brisse, H.; Jaimes, Camilo; Czech, Thomas; Dhall, Girish; Fangusaro, Jason; Faure‐Conter, Cécile; Fouladi, Maryam; Hargrave, Darren; Harreld, Julie H.; Mitra, Dipayan; Nicholson, James C.; Souweidane, Mark M.; Timmermann, Beate; Calaminus, Gabriele; Bartels, Ute; Bison, Brigitte; Murray, Matthew J.

doi:10.1016/s1470-2045(22)00063-8

Cited by 8 publications

(3 citation statements)

References 56 publications

(88 reference statements)

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“…Consensus recommendations set a cut off of the diameter of the inferior pituitary stalk of 3 mm [ 19 , 20 ]. Our study revealed that 27 out of the 64 (42.2%) measurable inferior stalks had a diameter within that range indicating that this is not a reliable indicator for early tumor detection.…”

Section: Discussionmentioning

confidence: 99%

“…Regarding bifocal GCT, it is important to note the recently published consensus recommendation on GCT in this context. When central diabetes insipidus is present with the absence of the PPBS on MRI, and the pituitary stalk or median eminence is not thickened, patients with pineal tumors and negative serum and cerebrospinal fluid α-fetoprotein and human chorionic gonadotropin markers should be considered as having sufficient evidence of bifocal germinoma [ 20 ].…”

Section: Discussionmentioning

confidence: 99%

“…According to current literature, there are no established limits for the thickness of the pituitary stalk in children. Since our cohort mainly consists of children, we have adhered to consensus recommendations, setting the cut-off at 3 mm [ 19 , 20 ]. Therefore, it was further evaluated how often the diameter did not exceed 3 mm.…”

Section: Methodsmentioning

confidence: 99%

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Imaging in malignant germ cell tumors involving the hypothalamo-neurohypophyseal axis: the evaluation of the posterior pituitary bright spot is essential

Stock,

Calaminus,

Weisthoff

et al. 2024

Neuroradiology

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Purpose Malignant intracranial germ cell tumors (GCTs) are rare diseases in Western countries. They arise in midline structures and diagnosis is often delayed. We evaluated imaging characteristics and early tumor signs of suprasellar and bifocal GCT on MRI. Methods Patients with the diagnosis of a germinoma or non-germinomatous GCT (NGGCT) who received non-contrast sagittal T1WI on MRI pre-therapy were included. Loss of the posterior pituitary bright spot (PPBS), the expansion and size of the tumor, and the expansion and infiltration of surrounding structures were evaluated. Group comparison for histologies and localizations was performed. Results A total of 102 GCT patients (median age at diagnosis 12.3 years, range 4.4–33.8; 57 males; 67 in suprasellar localization) were enrolled in the study. In the suprasellar cohort, NGGCTs (n = 20) were noticeably larger than germinomas (n = 47; p < .001). Each tumor showed involvement of the posterior lobe or pituitary stalk. A PPBS loss (total n = 98) was observed for each localization and entity in more than 90% and was related to diabetes insipidus. Osseous infiltration was observed exclusively in suprasellar GCT (significantly more frequent in NGGCT; p = .004). Time between the first MRI and therapy start was significantly longer in the suprasellar cohort (p = .005), with an even greater delay in germinoma compared to NGGCT (p = .002). The longest interval to treatment had circumscribed suprasellar germinomas (median 312 days). Conclusion A loss of the PPBS is a hint of tumor origin revealing small tumors in the neurohypophysis. Using this sign in children with diabetes insipidus avoids a delay in diagnosis.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Imaging in malignant germ cell tumors involving the hypothalamo-neurohypophyseal axis: the evaluation of the posterior pituitary bright spot is essential

Stock,

Calaminus,

Weisthoff

et al. 2024

Neuroradiology

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Monitoring pediatric CNS non‐germinomatous germ cell tumors via cerebrospinal fluid circulating tumor DNA

Zhang,

Jin,

Zhong

et al. 2024

Pediatric Blood & Cancer

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BackgroundAccurate molecular and clinical stratification of patients with central nervous system (CNS) non‐germinomatous germ cell tumors (NGGCTs) remains challenging, impeding the development of personalized therapeutic approaches. Herein, we investigated the translational significance of cerebrospinal fluid (CSF) circulating tumor DNA (ctDNA) in pediatric NGGCTs to identify characteristic features of CNS NGGCTs and to identify a subset of patients for whom the presence of residual disease is a risk factor and an indicator of shorter progression‐free survival (PFS) and overall survival (OS).MethodsMedical records of patients with CNS NGGCTs between January 1, 2018 and December 31, 2022 were reviewed retrospectively.ResultsThe cohort consisted of 11 male and six female patients. Tumor markers were elevated in four of the five people who underwent surgery. The remaining 12 patients were diagnosed with malignant NGGCTs according to elevated tumor markers. Among them, ctDNA before chemotherapy as well as ctDNA clearance were consistently associated with PFS and OS (p < .05). By setting a ctDNA positivity threshold of 6%, patients with high ctDNA (above the threshold) levels, which had limitation due to the selection based on optimal statistic from the survival analysis, had significantly inferior 5‐year PFS and OS compared to those with low levels (below the threshold). ctDNA or ctDNA clearance combined with the presence of residual disease predicted significantly worse OS and PFS (p < .05).ConclusionsCSF ctDNA might allow the study of genomic evolution and the characterization of tumors in pediatric NGGCTs. CSF ctDNA analysis may facilitate the clinical management of pediatric NGGCT patients, and aid in designing personalized therapeutic strategies.

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Time to dismiss boost? Outcomes of children with localized and metastatic germinoma

et al. 2023

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Purpose: To determine long-term outcomes of a cohort of children with germinoma treated with chemotherapy and radiation therapy without primary tumor boost even in the absence of complete response to chemotherapy.Methods: This retrospective study analyzed the outcome of patients with germinoma consecutively diagnosed and treated at a tertiary care center from January 2000 to December 2021. MRIs were reviewed by two radiologists, blinded to patient data. Tumor location at diagnosis, tumor response to chemotherapy and at completion of radiation therapy and site of relapse were assessed. Tumor response was assessed radiologically by determining the tumor size and response on diffusion-weighted imaging, in addition to biochemical, cytological parameters and neurological status.Results: Of 46 pediatric germinoma patients, 29 children (14 male; median age 12.8 years) received no primary tumor boost. Median follow-up was 63 months (range 9-187 months). Twenty-ve children had localized disease and tumor location was suprasellar (n= 11), pineal (n= 10), bifocal (n= 3) and basal ganglia (n= 1) while 4 children had metastatic disease at presentation. All patients completed multi-agent chemotherapy followed by either ventricular irradiation (VI) (23.4 Gy) (n = 23), whole brain (WBI) (23.4 Gy) (n = 5) or craniospinal radiation (CSI) (23.4 Gy) (n= 1). Two children, who had localized disease at presentation and received VI after chemotherapy, relapsed 9 months and 32 months after completion of treatment respectively. No patient had a local relapse. Location of relapse was distant, outside (n= 1) and inside (n= 1) the irradiation eld. Five-year PFS was 91% and overall survival (OS) was 100%.Conclusions: In this case series, excellent 5-year PFS and OS rates were achieved with chemotherapy followed by radiation therapy of 23.4 Gy delivered without primary tumor boost. No local relapse was observed despite omitting primary tumor boost in patients with localized and metastatic germinoma.

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Imaging response assessment for CNS germ cell tumours: consensus recommendations from the European Society for Paediatric Oncology Brain Tumour Group and North American Children's Oncology Group

Cited by 8 publications

References 56 publications

Imaging in malignant germ cell tumors involving the hypothalamo-neurohypophyseal axis: the evaluation of the posterior pituitary bright spot is essential

Imaging in malignant germ cell tumors involving the hypothalamo-neurohypophyseal axis: the evaluation of the posterior pituitary bright spot is essential

Monitoring pediatric CNS non‐germinomatous germ cell tumors via cerebrospinal fluid circulating tumor DNA

Time to dismiss boost? Outcomes of children with localized and metastatic germinoma

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