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Pineal anlage tumor: clinical and diagnostic features, and rationales for treatment

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Pineal anlage tumor: clinical and diagnostic features, and rationales for treatment. / Obrecht-Sturm, Denise; Pfaff, Elke; Mynarek, Martin; Bison, Brigitte; Rodehüser, Martina; Becker, Martina; Kietz, Silke; Pfister, Stefan M; Jones, David T; Sturm, Dominik; von Deimling, Andreas; Sahm, Felix; Kortmann, Rolf-Dieter; Schwarz, Rudolf; Pietsch, Torsten; Fleischhack, Gudrun; Rutkowski, Stefan.

In: J NEURO-ONCOL, Vol. 166, No. 2, 22.01.2024, p. 359-368.

Research output: SCORING: Contribution to journal › Case report › Research › peer-review

Harvard

Obrecht-Sturm, D, Pfaff, E, Mynarek, M, Bison, B, Rodehüser, M, Becker, M, Kietz, S, Pfister, SM, Jones, DT, Sturm, D, von Deimling, A, Sahm, F, Kortmann, R-D, Schwarz, R, Pietsch, T, Fleischhack, G & Rutkowski, S 2024, 'Pineal anlage tumor: clinical and diagnostic features, and rationales for treatment', J NEURO-ONCOL, vol. 166, no. 2, pp. 359-368. https://doi.org/10.1007/s11060-023-04547-5

APA

Obrecht-Sturm, D., Pfaff, E., Mynarek, M., Bison, B., Rodehüser, M., Becker, M., Kietz, S., Pfister, S. M., Jones, D. T., Sturm, D., von Deimling, A., Sahm, F., Kortmann, R-D., Schwarz, R., Pietsch, T., Fleischhack, G., & Rutkowski, S. (2024). Pineal anlage tumor: clinical and diagnostic features, and rationales for treatment. J NEURO-ONCOL, 166(2), 359-368. https://doi.org/10.1007/s11060-023-04547-5

Vancouver

Obrecht-Sturm D, Pfaff E, Mynarek M, Bison B, Rodehüser M, Becker M et al. Pineal anlage tumor: clinical and diagnostic features, and rationales for treatment. J NEURO-ONCOL. 2024 Jan 22;166(2):359-368. https://doi.org/10.1007/s11060-023-04547-5

Bibtex

@article{dc317ac10b704b0e97c363d9c651a906,
title = "Pineal anlage tumor: clinical and diagnostic features, and rationales for treatment",
abstract = "PURPOSE: To provide a treatment-focused review and develop basic treatment guidelines for patients diagnosed with pineal anlage tumor (PAT).METHODS: Prospectively collected data of three patients with pineal anlage tumor from Germany was combined with clinical details and treatment information from 17 published cases.RESULTS: Overall, 20 cases of PAT were identified (3 not previously reported German cases, 17 cases from published reports). Age at diagnosis ranged from 0.3 to 35.0 (median: 3.2 ± 7.8) years. All but three cases were diagnosed before the age of three years. For three cases, metastatic disease at initial staging was described. All patients underwent tumor surgery (gross-total resection: 9, subtotal resection/biopsy: 9, extent of resection unknown: 2). 15/20 patients were alive at last follow-up. Median follow-up for 10/15 surviving patients with available follow-up and treatment data was 2.4 years (0.3-6.5). Relapse was reported for 3 patients within 0.8 years after diagnosis. Five patients died, 3 after relapse and 2 from early postoperative complications. Two-year-progression-free- and -overall survival were 65.2 ± 12.7% and 49.2 ± 18.2%, respectively. All 4 patients who received intensive chemotherapy including high-dose chemotherapy combined with radiotherapy (2 focal, 2 craniospinal [CSI]) had no recurrence. Focal radiotherapy- and CSI-free survival rates in 13 evaluable patients were 46.2% (6/13) and 61.5% (8/13), respectively.CONCLUSION: PAT is an aggressive disease mostly affecting young children. Therefore, adjuvant therapy using intensive chemotherapy and considering radiotherapy appears to comprise an appropriate treatment strategy. Reporting further cases is crucial to evaluate distinct treatment strategies.",
keywords = "Adolescent, Adult, Child, Child, Preschool, Humans, Infant, Young Adult, Brain Neoplasms/diagnosis, Neoplasm Recurrence, Local/pathology, Pineal Gland/surgery, Pinealoma/diagnosis, Recurrence, Supratentorial Neoplasms/pathology, Treatment Outcome",
author = "Denise Obrecht-Sturm and Elke Pfaff and Martin Mynarek and Brigitte Bison and Martina Rodeh{\"u}ser and Martina Becker and Silke Kietz and Pfister, {Stefan M} and Jones, {David T} and Dominik Sturm and {von Deimling}, Andreas and Felix Sahm and Rolf-Dieter Kortmann and Rudolf Schwarz and Torsten Pietsch and Gudrun Fleischhack and Stefan Rutkowski",
note = "{\textcopyright} 2024. The Author(s).",
year = "2024",
month = jan,
day = "22",
doi = "10.1007/s11060-023-04547-5",
language = "English",
volume = "166",
pages = "359--368",
journal = "J NEURO-ONCOL",
issn = "0167-594X",
publisher = "Kluwer Academic Publishers",
number = "2",

}

RIS

TY - JOUR

T1 - Pineal anlage tumor: clinical and diagnostic features, and rationales for treatment

AU - Obrecht-Sturm, Denise

AU - Pfaff, Elke

AU - Mynarek, Martin

AU - Bison, Brigitte

AU - Rodehüser, Martina

AU - Becker, Martina

AU - Kietz, Silke

AU - Pfister, Stefan M

AU - Jones, David T

AU - Sturm, Dominik

AU - von Deimling, Andreas

AU - Sahm, Felix

AU - Kortmann, Rolf-Dieter

AU - Schwarz, Rudolf

AU - Pietsch, Torsten

AU - Fleischhack, Gudrun

AU - Rutkowski, Stefan

N1 - © 2024. The Author(s).

PY - 2024/1/22

Y1 - 2024/1/22

N2 - PURPOSE: To provide a treatment-focused review and develop basic treatment guidelines for patients diagnosed with pineal anlage tumor (PAT).METHODS: Prospectively collected data of three patients with pineal anlage tumor from Germany was combined with clinical details and treatment information from 17 published cases.RESULTS: Overall, 20 cases of PAT were identified (3 not previously reported German cases, 17 cases from published reports). Age at diagnosis ranged from 0.3 to 35.0 (median: 3.2 ± 7.8) years. All but three cases were diagnosed before the age of three years. For three cases, metastatic disease at initial staging was described. All patients underwent tumor surgery (gross-total resection: 9, subtotal resection/biopsy: 9, extent of resection unknown: 2). 15/20 patients were alive at last follow-up. Median follow-up for 10/15 surviving patients with available follow-up and treatment data was 2.4 years (0.3-6.5). Relapse was reported for 3 patients within 0.8 years after diagnosis. Five patients died, 3 after relapse and 2 from early postoperative complications. Two-year-progression-free- and -overall survival were 65.2 ± 12.7% and 49.2 ± 18.2%, respectively. All 4 patients who received intensive chemotherapy including high-dose chemotherapy combined with radiotherapy (2 focal, 2 craniospinal [CSI]) had no recurrence. Focal radiotherapy- and CSI-free survival rates in 13 evaluable patients were 46.2% (6/13) and 61.5% (8/13), respectively.CONCLUSION: PAT is an aggressive disease mostly affecting young children. Therefore, adjuvant therapy using intensive chemotherapy and considering radiotherapy appears to comprise an appropriate treatment strategy. Reporting further cases is crucial to evaluate distinct treatment strategies.

AB - PURPOSE: To provide a treatment-focused review and develop basic treatment guidelines for patients diagnosed with pineal anlage tumor (PAT).METHODS: Prospectively collected data of three patients with pineal anlage tumor from Germany was combined with clinical details and treatment information from 17 published cases.RESULTS: Overall, 20 cases of PAT were identified (3 not previously reported German cases, 17 cases from published reports). Age at diagnosis ranged from 0.3 to 35.0 (median: 3.2 ± 7.8) years. All but three cases were diagnosed before the age of three years. For three cases, metastatic disease at initial staging was described. All patients underwent tumor surgery (gross-total resection: 9, subtotal resection/biopsy: 9, extent of resection unknown: 2). 15/20 patients were alive at last follow-up. Median follow-up for 10/15 surviving patients with available follow-up and treatment data was 2.4 years (0.3-6.5). Relapse was reported for 3 patients within 0.8 years after diagnosis. Five patients died, 3 after relapse and 2 from early postoperative complications. Two-year-progression-free- and -overall survival were 65.2 ± 12.7% and 49.2 ± 18.2%, respectively. All 4 patients who received intensive chemotherapy including high-dose chemotherapy combined with radiotherapy (2 focal, 2 craniospinal [CSI]) had no recurrence. Focal radiotherapy- and CSI-free survival rates in 13 evaluable patients were 46.2% (6/13) and 61.5% (8/13), respectively.CONCLUSION: PAT is an aggressive disease mostly affecting young children. Therefore, adjuvant therapy using intensive chemotherapy and considering radiotherapy appears to comprise an appropriate treatment strategy. Reporting further cases is crucial to evaluate distinct treatment strategies.

KW - Adolescent

KW - Adult

KW - Child

KW - Child, Preschool

KW - Humans

KW - Infant

KW - Young Adult

KW - Brain Neoplasms/diagnosis

KW - Neoplasm Recurrence, Local/pathology

KW - Pineal Gland/surgery

KW - Pinealoma/diagnosis

KW - Recurrence

KW - Supratentorial Neoplasms/pathology

KW - Treatment Outcome

U2 - 10.1007/s11060-023-04547-5

DO - 10.1007/s11060-023-04547-5

M3 - Case report

C2 - 38253790

VL - 166

SP - 359

EP - 368

JO - J NEURO-ONCOL

JF - J NEURO-ONCOL

SN - 0167-594X

IS - 2

ER -