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Background <p>An effective strategy for salvage therapy in refractory or relapsed childhood brain tumours is yet to be established.</p> Methods <p>A national retrospective review of 135 children diagnosed with treatment refractory or recurrent MB from 2000 to 2021 at 5 academic Canadian pediatric neurosurgical centers was performed.</p> Results <p>The mean age at diagnosis was 7.09&#xa0;years (SD 4.37) with a male predominance (64.4%) and 17.8% under the age of 3&#xa0;years. Most of the tumour histology was classic MB. Just over a third of children experienced disease progression during or after adjuvant therapy with an average disease-free interval of 17.55&#xa0;months (SD 4.04). The majority of children experienced local disease progression (55.3%). Just over thirty percent of children succumbed to disease progression (<i>n</i>&#xa0;=&#xa0;47, 34.8%). The mean age of first srelapse was 10.85&#xa0;years (SD 7.10) and was identified on average 17.55&#xa0;months (SD 9.76) from diagnosis. Most children with progressive disease (82.9%) were deceased at the time of writing. The mean time from first and second relapse to death was 27.19&#xa0;months (SD 36.17) and 9.93&#xa0;months (11.92) respectively. The cause of death was most commonly disease progression followed by a medical complication. Initial response to salvage therapy with recurrence and biopsy at index surgery were independently associated with mortality. The children were followed up for a mean interval of 83.17 months (SD 54.15).</p> Conclusion <p>Recurrent disease in childhood medulloblastoma after provision of upfront therapy confers a very poor prognosis despite salvage therapy and is an unmet need for further study and therapeutic development.</p>

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National multicentered retrospective review of salvage therapy for pediatric medulloblastoma: a Canadian experience

  • 期刊论文

  • 发表日期：

    2025年12月1日

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  • Michelle M Kameda-Smith,
  • Melissa Lannon,
  • Alysa Almojuela,
  • Mark A MacLean,
  • Amit Persad,
  • Nicholas Sader,
  • Arani Kulamurugan,
  • Branavan Manoranjan,
  • Craig Erker,
  • Minoo Aminnejad,
  • Bruce Cooks,
  • Aleksander M Vitali,
  • Walter Hader,
  • Simon Walling,
  • P. Daniel McNeely,
  • Blake Yarascavitch,
  • Olufemi Ajani,
  • Forough Farrokhyar,
  • Adam Fleming,
  • Sheila K Singh

摘要

Background

An effective strategy for salvage therapy in refractory or relapsed childhood brain tumours is yet to be established.

Methods

A national retrospective review of 135 children diagnosed with treatment refractory or recurrent MB from 2000 to 2021 at 5 academic Canadian pediatric neurosurgical centers was performed.

Results

The mean age at diagnosis was 7.09 years (SD 4.37) with a male predominance (64.4%) and 17.8% under the age of 3 years. Most of the tumour histology was classic MB. Just over a third of children experienced disease progression during or after adjuvant therapy with an average disease-free interval of 17.55 months (SD 4.04). The majority of children experienced local disease progression (55.3%). Just over thirty percent of children succumbed to disease progression (n = 47, 34.8%). The mean age of first srelapse was 10.85 years (SD 7.10) and was identified on average 17.55 months (SD 9.76) from diagnosis. Most children with progressive disease (82.9%) were deceased at the time of writing. The mean time from first and second relapse to death was 27.19 months (SD 36.17) and 9.93 months (11.92) respectively. The cause of death was most commonly disease progression followed by a medical complication. Initial response to salvage therapy with recurrence and biopsy at index surgery were independently associated with mortality. The children were followed up for a mean interval of 83.17 months (SD 54.15).

Conclusion

Recurrent disease in childhood medulloblastoma after provision of upfront therapy confers a very poor prognosis despite salvage therapy and is an unmet need for further study and therapeutic development.