Pubertal development and primary ovarian insufficiency in female survivors of embryonal brain tumors following risk-adapted craniospinal irradiation and adjuvant chemotherapy

Mariko Dewire, Daniel M. Green, Charles A. Sklar, Thomas E. Merchant, Dana Wallace, Tong Lin, Tamara Vern-Gross, Larry E. Kun, Matthew J. Krasin, James M. Boyett, Karen D. Wright, Cynthia Wetmore, Alberto Broniscer, Amar Gajjar

Research output: Contribution to journalArticle

5 Citations (Scopus)

Abstract

Female survivors of central nervous system (CNS) tumors are at an increased risk for gonadal damage and variations in the timing of puberty following radiotherapy and alkylating agent-based chemotherapy. Procedure: Clinical and laboratory data were obtained from 30 evaluable female patients with newly diagnosed embryonal CNS tumors treated on a prospective protocol (SJMB 96) at St. Jude Children's Research Hospital (SJCRH). Pubertal development was evaluated by Tanner staging. Primary ovarian insufficiency (POI) was determined by Tanner staging and FSH level. Females with Tanner stage I-II and FSH>15mIU/ml, or Tanner stage III-V, FSH>25mIU/ml and FSH greater than LH were defined to have ovarian insufficiency. Recovery of ovarian function was defined as normalization of FSH without therapeutic intervention. Results: Median length of follow-up post completion of therapy was 7.2 years (4.0-10.8 years). The cumulative incidence of pubertal onset was 75.6% by the age of 13. Precocious puberty was observed in 11.1% and delayed puberty in 11.8%. The cumulative incidence of POI was 82.8%, though recovery was observed in 38.5%. Conclusions: Treatment for primary CNS embryonal tumors may cause variations in the timing of pubertal development, impacting physical and psychosocial development. Female survivors are at risk for POI, a subset of whom will recover function over time. Further refinement of therapies is needed in order to reduce late ovarian insufficiency.

Original languageEnglish (US)
Pages (from-to)329-334
Number of pages6
JournalPediatric Blood and Cancer
Volume62
Issue number2
DOIs
StatePublished - Feb 1 2015

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Craniospinal Irradiation
Primary Ovarian Insufficiency
Adjuvant Chemotherapy
Brain Neoplasms
Central Nervous System Neoplasms
Survivors
Delayed Puberty
Precocious Puberty
Alkylating Agents
Incidence
Recovery of Function
Therapeutics
Puberty
Radiotherapy
Drug Therapy
Research

Keywords

  • Medulloblastoma
  • Ovarian dysfunction
  • Ovarian insufficiency
  • Puberty

ASJC Scopus subject areas

  • Pediatrics, Perinatology, and Child Health
  • Hematology
  • Oncology

Cite this

Pubertal development and primary ovarian insufficiency in female survivors of embryonal brain tumors following risk-adapted craniospinal irradiation and adjuvant chemotherapy. / Dewire, Mariko; Green, Daniel M.; Sklar, Charles A.; Merchant, Thomas E.; Wallace, Dana; Lin, Tong; Vern-Gross, Tamara; Kun, Larry E.; Krasin, Matthew J.; Boyett, James M.; Wright, Karen D.; Wetmore, Cynthia; Broniscer, Alberto; Gajjar, Amar.

In: Pediatric Blood and Cancer, Vol. 62, No. 2, 01.02.2015, p. 329-334.

Research output: Contribution to journalArticle

Dewire, M, Green, DM, Sklar, CA, Merchant, TE, Wallace, D, Lin, T, Vern-Gross, T, Kun, LE, Krasin, MJ, Boyett, JM, Wright, KD, Wetmore, C, Broniscer, A & Gajjar, A 2015, 'Pubertal development and primary ovarian insufficiency in female survivors of embryonal brain tumors following risk-adapted craniospinal irradiation and adjuvant chemotherapy', Pediatric Blood and Cancer, vol. 62, no. 2, pp. 329-334. https://doi.org/10.1002/pbc.25274
Dewire, Mariko ; Green, Daniel M. ; Sklar, Charles A. ; Merchant, Thomas E. ; Wallace, Dana ; Lin, Tong ; Vern-Gross, Tamara ; Kun, Larry E. ; Krasin, Matthew J. ; Boyett, James M. ; Wright, Karen D. ; Wetmore, Cynthia ; Broniscer, Alberto ; Gajjar, Amar. / Pubertal development and primary ovarian insufficiency in female survivors of embryonal brain tumors following risk-adapted craniospinal irradiation and adjuvant chemotherapy. In: Pediatric Blood and Cancer. 2015 ; Vol. 62, No. 2. pp. 329-334.
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abstract = "Female survivors of central nervous system (CNS) tumors are at an increased risk for gonadal damage and variations in the timing of puberty following radiotherapy and alkylating agent-based chemotherapy. Procedure: Clinical and laboratory data were obtained from 30 evaluable female patients with newly diagnosed embryonal CNS tumors treated on a prospective protocol (SJMB 96) at St. Jude Children's Research Hospital (SJCRH). Pubertal development was evaluated by Tanner staging. Primary ovarian insufficiency (POI) was determined by Tanner staging and FSH level. Females with Tanner stage I-II and FSH>15mIU/ml, or Tanner stage III-V, FSH>25mIU/ml and FSH greater than LH were defined to have ovarian insufficiency. Recovery of ovarian function was defined as normalization of FSH without therapeutic intervention. Results: Median length of follow-up post completion of therapy was 7.2 years (4.0-10.8 years). The cumulative incidence of pubertal onset was 75.6{\%} by the age of 13. Precocious puberty was observed in 11.1{\%} and delayed puberty in 11.8{\%}. The cumulative incidence of POI was 82.8{\%}, though recovery was observed in 38.5{\%}. Conclusions: Treatment for primary CNS embryonal tumors may cause variations in the timing of pubertal development, impacting physical and psychosocial development. Female survivors are at risk for POI, a subset of whom will recover function over time. Further refinement of therapies is needed in order to reduce late ovarian insufficiency.",
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AU - Green, Daniel M.

AU - Sklar, Charles A.

AU - Merchant, Thomas E.

AU - Wallace, Dana

AU - Lin, Tong

AU - Vern-Gross, Tamara

AU - Kun, Larry E.

AU - Krasin, Matthew J.

AU - Boyett, James M.

AU - Wright, Karen D.

AU - Wetmore, Cynthia

AU - Broniscer, Alberto

AU - Gajjar, Amar

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N2 - Female survivors of central nervous system (CNS) tumors are at an increased risk for gonadal damage and variations in the timing of puberty following radiotherapy and alkylating agent-based chemotherapy. Procedure: Clinical and laboratory data were obtained from 30 evaluable female patients with newly diagnosed embryonal CNS tumors treated on a prospective protocol (SJMB 96) at St. Jude Children's Research Hospital (SJCRH). Pubertal development was evaluated by Tanner staging. Primary ovarian insufficiency (POI) was determined by Tanner staging and FSH level. Females with Tanner stage I-II and FSH>15mIU/ml, or Tanner stage III-V, FSH>25mIU/ml and FSH greater than LH were defined to have ovarian insufficiency. Recovery of ovarian function was defined as normalization of FSH without therapeutic intervention. Results: Median length of follow-up post completion of therapy was 7.2 years (4.0-10.8 years). The cumulative incidence of pubertal onset was 75.6% by the age of 13. Precocious puberty was observed in 11.1% and delayed puberty in 11.8%. The cumulative incidence of POI was 82.8%, though recovery was observed in 38.5%. Conclusions: Treatment for primary CNS embryonal tumors may cause variations in the timing of pubertal development, impacting physical and psychosocial development. Female survivors are at risk for POI, a subset of whom will recover function over time. Further refinement of therapies is needed in order to reduce late ovarian insufficiency.

AB - Female survivors of central nervous system (CNS) tumors are at an increased risk for gonadal damage and variations in the timing of puberty following radiotherapy and alkylating agent-based chemotherapy. Procedure: Clinical and laboratory data were obtained from 30 evaluable female patients with newly diagnosed embryonal CNS tumors treated on a prospective protocol (SJMB 96) at St. Jude Children's Research Hospital (SJCRH). Pubertal development was evaluated by Tanner staging. Primary ovarian insufficiency (POI) was determined by Tanner staging and FSH level. Females with Tanner stage I-II and FSH>15mIU/ml, or Tanner stage III-V, FSH>25mIU/ml and FSH greater than LH were defined to have ovarian insufficiency. Recovery of ovarian function was defined as normalization of FSH without therapeutic intervention. Results: Median length of follow-up post completion of therapy was 7.2 years (4.0-10.8 years). The cumulative incidence of pubertal onset was 75.6% by the age of 13. Precocious puberty was observed in 11.1% and delayed puberty in 11.8%. The cumulative incidence of POI was 82.8%, though recovery was observed in 38.5%. Conclusions: Treatment for primary CNS embryonal tumors may cause variations in the timing of pubertal development, impacting physical and psychosocial development. Female survivors are at risk for POI, a subset of whom will recover function over time. Further refinement of therapies is needed in order to reduce late ovarian insufficiency.

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