Severe gonadotoxic insult manifests early in young girls treated for Ewing sarcoma

TY - JOUR

T1 - Severe gonadotoxic insult manifests early in young girls treated for Ewing sarcoma

AU - Mörse, Helena

AU - Elfving, Maria

AU - Turkiewicz, Aleksandra

AU - Andersen, Claus Yding

AU - Øra, Ingrid

PY - 2016/8/1

Y1 - 2016/8/1

N2 - We prospectively investigated anti-Müllerian hormone (AMH) as a measure of ovarian insult in young females during and after treatment for Wilms tumor (WT), osteosarcoma (OS), and Ewing sarcoma (ES).Twenty-one female childhood cancer patients, with a mean age of 7.9 years (range 0.6-17), entered the study. Levels of AMH, follicle-stimulating hormone (FSH), and luteinizing hormone were monitored at diagnosis and every 3 to 4 months during, and regularly for a mean of 2.6 years after treatment.A profound decline in AMH was seen in the majority of the 21 study patients 3 to 4 months after the beginning of treatment, the exception being patients with WT, of whom 60% showed no such decline. During the remaining treatment, all patients except those with WT not treated with whole abdominal radiotherapy or stem cell transplantation (SCT) had AMH below detection limit.After completion of treatment, patients with OS and WT (without whole abdominal radiotherapy and SCT) recovered in AMH and had FSH in the normal range. In contrast, ES patients showed no AMH recovery and highly fluctuating FSH in the first years of follow-up, except for the 2 youngest patients, who had a late, slow AMH recovery.In conclusion, young female ES patients already showed signs of severe ovarian dysfunction during the first years after cancer treatment similar to patients treated with SCT and abdominal radiotherapy, in contrast to females with WT and OS. Fertility counseling and information concerning fertility preservation procedures should be considered before starting to treat young females with ES.

AB - We prospectively investigated anti-Müllerian hormone (AMH) as a measure of ovarian insult in young females during and after treatment for Wilms tumor (WT), osteosarcoma (OS), and Ewing sarcoma (ES).Twenty-one female childhood cancer patients, with a mean age of 7.9 years (range 0.6-17), entered the study. Levels of AMH, follicle-stimulating hormone (FSH), and luteinizing hormone were monitored at diagnosis and every 3 to 4 months during, and regularly for a mean of 2.6 years after treatment.A profound decline in AMH was seen in the majority of the 21 study patients 3 to 4 months after the beginning of treatment, the exception being patients with WT, of whom 60% showed no such decline. During the remaining treatment, all patients except those with WT not treated with whole abdominal radiotherapy or stem cell transplantation (SCT) had AMH below detection limit.After completion of treatment, patients with OS and WT (without whole abdominal radiotherapy and SCT) recovered in AMH and had FSH in the normal range. In contrast, ES patients showed no AMH recovery and highly fluctuating FSH in the first years of follow-up, except for the 2 youngest patients, who had a late, slow AMH recovery.In conclusion, young female ES patients already showed signs of severe ovarian dysfunction during the first years after cancer treatment similar to patients treated with SCT and abdominal radiotherapy, in contrast to females with WT and OS. Fertility counseling and information concerning fertility preservation procedures should be considered before starting to treat young females with ES.

KW - Adolescent

KW - Anti-Mullerian Hormone

KW - Bone Neoplasms

KW - Child

KW - Child, Preschool

KW - Combined Modality Therapy

KW - Female

KW - Follicle Stimulating Hormone

KW - Humans

KW - Infant

KW - Kidney Neoplasms

KW - Luteinizing Hormone

KW - Osteosarcoma

KW - Primary Ovarian Insufficiency

KW - Sarcoma, Ewing

KW - Wilms Tumor

KW - Journal Article

U2 - 10.1097/MD.0000000000004512

DO - 10.1097/MD.0000000000004512

M3 - Journal article

C2 - 27537574

SN - 0025-7974

VL - 95

JO - Medicine (Baltimore)

JF - Medicine (Baltimore)

IS - 33

M1 - e4512

ER -