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Pre- and postdiagnosis growth failure, adult short stature, and untreated growth hormone deficiency in radiotherapy-treated long-term survivors of childhood brain tumor

Anttonen Julia; Sirkiä Kirsti; Riikonen Pekka; Ojaniemi Marja; Pokka Tytti; Rantala Heikki; Lähteenmäki Päivi; Harila-Saari Arja; Arikoski Pekka; Lönnqvist Tuula; Arola Mikko; Remes Tiina

Pre- and postdiagnosis growth failure, adult short stature, and untreated growth hormone deficiency in radiotherapy-treated long-term survivors of childhood brain tumor

Anttonen Julia
Sirkiä Kirsti
Riikonen Pekka
Ojaniemi Marja
Pokka Tytti
Rantala Heikki
Lähteenmäki Päivi
Harila-Saari Arja
Arikoski Pekka
Lönnqvist Tuula
Arola Mikko
Remes Tiina
Katso/Avaa
LähteenmäkiEtAl2022Pre-AndPostdiagnosisGrowthFailure.pdf (1.071Mb)
Lataukset: 

Public Library of Science
doi:10.1371/journal.pone.0274274
URI
https://journals.plos.org/plosone/article?id=10.1371/journal.pone.0274274
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Julkaisun pysyvä osoite on:
https://urn.fi/URN:NBN:fi-fe2022102463169
Tiivistelmä

Purpose
Growth failure is common in radiotherapy-treated long-term survivors of pediatric brain tumors, but studies on longitudinal growth in this patient group are lacking. Here, the aim was to assess the changes in growth patterns before and after brain tumor diagnosis, the adult height, and the risk factors for compromised growth. The incidence and treatment practices of growth hormone deficiency were analyzed.

Methods
A cohort of 73 survivors of childhood brain tumor (median age 27.2 years, range 16.2 to 43.8 years) was studied after a median follow-up period of 20.4 years from diagnosis (IQR 14.9 to 22.9 years). Patients were treated in five university hospitals in Finland between 1970 and 2008. Growth curves, final height, and patient- and disease-related risk factors for compromised growth during different growth periods were analyzed. Laboratory analyses for IGF-1 and IGFBP-3 were performed at the follow-up.

Results
Growth failure was evident at diagnosis, with a mean height decline of -0.6 SDS (standard deviation score) from birth (95% CI -1.15 to -0.05). Mean height SDS decline after the diagnosis was -1.09 SDS (95%CI -1.51 to -0.66). At follow-up, 37% of the study subjects (27/73) had true short stature (height < -2 SDS). The mean height deficit corrected for target height was -1.9 SDS (95% CI -1.45 to -2.40). Growth failure was associated with the age at diagnosis, corticosteroid dose, radiotherapy modality and mean dose of irradiation in the thalamic area. Low IGF-1 level (below -2.0 SDS) was found in 32% (23/72), and untreated growth hormone deficiency in 40% (29/72) of the subjects.

Conclusion
Longitudinal growth impairment was common in radiotherapy-treated survivors of childhood brain tumor, resulting in compromised adult height. Loss of growth potential was evident already at diagnosis and further accelerated by the treatments. At young adulthood, unrecognized growth hormone deficiency was common.

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