Occurrence of neoplasms in individuals with congenital, severe GH deficiency from the Itabaianinha kindred

Cindi G. Marinho, Lívia M. Mermejo, Roberto Salvatori, João A. Assirati, Carla R.P. Oliveira, Elenilde G. Santos, Ângela C.G.B. Leal, Cynthia S. Barros-Oliveira, Nayra P. Damascena, Carlos A. Lima, Catarine T. Farias, Ayrton C. Moreira, Manuel H. Aguiar-Oliveira

Research output: Contribution to journalArticle

Abstract

Growth hormone (GH) and the insulin-like growth factor I (IGF-I) have cell proliferative and differentiation properties. Whether these hormones have a role in mutagenesis is unknown. Nevertheless, severe IGF-I deficiency seems to confer protection against the development of neoplasms. Here, we report five cases of adult patients with severe and congenital isolated GH deficiency (IGHD) due to the c.57+1G>A mutation in the GHRH receptor gene, who developed tumors. Four GH-naïve subjects presented skin tumors: a 42-year-old man with a fibroepithelial polyp, a 53-year-old woman and two men (59 and 56 years old) with epidermoid skin cancers. One of these died from it after three surgeries and radiotherapy. The fifth patient was a 25-year-old woman, who had intermittently received GH replacement therapy (GHRT) from age 11 to 18, who developed an ependymoma extending from the fourth ventricle to the end of the thoracic spine. She underwent three surgical procedures, without obvious evidence of tumor recurrence during the six years follow up. These observations suggest that severe IGHD does not protect completely from development of tumors.

Original languageEnglish (US)
JournalGrowth Hormone and IGF Research
DOIs
StateAccepted/In press - Jan 1 2018

Fingerprint

Growth Hormone
Pituitary Dwarfism
Neoplasms
Fourth Ventricle
Ependymoma
Hormone Replacement Therapy
Skin Neoplasms
Polyps
Insulin-Like Growth Factor I
Mutagenesis
Cell Differentiation
Spine
Radiotherapy
Thorax
Hormones
Recurrence
Skin
Mutation
Genes

Keywords

  • Ependymoma
  • Growth hormone
  • IGF-I
  • Neoplasm
  • Skin tumor

ASJC Scopus subject areas

  • Endocrinology, Diabetes and Metabolism
  • Endocrinology

Cite this

Marinho, C. G., Mermejo, L. M., Salvatori, R., Assirati, J. A., Oliveira, C. R. P., Santos, E. G., ... Aguiar-Oliveira, M. H. (Accepted/In press). Occurrence of neoplasms in individuals with congenital, severe GH deficiency from the Itabaianinha kindred. Growth Hormone and IGF Research. https://doi.org/10.1016/j.ghir.2018.03.004

Occurrence of neoplasms in individuals with congenital, severe GH deficiency from the Itabaianinha kindred. / Marinho, Cindi G.; Mermejo, Lívia M.; Salvatori, Roberto; Assirati, João A.; Oliveira, Carla R.P.; Santos, Elenilde G.; Leal, Ângela C.G.B.; Barros-Oliveira, Cynthia S.; Damascena, Nayra P.; Lima, Carlos A.; Farias, Catarine T.; Moreira, Ayrton C.; Aguiar-Oliveira, Manuel H.

In: Growth Hormone and IGF Research, 01.01.2018.

Research output: Contribution to journalArticle

Marinho, CG, Mermejo, LM, Salvatori, R, Assirati, JA, Oliveira, CRP, Santos, EG, Leal, ÂCGB, Barros-Oliveira, CS, Damascena, NP, Lima, CA, Farias, CT, Moreira, AC & Aguiar-Oliveira, MH 2018, 'Occurrence of neoplasms in individuals with congenital, severe GH deficiency from the Itabaianinha kindred', Growth Hormone and IGF Research. https://doi.org/10.1016/j.ghir.2018.03.004
Marinho, Cindi G. ; Mermejo, Lívia M. ; Salvatori, Roberto ; Assirati, João A. ; Oliveira, Carla R.P. ; Santos, Elenilde G. ; Leal, Ângela C.G.B. ; Barros-Oliveira, Cynthia S. ; Damascena, Nayra P. ; Lima, Carlos A. ; Farias, Catarine T. ; Moreira, Ayrton C. ; Aguiar-Oliveira, Manuel H. / Occurrence of neoplasms in individuals with congenital, severe GH deficiency from the Itabaianinha kindred. In: Growth Hormone and IGF Research. 2018.
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abstract = "Growth hormone (GH) and the insulin-like growth factor I (IGF-I) have cell proliferative and differentiation properties. Whether these hormones have a role in mutagenesis is unknown. Nevertheless, severe IGF-I deficiency seems to confer protection against the development of neoplasms. Here, we report five cases of adult patients with severe and congenital isolated GH deficiency (IGHD) due to the c.57+1G>A mutation in the GHRH receptor gene, who developed tumors. Four GH-na{\"i}ve subjects presented skin tumors: a 42-year-old man with a fibroepithelial polyp, a 53-year-old woman and two men (59 and 56 years old) with epidermoid skin cancers. One of these died from it after three surgeries and radiotherapy. The fifth patient was a 25-year-old woman, who had intermittently received GH replacement therapy (GHRT) from age 11 to 18, who developed an ependymoma extending from the fourth ventricle to the end of the thoracic spine. She underwent three surgical procedures, without obvious evidence of tumor recurrence during the six years follow up. These observations suggest that severe IGHD does not protect completely from development of tumors.",
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AU - Lima, Carlos A.

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