Skip to main content

Ontogenetic changes of craniofacial complex in Turner syndrome patients treated with growth hormone

Abstract

Objective

The present study assessed changes of craniofacial complex in Turner syndrome (TS) patients treated with growth hormone (GH) during development. The objective was to examine the growth rate and pattern of craniofacial structures and to establish effects of GH on craniofacial development.

Materials and methods

The study population consisted of 15 TS patients treated with GH aged 5–18.5 years (13.3 ± 4.4) and corresponding control group of 45 females aged 6.8–18.7 (11.4 ± 2.6). According to the stage of cervical vertebral maturation, subjects were categorized into pre-growth (5 TS and 15 controls) and growth (10 TS and 30 controls) subgroups. The cephalometric analysis comprised angular and linear variables, measured on lateral cephalometric radiographs.

Results

The mandibular corpus/anterior cranial base ratio increased significantly only in controls during development. In growth period, ramus/corpus ratio was significantly larger in TS group. SNA and SNB angles were significantly smaller in TS growth subgroup compared to corresponding controls. Among other variables, no statistically significant differences were revealed.

Conclusions

In TS patients treated with GH, growth capacities of cranial base and maxilla are adequate which can be attributed to GH treatment. Shape of mandible is altered due to decreased growth of corpus and overdeveloped ramus. Both maxillary and mandibular retrognathism are becoming more expressed during development.

Clinical relevance

Favorable influence of GH on craniofacial complex growth rate and altered growth pattern revealed in this study should be considered while planning both orthodontic treatment and retention.

This is a preview of subscription content, access via your institution.

Fig. 1
Fig. 2
Fig. 3
Fig. 4
Fig. 5
Fig. 6

References

  1. Nielsen J, Wohlert M (1991) Chromosome abnormalities found among 34,910 newborn children: results from a 13-year incidence study in Arhus, Denmark. Hum Genet 87:81–83

    PubMed  Article  Google Scholar 

  2. Damnjanovic T, Novakovic I, Milasin J, Bunjevacki V, Jekic B, Cvijeticanin S, Lukovic L (2007) X chromosome imprinting in Turner syndrome. Korean J Genetics 29:291–295

    Google Scholar 

  3. Djordjevic VA, Jovanovic JV, Pavkovic-Lucic SB, Drakulic DD, Djurovic MM, Gotic MD (2010) Cytogenetic findings in Serbian patients with Turner’s syndrome stigmata. Genet Mol Res 9:2213–2221

    PubMed  Article  Google Scholar 

  4. Ranke MB (1990) An introduction to Turner’s syndrome. Oxford Clinical Communications, Oxford

    Google Scholar 

  5. Ogata T, Matsuo N (1995) Turner syndrome and female sex chromosome aberrations: deduction of the principal factors involved in the development of clinical features. Hum Genet 95:607–629

    PubMed  Article  Google Scholar 

  6. Munns CJ, Haase HR, Crowther LM, Hayes MT, Blaschke R, Rappold G, Glass IA, Batch JA (2004) Expression of SHOX in human fetal and childhood growth plate. J Clin Endocrinol Metab 89:4130–4135

    PubMed  Article  Google Scholar 

  7. Ogata T, Matsuo N, Nishimura G (2001) SHOX haploinsufficiency and overdosage: impact of gonadal function status. J Med Genet 38:1–6

    PubMed  Article  Google Scholar 

  8. Clement-Jones M, Schiller S, Rao E, Blaschke RJ, Zuniga A, Zeller R, Robson SC, Binder G, Glass I, Strachan T, Lindsay S, Rappold GA (2000) The short stature homeobox gene SHOX is involved in skeletal abnormalities in Turner syndrome. Hum Mol Genet 9:695–702

    PubMed  Article  Google Scholar 

  9. Jensen BL (1985) Craniofacial morphology in Turner syndrome. J Craniofac Genet Dev Biol 5:327–340

    PubMed  Google Scholar 

  10. Babic M, Micic M, Jaksic N, Micic S (1991) An extra X chromosome effect on craniofacial morphogenesis in men. Eur J Orthod 13:329–332

    PubMed  Article  Google Scholar 

  11. Babic M, Scepan I, Micic M (1993) Comparative cephalometric analysis in patients with X-chromosome aneuploidy. Arch Oral Biol 38:179–183

    PubMed  Article  Google Scholar 

  12. Midtbo M, Wisth PJ, Halse A (1996) Craniofacial morphology in young patients with Turner syndrome. Eur J Orthod 18:215–225

    PubMed  Google Scholar 

  13. Babic M, Glisic B, Scepan I (1997) Mandibular growth pattern in Turner’s syndrome. Eur J Orthod 19:161–164

    PubMed  Article  Google Scholar 

  14. Gron M, Pietila K, Alvesalo L (1999) The craniofacial complex in 45, X/46, XX females. Arch Oral Biol 44:1077–1084

    PubMed  Article  Google Scholar 

  15. Perkiomaki MR, Kyrkanides S, Niinimaa A, Alvesalo L (2005) The relationship of distinct craniofacial features between Turner syndrome females and their parents. Eur J Orthod 27:48–52

    PubMed  Article  Google Scholar 

  16. Dumancic J, Kaic Z, Varga ML, Lauc T, Dumic M, Milosevic SA, Brkic H (2010) Characteristics of the craniofacial complex in Turner syndrome. Arch Oral Biol 55:81–88

    PubMed  Article  Google Scholar 

  17. Andersen E, Sonnesen L, Kjaer MS, Fischer Hansen B, Kjaer I (2000) The prenatal cranial base complex and hand in Turner syndrome. Eur J Orthod 22:185–194

    PubMed  Article  Google Scholar 

  18. Brook CG, Murset G, Zachmann M, Prader A (1974) Growth in children with 45, XO Turner’s syndrome. Arch Dis Child 49:789–795

    PubMed  Article  Google Scholar 

  19. Davenport ML, Punyasavatsut N, Stewart PW, Gunther DF, Savendahl L, Sybert VP (2002) Growth failure in early life: an important manifestation of Turner syndrome. Horm Res 57:157–164

    PubMed  Article  Google Scholar 

  20. Saenger P, Wikland KA, Conway GS, Davenport M, Gravholt CH, Hintz R, Hovatta O, Hultcrantz M, Landin-Wilhelmsen K, Lin A, Lippe B, Pasquino AM, Ranke MB, Rosenfeld R, Silberbach M (2001) Recommendations for the diagnosis and management of Turner syndrome. J Clin Endocrinol Metab 86:3061–3069

    PubMed  Article  Google Scholar 

  21. Bondy CA (2007) Care of girls and women with Turner syndrome: a guideline of the Turner Syndrome Study Group. J Clin Endocrinol Metab 92:10–25

    PubMed  Article  Google Scholar 

  22. Kjellberg H, Wikland KA (2007) A longitudinal study of craniofacial growth in idiopathic short stature and growth hormone-deficient boys treated with growth hormone. Eur J Orthod 29:243–250

    PubMed  Article  Google Scholar 

  23. Funatsu M, Sato K, Mitani H (2006) Effects of growth hormone on craniofacial growth. Angle Orthod 76:970–977

    PubMed  Article  Google Scholar 

  24. van Erum R, Mulier M, Carels C, Verbeke G, de Zegher F (1997) Craniofacial growth in short children born small for gestational age: effect of growth hormone treatment. J Dent Res 76:1579–1586

    PubMed  Article  Google Scholar 

  25. Forsberg CM, Krekmanova L, Dahllof G (2002) The effect of growth hormone therapy on mandibular and cranial base development in children treated with total body irradiation. Eur J Orthod 24:285–292

    PubMed  Article  Google Scholar 

  26. Hass AD, Simmons KE, Davenport ML, Proffit WR (2001) The effect of growth hormone on craniofacial growth and dental maturation in Turner syndrome. Angle Orthod 71:50–59

    PubMed  Google Scholar 

  27. Baccetti T, Franchi L, McNamara JA (2005) The cervical vertebral maturation (CVM) method for the assessment of optimal treatment timing in dentofacial orthopedics. Semin Orthod 11:119–129

    Article  Google Scholar 

  28. Enlow DH, Hans MG (1996) The plan of the human face. In: Essentials of facial growth. W. B. Saunders Company, Philadelphia, pp 146–165

Download references

Acknowledgments

This work was supported by the grant # 175075 of the Serbian Ministry of Education and Science.

Conflict of interest

The authors declare that they have no conflict of interest.

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Jovana Juloski.

Rights and permissions

Reprints and Permissions

About this article

Cite this article

Juloski, J., Glisic, B., Scepan, I. et al. Ontogenetic changes of craniofacial complex in Turner syndrome patients treated with growth hormone. Clin Oral Invest 17, 1563–1571 (2013). https://doi.org/10.1007/s00784-012-0844-8

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00784-012-0844-8

Keywords

  • Turner syndrome
  • Growth hormone therapy
  • Craniofacial development
  • Bimaxillary retrognathism