Craniofacial Stability in Patients with Crouzon or Apert Syndrome after Le Fort III Distraction Osteogenesis

Abstract

Objective

Le Fort III osteotomy with distraction osteogenesis (DO) is used to improve the retruded midface in patients with Crouzon or Apert syndrome. This study aimed to evaluate sagittal and vertical preoperative and postoperative cephalometric changes of DO of the midface in patients with Crouzon or Apert syndrome.

Design

Population-based case-control study.

Patients and Methods

Records of patients with the syndrome of Crouzon (N = 6) or Apert (N = 7) were compared, before and after Le Fort III DO, with a nonsyndromic untreated control group (N = 486).

Main Outcome Measures

Sagittal and vertical cephalometric maxillary landmarks and measurements were used to predict and measure midface advancement and rotation after Le Fort III DO. Cephalograms were taken before surgery (T0), 4 months after surgery at removal of the distraction device (T1), and 1 year after removal of the distraction device (T2).

Analysis

Z scores were performed to compare cephalometric measures of syndromic patients with control subjects.

Results

Cephalograms of 13 patients with Crouzon syndrome (N = 6) or Apert (N = 7) (age range 8.2 to 19.8 years) were evaluated. Treatment changes (T1-T2) showed statistically significant maxillary advancement, with no significant differences between the patients with the Crouzon or Apert syndrome.

Conclusions

DO of the midface in patients with Crouzon or Apert syndrome seems to be stable in the sagittal direction after follow-up. Although Crouzon and Apert differ after DO, anteroposterior craniofacial dimensions were significantly improved and were closer to patterns of normal subjects.

Keywords

Apert patients Crouzon patients distraction osteogenesis

Get full access to this article

View all access options for this article.

References

Allam

K.A.

Wan

D.C.

Khwanngern

Kawamoto

H.K.

Tanna

Perry

Bradley

J.P.

Treatment of Apert syndrome: a long-term follow-up study. Plast Reconstr Surg. 2011; 127: 1601–1611.

Bachmayer

D.I.

Ross

R.B.

Munro

I.R.

Maxillary growth following LeFort III advancement surgery in Crouzon, Apert, and Pfeiffer syndromes. Am J Orthod Dentofacial Orthop. 1986; 90: 420–430.

Baum

J.A.

Hanley

E.N.

Jr Pullekines

Comparison of halo complications in adults and children. Spine. 1989; 14: 251–252.

Cheung

L.K.

Chua

H.D.

Hägg

M.B.

Cleft maxillary distraction versus orthognathic surgery: clinical morbidities and surgical relapse. Plast Reconstr Surg. 2006; 118: 996–1008.

Connolly

J.P.

Gruss

Seto

M.L.

. Progressive postnatal craniosynostosis and increased intracranial pressure. Plast Reconstr Surg. 2004; 113: 1313–1323.

Costaras-Volarich

Pruzansky

Is the mandible intrinsically different in Apert and Crouzon syndromes?

Am J Orthod. 1984; 85: 475–487.

Dahlberg

Statistical Methods for Medical and Biological Students. New York: Interscience Publications; 1940: 184.

De Bastiani

Aldegheri

Renzi-Brivio

Trivella

Limb lengthening by callus distraction (callotasis). J Pediatr Orthop. 1987; 7: 129–134.

Fearon

J.A.

Halo distraction of the Le Fort III in syndromic craniosynostosis: a long-term assessment. Plast Reconstr Surg. 2005; 115: 1524–1536.

10.

Figueroa

A.A.

Polley

J.W.

Management of the severe cleft and syndromic midface hypoplasia. Orthod Craniofac Res. 2007; 10: 167–179.

11.

Figueroa

A.A.

Polley

J.W.

Friede

E.W.

Long-term skeletal stability after maxillary advancement with distraction osteogenesis using a rigid external distraction device in cleft maxillary deformities. Plast Reconstr Surg. 2004; 114: 1382–1392.

12.

Hopper

R.A.

Sandercoe

Woo

Watts

Kelley

Ettinger

R.E.

Saltzman

Computed tomographic analysis of temporal maxillary stability and pterygomaxillary generate formation following pediatric Le Fort III distraction advancement. Plast Reconstr Surg. 2010; 126: 1665–1674.

13.

Iannetti

Fadda

Agrillo

Poladas

Iannetti

Filiaci

LeFort III advancement with and without osteogenesis distraction. J Craniofac Surg. 2006; 17: 536–543.

14.

Jacobson

The “Wits” appraisal of jaw disharmony. Am J Orthod. 1975; 67: 125–138.

15.

Kolar

J.C.

Munro

I.R.

Farkas

L.G.

Patterns of dysmorphology in Crouzon syndrome: an anthropometric study. Cleft Palate J. 1988; 25: 235–244.

16.

Kreiborg

Aduss

Pre- and postsurgical facial growth in patients with Crouzon's and Apert's syndromes. Cleft Palate J. 1986; 23(suppl): 78–90.

17.

Kreiborg

Cohen

M.M.

Jr . Is craniofacial morphology in Apert and Crouzon syndromes the same? Acta Odontol Scand. 1998; 56: 339–341.

18.

Marchac

Arnaud

Midface surgery from Tessier to distraction. Childs Nerv Syst. 1999; 15: 681–694.

19.

Meazzini

M.C.

Mazzoleni

Caronni

Bozzetti

Le Fort III advancement osteotomy in the growing child affected by Crouzon's and Apert's syndromes: presurgical and postsurgical growth. J Craniofac Surg. 2005; 16: 369–377.

20.

Nout

Wolvius

E.B.

van Adrichem

L.N.

Ongkosuwito

E.M.

van der Wal

K.G.

Complications in maxillary distraction using the RED II device: a retrospective analysis of 21 patients. Int J Oral Maxillofac Surg. 2006; 35: 897–902.

21.

Panchal

Uttchin

Management of craniosynostosis. Plast Reconstr Surg. 2003; 111: 2032–2048.

22.

Pelo

Gasparini

Di Petrillo

Tamburrini

Di Rocco

Distraction osteogenesis in the surgical treatment of craniostenosis: a comparison of internal and external craniofacial distractor devices. Childs Nerv Syst. 2007; 23: 1447–1453.

23.

Posnick

J.C.

Ruiz

R.L.

The craniofacial synostosis syndromes: current surgical thinking and future directions. Cleft Palate Craniofac J. 2000; 37: 434.

24.

Prahl-Andersen

Controversies in the management of craniofacial malformations. Semin Orthod. 2005; 11: 67–75.

25.

Prahl-Andersen

Kowalski

C.J.

Heyendaal

PHJM

. A Mixed-Longitudinal Interdisciplinary Study of Growth and Development. New York: Academic Press; 1979.

26.

Reid

R.R.

Facial skeletal growth and timing of surgical intervention. Clin Plast Surg. 2007; 34: 357–367.

27.

Reitsma

J.H.

Ongkosuwito

E.M.

Buschang

P.H.

Prahl-Andersen

Facial growth in patients with Apert and Crouzon syndromes compared to normal children. Cleft Palate Craniofac J. 2012; 49: 185–193.

28.

Renier

Lajeunie

Arnaud

Marchac

Management of craniosynostoses. Childs Nerv Syst. 2000; 16: 645–658.

29.

Shetye

P.R.

Boutros

Grayson

B.H.

McCarthy

J.G.

Midterm follow-up of midface distraction for syndromic craniosynostosis: a clinical and cephalometric study. Plast Reconstr Surg. 2007; 120: 1621–1632.

30.

Shetye

P.R.

Davidson

E.H.

Sorkin

Grayson

B.H.

McCarthy

J.G.

Evaluation of three surgical techniques for advancement of the midface in growing children with syndromic craniosynostosis. Plast Reconstr Surg. 2010; 126: 982–994.

31.

Shrout

P.E.

Fleiss

J.L.

Intraclass correlations: uses in assessing rater reliability. Psychol Bull. 1979; 86: 420–428.

32.

Sloan

G.M.

Wells

K.C.

Raffel

McComb

J.G.

Surgical treatment of craniosynostosis: outcome analysis of 250 consecutive patients. Pediatrics. 1997; 100: E2.

33.

Swennen

Schliephake

Dempf

Schierle

Malevez

Craniofacial distraction osteogenesis: a review of the literature: Part I: clinical studies. Int J Oral Maxillofac Surg. 2001; 30: 89–103.

34.

Tanner

J.M.

Whitehouse

R.H.

Cameron

Marshall

W.A.

Healy

M.J.R.

Goldstein

Assessment of Skeletal Maturity and Prediction of Adult Height (TW2 method). London: Academic Press; 1983.

35.

Trpkova

Major

Prasad

Nebbe

Cephalometric landmarks identification and reproducibility: a meta analysis. Am J Orthod Dentofacial Orthop. 1997; 112: 165–170.

36.

Wah

P.L.

Cooke

M.S.

Hägg

Comparative cephalometric errors for orthodontic and surgical patients. Int J Adult Orthodon Orthognath Surg. 1995; 10: 119–126.

37.

Waitzman

A.A.

Posnick

J.C.

Armstrong

D.C.

Pron

G.E.

Craniofacial skeletal measurements based on computed tomography: Part II. Normal values and growth trends. Cleft Palate Craniofac J. 1992; 29: 118–128.