9th european craniofacial congress

9th European Craniofacial

CongressSalzburg (Austria), September 14-17, 2011

Editor

Peter Schachner

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Index

Front page .......................................................................................................................... I

Late results of functional treatment of patients with unilateral cleft lip and palate Kozelj v ............................................................................................................................... 1

Photometric evaluation of bclp patients after primary columella lengthening: 10 year follow-upMeazzini MC, DMD, MMSc, Tavecchia MG, DD, Brusati R, MD ............................................. 5

The Development of Molecular Therapies for Craniosynostosis.Mooney MP, Cray JJ Jr., Losee JE, Moursi AM, Siegel MI, Cooper GM ................................ 9

Speech Activity and Participation (ICF-CY) in Children with Cleft Palate Speech Neumann S. ........................................................................................................................ 19

Incidence of cleft lip and palate/craniofacial on saudi arabia-hospital basedAl Johar A, Kandasam R, Subhan S .................................................................................... 25

Surgical repositioning of downward displaced premaxilla in bilateral cleft lip and palate patients - our experienceChirila L, Tarhon RC ............................................................................................................. 29

Nasoalveolar molding with simple and esthetic appliancesDaratsianos N., Jäger A. ..................................................................................................... 35

Validation of the Use of Sphincter Pharyngoplasty in the Treatment of VPI with Sagittal Pattern ClosureElbarbary Amir, Ali Ahmed, Basiony Samia, Abou-El Saad Tamer, & Amer Ayman .............. 41

BSSO conducted by piezoelectric saw - review of 187 casesJagielak M., Michalski P., Jagielak A., Socha M., Rogus P. .................................................. 45

Morphological Characteristics of the Velum following Furlow or Push-back ProcedureSatoh K., Takai E., Nohara K., Yamanishi T., Ishihama K., Sakai T., Kogo M. ........................ 51

Author Index ....................................................................................................................... 57

©2011 by MEDIMOND s.r.l. 1N914R9009

Late results of functional treatment of patients with unilateral cleft lip and palate

Kozelj v

DEPARTMENT OF MAXILLOFACIAL SURGERY, University Hospital Ljubljana, Slovenia

SummaryReasons for the abnormal facial morphology of unilateral cleft lip and palate

(UCLP) are multiple: intrinsic developmental deficiencies, functional distortions and iatrogenic factors induced by the treatment. Treatment where proper function of the affected parts is respected should result in less distorted facial growth. We followed growth of 41 consecutive complete UCLP patients on dental casts and radiographs, clinically we evaluated oral posture and mode of breathing. After primary surgery 70% of patients retained proper oral posture and nasal breading at rest. At the end of growth they had harmonious skeletal relationship. The remaining 30% demonstrated combined oro-nasal breathing, habit of opened mouth with the tongue apart from the palate and majority of them needed orthognatic correction.

IntroductionEvery patient with orofacial cleft wants to look like an individual without it. The

reasons for abnormal facial morphology of these individuals are result of intrinsic developmental deficiencies, functional distortions affecting the position and growth of normal and abnormal parts, and iatrogenic factors induced by treatment. Intrinsic deficiencies are rarely of clinical significance. Superimposed on the intrinsic factors are functional growth factors, mainly from musculature activity, which cause distor-tions and are responsible for the apparent severity of the condition of the neonate (Ross, 1987). However, individuals with untreated UCLP have good facial growth in most usually measured parameters (Mars and Houston, 1990; Lambrecht et al, 2000; Shetye, 2004). Thus the inhibition or distortion of facial growth can be contributed to surgical treatment. Postoperative scars due to these treatments could be responsible for the abnormal facial morphology at the end of the treatment.

So far research has mainly focused on the evaluation of different treatment proto-cols to determine which results in the highest proportion of patients with appearance closest to the normal and who do not need orthognatic corrections. Between 10 to

http://www.ncbi.nlm.nih.gov/pubmed?term=%22Mars M%22%5BAuthor%5D

http://www.ncbi.nlm.nih.gov/pubmed?term=%22Houston WJ%22%5BAuthor%5D


2 9th European Craniofacial Congress

50 % of patients with UCLP need orthognatic correction (Daskalogiannakis and Mehta, 2009), attributed by most authors to facial growth inhibition seen at the end of growth due to scaring after surgery performed in childhood. There are few who believed that growth inhibition is related to improper posture of the tongue in the oral cavity developed in the course of surgical corrections when the treated child has lost the ability for unobstructed breathing through the nose (Delaire and Precious, 1986; Oblak, 1975a, 1975b, 1979, 1990; Precious et al, 2001; Talmant, 1982; 1993; 2002; 2004; 2007). According to them, good facial growth requires securing a child with a cleft to maintain proper oral posture present at birth also after each surgical procedure. Nasal breathing – reflecting normal nasal function - is jeopardized above all because of the deformation of the nasal septum. Combined nasal and oral breathing is in fact very common among children with unilateral cleft lip and palate (Warren and Hairfield, 1990).

In Ljubljana emphasis has been placed on maintaining nasal breathing and thus enabling normal use of nasal and oral cavity throughout the treatment. This is based on the hypothesis that maxillary growth is hindered with improper resting oral posture which is affected because of the obstructed nasal cavity. We follow the concept of functionally oriented treatment for more than 35 years. With the present study we wanted to verify what facial morphology of UCLP has been achieved.

We believe that the condition in the neonate with orofacial cleft reflects prenatal adaptation to the non-union of some embryonic parts and consequently united oral and nasal cavities (Oblak, 1975a); after birth the upper jaw grows without changing the shape and relations between maxillary segments until the beginning of treatment interventions (Kozelj, 1999). Neonate`s mouth is closed, the tongue is on the palate at rest, intruding from time to time between the alveolar segments to seal the cleft. The midpoint of the upper alveolar arch together with anterior nasal spine is shifted to the noncleft side, collumela is oblique, the alla is stretched; nasal septum is curved to the noncleft side and in cranio-caudal direction. These distortions cause partial obstruction of the nasal cavity on the noncleft side, but leave the opposite half broad enough to allow normal breathing. Transverse and sagittal dimension of the palate exceed dimensions of the noncleft palate. Without treatment the cleft palate grows similarly to the noncleft one and the described cleft conditions persist, as do those of the na-sal septum (Kozelj, 1991). Under these circumstances nasal patency might become insufficient after lip repair, especially when the child starts walking. With increased physical activity the child might start to breathe also through the mouth. When child is breathing through the mouth the tongue has to move apart from the palate, the lips are not in touch any more. The normal resting oral posture is compromised. When this posture becomes habitual, the equilibrium between the tongue which moulds the oral cavity from the inside and muscular forces acting from outside, is changed. This results in gradual collapse of the alveolar segments and in diminished growth of the palate. Growth of the palate which is in accordance with enlargement of the tongue if the tongue is on the palate at rest is diminished, and congruity between the tongue and the palate disappears. Presurgical passive plate which mimics the normal palate and has no extensions into the cleft does prevent the intrusion of the tongue between the segments and compression of the septum. Remodelling of the palate happens in a few months and results in considerable straightening of the nasal septum, cleft in alveolus is reduced to about 10 % of the initial width without the collapse of the

3Salzburg, Austria, 14-17 September, 2011

segments (Kozelj, 1999). More straight nasal septum increases a chance for unob-structed nasal breathing after surgical correction of the cleft.

Objective of this study was to observe and measure how facial morphology of patients treated according to the above principles differed from unaffected individuals of the same age.

Material and methodLongitudinal study was performed on dental casts and lateral radiographs of 41

subjects with nonsyndromic complete UCLP, born between 1987 and 1995 in Slove-nia. The follow up was from birth to age of 24; average age 19.2 years. Treatment received: passive presurgical orthopaedic plate from birth to 6 months, lip repair at 6 months of age with Tennison-Randall, intravelar veloplasty at 12 months, hard pa-late repair and mucoperiosteal closure of the alveolus at two and half years. All the surgical treatment was performed by two experienced senior surgeons. Growth of the upper jaw was evaluated on dental casts of patients at the time of early mixed den-tition (mean age 6,8 years) and compared to noncleft control group of the same age. Oral posture was assessed clinically. It was deemed proper if the lips were naturally together, the tongue leant against the palate and the patient was breathing through the nose. A lax and everted lower lip spoke against a closed mouth and it was possible that the child was breathing also through the mouth. Patients with proper posture of the tongue have concave shape of the palate and light pink palatal mucosa. Lateral radiographs of facial skeleton were taken when patients were more than 16 years old.

ResultsDental casts obtained in early mixed dentition showed that the transverse dimen-

sions of the patinets’ palates were within normal values in 66% and the sagittal in 48% of patients. Oral posture and breathing: in 70% of analysed children we found naturally closed mouth, breathing through the nose and concave palate in transverse plane. The remaining 30% of patients had difficulties with nasal breathing and they demonstrated signs of improper posture of the tongue.

When we analysed them as adults (mean age 19,2 years) 70% (29 persons) had good sagittal intermaxillary relation with mean SNA 75,4°, SNB 75,2° and Wits ap-praisal -1,2 mm. These individuals denied problems with breathing through the nose. The remaining 30% (12 persons) demonstrate diminished maxillary growth with the mean SNA 71,4°, SNB 75,5° and Wits -4,9 mm. They all had breathing difficulties detected already at the time of dental casts analysis in early mixed dentition. Eight of them required orthognatic correction.

ConclusionFollowing the protocol oriented to preserve nasal breathing and proper oral posture

throughout the rehabilitation time we were able to achieve harmonious intermaxillary relation and good aesthetic result in about 70 % of individuals treated for complete UCLP. Orthognatic correction was necessary in 20% of analysed persons, the remai-ning 10 % were borderline cases.


ReferencesDaskalogiannakis J, Mehta M. The need for orthognatic surgery in patients with repaired

complete UCLP and BCLP. Cleft Palate Craniofac J. 46:498-502;2009.Delaire J, Precious D. Influence of the nasal septum on maxillary growth in patients with

congenital labiomaxillary clefts. Cleft Palate J. 23: 270-277;1986.Lambrecht JT, Kreusch T, Schulz L. Position, shape, and dimension of the maxilla in unope-

rated cleft lip and palate patients: review of the literature. Clin Anat. 13:121-33; 2000.Kozelj V. Maxillary arch dimensions in patients with cleft of the lip, alveolus, and plate with

or without presurgical orthodontic treatment .In: Pfeifer G, ed. Craniofacial abnormalities and clefts of the lip, alveolus and palate. 4th Hamburg international symposium. Stuttgart: Georg Thieme Verlag, 1991.

Kozelj V. Changes produced by presurgical orthopedic treatment before cheiloplasty in cleft lip and palate patients. Cleft Palate Craniofac J. 36: 515-521;1999.

Kozelj V. The basis for presurgical orthopedic treatment of infants with unilateral complete cleft lip and palate. Cleft Palate Craniofac J. 37:26-32; 2000.

Kozelj V, Drevenšek M. Comparison of skeletal relationships in UCLP children in mi-xed dentition with standards for noncleft population. In: Lilja J, Elander A, Friede H, et al, editors. Transactions. 9th International congress on cleft palate and rela-ted craniofacial anomalies; 2001 Jun 25-29; Goeteborg. Goeteborg: 2001;397-401. Mars M, Houston WJ. A preliminary study of facial growth and morphology in unope-rated male unilateral cleft lip and palate subjects over 13 years of age. Cleft Plalate J. 27:7-10; 1990.

Oblak P. New concept of morphogenesis of clefts in the lip, alveolus, and palate. J Maxillofac Surg. 3:182-7: 1975.

Oblak P. New guiding principles in the treatment of clefts. J Maxillofac Surg. 3:231-9,1975.Oblak P. Das physiologische Wechselspiel der Mundhoehlenmuskeln und seine Wirkung auf die

Entwicklung der Spaltregion. 3. Int. Symp. fuer Lippen-Kiefer-Gaumenspalten, Hamburg 1979, Stuttgart, 1982; 121-123.

Oblak P. The importance of nasal airways in cleft patients. In: Bardach J, Morris HL, ed. Multi-disciplinary management of cleft lip and palate. Philadelphia: W.B. Saunders Company,1990. Precious DS, Goodday RH, Morrison AD, Davis BR. Cleft lip and palate: a review for dentists. J Can Dent Assoc. 67:668-73;2001

Ross RB. Treatment variables affecting facial growth in complete unilateral cleft lip and palate. Part. 1: treatment affecting growth. Cleft Palate J. 24:5-23;1987.

Shetye PR. Facial growth of adults with unoperated clefts. Clin Plast Surg. 31:361-371; 2004.Talmant JC. The nostril in unilateral cleft lip. Can aesthetics and normal breathing be recon-

ciled? Orthod Fr. 53:451-456; 1982.Talmant JC. Nasal malformations associated with unilateral cleft lip. Accurate diagnosis and

management. Scand J Plast Reconstr Surg Hand Surg. 27:183-191; 1993.Talmant J, Talmant C, Deniaud J. Fetal ventilation and craniomaxillary development. Orthod

Fr. 73.83-107; 2002.Talmant JC, Lumineau JP. Therapeutic approach to cleft lip-maxilla-palate: for normal facial

growth. A protocol and various techniques to restore nasal respiration. Orthod Fr. 75:297-319; 2004.

Talmant JC, Talmant JC, Lumineau JP. A functional approach in the primary treatment of labial-alveolar-velopalatine clefts for a minimum of sequels. Rev Stomatol Chir Maxillo-fac. 108:255-63;2007.

Warren DW, Hairfield WM. The nasal airway in cleft palate. In: Bardach J, Morris HL, ed. Multidisciplinary management of cleft lip and palate. Philadelphia: W.B. Saunders Com-pany,1990.

http://www.ncbi.nlm.nih.gov/pubmed?term=%22Lambrecht JT%22%5BAuthor%5D

http://www.ncbi.nlm.nih.gov/pubmed?term=%22Kreusch T%22%5BAuthor%5D

http://www.ncbi.nlm.nih.gov/pubmed?term=%22Schulz L%22%5BAuthor%5D

http://www.ncbi.nlm.nih.gov/pubmed/10679856

http://bswww.mf.uni-lj.si/pls/bs/BS_full_rec?lang=SLO&c_docid=13024






http://www.ncbi.nlm.nih.gov/pubmed?term=%22Mars M%22%5BAuthor%5D










http://www.ncbi.nlm.nih.gov/pubmed?term=%22Shetye PR%22%5BAuthor%5D











Photometric evaluation of bclp patients after primary columella lengthening: 10 year follow-up

Meazzini MC, DMD, MMSc1, Tavecchia MG, DDS, Brusati R, MD1

University of Milan, Regional Center for CLP, Department of Maxillo-Facial Surgery1, San Paolo Hospital, Milan, Italy

SummaryNasal stigmata in BCLP patients are a short columella and a flattened nasal tip. We

evaluated the nasal aesthetics of children with bilateral cleft lip and palate, operated with the Cutting primary columella lengthening technique, associated to a Grayson orthopedic naso-alveolar molding. Two groups of patients at 10 years of age were compared through normalized photogrammetry. Cutting Group: 11 patients treated with the Grayson-Cutting technique; control group: 39 age matched normal pre-school children. With the Cutting-Grayson technique the columella length and nasal tip protrusion are close to normal, while naso-labial angle and interalar distances are excessively wide. At this time none of the patients operated with this technique have needed secondary columella lenghthening.

IntroductionSecondary nasal deformity in bilateral cleft lip and palate patients has constrained

surgeons to perform secondary columella elongation with various techniques (Millard, 1958 and Brauer and Foerster, 1966). General dissatisfaction with the postoperative aspect of children with bilateral cleft lip and palate stimulated to find new methods to correct the nasal deformity simultaneously with the lip deformity (Mulliken, 1995). Therefore, new surgical approaches, based on the concept that “the columella is in the nose”, were designed. Cutting and Grayson presented a new approach to primary columella lengthening using presurgical naso-alveolar molding combined with a one stage lip, nose and alveolar repair, through an open rhinoplasty with a retrogade method to approximate the domes, approaching them from the septal angle. This technique seemed to produce a symmetrical lip and nose, with a good projection of the tip (Cutting et al., 1998).

Since 1998 in the Milano cleft lip and palate center the surgical protocol (Bru-sati, 1992) for the treatment of patients with complete bilateral cleft lip and palate


Fig. 1: Frontal view of a bilateral cleft lip and palate patient prior to pre-surgical orthopaedics (A).Frontal view of the patient during pre-surgical orthopaedics with the plate in place (B).Frontal view of the patient at the completion of pre-surgical orthopaedics. Note the lengthening of the columella and the symmetry of the premaxilla (C).Basal view of the patient at 3 years of age treated with Grayson-Cutting technique (D).Basal view of the patient at 11 years of age treated with Grayson-Cutting technique (E).


was changed to a primary columella lengthening technique according to the Cutting technique, associated to a Grayson pre-surgical naso-alveolar molding (Meazzini et al., 2010). (Fig 1)

The objective of this study was to evaluate the results in terms of nasal aesthetics of this new approach.

Materials and methodsSince 1998 63 patients with bilateral cleft lip and palate were treated in the Milano

CLP center with the Cutting-Grayson approach of primary columella lengthening.11 of the patients have reached or are older than 10 years of age and therefore

where included in the study. Inclusion criteria were: 1) Bilateral complete cleft lip and palate, 2) Non syndromic, 3) Only caucasian patients of Italian origin, 4) Ac-curate lateral, frontal and basal standardized photographs taken at 5 years of age, 5) Naso-alveolar molding performed by the same orthodontist (MCM), 6) Primary chei-lorynoplasty performed by the same surgeon with the same surgical technique (RB).

The study compared two samples. Cutting Group: 11 consecutive patents (avera-ge age 10.5±0.3 years) who had undergone bilateral cleft lip repair with columella elongation according to a modified Cutting method and presurgical orthopedics and nasal molding, according to a modified Grayson technique. Normal children: 39 normal Caucasian pre-school northern Italian children (average age 10.6±0.6 years) from a local school.

Photogrammetry was adopted as an alternative to anthropometric direct measurements to obtain distances between facial landmarks using two-dimensional images. Linear and angular measurements were carried out and compared with an unpaired T-Test.

ResultsThe length of the columella (sn-c’) in the Cutting group (5.8±0.6mm) was on

average very close to the length in a normal child (6.6±0.4mm). There was no sta-tistical difference between these two groups. In terms of ratio of columella (sn-c’) to nasal tip protrusion (sn-prn), the ratio in the Cutting group (44±5%) was lower than in normal children (48±5%), though the difference was not statistically significant.

Tip protrusion (sn-prn) was very similar to that of normal children in both surgi-cal groups.

Naso-labial angle (NLA) was significantly larger in the Cutting (137.1±6.3°) group compared to our control sample which had on average, a NLA of 107.6±10.1°.

Our Cutting sample had a nostril width of 37.8±3.6mm versus 29.1±1.8mm of width in normal children (significant difference p<0.01). The ratio between nasal width (al-al) and intercanthal width (en-en) in the Cutting group was 119±13% versus a 97±8% ratio in the non cleft sample (p<0.01), even though intercanthal width in cleft patients is slightly larger than in normal patients.

ConclusionsThe combination of the orthopaedic stretching of the tissues and the surgical primary

lengthening of the columella allows for an improvement in nasal aesthetics which


is maintained at 10 years of age. Although interalar distance and NLA are still too whole this procedure allows for the reduction of the surgical burden in BCLP patients.

References1. BRAUER RO, FOERSTER DW. Another method to lengthen the columella in the double

cleft patient. Plast. Reconstr. Surg.;38:27-31,1966.2. CUTTING C, GRAYSON B, BRECHT L, SANTIAGO P, WOOD R, KWON S. Presurgical

columellar elongation and primary retrograde nasal reconstruction in one-stage bilateral cleft lip and nose repair. Plast. Reconstr. Surg.;101:630-639,1998.

3. MILLARD DR JR. Columella lengthening by a forked flap. Plast. Reconstr. Surg. Trans-plant Bull.;22:454-457,1958.

4. MULLIKEN JB. Bilateral complete cleft lip and nasal deformity: an anthropometric analysis of staged to synchronous repair. Plast. Reconstr. Surg.;96:9-23,1995.

5. MEAZZINI MC, ROSSETTI G, MORABITO A, GARATTINI G, BRUSATI R. Photometric evaluation of bilateral cleft lip and palate patients after primary columella lengthening. Cleft Palate Craniofac J. Jan;47(1):58-65,2010

6. BRUSATI R, MANNUCCI N. The early gingivoalveoloplasty. Preliminary results. Scand. J. Plast. Reconstr. Surg. Hand Surg.;26:65-70,1992




The Development of Molecular Therapies for Craniosynostosis.

Mooney MP1, Cray JJ Jr. 2, Losee JE3, Moursi AM4, Siegel MI5, Cooper GM6

1 Departments of Oral Biology, Anthropology, Surgery-Division of Plastic and Reconstructive Surgery, Orthodontics, and Communication Science and Disorders, University of Pittsburgh, Pittsburgh PA, USA 2 Department of Oral Biology, Georgia Health Sciences University, Augusta, GA, USA 3 Department of Surgery, Division of Plastic and Reconstructive Surgery, University of Pittsburgh and Pediatric Craniofacial Biology Laboratory, Children’s Hospital of Pittsburgh, Pittsburgh, PA, USA 4 Department of Pediatric Dentistry, New York University, New York, NY, USA 5 Departments of Anthropology and Orthodontics, University of Pittsburgh, Pittsburgh PA, USA 6 Departments of Surgery, Division of Plastic and Reconstructive Surgery Oral Biology, and Bioengineering, University of Pittsburgh and Pediatric Craniofacial Biology Laboratory, Children’s Hospital of Pittsburgh, Pittsburgh, PA, USA

SummaryThe birth prevalence of craniosynostosis (premature suture fusion) is 300-500 per

1,000,000 live births. Surgical management involves the release of the synostosed suture. In many cases however, the suturectomy site rapidly reossifies, further restricts the growing brain, and alters craniofacial growth. This resynostosis requires additional surgery which increases patient morbidity and mortality. This paper selectively reviews recent studies from our laboratory in bone biology and molecular biochemical pathways involved with suture fusion and novel tissue engineering techniques to deliver growth factors to surgical sites. Such molecular based therapies may prove useful as surgical adjuncts to help to manage postoperative resynostosis in infants with craniosynostosis.

IntroductionCraniosynostosis is a pathological condition defined as the premature fusion of the

sutures of the skull and is associated with secondary deformities in the cranial vault, cranial base, and midface. Such skeletal deformities often result in significantly elevated intracranial pressure, optic nerve compression, papilledema, and, if left uncorrected, optic atrophy, blindness, cognitive disabilities, and mental retardation. These severe craniofacial, ocular, and neural abnormalities can lead to extensive, costly, and often recurrent clinical and surgical management problems. 1, 2


Current surgical managements involve the extirpation of the fused suture along with extensive cranial vault reshaping including radical cranioplasties and post-operative helmet therapies. 1, 2 Although current surgical strategies often allow for normal intra-cranial volume and pressure, and corrected cosmetic deformities, the suturectomy site frequently reossifies very rapidly after surgery (in 30–100% of reported cases and as early as six months after surgery) and results in “resynostosis.” of the surgery site.1-4 Pancraniosynostosis has also been reported following surgical correction for single suture synostosis.4 Such resynostosis can lead to a secondary increase in intracranial pressure and further restrict craniofacial growth, necessitating additional surgical procedures and increasing patient morbidity and mortality. 1-4

The molecular mechanisms underlying synostosis of the suture and resynostosis are poorly understood. These processes probably involve an interaction of normal bone wound healing events and abnormal bone signaling pathways responsible for the premature suture fusion. Several bone morphogenetic proteins (BMP2, BMP4, and BMP7) have been shown to induce bone formation and they are expressed during bony wound healing.1,5-8 Noggin has been identified as an extracellular antagonist to BMPs. 6 Furthermore, Noggin has been shown to inhibit ectopic bone formation and has been used to inhibit membranous bone healing.1,5-7 With regard to the cranial sutu-res, Noggin has been shown to inhibit normal suture fusion in the mouse, implicating a role for BMPs in initial suture fusion as well. 1,5-7 Because of the apparent role of

Figure 1: Suggested interactions between genes, growth factors, and suture fusion. 8


BMP signaling in bone induction and in normal suture fusion, perturbation of BMP activity may lead to inhibition of excessive bone healing and prevent resynostosis.

The presence of several growth factors have been identified in fusing sutures (Figure 1). 1,6-9 Fgf2 is localized to the dura and mature bone throughout suture morphogenesis and becomes highly expressed in the osteogenic bone fronts of fusing sutures. All three Tgf-ßs are present in the dura and in the osteoblasts lining the dura mater and periosteal surfaces of the cranial bones, throughout suture morphogenesis and after the suture is fully formed. The suture matrix of fusing sutures contain high levels of Tgf-ß1 and Tgf-ß2, while Tgf-ß3 is absent from the bone fronts of fusing sutures. In contrast, Bmp2, Bmp4 and Bmp7 are present in the osteogenic bone fronts of developing sutures, but it is unclear whether their presence is continuous throughout suture development. The Bmp antagonist Noggin, is present in the center of sutures destined to remain open, while it is absent in sutures destined to fuse.

Based on these findings it appears that there are a number of growth factors in the Fgf and Tgf-ß families that may be involved in both bony wound healing and prema-ture suture fusion and may also be responsible for postoperative resynostosis noted in individuals with craniosynostosis. This paper reviews a number of our recent studies that have manipulated various growth factor levels or functions following surgical release of a prematurely fused suture in a unique and well described rabbit model of familial craniosynostosis.9-14 The development of these techniques may allow us to design targeted and complementary molecular and gene therapy strategies to decrease complications following postoperative resynostosis in infants with craniosynostosis.7-9

Materials and MethodsRabbit model of craniosynostosis. Previous studies have described the breeding

demographics, pedigree analysis, and phenotypic variability of a colony of rabbits with coronal suture synostosis.10-11 Briefly, the craniosynostosis that occurs in this colony varies from partial (delayed- or postgestational onset) to complete unilateral or bilateral coronal suture synostosis (early- or prenatal onset) (Figure 2). Early-onset synostosis in this model occurs around the 27th day of the 31-day rabbit gestation. The primary synostotic event in these rabbits leads to secondary deformations in the skull, cranial base, neurocapsule, and brain. Because of the similarity between the onset and progression of craniosynostosis in these rabbits compared with craniosyno-stosis that occurs in humans, this rabbit colony is a good animal model for human nonsyndromic craniosynostosis. 9-14

Growth factor manipulations. Three molecular strategies were used to assess the efficacy of molecular manipulation to inhibit postoperative resynostosis: 1) the use of neutralizing anti-Tgf-ß2 antibodies in the surgical site which may interfere with increased levels of Tgf-ß2 and inhibit resynostosis 9, 12 (Figure 1); 2) the use of Noggin protein in the surgical site which blocks BMP activity and may inhibit bony wound healing and resynostosis;13 and 3) the use of Tgf-ß3 protein in the surgical site which may inhibit Tgf-ß2 activity by competing for limited receptor sites, retard bony wound healing, and prevent resynostosis. 14

Samples. Twenty-nine, 10 day old New Zealand White rabbits (Oryctolagus cuniculus) with early onset, bilateral coronal suture synostosis (Figure 2) were used in the anti-Tgf-ß2 antibody study. 9, 12 The rabbits were randomly assigned to three


groups of rabbits as follows: Group 1) Suturectomy with no treatment, which served as the surgical control group (n = 9); Group 2) Suturectomy with non specific, control IgG antibody (100μg/suture) in a slow release collagen vehicle, which served as the antibody control group (n=9); and Group 3) Suturectomy with anti-Tgf-b2 antibody (100μg/suture) in a slow release collagen vehicle, which served as the treatment group (n=11).

Thirty-one, 10 day old New Zealand white rabbits with bilateral, early onset coro-nal suture synostosis (Figure 2) were used in the Noggin protein study. 13 The rabbits were randomly assigned to three groups of rabbits as follows: Group 1) Suturectomy with no treatment, which served as the surgical control group (n = 13); Group 2) Suturectomy with nonspecific BSA (10mg/suture) in a slow release collagen vehicle, which served as the protein control group (n = 8); and Group 3) Suturectomy with Noggin protein (10mg/suture) in a slow release collagen vehicle, which served as the treatment group (n = 10).

Twenty-nine, 10 day old New Zealand white rabbits with bilateral, early onset co-ronal suture synostosis (Figure 2) were used in the Tgf-ß3 protein study. 14 The rabbits were randomly assigned to three groups of rabbits as follows: Group 1) Suturectomy with no treatment, which served as the surgical control group (n = 13); Group 2) Suturectomy with nonspecific BSA (10mg/suture) in a slow release collagen vehicle, which served as the protein control group (n = 8); and Group 3) Suturectomy with Tgf-ß3 protein (10mg/suture) in a slow release collagen vehicle, which served as the treatment group (n = 8).

All rabbits were born in our ongoing breeding colony of congenitally synostosed rabbits at the University of Pittsburgh, Department of Anthropology vivarium.9-14 These protocols were all approved by the Institutional Animal Care and Use Committee at the University of Pittsburgh.

Figure 2: Skulls from 42 day old wild-type (left) and synostosed (right) rabbits.


Surgical technique.At 10 days of age, all rabbits were anesthetized with an IM injection (0.59ml/kg)

of a solution of 9l% Ketaset (ketamine hydrochloride) and 9% Rompun (xylazine, 20 mg/ml). The scalps were then shaved, depilated, and prepared for surgery. The calvariae were exposed using a midline scalp incision and the skin reflected laterally to the supraorbital borders. All animals received postoperative IM injections (2.5 mg/kg) of Baytril as a prophylaxis for infection. Following marker implantation, a 3mm long by 15mm wide strip of frontal and parietal bones including the entire length and width of the synostosed coronal suture were extirpated and removed in one piece from pterion to pterion (Figure 3) using a cutting burr. Care was taken to preserve the meningeal (fibrous) layer of the dura, the regional vascularity, and the amalgam markers in the frontal and parietal bones.

In rabbits in the suturectomy control groups, only the suturectomy was performed (Figure 3). The periosteal and skin incisions were then closed with 4-0 vicryl suture. In rabbits in the Antibody/Protein Control and Experimental groups, the suturectomy sites were immediately filled with 0.1cc of a slow resorbing collagen gel mixed with either the control antibody or protein or the experimental antibody or protein. The collagen vehicle was a highly purified, slow-resorbing (>63 days in rabbit perisutural tissues), 9,15 bovine collagen type I gel and was provided by NeuColl, Inc., (Campbell, CA). The antibodies and proteins were commercially available (R&D Systems, Min-neapolis, MN). Following injections, the periosteal and skin incisions were closed with 4-0 vicryl suture.

Data Collection. Longitudinal 3-D head computed-tomographic (CT) scan data of the suturectomy sites were obtained from all rabbits at 10, 25, 42 and 84 days of age, an age at which approximately 80-90% of calvarial and brain growth is completed in the rabbit. The 3-D CT scans were taken with the rabbits tranquilized. The rabbits were CT scanned in the sagittal plane using a GE HiSpeed Advantage Scanner (DFOV = 24.0-18.0cm; mA = 120-150; kV = 120) at a thickness of 1mm. The suturectomy site was manually traced, reconstructed, and the remaining defect area was calculated using Allegro Software (ISG Technologies, Atlanta GA) on a Sun Workstation. All measurements were taken blind as to rabbit group identity and intra-observer, repeated measurement reliability was calculated (r = 0.941; p < 0.01) on a randomly drawn sample (20%) of rabbits.

At 84 days of age (74 days postoperatively), the rabbits were euthanized with an IV injection of pentobarbital and the suturectomy sites and adjacent parietal and frontal bones were harvested for histological examination. The specimens were fixed

Figure 3: Surgical pictures showing size of removed suture (left), the suturectomy site (middle), and delivery of the collagen vehicle (right).


in 10% buffered neutral formalin, demineralized in a formic acid solution (Calex II, Fisher Scientific), dehydrated in a series of alcohol washes, and embedded in paraffin. The specimens were sectioned in the sagittal plane in the middle of both the right and left coronal sutures at a thickness of 5-7μm and stained with hematoxylin and eosin for conventional, qualitative bright field light microscopy.

ResultsThree-dimensional CT reconstructions (Figure 4) showed very extensive reossifica-

tion of the coronal suturectomy site in the Suturectomy control group, and to a lesser extent, in the IgG and BSA control groups, compared to the three experimental groups at 84 days of age. The sites showed extensive new bone formation at the margins of the suturectomy which eventually resynostosed in the control groups by 84 days of age (74 days postop) (Figure 4). In contrast, rabbits receiving postoperative anti-Tgf-b2 antibody, Noggin Protein, or Tgf-b3 Protein therapies showed some resynostosis and bony bridging of the suturectomy site by 84 days of age but most rabbits still had sizeable, patent defects at this time (Figure 4).

Mean suturectomy site area in both the Suturectomy, IgG and BSA control groups rapidly decreased through 84 days of age, leaving only approximately 30% of the defect area patent by this time, as measured from the 3-D CT scan reconstructions (Figure 5). IgG and BSA control rabbits showed a little slower reossification than the Suturectomy control rabbits at 25 and 42 days of age (Figure 5). In contrast, mean suturectomy site area in rabbits in the Anti-Tgf-b2 Antibody, Noggin Protein, or Tgf-b3 Protein groups all showed an increase in defect area in the first two weeks postoperatively and approximately 60% of the defect was still patent by 84 days of age (Figure 5). It is interesting to note that from 25 to 84 days of the age, after the initial plateau, the reossification pattern in all three experimental groups paralleled the reossification pattern in the control groups through 84 days of age (Figure 5).

At 84 days of age, Suturectomy, IgG, and BSA treated control suturectomy sites had extensive reossification and resynostosis in the suturectomy sites (Figure 6) as seen with bright field light microscopy. The new bone was seen to extend both anteriorly from the parietal bones and posteriorly from the frontal bones and form extensive bridging throughout the original suturectomy site. The bone was similar in morphology to the bone in the original suturectomy site margins and it was extensively thickened on the endocranial (dural) surface. Most of the bone in the suturectomy site was fused in the middle of the defect and no coronal sutures (patent or obliterated) were

Figure 4: 3D CT scans showing the suturectomy site at 84 days of age (74 days postop). Note the extensive, still patent suturectomy sites in the three rabbits that received growth-factor therapy.


observed in any rabbit. Since many of the coronal suturectomy sites were resynostosed it was impossible to discern whether the new bone came from the frontal or parietal bones. Given the 3-D CT scan reconstructions presented in Figure 4, appeared that the suturectomy site was closing uniformly and circumferentially in both the anterior and posterior directions, neither region contributing more that the other. In contrast, suturectomy sites treated with Anti-Tgf-b2 Antibody, Noggin Protein, or Tgf-b3 Protein showed much less reossification and resynostosis than controls. The suturectomy site

Figure 5: Graph showing wound healing of the suturectomy sites. Note the greater patency of the suturectomy sites in the three rabbits that received growth-factor therapy especially the initial increase at 25 days of age and the then parallel healing rates through 84 days of age.

Figure 6: Histophotomicrographs of the suturectomy sites from the six groups under study. Note the extensive reossification and resynostosis of the suturectomy sites in the three controls groups (left column) and the fibrous tissue and relative patency of the suturectomy sites.


margins were thinner, had more tapered edges, and showed extensive fibrous tissue formation in the original suturectomy sites compared to controls (Figure 6).

DiscussionResults from the present study demonstrated that a short course of treatment with

either a neutralizing antibody (anti-Tgf-b2 antibody), a BMP inhibitory protein (Noggin Protein), or a receptor blocking protein (Tgf-b3 Protein) following surgical release of a prematurely fused coronal suture all resulted in significantly less reossification of the suturectomy site in a craniosynostotic rabbit model. Although the specific genetic mutation is still unknown in this rabbit model, these results suggest that a general interruption in the bone healing pathway may be enough to prevent postoperative resynostosis and the sequelae that accompany it.9,12-14

While the mechanisms underlying the functions of these various growth factors are still being elucidated, a number of causal mechanisms have been suggested for each therapy. 6-14 Tgf-b2 over-expression has been linked to premature suture fusion and resynostosis by significantly increasing cell proliferation and differentiation thereby increasing the number of osteoblasts in the suturectomy site and increased osteogenesis. The use of anti-Tgf-b2 antibody obviously neutralizes the function of Tgf-b2 which may result in normal bone would healing and fibrous nonunions as seen histologically in synostotic rabbits treated with anti-Tgf-b2 antibody therapy. In contrast, BMP2, -4, and -7 have been found to be potent bone-inducing proteins that are expressed during normal bone healing which may result in increased postoperative osteogenesis and subsequent resynostosis. Noggin protein inhibits the ability of BMPs to produce bone and therefore retard postoperative osteogenesis and subsequent resynostosis which was seen in synostotic rabbits treated with Noggin protein therapy. Lastly, Tgf-b3 may have an inhibitory effect on suture fusion or postoperative osteogenesis and subsequent resynostosis by competing with or inhibiting the binding of Tgf-b2 to its receptor (Tbr-I). Down regulating Tbr-I binding or expression by Tgf-b3 may limit receptor access thereby reducing the mitogenic and osteogenic effects of Tgf-b2 and inhibiting postoperative osteogenesis and subsequent resynostosis as seen in synostotic rabbits treated with Tgf-b3 protein therapy.

All three therapies, delivered once within a slowly resorbing collagen gel, showed an effective inhibition of bone healing and delayed suturectomy site resynostosis in the rabbit model, as evidenced by the long-term persistence of defects in the calva-ria. It must be noted that the resultant defect was not the same size or shape of the original suturectomy (3x15 mm). In fact, the lateral margins of growth factor-treated defects were reossified and resynostosed. These data suggest that, although these growth factors were able to inhibit bone healing in this model, the delivery techni-que must be improved to inhibit bone formation in a more controlled and patterned manner. The inhibition of bone healing by growth factor therapy occurred primarily in the first two weeks after surgery, followed by the resumption of bone healing at rates similar to control defects. Notice that the reossification lines in the growth factor-treated groups closely paralleled the lines in the three control groups for the last three time points (Figure 6). The only difference is that the defects in the growth factor-treated groups increased in size between 10 and 25 days of age (indicative of normal neurocranial expansion), starting their normal healing from a different defect


size than the two control groups. The resumption of normal healing after 25 days of age in the growth factor-treated group may reflect the loss of growth factor function caused by its release and degradation from the collagen gel vehicle over time.

The data presented here show that a single dose of treatment with any of these growth factors can have lasting effects on bone formation despite the finding that the therapy had a limited temporal activity. In rabbits, 90% of the total brain growth is completed by 84 days of age, whereas, in humans, it takes nearly six years to complete 90% of brain growth. Therefore, therapies developed for use in humans must have a much longer period of effect than those studied in our rabbit model of simple, nonsyndromic craniosynostosis. For this reason, our group is also exploring other delivery mechanisms for protein-based and gene-based therapies. Although the genetic mutation responsible for craniosynostosis in this rabbit model has not been identified yet, data obtained from this model suggest that postoperative resynostosis probably occurs via a number of highly conserved signaling pathways. This should make the design and development of other biologically based therapies much easier, especially if the goal is to interrupt downstream signaling and reduce osteogenesis at the surgical site and not treat the primary genetic etiology of craniosynostosis. The combination of surgery and biologically-based therapy for the treatment of cra-niosynostosis promises to be an exciting endeavor in the next decade, especially once the technology in tissue engineering and gene therapy are improving dramatically.

References1. COHEN MM Jr, MACLEAN RE (eds.). Craniosynostosis: Diagnosis, Evaluation, and

Management. New York, NY, USA, Oxford University Press, 2000.2. PANCHAL J, UTTCHIN V.Management of craniosynostosis. Plast Rec Surg 111; 2032–2048:

2003.3. NORWOOD CW, ALEXANDER E JR, DAVIS CH JR, KELLY DL JR. Recurrent and

multiple suture closures after craniectomy for craniosynostosis. J Neurosurg 41; 715–719: 1974.

4. GREENE CS Jr: Pancraniosynostosis after surgery for single sutural craniosynostosis. Pediatr Neurosurg 29; 127-132: 1998.

5. SPECTOR JA, LUCHS JS, MEHRARA BJ, GREENWALD JA, SMITH LP, LONGAKER MT. Expression of bone morphogenetic proteins during membranous bone healing. Plast Rec Surg 107;124–134: 2001.

6. WARREN SM, BRUNET LJ, HARLAND RM, ECONOMIDES AN, LONGAKER MT. The BMP antagonist noggin regulates cranial suture fusion. Nature 422: 625–629: 2003.

7. RICE DP (ed.). Craniofacial Sutures: Development, Disease, Treatment. Basel, Karger, 2008.

8. OPPERMAN LA, OGLE RC: Molecular Studies of Craniosynostosis: Factors Affecting Cranial Suture Morphogenesis and Patency. In: Mooney MP, Siegel MI (Eds.). Under-standing Craniofacial Anomalies: The Etiopathogenesis of Craniosynostoses and Facial Clefting. New York, John W. Wiley and Sons, 2002.

9. MOONEY MP, MOURSI AM, OPPERMAN LA, SIEGEL MI. Cytokine therapy for cra-niosynostosis. Exper Opin Biol Ther, 4; 279-299: 2004.

10. MOONEY MP, LOSKEN HW, SIEGEL MI, LALIKOS JF, LOSKEN A, SMITH TD, BURROWS A. Development of a strain of rabbits with congenital simple, nonsyndromic coronal suture synostosis: Part I. Breeding demographics, inheritence pattern, and cranio-facial anomalies. Cleft Palate-Craniofac J, 31; 1-7: 1994.


11. MOONEY MP, LOSKEN HW, SIEGEL MI, LALIKOS JF, LOSKEN A, BURROWS A, SMITH TD. Development of a strain of rabbits with congenital simple, nonsyndromic coronal suture synostosis: Part II. Somatic and craniofacial growth. Cleft Palate-Craniofac J,31:8-6:1994.

12. MOONEY MP, LOSKEN HW, MOURSI A, BRADLEY J, AZARI K, ACARTURK O, COOPER GM, THOMPSON B, OPPERMAN LA, SIEGEL MI. Anti-Tgf-β2 antibody therapy inhibits postoperative resynostosis in craniosynostotic rabbits. Plast Reconstr Surg,119:1200-1212; 2007.

13. COOPER GM, CURRY C, BARBANO T, BURROWS AM, VECCHIONE L, CACCAME-SE J, NORBUTT C, COSTELLO BJ, LOSEE JE, MOURSI AM, HUARD J, MOONEY MP. Noggin inhibits postoperative resynostosis in craniosynostotic rabbits. J Bone Miner Res, 22:1046-1054: 2007.

14. KARSKI M, BARBANO TE, SMITH TD, CASS JM , NORBUTT C, DUDZIAK M , COSTELLO BJ, CRAY JJ, COOPER GM, LOSEE JE, SIEGEL MI, MOURSI AM, MOONEY MP. Suturectomy Site Healing Following Postoperative TGF-b3 Therapy in Craniosynostotic Rabbits. J Dent Res, 87, Special Issue A: 816; 2008.

15. PREMARAJ S, MUNDY B, MORGAN D, WINNARD PL, MOONEY MP, MOURSI AM. Sustained delivery of bioactive cytokine using a dense collagen gel vehicle. Arch Oral Biol, 51: 325-333; 2006.


Speech Activity and Participation (ICF-CY) in Children with Cleft Palate Speech

Neumann S.1,2

1 Speech-Language Pathology, Faculty of Human Sciences, University of Cologne, Germany 2 Institute of Neuroscience and Medicine (INM-3), Research Centre Juelich, Germany

Summary Since the publication of the ICF-CY in 2007, the WHO offers a biopsychosocial

classification of health states, which facilitates interprofessional exchange through the use of standardized language/codes. The ICF-CY implements the new components Activities, Participation and Context Factors. Children with cleft palate speech are at risk of having problems with speech-related social participation, which they are also aware of. The paper gives first insights into the subjective experience of affected children concerning their speech.

IntroductionChildren with cleft lip and/or palate (CL/P) are at an increased risk of a range of

physiological and psycho-social impairments related to their cleft condition (Howard and Lohmander 2011). To classify those health-related problems, the World Health Organization (WHO) urges member states to apply the International Classification of Functioning, Disability and Health - Children and Youth Version (ICF-CY) in clinical practice and research (WHO 2007).

International Classification of functioning, disability and health - children and youth version (ICF-CY)

The International Classification of Functioning, Disability and Health - Children and Youth Version (ICF-CY) for use with children and adolescents aged 0 to 18 was first published in November 2007 (WHO 2007). It provides a biopsychosocial view and a scientific basis for understanding health-related states, outcomes and determi-nants establishing a common language using standardized codes. This may improve communication among users of different health care professions around the world.


The Children and Youth Version reflects the further development of the ICF (WHO 2001) and covers the developmental aspects of childhood. Currently it is used and implemented by 191 countries in research and for documentation purposes. The ICF-CY implements more than 1500 codes, subdivided into four components: Body Structures, Body Functions, Activities and Participation, and Environmental Factors. Ueda and Okawa (2003) emphasized the importance of reflecting the individual subjective experience of a disability referring to each of the classified domains (Fig. 1). The components of Activities and Participation consider the psychosocial part of the classification (Fig. 2).

Speech Activities and Participation in children with CL/PLittle research on CL/P has been explicitly designed in accordance with the com-

ponents of the ICF. Even in the domains of Activities and Participation, no studies investigated occurrence of activity limitations and participation restrictions caused by cleft palate speech. For this, the author likes to present preliminary results of a retrospective analysis of speech activities and participation and their subjective im-pressions in ten children with CL/P. To get an impression of the impact of speech difficulties on children’s lives we used the Speech Participation and Activity of Children Questionnaire, version 2.0 (SPAA-C, McLeod 2003), Part A: Questions for the child (27 items). Ten children with repaired CL/P aged 6;0-9;11 yrs. (mean age of 8;3 yrs), five boys (age range: 6;7-9;10) and five girls (age range: 6;1-9;3) acted as subjects. All of them showed moderate to severe hypernasality, nasal emission, nasal turbu-lence (4/10), orofacial dysfunctions, cleft type characteristics (CTCs), slightly or moderately reduced intelligibility. Analyzing the results, all children indicated that they were concerned about their manner of speaking. Some of them (n=3) mention,

Figure 1: Overview of the ICF-CY components in correlation to subjective dimension


that they say words (n=2), or ‘letters’ (n=1) in a wrong way, one boy indicated that he felt like he had a different voice and missed out some ‘letters’ while speaking, one girl said she was inarticulate. Eight of the ten children indicated that they were asked to say things again and that they either felt angry (n=4) or sad (n=4) (Fig. 3). Asking them about their individual coping strategies, five children specified that they repeat the wrong words or phrases and try to speak more intelligibly, while three of them reported reacting in an aggressive way. One boy frequently asks his brother or friend to speak for him. These preliminary results show, that children with cleft palate speech might manifest speech-related limitations/restrictions in activity and communicative participation.

Consequences for future implementation of ICF-CY in children with CL/P Adopting the ICF-CY in children with CL/P their speech-language disability has to be seen as a result or consequence of a complex interaction between the health condition of the affected child with cleft palate speech, its personal factors and the environmental factors, which reflect the conditions under which the child grows up. We need to determine risk factors of activity limitations and participation restrictions correlating with the experience of growing up with a cleft-related speech impairment. For this, it is important to identify individual barriers/facilitators (Environmental Fac-tors) and also predisposing factors for participation restrictions directed by Personal Factors. A field-testing of published ICF-orientated assessments or qualitative methods in CL/P should start, like the SPAA-C (McLeod 2003), the Focus on the Outcomes of Communication Under Six (FOCUS, Thomas-Stonell et al. 2010) or using child

Figure 2: Characteristics of Activities and Participation domains


drawings (Mosaic Approach, Holliday et al. 2009). Ideally, new ICF-assessments especially for speech activity and participation in children with cleft palate speech will have to be developed. To implement the subjective dimension of limitations, new self-rating questionnaires might provide information about the subjective experience of activities & participation and perceived barriers concerning contextual factors. Such assessments will enable Speech-Language Pathologists to detect negatively influencing conditions, which may then be modified in the current environment of the individual

Figure 3: Results of Likert-scale about feelings about their speaking of children with cleft lip and palate using the SPAA-C (Neumann 2011, 191)


child. Intervention and goal setting can then be planned in a broader biopsychosocial approach in cooperation with the child and family. This development will then change Speech-Language Pathologists’ work from micro-praxis to macro-praxis (Cruice 2010). Assessment and therapy of children with cleft palate speech has to go beyond inter-vention focused solely on the child, but also include the family, siblings and friends.

ConclusionsAdopting the ICF-CY as a basic framework in Speech-Language Therapy in children

with CL/P may aid experts in identifying appropriate starting points for assessment, counseling, and therapy. Also it will hopefully expand the traditional way of inter-vention to a new therapy model, which focuses on speech activity, participation and the child’s environment.

ReferencesCRUICE, M. Considering the ICF as a conceptual framework for understanding quality of

life of adults with communication disorders: Strengths and limitations. Paper presented at the 28th World Congress of the International Association of Logopedics and Phoniatrics (IALP), 22-26 August, Athens, Greece. 2010

HAVSTAM C, LAAKSO K, LOHMANDER A, RINGSBERG K. Taking Charge of Commu-nication: Adults’ Descriptions of Growing up with a Cleft-Related Speech Impairment. Cleft Palate-Craniofac J. 48, 717-726, 2011

HOLLIDAY EL, HARRISON LJ & MCLEOD S. Listening to children with communication impairment talking through their drawings. J Early Child Res, 7, 244-263, 2009

HOWARD, S & LOHMANDER, A. eds. Cleft Palate Speech. Assessment and Intervention. West Sussex, Wiley-Blackwell, 2011

MCLEOD, S. Speech Participation and activity of children (SPAA-C) (version 2.0), 2003, Available at: http://athene.riv.csu.edu.au/~smcleod/ SPAAC2.pdf, Assessed January 14, 2011

NEUMANN, S. Listening to children with cleft lip and palate in Germany. In: ROULSTONE, S. & MCLEOD, S. (Eds.): Listening to children and young people with speech, language and communication needs. London, J&R Press, 183-189, 2011

NEUMANN, S & ROMONATH, R. Application of the International Classification of Fun-ctioning, Disability and Health - Children and Youth Version (ICF-CY) to Cleft Lip and Palate. Cleft Palate-Craniofac J, in press 2012

THOMAS-STONELL N, ODDSON B, ROBERTSON B. & ROSENBAUM P. Development of the FOCUS (Focus on the Outcomes of Communication Under Six): A Communication Outcome Measure for Preschool Children. Develop Med Child Neurol, 52, 47-53, 2010

UEDA, S & OKAWA, Y. The subjective dimension of functioning and disability: what is it and what is it for? Disab and Reh. 25, 596-601, 2003

WHO (WORLD HEALTH ORGANIZATION). International Classification of Functioning, Disability and Health, Children and Youth Version. Geneva, WHO, 2007

©2011 by MEDIMOND s.r.l. 25N914C0006

Incidence of cleft lip and palate/craniofacial on saudi arabia-hospital based

Al Johar A, Kandasam R, Subhan S

King Faisal Specialist Hospital and Researh Centre- Riyadh, KSA

Abstract: Cleft lip and palate are known to be the most common craniofacial defects throu-

ghout the world. The prevalence of individual conditions varies across geographic area and ethnicity groups. In Saudi Arabia, like most of Middle Eastern countries, the exact number of children born with oral cleft is unknown due to lack of national registration system and epidemiological studies.

The focus of this presentation is to present the prevalence of oral cleft and cra-niofacial anomalies in Saudi Arabia based on a tertiary care hospital experience, also the influence of consanguinity on the occurrence and recurrence of cleft.

More than 1400 patients diagnosed with oral cleft or craniofacial abnormalities were included in this presentation with great emphasis on classification and association of other congenital abnormalities. The results show male predominance in CLP(1.5:1), and female predominance in CP(0.9:1). About 33.6% of patients with CLP reported to have family history of clefts.

In general more than 50% of patient’s parents were consanguineously married. The incidence of oral cleft are more in the family of consanguineously married than non-consanguineously married.

Background: King Faisal Specialist Hospital and Research Centre established the Cleft lip and

Palate/Craniofacial registry in June 1999. The fact that orofacial clefts are easily de-scribed in the newborn, as compared to some other birth defects, makes their inclusion in the registry easy and complete.

Over the twelve year period (1999-2010), the registry registered a total of 1,418 patients (Male -783; Female - 635). The incidence and distribution of deformity clearly explained in the following tables:


Distribution of Cleft lip and Palate There were 699 males and 559 females with CLP. Cleft palate was most common

deformity followed by unilateral CLP and bilateral CLP. Overall, the male to female ratio was 1.25:1. However, a female preponderance was observed among cleft palate; lip and alveolar cleft; midline cleft (Table 1).

Table 1: Distribution of Cleft Deformities by Gender

Table 2: Number of Cases by Cranial/Facial Status and Gender

Distribution of Craniofacial AnomaliesOut of the 484 total craniofacial patients there were 263 male and 221 female

patients with a male to female ratio of 1.19.1. More than half of the patients (58.5%) had only facial, 24.8% had only cranial and 16.7% had both the anomalies (Table 2). There were 41 patients (cranial only 8; cranial and facial 33) having conditions like microcephaly, hydrocephaly, widely opened anterior fontanelle, etc. for whom the syndromic status was not applicable.


Family HistoryFamily history of orofacial/craniofacial deformities was reported by 404(28.5%)

patients and for 38 (2.7%) patients the information was unknown. Out of the 404 patients reported having a positive family history, 216 were male and 188 were fe-male. Table 3 shows status of familial history by major classification of anomalies.

Consanguinity by Family History Among consanguineously married 31.8% (247/777) had familial history whereas

among not related only 25.4% (152/599) had family history of anomalies.

Table 3: Family History by major Classification of Anomalies

Table 4: Shows the consanguinity in relation to classification of anomalies

Table 6 shows relationship of affected family members to the patient. It can be seen that, among first degree relatives, siblings had history of deformities more than any other relations. Siblings of 97 male patients and 81 female patients were affected either by orofacial or craniofacial.

Table 5: Number of Cases by Consanguinity and Family History

Table 6: Relationship to Patient among Positive Family History by Gender


Conclusion 1. There is a great need for national health educational programs on the impact

of consanguinity and facial deformity.2. To reorganized the health system in order to improve the medical care for

craniofacial patients.

References

3. JOHAR AA, RAVICHANDRAN K, SUBHANI SN, JARBA EA., Cleft lip and Palate and Craniofacial Anomalies Registry Commutative Report 1999-2010, Riyadh, Saudi Arabia, King Faisal Specialist Hospital and Research Centre, Riyadh, July 2011

4. AL JOHAR A, RAVICHANDRAN K, SUBHANI S., Pattern of cleft lip and palate in hospital-based population in Saudi Arabia: Retrospective study. Cleft Palate Craniofac J. 2008;45:592-6.

5. AL JOHAR A, RAVICHANDRAN K, SUBHANI S. Pattern of craniofacial anomalies seen in a tertiary care hospital in Saudi Arabia: Annals of Saudi Medicine 31(5), 488-492, 2011.


Surgical repositioning of downward displaced premaxilla in bilateral cleft lip and palate patients - our experience

Chirila L*, Tarhon RC**

*DDS, MD, PhD Department of Oral and Maxillofacial Surgery, Clinical Hospital OMF Dan Theodorescu Bucuresti, University of Medicine and Pharmacy Carol Davila Bucuresti Romania **trainee OMF surgery, Clinical Hospital Dan Theodorescu OMF Bucuresti, Romania

Summary Downward displacement of premaxilla (DDP) in bilateral cleft lip and palate

(BCLP) patients is not a common finding, but a somehow puzzling one. The medical litterature is rather vague on this topic and the guidelines are rare. We present, in this article, our experience in managing such cases, expecially the surgical repositioning of downward displaced premaxilla in bilateral cleft lip and palate patients.

IntroductionDownward displacement of premaxilla (DDP) in bilateral cleft lip and palate (BCLP)

patients leads to controversial problems. Vertical downward movement seem to appear due to overgrowth to the level of premaxilla- vomer suture and after cheiloplasty with increase tension of the upper portion of lip. We find this condition in older patients, after 8 years old. In our clinical practice that condition was not a common finding.

Material and method.Our experience in manage DDP is based on small number of patients ( 5 patients

aged 10 – 22 years old) . We performed premaxilla osteotomy and intrusion usually when vertical premaxilla excess is more than 6 mm. Fig.1. Fig. 2.

We perform surgical intervention via bucal approach.Vestibular abord was prefered because of improved visibility, facile condition for internal fixation device and bone grafting, and favorable trofic apport from palatinal area.Fig.3.

Bone segment was fixed with osteosinthesis material (Stryker).Fig 4.Fig 5.Postsurgical we used acrilic plate covering teeth or orthodonthic splint in order

to improve premaxilla rigidity, in special in cases were not performed bone grafting


Fig.1.Vertical excess and orthognathic device fixed on premaxilla in 10 y BCLP girl.

Fig. 2. Intraoral view of downward displacement of premaxila and severe colaps of alveolar segments resistant to orthognathic treatment

Fig.3. Bucal access and premaxilla subspinal osteotomy. Notice good visibility, and secure manipulation of tissues


at the same time with surgical intrusion of premaxilla. In two patients bilateral bone grafting with iliac bone block was realised at the time with premaxila repositioning.

Results.We did not have any major complications after surgery. We noticed a good blood

circulation in the premaxilla, which was the premise for the good integration of the bone grafts. No wound dehiscence, infection or exposed bone grafts was recorded. The surgery markedly improved the premaxilla position, occlusion and facial appearance. Both functional and aesthetical results were acceptabile in all five cases.

Discussion.The downward displacement of the premaxilla leads not only to occlusal and

functional problems, but also to aesthetical issues, with a direct psychological reper-

Fig 4 .Premaxilla secured in place with osteosinthesis material. Small amount of bone and incisors apex make screws placement more difficult

Fig 5. Postoperative view of repositioned premaxila and suplimentary stabilised with orthodontic splint


cussion. Furthermore we rally to the considerents of Miyasaka et al., concerning the use of surgical treatment in children under 7 years of age, i.e.before school. However, we didn’t have any patient under 7 y.o.in our study group. Another important aspect resides in the type on dentition. Liou’s technique of premaxilla orthopedic intrusion, correct the premaxilla deformity, mainly from orthopedic intrusion ( about 3 mm), dental orthodontic intrusion of maxillary incisors( about 3,5 mm) and shortening of the premaxillary dentoalveolar height.This technique is suitable for deciduous dentition.

None of our patients had decidous dentition. The treatment plan was more chal-lenging because of the inconsistent orthognathic treament done in the past, and the ”negative fillings” regarding dental hygene in some of our patients.

Last, but not least, one should consider the extent of the vertical excess. Even though the literature data are very scarce, we embrace the protocol postulated by Meazzini et al.: orthodontic intrusion is a method suitable in the mixed dentition, when the vertical excess doesn’t exceed 7-8 mm. The surgical intrusion is used in mixed dentition when the vertical excess is larger than 8 mm. Also, it is the method of choice in permanent dentition. Four of our cases showed a vertical excess larger than 8 mm, which is consistent to Meazzini’s criteria. One subject had a vertical excess of only 6 mm., but anamnestically we noticed a lack of compliance to the orthodon-tic treatment. Even more the subject was 22 y.o. Based on these considerations we choosed the surgical treatment, which proved to be a good solution.

Regarding the surgical approach, we favoured the vestibular abord because of improved visibility, facile condition for inserting the internal fixation device and bone grafting, and an excellent trofic apport from the palatinal area. The bone fragment was fixed with osteosinthesis material (Stryker) in all five cases. The dificulty appears to reside in stabilisating the premaxilla with miniplates only, especially in cases with delayed bone graft. Use of bipolar fixation, oseous and alveolar is mandatory. The adult patients have no risk of tooth germ damage. All five subjects in our study were over 10 y.o. Their developement allowed the internal fixation with miniplates. Also, the literature data sugest an impairment on maxillary growth as a side effect of the plates. Surgical premaxilla osteotomy with internal fixation appears to be harmless to subsequent growth in our series.

The iliac bone grafting performed in the same surgical step with the osteotomy offers a bone bridge for premaxillary fixation. Also, it helps alligning the permanent teeth. We used bilateral bone grafting with iliac bone block at the same time with premaxilla repositioning in two of our patients. We did not record any bone graft rejection, necrosis or any sign of infection. Mishimmaki et al.(cit.by.Miyasama) also reports 100% success in six cases. The key here is the ability to mainain an ade-quate blood supply to the premaxilla. The vestibular abord we used allowed a good trofic apport from the palatinal area. Nevertheless, to prevent poor blood supply in the premaxilla, the literature data emphasise that grafting with osteotomy should be limited to complete BCLP children whose alveolar clefts are not too wide, who do not have many scars associated with the initial surgery and whose periosteum is avaible for graft beds.

Post surgery, we used acrilic plate covering the teeth or orthodonthic splint in or-der to improve the rigidity of premaxilla, especially in those cases where we did not performe bone grafting at the same time with surgical intrusion of premaxilla. Albeit Miyasaka et al.criticise the bite splint for deviating the segment from its intended


position, we did not encounter this problem. However, it was necesary to rebuild the alveolar splint due to fracture one week after surgical intrusion in three cases.

Conclusions.Facial dismorphysm in BCLP due to DDP could increase psychologic problems,

make bone grafting difficult and impare orthodonthic treatment.Orthopedic and ort-hodontic intrusion are suitable for deciduous or mixed dentition with vertical excess 6-7 mm or less. In our cases we met higher vertical excess or resistant cases to any previous orthopedic-orthodontic movement. Surgery is advocated in cases where the vertical excess is over 6 - 8 mm. and for definitive dentition.Surgical premaxilla osteotomy appears to be harmless to subsequent growth in our series( aged from 10 years old). For increased rigidity of intruded premaxilla we recomand bipolar fixation, osseous and dentoalveolar splint.

References:FRIEDE H., PRUZANSKY S.: ”Long-term Effects of Premaxillary Setback on Skeletal Profile

in Complete Bilateral Cleft Lip and Palate” in Cleft Palate Journal, Vol.22, No.2, pp.97-105, April 1985

LIOU EJ, CHEN PK, HUANG CS, CHEN YR.: ”Orthopedic intrusion of premaxilla with distraction devices before alveolar bone grafting in patients with bilateral cleft lip and palate” in Plast Reconstr Surg. 2004 Mar;113(3)

MEAZZINI M.C., LEMATTI L., MAZZOLENI F., RABBIOSI D., BOZZETTI A., BRUSA-TI R.: ”Vertical Excess of the Premaxilla in Bilateral Cleft Lip and Palate Patients: A Protocol for Treatment” in The Journal of Craniofacial Surgery, Volume 21, Number 2, pp.499-502, March 2010

MIYASAKA M., AKAMATSU T., YAMAZAKI A., OCHIAI T., TANAKA R., SAKUMA Y., SAKATA N., NIIKRA R.: “A New Device for Fixing the Premaxilla with Osteotomy in a Child with Complete Bilateral Cleft Lip and Palate in Mixed Dentition Period – A Preliminary Case Report” in Tokai J.Exp.Clin.Med., Vol.33, No.1, pp.21-27, 2008

E J.W. LIOU, P K.T. CHEN, Management of Maxillary Deformities in Growing Cleft Patients, in Cleft Lip and Palate 2nd Edition, Editor Samuel Berkowitz, Springer-Verlag Berlin Heidelberg 2006

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Nasoalveolar molding with simple and esthetic appliances

Daratsianos N., Jäger A.

Department of Orthodontics, University of Bonn, Germany

SummaryThe aim of this presentation is to show the effects of presurgical infant orthopedics

with simple appliances on the development of the alveolar, nasal and lip tissues of cleft patients. Presurgical infant orthopedics of the alveolar tissues (alveolar molding) with palate plates has been used for decades in many cleft centers with controversial long-term effects for the patient. In the last years a method of presurgical molding of the alveolar and nasal tissues (nasoalveolar molding) was developed. It promises benefits for the surgical procedures as well as superior nasal symmetry and facial esthetics with evidence based middle-term effects. In the cleft center of the University Hospital of Bonn nasoalveolar molding with simple and esthetic appliances is used before and after lip surgery. The presentation focuses on the appliances and case reports. Nasoalveolar molding is a powerful and simple tool for the therapy of cleft patients and can help the surgeon to achieve superior facial esthetics and functional results.

IntroductionPresurgical infant orthopedics of the alveolar tissues (alveolar molding) with palate

plates has been used for decades in many cleft centers with controversial long-term effects for the patient [1-5]. The concept of an intraoral device to reposition the cleft alveolar segments was first introduced by McNeil in 1950 [6]. Latham introduced a pin-retained appliance to simultaneously retract the premaxilla and expand the po-sterior segments [7]. Hotz described the use of a passive orthopedic plate to slowly align the cleft segments without retraction of the premaxilla [8].

In the last decades a method of presurgical molding of the alveolar and nasal tissues (nasoalveolar molding) was developed [9-11]. It promises benefits for the surgical procedures as well as superior nasal symmetry and facial esthetics with evidence based middle-term effects [9, 12, 13]. The appliances usually consist of an intraoral plate and extraoral attachments such as tapes, tapes and springs. They attempt to mold the maxillary arch, reshape the nasal structures and reduce post surgical scarring [14].


Figure 1: A NAM-device for a unilateral cleft. The device consists of an intraoral passive plate and an extraoral stent covered with soft acrylic. A little loop allows an easier activation.

Figure 2: Example of a treatment with NAM on a patient with unilateral cleft lip and palate. A: Initial photo-graph. B: Begin of the NAM-treatment. The extraoral stent pushes the nose forward and upward increasing the convexity of the affected nostril. The tape pulls the lip parts together. C: Result of the NAM-treatment before lip surgery: The convexity of the affected nostril and the symmetry of the nose are increased and the cleft is smaller. D: The patient approx. one year after lip surgery with a relatively stable result.


The aim of this presentation is to show the effects of presurgical infant orthopedics with simple appliances on the development of the alveolar, nasal and lip tissues of cleft patients.

Case presentationsIn the cleft center of the University Hospital of Bonn nasoalveolar molding (NAM)

is used as a standard procedure before and after surgery. Unlike in other centers, where complicated appliances are used incorporating excessive extraoral tapeping, we have focused on using simple and esthetic devices to increase the compliance of the parents.

A typical device for a unilateral cleft is presented in Figure 1. The device consists of an intraoral passive plate, which is grinded to allow the alveolar segments to deve-lop. Adhesive gel is used for anchorage. An extraoral stent covered with soft acrylic is fixed at the anterior part of the device and activated to push the nasal structures to a normal position. A little loop allows an easier activation. At the same time a skin-colored tape is used to stretch the parts of the lip and bring them together. The biomechanics of the device are presented in Figure 2b. The stent is hidden inside the lip cleft behind the tape. The device allows the cleft to ostensibly disappear (Figure

Figure 3: Extraoral photographs of the patient in Figure 2. A: Initial photograph. B: Begin of the NAM-treatment. The extraoral stent pushes the nose forward and upward increasing the convexity of the affected nostril. The tape pulls the lip parts together. The cleft is well camouflaged. C: Result of the NAM-treatment before lip surgery: The convexity of the affected nostril and the symmetry of the nose are increased and the cleft is smaller. D: The patient approx. one year after lip surgery with a favorable and relatively stable result.


3b). The patient can therefore be better socialized and the compliance of the parents is generally high.

The treatment allows the alveolar and lip cleft to get smaller, the convexity of the cartilage of the affected nostril to increase and the nose to become symmetrical in all. As a result, the patient shows less scaring and agreeable nose / lip configuration postoperatively (Figures 2 and 3).

The procedure can be also applied to patients with bilateral clefts. The appliance has two stents (Figure 4b and 5b), that are connected to each other with a soft-acrylic bridge at a later stage of the treatment (Figure 4b and 5c). While the two stents push the nostrils forward and upward, the acrylic bridge applies a posterior force vector on the subnasal point. A vertical adhesive tape pulls the prolabium downwards to the plate and stretches it. The horizontal tape brings the separated lip parts together. Adhesive gel and the vertical tape contribute to the anchorage of the device. The biomechanics are presented in Figure 5c. The stents are hidden behind the adhesive tapes. The device and the extraoral tapes contribute to the camouflage of the cleft and can be easily handled by the parents.

The treatment allows the alveolar and lip clefts to get smaller, the convexity of the cartilage of the nostrils and the length of the columella to increase (Figure 5d).

Figure 4: A patient with bilateral cleft lip and palate. A: Initial photograph. B: Intermediate stage of the NAM-treatment. The extraoral stents push the nose forward and upward. The stents are connected to each other with a soft-acrylic bridge which applies a posterior force vector on the subnasal point. A vertical adhesive tape pulls the prolabium downwards to the plate and stretches it. The horizontal tape brings the separated lip parts together. The stents are hidden behind the adhesive tapes. The device and the extraoral tapes contribute to the camouflage of the cleft.


DiscussionPresurgical nasoalveolar molding is emerging as a standard in contemporary ma-

nagement of cleft lip palate deformity repair in many centers. However the long term benefits are controversial. While most studies about alveolar molding with intraoral plates show no significant long term effects on any parameter [1-4, 15-17], the use of NAM-devices seem to have a lasting benefit for nasal esthetics [9, 12, 13].

The use of NAM-devices can be easily integrated in a cleft center, especially when using relatively simple devices. The fabrication is quick and many steps can be accomplished chair-side. The compliance of the parents is improved because of the easy handling and the apparent and immediate camouflage of the deformity.

Avoiding complicated extraoral taping for anchorage of the NAM-device contribu-tes to the easy handling and good extraoral esthetics. Apart from that, only a small posterior force vector on the nasomaxillary complex is applied, probably contributing to a more favorable growth. However, the simple NAM-procedure as shown here can have some limitations concerning the results, especially when evaluating the length of the columella of patients with bilateral clefts (Figure 5d). The posterior force vector on the subnasal point could be increased by using extraoral tapes pulling the whole device backwards, contributing to a better definition of the subnasal point and incre-ased lengthening of the columella [13], but also making the handling more difficult and the devices less esthetic.

Figure 5: Intraoral photographs of the patient in Figure 4. A: Initial photograph. B: Begin of the NAM-treatment. Two extraoral stents push the nose forward and upward. A vertical adhesive tape pulls the prolabium downwards to the plate and stretches it. The horizontal tape brings the separated lip parts together. C: At a later stage of the treatment a soft-acrylic bridge is added between the stents to apply a backward force vector on the subnasal point. The biomechanics of the treatment is shown. D: Result of the NAM-treatment before lip surgery. The length of the columella has increased.


ConclusionsNasoalveolar molding is a powerful tool for the therapy of cleft patients and can

help the surgeon achieve superior facial esthetics and functional results. Using simple NAM-devices can contribute to an easy integration in the clinic, improved compliance and good immediate extraoral esthetics.

References1. PRAHL, C., et al., A randomised prospective clinical trial into the effect of infant ortho-

paedics on maxillary arch dimensions in unilateral cleft lip and palate (Dutchcleft), Eur J Oral Sci, 5, p. 297-305, 2001

2. PRAHL, C., et al., A randomized prospective clinical trial of the effect of infant ortho-pedics in unilateral cleft lip and palate: prevention of collapse of the alveolar segments (Dutchcleft), Cleft Palate Craniofac J, 4, p. 337-42, 2003

3. BONGAARTS, C.A., et al., Infant orthopedics has no effect on maxillary arch dimen-sions in the deciduous dentition of children with complete unilateral cleft lip and palate (Dutchcleft), Cleft Palate Craniofac J, 6, p. 665-72, 2006

4. PRAHL, C., et al., Infant orthopedics in UCLP: effect on feeding, weight, and length: a randomized clinical trial (Dutchcleft), Cleft Palate Craniofac J, 2, p. 171-7, 2005

5. KONST, E.M., et al., Cost-effectiveness of infant orthopedic treatment regarding speech in patients with complete unilateral cleft lip and palate: a randomized three-center trial in the Netherlands (Dutchcleft), Cleft Palate Craniofac J, 1, p. 71-7, 2004

6. MCNEIL, Orthodontic procedures in the treatment of congenital cleft palate., Dent Rec., 70, p. 126-32, 1950

7. GEORGIADE, N.G. and LATHAM, R.A., Maxillary arch alignment in the bilateral cleft lip and palate infant, using pinned coaxial screw appliance, Plast Reconstr Surg, 1, p. 52-60, 1975

8. HOTZ, M. and GNOINSKI, W., Comprehensive care of cleft lip and palate children at Zurich university: a preliminary report, Am J Orthod, 5, p. 481-504, 1976

9. MAULL, D.J., et al., Long-term effects of nasoalveolar molding on three-dimensional nasal shape in unilateral clefts, Cleft Palate Craniofac J, 5, p. 391-7, 1999

10. DOGLIOTTI PL, B.R., LOSOVIZ E, Tratamiento no quirúrgico de la deformidad nasal en el fisurado., Revista del Ateneo Argentino de Odontologia, 1, p. 31-5, 1991

11. GRAYSON, B.H., CUTTING, C., and WOOD, R., Preoperative columella lengthening in bilateral cleft lip and palate, Plastic and reconstructive surgery, 7, p. 1422-3, 1993

12. BARILLAS, I., et al., Nasoalveolar molding improves long-term nasal symmetry in com-plete unilateral cleft lip-cleft palate patients, Plast Reconstr Surg, 3, p. 1002-6; discussion 1007-9, 2009

13. LEE, C.T., et al., Nasoalveolar molding improves appearance of children with bilateral cleft lip-cleft palate, Plast Reconstr Surg, 4, p. 1131-7, 2008

14. GRAYSON, B.H. and CUTTING, C.B., Presurgical nasoalveolar orthopedic molding in primary correction of the nose, lip, and alveolus of infants born with unilateral and bilateral clefts, Cleft Palate Craniofac J, 3, p. 193-198, 2001

15. ROSS, R.B., Treatment variables affecting facial growth in complete unilateral cleft lip and palate, Cleft Palate J, 1, p. 5-77, 1987

16. ROSS, R.B. and MACNAMERA, M.C., Effect of presurgical infant orthopedics on facial esthetics in complete bilateral cleft lip and palate, Cleft Palate Craniofac J, 1, p. 68-73, 1994

17. SHAW, W.C., et al., A six-center international study of treatment outcome in patients with clefts of the lip and palate: Part 5. General discussion and conclusions, Cleft Palate Craniofac J, 5, p. 413-8, 1992

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Validation of the Use of Sphincter Pharyngoplasty in the Treatment of VPI with Sagittal Pattern Closure

Elbarbary Amir*1, Ali Ahmed*1, Basiony SamiaΦ1, Abou-El Saad TamerΦ2, & Amer AymanΦ2

Departments of Plastic Surgery* & PhoniatricsΦ, Ain-Shams1 & Mansoura2 Universities, EGYPT

Introduction/Background:Velopharyngeal insufficiency occurs in a considerable number of patients following

cleft palate repair1-4. Substantial uncertainty occurs in choosing between sphincter pharyngoplasty and pharyngeal flap in restoring the velopharyngeal function5-6. Pharyngeal flaps have been recommended when the residual velopharyngeal gap is sagittal, while sphincter pharyngoplasty is indicated when the residual velopharyngeal gap is coronal7-8. However, supporters of the sphincter pharyngoplasty find that the pharyngeal flap divides the velopharyngeal area into two lateral ports. They believe that it disturbs the palatopharyngeus–superior constrictor sphincter with loss of its mechanical advantage and weakens the posterior and lateral pharyngeal wall movement ultimately defeating the purpose of its use9.

Materials & Methods:This prospective blinded randomized study sought to assess the treatment outco-

me of sphincter pharyngoplasty as compared to superiorly based pharyngeal flaps in children suffering residual velopharyngeal insufficiency with sagittal gaps following palatal repair. The velopharyngeal gap areas were more than 5 mm with moderate to severe degree of open nasality. The study population were randomly divided into two groups, the first underwent sphincter pharyngoplasty while the second a superiorly based pharyngeal flap. Preoperatively, patients underwent in-depth auditory perceptual assesment, nasometric, nasoendoscopic, and videofluoroscopic evaluations for sym-ptoms of velopharyngeal insufficiency. To ensure reliable results, patients were chosen so that there was no statistical significance among all of the preoperative variables between the two groups as well as the age of patients at the time of primary palatal and secondary velopharyngeal repair.


Fig. (1) Preoperative nasoendoscopic examination in group I. (A) at rest and (B) during phonation of /a/ sound

Fig. (2): Postoperative nasoendoscopic examination in group I. (A) at rest and (B) during phonation of /a/ sound

Fig. (3) Preoperative nasoendoscopic examination in group II. (A) at rest and (B) during phonation of /a/ sound


Results:Six to twelve weeks following the surgical procedure the children underwent the

same thorough evaluation. Although postoperative evaluation in both groups prior to speech therapy were comparable in the outcome, the degree of intelligibility impro-ved significantly in patients whom underwent sphincter pharyngoplasty as compared to the pharyngeal flap group (Table 1). Furthermore, the velopharyngeal gap was found to be significantly narrower at rest with the pharyngeal flap as compared to the sphincter pharyngoplasty, which could explain why patients in the pharyngeal flap group are more prone to airway obstruction complications. Finally, the dynamic property of the sphincter pharyngoplasty could be verified based on the significant reduction of velopharyngeal gap between the resting position & that at the time of phonation (Table 2).

Fig. (4): Postoperative nasoendoscopic examination in group II. (A) at rest and (B) during phonation of /a/ sound

Table (1): Postoperative descriptive data of APA for both groups. The degree of intelligibility improved significantly (P value 0.025) in patients whom underwent sphincter pharyngoplasty as compared to the pharyngeal flap group


Conclusion:The results of this study confirm that sphincter pharyngoplasty can be used effi-

ciently in patients suffering residual velopharyngeal insufficiency with sagittal gaps following palatal repair where pharyngeal flaps are originally indicated.

References: 1. Shprintzen RJ, Golding-Kushner KJ: Evaluation of velopharyngeal insufficiency. Otolarngol

Clin North Am 22: 519, 1989.2. Pamplona M, Ysunza A, Guerrero M, Mayer I, García-Velasco M.: Surgical correction

of velopharyngeal insufficiency with and without compensatory articulation. Int J Pediatr Otorhinolaryngol 34(1-2): 53, 1996.

3. Kuehn DP, Moller KT: Speech and language issues in the cleft palate population: The state of the art. Cleft Palate Craniofac J 37: 348, 2000.

4. Ysunza A, Pamplona M, Mendoza M, Molina F, Martinez P, García-Velasco M, Prada N: Surgical treatment of submucous cleft palate: a comparative trial of two modalities for palatal closure. Plast Reconstr Surg 107(1): 9, 2001.

5. de Serres LM, Deleyiannis FWB, Eblen LE, Gruss JS, Richardson MA , Sie KCY: Results with sphincter pharyngoplasty and pharyngeal flap. Int J of Ped Otorhinolaryng 48: 17, 1999.

6. Abyholm F, D’Antonio L, Davidson-Ward SL, Kjoll L, Saeed M, Shaw W, Sloan G, Whitby D, Worthington H, Wyatt R, VPI Surgical Group: Pharyngeal flap and sphincterplasty for velopharyngeal insufficiency have equal outcome at 1 year postoperatively: results of a randomized trial. Cleft Palate Craniofac J 42(5): 501, 2005.

7. Sloan GM: Posterior pharyngeal flap and sphincter pharyngoplasty: the state of the art. Cleft Palate Craniofac J 37(2): 122, 2000.

8. Marsh JL: Management of velopharyngeal dysfunction: differential diagnosis for differential management. J Craniofac Surg 14(5): 621, 2003.

9. Huang M H, Lee ST, Rajendran K: Anatomic Basis of Cleft Palate and Velopharyngeal Surgery: Implications from a Fresh Cadaveric Study. Plast Reconstr Surg 101 (3): 613, 1998.

Table (2): Postoperative descriptive data of videofluroscopy for both groups. Interpretation of data reveals a wider airway at rest with the sphincter pharyngoplasty along with dynamic property during phonation. The velopharyngeal gap is significantly narrower at rest (p value 0.004) with the pharyngeal flap group as com-pared to sphincter pharyngoplasty group which could explain why patients with pharyngeal flaps are more prone to airway obstruction. The velopharyngeal gap in the sphincter pharyngoplasty group is significantly reduced (P value 0.002) at the time of phonation as compared to rest which indicated the dynamic nature of sphincter pharyngoplasty. The succes of pharyngeal flap relies on elevating a large static flap that closes the velopharyngeal gap at the expense of a narrow airway at rest.

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BSSO conducted by piezoelectric saw - review of 187 cases

Jagielak M.1, Michalski P.1, Jagielak A.2, Socha M.3, Rogus P.3

1Mother and Child Institute, Warsaw, 2 Private orthodontic practice, Raszyn, 3 Cranio-Maxillo-Facial Surgery The Infant Jezus Hospital, Warsaw, Poland

Introduction:During the last few years the application of piezosurgical instrumentiation in the

field of craniomaxillofacial surgery gained much of interest and on the other hand have generated discussions concerning its usefulness and practicality comparing to the traditional and well recognized armamentarium1,8. In our study we evaluated the advantages of the clinical application of piezoelectric saw for the mandibular ramus sagittal split osteotomy (mostly bilateral – BSSO). Three different piezoelectrics sy-stems were compared. In the years 2007-2011 we performed 324 procedures using piezoelectric equipment from various manufacturers - Mectron, NSK, Esacrom.

Fig1. Various piezoelectric sets


Material:In 187 cases the osteotomies were performed with piezoelectric saw, in 137 ca-

ses piezosurgery was used as a complementary technique, generally in guiding the lines of osteotomies and in situations with the increased probability of complication occurrence (injuries to vessels, nerves). In majority of cases (85%) the BSSO was a part of bimaxillary procedures.

Methods:With the increasing number of the performed osteotomies by means of piezoelectric

saw we observed the major shortening of the operation time equaling the traditional method. The new method proved to be very effective in cases with the bad split complication - incomplete mandible splitting or mobilization of the neurovascular

Fig 2. The mandibular ramus osteotomy performed traditionally – rose and Linemann burrs in use

Fig 3. Osteotomy performed with the use of different pieces of piezoelectric saw – thin an smooth line of the osteotomy


bundles4,7. During the first operations performed by piezo-saw few saw blades were broken due to leak of experience and using to high forces. No disruption of the man-dibular nerve was reported. Time of lack of sensation and paresthesias were shorter and less frequent. The marked decrease in the blood loss (average 410 ml versus 1280 ml with rotating tools in bimaxillary osteotomies) and in the postoperative swelling was observed. The rotating pieces was used as a support in creating the notch for piezoelectric saw above the foramen of the mandible to avoid slipping of the bone surface and thus eliminating the risk of undesired damage to the adjacent soft tissues. Additionally reciprocating saws were used for smoothening sharp edges of the bone created at the interface of osteotomy lines. Straight and curved chisels were used for widening osteotomy line and to achieve split. In cases where inferior alveolar nerve remained within ramus of the mandible after the split piezoelectric allowed to cut the bone around and release and vessel harmlessly. In cases of rotation of the distal fragment of mandible piezoelectric saw was used to improve the overlapping and contact of the osteotomised bone fragments by cutting of the portion of bone in retromandibular region. In cases of retained 3rd molars in mandible piezoelectric saw enable to performed the simultaneous tooth extraction and the sagittal osteotomy without the increased risk of bad split.

Fig 4. BSSO in the retained 3-rd molars case

Results:The application of piezoelectric saw for mandibular osteotimized diminished

blood loss, soft tissue damage complications (especially injuries to inferior alveolar nerve bundle), allowed for performing the mandibular split in cases with retained 3rd molars thus shortening the overall treatment time. We observed that with the conse-cutive gaining of experience how to use piezoelectric pieces the time of procedures was shortening and finally equaled the time of traditionally performed osteotomies. On the other hand there are still places where the use of reciprocating and rotating instruments is very helpful1,8.

Conclusions:Piezoelectric devices create the breakthrough in the surgery of human skeleton

deformities. The future trends in improving the quality of piezoelectric instrumen-tation should be aimed at increasing their power, which could shorten the operation time. The research should be aimed of increasing their power and their applications.


Fig5. Hemifacial hypertrophy - ostectomies and ostotomies performed by piezoelectric saw.


References:1. Critical Evaluation of Piezoelectric Osteotomy in Orthognathic Surgery: Operative Tech-

nique, Blood Loss, Time Requirement, Nerve and Vessel Integrity Journal of Oral and Maxillofacial Surgery Volume: 66, Issue: 4, April, 2008, pp. 657-674 Landes, Constantin A.; Stübinger, Stefan; Rieger, Jörg; Williger, Babett; Ha, Thi Khanh Linh; et. al.

2. Comparison of postoperative pain: piezoelectric device versus microdrill European Archi-ves of Oto-Rhino-Laryngology Volume: 268, Issue: 9, September 2011, pp. 1279 - 1282 Crippa, Barbara; Salzano, Francesco Antonio; Mora, Renzo; Dellepiane, Massimo; Salami, Angelo; et. al.

3. Ultrasonic piezosurgery in orthognathic surgery and distractions osteogenesis International Journal of Oral & Maxillofacial Surgery Volume: 36, Issue: 11, November, 2007, pp. 1018 Majeed, R.; Sader, R.

4. 4.Piezosurgery-assisted sliding genioplasty: a method for reduction of complications. Re-view and case report European Journal of Plastic Surgery Volume: 33, Issue: 4, August 2010, pp. 183 - 187 Peter, Burghard

5. Bone Healing After Piezosurgery and its Influence on Clinical Applications Journal of Oral and Maxillofacial Surgery Volume: 65, Issue: 9, Supplement, September, 2007, pp. 39.e7-39.e8 Stübinger, Stefan

6. Assessment of Nerve Damage Using a Novel Ultrasonic Device for Bone Cutting Journal of Oral and Maxillofacial Surgery Volume: 66, Issue: 3, March, 2008, pp. 593-596 Schaeren, Stefan; Jaquiéry, Claude; Heberer, Michael; Tolnay, Markus; Vercellotti, Tomaso; et. al.

7. Use of piezosurgery in calvarial bone grafts and for release of the inferior alveolar nerve in sagittal split osteotomy: technical note British Journal of Oral & Maxillofacial Surgery Volume: 49, Issue: 8, December, 2011, pp. 668-669 Nusrath, M.A.; Postlethwaite, K.R.

8. Piezoosteotomy in orthognathic surgery versus conventional saw and chisel osteotomy Oral and Maxillofacial Surgery Volume: 12, Issue: 3, September 2008, pp. 139 - 147 Landes, C. A.; Stübinger, S.; Ballon, A.; Sader, R.

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Morphological Characteristics of the Velum following Furlow or Push-back Procedure

Satoh K.1,2, Takai E.1,2, Nohara K.2, Yamanishi T.2, Ishihama K.2, Sakai T.2, Kogo M.2

1Department of OMFS, Saiseikai Matsusaka General Hospital, Tsu city, Mie prefecture. 2Cleft Palate Team, Osaka University, Graduate School of Dentistry, Suita city, Osaka prefecture, Japan.

IntroductionFurlow procedure has been developed as an alternative to Push-back procedure

in the last decade, however, there seems to be little discussion about morphological characteristics of the velum in patients with repaired cleft palate following Furlow or conventional Push-back procedure.

The objectives of this study were to describe morphological characteristics of the velum in patients following Furlow or Push-back procedure at 4 and 8 years of age, and to make a morphological comparison of the velum between patients who main-tained the normal velopharyngeal function and patients who showed the deterioration of velopharyngeal function at 8 years of age.

Subjects and methodsSubjects were 50 patients with repaired cleft palate, and the complete velopha-

ryngeal closure was confirmed by perceptual and nasal endoscopic evaluations in all subjects at 4 years of age (Table 1). Twenty-four patients had Furlow procedure (F group) and 26 patients had Push-back procedure (P group). Seven subjects in the F group and 5 subjects in the P group showed the subsequent deterioration of velo-pharyngeal function.

In subjects with deteriorated velopharyngeal function, the velum could touch the posterior pharyngeal wall but nasality was found.

Lateral cephalograms were taken with subjects at rest and during blowing, and cephalometric landmarks were established in the nasopharyngeal area (Figure 1). Using these landmarks, linear and angular variables were measured. Measurement variables at rest were Velar length, Pharyneal depth, Adequate ratio, Velar thickness, and Velar position, and Measurement variables during blowing were Velar length, Contact length, Velar position, Velar flexion, and Velar ascent.


FIGURE 1. Cephalometric landmarks: ANS; anterior nasal spine, PMP; posterior maxillary point, point cre-ated by dropping a perpendicular from Ptm to the palatal plane, PPW; posterior pharyngeal wall at the intersection with the palatal plane(PPWa; at rest, PPWb; during blowing), V; tip of the velum(Va; at rest, Vb; during blowing), VPC; velar-pharyngeal contact point(VPCs; superior, VPCi; inferior), X; midpoint of the velum(Xa; at rest, Xb; during blowing). Measurement variables at rest: Velar length; distance from PMP to Va, Pharyngeal depth; distance from PMP to PPWa, Adequate ratio; ratio of velar length to pharyngeal depth, Velar thickness; distance from nasal to oral side of the velum at Xa, Velar position; velar angle to the Palatal plane(Xr-PMP-ANS). Measurement variables during blowing: Velar length; the total length of PMP-Xb and Xb-Vb, Contact length; length of velar-phayrngeal contact(distance from VPCs to VPCi), Velar position; velar angle to the palatal plane(Xb-PMP-ANS), Velar flexion; angle of the anterior and posterior velum(PMP-Xb-Vb), Velar ascent; paired difference of the velar position during blowing and at rest.


ResultsDifferences in measured variables between the Furlow and Push-back groups and

between subjects with normal VPF and deteriorated VPF at 8 years of age in each group were evaluated by t-test (Table 2-5).

At 4 years of age at rest (Table 2), Velar length and Velar thickness were si-gnificantly larger in the F group than in the P group, and there were no significant differences in the other variables. At 4 years of age during blowing (Table 2), Velar length, Contact length, and Velar flexion were significantly larger in the F group than in the P group. Velar ascent were significantly larger in the P group than in the F group, and there was no significant difference in Velar position.

At 8 years of age at rest (Table 3), as same as at 4 years of age, Velar length and Velar thickness were significantly larger in the F group than in the P group. At 8 years of age during blowing (Table 3), as same as at 4 years of age, Velar length and Contact length were significantly larger in the F group than in the P group. Velar ascent were significantly larger in the P group than in the F group,

At 8 years of age following Furlow procedure, Velar length and Contact length were significantly smaller in the subjects with deteriorated VPF than in the subjects with normal VPF. There were no significant differences in the other variables (Table 4). At 8 years of age following push-back procedure, Contact length, Velar flexion,


and Velar ascent were significantly smaller in the subjects with deteriorated VPF than in the subjects with the normal VPF. There were no significant differences in the other variables (Table 5).

ConclusionsFrom comparisons of measurement values at 4 years of age, the velar contact

against the posterior pharyngeal wall was widely achieved with long and thick velum in the Furlow group and was achieved with the higher elevation of velum in the Push-back group. This tendency stayed the same at 8 years of age. Contact length was significantly smaller in the subjects with deteriorated velopharyngeal function than in the subjects with normal velopharyngeal function in both groups. The smaller Contact length resulted from the smaller Velar length in the Furlow group and from the smaller Velar ascent in the Push-back group.

This study described the different patterns of velopharyngeal closure. Velar length and Velar thickness in the Furlow group, Velar ascent in the Push-back group were seemed to be crucial factors to maintain the normal velopharyngeal function. It se-emed reasonable to suppose that the secondary procedure to correct the deteriorated velopharyngeal function would be different when the primary procedure was different.

©2011 by MEDIMOND s.r.l. 57

Author Index

Abou-El Saad Tamer, 41Al Johar A, 25Ali Ahmed, 41Amer Ayman, 41Basiony Samia, 41Brusati R, MD, 5Chirila L, 29Cooper GM, 9Cray JJ Jr., 9Daratsianos N., 35Elbarbary Amir, 41Ishihama K., 51Jäger A., 35Jagielak A., 45Jagielak M., 45Kandasam R, 25Kogo M., 51Kozelj v, 1

Losee JE, 9Meazzini MC, DMD, MMSc, 5Michalski P., 45Mooney MP, 9Moursi AM, 9Neumann S., 19Nohara K., 51Rogus P., 45Sakai T., 51Satoh K., 51Siegel MI, 9Socha M., 45Subhan S, 25Takai E., 51Tarhon RC, 29Tavecchia MG, DD, 5Yamanishi T., 51

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