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YIJOM-4705; No of Pages 7

Int. J. Oral Maxillofac. Surg. 2019; xxx: xxx–xxx


https://doi.org/10.1016/j.ijom.2021.04.012, available online at https://www.sciencedirect.com

Clinical Paper
Cleft lip and palate

Non-surgical treatment of M. C. Meazzini1, N. Cohen1,


L. Autelitano2, J. Radojicic3
1
Smile House, Orthodontic Service, Regional

vertical excess of the premaxilla Centre for CLP, Department of Maxillofacial


Surgery, Santi Paolo and Carlo Hospital,
Milan, Italy; 2Smile House, Regional Centre
for CLP, Department of Maxillofacial Surgery,

in growing bilateral cleft lip and Santi Paolo and Carlo Hospital, Milan, Italy;
3
Department of Orthodontics, University
Clinical Centre of Ni, Ni, Serbia

palate patients
M.C. Meazzini, N. Cohen, L. Autelitano, J. Radojicic: Non-surgical treatment of
vertical excess of the premaxilla in growing bilateral cleft lip and palate patients. Int.
J. Oral Maxillofac. Surg. 2019; xxx: xxx–xxx. ã 2021 International Association of
Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.

Abstract. Patients with bilateral complete cleft lip and palate (BCLP) may present a
vertical excess of the premaxilla in childhood. This is a severe functional and
aesthetic problem, where bone grafting is more challenging. The aim of this study
was to describe a simple and reproducible non-surgical orthopaedic treatment for
vertical excess of the premaxilla in the deciduous/early mixed dentition phase in
BCLP patients. Six growing patients with complete BCLP with a severe vertical
excess of the premaxilla were included. An intrusion device associated with a
bonded rapid palatal expander was applied to intrude the premaxilla. Radiographic
and photographic records obtained before and at the end of the orthodontic
intrusion, at short- and long-term follow-up, were available. A flattening of the
occlusal plane was achieved in all patients. Normalization of the position of the
maxillary incisors and gingival display in relation to the upper lip was obtained, and
an improvement in anterior nasal spine position was also observed in all cases. The
novel technique described might be of assistance in treating BCLP children with Key words: Cleft Palate; Maxillary Bone; over-
vertical excess of the premaxilla during the deciduous/early mixed dentition phase. bite; orthodontics; osteotomy.
This simplified, easily reproducible method may allow the burden of care of this rare
but complex problem affecting BCLP patients to be reduced significantly. Accepted for publication 26 April 2021

Vertical excess of the premaxilla in bilat- Sagittal dental and skeletal maxillary patients with BCLP will require midface
eral cleft lip and palate (BCLP) patients is protrusion is physiological and desirable advancement at skeletal maturity3–5.
a major challenge for both orthodontists in BCLP during childhood and typically Therefore, it is generally not prudent to
and maxillofacial surgeons. Both vertical does not need any correction, because reduce sagittal protrusion during growth.
and sagittal excess are induced by an there is a progressive and spontaneous Vertical excess of the premaxilla in
overgrowth at the premaxillary-vomerine reduction of maxillary projection during BCLP, on the other hand, usually does
suture1,2. growth, and a significant number of not improve spontaneously with growth.

0901-5027/000001+07 ã 2021 International Association of Oral and Maxillofacial Surgeons. Published by Elsevier Inc. All rights reserved.

Please cite this article in press as: Meazzini MC, et al. Non-surgical treatment of vertical excess of the premaxilla in growing bilateral
cleft lip and palate patients, Int J Oral Maxillofac Surg (2021), https://doi.org/10.1016/j.ijom.2021.04.012
YIJOM-4705; No of Pages 7

2 Meazzini et al.

Table 1. Case series with skeletal and dental measurements, before the orthodontic intrusion (T0) and at the end of the orthodontic intrusion (T1).
Patient number 1 2 3 4 5 6 Average Paired t-test
Sex F F M M M F difference (T0 vs T1)
Age at T0 (years) 4.9 5.1 4 7 5.3 5.4 T1–T0 (mm)
T0 T1 T0 T1 T0 T1 T0 T1 T0 T1 T0 T1
Vertical excess of the 9 0 8 1 10 2 8 0 8 1 12 3 8.3 0.000002
premaxilla (mm)
Vertical intrusion of 62 54 60 55 61 59 63 61 55 52 60 52 4.7 0.01
incisors (mm)
Vertical intrusion ANS (mm) 41 37 38 36 39 36 38 37 34 31 33 27 3.2 0.006
Occlusal plane ( ) 28 5 20 10 20 10 17 10 20 9 27 11 12.8 0.003
Overjet (mm) 6 1 5 2 7 2 5 2 6 2 18 5 5.5 0.02
Overbite (mm) 11 0 8 1 10 2 8 0 8 1 12 3 8.7 0.00002
SNA change T0–T1 ( ) 1 2 0 2 0 5 1.7
Rotation of the premaxilla No No No No Yes No No No No No Yes No
Posterior crossbite Yes No Yes No Yes No Yes No Yes No Yes No
Type of surgery pre/post ESGAP ESGAP SBG SBG Waiting Waiting
intrusion (age, years) (2) (5.9) (5.4) (11.9) for SBG for SBG
Follow-up time (years) 13.6 13.2 5.3 7.2 3.3 1.1
Age at longest follow-up 19.8 19.1 10.1 15.3 8.1 7.2
*(T0) initial records; (T1) records at the end of the orthodontic intrusion; Vertical excess of the premaxilla(mm): distance between the incisal edge
of the upper incisor (U1) and an occlusal plane obtained between the molar and the canine occlusal point; Vertical Intrusion of incisors(mm):
distance between U1 and a horizontal reference line constructed down anteriorly 7 degrees to the Sella-Nasion plane(S-N); Vertical intrusion of
Anterior nasal Spine(mm): distance between Anterior nasal Spine (ANS) and a horizontal reference line constructed down anteriorly 7 degrees to
the Sella-Nasion plane(S-N); occlusal plane( ):angle between the occlusal plane and the Frankfurt plane; ESGAP: Early Secondary Gingivo
Alveolo Plasty; SBG: Secondary autogenous bone grafting.

It represents a severe aesthetic and psy- The aim of this study was to describe a as lip closure at the time of intrusion. The
chosocial problem. Bone grafting in these simple and reproducible non-surgical or- case series with skeletal and dental data is
patients is also more challenging. There- thopaedic approach for the treatment of reported in Table 1. Five patients were in
fore, correction of the vertical premaxil- vertical excess of the premaxilla in BCLP full deciduous dentition between 4 and 5
lary excess during childhood is highly patients in the deciduous/early mixed den- years of age before intrusion (T0). One
recommended. tition phase. A description of the various patient was 7 years old at T0.
In the 1960s and 1970s, premaxillary steps in the protocol is provided. All patients presented a severe aesthetic
osteotomies were often performed to in- disharmony at rest and during smile, with
trude and set back the premaxilla. How- a prominent and downward displaced pre-
ever, severe growth impairment induced Patients and methods maxilla, interposed between the upper and
by premaxillary osteotomy was repor- lower lip, with excessive premaxillary
Patients
ted3,6. Padwa et al. in 1999 suggested that gingival display (Fig. 1a–f). The overbite
premaxillary osteotomies performed be- Six growing patients with complete BCLP ranged from 8 mm to 12 mm and the
tween 6 and 8 years of age do not harm (three female and three male) presenting a overjet ranged from 5 mm to 18 mm at
subsequent growth8. Regrettably, the lon- severe vertical protrusion of the premax- T0 (Table 1). None of the patients treated
gest follow-up in that study only reached illa were included in this study. The study with the technique described in this report
age 11 years, which is too far from the end was approved by the internal ethics com- were excluded from the sample.
of growth to draw any conclusions regard- mittee of the hospital (N.0001191/2019). Photographic records were obtained im-
ing growth in BCLP9. The study followed the current national mediately before the orthodontic intrusion
The current evidence in the literature and international laws and regulations (T0) and at the end of the orthodontic
does not recommend a surgical approach governing the use of human subjects (Dec- intrusion (T1) and, when available, at
with premaxillary osteotomy in patients laration of Helsinki II). Informed consent the longest follow-up.
younger than 9–11 years of age2,11. Nev- was obtained from all of the participants
ertheless, there are no clear guidelines included in the study.
Premaxillary intrusion device
regarding the treatment of vertical pre- As only one of the patients had undergone
maxillary excess in the literature. Based primary surgery at the study centre, there An efficient, but relatively complex device
on the age of the patient and the severity was a high heterogeneity in surgical proto- was developed by Liou et al.14 and modi-
of the vertical premaxillary protrusion, cols. All patients had undergone cheilo- fied by Meazzini et al.12. These authors
Meazzini at al. proposed a protocol with plasty at a younger age. The patient used an intraoral tooth-borne appliance,
three different treatment approaches that treated at the study centre had been sub- which delivered heavy intermittent intru-
included orthopaedic, orthodontic, and jected to lip and soft palate closure at 6 sive forces to the premaxilla through two
surgical intrusion. The rigid orthopaedic months and an early secondary gingivoal- lateral rigid lever arms. The orthodontic
intrusion described for very young veoloplasty (ESGAP) together with hard techniques applied are typical intrusion
patients (deciduous/early mixed denti- palate closure at 20 months of age13. Two mechanics15. In the present study, instead
tion), though effective, raised various pro- patients had had cheiloplasty and soft palate of using a rigid system, which many ortho-
blems in terms of patient compliance and closure only. Three patients had already dontists find complex and which is not
orthodontic management12. received soft and hard palate closure as well easily accepted by the young patients, a

Please cite this article in press as: Meazzini MC, et al. Non-surgical treatment of vertical excess of the premaxilla in growing bilateral
cleft lip and palate patients, Int J Oral Maxillofac Surg (2021), https://doi.org/10.1016/j.ijom.2021.04.012
YIJOM-4705; No of Pages 7

Non-surgical treatment of vertical excess in BCLP 3

Fig. 1. Female BCLP patient with a severe vertical and horizontal excess of the premaxilla (case 6). Images a–f: initial lateral X-ray, extraoral and
intraoral frontal, lateral, and palatal photographs before the orthodontic intrusion (T0). Images g–l: lateral X-ray, extraoral and intraoral frontal,
lateral, and palatal photographs at the end of the orthodontic intrusion (T1).

bonded expander was applied. This appli- intrude the premaxilla. Intrusion was The TMA was subsequently substituted
ance exerts a double effect that includes an started directly with rectangular wires. A with pre-formed wires, in sequence,
expansion of the lesser segments that are titanium molybdenum steel (TMA) 0.017 straight 0.017  0.025 nickel–titanium
usually constricted in these patients and a  0.025 inch wire was initially applied, (NiTi) and then reverse curve 0.017 
solid anchorage for the orthodontic intru- with active bends added between the pre- 0.025 NiTi rectangular wires. In the five
sion wires. The orthodontic wires deliver maxilla and the lesser segments in cases of patients in deciduous dentition, an acrylic
low and continuous forces to gradually rotation and asymmetry of the premaxilla. cap with brackets was applied instead of

Please cite this article in press as: Meazzini MC, et al. Non-surgical treatment of vertical excess of the premaxilla in growing bilateral
cleft lip and palate patients, Int J Oral Maxillofac Surg (2021), https://doi.org/10.1016/j.ijom.2021.04.012
YIJOM-4705; No of Pages 7

4 Meazzini et al.

Fig. 2. Female BCLP patient with a severe vertical and horizontal excess of the premaxilla (case 6). Images a–d: photographs of the intrusion
device and intraoral frontal, lateral, and palatal photographs with the intrusion device in situ.

Fig. 3. Cephalometric landmarks and measurements.

brackets directly bonded on the teeth, in patients. Lateral cephalometric tracings Results
order to reduce the risk of debonding, at T0 and T1 were superimposed on
The rotation and asymmetry of the pre-
emergency visits, and total chair time the anterior cranial base, orienting on
maxilla observed at T0 in two patients
(Fig. 2). the sella–nasion line. For linear mea-
(cases 3 and 6) were normalized at T1.
Patients were checked after 2 months surements, a horizontal reference line
A flattening of the occlusal plane was
and every 3–4 months subsequently, for a constructed 7 down and anteriorly to
obtained in all patients (below 11 at T1).
total treatment time of 10–16 months. the sella–nasion plane (SN) was
Superimposition of the lateral cephalo-
depicted, as described previously
metric X-rays showed that the premaxil-
(Fig. 3)16. The differences between
Cephalometric analysis lary dentoalveolar height was significantly
T0 and T1 values were analysed with
decreased and within the normal range at
Lateral cephalometric radiographs tak- a paired t-test (P-value for significance
T1 (from 1 mm to 3 mm). One patient
en at T0 and at T1 were obtained for all set at 0.05).

Please cite this article in press as: Meazzini MC, et al. Non-surgical treatment of vertical excess of the premaxilla in growing bilateral
cleft lip and palate patients, Int J Oral Maxillofac Surg (2021), https://doi.org/10.1016/j.ijom.2021.04.012
YIJOM-4705; No of Pages 7

Non-surgical treatment of vertical excess in BCLP 5

Fig. 4. Female BCLP patient with a severe vertical excess of the premaxilla (case 2). Images a–c: initial lateral X-ray and intraoral frontal and
lateral photographs before the orthodontic intrusion (T0). Images d–f: lateral X-ray and intraoral frontal and lateral photographs at the end of the
orthodontic intrusion (T1). Images g–i: lateral X-ray and intraoral frontal and lateral photographs after 13 years of follow-up (at 19 years of age).

(case 5) showed an overcorrection of 1 mm bone grafting (SBG) immediately after the a progressive and spontaneous reduc-
above the occlusal plane between the molar orthodontic intrusion and two patients are tion of maxillary protrusion (SNA an-
and canine occlusal points. The anterior still waiting for SBG, using a night-time gle) during growth in BCLP patients, as
nasal spine was slightly lifted in all patients, acrylic appliance to aid retention of the demonstrated by Vargervik3 , Smahel4
by an average value of 3.2 mm, ranging newly positioned premaxilla during the and Trotman and Ross 5. Therefore, it
from 1 mm to 6 mm (P = 0.006). The first year post-intrusion. Only two out of is not prudent to correct a sagittal pro-
nasolabial morphology was mildly im- six patients were followed-up in the long- trusion during growth, unless there are
proved as a consequence of the raising of term post intrusion, until 19.8 years and severe psychological problems17. In
the anterior nasal spine. There was also 19.1 years of age, at the completion of those cases where the premaxillary sag-
extrusion of the posterior segments in all growth. These patients showed a stable ittal protrusion does not allow for a
patients, which is usually desirable because occlusal result, with no further need for bone graft, it is possible to orthodonti-
of the vertical maxillary defect, as noted in orthodontic intrusion or any surgical treat- cally protract the posterior segments
T0 and T1 lateral X-rays (Fig. 1a vs g, and ment (Fig. 4). The results are shown in instead of surgically retracting the
Fig. 4a vs d). Table 1. premaxilla18. In contrast, vertical dis-
The patient who had ESGAP at 2 years placement of the premaxilla needs to be
of age did not need further alveolar corrected during growth, since no spon-
Discussion
surgery after intrusion. One patient taneous improvement occurs2 .
underwent hard palate closure together Sagittal maxillary protrusion in BCLP The origin of premaxillary vertical
with ESGAP immediately after intrusion. during childhood typically does not outgrowth is not precisely known. Given
Two patients had a secondary autogenous need any correction, because there is the fact that we had a very heterogeneous

Please cite this article in press as: Meazzini MC, et al. Non-surgical treatment of vertical excess of the premaxilla in growing bilateral
cleft lip and palate patients, Int J Oral Maxillofac Surg (2021), https://doi.org/10.1016/j.ijom.2021.04.012
YIJOM-4705; No of Pages 7

6 Meazzini et al.

group from a surgical standpoint, we this observation. The significant decrease Acknowledgements. We wish to thank
have tried to find a common denomina- in the premaxillary dentoalveolar height Fabio Carta, specialized orthodontic tech-
tor. The only common factor between might be related to a sutural contraction/ nician, for the construction and donation
these six patients, even considering the osteolysis in the vomero-premaxillary su- of two of the appliances used in the
patients treated in the past with the pre- ture, combined with a slight vertical dis- sample.
vious intrusion technique12, was the con- placement of the vomero-nasal complex
stant interposition of the premaxilla and of the nasal bones and a mild lifting
between the upper and lower lip since of the anterior nasal spine.
early infancy. Poor lip repair did not The results of this study are based on a References
seem to be the main factor in this group, small series of heterogeneous patients,
[1] Pruzansky S. The growth of the premaxil-
although it cannot be excluded as a since this anomaly is fortunately rarely lary-vomerine complex in complete bilat-
cofactor. observed. Therefore, this study has no eral cleft lip and palate. Tandlaegebladet
Furthermore, premaxillary sagittal and presumption of giving any scientific ev- 1971;75:1157–69.
vertical protrusion are often combined, idence: it is the suggestion of an ex- [2] Heidbuchel KL, Kuijpers-Jagtman AM,
and a moderate amount of premaxillary tremely simple and repeatable non- Freihofer HP. An orthodontic and ceph-
orthopaedic retraction is required to allow surgical approach that may be an addi- alometric study on the results of the
lip competence (Fig. 1). tional tool for the orthodontist in order to combined surgical–orthodontic ap-
Although rigid orthopaedic intrusion, help the surgeon in the treatment of these proach of the protruded premaxilla in
as described previously, has been demon- rare, but psycho-socially handicapping bilateral clefts. J Craniomaxillofac Surg
strated to be very effective in correcting deformities. 1993;21:60–6.
the severe aesthetic and functional pro- In conclusion, a multidisciplinary [3] Vargervik K. Growth characteristics of the
blems without harming subsequent approach allows the cleft team to select premaxilla and orthodontic treatment prin-
growth12,14, this requires a technically the most appropriate method of treat- ciples in bilateral cleft lip and palate. Cleft
complicated appliance, increased chair ment, depending on the age of the patient Palate J 1983;20:289–302.
time, and high compliance, and there is and the severity of the disharmony and [4] Smahel Z. Craniofacial morphology in
a high risk of emergencies; therefore, the surgical treatment previously per- adults with bilateral complete cleft lip
there is a higher burden of orthodontic formed. The technique described in this and palate. Cleft Palate J 1984;21:
159–69.
care. The aim of this paper was to de- study might be of benefit in treating
[5] Trotman CA, Ross RB. Craniofacial growth
scribe a simple, approachable, and easily BCLP children with vertical excess of
in bilateral cleft lip and palate: ages six to
reproducible orthodontic method for the the premaxilla during the deciduous den-
adulthood. Cleft Palate Craniofac J
treatment of premaxillary vertical excess tition or the early mixed dentition phase. 1993;30:261–73.
in patients in the deciduous/early mixed It is a simple, reproducible method, with [6] Friede H, Pruzansky S. Longitudinal study
dentition phase, which the orthodontist in a low need for compliance. This simpli- of growth in bilateral cleft lip and palate,
the team may propose as an alternative to fied method may allow the burden of from infancy to adolescence. Plast Reconstr
a surgical premaxillary intrusion. The care of this rare but complex and Surg 1972;49:392–403.
authors are not aware of any previous psychologically distressing problem af- [8] Padwa BL, Sonis A, Bagheri S, Mulliken
publications on the same simple ap- fecting BCLP patients to be reduced JB. Children with repaired bilateral cleft
proach. The method was well tolerated significantly. lip/palate: effect of age at premaxillary
by all patients during early childhood. osteotomy on facial growth. Plast Reconstr
The use of a bonded expander and bonded Surg 1999;104:1261–9.
anterior acrylic cap reduced the initial Funding [9] Semb G. A study of facial growth in patients
total visits for appliance delivery from with bilateral cleft lip and palate treated by
three to two and reduced the risk of This research did not receive any specific the Oslo CLP Team. Cleft Palate Craniofac J
debonding, emergency visits, and total grant from funding agencies in the public, 1991;28:22–39. http://dx.doi.org/10.1597/
chair time. commercial, or not-for-profit sectors. 1545-1569_1991_028_0022_asofgi_2.3.
The technique of non-surgical intru- co_2. discussion 46–48.
sion with orthodontic wires during [11] Geraedts CT, Borstlap WA, Groenewoud JM,
Borstlap-Engels VM, Stoelinga PJ. Long-
growth described in this study utilizes Competing interests
term evaluation of bilateral cleft lip and
low forces that allow the relationship
The authors declare no potential conflicts palate patients after early secondary closure
between the anterior teeth and upper
of interest with respect to the research, and premaxilla repositioning. Int J Oral
lip to be improved, by both contracting
authorship, and publication of this article. Maxillofac Surg 2007;36:788–96. http://dx.
the overgrowth that has occurred doi.org/10.1016/j.ijom.2007.04.010. Erra-
at the vomero-premaxillary suture and tum in: Int J Oral Maxillofac Surg 2008:
controlling the vertical premaxillary 37: 202.
growth, while allowing a posterior max- Ethical approval
[12] Meazzini M, Lematti L, Mazzoleni F, Rab-
illary growth, often deficient in BCLP The study was approved by the internal biosi D, Bozzetti A, Brusati R. Vertical ex-
patients. The biological effect obtained ethics committee of the hospital cess of the premaxilla in bilateral cleft lip and
seems to be, in part, an actual bony (N.0001191/2019). palate patients: a protocol for treatment. J
contraction19. Craniofac Surg 2010;21:499–502. http://dx.
This approach normalized the position of doi.org/10.1097/SCS.0b013e3181cffb4d.
the maxillary incisors in relation to the [13] Brusati R, Mannucci N. The early gingi-
Patient consent voalveoloplasty. Preliminary results. Scand
occlusal plane and to the upper lip, as shown
in Figures 1 and 4 and in Table 1. Lateral Informed consent was obtained from all of J Plast Reconstr Surg Hand Surg
cephalometric superimposition confirmed the participants included in the study. 1992;26:65–70.

Please cite this article in press as: Meazzini MC, et al. Non-surgical treatment of vertical excess of the premaxilla in growing bilateral
cleft lip and palate patients, Int J Oral Maxillofac Surg (2021), https://doi.org/10.1016/j.ijom.2021.04.012
YIJOM-4705; No of Pages 7

Non-surgical treatment of vertical excess in BCLP 7

[14] Liou EJW, Chen PKT, Huang S, Chen YR. [17] Hayward JR. Management of the premax- Address:
Orthopedic intrusion of premaxilla with illa in bilateral clefts. J Oral Maxillofac Smile House
distraction devices before alveolar bone Surg 1983;41:518–24. Orthodontic Service
grafting in patients with bilateral cleft [18] Meazzini MC, Cohen N, Battista VMA, Regional Centre for CLP
lip and palate. Plast Reconstr Surg Incorvati C, Biglioli F, Autelitano L. Ortho- Department of Maxillofacial Surgery
2004;113:818–26. dontic-pre grafting closure of large alveolar Santi Paolo and Carlo Hospital
[15] Burstone CR. Deep overbite correction bony and soft tissue gaps: a novel non- Via di Rudinı̀ 8
by intrusion. Am J Orthod 1977;72: surgical protraction of the lesser segments
20142
Milan
1–22. in growing cleft lip and palate patients.
Italy
[16] Meazzini MC, Basile V, Mazzoleni F, Cleft Palate J )2021;(Apr 13). http://dx.
Tel: +39 3477058952
Bozzetti A, Brusati R. Long- term fol- doi.org/10.1177/10556656211007697. ortodonziasmilehouse@asst-santipaolocarlo.
low-up of large maxillary advancement Epub ahead of print. it,
with distraction osteogenesis in growing [19] Castello JR, Olaso AS, Chao JJ, McCarthy JG, noah.cohen@asst-santipaolocarlo.it,
and non-growing cleft lip and palate Molina F. Craniofacial shortening by contrac- luca.autelitano@asst-santipaolocarlo.it,
patients. J Plast Reconstr Aesthet Surg tion osteogenesis: an experimental model. dr.julija.radojicic@gmail.com
2015;68:79–86. Plast Reconstr Surg 2000;105:617–25.

Please cite this article in press as: Meazzini MC, et al. Non-surgical treatment of vertical excess of the premaxilla in growing bilateral
cleft lip and palate patients, Int J Oral Maxillofac Surg (2021), https://doi.org/10.1016/j.ijom.2021.04.012

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