Repair of complete bilateral cleft lip with severely protruding premaxilla performing a premaxillary setback and vomerine ostectomy in one stage surgery
Abstract
Background: The authors present a technique for selected cases of CBCL. The primary repair of the CBCL with a severely protruding premaxilla in one stage surgery is very difficult, essentially because a good muscular apposition is difficult, forcing synchronously to do a premaxillary setback to facilitate subsequent bilateral lip repair and, thus, achieving satisfactory results. We achieve this by a reductive ostectomy on the vomero- premaxillary suture. Material and Methods: 4 patients with CBCL and severely protruding premaxilla underwent premaxillary setback by vomerine ostectomy at the same time of lip repair in the past 24 months. The extent of premaxillary setback varied between 9 and 16 mm. The required amount of bone was removed anterior to the vomero-premaxillary suture. The authors did an additional simultaneous gingivoperiosteoplasty in all patients, achieving an enough stability of the premaxilla in its new position, to be able to close the alveolar gap bilaterally. The authors have examined the position of premaxilla and dental arch between 6 and 24 months. We did not do the primary nose correction, because this increased the risk of impairment of the already compromised vascularity of the philtrum and premaxilla. Results: The follow-up period ranged between 6 and 24 months. None of the patients had any major complication. During follow-up, the premaxilla was minimally mobile. We achieved a good lip repair in all cases: adequate muscle repair, symmetry of the lip, prolabium and Cupid’s bow, as well as good scars. Cloncusions: To our knowledge, there are few reports of one stage surgery with vomerine ostectomy to repair CBCL with severely protruding premaxilla. Doing this vomerine ostectomy, we don’t know how it will affect the subsequent growth of the premaxila and restrict the natural maxillary growth. Applying this alternative treatment for children with CBCL and protruded premaxilla without any preoperative orthopedic, we can successfully perform, in a single-stage surgery, a good primary lip repair at our center. Further confirmations of this surgery with follow up and anthropometric studies of these patients during childhood and adolescence are required.
Full text
Med O al Pa ol O al Ci Bucal. 2015 Jul 1;20 (4):e500-7. Repai o comple e bila e al cle lip
e500
Jou nal sec ion: O al Su ge y
Publica ion Types: Re iew
Repai o comple e bila e al cle lip wi h se e ely p o uding p emaxilla
pe o ming a p emaxilla y se back and ome ine os ec omy in one s age su ge y
Nabil Fakih-Gomez, Ma a Sanchez-Sanchez, Fe nando Iglesias-Ma in, Albe o Ga cia-Pe la-Ga cia,
Rodol o Belmon e-Ca o, Luis-Miguel Gonzalez-Pe ez
Depa men o O al and Maxillo acial Su ge y, Vi gen del Rocio Uni e si y Hospi al, Se ille, Spain
Co espondence:
Depa men o Maxillo acial Su ge y
Vi gen del Rocio Uni e si y Hospi al
Se ille, Spain
d nabil [email protected]
Recei ed: 25/12/2014
Accep ed: 26/02/2015
Abs ac
Backg ound: The au ho s p esen a echnique o selec ed cases o CBCL. The p ima y epai o he CBCL
wi h a se e ely p o uding p emaxilla in one s age su ge y is e y di icul , essen ially because a good muscula
apposi ion is di icul , o cing synch onously o do a p emaxilla y se back o acili a e subsequen bila e al
lip epai and, hus, achie ing sa is ac o y esul s. We achie e his by a educ i e os ec omy on he ome o-
p emaxilla y su u e.
Ma e ial and Me hods: 4 pa ien s wi h CBCL and se e ely p o uding p emaxilla unde wen p emaxilla y se back
by ome ine os ec omy a he same ime o lip epai in he pas 24 mon hs. The ex en o p emaxilla y se back
a ied be ween 9 and 16 mm. The equi ed amoun o bone was emo ed an e io o he ome o-p emaxilla y
su u e. The au ho s did an addi ional simul aneous gingi ope ios eoplas y in all pa ien s, achie ing an enough
s abili y o he p emaxilla in i s new posi ion, o be able o close he al eola gap bila e ally.
The au ho s ha e examined he posi ion o p emaxilla and den al a ch be ween 6 and 24 mon hs. We did no do he
p ima y nose co ec ion, because his inc eased he isk o impai men o he al eady comp omised ascula i y o
he phil um and p emaxilla.
Resul s: The ollow-up pe iod anged be ween 6 and 24 mon hs. None o he pa ien s had any majo complica ion.
Du ing ollow-up, he p emaxilla was minimally mobile. We achie ed a good lip epai in all cases: adequa e
muscle epai , symme y o he lip, p olabium and Cupid’s bow, as well as good sca s.
Cloncusions: To ou knowledge, he e a e ew epo s o one s age su ge y wi h ome ine os ec omy o epai
CBCL wi h se e ely p o uding p emaxilla. Doing his ome ine os ec omy, we don’ know how i will a ec he
subsequen g ow h o he p emaxila and es ic he na u al maxilla y g ow h. Applying his al e na i e ea men
o child en wi h CBCL and p o uded p emaxilla wi hou any p eope a i e o hopedic, we can success ully
Fakih-Gomez N, Sanchez-Sanchez M, Iglesias-Ma in F, Ga cia-Pe la-Ga -
cia A, Belmon e-Ca o R, Gonzalez-Pe ez LM. Repai o comple e bila e al
cle lip wi h se e ely p o uding p emaxilla pe o ming a p emaxilla y
se back and ome ine os ec omy in one s age su ge y. Med O al Pa ol
O al Ci Bucal. 2015 Jul 1;20 (4):e500-7.
h p://www.medicinao al.com/medo al ee01/ 20i4/medo al 20i4p500.pd
A icle Numbe : 20568 h p://www.medicinao al.com/
© Medicina O al S. L. C.I.F. B 96689336 - pISSN 1698-4447 - eISSN: 1698-6946
eMail: [email p o ec ed]
Indexed in:
Science Ci a ion Index Expanded
Jou nal Ci a ion Repo s
Index Medicus, MEDLINE, PubMed
Scopus, Embase and Emca e
Indice Médico Español
doi:10.4317/medo al.20568
h p://dx.doi.o g/doi:10.4317/medo al.20568
Med O al Pa ol O al Ci Bucal. 2015 Jul 1;20 (4):e500-7. Repai o comple e bila e al cle lip
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pe o m, in a single-s age su ge y, a good p ima y lip epai a ou cen e . Fu he con i ma ions o his su ge y wi h
ollow up and an h opome ic s udies o hese pa ien s du ing childhood and adolescence a e equi ed.
Key Wo ds: P o uding p emaxilla, bila e al cle lip, ome ine os ec omy, one s age su ge y, Milla d II echnique.
In oduc ion
P ominen p emaxilla is a cha ac e is ic o in an s wi h
comple e bila e al cle lip. I is ela ed o he a achmen
o p emaxilla o he ome and he nasal sep um ia he
sep o-p emaxilla y ligamen in he absence o la e al
s ains. The p o uding wis ed p emaxilla adds he
p oblems o su gical managemen o bila e al comple e
cle o lip pa ien s (CBCL). P emaxilla is un es ained
by ei he o he maxilla y al eoli and only a ached o
nasal sep um by sep omaxilla y ligamen . In no mal
child en, he ca ilaginous sep um mus slide o wa d in
ela ion o he p emaxilla y egion owing o he es ain
on he p emaxilla by he lip and la e al maxilla y
segmen s. In bila e al cle , he p emaxilla is ca ied
o wa d a he same a e as ha o he g owing sep um
o which i is i mly held. The p emaxilla has only one
es aining connec ion, he ome . This es ain is
ealized as a ension be ween hese bones bo ne by he
ome o-p emaxilla y su u e, hus c ea ing he condi ion
o bone o ma ion (1,2).
Va ious appliances o p emaxilla y se back such
as ex ao al head cap, elas ic s aps, apes, La ham
appliance and Bu s on pla e ha e been success ul a e
a ious su ge ies and long ime ea men (3-6). The
li e a u e has men ioned abou he lip adhesion as he
ea men o p o uding p emaxilla; i is no always
success ul o mold p emaxilla wi hou muscle epai .
In addi ion, i adds o one mo e su ge y and also adds
sca s o he lip making he nex epai mo e di icul .
The p oblems a e compounded in BCLP by p o uding
p emaxilla (7).
So condi ioned on amilia demand and lack o
p eope a i e o hopedic ea men o ce us o use one
s age su ge y o ea hem. This p ima y epai o
CBCL wi h a se e ely p o uding p emaxilla in one
s age su ge y is e y di icul , essen ially because a good
muscula apposi ion is di icul , o cing synch onously
o do a p emaxilla y se back o acili a e subsequen
bila e al lip epai .
While ollowing he p incipals desc ibed by Milla d in
1977 (8,9), we added he ome ine os ec omy echnique o
sui he local ci cums ances. So by his educ i e os ec omy
on he ome o-p emaxilla y, we can pe o m i in one
s age su ge y. A he same ime as p emaxilla y se back,
he blood supply o he p emaxilla is impai ed, wi h he
isk o p emaxilla y nec osis. The e a e ew epo s o one
s age su ge y wi h ome ine os ec omy o epai CBCL
wi h se e ely p o uding p emaxilla. (10-13). To ou
knowledge, his is he i s epo using bo h combina ions
ome os ec omy and Milla d II echnique.
Pa ien and Me hods
- Su gical Technique:
Unde gene al anes hesia, supine posi ion and minimal
ex ension o he pa ien ’s neck, he p ocedu e begins
wi h ome ine os ec omy. The equi ed o bone was
emo ed an e io o he ome o-p emaxilla y su u e.
We did addi ional simul aneous gingi ope ios eoplas y,
achie ing an enough s abili y o he p emaxilla in
i s new posi ion, o be able o close he al eola gap
bila e ally, ollowed by su u ing he o biculus muscle
hen using Milla d II echnique o epai he lip. We did
no do p ima y nose co ec ion, because his inc eased
he isk o impai he al eady comp omised ascula i y
o he phil um and p emaxilla. The ala ca ilages a e
eadjus ed h ough im incisions.
- Clinical Cases:
Be ween Decembe 2011 and Ap il 2013, 4 pa ien s wi h
comple e bila e al cle lip wi h p o uding p emaxila
unde wen p emaxilla y se back by ome ine os ec omy
a he same ime o lip epai . All pa ien s had pala ine
cle associa ed who hey unde wen a second ime
su ge y o epai he pala ine cle . Pa ien ages anged
be ween 7 mon hs and 4 yea s 5 mon hs wi h a mean age
o 19 mon hs. The e we e 2 males and 2 emales. None
o he child en had unde gone any p e ious o hodon ic
ea men .
- Case 1: A 4-yea -old male pa ien wi h no amily
his o y no isk ac o s, no associa ed anomalies
and synd ome, ha a e being ea ed se e al imes
p e iously in o he cen e s, e e ed o ou cen e o
esol e he pala e cle and he p ojec ed p e- ea ed
p emaxilla wi h he closu e o he cle lip. Only in his
pa ien we epai ed a he same ime he pala e cle wi h
he ome ine os ec omy ollowed by a new lip epai 4
mon hs la e . We unde wen p emaxilla y se back a he
ime o pala e epai because he had unde wen o he lip
su ge ies in o he clinics wi hou ea ing he p ojec ed
p emaxila (Fig. 1).
- Case 2: A male pa ien o 6 mon hs wi h no amily
his o y no isk ac o s, no associa ed anomalies and
synd ome (Fig. 2).
- Case 3: A emale pa ien o 9 mon hs wi h amilia
his o y (pa e nal uncle) wi h cle lip, wi h mo he
his o y o d ug in ake and no associa ed anomalies no
synd ome (Fig. 3).
Med O al Pa ol O al Ci Bucal. 2015 Jul 1;20 (4):e500-7. Repai o comple e bila e al cle lip
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- Case 4: A emale pa ien o 7 mon hs wi hou amily
his o y o isk ac o s and bila e al hypoacusis (Fig. 4).
The h ee o he cases unde wen he su gical echnique
desc ibed be o e. Case 2 had cle pala e epai 13
mon hs la e . The o he wo emaining cases a e
awai ing su gical in e en ion o pala e cle .
The ex en o p emaxilla y se back a ied be ween 9
and 16 mm. The equi ed amoun o bone was emo ed
an e io o he ome o-p emaxilla y su u e. Only in case
1, being olde , elas ic ac ion is pe o med o g adually
e ac he p emaxilla. To achie ing an enough s abili y
o he p emaxilla in i s new posi ion, and o be able o
close he al eola gap bila e ally, we did an addi ional
simul aneous gingi ope ios eoplas y.
Resul s
The ollow-up pe iod anged be ween 6 and 24 mon hs.
We examined he den al a ch and posi ion o he
p emaxilla clinically and documen ed pho og aphically
in all e isions. None o he pa ien s had any majo
complica ion, such as loss o he p emaxilla o any
ischemic episode o ascula comp omise o he
p emaxilla o skin dehiscence o he lip. Du ing ollow-
up, i was no ed ha he p emaxilla was minimally
Fig. 1. (case 1): A 4-yea -old male wi h CBCL wi h p o uding p emaxilla and pala e cle . (A) P eope a i e an e io and
la e al iew. (B) CT scan showing he ex eme p o usion o he p emaxilla wi h a e ical excess ; No ice he leng h o he
ome ine bone needed o be excised on CT scan. (C) Occlusal in a-o al iew, showing he un es ained an e io nasal sep al
and ome o-p emaxilla y su u e a e wedge os ec omy o he ome . (D) Pos ope a i e esul s on la e al iew (18 mon hs
pos ope a i ely).
AB
D
C
Med O al Pa ol O al Ci Bucal. 2015 Jul 1;20 (4):e500-7. Repai o comple e bila e al cle lip
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mobile in all pa ien s. The ex en o p emaxilla y se back
a ied be ween 9 and 16 mm. We achie ed a good lip
epai in all cases wi h adequa e muscle epai , wi h an
excellen symme y o he lip, p olabium and Cupid’s
bow as well as good sca s. No is ulas appea ances we e
documen ed. Re e ing o case 1, speech e alua ion
showed imp o emen wi h ega d o in elligibili y,
hype nasali y, and a icula ion e o . Howe e , as
expec ed, i was no no mal because o ob ious ac o s,
such as la e pala e epai and habi ual pa e ns. In he
o he cases, a e s ill young o eco d in elligibili y and
wo o hem is s ill awai ing pala e cle epai .
Discussion
I is bes o ini ia e ea men as ea ly as possible
because su gical sca s a e less isible ollowing su ge y
in in an s (14). In addi ion, he bone and ca ilage
issues a e so and moldable in in an s acila ing mo e
he su gical co ec ion, and no mal o al unc ion can be
es ablished once he lip s uc u es ha e been epai ed
ana omically. Psychological p oblems o he baby and
amily a e also minimized (15).
P o usion o he p emaxilla in CBCL can be seen as
ea ly as 10 weeks o ges a ion (1). Un es ained an e io
nasal sep al and ome o-p emaxilla y su u e g ow h,
combined wi h lack o bony and so issue con inui y,
and dis up ion o balance be ween he ci cumo al
muscula u e and he ongue a e hough o p oduce he
classic bila e al p emaxilla y de o mi y (16,17). This
p emaxilla y ela ionship may esul in signi ican
unc ional p oblems such as absence o p ope an e io
occlusion, la e al mobili y o he p emaxilla y segmen
and labial o pala al o onasal is ulae wi h consequen
p oblems in speech and o al hygiene. P ominence o
e ical o e de elopmen o he p emaxilla also may
esul in signi ican psychological ha m du ing a child’s
Fig. 2. (case 2): A 6 mon hs male wi h CBCL wi h p o uding p emaxilla and pala e cle . (A) P eope a i e an e io iew. (B) Oc-
clusal in a-o al iew, showing ome o-p emaxilla y su u e and he si e o he wedge os ec omy o he ome wi h a bone cu e .
(C) Occlusal in a-o al iew, showing he gap a e he wi hd awal o he wedge os eo omized ome . (D) Lip epai and immedia e
pos ope a i e esul s on an e io iew a e su u ing he o bicula is muscle and he gingi ope ios eoplas y.
AB
D
C
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o ma i e yea s. Bila e al cle lip epai wi h p o uding
p emaxila emains one o he mos challenging p oblems
o su geons. To ou knowledge, he e a e ew epo s
o one s age su ge y wi h ome ine os ec omy o epai
CBCL wi h se e ely p o uding p emaxilla (10-13).
Co ec ion o he displaced p emaxilla by o hodon ic
ea men alone is impossible. On he o he hand,
su gical eposi ioning is echnically demanding and
may in e e e wi h he blood supply o he p emaxilla.
Two-s age p ocedu e was ad ised by some au ho s (18),
bu os eo omy o he p emaxilla, in combina ion wi h
he co ec ion o he lip cle , has been also epo edly
success ul in selec ed cases, especially en olde
pa ien s. This p ocedu e enables closu e o la ge is ulae
(inc easing he chances o success o lip econs uc ion
wi hou ension in he lip skin), acili a es o e je
and o e bi e co ec ion, and may ha e psychosocial
ad an ages o he child and his amily which demand
one ime su ge y and whe e we can no o e hem
nasoal eola moldings (NAM) o p e ious o hodon ic
ea men . Al hough ea ly esul s wi h p eope a i e
NAM ha e good esul , objec i e analysis o long- e m
ou come has no ye been epo ed (19,20).
Mul iple s udies compa ed un epai ed adul cle lip and
cle pala e maxilla y mo phology o ha in adul s wi h
epai ed cle s (21-26). The esul s a e well- documen ed:
adul s wi h un epai ed cle lips o pala es ha e maxillas
wi h a no mal o sligh ly p ogna hic cephalome ic
posi ion an e io -pos e io ly, and a no mal o widened
maxilla y ans e se wid h. Based on hese s udies, i is
e iden ha he acial cha ac e is ics o an un epai ed
adul cle lip and pala e pa ien a e unique: a no mal
Fig. 3. (case 3): A 9 mon hs emale wi h CBCL wi h p o uding p emaxilla and pala e cle . (A) P eope a i e an e io iew.
(B) Occlusal in a-o al iew, showing he wedged os eo omized ome . (C) Lip epai and immedia e pos ope a i e esul s on
an e io iew. (D) Pos ope a i e esul s on an e io iew (1 mon h pos ope a i ely).
AB
CD
Med O al Pa ol O al Ci Bucal. 2015 Jul 1;20 (4):e500-7. Repai o comple e bila e al cle lip
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o sligh ly p o usi e uppe jaw, p o uded maxilla y
an e io den i ion, and a no mal o sligh ly hypoplas ic
mandibula ela ionship. In addi ion, he cle lip and
cle pala e dimensions a e ha much g ea e , because
he cle g ows p opo iona ely wi h he su ounding
ana omy. Padwa e al. (27) sugges ed ha a p o usi e
p emaxilla could be su gically eposi ioned a e 6 o 8
yea s wi hou dele e ious e ec s on mid acial g ow h.
F eiho e e al. (28) also no ed ha by his age (8 o 13
yea s), he de elopmen o he maxilla is a ad anced
and he g ow h dis u bance a his age by p emaxilla y
se back has only a ela i ely es ic ed nega i e
in luence. Howe e , F iede and P uzansky (29) no ed
ha he majo i y o pa ien s wi h bila e al comple e cle
lip and pala e who ha e unde gone p emaxilla y se back
ha e conca e aces because o maxilla y e usion.
Al hough his is a eal side ec , i is essen ial o b ing
he p emaxilla y se back in alignmen wi h he la e al
segmen so ha subsequen al eola bone g a ing and
o hogna hic p ocedu es can be pe o med. Mid acial
g ow h is easily documen ed by la e al cephalome y
and by analysis o plas e den al cas s. Many den al
specialis s emphasize ha he deg ee o maxilla y
e usion is a e lec ion o he su gical p o ocol o he
ope a i e echnique. Some specialis s belie e ha
closu e o he seconda y pala e should be delayed un il
childhood, despi e s udies o show ha his s a egy can
esul in poo speech. I has been clea ly documen ed
ha adul s wi h un epai ed cle lip/pala e exhibi
no mal acial g ow h (30,31). Repai o bila e al cle
lip/pala e inhibi s maxilla y g ow h, ye he deba e
con inues whe he labial o pala al closu es, o bo h,
Fig. 4. (case 4): A 7 mon hs emale wi h CBCL wi h p o uding p emaxilla and pala e cle . (A) P eope a i e an e-
io iew. (B) Occlusal in a-o al iew, showing ome o-p emaxilla y su u e and he si e o he wedge os ec omy
o he ome . (C) Occlusal in a-o al iew, showing he wedged os eo omized ome . (D) Pos ope a i e esul s on
an e io iew (1 mon h pos ope a i ely).
AB
D
C
Med O al Pa ol O al Ci Bucal. 2015 Jul 1;20 (4):e500-7. Repai o comple e bila e al cle lip
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a e esponsible. Also Be kowi z e al. (32) has epo ed
ha ea ly p eope a i e o hopedics leading buccal bi e
o he p emaxilla wi h conca e acial p o iles wo sen
wi h ime.
Wi h limi ed in o ma ion in he li e a u e abou
ollow-up s udies o CBCL wi hou any p eope a i e
o hopedics, his s udy showed he ou come posi ion o
he p emaxilla and he lip epai esul s. In all ou ou
pa ien s, applying his al e na i e ea men o child en
wi h CBCL and p o uded p emaxilla wi hou any
p eope a i e o hopedic, we can success ully pe o m,
in a single-s age su ge y, a good p ima y lip epai a
ou cen e . Single-s age p ima y ope a ion is ela i ely
easie o pe o m, as he e a e no sca ed issues. The
ome ine os ec omy acili a e subsequen bila e al lip
epai and adequa e muscula epai , which is i al o
labial unc ion. Simul aneous gingi ope ios eoplas y on
bo h sides p o ides be e s abili y and alignmen o he
a ches in addi ion o he bila e al closu e o he al eola
gap. Using Millad II echnique achie es an excellen
symme y o he lip, p olabium and Cupid’s bow as
well as good sca s. No complica ions we e documen ed.
Doing his ome ine os ec omy, we don’ know how i
will a ec he subsequen g ow h o he p emaxila and
es ic he na u al maxilla y g ow h, bu as desc ibed
be o e, i is essen ial o b ing he p emaxilla y
se back in alignmen wi h he la e al segmen so ha
subsequen al eola bone g a ing and o hogna hic
p ocedu es i equi ed can be pe o med. In addi ion
many adolescen s wi h epai ed bila e al cle lip need
maxilla y ad ancemen o imp o e p ojec ion o he
nasal ip, o p o ude he uppe lip, and o a ain no mal
sagi al skele al ha mony. Howe e , we a e suppo e s o
pe o ming ea ly su ge y, assuming he sequels o be
co ec ed la e bu a o ing he be e de elopmen o
language and aes he ics du ing childhood (14,15,29).
Conclusions
Applying his al e na i e ea men o child en
wi h CBCL and p o uded p emaxilla wi hou any
p eope a i e o hopedic, we can success ully pe o m,
in a single-s age su ge y, a good p ima y lip epai a
ou cen e . Fu he con i ma ions o his su ge y wi h
ollow up and an h opome ic s udies o hese pa ien s
du ing childhood and adolescence a e equi ed.
Re e ences
1. La ham RA. De elopmen and s uc u e o he p emaxilla y
de o mi y in bila e al cle lip and pala e. B J Plas Su g. 1973;26:1-
11.
2. Delai e J, P ecious D. In luence o he nasal sep um on maxillonasal
g ow h in pa ien s wi h congeni al labiomaxilla y cle . Cle Pala e
J. 1986;23:270-7.
3. Bu s on WR. The ea ly o hodon ic ea men o cle pala e
condi ions. Den P ac . 1958;9:41.
4. Geo giade NG, La ham RA. Maxilla y a ch alignmen in he
bila e al cle lip and pala e in an , using pinned coaxial sc ew
appliance. Plas Recons Su g. 1975;56:52-60.
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