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\begin{document}

\title{AN INNOVATIVE APPROACH IN THE MANAGEMENT OF ALVEOLAR CLEFTS WITH BONE
GRAFT HARVEST FROM MAXILLARY TUBEROSITY AND MANDIBULAR WISDOM MOLAR
ODONTECTOMY SITES- A CASE REPORT}



\author[1]{Symon Guthua}%
\author[1]{Peter Ng'ang'a}%
\author[1]{Krishan Sarna}%
\author[1]{Martin Kamau}%
\affil[1]{University of Nairobi}%


\vspace{-1em}



  
  \date{May 27, 2024}


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\selectlanguage{english}
\begin{abstract}
Alveolar bone grafting is a complex procedure utilized in alveolar cleft
repair, however, the ideal site of bone graft material remains highly
debated. In this paper, we describe the management of a 14 year old
female with bilateral alveolar clefts using alternative intra-oral donor
sites for bone graft harvest.%
\end{abstract}%



\sloppy


\textbf{AN INNOVATIVE APPROACH IN THE MANAGEMENT OF ALVEOLAR CLEFTS WITH
BONE GRAFT HARVEST FROM MAXILLARY TUBEROSITY AND MANDIBULAR WISDOM MOLAR
ODONTECTOMY SITES- A CASE REPORT}

Symon Guthua \emph{BDS, M.MED.Sc, D.O.M.S \textsuperscript{1}}

Peter M. Ng'ang'a \emph{BDS, MSD, PhD \textsuperscript{2}}

Krishan Sarna \emph{BSc Anatomy\textsuperscript{3}}

Martin Kamau \emph{BDS, MDS-OMFS \textsuperscript{1,3}}

\begin{enumerate}
\tightlist
\item
  Department of Oral and Maxillofacial Surgery, Oral Pathology and Oral
  Medicine, Division of Oral and Maxillofacial Surgery, University of
  Nairobi, Kenya
\item
  Retired professor at the Department of Pediatric Dentistry and
  Orthodontics, University of Nairobi, Kenya (currently in private
  practice of orthodontics - Nairobi)
\item
  Department of Human Anatomy, University of Nairobi, Kenya
\end{enumerate}

\textbf{Address for correspondence}

Krishan Sarna

Department of Human Anatomy,

University of Nairobi, Kenya

krishnasarna@gmail.com

+254790536570

\textbf{CONSENT:} Written informed consent was obtained from the
patient's parents to publish this report in accordance with the
journal's patient consent policy

\textbf{ABSTRACT}

Alveolar bone grafting is a complex procedure utilized in alveolar cleft
repair, however, the ideal site of bone graft material remains highly
debated. In this paper, we describe the management of a 14 year old
female with bilateral alveolar clefts using alternative intra-oral donor
sites for bone graft harvest.

\textbf{KEY CLINICAL MESSAGE}

The maxillary tuberosity and odontectomy sites of wisdom molars are
overlooked donor sites which may be used in alveolar cleft
reconstruction. A pre-operative assessment is recommended to evaluate
their viability as potential bone harvest sites.

Key words: Alveolar bone grafting, Alveolar Clefts, Maxillary
tuberosity, Wisdom molars, Odontectomy sites



\textbf{INTRODUCTION}

Alveolar clefts are one of the principle sequelae of cleft lip and
palate and are defined as a discontinuity of the dental arch, having an
incidence of 0.18 to 2.50 per 1000 births.\textsuperscript{1,2} The
region of the alveolus most commonly affected lies between the lateral
incisor and canine teeth and result in the appearance of a `floating'
premaxillary segment if present bilaterally. Alveolar clefts occur in
response to divergence from normal embryological development during
frontonasal and maxillary prominence growth, contact and
fusion.\textsuperscript{3} A clinical picture comprising of facial
growth disturbances, nasal reflux, chronic periodontal inflammation,
speech disturbances and an unsightly aesthetic appearance is frequently
observed.\textsuperscript{4,5} With recent advancements and innovative
techniques in reconstructive surgery aimed at correcting such defects,
the quality of life of such patients can be improved significantly.

Alveolar bone grafting (ABG) was described at the beginning of the
20\textsuperscript{th} century as a surgical intervention to be employed
in reconstruction of alveolar clefts.\textsuperscript{6} However,
success rates were initially low and varied tremendously depending on
the age at which it was performed, the type and source of bone used and
the surgeons expertise. With familiarity of the procedure increasing in
recent times, ABG is now recognized as the treatment of choice in
patients with alveolar clefts secondary to cleft lip and
palate.\textsuperscript{5} It remains a complex procedure requiring
meticulous planning and flawless execution by a multidisciplinary team
consisting partly of craniofacial orthodontists and oral and
maxillofacial surgeons.\textsuperscript{7} ABG involves reconstruction
of the alveolar defect through provision of bone (ideally autologous)
from a donor site that is then transferred to `fill' in the deficiency
at the recipient site.\textsuperscript{8} Despite great developments and
improvements in the surgical technique the matter of the ideal source of
bone graft material still remains controversial to this
day.\textsuperscript{9}

Presently, bone from the iliac crest is widely considered the `gold
standard' due to its potential to supply large quantities of
endochondral cancellous bone for reconstruction of large alveolar
defects.\textsuperscript{10} However, many authors report significant
donor site morbidity, thus leading to the search for alternative
intra-oral donor sites that have easy accessibility, rapid harvesting
time and a low donor site morbidity.\textsuperscript{11} In line with
this, intramembranous bone grafts from the mental symphysis and
mandibular ramus have been used, albeit
sparingly.\textsuperscript{5}Even though endochondral bone grafts remain
more popular than intramembranous grafts, literature reveals
endochondral grafts take a far longer time to achieve complete
osseointegration and may undergo up to 65\% volume loss
post-operatively. In comparison, intramembranous bone grafts have shown
promising results with rapid healing, revascularization and negligible
bone loss.\textsuperscript{12}

The maxillary tuberosity is recognized as an alternative intra-oral
donor site, however its use has been limited to minor maxillary and
mandibular alveolar ridge augmentation prior to fabrication of
prosthesis and sub antral augmentation.\textsuperscript{13} Bone from
this region and other intra-oral donor sites develop by intramembranous
ossification, similar to the mode of ossification of the alveolar ridge.
Indeed, studies on animal subjects show rapid osseointegration and
healing when `like is replaced with like'.\textsuperscript{14} A paucity
of information exists regarding the use of bone from the maxillary
tuberosity in reconstruction of bilateral alveolar clefts secondary to
cleft lip and palate. Additionally, the possibility of odontectomy
(disimpaction) sites of the 3\textsuperscript{rd} molars being a donor
site for ABG has not been discussed previously. In this paper, we report
the management of a 14 year old female presenting with bilateral cleft
lip and palate by a combination of orthodontic treatment and surgical
reconstruction of the alveolar ridge using bone harvested both from the
maxillary tuberosity and odontectomy sites of the
3\textsuperscript{rd}molars.

\textbf{CASE PRESENTATION}

A 14-year-old African female was referred to the Nairobi hospital,
Kenya, seeking treatment for secondary defects of bilateral cleft lip
and palate together with oro-nasal fistulae. The chief complaint was
misalignment of anterior teeth, creating an unsatisfactory aesthetic
appearance for the patient. In addition, the patient reported difficulty
in feeding due to oro-nasal regurgitation, especially while consuming
fluids. The patient had unsuccessfully undergone previous cheiloplasty
and several attempts at cleft palate repair prior to referral.
Extra-oral examination revealed a whistling deformity characterized by
an unsightly central vermillion notching and residual scars on the upper
lip consistent with past surgical procedures. Upon intra-oral and dental
cast analysis, it was verified that the patient had a collapse of the
maxilla characterized by a class III skeletal relationship, anterior
crossbite, bilateral posterior crossbite as well as palato-nasal and
labio-nasal fistulae. Additionally, the maxillary lateral incisors (12
and 22), maxillary canine (13) and maxillary
2\textsuperscript{nd}premolar (25) were clinically absent \emph{(Figure
1)}. Through the panoramic radiograph, absence of 12 was confirmed while
13, 18, 22, 28, 38 and 48 were all impacted. A computed tomography (CT)
scan revealed a bilateral discontinuity of the maxillary alveolar ridge
resulting in a floating anterior maxillary segment with attachment
solely to the nasal septum. In addition, a total of three round/oval
oro-nasal fistulae were visualized \emph{(Figure 2)}. The patient was
managed with a multidisciplinary approach in three well-defined phases.
The first phase consisted of pre-surgical orthodontic treatment and
involved use of a hyrax rapid maxillary expander. The screw was turned
one quarter of a turn once a day for a total of 5 weeks. At the end of
expansion, the device was kept in place for another 5 months, after
which, upper and lower orthodontic fixed appliances were bonded. The
lower 1\textsuperscript{st} premolars were extracted in an attempt to
balance the occlusion. The final step in the first phase of treatment
involved right maxillary ABG to reconstruct the cleft of the alveolus.
Surgical exposure and consequent orthodontic traction was then employed
to align the 13 into occlusion \emph{(figure 3)}. The second phase of
treatment entailed left maxillary ABG and closure of the oro-nasal
fistulae. It was noted that the wisdom teeth were impacted (see
\emph{figure 2}) and upon recommendation from the orthodontist, they
were removed. Hence proper planning of the surgery was imperative which
comprised of surgical odontectomies (disimpactions) of 18, 28, 38 and 48
followed by harvesting of the particulate cortico-cancellous bone from
the maxillary tuberosity, distal to 18 and 28 and from the retromolar
area distal to 38 and 48. The volume of bone harvested in this case was
15 cc in total. After the bone was obtained, a buccal flap was raised to
expose the cleft region, followed by disimpaction of the unfavorably
positioned 22 present within the cleft. Nasal floor soft tissue repair
was then performed followed by packing of the particulate bone into the
cleft \emph{(figure 4).} Due to the heavy scarring resulting from
multiple unsuccessful palatal surgeries performed previously, a poor
soft tissue profile (deficiency) was noted around the oro-nasal fistulae
(\emph{figure 3(b)}). This prevented adequate local soft tissue closure
necessitating the use of an anteriorly based, left dorsal tongue flap.
The flap was designed and elevated with a 5 mm thickness and adequate
pedicle length that was enough to allow suturing to the palate without
any tension. Post-operatively, the patient was fed via nasogastric tube
for 5 days after which the oral feeding resumed albeit, on a pureed
(blenderized) diet. Three weeks later, the flap was divided and the rest
returned to the donor site \emph{(figure 5).}Postoperative pain was
managed using a combination of paracetamol and diclofenac. Antibiotic
cover consisted of Augmentin 1.2g, IV for 3 days and then 1g peroral
twice a day for 4 days. Clinical evaluation after discharge was
undertaken at 2 weeks, 1, 3 and 6 months duration. In order to assess
graft survival and dental arch stability, intraoral periapical (IOPA)
and a digital orthopantomogram (OPG) were taken after 6 months (figure
6). The third phase of treatment comprised of post-surgical orthodontics
in order to close spaces and coordinate the occlusion. Overall, the
orthodontic treatment took 47 months to complete.

\textbf{DISCUSSION}

Cleft lip and palate is considered the most prevalent congenital
craniofacial birth defect and is the second most common congenital
malformation of the human body, second only to
clubfoot.\textsuperscript{15} Fusion of several structures and processes
of the neonatal face result in development of both the lip and palate
between the 4\textsuperscript{th} to 12\textsuperscript{th} week of
gestation. A failure of fusion due to genetic or environmental causes
may lead to the development of cleft lip and palate.\textsuperscript{16}

ABG forms a fundamental component of the treatment protocol of alveolar
clefts in patients with cleft lip and palate. The main objectives of ABG
are to: establish continuity of the dental arch, facilitate closure of
oro-nasal fistulae, correction of the nasal alar bases and to provide
solid bone for tooth migration and dental implant placement. Although
its use has increased, certain aspects of the surgical technique are
shrouded in controversy.\textsuperscript{5} The timing at which ABG is
performed is one such dilemma with two possible approaches having been
proposed: primary bone grafting during infancy or secondary bone
grafting during the mixed dentition period.\textsuperscript{17}Recently,
some consensus seems to have been reached with most surgeons opting for
secondary ABG between 8 to 10 years of age due to lower incidences of
complications such as maxillary growth restriction which have been
reported frequently after primary ABG.\textsuperscript{9,18}However, the
current debate revolves around the choice of ideal source of bone graft
material which may be even more controversial than the timing issue of
ABG.

The ideal bone graft sites can be grouped into either extra-oral sites
such as the iliac crest, proximal tibia and ribs or intra-oral sites
such as the mandibular symphysis and mandibular
ramus.\textsuperscript{5} The selection of a particular donor site is
dependent upon the size of the defect being repaired, ease of harvest,
donor site morbidity and the experience and preference of the
surgeon.\textsuperscript{11} The various donor sites also provide the
surgeon with a choice of either endochondral cancellous bone (extra-oral
sites) or intramembranous cortico-cancellous bone (intra-oral
sites).\textsuperscript{9} For many years it has been believed that
endochondral bone is far more superior to intramembranous
bone.\textsuperscript{9,11} However, due to the increased cortical bone
content in intramembranous bone harvested from intra-oral sites, it
undergoes delayed resorption and therefore maintains its volume for a
prolonged period of time compared to endochondral
bone.\textsuperscript{14} Additionally, intramembranous grafts have
shown to develop up to 166\% more new bone around the graft site, which
is significantly higher than endochondral grafts.\textsuperscript{12}

The maxillary tuberosity contains an appreciable amount of
intramembranous bone which can be used to reconstruct small to medium
alveolar clefts. After careful patient assessment, the amount of bone
obtained can be enhanced further by also harvesting bone from
odontectomy sites of the wisdom molars. If all these sites are utilized,
there is a potential of harvesting up to 30 cc of bone, which can
satisfy extensive grafting requirements.\textsuperscript{19} Other
salient advantages of using these sites as a source of bone graft lies
in their convenient anatomical location, a single surgical site in the
same region of the body as opposed to two sites away from one another,
minimal post-operative complications, hidden scars and a much shorter
hospital stay.\textsuperscript{5}

We therefore strongly recommend that clinical examination of these
regions be a part of the routine evaluation of patients when selecting a
donor site for ABG.\textsuperscript{13} During pre-operative assessment,
Cone Beam Computed Tomography (CBCT) can be implemented to make an
accurate 3-dimensional analysis of the maxilla and mandible for the best
sites of bone graft material. Additionally, the timing of removal of the
wisdom teeth should ideally coincide with repair of the clefts. It seems
that the use of these sites can be a simple and valuable alternative
technique for alveolar cleft reconstruction with fewer intraoperative
difficulties and post-operative complications. In the event that the
maxillary antrum is exposed during bone harvesting, primary immediate
repair can be done.\textsuperscript{19} Since some of these patients
present after having undergone unsuccessful repair of oro-nasal
fistulae, advancement of local flaps to close the defects will not be
successful. A well designed pedicled tongue flap is the best alternative
for soft tissue repair of such defects.

In the present case, ABG was successful based on the clinical and
radiographic findings \emph{(figure 6).} There was establishment of good
maxillary arch form with stabilization of the premaxillary segment.
There was also complete closure of oro-nasal fistulae, significant
improvement in the patient's occlusion and facial profile. Overall, a
satisfactory aesthetic outcome was achieved.

\textbf{CONCLUSION}

ABG is a complex procedure requiring interdisciplinary effort in order
to achieve the intended results. We have presented a case where ABG was
performed using unconventional donor sites such as the maxillary
tuberosity and mandibular wisdom molar odontectomy sites to harvest
significant amounts of bone. The intramembranous bone harvested from
these sites achieved complete osseointegration with minimal resorption
after grafting. Therefore, these could be identified as alternative
donor sites during the pre-operative assessment of patients requiring
ABG secondary to cleft lip and palate. Furthermore, where there is
immense scarring making local tissues unsuitable oro-nasal fistula
repairs, a tongue flap may be a good alternative to close the fistulae.

\textbf{FUNDING}

This research received no specific grant from any funding agency in the
public, commercial, or not-for-profit sectors.

\textbf{CONFLICT OF INTEREST}

The Author(s) declare(s) that there is no conflict of interest.

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\textbf{FIGURE LEGENDS}

\textbf{Figure 1:} Dental casts showing the maxillo-mandibular
relationship \textbf{(a)} and arch form \textbf{(b)} of the patient and
intraoral photograph showing the rotated anterior maxillary segment,
anterior crowding and a centrally placed oro-nasal fistula \textbf{(c)}
.

\textbf{Figure 2: \emph{(a)}} Panoramic radiograph at the beginning of
treatment showing anterior maxillary crowding with impacted
teeth\emph{(green arrows)} . \emph{(b)} Axial section CT scan revealing
a discontinuity of the maxillary alveolus accompanied by fistulae
\emph{(yellow arrows). \textbf{(c)}} 3D reconstruction showing the
floating anterior maxillary segment.

\textbf{Figure 3:} Surgical exposure of 13 and orthodontic traction in
order to align the tooth in occlusion (a) . Results by the end of phase
one treatment \emph{\textbf{(b)}.}

\textbf{Figure 4:} Intraoperative photographs showing exposure of the
cleft segment and soft tissue repair of the nasal floor (a) followed by
packing of the bone graft to reconstruct the alveolus
\emph{\textbf{(b)}.} This was followed by adequate soft tissue cover
using a mucoperiosteal flap \emph{\textbf{(c)}.}

\textbf{Figure 5:} The anteriorly based, left dorsal tongue flap
design\emph{\textbf{(a)}.} Suturing of flap to palate with adequate
pedicle length while its base remained attached to the tongue, promoting
vascularization and healing \emph{\textbf{(b)}.} Adequate closure of all
oro-nasal fistulae was achieved with no futher complications

\emph{\textbf{(c)}.}

\textbf{Figure 6:} Patient's final intraoral appearance, after
completion of treatment \emph{\textbf{(a)}.} The IOPA shows complete
osseointegration at the recipient site with adequate bone stock between
21 and 23 \emph{\textbf{(b)}.}\selectlanguage{english}
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