ARTICLE IN PRESS

Vol. 00 No. 00 && 2021

Potential role of active decompression and distraction

sugosteogenesis for the management of ameloblastomas:
Report of two cases and review of the literature
Andr
es Wiscovitch, DMD, MSD,a Jos
e S. Sifuentes-Cervantes, DDS,b Juan-Pablo Porte, DDS, MSD,c
u~
Jaime Castro-N
nez, DMD, MSD,d,e Jairo Bustillo, DDS,f Pedro Moreno-Rodr
ıguez, DDS,g and
Lidia M. Guerrero, DMD, FACSh

Ameloblastomas are aggressive odontogenic entities well-known for their high tendency to recur. Clinical presentation includes
lesions discovered on routine examination or radiographs, pathologies causing facial swelling, pain, cortical expansion, tooth
mobility, root resorption, and paresthesia. Radiographic findings comprise large unilocular or multilocular radiolucencies with
well-defined borders associated to an impacted tooth. Ameloblastomas are classified as unicystic, multicystic/solid, and periph-
eral. Treatment options include marsupialization, decompression, enucleation, or curettage with or without adjuvant measures
such as Carnoy’s solution, marginal resection, and segmental resection. Recently, active decompression with distraction sugos-
teogenesis (ADDS) was introduced for the conservative management of odontogenic cystic conditions. The purpose of this paper
is to present 2 cases of a conventional ameloblastoma treated by means of ADDS. The purpose of this novel approach is to signifi-
cantly reduce the amount of time required to decompress cystic-like lesions. In these cases, ADDS proved to be a viable treatment
because it demonstrated a reduction in size of the initial lesion by new osseous formation within 2 weeks of placement of the
device. The cases presented in this paper demonstrate that ADDS could be a valuable treatment modality for this type of amelo-
blastoma, although further research is necessary to validate this philosophy. (Oral Surg Oral Med Oral Pathol Oral Radiol
2021;000:e1 e10)

Active decompression and distraction sugosteogene-
sis (ADDS) is an emerging philosophy developed to
treat odontogenic cystic lesions by means of active
intracystic negative pressure.1-5 The technique employs
a device called the evacuator for odontogenic cysts
(Evocyst), and it is based on the rationale that negative
Received for publication July 26, 2021; returned for revision Septem-
ber 22, 2021; accepted for publication September 28, 2021.
a
PGY III, Oral and Maxillofacial Surgery Residency Program,
School of Dental Medicine, University of Puerto Rico, Medical Sci-
ences Campus, San Juan, Puerto Rico.
b
PGY I, Oral and Maxillofacial Surgery Residency Program, School
of Dental Medicine, University of Puerto Rico, Medical Sciences
Campus, San Juan, Puerto Rico.
c
PGY III, Oral and Maxillofacial Surgery Residency Program,
School of Dental Medicine, University of Puerto Rico, Medical Sci-
ences Campus, San Juan, Puerto Rico.
d
PGY III, Oral and Maxillofacial Surgery Department, School of
Dental Medicine, University of Puerto Rico, Medical Sciences Cam-
pus, San Juan, Puerto Rico.
e
Research Department, Instituci
on Universitaria Colegios de Colom-
bia, Bogota, Colombia.
f
Professor, Oral and Maxillofacial Pathology Department, School of
Dentistry, Universidad El Bosque, Bogot
a, Colombia.
g
Oral and Maxillofacial Surgery Care Center, Medell
ın and Quibd
o,
Colombia.
h
Program Director, Oral and Maxillofacial Surgery Residency Pro-
gram, School of Dental Medicine, University of Puerto Rico, Medi-
cal Sciences Campus, San Juan, Puerto Rico.
Received for publication Jul 26, 2021; returned for revision Sep 22,
2021; accepted for publication Sep 28, 2021.
Ó 2021 Published by Elsevier Inc.
2212-4403/$-see front matter
https://doi.org/10.1016/j.oooo.2021.09.014
pressure has the capacity to both stimulate osteoblasts
to accelerate the production of bone (sugosteogenesis)
and to evacuate, at least partially, harmful products
secreted by the cyst.4 By employing negative pressure,
this method decompresses cystic lesions actively
(active decompression), instead of using passive drain-
age systems well-known in clinical oral and maxillofa-
cial surgery. Because the negative pressure exerted
over the cavity actually “distracts” the bone, the term
“distraction sugosteogenesis” is usually employed to
describe the technique.
The Evocyst is composed of intra- and extraoral
units, which are interconnected to satisfy not only ther-
apeutic goals, but also patients’ needs. The intraoral
unit is a two-way system composed of irrigation and
decompression tubes. The first tube is used to irrigate
the entity, and the latter serves as a suction or decom-
pression conduit that connects to the extraoral unit.
The extraoral unit is an active negative pressure closed
drainage system capable of providing negative pressure
of about 45 mmHg. Research on the subject has
described the device in detail and reported bone forma-
tion as early as 2 weeks postoperatively.1-5
Ameloblastomas are aggressive, slow-growing
odontogenic pathologies that may present as a tumor or
cyst, particularly known for their high tendency to
recur.6 As an odontogenic entity, its epithelial lining
has its roots in the remnants of the dental lamina,
reduced enamel epithelium, or the basal epithelial cells
of the oral mucosa.7 Clinical presentation ranges from

e1
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e2 Wiscovitch et al.
Fig. 1. Patient’s clinical appearance at presentation.
unnoticeable conditions discovered on routine dental
examination/radiographs to entities causing facial
swelling, pain, cortical expansion with or without fen-
estration, tooth mobility, rhizolysis, and paresthesia.
Radiographic findings usually include large unilocular
or multilocular radiolucencies with well-defined bor-
ders associated with an impacted tooth.6-8
According to the Classification of Head and Neck
Tumors8 performed by the World Health Organization
(WHO) in 2017, ameloblastomas present in the follow-
ing 4 different types: conventional (acanthomatous,
basal cell, desmoplastic, follicular, granular cells, and
plexiform histologic subtypes); unicystic (mural, lumi-
nal, and intraluminal histologic subtypes); extraoss-
eous/peripheral; and metastasizing (malignant)
ameloblastoma. Of note, the term solid/multicystic was
eliminated because most conventional ameloblastomas
show cystic degeneration with no biologic differences.
This classification is not only of academic importance,
but it also yields to significant therapeutic and prognos-
tic considerations, which will be discussed later.
Although no consensus has been reached regarding the
best treatment modality for this entity, it is clear that varia-
bles such as patient’s age, clinical presentation, radio-
graphic characteristics, and histopathologic features must
be taken into consideration when proposing a treatment
plan.7 Historically, surgeons have advocated aggressive
methods to treat these lesions, with the intraluminal unicys-
tic form being an exception due to its lesser tendency to
recur when treated conservatively; this approach,
Fig. 2. Initial panoramic X-ray.
OOOO
&& 2021
nevertheless, is seldom chosen in clinical practice.6-7 Treat-
ment for this aggressive entity ranges from conservative
(marsupialization, decompression, enucleation or curettage
with or without adjuvant measures such as Carnoy solu-
tion) to aggressive philosophies (marginal or segmental
resection).8-12
Although the long-term results of ADDS are
unknown, initial findings are promising and have
shown it to be a predictable manner to treat odonto-
genic cysts. The purpose of this paper is to present 2
cases of ameloblastomas treated by means of ADDS.
CASE 1
This is the case of an otherwise healthy 18-year-old
Hispanic female patient with no prior history of surgi-
cal/medical intervention and no known drug/food aller-
gies. The patient presented to our institution with right-
sided facial edema, trismus, dysphagia, and odynopha-
gia (Figure 1). The patient’s primary concern was the
swelling and pain that impeded her ability to eat.
Before this emergency room (ER) visit, the patient
had not sought treatment for this lesion. Intraoral
findings included purulent discharge from the area
of an unerupted lower right third molar, tenderness,
swelling, and buccal-lingual expansion. Her vital
signs were within normal limits. Laboratory studies
showed WBC: 10.54, RBC: 3.39, HGb 8.6, HCt
27.4, ESR: 45; CRP: 116.5.
A panoramic X-ray, which revealed an impacted
third molar associated to a multiloculated radiolucent
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Volume 00, Number 00 Wiscovitch et al. e3

Fig. 3. Initial CT maxillofacial scan. (A) Lateral view. (B) Frontal view. (C) Sagittal view. (D) Coronal view.

lesion, was taken. The entity involved the distal area of
the lower right second premolar, the adjacent ipsilateral
first and second molars, angle of the mandible, and ver-
tical ramus (Figure 2). Subsequently, a computed
tomography (CT) with contrast was taken at the ER,
which revealed a large, well-defined expansile lesion
measuring 5.4 cm antero-posteior (AP) £ 3.3 cm
transverse £ 4.1 cm cranio-caudally involving the right
mandibular body, angle, and vertical ramus (Figure 3).
Involvement of the ipsilateral alveolar ridge was noted.
Additionally, the image demonstrated asymmetric
edema within the right facial soft tissues, mainly
involving the right buccal space, with overlying thick-
ening of the skin and platysma muscle and diffuse
subcutaneous fat stranding. No fluid collection was
present on imaging.
Patient was admitted to the hospital, and intravenous
antibiotic therapy and analgesics were initiated. Before
the treatment of the lesion, all conventional treatment
options were discussed with patient/mother, and they
decided to proceed with ADDS. Then, the patient was
taken to the oral and maxillofacial surgery clinic,
where she underwent incisional biopsy and ADDS.
The intracystic content available inside of the extraoral
unit was examined histologically. The smear revealed
presence of isolated mature squamous cells, few foamy
macrophages, abundant neutrophils, many lympho-
cytes, and few red blood cells. Once clinical and

Fig. 4. Follow-up panoramic X-ray 2 weeks after ADDS.

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e4 Wiscovitch et al. && 2021

Fig. 5. Follow-up panoramic X-ray 9 months after ADDS.

paraclinical parameters improved, patient was dis-
charged home.
As per protocol, she returned for a follow-up visit
with a panoramic X-ray 2 weeks later. The 2-week
radiography revealed dramatic reduction of the entity
with remarkable bone formation (Figure 4). On histo-
pathologic review, the components of the lesion were
most consistent with a conventional ameloblastoma,
plexiform subtype. Since this lesion demonstrated a
positive response to ADDS, the decision was made to
continue with treatment. ADDS lasted 4 weeks, and the
intraoral unit was removed without complications.
Following ADDS, the associated third molar was
extracted under local anesthesia.
Because there are no published reports regarding the
use of ADDS in ameloblastoma cases, this team fol-
lowed the patient every month to watch for signs of
deterioration. A panoramic X-ray taken at 9 months
revealed a small periapical radiolucency mainly in rela-
tionship with the distal root of the lower right second
molar (Figure 5). Also, a maxillofacial CT, which dem-
onstrated a remaining 1.3 £ 1.1 £ 2.3-cm lesion
involving the right mandibular body with bone present-
ing ground glass density (sclerotic changes) and no

Fig. 6. CT Maxillofacial scan at 9 months. (A) Lateral view. (B) Frontal view. (C) Sagittal view. (D) Coronal view.
 ARTICLE IN PRESS
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Volume 00, Number 00 Wiscovitch et al. e5

Fig. 7. Histopathology results consistent with ameloblastoma. Hematoxylin and eosin staining 4£.

Fig. 8. Clinical appearance.

periosteal reaction (Figure 6), was taken. The rest of CASE 2

the bone, compared with the lesion at presentation,
showed a homogenous appearance. Final enucleation
and extraction of tooth number 31 was performed
under general anesthesia. The final histopathologic
diagnosis was consistent with conventional ameloblas-
toma, plexiform subtype (Figure 7). This patient
presents no signs of recurrence 24 months after ADDS.
She has been placed on a 10-year follow-up plan with
visits twice a year during the first 5 years and annually
thereafter.
A 24-year-old female patient presented to clinic with the
chief complaint of a growing mass in her right cheek
(Figure 8). After panoramic X-ray evaluation, a unilocular
radiolucent lesion measuring 3.0 £ 2.5 cm and involving
the right mandibular premolars was noted (Figure 9). A
CT scan was ordered, and lesion size was established at
4.0 £ 3.0 £ 3.0 cm (Figure 10). Incisional biopsy was
indicated, and the ADDS protocol was started at the same
appointment. Biopsy result was consistent with follicular
ameloblastoma (Figure 11). After 6 weeks of ADDS,

Fig. 9. Initial panoramic X-ray.

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e6 Wiscovitch et al. && 2021

Fig. 10. Initial CT maxillofacial scan. (A) Coronal view. (B) Axial view.

Fig. 11. Histopathology demonstrating follicular islands of odontogenic epithelium characteristic of this subtype type. Hematoxy-
lin and eosin staining 4£.

Fig. 12. Follow-up 6 weeks later, panoramic X-ray.

DISCUSSION
noticeable radiologic changes were noted (Figure 12).
The purpose of the present article was to report 2 cases of
Once the lesion measured less than 2 cm, final enucleation
ameloblastomas treated by means of ADDS, an emerging
was performed under general anesthesia. At the 7-month
technique designed to treat odontogenic cystic lesions
follow-up radiography, substantial lesion obliteration and
employing the Evocyst (Figures 15 and 16). The method
considerable bone formation were noted (Figure 13). After
we used demonstrated osseous response/cystic shrinkage
3 years of follow-up, this patient presents no signs of recur-
as early as 2 weeks after the initiation of ADDS. Note that,
rence, and the CT scan shows remarkable improvement
as shown in Figure 4, it took 14 days of active decompres-
(Figure 14). Patient has been placed at a 10-year follow-up
sion for the initial lesion to reduce in size about 50% and
schedule.
to stimulate bone formation. Moreover, osteogenesis
 ARTICLE IN PRESS
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Volume 00, Number 00
Fig. 13. Follow-up 7 months later, panoramic X-ray.
Fig. 14. CT maxillofacial scan follow-up 7 months later. (A) Lateral view. (B) Frontal view. (C) Sagittal view. (D) Axial view.
continued after the period of mechanical stimulation had
ceased. Although the long-term results of this method
remain to be investigated, it appears to be an alternative to
conventional treatment modalities.
Currently, the management of this entity remains con-
troversial, but in general terms, it ranges from conservative
approaches to resection, whether segmental or marginal.12
Surgical resection of ameloblastomas, however, has been
the mainstay of treatment since the times of Cusack13 in
the early 1800s, with conservative approaches being
described after the mid-1900s, when Seldin14 and Seldin15
decompressed ameloblastomas in children. Note that, in
this specific population, the management is further chal-
lenged by the presence of tooth buds or unerupted teeth,
continuing facial growth, bone physiology (greater percent-
age of cancellous bone, increased bone turnover, and
CASE REPORT
Wiscovitch et al. e7
reactive periosteum), and social issues. Retention of nor-
mal facial contour and absence of disfiguring scars with
improved social adaptation are important factors in the
treatment algorithm and therefore add credence to conser-
vative options.
Recurrence, another significant factor, should be
weighted carefully in children. In 2010, Zhang et al.16
reported that the rate of recurrence after radical treat-
ment was lower than the rate of recurrence after con-
servative techniques. Although recurrence plays a
significant role in the management of ameloblastoma,
aggressive techniques should be avoided in growing
patients due to the long-term sequelae. In line with cur-
rent literature17,18 and within the context of our
patients, we proposed conservative or minimally inva-
sive methods (ie, marsupialization, passive
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ORAL AND MAXILLOFACIAL SURGERY
e8 Wiscovitch et al.
Fig. 15. The Evocyst.
Fig. 16. Intraoral unit in place.
decompression, and ADDS with final enucleation once
the lesions measured less than 2 cm).
The osseous response to negative pressure has been
documented both clinically1-5 and experimentally.19,20
Our cases demonstrate that, 2 weeks after ADDS, both
ameloblastomas had reduced more than 50% of their
original size, with a great amount of newly formed
bone surrounding the shrunk entities. In contrast to
ADDS, passive decompression would have taken
approximately 8 to 12 months to exert its effects,
according to current literature.21
In 1827, James W. Cusack published in the Dublin
Hospital Reports and Communications in Medicine
and Surgery a series of 7 cases in which he described
the resection of portions of the mandible due to
“cancerous affections [. . .] or morbid growth,” which
are now believed to be ameloblastomas.13 The opera-
tion, by then familiar in France, Great Britain, and
OOOO
&& 2021
America, had been previously reported by Lallemand
and McClelland.14 In 1885, Louis-Charles Malassez22
called it adamantinoma, and in 1930, Ivy and Church-
ill23 proposed the term ameloblastoma. Since then, sur-
geons and pathologists have devoted a large body of
literature to this entity. Currently, ameloblastomas are
characterized in light of their clinical, radiographic,
and histopathologic characteristics, all of which have
significant therapeutic and prognostic implications.24
Ameloblastomas’ radiographic features are of capital
importance because they determine whether the entity
is unilocular, a necessary parameter for unicystic ame-
loblastoma, as originally described by Robinson and
Martinez25 in 1977. The 2017 WHO Classification of
Head and Neck Tumors8 recognized once again that
the unicystic ameloblastoma is a separate entity having
3 histopathologic subtypes (intraluminal, luminal, and
mural) depending on whether solely the cystic lining is
affected (luminal) or if a solid neoplasm could be rec-
ognized extending into the lumen (intraluminal) or
infiltrating the wall of the cyst (mural).26-28
Currently, surgeons and pathologist seem to agree upon
the fact that the type which proliferates into the lumen has
a better prognosis than other types of ameloblastomas.26
The prognosis for a unicystic ameloblastoma depends on
whether its lining proliferates into the cystic cavity or into
the wall/surrounding tissues. When malignant cells spread
into the cystic wall or surrounding bone, conservative tech-
niques seem to be less effective or lead to unnecessary
recurrences. Marx and Stern27 have illustrated the microin-
vasive and invasive capacity of these malignant cells and
proposed a different terminology/classification to reflect
such behavior, although it has not gained widespread
acceptance and was not considered in the 2017 WHO Clas-
sification of Head and Neck Tumors.
 ARTICLE IN PRESS
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Volume 00, Number 00
Regarding the conventional ameloblastoma (solid/
multicystic, dropped terms in 2017), as the one our first
patient presented with, the initial radiography revealed
a multiloculated pattern with an associated third molar.
After incisional biopsy, the histopathology was consis-
tent with the plexiform subtype. Histologically, no
invasion to the wall or surrounding tissues was
observed. The fact that this conventional ameloblas-
toma was in the cystic lumen without spreading into
the wall/surroundings tissues represents an important
prognostic factor. This finding, however, does not pre-
clude the necessity of long-term follow-ups because
ameloblastomas are known for recurrence, with estima-
tions at 3.6% for wide resection, 30.5% for enucleation,
and 16% for enucleation after Carnoy solution.28
Although controversial, conservative approaches for
ameloblastomas are employed when present in chil-
dren, despite the potential for recurrence. A myriad of
authors will contend this perspective. However, when
recurrence rate is the only consideration, surgeons are
inclined to select aggressive methods and to overlook
the biological, social, and financial impact of this deci-
sion in these patients. Nevertheless, when it recurs, a
second surgery should be more aggressive. It is the
opinion of the authors that conservative methods,
which consist of marsupialization or decompression
(passive or active) followed by enucleation or enucle-
ation alone, should constitute the main treatment in
growing patients. Therefore, health professionals
should consider this option because major surgery can
cause facial deformity and dysfunction, have a negative
influence on facial growth, and effect the patient psy-
chologically. The importance of patient individualiza-
tion cannot be overstressed, and no single technique
will fit all types of ameloblastomas. Often, a combina-
tion of 2 or more methods will be required. Every case
must be studied carefully and all factors weighted
appropriately in benefit of the patient.
Along with the radiographic and histopathologic charac-
teristics of this malignancy, clinical signs and symptoms
play an equally important role. Size, cortical expansion,
associated tooth, root resorption, and anatomic location,
among others, are important characteristics having an
impact on the overall course of treatment.
Ameloblastomas are more common in the mandible
than in the maxilla. In the former anatomic location,
they frequently involve the molar/mandibular angle
(66%), premolar area (11%), and anterior region
(10%).24 The importance of the anatomic location is
unquestionable. When an ameloblastoma develops in
the symphysis area, it should prompt the clinician to
consider the desmoplastic subtype, a variant described
by Eversole et al.29 in 1984. Accounting for the signifi-
cant differences in anatomic site (predilection for the
anterior mandible), imaging (mixed radiopaque-
CASE REPORT
Wiscovitch et al. e9
radiolucent lesion), and histopathologic features (lack
of capsule, extensive stromal desmoplasia, and dense
collagenization with highly variable odontogenic epi-
thelium islands and cords of various sizes), the 2005
WHO Histopathological Typing of Odontogenic
Tumors considered it a separate clinicopathologic
entity. In the 2017 classification, however, the desmo-
plastic type was considered a histopathologic subtype
of the conventional ameloblastoma.8 Considering it a
subtype instead of a separate entity does not change
either its histopathologic features or its biologic behav-
ior. The entity still differs from the other subtypes in
that it is more frequently seen in the symphysis, and it
is composed of a mixed radiolucent radio-opaque
appearance that often resembles a benign fibro-osseous
lesion.29 Furthermore, resection is an accepted method
of treatment to prevent recurrence, which is probably
due to the lack of capsule and precise limits.30
To conclude, given our patients’ ages, clinical find-
ings, location of the entity, radiologic features, and the
histologic subtypes, ADDS was chosen as treatment
modality because it included not only a faster bone
healing, but also less patient morbidity with concomi-
tant long-time effects on their quality of life. Neither of
the patients developed paresthesia. Both patients are
being followed up, as stated previously, to verify any
signs of recurrence. Although complete removal of the
entity was performed, recurrence cannot be completely
ruled out. Long-term follow-up is warranted for them.
Disclosure: We confirm that this manuscript, nor
any part of it, has not been submitted or published and
will not be submitted elsewhere for publication while
under consideration by OOOO. We disclose no previ-
ous presentation of abstracts at meetings and no posting
of any part of it or all of the content on a website, even
in draft form.
Funding: No funding received.
ACKNOWLEDGEMENT
The authors wish to thank Drs. Cristina Ortiz-D
ıaz, Fran-
cisco Carrillo-Morales, Brayann Alem
an, Sona Rivas-
Tumanyan, and Augusto Elias-Boneta for their support.
Special acknowledgment to our attending, Dr. Bonifacio
Rivera, for his guidance. Many thanks to Dr. Jose Wisco-
vitch for sharing his knowledge on odontogenic entities.
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759.
Reprint requests:
Dr. Andr
es Wiscovitch
Oral and Maxillofacial Surgery Department
University of Puerto Rico
Medical Sciences Campus
Paseo Dr. Jos
e Celso Barbosa
San Juan
PR
00921.
wiscovitch@upr.edu