Oral Oncology 47 (2011) 145146

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Oral Oncology
journal homepage: www.elsevier.com/locate/oraloncology

Management of recurrent ameloblastoma of the jaws; a 40-year single

institution experience
Doenja Hertog a, Engelbert A.J.M. Schulten a, C. Ren Leemans b, Henri A.H. Winters c, Isac Van der Waal a,
a
Department of Oral and Maxillofacial Surgery/Oral Pathology, VU University Medical Center and Academic Centre for Dentistry Amsterdam (ACTA), Amsterdam, The Netherlands
b
Department of Otolaryngology Head and Neck Surgery, VU University Medical Center, Amsterdam, The Netherlands
c
Department of Plastic and Reconstructive Surgery, VU University Medical Center, Amsterdam, The Netherlands

a r t i c l e i n f o s u m m a r y

Article history: Ameloblastoma is a histologically almost always benign odontogenic tumor with a high rate of recur-
Received 27 October 2010 rence if not removed completely. Therefore, radical surgery is the treatment of choice of a primary ame-
Received in revised form 10 November 2010 loblastoma. Of 18 patients with a recurrent ameloblastoma, previously treated by enucleation, radical
Accepted 11 November 2010
surgery was deemed impossible in three because of the extent of the tumor or because of a poor general
Available online 14 December 2010
condition of the patient. Of the remaining 15 patients three refused to undergo radical surgery and have
been treated for their recurrence by enucleation again.
Keywords:
In none of the twelve remaining patients treated by radical surgery a recurrence was observed in a
Odontogenic tumors
Recurrent ameloblastoma
mean follow-up period of 10.5 years. In one of these patients a metastatic cervical lymph node was
Ameloblastoma detected during the primary reconstruction of the mandibular defect. The absence of recurrences in
patients treated by radical surgery should be looked at with some reservation, since recurrences may still
show up after 10.5 years. The three patients who refused radical surgery all developed one or more new
recurrences.
2010 Elsevier Ltd. All rights reserved.

Introduction at the Department of Oral and Maxillofacial Surgery of the VU Uni-

Ameloblastoma is a rather rare odontogenic tumor. The esti-
mated incidence is approximately 0.5 per million population per
year, with no gender predilection. Most patients are aged between
30 and 60 years at the time of diagnosis. Almost all ameloblasto-
mas are histologically benign and incomplete removal usually
leads to recurrence. In the 2005, World Health Organization Classi-
cation of Odontogenic Tumours four subtypes of benign amelo-
blastoma are recognized, being (1) the solid/multicystic type, (2)
the desmoplastic type, (3) the unicystic type, and (4) the extraos-
seous/peripheral type.[1] Radical surgery is the treatment of choice
with low recurrence rates both in case of a primary and a recurrent
ameloblastoma.
In this study the experience with the management of recurrent
ameloblastoma over a 40-year period is reported.
Patients and methods
In the period between 1969 and 2009, 18 patients have been
diagnosed with a histologically benign, recurrent ameloblastoma
Corresponding author. Address: Department of Oral and Maxillofacial Surgery/
Oral Pathology, VU University Medical Centre (VUMC)/Academic Centre for
Dentistry Amsterdam (ACTA), P.O. Box 7057, 1007 MB Amsterdam, The Nether-
lands. Tel.: +31 20 444 4039; fax: +31 20 444 4046.
E-mail address: i.vanderwaal@vumc.nl (I. Van der Waal).
versity Medical Centre in Amsterdam, the Netherlands. There were
eight men and ten women. Seven patients had been treated by enu-
cleation in our own institution on a single occasion. The remaining
eleven patients had been treated elsewhere for their primary ame-
loblastoma by enucleation. Recurrence has been dened as a re-
lapse after a minimum disease free period of 1 year. The mean
number of recurrences was 1.8 (range 13). The information as
being retrieved from the les included demographics, localization,
histopathological subtype, and type of previous surgery (Table 1).
Of the 18 patients with recurrent ameloblastoma radical sur-
gery was considered impossible in three because of the extent of
the tumor or a poor general condition. Of the remaining 15 patients
three refused to undergo radical surgery and have been treated
conservatively. In one of these three patients radical surgery was
performed after having experienced ve recurrences. Radical sur-
gery in the remaining 12 patients consisted of resection with a
margin of 1.52 cm. All defects were immediately reconstructed,
except for 4 patients in the rst part of the study period when
no free vascularised aps were used for reconstruction of the man-
dible. In two of the three maxillary cases rehabilitation consisted of
an obturator, in 1 patient followed by delayed reconstruction with
a free vascularised bula transplant. In the last decades, recon-
struction included prosthetic rehabilitation using dental implants.
Follow-up was performed annually by clinical and radiographic
examination, supplemented by CT or MRI in case of maxillary

1368-8375/$ - see front matter 2010 Elsevier Ltd. All rights reserved.
doi:10.1016/j.oraloncology.2010.11.008
146 D. Hertog et al. / Oral Oncology 47 (2011) 145146

Table 1 surgery. At present, 45 years after the rst treatment of his amelo-
Characteristics of 18 patients with recurrent ameloblastoma. blastoma the patient is doing well. Another patient repeatedly re-
Gender fused radical surgery for his recurrence and has been treated ve
Male 8 times for such a recurrence in a period of 30 years. At present, he
Female 10 is asymptomatic in spite of having a new recurrence in his mandi-
Age (in yrs) 26.5 (range 1253) ble. The third patient initially refused to undergo radical surgery
Localization for her primary ameloblastoma. Only when she experienced a fth
Mandible 15 recurrence, within a period of 11 years, she consented in having
Maxilla 3 radical surgery.
Histological subtype In one case we had the experience of a radiographically con-
Follicular 1 vincing recurrence, while after exploration no tumor could be
Plexiform 2
Follicular/plexiform 2
demonstrated upon histopathological examination. Therefore, we
Desmoplastic 1 strongly recommend to have a biopsy taken before performing rad-
Unicystic 5 ical surgery in what seems clinically and radiographically an obvi-
Acanthomatous 2 ous recurrent ameloblastoma.
Mixed pattern or difcult to assess 5
As described in a previous paper on the treatment of primary
Type of previous surgery ameloblastoma, half of the patients who had been treated by
Enucleation 18
enucleation alone, developed a recurrence during a mean follow-
up of 9.9 years.[3] All patients who consented in having radical sur-
gery have been successfully treated, the mean follow-up being
involvement, up to a period of at least 10 years. The minimum fol- 7.4 years. The 3 patients who refused radical surgery for their rst
low-up period was 3 years. recurrence, being treated by enucleation again, all experienced a
second recurrence in a mean period of 9.5 years.
Results Recurrences have been suggested to be correlated to the histog-
ical subtype of the ameloblastoma, such as follicular, plexiform,
None of the 12 patients with recurrent ameloblastoma showed desmoblastic and unicystic subtype. However, the general view is
a recurrence after radical surgery in a mean follow-up period of that the radicality of surgery by far outweights histopathological
10.5 years. Consequently, no statistical analysis could be per- typing with regard to outcome of the treatment, as was apparently
formed with regard to demographics, localization and histopathol- also the case in the present study. It is generally recommended to
ogical subtype. In one of the patients a metastatic cervical lymph take a margin of 1.52.0 cm of uninvolved bone around the tumor,
node in level I was detected during the resection. This patient if feasible.
has been described elsewhere in more detail.[2] The absence of recurrences in our patients treated by radical
The 3 patients, who initially refused radical surgery for their surgery should be looked at with some reservation, since recur-
recurrence, all developed a new recurrence during a mean fol- rences may still show up after the 10.5 year follow-period in the
low-up of 10.5 years (range 8.713.6 years); 2 of them were again present study.[4,5] Nevertheless, the question may be raised
treated by enucleation because of persistent refusal to undergo whether follow-up after more than ten uneventful years post-
radical surgery. The third patient agreed to undergo radical surgery treatment is indicated.
after having experienced ve recurrences.
Conict of interest
Discussion
None declared.
The demographic data of the presently reported patients are
more or less similar as in other studies of recurrent ameloblas- References
toma. Six out of the 11 patients who had been treated for their pri-
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