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Bangladesh Journal of Medical Science Vol. 14 No. 02 April’15

Case report
Ameloblastic carcinoma of the mandible 

Purmal K1, Alam MK2, Pohchi A3, Rahman SA4

Abstract:
Ameloblastic	 carcinoma	 is	 a	 highly	 malignant	 tumour	 and	 requires	 aggressive	 treatment.	
This	case	report	describes	an	aggressive	ameloblastic	carcinoma	that	infiltrated	the	mandible.	
Mandibulectomy	with	right	functional	radical	neck	dissection	and	left	supra	omohyoid	dissection	
was	followed	by	primary	reconstruction	with	a	single	free	vascularised	fibula	flap.		The	post-
operative	course	was	uneventful.	The	2	year	regular	follow	up	revealed	no	signs	of	recurrent	
tumour or metastasis.  Future reporting of this rare condition is encouraged in lieu of limited 
information in its clinical course and prognosis. 
Keywords: ameloblastic	 carcinoma;	 mandibulectomy;	 radical	 neck	 dissection;	 malignant	
tumour;	metastasis

Corresponds to: Dr. Mohammad Khursheed Alam, Orthodontic Unit, School of Dental Science, 
Universiti Sains Malaysia. Kubang Kerian 16150, Kota Bharu, Kelantan, Malaysia. Email: 
dralam@gmail.com, dralam@usm.my. 

1. Kathiravan Purmal, Resident, Oral and Maxillofacial surgery Department,
2. Mohammad Khursheed Alam, Senior Lecturer, Orthodontic Unit.
3. Abdullah Pohchi,  Senior Lecturer, Oral and Maxillofacial surgery Department.
4. Shaifulizan Abdul Rahman, Senior Lecturer, Oral and Maxillofacial surgery Department.
School of Dental Science, Universiti Sains Malaysia, Kota Bharu, Kelantan, Malaysia.

Introduction
Ameloblastoma is the most common epithelial 
odontogenic	 tumour,	 representing	 1%	 of	 all	 oral	
neoplasia	 with	 incidence	 of	 80%	 in	 the	 mandible	
and	20%	in	the	maxilla.1This benign neoplasma is 
locally	 invasive	 and	 shows	 considerable	 tendency	
to	recur,	but	rarely	behaves	aggressively	or	shows	
metastatic dissemination.2 Malignant ameloblastoma 
on the other hand has a typical benign histological 
appearance but gives rise to nodal or pulmonary 
metastasis. Both the primary and metastatic lesions 
retain their benign histologic appearances.  Therefore 
the ability of ameloblastoma to metastasize cannot 
be predicted by the histology alone.3, 4
Ameloblastic	 carcinoma	 which	 is	 different	
entity form malignant ameloblastoma is a rare 
malignant odontogenic tumour.5 In fact in 2005, 
The	 World	 Health	 Organization	 have	 reclassified	
odontogenictumours	and	defined	it	as	anodontogenic	
malignancy that combines the histological features 
of	 ameloblastoma	 with	 cyctologicatypia,	 even	 in	
the absence of metastases.  It may develop de no vo 
(primary	type)	or	by	malignant	transformation	of	an	
ameloblastoma	 (secondary	 type)	 with	 a	 distinction	
between	intraosseous	and	periphery	ameloblastoma.6
Ameloblastic	 carcinoma	 shows	 many	 histologic	

DOI: http://dx.doi.org/10.3329/bjms.v14i2.20931 
Bangladesh Journal of Medical Science Vol.14(2) 2015 p.196-202

Figure 1. Extra oral presentation of the swelling.



197

Ameloblastic carcinoma

features	 of	 malignancy	 like	 dysplasia,	 increased	
mitosis,	 and	 infiltration	 of	 surrounding	 tissues.		
However	the	common	features	of	ameloblastoma	like	
having	epithelium	in	which	the	basal	cells	contains	
columnar or palisaded cells that have a tendency for 
the nucleus to move from the basement membrane to 
the	opposing	end	of	the	cell	(reverse	polarization)	is	
still	present.		The	epithelium	is	also	known	to	in	the	
form of islands, strands and medullary arrangements 
against	a	background	stroma	of	fibrous	connective	
tissue	(follicular	pattern)	or	the	epithelium	proliferates	
in	a	mesh	arrangement	(plexiform	pattern).	Clinical	
presentation	of	this	lesion	is	the	rapid	growth,	causing	
pain and may even perforate the cortex. Although 
rare,	these	lesions	have	been	known	to	metastasize	
mostly to the lung or regional lymph nodes.7
Case Report
A	53	year	old	Malay	fisherman	came	to	the	School	
of	Dental	Sciences	University	Sains	Malaysia	with	
complaints	 of	 swelling	 in	 the	 lower	 jaw.	 He	 had	
first	noticed	the	swelling	about	6	months	ago.	The	
swelling	 has	 been	 increasing	 in	 size	 with	 bilateral	
lower	lip	parathesia.	It	was	also	associated	with	pain	
and trismus. 
His	significant	medical	history	includes	pulmonary	
tuberculosis	diagnosed	one	year	ago.	He	has	since	
completed the regime of anti-tuberculosis drugs and 
currently	on	regular	follow-up	with	the	respiratory	
team.  
Clinical	examination	revealed	a	fit	man	with	no	signs	
and	 symptoms	 elsewhere.	 There	 was	 a	 bony	 hard	
swelling	from	the	right	ramus	to	the	left	ramus.	The	
size	of	the	swelling	was	6cm	by	5	cm.	The	overlying	

skin	was	normal	and	not	fixed	to	the	lesion	(Figure	
1).	There	was	reduced	mouth	opening	about	3cm.
On	the	neck	the	right	submandibular	lymph	nodes	
(Level	I)	and	right	upper	cervical	lymph	nodes	(Level	
II) were	palpable.	The	size	of	both	the	lymph	nodes
was	1cm	by	1cm.	The	lymph	nodes	were	not	tender	
or	fixed	to	the	overlying	skin.	The	bony	lesion	had	
perforated	the	oral	mucosa	of	the	right	side	which	
is	highly	suggestive	of	malignancy	(Figure	2	and	3).
Intraoral	 examination	 showed	 a	 partial	 edentulous	
arch	with	mobile	(grade	3)	teeth.		Bony	expansion	
was	evident	bilaterally.	
Orthopanthomogram	 radiograph	 showed	 a	
multilocularosteolytic	 lesion	 with	 distinct	 margin	
extending from right to the left angle of mandible 
(Figure	4).	There	is	resorption	of	root	of	lower	first	
left molar in the mandible. No lesion detected in the 
maxilla. There is multiple retain roots and vertical 
bone loss around the teeth present.
The	 posterior	 anterior	 chest	 radiograph	 shows	 left	
upper	lobe	fibrotic	changes	(Figure	5).
CT	scans	of	from	the	base	of	skull	to	the	abdomen	

Figure 2. Right side of the lesion. Figure 3. Left side of the lesion.

Figure 4. Orthopantomogram radiograph showing 
extension of the lesion.



198

Purmal K, Alam MK, Pohchi A, Rahman SA

showed	the	lesion	is	confined	to	the	mandible.	CT	
guidedpercutaneous	lung	biopsy	was	done.	It	showed	
no ameloblastic carcinoma cells on the left upper 
lobe.  Fine needle aspiration cytology of the palpable 
lymph	nodes	was	done.	That	showed	ameloblastic	
cells	infiltration.	TNM	staging	T4N2bM0
Based	 on	 these	 findings,	 the	 diagnosis	 was	
ameloblastic carcinoma and the differential 
diagnosis	 was	 intraosseous	 carcinoma,	 squamous	
odontogenictumour, calcifying epithelial odontogenic 
tumour, salivary gland neoplasia or other metastatic 

carcinoma	to	the	jaws	from	other	primary	locations.
Histopathology
Incisional	biopsy	revealed	the	tumour	was	composed	
of islands and sheets of odontogenic epithelium 
exhibiting basal cell palisading and hypercellular 
stellate	reticulum	like	cells	having	vacuolated	nuclei	
with	 prominent	 nucleoli.	 The	 periphery	 of	 cells	
nests exhibited a columnar morphology. These cells 
contained	pleomorphic	nuclei	with	mitotic	figures.		
Squamous	 metaplasma	 together	 with	 infiltrating	
well-differented	 squamous	 cell	 carcinoma	 islands	
were	present	in	these	areas	(Figure	6	a-d)
Treatment
Surgical	excision	(2cm	clear	margin)	with	primary	
reconstruction	of	the	defect	was	the	mode	of	treatment	
chosen	for	this	patient	(Figure	7).Tracheostomy	and	
reconstruction	 with	 osteomyocutanous	 free	 fibula	
flap	was	planned.	The	mandible	was	resected	from	the	
right body to the left body preserving both condyles. 
All	 the	upper	 teeth	which	had	poor	prognosis	and	
retain	 roots	 were	 also	 extracted.	 Right	 functional	
neck	 dissection	 and	 left	 supraomohyoid	 dissection	
was	done.

Figure 4. Orthopantomogram radiograph showing 
extension of the lesion.

Figure 6. Histopathology: a. Sheets of odontogenic epithelium with palisading basal cell and hypercellular 
stellate reticulum like cells with vacuolated nuclei. b. Higher magnification showing cells containing pleo-
morphic nuclei with mitotic figures. c. Squamous cell metaplasia with infiltrating squamous cell carcinoma. 
d. Higher magnification showing tumour infiltrating the muscle tissues.



199

Ameloblastic carcinoma

A	23	cm	length	fibula	graft	with	overlying	skin	was	
harvested	from	the	right	leg.		The	fibula	graft	was	
fixed	with	 the	mandible	with	 reconstruction	plates	
and	titanium	screws.		The	vessels	were	anastomosed	
to	 the	 facial	 artery	 and	 veins	 while	 the	 skin	 from	
the	 dorsum	 of	 the	 thigh	 was	 used	 to	 provide	 the	
intraoral	 cover.	 Nasogastric	 tube	 was	 inserted	 to	
facilitate	feeding	(Figure	8).The	graft	and	donor	site	
healed	without	any	complications	(Figure	9).	Post-
operativeOrthopanthomogram	radiograph	shows	two	
reconstruction	plate	with	the	fibula	graft	(Figure	10).	
The	resected	mandible	and	lymph	nodes	were	sent	
for	histopathological	study.	 	The	results	confirmed	
our	diagnosis	of	ameloblastic	carcinoma	with	2	cm	
clear margin of the lesion and the remaining lymph 
nodes free from any lesions.
The	patient	was	sent	for	radiotherapy	after	4	weeks	
to eliminate any residual cancer cells. The dose given 
was	60	Gy	over	a	6	weeks	period	with	radiation	5	
days	a	week.	Post	radiation	shows	area	of	fibrosis	on	
the	right	and	left	neck	area	(Figure	11	and	12).	Noted	

wound	breakdown	on	left	side	of	the	neck	(Figure	
12).	The	breakdown	was	managed	by	daily	dressing	
and	 cleaning	 with	 chlorhexidine	 solution	 weekly.	
The	wound	healed	uneventfully.	He	also	complained	
about	the	hair	growth	intraorally	which	was	trimmed	
regularly.	Retrospectively	a	skin	graft	from	non-	hair	
bearing area might have avoided this problem.
After	six	months,	the	patient	was	fitted	with	upper	and	
lower	full	dentures.		He	is	able	to	lead	a	reasonable	
normal life after the rehabilitation. 
After 2 years our patient continues to remain free of 
local, regional or distant metastasis as he remains on 
active	follow-up	with	periodic	radiographic	follow	
up	(Figure	13).
Discussion
Our	 patient	 was	 diagnosed	 as	 having	 pulmonary	
tuberculosis one year before seeing us. As a 
consequence,	the	lesion	seen	in	the	chest	radiograph	
(Figure	5)	and	the	palpable	lymph	node	could	be	also	
due to the metastasis or pulmonary tuberculosis. Datta 
et al8 have mentioned that ameloblastic carcinoma is 

Figure 7. Mandible resected and recon plate 
being contoured.

Figure 9. Intra oral view of the reconstructed 
flap and skin cover.

Figure 8. Immediate post op view with 
tracheostomy and Nasogastric tube.



200

Purmal K, Alam MK, Pohchi A, Rahman SA

an aggressive neoplasm that is locally invasive and 
can spread to regional lymph nodes or distant sites 
such as lungs and bones.  Fine needle aspiration 
cytology	confirmed	the	palpable	lymph	nodes	were	
infiltrated	with	ameloblastic	cells	and	biopsy	of	the	
lungs	 showed	 no	 ameloblastic	 cells	 in	 the	 lungs.		
Even though this case demonstrated unusual spread 
within	short	period	time	(6	months),	we	had	to	delay	
the treatment because of the patient’s pulmonary 
tubercolosis.	We	waited	until	his	sputum	tests	were	
negative	for	3	consecutive	weeks	before	doing	the	
operation to reduce the chances of cross infection.  
The diagnosis criteria of ameloblastic carcinoma 
from ameloblastoma or malignant ameloblastoma 
are based on cytologicatypia and increased mitotic 
activity.9 Most ameloblastic carcinoma arise de 
novo	and	less	than	1%		of	ameloblastoma	undergo	

malignant transformation.10 Some studies have 
recorded	widespread	metastatsis	to	the	lung,	bones	
through lymphatics and bloodstream.11, 12These 
lesions should be distinguished from metastatic 
ameloblastoma	 which	 is	 defined	 as	 a	 histoloigical	
benign	appearing	ameloblastoma	with	metastasis.	
From the differential diagnosis, intraosseous 
carcinoma	is	difficult	to	differentiate	radiographically,	
histologically or clinically from ameloblastic 
carcinoma. In fact the intra osseous carcinoma may 
represent	a	less	differentiated	non	keratinizing	form	
of ameloblastic carcinoma both being deprieved from 
odontogenic epithelial remants.3, 13 Nonethess, typical 
features	of	ameloblasticdfferentiation	which	would	
justify	 a	 diagnosis	 of	 ameloblastomic	 carcinoma	
are	 usually	 lacking	 in	 intraosseous	 carcinoma.14 
Squamous	 odontogenic	 tumour	 is	 composed	 of	
islands	of	squamous	cells	and	epithelial	odontogenic	
tumour	contains	sheets	of	epithelial	and	amyloid	like	
material	with	concentric	ring	calcification	which	can	
differentiate it from ameloblastic carcinoma.  Salivary 
gland neoplasm or secondary from other sites may 
invade	 the	 jaws	 and	 can	 be	 differentiate	 from	 the	
history and clinical symptoms.15,	16	Marsuzaki	et	al17 
have reported that immunohistochemistry study of 
ameloblastic	carcinoma	will	reveal	high	positive	rates	
of	P53	and	Ki67	to	aid	in	the	diagnosis	of	this	lesion.	
Chromosal	 imbalances	 in	 ameloblastomawhith	
losses in choromsoam 22 and 10 have been reported. 

Figure 10. Post surgery OPG with two reconstruc-
tion plate and the fibula graft.

Figure 11. Post radiation right side of the neck.
Figure 12. Left side of the neck post radiation. 
Wound breakdown noted at angle of the mandible.



201

Ameloblastic carcinoma

In additional to that aneuploidy is more common 
in ameloblastic carcinoma and may predict its 
malignant potential.18
In	 our	 patient,	 the	 histology	 showed	 features	 of	
malignancy	like	higher	mitotic	activity,	ell	atyplia,	
hyperchromatic nuclear, invasion of bone and 
muscles.	 	 Clinically	 ameloblastoma	 have	 a	 slow	
growth	with	27	months	as	the	mean	duration	before	
any symptoms.19	This	case	shows	growth	in	only	6	
months.  Moreover there is perforation of the cortex 
of mandible intraorally.  Ameloblastoma hardly 
perforates the cortex unless left undetected for a very 
long time. Pain and parathesia is also not a common 
finding	in	ameloblastoma.19 From the demographics, 
the mean age for ameloblastic carcinoma is 52, 
male	to	female	ratio	is	2:	1	and	66%	of	the	times	the	
lesion is located in the mandible.7 This information is 
consistent	with	our	patient	presented	here.
Most of the cases of ameloblastic carcinoma in 
the	 literature	 was	 treated	 with	 extensive	 surgery	
with	 margins	 of	 2-3	 cm.	 	 Radiotherapy	 was	 used	
after	 the	 surgery	 for	 cases	 with	 close	 resection	
margin, extracapsular or perineural invasion.6,	 20,	 21 
Chemotherapy	did	not	show	any	promising	results.6,
22 Radiotherapy alone is also not recommended given 
its intraosseous location.23 Recently Perera et al. 
have	reported	on	the	use	of	gamma	knife	stereotatic	
radiosurgery in the management of recurrent 
ameloblastic	carcinoma	lesion.	The	authors	showed	
promising results for recurrent lesions after 2 years 
of post-resection.  Based on the above information, 
we	 decided	 surgical	 option	 with	 concomitant	
radiotherapy.  We managed to preserve the condyle in 
this	patient.		Keeping	the	mandibular	condyle	allows	
the	patient	to	preserve	the	temporo-	mandibular	joint	
which	gives	better	mobility	to	the	rest	of	the	jaws	and	
also diminishes post-op pain.
The	 task	 of	 restoring	 the	 mandibular	 defect	 is	 a	
challenging problem for surgeons after ablation of 
the	 tumour.	 	 Microvascular	 techniques	 to	 transfer	
vascularized	bone	to	the	head	and	neck	region	have	
been successfully applied to solve the problem of 
various segmental mandibular defects.24, 25 In our 
case,	 almost	 the	 entire	 mandible	 was	 successfully	
reconstructed	with	a	vascularised	fibula	flap.
Local	 recurrence	 have	 been	 reported	 between	 5	
to 151 months.20,	 26 Distant metastasis has also 
been reported as early as 4 months and as late as 
47 months after surgery.20, 27 Therefore long term 
follow-up	is	mandatory	to	detect	the	late	recurrence	
or	metastasis.	The	5	and	10	year	survival	rates	were	
72.9%	and	56.8%	respectively.21 It is important that 

people	should	be	fortified	to	immediately	report	to	
a	surgeon	whenever	they	see	any	unusual	swelling	
any discomfort. Management should also include 
the	awareness	of	the	people	of	their	responsibilities	
regarding	 their	 own	 health,	 and	 not	 wait	 for	 the	
swelling	 to	 grow	 and	 cause	 great	 discomfort	 and	
difficulty	 to	 themselves	 in	 future.28	 Nonetheless, 
it	 is	 generally	 known	 that	 there	 were	 significant	
association	 between	 histological	 subtype	 and	
recurrence	 outcome	 and	 between	 treatment	 and	
recurrence of ameloblastoma.29
Conclusion
Ameloblastoma	 shows	 a	 spectrum	 of	 histology	
and biologic behaviour ranging from benignity at 
one end to malignancy at the other end.  Cases of 
ameloblastoma should thus be carefully analysed to 
detect subtle changes in the histology that may predict 
its aggressive behaviour. We have reported a rare case 
of primary ameloblastic carcinoma of the mandible 
in	a	53	year	old	man.	Aggressive	wide	excision	and	
radiotherapy	with	primary	reconstruction	has	given	a	
successful	outcome.	However	long	term	follow	up	is	
necessary to detect the late recurrence or metastasis.

Conflict of interest: None declared.

Figure 13. After 2 years follow up review.



202

Purmal K, Alam MK, Pohchi A, Rahman SA

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