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 ( ISSN: 2814-2098 )                                                                                    Fahasoavana Rohamah Njatosoa¹* 
https://ijojournals.com/                                                     Volume 07 || Issue 02 || February, 2024 || 
 

 

 

Ameloblastoma: from benign to malignant about a case report at the Hospital Center 

Soavinandrina Antananarivo (CENHOSOA) 
 

Fahasoavana Rohamah Njatosoa¹*, Jacques Adrien Ramangason¹, Lauricia Gaël Lalanirina¹, 

Ramarosahanina Harivola Andrianantenaina¹, John Alberto Bam Rakotondrabe², Emmanuel 

Andrianony Rakotoarivony¹, Aurélien Richard Rakotoarison³. 

¹ Department of Maxillo-facial surgery in University Hospital Centrer Professor Zafisaona 

Gabriel Androva (CHU-PZaGa), Mahajanga, Madagascar  

² Department of Maxillo-facial surgery in University Hospital Centrer Joseph Dieudonné 

Rakotovao (CHU-JDR) – Befelatanana Antananarivo 

³ Department of Maxillo-facial surgery in Hospital Centrer Soavinandrina (CENHOSOA), 

Antananarivo 

 

    
   

  
   
 

Abstract  

Ameloblastoma is a common odontogenic tumor of the maxillae in developing countries such as 

Africa. It is a benign tumor, but highly invasive, recurrent and potentially malignant. The aim of 

this study was to share clinical experience of a rare case of ameloblastoma that had become 

malignant, and to determine the probable causes of this transformation. This is a retrospective 

study covering a 20-year period from 2002 to 2021. Six cases were identified, but only one met 

our inclusion criteria. All parameters were reported for comparison with the literature. Our 

patient was a 48-year-old adult who came to us in 2002 with a recurrence of right mandibular 

ameloblastoma. A total of six operations were performed on this patient, and on each occasion, 

the technique adopted was enucleation, which is a conservative surgery. The clinic is 

characterized by the absence of pain at its onset, becoming painful at the cancerous stage. The 

initial CT scan suggested well-limited, monogeneodic tissue osteolysis, then mixed polygeodic, 

then poorly limited with invasion into neighbouring tissues, and finally with lymph node and lung 

metastases. Chemotherapy was administered, but the patient did not survive. When faced with a 

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Correspondant author : Names :Fahasoavana Rohamah Njatosoa
Adress : Centre Hospitalier Universitaire PZaGa, Androva Majunga Madagascar.

mailto:njatosoas@outlook.com


IJO - INTERNATIONAL JOURNAL OF HEALTH SCIENCES AND NURSING  

 ( ISSN: 2814-2098 )                                                                                    Fahasoavana Rohamah Njatosoa¹* 
https://ijojournals.com/                                                     Volume 07 || Issue 02 || February, 2024 || 
 

 

 

 

  

 

 

 

 

 

 

 

  

 

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recurrent ameloblastoma, radical surgery of the interrupting osteotomy or non-interrupting type 

would be the best indication to avoid multi-recurrence and malignant transformation.

Key words: Ameloblastoma, Surgery, Recurrence, Malignant transformation.

INTRODUCTION

he overall aim of this study is to share a clinical experience on a case of historical ameloblastoma 

that became malignant.

Ameloblastoma is a benign, locally invasive odontogenic epithelial tumour.In 80% of cases, it is 

located in the posterior mandible.Its annual incidence in the general population is 0.5 cases per 

1,000,000 inhabitants, and more frequently in developing countries [1].This type of tumor is not 

an emergency pathology, but its seriousness stems from its insidious invasive evolution.Their 

histopathological types can vary according to the WHO classification.The most common is the 

follicular plexiform type [2].Clinically, an ameloblastoma remains asymptomatic and evolves 

slowly before the discovery of a swelling deforming the face, tooth mobility and/or falls [3].On 

radiography, it presents as a uni- or multilocular osteolytic lesion with a cystic and/or tissue-like 

appearance.Sometimes, intra-lesional bone septa are visible, giving a honeycomb-like appearance 

[3].Diagnosis is confirmed anatomically and pathologically [1, 2].

Prognosis depends mainly on how early the disease is diagnosed and managed [3]. Treatment is 

purely surgical, with a wide excision margin to avoid recurrence, which is very common.

Malignant transformation is possible after several recurrences [3, 4].

PATIENT AND METHOD

Our study was carried out at the Stomatology and Maxillofacial Surgery Department of the 

Soavinandriana Hospital (CENHOSOA) in Antananarivo. This is a rare case report. All the 

variables mentioned in this case are compared with the data in the literature. Over a study period 

extending from May 2002 to October 2021, i.e. 20 years, six cases were identified, but only one 

case met our inclusion criteria. This was the case of a patient with a multi-recurrent 

ameloblastoma who had undergone iterative surgery at CENHOSOA, and who developed a 

malignant transformation.

RESULTS

This is a 48-year-old man who presented with a benign mixed variant ameloblastoma with tissue 

predominance at onset. This patient had several recurrences. Indeed, five recurrences were



IJO - INTERNATIONAL JOURNAL OF HEALTH SCIENCES AND NURSING  

 ( ISSN: 2814-2098 )                                                                                    Fahasoavana Rohamah Njatosoa¹* 
https://ijojournals.com/                                                     Volume 07 || Issue 02 || February, 2024 || 
 

 

 

observed over 20 years. Right genital swelling was the reason for our patient's consultation. It 

was painless to palpate and caused facial asymmetry and even significant deformity of the right 

face. Dental mobility and displacement were also observed at the third recurrence in 2006. In the 

malignant stage, signs of odynophagia were mentioned. The pathological findings after the fifth 

operation suggested malignant ameloblastoma with cervical and pulmonary lymph node 

metastasis. 

The following table shows the evolution of our case. 

.

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IJO - INTERNATIONAL JOURNAL OF HEALTH SCIENCES AND NURSING  

 ( ISSN: 2814-2098 )                                                                                    Fahasoavana Rohamah Njatosoa¹* 
https://ijojournals.com/                                                     Volume 07 || Issue 02 || February, 2024 || 
 

 
 

Evolution 2002 2006 2009 2013 2014 

Mainspring for 
consultation 

Painless 
tumefaction of the 
right genicus 

Painless 
tumefaction of the 
right goniac angle 

Painless 
tumefaction of the 
right ramus 

Tumefaction of the 
right maxillary 
hemi-arcade 

Painful tumefaction 
of the right hemi-
face 

 

Clinical features 
Functional 
discomfort 
 
Facial asymmetry 

Painless 

dental mobility and 
displacement -  

 

 - Odynophagia 

- Suspicious sign 
of malignancy 

- Chest pain 

- Odynophagia 

- Chest pain 

Cervico-facial 
ultrasound 

Mixed voluminous 
mandibular mass 
with tissue 
predominance 

  Palpable and 
visible homolateral 
adenopathies 

Submental 
adénomégaly 

Computed 
tomography 

Right latero-
mandibular bone 
lysis 

Mixed-content 
polygeodic 
osteolysis 

Floor and 
pharyngeal 
compression 

Multiple thoracic 
nodules  

Invasions: 

- Submental nodes 

-  Right maxillary 
sinus 

- Right nasal fossa 

- Lungs 

Histology Améloblastoma 
follicular  

Améloblastoma 
follicular  

Améloblastoma 
follicular  

 Améloblastoma 
malignant 

 

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IJO - INTERNATIONAL JOURNAL OF HEALTH SCIENCES AND NURSING  

 ( ISSN: 2814-2098 )                                                                                    Fahasoavana Rohamah Njatosoa¹* 
https://ijojournals.com/                                                     Volume 07 || Issue 02 || February, 2024 || 
 

 

 

The management of our case of ameloblastoma was always conservative surgery by enucleation. 

After diagnosis of malignancy, chemotherapy was received. By the sixth operation, the prognosis 

had become bleak. This was caused by respiratory distress probably secondary to pulmonary 

metastasis. Also, chemotherapy was abandoned due to intolerance linked to poor general 

condition. 

 

DISCUSSION  

Embryologically, during genesis, the epithelial cells of the dental follicle differentiate into several 

cells. The ameloblastic cells in turn disappear physiologically through apoptosis. The persistence 

of these cells is the origin of ameloblastoma, which develops within a fibrous stroma. It is 

characterized by being clinically benign locally, but invasive with a high risk of recurrence [5, 6]. 

Malignant ameloblastoma is defined by the WHO as a tumor that presents architectural features 

of benign ameloblastoma and cytological signs of malignancy in the primary tumor and/or 

metastases [7, 8]. Two percent of ameloblastic tumours are at risk of developing into malignancy 

[9]. It has a characteristic known as "clinical silence", which leads to neglect of patients who only 

come for consultation at an advanced stage of the tumor, causing aesthetic problems and 

functional problems such as dysphagia and dysarthria. It should be noted that the presence of pain 

can be attributed to a possible superinfection or, at worst, to a malignant form of the tumor. 

Our case fits the profile of the type described in the literature. Indeed, our patient belongs to the 

fourth decade of life and is male. The tumor started in the mandible and progressed to the 

maxilla. It is a follicular ameloblastoma recurring after repeated enucleations. However, although 

rare, this tumor has become malignant after several recurrences. The odontostomatologist has an 

important role to play in the early diagnosis of this tumoral pathology. He or she should consider 

it if mobile or displaced teeth are present, or if an osteolytic image is observed incidentally on an 

orthopantomogram or CT scan (see figures B, C, D). 

Our study has been confirmed in the literature, which suggests an age of onset of between 30 and 

50 years [1]. Rammeh reported ameloblastic tumor involvement at the age of 64 [6]. Burcu 

Senguven reported a case in a 9-year-old child. The age of onset is therefore relative [10]. As for 

extension to the maxilla, our study corroborates the study done by Faras in 2017 [11]. However, 

Filizzola and Siriwardena invalidated this result [12, 13].  

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IJO - INTERNATIONAL JOURNAL OF HEALTH SCIENCES AND NURSING  

 ( ISSN: 2814-2098 )                                                                                    Fahasoavana Rohamah Njatosoa¹* 
https://ijojournals.com/                                                     Volume 07 || Issue 02 || February, 2024 || 
 

 

 

Our patient is male. In general, there is no significant difference between the genders, whereas 

the predominance of the male gender is mentioned in a study by Raharison in Antananarivo in 

2004 [14], and by Ahlem in Tunisia in 2015 [15]. However, Faras and Li have demonstrated 

cases to the contrary [11].  

Our histo-pathological finding of follicular type is similar to that of Soyele in Nigeria [16]. In 

contrast, Intapa in Thailand reported a high frequency of the plexiform type in 2017 [17].  

The suggestive clinical signs (facial asymmetry, tooth mobility and displacement) were 

confirmed by Raharison's study [14] (see Figure A). and that of Bel Hadj Hassine in 2017 [9]. 

Painful lymph node metastases appearing in 2009 were also a sign suggestive of malignancy 

according to the literature; as was the presence of chest pain evoked in our study, like the case 

mentioned by Salami in 2018 [18]. Indeed, malignant transformation is rarely evoked by authors, 

and not all multi-recurrences lead to malignancy as in our study and that of Faras in 2017 [11]. 

Treatment of this tumor is purely surgical, with a wide margin of resection. For the malignant 

form, additional cervical lymph node dissection is essential, particularly if lymph node 

metastases are observed on imaging. The enucleation technique caused recurrent tumor 

recurrence in our patient. Ruslin in 2018 [19] and Bal in 2016 [20] also adopted this enucleation 

technique; as did Collins in 2021 [21]. Indeed, the risk of recurrence is high due to the 

impossibility of exeresis margins in enucleation. Interruptive or non-interruptive radical surgery 

is indicated in cases of recurrence and for polygeodic forms. According to Amzerin in 2011 [22] 

and Li in 2019 [23], complementary radiotherapy can give excellent results, and chemotherapy is 

very well indicated in the case of distant (pulmonary) metastasis. as in our case.  

Thus, a healthcare system that promotes in-depth research to find ways of preventing this 

pathology, and a choice of appropriate surgery to limit recurrence, are essential and fundamental. 

The completion of this study would improve the attitude of the two main stakeholders, patient 

and practitioner, towards this pathology. 

CONCLUSION  

This study reports a rare clinical case of a muti-recurrent ameloblastoma at CENHOSOA 

Antananarivo, from its benign to malignant stages. The tumor started in the mandible and 

invaded the maxilla after several recurrences. Although very rarely observed in the literature, 

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IJO - INTERNATIONAL JOURNAL OF HEALTH SCIENCES AND NURSING  

 ( ISSN: 2814-2098 )                                                                                    Fahasoavana Rohamah Njatosoa¹* 
https://ijojournals.com/                                                     Volume 07 || Issue 02 || February, 2024 || 
 

 

 

malignant transformation is possible, as in our case. Adjuvant chemotherapy and radiotherapy are 

then adopted, but the prognosis is guarded at this malignant stage. 

Diagnosis is confirmed anatomically and pathologically. The earlier the diagnosis, the earlier the 

treatment, and therefore the better the prognosis. Treatment should be radical surgery with an 

interrupting or non-interrupting osteotomy to avoid recurrence. 

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IJO - INTERNATIONAL JOURNAL OF HEALTH SCIENCES AND NURSING  

 ( ISSN: 2814-2098 )                                                                                    Fahasoavana Rohamah Njatosoa¹* 
https://ijojournals.com/                                                     Volume 07 || Issue 02 || February, 2024 || 
 

 

 

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IJO - INTERNATIONAL JOURNAL OF HEALTH SCIENCES AND NURSING  

 ( ISSN: 2814-2098 )                                                                                    Fahasoavana Rohamah Njatosoa¹* 
https://ijojournals.com/                                                     Volume 07 || Issue 02 || February, 2024 || 
 

 

 

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IJO - INTERNATIONAL JOURNAL OF HEALTH SCIENCES AND NURSING  

 ( ISSN: 2814-2098 )                                                                                    Fahasoavana Rohamah Njatosoa¹* 
https://ijojournals.com/                                                     Volume 07 || Issue 02 || February, 2024 || 
 

 

 

Metastatic ameloblastoma responding to combination chemotherapy: case report 

and review of the literature. J Med Case Reports. 2011;5:491. 

 

23. Li D, Xu S, Sun M, Qiao L. MAID chemotherapy regimen as a treatment strategy 

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Figure A :asymmetrical patient face on after the other five invasive surgical procedures 
Figure B :polygeodic image with mixed fluid content (tissue and fluid) 
(CENHOSOA, 2002 à 2006) 
 

B A 

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https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3198715/
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 ( ISSN: 2814-2098 )                                                                                   

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Figure C : reflux or the buccal floor and pharynx 
Figure D :Invasion of maxillary sinus and right nasal cavity

(CENHOSOA, 2009 et 2014) 

C 

INTERNATIONAL JOURNAL OF HEALTH SCIENCES AND NURSING 

                                                                   Fahasoavana Rohamah 
                                          Volume 07 || Issue 02 || February

lux or the buccal floor and pharynx with suspicious appearance of malignancy
Invasion of maxillary sinus and right nasal cavity 

D 

AND NURSING  

Fahasoavana Rohamah Njatosoa¹* 
February, 2024 || 

 

suspicious appearance of malignancy 

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