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J Clin Exp Dent. 2010;2(4):e204-6.	         		                       	             		                                Desmoplastic Ameloblastoma.




Journal section: Oral Medicine and Pathology                                         			                                     doi:10.4317/jced.2.e204
Publication Types: Case report


                       Desmoplastic ameloblastoma of maxilla- a case report



Lalitkumar Gade 1, Sangeeta Patankar 2, Komal Khot 3, Sheetal Korde 4, Sheeba Alex 5

1
  P.G. student, Y.M.T. Dental College & Hospital, Navi Mumbai, India.
2
  Professor. & H.O.D., Y.M.T. Dental College & Hospital, Navi Mumbai, India.
3
  Professor, Y.M.T. Dental College & Hospital, Navi Mumbai, India.
4
  Senior Lecturer, Y.M.T. Dental College & Hospital, Navi Mumbai, India.
5
  Reader, Y.M.T. Dental College & Hospital, Navi Mumbai, India.




Correspondence:
Department of Oral and Maxillofacial Pathology.
YMT Dental College & Hospital, Industrial area, Sect-4
Navi Mumbai, Maharshtra, India.
E-mail address- drlalitkumargade@indiatimes.com




Received: 23/06/2010
Accepted: 25/07/2010




                                                            Gade L, Patankar S, Khot K, Korde S, Alex S. Desmoplastic ameloblasto-
                                                            ma of maxilla- a case report. J Clin Exp Dent. 2010;2(4):e204-6.
                                                            http://www.medicinaoral.com/odo/volumenes/v2i4/jcedv2i4p204.pdf

                                                             Article Number: 50342       http://www.medicinaoral.com/odo/indice.htm
                                                             © Medicina Oral S. L. C.I.F. B 96689336 - eISSN: 1989-5488
                                                             eMail: jced@jced.es




    Abstract
    Ameloblastoma is the most common neoplasm affecting the jaws, arising from the odontogenic epithelium. Despite
    its locally aggressive nature, it is considered to be benign. The chief histopathological variants of ameloblastoma
    are the follicular and plexiform types, followed by the acanthomatous and granular cell types. Uncommon variants
    include desmoplastic, basal cell, clear cell ameloblastoma, keratoameloblastoma and papilliferous keratoamelo-
    blastoma. When the desmoplastic type co-exists with other types, it is called as “hybrid” ameloblastoma.
    There are significant anatomic, histopathological and radiological differences between desmoplastic ameloblasto-
    ma and the classical type. The purpose of this article is to report a case and to review the relevant literature, empha-
    sizing peculiar aspects of this unusual lesion.

    Key words: Odontogenic neoplasm, desmoplastic ameloblastoma, desmoplasia.



                                                                e204
J Clin Exp Dent. 2010;2(4):e204-6.	            		                  	          		                         Desmoplastic Ameloblastoma.




Introduction
Ameloblastoma is the most common benign odontoge-
nic neoplasm of epithelial origin occurring in the oral
cavity, mostly in third molar region and ascending ramus
of a mandible(1). It is prevalent in the third and fourth
decade equally affecting both genders with no predilec-
tion for any race.
Desmoplastic ameloblastoma is an aggressive variant
of ameloblastoma which needs special attention by the
clinician, owing to its locally destructive nature. It repre-
sents 4-5% of all ameloblastomas(2). On a radiograph,                  Fig. 2. Mixed lesion in upper left premolar and molar
this lesion rarely suggests the diagnosis ameloblastoma.               region.
Many a times it is similar to a fibro-osseous lesion due to
                                                                  Histopathologically the section showed strands, islands
its mixed radiolucent-radiopaque appearance.
                                                                  and a few small sheaths of odontogenic epithelium in a
                                                                  dense collagenous fibrous tissue stroma with stretched
Case report
                                                                  out kite-tail appearance (Fig.3). Under high power folli-
A 35-year-old female reported to the Yerala Medical
                                                                  cles with typical peripheral pre-ameloblast like cells and
Trust’s Dental College and Hospital with a chief com-
                                                                  central stellate reticulum like cells were seen
plaint of swelling in the left maxillary anterior region.
                                                                  scattered in the connective tissue stroma(Fig.3). A his-
The swelling had been present since 1 year, growing
                                                                  tological diagnosis of desmoplastic ameloblastoma was
slowly and was not associated with pain. She also gave
                                                                  made with respect to the above findings.
history of traumatic extraction of 26 about 1 and ½ year
ago. A few months post extraction, she noticed swelling
in the maxillary anterior region.
Extraoral examination reveled an infraorbital swelling
extending from ala of nose towards angle of mouth cau-
sing facial asymmetry on left side of face. On intra-oral
examination, a well-defined swelling of 4x3 cm was seen
in the upper left posterior region extending from 23 to 26
obliterating the vestibule. Swelling was oval, firm, non-
tender, non-fluctuant and was attached to the underlying          Fig. 3. Right side10X view showing Desmoplastic stroma with
structures (Fig.1). The overlying mucosa was normal.              odontogenic follicles having kite-tail appearance. Left side 40X view
                                                                  showing odontogenic follicle with peripheral. preameloblast like
                                                                  cells.
                                                                                   		
                                                                  Discussion
                                                                  Eversole et al (1) in 1984 were the first to describe three
                                                                  cases of desmoplastic ameloblastoma. Later, Waldron
                                                                  and El Mofty(3) reported additional 14 cases . This
                                                                  unusual variant is characterized histologically by exten-
                                                                  sive stromal collagenation or desmoplasia with small
                                                                  nests and strands of odontogenic epithelium.
                                                                  Immunohistochemical studies suggest that the desmo-
                                                                  plasia originates de novo from synthesis of extracellular
                                                                  matrix proteins (4).
   Fig.1. Swelling in upper left posterior region.
                                                                  Phillipson, et al.(5) reported 100 cases of desmoplastic
                                                                  ameloblastoma from 1984 to 2001. Till date 145 cases
Radiographically intraoral periapical, occlusal X-ray             have been reported in Japanese, Chinese, Malaysian,
and orthopantomography showed mixed lesion with ra-               Western, and African population with very few cases
diopaque flecks, diffuse lesion in left maxillary premolar        described in the Indian population(5,6). The knowledge
and molar regions with retained root piece of 26 (Fig.2).         regarding the clinico-radiological presentation and pa-
All laboratory investigations were carried out before the         thology of desmoplastic ameloblastomas has led to its
surgical procedure and were found to be within normal             categorization as a distinct variant of ameloblastoma in
limits.                                                           the World Health Organization classification of odonto-
Excised gross specimen was hard in consistency with an            genic tumors in 2005.
irregular surface.
                                                           e205
J Clin Exp Dent. 2010;2(4):e204-6.	            		                            	         		    Desmoplastic Ameloblastoma.




Desmoplastic ameloblastoma is a rare and infrequent                         1993;76:525-9.
tumor characterized histologically by marked stromal
desmoplasia. It is common in the third to fifth decade
with a male predominance.
More than 70% of the desmoplastic ameloblastoma cases
are seen in the anterior region of the maxilla, as against
conventional ameloblastomas, which are usually found
in the mandibular posterior region (7). In 15 cases of
desmoplastic ameloblastoma analyzed, 11 lesions were
located in the maxilla (73%) and only 4 in the mandible
(27%), with most cases reaching anterior region. Howe-
ver, in another study of 10 cases of desmoplastic ame-
loblastoma reported by Kishino et al.(8) in 2001, 40%
involved the anterior region of the maxilla and 60% in
the posterior region of the mandible. Our case findings
are with Takata et al (7).
Radiographically, desmoplastic ameloblastoma may
show either a multilocular, mixed radiolucent-radiopa-
que appearance or multilocular appearance of
minute flecks of bone similar to that seen in benign fi-
bro-osseous lesions (9). As per the reviewed cases, a ma-
jority of desmoplastic ameloblastomas showed mixed
radiolucent-radiopaque appearance which can be due to
infiltrative growth pattern of tumor cells into surroun-
ding marrow spaces and simultaneous vigorous osteo-
blastic activity leading to number of bony flecks (8).
The histopathological diagnostic criteria have been es-
tablished by Kishino et al. (8) such a lesion is characte-
rized by small nests and cords of odontogenic tumoral
epithelium, organized in an abundant densely collage-
nized stroma which makes the tumoral islands compres-
sed. Extensive collagenization, also called desmoplasia,
is the hallmark of this lesion.

References
1. Yazdi I, Seyedmajidi M, Foroughi R. Desmoplastic ameloblastoma
(a hybrid variant): report of a case and review of the literature. Arch
Iran Med. 2009;12:304-8.
2. Manuel S, Simon D, Rajendran R, Naik BR. Desmoplastic amelo-
blastoma: a case report. J Oral Maxillofac Surg. 2002;60:1186-8.
3. Waldron CA, el-Mofty SK. A histopathologic study of 116 amelo-
blastomas with special reference to the desmoplastic variant. Oral Surg
Oral Med Oral Pathol. 1987;63:441-51.
4. Philipsen HP, Reichart PA, Takata T. Desmoplastic ameloblasto-
ma (including “hybrid” lesion of ameloblastoma). Biological profi-
le based on 100 cases from the literature and own files. Oral Oncol.
2001;37:455-60.
5. Desai H, Sood R, Shah R, Cawda J, Pandya H. Desmoplastic ame-
loblastoma: report of a unique case and review of literature. Indian J
Dent Res. 2006;17:45-9.
6. Sivapathasundharam B, Einstein A, Syed RI. Desmoplastic ame-
loblastoma in Indians: report of five cases and review of literature.
Indian J Dent Res. 2007;18:218-21.
7. Takata T, Miyauchi M, Ogawa I, Zhao M, Kudo Y, Sato S, et al.
So-called ‘hybrid’ lesion of desmoplastic and conventional ame-
loblastoma: report of a case and review of the literature. Pathol Int.
1999;49:1014-8.
8. Kishino M, Murakami S, Fukuda Y, Ishida T. Pathology of the des-
moplastic ameloblastoma. J Oral Pathol Med. 2001;30:35-40.
9. Kaffe I, Buchner A, Taicher S. Radiologic features of desmo-
plastic variant of ameloblastoma. Oral Surg Oral Med Oral Pathol.
                                                                          e206

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Desmoplastic ameloblastoma of maxilla a case report

  • 1. J Clin Exp Dent. 2010;2(4):e204-6. Desmoplastic Ameloblastoma. Journal section: Oral Medicine and Pathology doi:10.4317/jced.2.e204 Publication Types: Case report Desmoplastic ameloblastoma of maxilla- a case report Lalitkumar Gade 1, Sangeeta Patankar 2, Komal Khot 3, Sheetal Korde 4, Sheeba Alex 5 1 P.G. student, Y.M.T. Dental College & Hospital, Navi Mumbai, India. 2 Professor. & H.O.D., Y.M.T. Dental College & Hospital, Navi Mumbai, India. 3 Professor, Y.M.T. Dental College & Hospital, Navi Mumbai, India. 4 Senior Lecturer, Y.M.T. Dental College & Hospital, Navi Mumbai, India. 5 Reader, Y.M.T. Dental College & Hospital, Navi Mumbai, India. Correspondence: Department of Oral and Maxillofacial Pathology. YMT Dental College & Hospital, Industrial area, Sect-4 Navi Mumbai, Maharshtra, India. E-mail address- drlalitkumargade@indiatimes.com Received: 23/06/2010 Accepted: 25/07/2010 Gade L, Patankar S, Khot K, Korde S, Alex S. Desmoplastic ameloblasto- ma of maxilla- a case report. J Clin Exp Dent. 2010;2(4):e204-6. http://www.medicinaoral.com/odo/volumenes/v2i4/jcedv2i4p204.pdf Article Number: 50342 http://www.medicinaoral.com/odo/indice.htm © Medicina Oral S. L. C.I.F. B 96689336 - eISSN: 1989-5488 eMail: jced@jced.es Abstract Ameloblastoma is the most common neoplasm affecting the jaws, arising from the odontogenic epithelium. Despite its locally aggressive nature, it is considered to be benign. The chief histopathological variants of ameloblastoma are the follicular and plexiform types, followed by the acanthomatous and granular cell types. Uncommon variants include desmoplastic, basal cell, clear cell ameloblastoma, keratoameloblastoma and papilliferous keratoamelo- blastoma. When the desmoplastic type co-exists with other types, it is called as “hybrid” ameloblastoma. There are significant anatomic, histopathological and radiological differences between desmoplastic ameloblasto- ma and the classical type. The purpose of this article is to report a case and to review the relevant literature, empha- sizing peculiar aspects of this unusual lesion. Key words: Odontogenic neoplasm, desmoplastic ameloblastoma, desmoplasia. e204
  • 2. J Clin Exp Dent. 2010;2(4):e204-6. Desmoplastic Ameloblastoma. Introduction Ameloblastoma is the most common benign odontoge- nic neoplasm of epithelial origin occurring in the oral cavity, mostly in third molar region and ascending ramus of a mandible(1). It is prevalent in the third and fourth decade equally affecting both genders with no predilec- tion for any race. Desmoplastic ameloblastoma is an aggressive variant of ameloblastoma which needs special attention by the clinician, owing to its locally destructive nature. It repre- sents 4-5% of all ameloblastomas(2). On a radiograph, Fig. 2. Mixed lesion in upper left premolar and molar this lesion rarely suggests the diagnosis ameloblastoma. region. Many a times it is similar to a fibro-osseous lesion due to Histopathologically the section showed strands, islands its mixed radiolucent-radiopaque appearance. and a few small sheaths of odontogenic epithelium in a dense collagenous fibrous tissue stroma with stretched Case report out kite-tail appearance (Fig.3). Under high power folli- A 35-year-old female reported to the Yerala Medical cles with typical peripheral pre-ameloblast like cells and Trust’s Dental College and Hospital with a chief com- central stellate reticulum like cells were seen plaint of swelling in the left maxillary anterior region. scattered in the connective tissue stroma(Fig.3). A his- The swelling had been present since 1 year, growing tological diagnosis of desmoplastic ameloblastoma was slowly and was not associated with pain. She also gave made with respect to the above findings. history of traumatic extraction of 26 about 1 and ½ year ago. A few months post extraction, she noticed swelling in the maxillary anterior region. Extraoral examination reveled an infraorbital swelling extending from ala of nose towards angle of mouth cau- sing facial asymmetry on left side of face. On intra-oral examination, a well-defined swelling of 4x3 cm was seen in the upper left posterior region extending from 23 to 26 obliterating the vestibule. Swelling was oval, firm, non- tender, non-fluctuant and was attached to the underlying Fig. 3. Right side10X view showing Desmoplastic stroma with structures (Fig.1). The overlying mucosa was normal. odontogenic follicles having kite-tail appearance. Left side 40X view showing odontogenic follicle with peripheral. preameloblast like cells. Discussion Eversole et al (1) in 1984 were the first to describe three cases of desmoplastic ameloblastoma. Later, Waldron and El Mofty(3) reported additional 14 cases . This unusual variant is characterized histologically by exten- sive stromal collagenation or desmoplasia with small nests and strands of odontogenic epithelium. Immunohistochemical studies suggest that the desmo- plasia originates de novo from synthesis of extracellular matrix proteins (4). Fig.1. Swelling in upper left posterior region. Phillipson, et al.(5) reported 100 cases of desmoplastic ameloblastoma from 1984 to 2001. Till date 145 cases Radiographically intraoral periapical, occlusal X-ray have been reported in Japanese, Chinese, Malaysian, and orthopantomography showed mixed lesion with ra- Western, and African population with very few cases diopaque flecks, diffuse lesion in left maxillary premolar described in the Indian population(5,6). The knowledge and molar regions with retained root piece of 26 (Fig.2). regarding the clinico-radiological presentation and pa- All laboratory investigations were carried out before the thology of desmoplastic ameloblastomas has led to its surgical procedure and were found to be within normal categorization as a distinct variant of ameloblastoma in limits. the World Health Organization classification of odonto- Excised gross specimen was hard in consistency with an genic tumors in 2005. irregular surface. e205
  • 3. J Clin Exp Dent. 2010;2(4):e204-6. Desmoplastic Ameloblastoma. Desmoplastic ameloblastoma is a rare and infrequent 1993;76:525-9. tumor characterized histologically by marked stromal desmoplasia. It is common in the third to fifth decade with a male predominance. More than 70% of the desmoplastic ameloblastoma cases are seen in the anterior region of the maxilla, as against conventional ameloblastomas, which are usually found in the mandibular posterior region (7). In 15 cases of desmoplastic ameloblastoma analyzed, 11 lesions were located in the maxilla (73%) and only 4 in the mandible (27%), with most cases reaching anterior region. Howe- ver, in another study of 10 cases of desmoplastic ame- loblastoma reported by Kishino et al.(8) in 2001, 40% involved the anterior region of the maxilla and 60% in the posterior region of the mandible. Our case findings are with Takata et al (7). Radiographically, desmoplastic ameloblastoma may show either a multilocular, mixed radiolucent-radiopa- que appearance or multilocular appearance of minute flecks of bone similar to that seen in benign fi- bro-osseous lesions (9). As per the reviewed cases, a ma- jority of desmoplastic ameloblastomas showed mixed radiolucent-radiopaque appearance which can be due to infiltrative growth pattern of tumor cells into surroun- ding marrow spaces and simultaneous vigorous osteo- blastic activity leading to number of bony flecks (8). The histopathological diagnostic criteria have been es- tablished by Kishino et al. (8) such a lesion is characte- rized by small nests and cords of odontogenic tumoral epithelium, organized in an abundant densely collage- nized stroma which makes the tumoral islands compres- sed. Extensive collagenization, also called desmoplasia, is the hallmark of this lesion. References 1. Yazdi I, Seyedmajidi M, Foroughi R. Desmoplastic ameloblastoma (a hybrid variant): report of a case and review of the literature. Arch Iran Med. 2009;12:304-8. 2. Manuel S, Simon D, Rajendran R, Naik BR. Desmoplastic amelo- blastoma: a case report. J Oral Maxillofac Surg. 2002;60:1186-8. 3. Waldron CA, el-Mofty SK. A histopathologic study of 116 amelo- blastomas with special reference to the desmoplastic variant. Oral Surg Oral Med Oral Pathol. 1987;63:441-51. 4. Philipsen HP, Reichart PA, Takata T. Desmoplastic ameloblasto- ma (including “hybrid” lesion of ameloblastoma). Biological profi- le based on 100 cases from the literature and own files. Oral Oncol. 2001;37:455-60. 5. Desai H, Sood R, Shah R, Cawda J, Pandya H. Desmoplastic ame- loblastoma: report of a unique case and review of literature. Indian J Dent Res. 2006;17:45-9. 6. Sivapathasundharam B, Einstein A, Syed RI. Desmoplastic ame- loblastoma in Indians: report of five cases and review of literature. Indian J Dent Res. 2007;18:218-21. 7. Takata T, Miyauchi M, Ogawa I, Zhao M, Kudo Y, Sato S, et al. So-called ‘hybrid’ lesion of desmoplastic and conventional ame- loblastoma: report of a case and review of the literature. Pathol Int. 1999;49:1014-8. 8. Kishino M, Murakami S, Fukuda Y, Ishida T. Pathology of the des- moplastic ameloblastoma. J Oral Pathol Med. 2001;30:35-40. 9. Kaffe I, Buchner A, Taicher S. Radiologic features of desmo- plastic variant of ameloblastoma. Oral Surg Oral Med Oral Pathol. e206