Resumé
Background: Presence of different cell lines in
the head and neck region allows for the rare
possibility of different neoplastic processes
occurring simultaneously. The occurrence of these
co-existing lesions could just be coincidental but
occasionally may be due to a local or systemic
cause. To the best of the authors’ knowledge, there
are no previous reports of co-existing ossifying
fibroma and pleomorphic adenoma in the head and
neck region in the English literature.
Case presentation: A 39-year old female
presented with a chief complaint of recurrent
swelling on the right side of the neck of nine years’
duration and left maxillary swelling of eight and a
half years duration both of which were spontaneous
in onset. The swellings had slowly increased in
size to the dimension at presentation with no other
significant findings in the medical and family
history. Following other relevant history,
examination and investigations a diagnosis of left
maxillary ossifying fibroma and right recurrent
parotid pleomorphic adenoma was made. Surgical
excision of the two lesions were done with good
outcome. We present a case of concurrent
ossifying fibroma and pleomorphic adenoma
occurring in the head and neck region. We also
discussed the most important features, biologic
behaviour and treatment of these two lesions and
reviewed the literature.
Conclusion: Although the co-existence of these
two lesions presented appear to be coincidental,
however, the possibility of an underlying systemic
link cannot be totally ruled out. Further reporting
of similar cases may help to throw more light for
better understanding of these multiple lesions.
Referencer
Cavaco BM, Barros L, Pannett AA, et al. The
hyperparathyroidism-jaw tumour syndrome in
Portuguese kindred. QJM. 2001 Apr;94(4):213-
doi: 10.1093/qjmed/94.4.213. PMID:
Akcam T, Altug HA, Karakoc O, et al.
Synchronous ossifying fibromas of the jaws: a
review. Oral Surg Oral Med Oral Pathol Oral
Radiol 2012;114 Suppl 5:120-125
Kaya Gª, Özalp Ö and Özbudak ÝH.
Synchronous occurrence of multiple distinct jaw
lesions in Simpson-Golabi-Behmel Syndrome: A
case report. Journal of Stomatology, Oral and
Maxillofacial Surgery. 2019;120(5):483-488. DOI:
1016/j.jormas.2018.12.001.
Khan SA, Sharma NK, Raj V and Sethi T.
Ossifying fibroma of maxilla in a male child: report
of a case and review of the literature. Natl J
Maxillofac Surg. 2011; 2(1): 73-79.
Levine P, Wiggins R, Archibald R and Britt R.
Ossifying fibroma of the head and neck:
Involvement of the temporal bone, an unusual
and challenging site. Laryngoscope 1981;91: 720-
Çakir B and Karadayi N. Ossifying fibroma in
the nasopharynx a case report. Clin Imaging.
;15: 290–292. DOI: 10.1016/0899-
(91)90122-C.
Thomas GK and Kasper KA. Ossifying fibroma
of the frontal bone. Arch Otolaryngol 1966; 83:
-46.
Mitrani M, Remsen K, Lawson W and Biller H.
Giant ossifying fibroma of the paranasal sinuses.
Ear Nose Throat J 1988; 67: 186-192
Gatt WM and Cronin JJ. Ossifying fibroma of
the sphenoid sinus. EENT Monthly 1975; 54: 67-
Margo C, Weiss A and Habal M. Psammomatioid
ossifying fibroma. Arch Ophthalmol
;104:1347-1351.
Bertolini F, Caradonna L, Bianchi B and Sesenna
E. Multiple ossifying fibroma of the jaws: a case
report. J Oral Maxillofac Surg. 2002; 60: 225–229.
Regezzi JA and Sciubba JJ. Oral Pathology. 3rd
ed. Philadelphia: W.B. Saunders; 1999. p. 357-
Neville BW, Damm DD, Allen CM and Bouquot
JE. Oral and maxillofacial pathology. 3rd ed.
Philadelphia: Saunders; 2009. p. 647.
Titinchi F and Morkel J. Ossifying fibroma:
analysis of treatment methods and recurrence
patterns. J Oral Maxillofac Surg. 2016; 74: 2409-
Rajendran S and Sivapathasundaram S. Shafer’s
textbook of oral pathology 6th ed. New Delhi,
Elsevier, 2009. p. 219224
Cawson RA and Odell EW. Cawson’s essentials
of oral pathology and oral medicine. 7th ed.
Edinburgh, Churchill Livingstone. 2008. p. 247-
Clark K. Etiology and familial inheritance of
pleomorphic adenomas Dentistry 3000. 2017;
(1). 4 pages. doi:10.5195/d3000.2017.70
Available from: https://www.researchgate.net/
p ub lica tion/ 31 75 94 60 8_ E tiology_ and
_familial_inheritance_of _pleomorphic_
adenomas [accessed Oct 02 2020].
Whatley WS, Thompson JW and Rao B. Salivary
gland tumours in survivors of childhood cancer.
Otolaryngol Head Neck Surg. 2006; 134(3): 385-
doi: 10.1016/j.otohns.2005.10.022. PMID:
Martinelli M, Martini F, Rinaldi E, et al.
Simian Virus 40 Sequences and expression of
the viral large T-antigen oncoprotein in Human
Pleomorphic Adenomas of Parotid Glands.
; 161(4): 1127–1133. PMID: PMC1867276
Sadetzki S, Chetrit A, Jarus-Hakak A, et al.
Cellular phone use and risk of benign and
malignant parotid gland tumors—a nationwide
case-control study. Am J Epidemiol.
;167(4):457-467. doi: 10.1093/aje/kwm325.
Epub 2007 Dec 6. PMID: 18063591.
Sunil S and Gopakumar D. Pleomorphic adenoma.
A case report and review of literature. Int. J.
Odontostomat. 2013; 7(2):171-174.
Naeim F. Forsberg MI. Waisman J and Coulson
WF. Mixed tumours of the salivary glands: growth
pattern and recurrence. Arch Pathol Lab Med.
; 100: 271–275
Dulguerov P, Todic J, Pusztaszeri M and
Alotaibi NH. Why Do Parotid Pleomorphic
Adenomas Recur? A Systematic Review of
Pathological and Surgical Variables. Front. Surg.
; 4:26. doi: 10.3389/fsurg.2017.00026
Andreasen S, Therkildsen MH, Bjorndal K and
Homoe P. Pleomorphic adenoma of the parotid
gland 1985-2010: a Danish nationwide study of
incidence, recurrence rate, and malignant
transformation. Head Neck. 2016; 38 Suppl 1:
E1364- E1369
Hua Yan, Kai Luo, BaoLong Liu and Jiamin Kang.
A solitary fibrous tumour with concurrent
meningioma at the same site: A case report and
review of the literature. Oncol Lett.
;11:3655–3659. doi: 10.3892/ol.2016.4486.
Murgod S, Girish HC, Savita JK and Varsha VK.
Concurrent central odontogenic fibroma and
dentigerous cyst in the maxilla: A rare case report.
J Oral Maxillofac Pathol. 2017; 21: 149–153.
Munde A, Modi P, Karle R, Wankhede P and
Shoeb S. Nonsyndromic Synchronous Multifocal
Central Giant Cell Granulomas of the
Maxillofacial Region: Report of a Case. Journal
of Dentistry (Tehran, Iran). 2015;12(2):157-162.
Haven CJ, Wong FK, van Dam EW, et al. A
genotypic and histopathological study of a large
Dutch kindred with hyperparathyroidism-jaw
tumour syndrome. J Clin Endocrinol Metab.
;85(4):1449-1454.
Yamashita Y, Akiyama T, Mizusawa N, Yoshimoto
K and Goto MA. A case of hyperparathyroidismjaw tumour syndrome found in the treatment of
an ossifying fibroma in the maxillary bone. Int J
Oral Maxillofac Surg. 2007;36:365–369.
Bradley ES and Leake D. Ossifying fibroma
involving the maxilla and mandible. Oral Surg Oral
Med Oral Pathol. 1968; 26: 605–614.
Yih W, Pederson GT and Bartley MH. Multiple
familial ossifying fibromas/ : Relationship to other
osseous lesions of the jaws. Oral Surg Oral Med
Oral Pathol. 1989; 68: 754–758.
Wang T, Zhang R, Wang L, et al. Two cases of
multiple ossifying fibromas in the jaws. Diagn
Pathol 2014; 9(1): 75. https://doi.org/10.1186/
-1596-9-75
Bernard EK, Akama MK and Vilembwa A.
Surgical management of aggressive synchronous
jaw central giant cell granuloma/ : case report.
East Afr Med J. 2012; 89: 282–284.
Horisk N, Stephenson E, Sayers C and Reid J.
Incidental finding of synchronous pleomorphic
salivary adenoma and Warthin’s tumour within a
TO Aladelusi, OO Gbolahan and AO Fasola
parotid gland. BMJ Case Rep. 2019; 12: e228675.
DOI: 10.1136/bcr-2018-228675.
Filippo R, Angelo P, Flavia O, et al. A Rare Case
of Synchronous Bilateral Pleomorphic Adenoma
of the Parotid Gland. Case Rep Clin Med. 2014;
: 387–391.
Siar CH and Ng K. Combined ameloblastoma
and odontogenic keratocyst’ or ‘keratinising
ameloblastoma. Br J Oral Maxillofac Surg
;31:183–186.
Fregnani ER, da Cruz Perez DE, Soares FA and
Alves FA. Synchronous ameloblastoma and
orthokeratinized odontogenic cyst of the mandible.
J Oral Pathol Med. 2006;35(9):573-575. doi:
1111/j.1600-0714.2006.00445.x. PMID: