Lingual osteoma: case report and literature review.
ABSTRACT Lingual osteomas are extremely rare. Fewer than 100 cases have been reported in the medical literature. This article reports a case of lingual osteoma, together with a literature review of its clinical features and pathogenesis so as to enhance our understanding of this unusual osseous lesion.
Article: Lingual osteoma: a debatable entity.[Show abstract] [Hide abstract]
ABSTRACT: This article analyzes osseous lesions in the tongue with regard to the clinical and histologic features and discusses the pathogenesis and terminology of these lesions. Thirty-eight cases of lingual osseous lesions were retrieved from the English language literature, and two new cases were added. The lesions had a female predilection, with a female-to-male ratio of 2.3:1. Mean age of the patients at time of diagnosis was 31.4 years. The lesions were located in the posterior third of the dorsum of the tongue in 67.5% of the cases, in the lateral margins in 25%, and in the mid-third of the tongue in 7.5%. Size ranged from 0.5 to 5.0 cm. Duration of the lesions ranged from several months to 50 years. In eight cases, there was information that the lesions had increased in size. Histologically, the lesions appeared a well-circumscribed mass of vital bone under the surface epithelium. The two new cases, when examined by polarized light, showed a mixture of woven and mature bone. The osseous lesions of the tongue represent a heterogeneous group of lesions in which some may represent a developmental malformation, and others may be a reactive lesion after trauma or irritation. Some lesions behave as true neoplasms and others as choristomas. It is suggested that the descriptive term "osseous tumorlike lesion of the tongue" may be more appropriate for this group.Journal of Oral and Maxillofacial Surgery 02/1998; 56(1):9-13; discussion 14. · 1.28 Impact Factor
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ABSTRACT: A choristoma is a tumor-like mass of normal tissue in an "abnormal" location. Lingual osseous choristoma, previously known as osteoma of the tongue, is a rare entity: we found documentation of eight cases at our center during the 10-year period between 1987 and 1996. These cases were analyzed along with 50 others reported in the English language literature between 1913 and 1996. Lingual osseous choristoma frequently occurs during the third and fourth decades of life, and predominantly affects women (81%). Most of the osseous choristomas in our review were located in the posterior third of the tongue, primarily at or close to the foramen cecum or circumvallate papillae (87.9%). While 39.7% of the patients were asymptomatic, the remaining patients complained of symptoms including a lump in the throat (25.8%), dysphagia (6.9%), gagging (5.1%), nausea (3.4%) and irritation (3.4%). Treatment of lingual osseous choristoma consists of simple excision. The tumor's origin has been discussed elsewhere.Ear, nose, & throat journal 05/1998; 77(4):316-8, 320, 325. · 0.88 Impact Factor
- Journal of otolaryngology - head & neck surgery = Le Journal d'oto-rhino-laryngologie et de chirurgie cervico-faciale 05/2009; 38(2):E78-81. · 0.71 Impact Factor
Singapore Med J 2011; 52(10) : e198
C a s e R e p o r t
Lingual osteoma: case report and
Liu J Y, Tan K K H
Lingual osteomas are extremely rare. Fewer
than 100 cases have been reported in the medical
literature. This article reports a case of lingual
osteoma, together with a literature review
of its clinical features and pathogenesis so as
to enhance our understanding of this unusual
Keywords: glossal, lingual, osseous choristoma,
Singapore Med J 2011; 52(10): e198-e200
Osteomas are benign bony neoplasms consisting of
normal mature osseous tissue. They are uncommonly
found in the head and neck region, and rarely occur
in intraoral tissues. Here, we report a case of lingual
osteoma. The original description of this lesion is
attributed to Monserrat in 1913; since then, fewer than
100 cases have been reported.(1) As this is a rare lesion,
its pathogenesis remains a topic of controversy.
A 17-year-old boy presented with a painless mass
measuring 0.5 cm in diameter on his dorsal tongue for
eight years. It had remained the same size throughout
the years. There was no history of trauma, and the
patient had never experienced bleeding from the mass
or dysphagia. Examination revealed a pedunculated
mass covered with normal mucosa on the dorsal tongue,
posterior to the line of circumvallate papillae and to
the right of the foramen caecum. There was no cervical
lymphadenopathy. The thyroid gland was normal. The
lesion was excised under general anaesthesia via a per-
oral approach. It measured 0.7 cm × 0.5 cm × 0.3 cm
and was hard to palpate (Fig. 1). Histology confirmed a
diagnosis of osteoma with dense sclerotic lamellar bone
underneath the tongue epithelium (Fig. 2).
Lingual osteomas are extremely rare. To date, less than
100 cases have been reported since its first description
by Monserrat in 1913.(1) Most occur in women, with a
female to male ratio of 3.25 to 1.00.(2) The age of diagnosis
is 5–73 years, but lingual osteomas are most commonly
seen in the third and fourth decades of life. The most
common location of the lesion is in the posterior third
of the dorsal tongue. In most cases, the patients are
asymptomatic but may occasionally experience a foreign
body sensation in their throat (25.8%), dysphagia (6.9%),
gagging (5.1%), nausea (3.4%) and irritation (3.4%).(3)
Reported lesions are 0.5–2.0 cm in diameter, with most
being pedunculated. All cases have been unilateral,
except for a case of two osteomas, one on either side of
the foramen caecum, reported by Jung.(4) Histologically,
these lesions are well circumscribed and are composed
of mature compact bone with Haversian systems.
Osteoblastic cellular activity has been reported in only
four cases, three of which had a history of the lesions
increasing in size.(5) Treatment in all cases was surgical
excision, with no reported cases of recurrence.
The origin of lingual osteomas is uncertain,
although three pathogenetic mechanisms have been
proposed: post-traumatic reaction,(3) developmental
malformation(1,3,5) and ossification of an embryologic
thyroid remnant.(6,7) The first hypothesis of post-traumatic
reaction suggests that the posterior third of the tongue is
a reactive centre of ossification, since it is susceptible to
trauma and irritation from swallowing.(3) Bony metaplasia
then occurs due to previous injury, haematoma or chronic
inflammation; this is similar to the pathologic process
seen in post-traumatic myositis ossificans.(3) However,
this theory fails to explain why lingual osteomas are all
composed of well-developed mature bone, rather than
an inflammatory irregular bony pattern, which is to be
expected in a traumatic lesion.
The second hypothesis of developmental
malformation was proposed by Monserrat. Lingual
osteomas are thought to be ossified branchial arch
remnants due to their close proximity to the foramen
caecum. Foramen caecum represents the junction between
the first and third branchial arch derivatives, and is also
the site where the second branchial arch disappears.
Osteomas arise from either a congenital remnant of the
second branchial arch or from the first or third arch
remnants.(7) During embryological development, multi-
potential mesenchymal cells may be entrapped in this
Khoo Teck Puat
100 Yishun Central,
Liu JY, MMed,
KK Women’s and
100 Bukit Timah
Tan KKH, MBBS,
Dr Liu Jiaying
Tel: (65) 8121 1670
Fax: (65) 6295 6339
Singapore Med J 2011; 52(10) : e199
Fig. 1 Intraoperative photographs show the osteoma located at the posterior third of the tongue
pre- and post excision.
Fig. 2 Photomicrograph of the specimen shows the nodule
of lamellar bone beneath the attenuated tongue mucosa
(Haematoxylin & eosin, × 40)
region, and by some stimulus in the milieu, give rise
to osteomas over time.(5) This hypothesis explains the
anatomic location but does not account for its predilection
The final theory involves thyroid remnant
calcification.(6,7) It is an attractive theory, as it accounts
for the distinct anatomic relationship between these
osteomas and the foramen caecum, as well as the higher
predilection seen in females. Foramen caecum is also the
site where the thyroid anlage arises and descends into
the neck. Remnants of undescended intraglossal thyroid
tissue can lead to the development of this osseous lesion.(8)
Moreover, lingual thyroid remnants and embryologically
displaced intralaryngeal thyroid tissue are all found
predominantly in females.(9) Metaplastic ossification in
thyroid tissue is also known to occur in colloid goitres
and thyroid cysts. Hence, lingual osteomas may be
ossified derivatives of thyroid remnants.
Apart from aetiology, controversy pertaining to the
correct terminology for this group of lesions also exists.
Krolls et al(10) believed that the term lingual choristoma
was more appropriate than lingual osteoma. They
argued that osteomas are benign, progressively enlarging
neoplasms originating from osteogenic tissue, and thus
closely associated with the skeleton. Lingual osteomas
are neither closely related to the skeleton nor proven to
be of osteogenic origin. Krolls recommended the term
choristoma, as it applies to a cohesive, tumour-like mass
consisting of normal cells in an abnormal location.(10,11) In
spite of the debate over its pathological features, the terms
‘lingual osteoma’ and ‘lingual choristoma’ continue to be
used interchangeably in the medical literature.
In conclusion, lingual osteomas are rare benign
lesions commonly found in the posterior third of the
dorsal tongue, close to the foramen caecum. They
usually occur in females and have varied presentations.
It is suggested that they represent either a developmental
malformation or a post-traumatic reactive lesion.
Definitive management is by surgical excision, with no
cases of recurrence having been reported to date.
1. Monserrat M. Osteome de la langue. Bull Soc Anat 1913; 88:282-3.
2. Liu SC, Su WF, Nieh S, Lin DS, Chu YH. Lingual Osteoma. J Med
Sci 2010; 30:97-9.
Singapore Med J 2011; 52(10) : e200
3. Supiyaphun P, Sampatanakul P, Kerekhanjanarong V,
Chawakitchareon P, Sastarasadhit V. Lingual osseous choristoma:
a study of eight cases and review of the literature. Ear Nose Throat
J 1998; 77:316-8,320,325.
4. Jung G: Ube rein Osteom der Zunge. Beiträge zur Klinischen
Chirugie 1931; 154:167.
5. Vered M, Lustig JP, Buchner A. Lingual osteoma: a debatable
entity. J Oral Maxillofac Sur 1998; 56:9-13.
6. Lee DL, Wong KT, Mak SM, Soo G, Tong MC. Lingual osteoma:
case report and literature review. Arch Otolaryngol Head Neck
Surg 2009; 135:308-10.
7. Jahnke V, Daly JF. Osteoma of the tongue. J Laryngol Otol 1968;
8. Cataldo E, Shklar G, Meyer I. Osteoma of the tongue. Arch
Otolarngol 1967; 85:202-6.
9. Markaki S, Gearty J, Markakis P. Osteoma of the tongue. Br J Oral
Maxillofac Surg 1987; 25:79-82.
10. Krolls SO, Jacoway JR, Alexander WN. Osseous choristomas
(osteomas) of intraoral soft tissues. Oral Surg Oral Med Oral
Pathol 1971; 32:588-95.
11. Chou L, Hansen LS, Daniels TE. Choristomas of the oral cavity: a
review. Oral Surg Oral Med Oral Pathol 1991; 72:584-93.