ABSTRACT

Soft tissue osteoma is a rare entity having a strongpredilection for the head and neck region, mainly pos-terior region of the tongue. The so-called lingual osteo-ma is mostly manifested as an asymptomatic exophyticlesion. It can be diagnosed by physical or radiologicalexaminations.

We represent a patient with undiagnosed lingual osteo-ma, accidentally detected during laryngoscopy for intu-bation for a gynecologic surgery. General anesthesiawas planned for a 52 year-old undergoing gynecologicsurgery. Before surgery a laryngoscopy was performedfor intubation. During this procedure a pedunculatedmass was seen in the posterior region of the tongue.Although the pathogenesis and terminology is contro-versial, surgical excision is the preferred treatmentmodality. We aimed to present an osseos lesion intongue, to review the literature in regard to relevantclinical, histological features and to discuss the patho-genesis and terminology involved.

Key words: Tongue, osteoma, intubation

ÖZET

Yumuflak doku osteomu ender bir durum olup, bafll›cadilin arka k›sm› olmak üzere bafl-boyun bölgesindegörülmeye güçlü bir yatk›nl›k gösterir. Lingual osteomad› verilen bu durum s›kl›kla yak›nmas›z d›fla do¤rugeliflmifl bir lezyon olarak görülür. Tan›s› fizik muayeneya da görüntüleme yöntemleriyle konur.

Sundu¤umuz hasta daha önce tan› almam›fl, jinekolojikameliyat öncesi entübasyon s›ras›nda tesadüfen farkedilen bir lingual osteom olgusudur. Elli iki yafl›ndakad›n hastan›n jinekolojik cerrahi için genel anestezialmas› planlanm›flt›r. Cerrahi öncesi entübasyon içinlaringoskopi yap›lmas› esnas›nda dilin arka k›sm›ndasapl› bir kitle görülmüfltür. Patogenez ve terminolojikonusu tart›flmal› olsa da, tedavi yönteminde seçim cer-rahidir. Bu makaledeki amac›m›z dildeki kemiksi birlezyonu sunarken, klinik ve histolojik özellikleridikkate olarak literatürü gözden geçirmek ve patogene-zle birlikte terminolojiyi tart›flmakt›r.

Anahtar sözcükler: Dil, osteom, entübasyon

INTRODUCTION

Extraosseous osteoma in the tongue is a ra-re entity with a typical location in the posteriorthird close to foramen caecum and posterior tocircumvallate papillae but it also can be locatedin the lateral margins and in the mid-third of the

tongue. It can be pedunculated or sessile (1).The patients are usually young women having ahard mass arising from posterior part of the ton-gue with no symptoms. Difficulty in swallowingis the most common complaint in symptomaticcases. Osteomas are two to three times moreprevalent in women than in men (1,2,3). Altho-ugh reports about patients having lingual osteo-mas at the time of birth and in early childhood

An unexpected diagnosis during laryngeal intubation: osseous polypoid lesion of thetongue: osteoma or choristoma?

Laringeal entübasyon s›ras›nda tesadüfi tan›: lingualosseöz polipoid lezyon: osteom veya koristom?

Ertap AKO⁄LU1, Esin AT‹K2, Sinem KARAZ‹NC‹R3, fiemsettin OKUYUCU1, Ça¤la ÖZBAKIfi4,Ali BALCI3, Ramazan GÜMÜfi1, fiafak DA⁄LI1

Mustafa Kemal University Tayfur Ata Sokmen School of Medicine, Departments of Otolaryngology Head and Neck Surgery1,Pathology2, Radiology3, and Anesthesiology and Reanimation4, HATAY

Corresponding author: fiemsettin Okuyucu, Mustafa KemalÜniversitesi Araflt›rma Hastanesi 31100, Antakya, Hatay

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have been cited in literature, the average agerange is the third and fourth decades (4). Thepathogenesis and terminology of the lesion re-mains uncertain (3,4). According to our know-ledge this is the first case that probable diagno-sis is established during intubation.

CASE REPORT

General anesthesia was planned for a 52year-old undergoing gynecologic surgery. Befo-re surgery a laryngoscopy was performed for in-tubation. During this procedure a pedunculatedmass was seen in the posterior region of the ton-gue that didn't restrict intubation. After the pati-ent had recovered from her gynecologic comp-laints, a physical examination was made by anotolaryngologist. The patient was aware of thismass with no symptoms so the duration of thelesion is unknown. Examination showed a4x2x2 cm firm bony hard swelling with a nor-mal color and aspect with the overlying mucosapedunculated to the dorsum of the tongue justleft to the foramen caecum (Figure 1). Compu-terized tomographic (CT) scans revealed a den-se lesion consisting of hypodense areas with awell-demarcated border at the base of the ton-gue (Figure 2). She underwent a surgical excisi-on under general anesthesia with a provisional

diagnosis of osteoma.Macroscopic examination showed a yel-

low-beige colored, polypoid osseous mass me-asuring 4x2x2 cm. After decalcification, thesamples underwent routine tissue processingand the paraffin blocks were prepared. Five mic-ron sections retrieved from the paraffin blocksstained with hematoxylin eosin were examinedunder light microscope.Figure 1. Lingual osteoma on the dorsum of the tongue.

Figure 2. CT image revealed a well-demarcated hyperdenselesion with hypodense areas at the base of the tongue.

Figure 3. Dense lamellar bone including fibroadipose tissuewith rare osteoblasts (HE x400).

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An unexpected diagnosis during laryngeal intubation: osseous polypoid lesion of the tongue: osteoma or choristoma?

Microscopic examination revealed mostlydense lamellar osteoid tissue with rare osteob-lasts, fibroadipose tissue and bone marrow(Figure 3-4).

DISCUSSION

There are different theories related to thepathogenesis of the osseous lesions of the ton-gue. Two main proposals are embryologic deve-lopmental malformation and reactive or posttra-umatic theory. Monserrat was the first who exp-lained the embryologic developmental theoryrelated to osseous lesions in the tongue (3). Therational of his theory depends on the fact thattwo thirds of the tongue derives and the posteri-or third originates from the third branchial arch.First and third branchial arches meet in the fora-men caecum. It is known that bony structuressuch as malleus and incus develop from the firstand hyoid bone from the third branchial archthrough enchondral ossification. Therefore, the-se branchial arches have the capability to to enc-lave mesenchymal pluripotent cells for the sub-sequent development of an osseous lesion in thetongue. Cataldo et al. and Jankhe and Daly haveproposed an alternative developmental theory,which is associated with the remnants of thyro-id tissue (5,6). In embryologic life the anlage ofthe thyroid gland descends from the foramencaecum to the neck. They suggested that the pri-mordial endodermal or differentiated thyroid

parenchymal cells developing from undescen-ded intraglossal thyroid remnants can produceunusual osseous proliferation later in life (3).

The second theory suggests that osseouslesions of the tongue represent a reactive orposttraumatic center for ossification. 'Myositisossificans' is the term used for this kind of lesi-ons in other muscles of the body. Chronic inf-lammation due to trauma or irritation is a com-mon finding at the posterior third of the tongue.Inflammatory and posttraumatic lesions have ir-regular areas of ossification, with neither haver-sian systems nor normal bone architecture (3).Controversially osseous lesions in the tongueare composed of well-developed mature bonethat could not be associated with trauma. On theother hand two cases was cited in literature thatshowed diffuse foci of ossification without awell-circumscribed osseous lesion (7,8). Reacti-ve responses of the tissue to trauma and irritati-on may differ from chronic inflammation to me-taplasia, and also osteoma which might be themost mature stage of metaplastic process (9).

Our case was reported as osteoma. Diffe-rential diagnosis includes sialolithiasis, lipomawith osseous metaplasia, osteo-cartilaginouschoristoma, metastatic osteosarcoma, liposarco-ma with metaplasia, and post-traumatic chondri-fication (9,10,11).

There is not a consensus on the termino-logy for these lesions. Osteoma defines a be-nign, progressively enlarging neoplasm of boneoriginating from osteogenic tissue and it is clo-sely associated with the skeleton. Lingual osteo-ma doesn't fulfill these criteria because the ton-gue is not associated with skeleton and it is notan osteogenic tissue. “Lingual choristoma” issuggested as an alternative term because it des-cribes a cohesive tumor like mass consisting ofnormal cells in an abnormal location. As someof the lesions have been reported to increase insize, the term choristoma fails to fit these defini-tions. Also not widely used 'osseous tumor likelesions of the tongue' is a descriptive term forthis kind of lesions (12).

Figure 4. Dense lamellar osteid tissue (HE x40).

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Even it may be asymptomatic, surgical ex-cision is the preferred treatment modality. His-topathologic examinations are necessary for thediagnosis. After removing the lesion, recurrenceis not expected.

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3. Vered M, Lustig JP, Buchner A. Lingual osteoma: adebatable entity. J Oral Maxillofac Surg 1998;56:9-13.

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G,Chauderhy AP. Lingual osseous choristoma. J OralMed 1983;38:87-89.

8. Maqbool M, Ahmad R, Ahmad R. Osteoma of the ton-gue. Indian Pediatr 1992;29:1429-1431.

9. Kransdorf MJ, Meis JM. Extraskeletal osseous and car-tilaginous tumors of the extremities. Radiographics1993;13:853-884.

10. Turkoz H K, Varnali Y, Comunoglu C. A case of oste-olipoma of the head and neck area. Kulak Burun BogazIhtis Derg 2004;13:84-86.

11. Piatelli A, Fioroni M, Iezzi G, Rubini C. Osteolipomaof the tongue. Oral Oncology 2001;37:468-470.

12. Chou L, Hansen LS, Daniels TE. Choristomas of theoral cavity: a review. Oral Surg Oral Med Oral Pathol1991;72:584-93.

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An unexpected diagnosis during laryngeal intubation: osseous polypoid lesion of the tongue: osteoma or choristoma?