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avis JM, Campbell LA. Fatal air embo-ism during dental implant surgery: a reportf three cases. Can J Anaesth 1990: 37:12–121.azaridis N, Tilaveridis I, Venetis G,azaridou M. Maxillary sinus osteo-lasty with vascularized pedicledone flap. Oral Surg Oral Med Oralathol Oral Radiol Endod 2008: 106:28–832.opatin AS, Sysolyatin SP, Syso-

ayatin PG, Melnikov MN. Chronicaxillary sinusitis of dental origin:

s external surgical approach mandatory?aryngoscope 2002: 112: 1056–1059.

5. Mason ME, Triplett RG, Alfonso WF.Life-threatening hemorrhage from place-ment of a dental implant. J Oral MaxillofacSurg 1990: 48: 201–204.

6. Mason ME, Triplett RG, Van Sickels

JE, Parel SM. Mandibular fracturesthrough endosseous cylinder implants:report of cases and review. J Oral Max-illofac Surg 1990: 48: 311–317.

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Address:Seog-Kyun MunDepartment of Otorhinolaryngology-Head

and Neck SurgeryChung-Ang UniversityYongsan HospitalHangangno 3-gaYongsan-guSeoul 140-757Republic of KoreaTel: +82 2 748 9847Fax: +82 2 792 6642.E-mail: entdoctor@cau.ac.kr

Hong and Mun

i:

Head and Neck Oncology

. Lopez-Quiles1, E. Ferreira1,

. A. Jimenez-Heffernan2,. Del Canto1

epartment of Oral and Maxillofacialurgery, Hospital La Zarzuela, Madrid, Spain;epartment of Pathology, Hospital La

arzuela, Madrid, Spain

do

lear cell carcinoma of theajor salivary glands in anIV-infected patientopez-Quiles, E. Ferreira, J. A. Jimenez-Heffernan, M. Del Canto: Clear cell

cinoma of the major salivary glands in an HIV-infected patient. Int. J. Oralxillofac. Surg. 2011; 40: 760–763. # 2011 International Association of Oral andxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.

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tract. Clear cell carcinoma is a rare type of salivary gland carcinoma. It has a low

ree of malignancy and long-term prognosis is favourable after surgical removal.

authors describe the case of a human immunodeficiency virus (HIV) infected 43-r-old woman who presented with a tumour on the floor of the mouth. After biopsy,

suprahyoid lymph node dissection and removal of the submandibular and

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10.1016/j.ijom.2010.11.027

Case Report

lingual glands was performed, followed byour presented the characteristic features oned as a variant of clear cell carcinoma bysification. Hyalinizing clear cell carcinom

tern and, to date, there is insufficient informave similarly or differently. The present caour variant occurring in a HIV-infected pan described in the medical literature. The lowour demands a precise diagnosis and com

an immunodeficiency virus (HIV)cted patients often experience anease in the size of the salivary glands,

to a wide range of conditions, includ-

inanthof

radiotherapy. Histologically, thehyalinizing clear cell carcinoma,e latest World Health Organizationhas a characteristic histologicalion to determine whether both formse illustrates a highly uncommonnt. To date, this association has not

grade of malignancy reported for thisete tumoral excision.

Kno

AAv

inflammatory processes, infections neoplasms. In some patients, it is

first sign of disease. An understandingthe underlying salivary pathology is

nHcha

words: clear cell carcinoma; human immu-eficiency virus; HIV; major salivary glands.

epted for publication 1 February 2011ilable online 15 March 2011

essary to guarantee correct treatment3. patients have approximately a 40%

nce of developing a malignant disease,te that is expected to rise due to their

reli-

erssInheceralfa-

Sublingual CCC in a HIV-infected patient 761

increased survival4. The most frequenhead and neck tumours in HIV-infectepatients are Kaposi’s sarcoma (over 15%and non-Hodgkin’s lymphoma (3–10%)4,7

During the last few years, the risk odeveloping oral squamous cell carcinomhas increased4.

Clear cell carcinoma (CCC) is a rarsalivary gland tumour that shows a lowgrade of malignancy1,2. It has a certaipotential for recurrence and metastasisbut long-term prognosis is favourablafter removal, with or without radiotherapy1. It most often affects minor salivarglands5 and occurs predominantly iwomen as a painless, submucosamass1,9. The latest WHO classificatiohas defined it as a malignant epithelianeoplasm composed of a populatioof monomorphic cells having cleacytoplasm5. Many types of salivargland neoplasms may contain a component of clear cells, but CCC is distinguished by the absence of the typicacharacteristics of other neoplasms and itmonomorphic population of clear cells5

Hyalinizing CCC (HCCC) is considerea variant of CCC, distinguished by a densely hyalinized stroma. Both tumours havsimilar clinical characteristics5.

Fig. 1. MRI showing a tumour on the left s

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The authors describe the case of a HIVpositive female patient who developedHCCC in a sublingual location. It is interesting not only because of the relationbetween HIV infection and the tumou

ide of the floor of the mouth.

Fig. 2. The surgical sample had two

3.9 cm � 3.1 cm � 2 cm) attached by a thin

but also because it concerns a ramorphological variant and a major savary gland.

Case report

A 43-year-old woman was referred by hdentist for the evaluation of a painletumour on the floor of the mouth.

1995, during a laboratory analysis swas diagnosed HIV-positive, and sinthen she has been receiving antiretrovitherapy (didanosine, lamivudine and e

-

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r

virenz). She has been asymptomatic withno AIDS-related disorders. The tumourwas detected 1 month before surgery.Laboratory analysis confirmed the HIVpositivity and showed a normal CD4count. Complementary studies includedorthopantomography, which ruled outbone involvement, and MRI, whichshowed a tumour on the left side of thefloor of the mouth (Fig. 1). No local orregional adenopathies were present. Adiagnostic biopsy was carried out and adiagnosis of CCC of salivary gland wasobtained. It was decided to perform a leftsuprahyoid lymph node dissection and toremove the submandibular and sublingualglands. The surgical sample had two nod-ular areas (of 3.5 and 3.9 cm) attached by athin fibrous band (Fig. 2). The smallest hada firm, irregular, solid aspect with inter-mixed adipose tissue whilst the largest onecorresponded to the submandibular sali-vary gland.

Microscopically, the tumoral noduleshowed salivary gland parenchyma (sub-lingual) extensively infiltrated by a neo-

nodular areas (3.5 cm � 3.4 cm � 1.5 cm and fibrous band.

plasgrecellandstrowidfibrpho

cytoplasm. No squamous, acinar or muci-nous differentiation was seen. There was nonecrosis, vascular or neural invasion. Theneoplasia was not encapsulated and showedan infiltrative growth pattern with focal,microscopic involvement of the surgicallimits. The submandibular gland showedno abnormalities. The surgical specimenincluded two small periglandular lymphnodes that showed no metastases. Tumoralcells showed a positive reaction with PASstaining and immunohistochemical expres-sion of cytokeratins (AE1/AE3). There wasno expression of S-100 protein, smoothmuscle actin, muscle-specific actin, carci-noembrionic antigen or calponin. Isolatedcelgenwesubrecrad504dibnodfrac

Fig tic cells are seen in a dense, fibrous stromacon fibrous tissue (haematoxylin–eosin, 20�).

Figwith

762 Lopez-Quiles et al.

. 3. Variably sized solid nests of clear neoplassisting of wide areas of hyalinizing connective

tic epithelial growth. It showed aatest dimension of 2.7 cm. Tumorals formed variably sized solid nests irregular cords. Characteristically, thema was dense and fibrous consisting ofe areas of hyalinizing connectiveous tissue with few fibroblasts and lym-cytes (Fig. 3). The tumoral cells were

large, polygonal and the majority showedclear, empty cytoplasm with well definedcytoplasmic membranes (Fig. 4). The cellswere moderately pleomorphic with occa-sional nucleoli and mitotic figures. In addi-tion to clear cells, a few nests showed apopulation of peripherically distributed,smaller cells with slightly eosinophilic

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. 4. Tumoral cells were large, polygonal and the majority showed a clear, empty cytoplasm well defined cytoplasmic membranes (haematoxylin–eosin, 40�).

ls expressed epithelial membrane anti-. Histology and immunohistochemistry

re characteristic of HCCC involving thelingual gland. After surgery, the patienteived radiotherapy with an externaliation cycle over 6 weeks. She was given0 cGY in 28 fractions to the subman-ular, cervical and supraclavicular lymphes, bilaterally, and 6120 cGY in 34tions to the floor of the mouth. There

been no recurrence or metastasis in themonths since the first operation.

cussion

C is a rare form of adenocarcinoma of salivary glands. Based on the experi-e of the Army Forces Institute ofhology, CCC accounts for 0.7% ofsalivary gland tumours (2% of the

lignant)5. In an English series, 14 of salivary gland tumours (2%) wereposed predominantly of clear cells,

sing great difficulties in diagnosis5.pproximately two-thirds of CCC

ur in the minor salivary glands, gen-lly in the palate, tongue and floor of theuth. More rarely they involve the par-

gland, submandibular gland or lar-5. Patients typically present with a

nless mass, and symptoms may lastm a few months to several years5.spite its good prognosis, isolated casesh cervical lymph node metastasis haven reported5,6,8. The present case isvant because it involved an HIV-cted patient and a major salivary

nd. It is well known that HIV infectionults in an increased risk of malignantease4. Since no other descriptions of

precise association have been reported authors cannot rule out a casual rela-. The tumour corresponded to thesual variant of CCC known as HCCC.s entity was first proposed in 1994 bylchgrup et al. as a subtype of CCC5,6,8.hows a low grade of malignancy and nooepithelial phenotype. It is character-

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Sublingual CCC in a HIV-infected patient 763

ized by nests and narrow strings of clecells in a densely hyalinized stroma. Tpredominant cell population is charactized by its polygonal shape and abundaclear cytoplasm5. From a histopatholocal point of view, the present case wsimilar to those described by Milchgret al.; the tumour cells formed solid neand irregular strings, in a densely hyanized stroma.

CCC and its variants are diagnoses

exclusion. Amongst the other salivaneoplasms that may present with clecells, acinic cell carcinoma, mucoepidmoid carcinoma, myoepithelial carnoma, squamous cell carcinomoncocytoma and epithelial–myoepithelcarcinoma should be considered5,8. Tpossibility of metastases from carcinomwith a clear cell component, mainly rencell carcinoma, should also be consered9. Thorough histological examinatiof the whole tumour is mandatory befoaccepting a diagnosis of CCC. Immunhistochemistry is an important aid in dianosis2. In this patient, the tumour showno evidence of myoepithelial markers,those usually expressed by renal clear ccarcinoma (vimentin and CD10). Histogically, HCCC shows characteristic fetures that permit a precise recognitioTumoral stroma is abundant, dense, hyline and desmoplastic5,6,8. A second poplation of eosinophilic cells is acommonly seen5. An important differe

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tial diagnosis in the present case was osquamous cell carcinoma that may havclear cell component and may affect HIinfected individuals, but no histologievidence of squamous differentiatiwas seen.

Treatment of HCCC is surgical, aimifor complete excision. In this patient,

months have passed since surgery athere is no evidence of recurrence

metastasis. In conclusion, the present caillustrates a highly uncommon tumovariant occurring in a HIV-infectpatient. To date, this association has nbeen described in the medical literatuThe low grade of malignancy reported

this tumour demands a precise diagnoand complete tumoral excision.

Competing interest

None declared.

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9. Wang B, Brandwein M, Gordon

Robinson R, Urken M, Zarbo RJ. Pmary salivary clear cell tumors—a dianostic approach. Arch Pathol Lab M2002: 126: 676–685.

Address:Juan Lopez-QuilesDepartment of Oral and Maxillofacial

SurgeryHospital La Zarzuela28040 MadridSpainTel.: +34 913941929fax: +34 913941929E-mail: jlopezquiles@odon.ucm.es

doi:10.1016/j.ijom.2011.02.009