口腔病理診斷科 Clinicopathological conference Intern Case Report

口腔病理診斷科Clinicopathological conferen

ce

Intern Case Report指導老師 : 林立民醫師 陳玉昆醫師

王文岑醫師 陳靜怡醫師

D 組：楊駿恒‧鄭婉伶‧曾偉哲‧蔡馥慧‧李佩昕

95,02,07

Clinicopathological conferenceClinicopathological conference

clinicalclinical radiologyradiology pathologypathology

A case reportA case report

General featuresGeneral features

conventional filmconventional film MRIMRICTCT

treatmenttreatment

prognosisprognosis

• Name : 金 XX• Gender : female• Age : 13• Native : 高雄市• Occupation : 學生• First visit : 94/09/24• Attending V.S. : 黃逸岳醫師

General data

Chief Complaint

Unerupted lower left second molar

Present Illness

• The 13 y/o female p’t suffered from 37 unerupted tooth. She went to see Dr. 謝尚廷 10 months ago. The dentist found there was a dentigerous cyst and referred her to Dr. 柯政全 for help.

• Dr. 柯 referred her to our OPD for examination and treatment.

Past History

• Past medical history– Denied any systemic disease– Denied any drug or food allergy

• Hospitalization : – Nil

• Past dental history– Attitude ： afraid– Extraction– O.D

Personal History

Alcohol : (-)Betel nut : (-)Cigarette : (-)

Intraoral Examination• 37 unerupted tooth

• Mild swelling on 37 area

• Mild expansion

• No purulent drainage was presented

• Overlying mucosa: normal color

• No loosening teeth

Panorex(940924)

18, 28, 38, 48, 37 uneruptionA well-defined ovoid-shaped radiolucent lesion with regular corticated margin over

37 coronal part to the root trunk, measuring 3x2.5 cm.The superior border of mandible body over 37 area seems absent.The left mandibular canal is narrowed and indented over 37 apical area.37 is well-developed and submerged down to the level 1 cm below the superior alv

eolar ridge.

Lateral view

PA view

Water’s view

• Inflammation or Neoplasm?

• Intrabony cystic lesion?

Benign or Malignant?Benign or Malignant?

Inflammation or Neoplasm?

• No purulent drainage was presented• No fever• No complaint of pain or other

uncomfortable symptom• Overlying mucosa: normal color

NeoplasmNeoplasm

Benign or Malignant?

• A well-defined R-L bony lesion with regular, corticated border.

BenignBenign

可能為一個可能為一個

Intrabony benign tumorIntrabony benign tumor

Intrabony cystic lesion?• Overlying mucosa: normal color

• A well-defined R-L lesion with corticated border

可能為一個可能為一個Intrabony cystic lesionIntrabony cystic lesion

Working Diagnoses

Intrabony benign tumor• Unicystic ameloblastoma• Ameloblastoma• Ameloblastic fibroma• Odontogenic fibroma• Odontogenic myxoma• Adenomatoid odontogenic tumor (AOT)

Intrabony cystic lesion• Dentigerous cyst• Odontogenic keratocyst

Unicystic ameloblastomaHigh compatible Low compatible

Female predilection Usually with an impacted third molar

Age ： 10-20Well-defined Radiolucencyposterior region of mandible

Expansion of cortical plateto the point of destruction

AmeloblastomaHigh compatible Low compatible

Mandible, molar-ascending ramus area

Usually older than 20

Asymptomatic, painless swelling

Multilocular radiolucent lesion, soap bubble, honeycombed

An unerupted tooth Irregular scalloping margin

Ameloblastic fibromaHigh compatible Low compatible

Age ： 5~15yrs slightly more common in males

A missing tooth or an unerupted tooth(75%) is associatedPremolar-molar area of mandible

～ unilocular or multilocular～ Well defined and often corticated in a manner similar to that of a cyst

Odontogenic fibromaHigh compatible Low compatible

Female predilection Mean age 40 y/o

Mandible ： posterior to 1st molar

Accompanied with impacted tooth

Well defined R-L lesion

Odontogenic myxomaHigh compatible Low compatibleUsually is well definedcorticated margin

honeycomb 、 tennis racket 、 soap bubble

Age ：１ 5 ~ 30 Usually multilocular

With an impacted tooth cystlike ， unilocular ，may have a mixed R-L, R-O imageMand.>Max.

Adenomatoid odontogenic tumor (AOT)

High compatible Low compatible

Female > Male Ant. portion of jaws

follicular, sometimes accompany with impacted Tooth.

Max.>Mand.

Slow growing, benign, and does not infiltrate bone

Dentigerous cyst

High compatible Low compatible

With impacted tooth Cyst attaches at the CEJ

R-L, Well defined Lower third molaror upper canine

Corticated margin

Unilocularcurved or circular outli

ne

Clinical Impression

• Dentigerous cyst, 37 area• Unicystic ameloblastoma, left mandibul

ar body over 37 area

Treatment Course• Incisional Biopsy on 940924

HP report ： Odontogenic myxoma with feat

ures of cystic odontoma over 37 area

Treatment Course cont~

• OP on 94/10/21 – (Hospitalization:10/20~10/25)

• Extraction of 36 37 38 48

• Excision + bone trimming

• Bone graft (Bio-oss x 3 BT)

• Primary closure

Treatment Course cont~ 94/10/22

Treatment Course cont~

Treatment Course cont~

Treatment Course cont~

Treatment Course cont~

Treatment Course cont~

Treatment Course cont~

HP report—• Odontogenic myxoma 37 area• Root resorption 36 37

Treatment Course cont~

OPD follow up—• 941029 No numbness and parest

hesia Wound dehiscence

• 941105 Re-suture

Treatment Course cont~ 94/11/19

Discussion

odontogenic myxoma

Definition• World Health Organization’s classificati

on for histological typing of odontogenic tumors:

“A benign tumor, which is of ectomesenchymal origin and is

a locally invasive neoplasm consisting of rounded and angular cells lying in an abundant mucoid stroma”

odontogenic myxoma Relative Prevalence

• The relative prevalence of odontogenic myxoma is 0.2–17.7% of all odontogenic tumors.

DISCUSSION

Odontogenic myxomaOdontogenic myxoma

clinicalclinical radiologyradiology pathologypathology

A case reportA case report

General featuresGeneral features

conventional filmconventional film MRIMRICTCT

treatmenttreatment

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Clinical Features

• About 6% of all odontogenic tumors of the jaw

• Predominantly in young adults– Occur over a wide age group

• Average age ： 25 ~ 30• No sex predilection• Almost any area of the jaws• Mandible > Maxilla

Clinical Features• Slow growing• May be associated with an impacted t

ooth• Small lesions ： asymptomatic• Larger lesions ： painless expansion

of bone• Loosening and migration of teeth• In some instances may grow rapidly

– Accumulation of myxoid ground substance

Odontogenic myxomas

in the Hong Kong Chinese:clinico-radiological presentation

and systematic review

DS MacDonald-Jankowski, R Yeung2, KM Lee and TKL LiDentomaxillofacial Radiology (2002) 31, 71 ~83

Aim

• to determine the clinical and radiological presentation on plain films of central odontogenic myxomas (OM) in the Hong Kong Chinese

• compare them to other reported series by a systematic review (SR).

Methods:

• The files of the Department of Oral and Maxillofacial Surgery of the University of Hong Kong between 1989 and 2000 were reviewed for OM cases.

• The relevant literature was identified by electronic databases

Results:

• The 10 Hong Kong cases were broadly consistent with the predilections for females and the mandible of other reports.

• The mean age at first presentation in the present report is 36.9,older than the other reports.

• Most lesions appear to be larger than those in many other reports.

• Although all OMs in the present study are still being followed up after surgery, none have recurred.

Discussion

‧presentation of larger lesions in the older Chinese could in part be explained – by attitudes rooted in traditional

medicine• in spite of the widespread availability of

modern medical care in Hong Kong

• In comparison to other industrialised nations – Hong Kong has a moderately high mortality

rate for cervical cancer• Hong Kong Chinese women fail to make use of t

he almost ubiquitous cervical screening services.

• Nevertheless, it would appear that long term if not life-long follow-up of OM is merited. – One case, followed for 35 years, recurred

• after prolonged remissions of 20 and then 10 years

Odontogenic myxomaOdontogenic myxoma

clinicalclinical radiologyradiology pathologypathology

A case reportA case report

General featuresGeneral features

conventional filmconventional film MRIMRICTCT

treatmenttreatment

prognosisprognosis

Conventional image

• Radiolucency • The lesion is usually well-defined and it often

has a corticated margin. -- However , the outline of some lesion , especially in maxilla , is po

orly defined.• When it occurs pericoronally with an impacted

tooth , it is most likely to have a cystlike , unilocular outline , although it may have a mixed radiolucent-radiopaque image.

• It may be unilocular or multilocular (honeycomb , tennis racket , soap-bubble)

Conventional image cont~

• When the tumor expands in a tooth-bearing area , it displaces and loosens teeth , but root resorption is rare.

• The lesion also frequently scallops between the roots of adjacent teeth.

Conventional image cont~

Benefits of CT & MRI

• CT and MRI should be performed for the determination of tumor extent and margins before resection.

→Reduce the recurrence rate, especially for a tumor spreading widely into the soft tissues.

• Analyses based on CT features may enable a consensus on interpretation and expression of findings of odontogenic myxoma on conventional radiographs.

Computed Tomography

Features:1. Locularity: • The multilocular appearance was three-dime

nsionally defined as a finding with compartments truly separated by bony septa.

(On conventional radiographs, intralesional trabeculations might be projected two-dimensionally onto films, forming the multiple compartments.)

• CT analyses could contribute to resolving this discrepancy.

T Koseki et al / Dentomaxillofacial Radiology, 2003

Computed Tomography (cont.)

2. Border:• As many bony structures are superimposed o

n the maxilla, conventional radiography may not clearly depict the border.

• Most of maxillary tumors with diffuse borders were relatively large and spread into the maxillary sinus.

• Both maxillary and mandibular lesions on CT are well defined.

• Even in large tumors without cortical continuity and with direct contact with the surrounding soft tissue, the tumor margin was observed to be smooth and clear on CT images.

Computed Tomography (cont.)

3. Density - “tennis racket” appearance”:

• The fine straight trabeculations form square or triangular compartments appearance on conventional radiographs.

• “soap bubble” or “honeycomb” appearances suggesting other lesions, including ameloblastoma or odontogenic keratocyst.

• This feature was observed definitely on the inner side of the cortical margins in several cases.

• This typical appearance cannot be observed in all patients, but this CT feature would have significantly contributed to allowing a diagnosis of odontogenic myxoma.

Magnetic Resonance Imaging

• MRI showed soft tissue image.• Myxoma typically:

Intermediate signal intensity on T1-weighted imageHigh signal intensity on T2-weighted image.

• In contrast T1-weighted image:– Peripheral with large collagen bundles → enhanced– Central with cellular mucoid component → not enha

nced→MRI and enhanced MRI corresponded to the gro

ss features and the microscopic features of the resected tumor.

J. Asaumi et al. / European Journal of Radiology, 2002Kawai et al. / ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY, 1997

MRI (cont.)

• MRI showed well the erosive extension into the adjacent structures and the invasion into the interroots of the teeth.

→This feature indicated that the lesion was not a consistent mass lesion such as ameloblastoma because the lesion did not absorb or move the roots of the teeth.

• Recurrent tumors can be detected by their high signal in T2-weighted MRI scans.

dynamic Magnetic Resonance Imaging

• Difficult to radiographically distinguish odontogenic myxomas from ameloblastomas.

→ Noting that although both the gross and microscopic features are visible by MRI, the signal intensities are not characteristic of odontogenic myxomas alone.

→ dynamic MRI- Contrast medium: Gd–DTPA

J. Asaumi et al. / European Journal of Radiology, 2002

Dynamic MRI (cont.)

• The contrast index (CI): (signal intensity (postcontrast)−signal intensity (precontrast))/signal intensity (precontrast). The time course of the CI (CI curves) was obtained by plotting the CI on a time course.

dynamic MRI (cont.)

• The CI curves represent the blood behavior, therefore the enhancement pattern of dynamic MRI may reflect the intratumoral angiogenesis.

• CI curves of benign tumors increase gradually, while malignant tumors increase rapidly.

• The CI curve: Pleomorphic adenomas gradual increased

Warthin's tumors rapid enhancement, reaching a maximum CI, decreasing rapidly, and then undergoing a gradual wash-out.

• dynamic MRI enhancement have characteristic features, and may be useful in making a differential diagnosis.

Odontogenic myxomaOdontogenic myxoma

clinicalclinical radiologyradiology pathologypathology

A case reportA case report

General featuresGeneral features

conventional filmconventional film MRIMRICTCT

treatmenttreatment

prognosisprognosis

• A loose,myxomatous tumor can be seen

• filling the bone marrow spaces between the bony trabeculae

• The inset shows stellate-shaped cells and fine collagen fibrils.

(i)gelatinus ,loose structure of the myxoma,is observed.

(ii)composed of haphazardly arranged stellate,spindle-shaped,and round cells in an abundant ,loose myxoid stroma that contains only a few collagen fibrils.

histopathologic features

(iii)small islands of inactive-appearing odontogenic epithelial rests may be scattered throughout the myxoid ground substance.(not required for diagnosis and not in most cases)

but….

• Odontogenic Myxoma Showing Active Epithelial Islands With Microcystic

Features J Oral Maxillofac Surg 59:1226-1228, 2001

(iv)(in some patients)

the tumor may have a greater tendency to form collagen fibers;such lesions are sometimes designated as fibromyxomas or myxofibromas.

histochemical studythe ground substance is composed ofglycosaminoglycans,chiefly hyaluronic aci

d and chondroitin sulfate.

Immunohistochemical study

the myxoma cell show diffuse immunoreactivity with antibodies directed against vimentin,with focal reactivity for muscle-specific actin

• Less than 1% of tumor and control cells were positive for Ki-67.

• Odontogenic myxoma tumor cells did not show an increase in cell division.

The Expression of Apoptotic Proteins and Matrix Metalloproteinases in Odontogenic Myxomas J Oral Maxillofac Surg 61:1463-1466, 2003

Bcl-2

Bcl-X Ki-67

MMP-2

MMP-3

MMP-9

Bak&Bax(proapototic protein)

Specimen

(+),

6.5

%

(+),

10.4%

<1%

90%

- -

Not

detected

Control tissue

(+),

1.1

%

(+),1.2%

<1%

90% - -

Not

detected

• Odontogenic myxoma tumor cells showed increased expression of antiapoptotic proteins (Bcl-2 and Bcl-X) and the mat

rix metalloproteinase MMP-2.

• This study suggests that 2 mechanisms of disease progression used by the odontogenic myxoma are the production of antiapoptotic proteins and the secretion of matrix metalloproteinases.

The Expression of Apoptotic Proteins and Matrix Metalloproteinases in Odontogenic Myxomas J Oral Maxillofac Surg 61:1463-1466, 2003

• Irregularly shaped epithelial cells showing a positive reaction with CK 19

• The microcystic spaces were lined with CK 19-positive flattened cells

--supports the odontogenic origin

Odontogenic Myxoma Showing Active Epithelial Islands With Microcystic Features J Oral Maxillofac Surg 59:1226-1228, 2001

• An increased number of Ki-67-labeled active cells

Odontogenic Myxoma Showing Active Epithelial Islands With Microcystic

Features J Oral Maxillofac Surg 59:1226-1228, 2001

• some of the epithelial islands were not only active morphologically, but also functionally.

couclusion~• From histopathologic study: 1.not encapsulated,tumor cells fill the bone marrow spaces • From Immunohistochemical study: 1.odontogenic origin 2.antiapoptotic proteins production, matrix metalloproteinases secretion.=>recurrence rates approximately 25%

Odontogenic myxomaOdontogenic myxoma

clinicalclinical radiologyradiology pathologypathology

A case reportA case report

General featuresGeneral features

conventional filmconventional film MRIMRICTCT

treatmenttreatment

prognosisprognosis

Treatment

• Small myxomas are generally treated by curettage, but careful periodic reevaluation is necessary for at least 5 years.

• For larger lesions, more extensive resection may be required because myxomas are not encapsulated and tend to infiltrate surrounding bone.

• Complete removal of a larger tumor by curettage is often difficult to accomplish, and lesion of the maxilla, in particular, should be treated more aggressively in most instances.

Slootweg and Wittkampf (Myxoma of the jaws. An analysis of 15 cases. J Maxillofac Surg 1986;14:46-52.)

• site of the myxoma – management plans

• Mandible: – easier to extirpate all visible tumor tissue by enuc

leation with thorough curettage. • Maxilla:

because of the more complex anatomy vicinity of vital structures greater risk of infiltrative spread being undetected m

acroscopicallybecause of the greater risk of recurrence.

precludes the more conservative approach

• Complete surgical removal can be difficult, especially in the maxilla – myxomatous tissue infiltrates cancellous bo

ne at an early stage, – impossible to clinically determine the exten

t of this infiltration if no visible bony destruction has occurred.

Management techniques for myxomas of the jaws

(Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2000;90:348-53)

• enucleation and curettage or wide excision and resection.

• most recently the trend is toward more radical surgery, aiming to prevent recurrence.

• Advocates of radical surgery,promote the use of wide excision or resection of the lesion together with adjacent tissue.

Barros et al reviewed a series with a recurrence rate of 75% in those cases managed conservatively.

Harder reported that 6 out of 10 lesions treated conservatively had recurred in their series

However, advocates of the more conservative regime argue that, despite a notable recurrence rate, a significant number of myxomas are treated conservatively (by excision or curettage) with no recurrence.

Kangur TT, Dahlin DC, Turlington EG. Myxomatous tumors of the jaws. J Oral Surg 1975;33:523-8.

• Therefore, a permanent cure does not

always require extensive surgery, removing adjacent uninvolved bone.

Management techniques for myxomas of the jaws

• enucleation and curettage or wide excision and resection.

• the trend is toward more radical surgery,aiming to prevent recurrence.

To this end, Alphin et al believe an initial conservative approach sparing uninvolved structures could be used to allow maximal preservation of function reserving the more radical approach only for recurrences.

• However, it is important to balance the use of the radical approach with maintaining the presence of vital or uninvolved structures to preserve function as fully as possible.

Prognosis of myxomas of the jawPrognosis of myxomas of the jaw is generally good, although recurrence has been described over 30 years after original surgery.

It is suggested that patients be followed closely for at least 2 years because this is the most likely time for recurrence.Bucci E. Odontogenic myxoma: report of a case with peculiar features. J Oral Maxillofac Surg 1991;49:91-4.

Recurrence & Prognosis

• Because of its infiltrative character, this lesion is difficult to be curettaged, and this explains its high recurrence rate.

• Recurrence rates from various studies average approximately 25%.

• In spite of local recurrences, the overall prognosis is good, and metastases do not occur.

Cryotherapy as an adjunct procedure to curettage can be used as such technique minimizes the risk of recurrence.Raphael N. Aquilino *, Fabr ´cio M. Tuji, Nayene L.M. Eid,Omar F. Molina, Hea Y. Joo, Franciısco Haiter NetoGurupi Regional University—UNIRG, Gurupi, Tocantins, BrazilPiracicaba Dental School—UNICAMP, Piracicaba, Sa o Paulo, Brazil˜Received 3 October 2005; accepted 5 October 2005

Clinicopathological conferenceClinicopathological conference

clinicalclinical radiologyradiology pathologypathology

A case reportA case report

General featuresGeneral features

conventional filmconventional film MRIMRICTCT

treatmenttreatment

prognosisprognosis

Thank you for your attention!