ƒ Gender : male

指導老師：林立民、陳玉昆、王文岑、陳靜怡醫師

報告者：Intern  G 組 張啟源、鄧聖霖、戴宇昀、何盈興

General Data

ƒ Name : 蔡昇峰

ƒ Gender : male

ƒ Age : 23

ƒ Native : Taiwan

ƒ Occupation : 勞工

ƒ Attending V.S. :王文岑醫師

ƒ First visit :2007/02/14

Chief Complaint

ƒ Pain over L’t lower molar area since

February 2007.

Present Illness

ƒ This 22 y/o male suffered from episode for 2~3 days. He said his tooth fractured during chewing.

Then teeth over L’t lower posterior area became pain .He came to LDC, then the doctor suggested him to our OPD for further exam.

ƒ OPD had biopsy of L’t edentulous ridge showed osteomyelitis(2.14.2007)and inflammatory fibrous hyperplasia(2.23.2007). Clindamycin was

prescribed.

ƒ However, he felt teeth pain aggravated. Biopsy

was performed again over bilateral mandible and

maxilla(3.13.2008).

Past History

ƒ Past Medical History

y Aspirin allergy

ƒ Past Dental History

日期 CC Dx Specimen

20070214 L’t Md. pain Osteomyelitis, 35,36 ■

20070221 Extraction of Retained

dental root (distal root of 36)

■

20070227 Other disturbances of oral epithelium, including tongue

20070416 Periodontitis

20070418、25 Periodontitis

H-P: Ulcer with subacute inflammation (edentulous ridge of 36) Inflammatory fibrous hyperplasia (edentuloous ridge of 36)

Personal History

ƒ Alcohol ：(+)

ƒ Betel nut ： (+)

ƒ Cigarette ： (+)

y 1 package / day

Intraoral Examination

ƒ 16 Elongation

ƒ 16 Root exposure

ƒ 16 17 Dehiscence

ƒ Gingival swelling

ƒ Gingival inflammation

ƒ Alveolar bone resorption

^2008.03.13

Intraoral Examination (cont.)

ƒ 17 un-healing tooth socket located between tooth 16 &18 measuring 1.5X1.5cm

ƒ Color：pink

ƒ No pus discharge

ƒ Tooth 15 lingual tipping

2008.03.13

Physical Examination

ƒ Pain (+)

ƒ Swallowing pain (-)

ƒ Fever or local heat (-)

ƒ Indurations (?)

ƒ Tenderness (?)

ƒ Lymphadenopathy (?)

X-ray finding：Panorex

2008.03.13 Lesion 1

Lesion 2

X-ray finding

Panorex (cont.)

ƒ Lesion 1

is located in the L’t mandibular ramus

ƒ

There is an

ill-defined

and irregular radiolucency without a corticated margin over the left mandibular body extending from 37 distal root mesial aspect to 34 root and extending from alveolar ridge down to mandibular canal measuring 4X2.5 cm

ƒ

No root resorption.

ƒ Tooth floating in the air

over tooth 34 35

ƒ PDL destruction

over 34 35

X-ray finding Panorex (cont.)

ƒ Lesion 2

is located over R’t maxilla tuberosity

ƒ

There is an

ill-defined

and irregular radiolucency without a corticated margin over R’t maxilla tuberosity extending

from tooth 18 distal buccal root to 15 root mesial aspect and extend from hard palate down to 16 furcation,

mesauring about 3X1.5 cm

ƒ No root resorption

but 16 elongation

ƒ PDL destruction

over tooth 16

Dental X-ray finding

ƒ

Missing tooth: 11 21 22 17 36(R.R. 2007.02.21) 38 48

ƒ

residual root: 46

ƒ

caries:18 27 28 47

ƒ

periodontal condition: poor

ƒ

endodontic condition:37

ƒ

restoration (e.g. C & B):12XXX2324

ƒ

filling: 16 25 26 resin filling ,37 amalgam filling

ƒ

Other dental finding：47 distal root resorption

ƒ

Tooth floating in air over tooth 16

ƒ

PDL destruction over tooth 16 31 32

ƒ

PDL widering over tooth 15

ƒ

There is an

ill-defined

and irregular

radiolucency

without a corticated margin over mandibular symphysis extedning from tooth 31 root mesial aspect to 32 root distal aspect and extending from 31 root apex down to

mental spine ,measuring about 1X1cm.

Internal root resorption of 31 was noted.

X-ray finding：Periapical film

X-ray finding

Lesion 1

:

34 floating in air Bone remodeling

Lesion 2

:

17 missing

2007.02.14

2008.03.13 Lesion 1 Lesion 1 Lesion 1 Lesion 2

Lesion 2

2007.02.13 2008.03.20

X-ray summary

ƒ

RL with ill-defined margin over 34-37 area

ƒ Bone remodeling

ability over 34-37 can be seen

ƒ

Incisor lesion –

PDL remained

upon 2/3 root length

ƒ 24 apex area bone density decreased

ƒ

16-18 area bone density decreased

ƒ

--

Multiple area

ƒ

--

Ill-defined margin

ƒ

--

Tooth floating in the air

– PDL disappear

ƒ

--

Bone remodeling character

1. Disease origin : Bony or periodontal origin

2. Infection or neoplasm

3. Benign or malignant

Neoplasm or Infection ?

ƒ Fever or Local heat： (-)

ƒ Pus discharge : (-)

ƒ Duration：14 months

ƒ Lymphadenopathy： (？)

ƒ Induration：(？)

Neoplasm

Benign or malignant?

ƒ Margin：poor-defined

ƒ Pain：(+)

ƒ Ulceration: (+)

ƒ Duration : 14 months ⁺

Malignant

Working diagnosis

1. Multiple myeloma

2. Langerhan cell disease

3. Metastatic carcinoma

Multiple myeloma

Our case Multiple myeloma

Gender Male

Age 23 35-70 y/o (60 y/o average)

Frequency Contineous pain Symptom & Sign Dull bone pain

Tooth mobility

Bone pain, Bone swelling Pain paraesthesia

Tooth mobility Location L’t ramus , R’t tuberosity Jaw bone (30%)

Size 4x2.5cm , 3x1.5cm

Number 3, Multiple Multiple

Margin Ill-defined Well-defined

Shape (X-ray) Radioluency Punched out radiolucency Tooth involvement Floating in the air

Our case

Multiple myeloma

Langerhan cell disease

Our case Langerhan cell disease

Gender Male Male prediction

Age 23 < 10 y/o (50%), All aged

Frequency Contineous pain Symptom & Sign Dull bone pain

Tooth mobility

Dull pain Tenderness

Location L’t ramus , R’t tuberosity Mandible posterior

Number 3, Multiple Monostotic, polyostotic

Osteolytic bone lesion

Margin Ill-defined No corticated rim

Ill-defined radiolucency Shape (X-ray) Radioluency punched out radiolucency

Scoope out

Tooth involvement Floating in the air Floating in the air Tooth mobility, loss Severe periodontitis

Our case

Langerhan cell disease

Metastastic tumor

Our case Metastastic tumor

Gender Male Male inclination

Age 23 Middle age to older adult

Frequency Contineous Unknown

Symptom & Sign Dull bone pain Mucosa ulceration Location L’t ramus , R’t tuberosity Gingival>50%

Tongue 25%

Number 3, Multiple Uncertain

Shape (X-ray) Radioluency Ill-defined bony destruction Tooth involvement Floating in the air uncertain

But p’t didn’t have medical history of origin primary tumor

◎

Langerhan cell disease

Other test and examination:

※染色體檢查報告:

Date : 2008/4/21

Specimen site: Buccal mucosa

Result : No chromosome abnormality

was found

Biopsy :

Date: 2008/3/27

Site: bilateral maxillary & mandibular: A 34-36 area;

B14-16 area

Pathologic diagnosis: A &B all showed Langerhan cell disease with feature of lymphoproliferative disorder

Date: 2008/4/24

Site: bilateral maxillary & mandibular: A 34. 35 area;

B15,16 area

Pathologic diagnosis: A &B all showed Langerhan cell disease with feature of lymphoproliferative disorder

MRI examination(2008/4/9):

Tc -99m MDP whole body bone scan Impression:

High probability of local bone invasion

from oral cancer to facial bones and

distant bone metastasis to spine

Chest X­ray examination (lung windows) 2008/4/21 Specific findings:

Multiple lung cyst (multiple cyst ）

Compatable with langher cell hisiocytosis

Lung cyst

Final diagnosis :

Langerhan cell disease

References

ƒ Oral and maxillofacial pathology Second edition

ƒ Different diagnosis of oral and maxillofacial lesions fifth edition

ƒ Essentials of ORAL PATHOLOGY AND ORAL MEDICINE ; R.A. CAWSON, E.W. ODELL, CHURCHILL LIVINGSTONE, Sixth edition

ƒ Langerhan cell granulomatosis with unusual FNAC findings. SenGupta SK, Vince JD, Chakravorty P, Sinha SK. Department of Pathology, Faculty of Medicine, University of Papua New Guinea, Papua New Guinea.

ƒ Histiocytoses in children: analysis of 120 cases and the bone marrow findings in infection-induced hemophagocytic syndrome vs malignant histiocytosis. Hathirat P, Chuansumrit A, Nitiyanant P, Kraipibool P, Ruangdaraganon N, Mahaphan W, Daengprasert S, Isarangkura P.

Department of Pediatrics, Faculty of Medicine, Ramathibodi Hospital, Mahidol University, Bangkok, Thailand.

ƒ Osteomyelitis caused by mycobacterium fortuitum. Ohry A, Brooks M, Steinbach T, Rozin R.

Thanks for your attention