大韓放射*영양홉장F 會註‘ 第 22 卷 第 6 號 pp. 1066 -1071, 1986 Journal of Korean Radiological Society, Vol. 22 , No.6, 1986

An Aggressive Osteoblastoma in the Left Iliac Bone

- A Case Report -

Myung Joon Kim, M.D., Seoung Oh Yang, M.D., Han Gi Jo, M.D., Chul Koo Cho, M.D., In Woo Ro, M.D.

Departmeη t 01 Radiology, CaPital Armed Forces Geη eral Hospital

〈 국문초록〉

조}측 장콜에 발생한 침습적 콜아세포종으 I 1 예 보고

국군수도뱅원 땅사선과

김명준·양승오·조한기·조철구 · 노인우

골아세 포종은 양성 골종양으로 발생 빈도는 약 1% 로 매우 낮마. 특히 장골에 발생 한 침습적 골 아세 포종응 발생 인도가 더욱 낮응 것무로 알려져 있다.

저자들은 국군수도뱅원에서 좌측 :o

Aggressive osteoblastoma is a very rare primary bone tumor having locall y aggressive and destructive natures But distant metastasis is not we ll occurred. Aggressive osteoblastoma may be similar to osteoblastoma radiological Iy, but has different pathological leatures We experienced one case 01 aggressive osteoblastoma arising Irom lelt ilium and resport this case with review 01 the literatures

among the osteoblastomas.'.8 Introduction Aggressive osteoblastoma is very rare and bi 이 ogical ， pathological features of it is different frorn Osteoblastoma is an uncommon primary neoplasm the conventional osteoblastoma. We experienced one of bone, accounting for 1070 of primary bone case of aggressive osteoblastoma in the left ilium and tumors. 12.3.4 Osteoblastornas are benign bone tumors, describes the radiological, pathological findings of an but recurrences and malignant transformation of aggressive osteoblastoma with review of the literatures.

S 6 7 osteoblastoma were reported. . . So Jaffe and Dorf man proposed the term of “ aggressive osteoblastoma" Case Report in which the tumor had intermediate biological natures A 45 year old man was admitted to the Capital Received September 23; accepted r\Jovember 3, 1986 Armed Forces General Hospital in June 1986 com

- 1066 Myung ]oon Kim , et al.: An Aggressive Osteoblastoma in the Left Iliac Bone

plaining of pain and palpable mass in the left iliac area He was well until 10 years ago, when he experienced pain in the left iliac area, which was initially mild and intermittent, but the symptom was aggravated since April 1986. On physical examination there was an egg sized, hard, non-movable, tender mass in the left anterior superior iliac spine. Radiograph of the left ilium showed an osteolytic mass having mottled and dense ossifications. There was also thin calcified shell in the inferior margin of mass and interspersed radiolucent areas. (Fig. 1) Computerized tomograph of the pelvis discolsed a conglomerated calcific density and scat­ tered low densities within the osteolytic mass, and moderate cortical break-down. And soft tissue involve­ ment was suspected. (Fig 2 A , B) A 99mTc methylene diphosphonate bone scan demonstrated a focus of increased activity in the left iliac bone. (Fig 3) The patient underwent wide exci­ sion. The mass located in the left iliac wing was about

8 x 5 cm sized, hard and nodular. Perforation of in­ Fig. 1. Radiograph of left ilium shows oste 이ytic mass ner cortex was also demonstrated. (Fig 4) Foci of the having central dense sclerotic area with thin calcified rim in the inferior margin. cortical break-down with necrotic areas were seen on the cut surface. (Fig 5) Microscopic examination revealed osteoid rimm­ was heavily calcified. The calcification was irregular, ed trabeculae in a vascular fibrous stroma. The osteoid dense, jagged calcification so called spiculated blue

A B

Fig, 2. A.B. CT of left ilium shows conglomerated calcific density and scattered low densities within the mass Cortical breakdown and soft tissue involvement were suspected.

- 1067- - The Journal of the Korean Radiological Society, Vol. 22 , No. 6, 1986 -

~-

.. ‘ ?‘ (ημ I I에.A. F.‘:J . l“ PA!1'fI s..a앙!엉;‘ 1 ____ • l!iIil‘ I>l - - - ,, - _.""" ._..~‘ “ liïdWi 킥M - L •-• 一l Fig. 5. Photograph of surgical specimen shows about 10 x 10 x 5cm sized mass. Cut surface of bony tissue shows foci of the cortical break-down and necrobc areas

bone. The osteoblasts showed large, plump nuclei and abundant pale cytoplasms. (Fig 6 A,B,C)

Fig. 3. Whole body bone scan shows a focal increased Discussion activity in the left ilium. Osteoblastoma is an uncommon primary bone tumor and the incidence is about 1070 of primary bone

tumors. I.2.3.4.9) Osteoblastomas are benign bone tumor. But rarely osteoblastoma may behave in locally ag­ gressive fashion. And the recurrence of osteoblastoma (15%) and malignant transformation to osteosarcoma

were reported. 4 . 5 . 7.1 0.11 ) There were unusual cases in which the distinction 2 11 from the low grade osteosarcoma was difficult. . . 13) So some authors have referred to such tumors hav­ ing intermediate natures as malignant osteoblastoma

13 or pseudomalignant osteoblastoma. 'O . ) Schajowicz

and Lemos 'O ) used the term of malignant osteoblastoma which was locally aggressive but separated from the osteosarcoma because of different c1 inical, radiological and peculiar histologic pattern. Osteoblastoma with typical benign radiological ap­ pearance and demonstrating bizarre cytologic aberra­ tions was called pseudomalignant osteoblastoma by

κ1irra et al. 13) But now the designation of an aggressive Fig. 4. Radiograph of resected iliac bone shows more dense sclerotic area in central portion and thin osteoblastoma, as a transitional form, is used since , 8 calcified rim in the inferior margin‘ of the mass. it has been proposed by Jaffe and Dorfman. . ) So call

- 1068 - - Myung Joon Kim , et a1.: An Aggressive Osteoblastolll a in th e Left lIiac Bone -

B

Fig. 6. A.B. Photolllicroscopy (H-Ex 40 , 100) shows highly cellular area fOr!lling cOlllpact group of tumor cells of osteoblastic type with calcified spiculated blue bone. C. Photolllicroscopy (H-E x200) shows in­ divi dual tUlllor cells having large pale nuclei with abundant pale eosinop hil ic cytoplaslll , and occasional prolllÍnent nucl eoli.

ed aggressie osteoblastoma is the osteoblastoma which bone are also involved. Aggressive osteoblastoma of recurs and invades locally but does not metastasize. the pelvis is very rare.IO .I1 .15 . 16 But recently metastatic cases were reported . 5 . 1 이 There is much variable radiological features in ag- These features raise the controversial point, that gressive osteoblastoma. Some tumors are seen with is, aggressive osteoblastoma is really osteosarcoma equivocal roentgenographic picture of osteosarcoma

9 which resembles osteoblastoma with indefinite verus typical osteoblastoma. ) About 25070 of histologic features of osteosarcoma or transformed to osteoblastomas may have a roentgenographic ap-

9 osteosarcoma. by histological change. ) pearance suggestive of malignancy. 2) Advanced or The age of patients with aggressive osteoblastomas recurrent lesions showed extensive soft tissue invasion 8 has ranged from 6 to 67 years. About half have oc- and destruction of the cortex of the involved bone. ) curred in individuals less than 30 years 01d. 15 In this Most osteoblastomas have the destructive and ex- case the patient was 45 year old man. Symptoms are pansile appearance and the cortex of the lesion site usually pain and palpable mass, and present for 3 to is thinned. This osteolytic lesion is well outlined and

15 5 months. This patient symptom was same as the may have a fine caIcific shell.l.2.I2. 15 . 17) above and aggravated 2 months before admission. But The tumor matrix shows a variable amount of mild and intermittent pain in the left iliac area has been caIcifications and may be radiolucent, contain some present since 1975. spotted caIcifications. And the tumor may be an ex- In contrast to conventional osteoblastoma the ax- pansile, amorphous or densely caIcified mass. In such ial skeletons, such as femur, tibia, fibula are mo re cases Robert Freiberger used a term “ cumulus cloud often involved. But the vertebra, pelvis, metatarsal configuration."13 Longstanding tumor becomes heavi-

- 1069 - - The Journal of the Korean Radiological Society, Vo l. 22 ‘ No. 6, 1986 •

ly ossified when it reaches full maturity. 1 ) In some osteoblastoma has the basic pattern of conventional cases there may be cortical break-down and form a osteoblastoma. But almost aggressive osteoblastoI1ì a

17 soft tissue component. 2.17) are cytologically atypical. ) The histologiaal hallm:ark In conventional radiograph of this case the tumor of aggressive osteoblastoma and characteristics of showed an expansile osteolytic masscontaining dense osteosarcoma are summarized in followil ~g table 1) 9) ossification. In the inferior margin of the mass 11) peripheral caIcified rim was revealed. The computeriz- Confluent osteoid and heavily calcified !i)steoid can ed tomograph demonstrated conglomerated and be found. This calcification has been emp:J, asized by some spotted caIcific densities and scattered low den- “ spiculated blue bone'’, which is similar to that seen

15 sities within the mass. Moderate cortical break-down in some osteosarcoma.IO. ) In the diagnosis of ag- and adjacent soft tissue involvement were suspected. gressive osteoblastoma differentÎation from osteosar- The radiological findings of this case, especiall y coma is most important and may be diffícult. peripheral thin caIcified rim, central dense ossifica- The radiological differential diagnosis ln this case tion, and cortical break-down may be compatible incI udes chondrosarcoma, osteosarcoma .malignant with aggressive osteoblastoma. change of osteochondroma. Osteoid osteoma is less Osteoblastoma is an osteoid producing benign bone possible tumor. The histological characteristics of The osteolytic and expansile lesion with central osteoblastoma are the trabec 비 ae of osteoid tissue, calcifications in a 45 year old male patient involving usually surrounded by osteoblasts, interspersed with the pelvic bone is raised the possibility of chondrosar- a highly vascularized stroma of fibrous connective coma. Coalescecj caIcifications are unusual, but can tissue. 1) But in occasional instances differentiation be found in the chondrosarcoma. And caIcifications from the low grade osteosarcoma may be difficult. So of chondrosarcoma are punctate, snowflake type or

'9 Dorfman and Weiss 'B ) devided those osteoblastic ring shaped. ) Thin calcified rim is not usual pattern tumors having malignant potentia l into 4 groups; 1) of chondrosarcoma and soft tissue mass is not definite low grade osteosarcoma resembling osteo

Aggressive osteoblastoma OsteosarcoO'l a

epithelioid osteoblast • Osteoid trabecular or sheet like lace )ike • Intertrabecular tissue loose, vascular solicl. cellular • Mitotic rate low high • 、 A typical mitosis no yes • Sheets of cells no yes without bone production • Tumor cartilage formation no yes • Zonation yes no

* zonation; presence of peripheral, well formed bony trabeculae

- 1070 - Myung Joon Kim, et al.: An Aggressive Osteoblastoma in the Left Iliac Bone portion and thin cartilage cap is demonstrated. The 7. Merryweather R, Middlemiss JH, 5anerkin NC: Malignant outlines are well demarcated. But large osteochon- Transformation of Osteoblastoma. j 80ne joint 5u영 droma with central ossification and peripheral car- 62-8:381-384, 1980 tilage cap may show similar radiological appearance 8. Revell PA, 5choltz CL: Aggressive Osteoblastoma. j Pathol 10 this case. Interrupted demarcation may be 127:195-198, 1979 demonstrated in the malignant change of 9. Bertoni F, Unni KK, McLeod RA et al: Osteosarcoma osteochondroma. resembling Osteoblastoma. Cancer 55:416-426, 1985

Osteoid osteoma in the pelvic bone is very rare. 16) 10. 5chajowicz F, Lemos C: Malignant Osteoblastoma. j 80ne And osteoid osteoma is more small sized lesion and joint 5urg 58-8:202-2 11, 1976 has prominent surrounding sclerosis. But Pieterse'O) 11. Dorfman HD: Case Reports of the Massachusetts Ceneral

S and Seki ) reported the osteoid osteoma transforrri- Hospital, case 40. N Engl j Med 303:866-873, 1980 ing to aggressive osteoblastoma. 12. Kenan 5, Floman Y, Robin CC et al ‘ Aggressive Osteoblastoma-A Case Report and Review of the

Summary Literature. C1in Orthop 195:294-298, 1985‘ 13. Mirra JM, Kendrick RA, Kendrick RE : Pseudomalignant We experienced one case of aggressive Osteoblastoma Verus Arrested Osteosarcoma. Ca ncer osteoblastoma of left ilium in a 45 year old man and 37:2005-2014, 1976 described radiological and pathological findings of ag 14. Mitchell ML, Ackerman LV ι1etastat i c and gressive osteoblastoma. And we will perform a follow Pseudomalignatn Osteoblastoma; A Report of Two up study for the evaluation of loca“ recurrence, Unusual Cases. 5keletal Radiol 15:213-218, 1986. metastasis or sarcomatous change. 5. 5pjut Hj, Fechner RE, Ackerman LV: Tumors of 80ne and

Cartilage. Atlas of tumor pathology, 5econd 5eri eε Fasci­ REFERENCES C/e 5, 5upplement. Armed Forces Institute of Pathology, Washington: 19-23, 1981 1. Abdelwahab IF, Frankel VH , Klein MJ: Case Report 351 16. Huvos AC: 80ne Tumors; Oiagnosiε Treatment and Pro­ 5keletal Radiol 15:164-169, 1986 gnosis. 1st Ed: 18-32, 1979 2. McLeod RA, Dahlin DC, Beabout JW: The 5pectrum of 17. Creenfield CB: Radiology of Bone 9isease. 4th Ed: 625-629, Osteoblastom. AjR 126:321-345, 1976 1986. 3. Cellad FE, Hafiz MA, 81anchard CL: Osteoblastoma of the 18. Dorfman HD, Weiss 5W: 80derline Osteoblastic Tumors; Temporal80ne; CT Findings- Case Report. j Comput Assist Problems in the Oifferential Oiagnosis of Aggressive Tomogr 9:577-579, 1985 Osteoblastoma and Low Crade Osteosarcoma. 5em Oiag 4. Marsh BW, Bonfiglio M, Brady LP et al: 8enign Pathol 1:215-234, 1980 Osteoblastoma; Range of Manifestations. j 80ne joint 5urg 19. Edeiken J: Roentgen Oiagnosis of Oisease of 80ne. 3rd Ed

57-A ’ 1-10, 1975. Vol 1:54-62, 1981 5. 5eki T, Fukuda H, Ishii Y et al: Malignant Transformation 20. Pieterse A5, Roberts BV, Paterson DC et al: Osteoid of 8enign Osteoblastoma- A Case Report. j 80ne joint 5urg osteoma transforming to aggressive (Iow grade malignant)

57-A ’ 424-426, 1975. osteoblastoma; a case report and literature review

6. Jackson JR, Bell ME: 5purious 영enign Osteoblastoma" A Histopathology 7:789-800, 1983. Case Report. j 80ne joint 5urg 59-A:397-401, 1977.

1071