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Turkish Neurosurgery 6: 88 - 92, 1996 Durmaz: Osteob/astoma of tlze Cervica/ Spiiie Osteo blastoma Histopathological Of and The Cervical Radiological Spine. Correlation Servikal Omurga Osteoblastomu. Bulgu Radyolojik Iliskisi ve Histopatolojik RAMAZAN DURMAZ, HAKAN BOZOGLU, SARE KABUKÇUOGLU, ERKAN VARDARELI, ESREF TEL Osmangazi University Faculty of Medicine, Oepartments of Neurosurgery (RO,HB,ED, Pathology (SK), and Nuclear Medicine (EV),Eskisehir, Turkey Abstract: This is the case report of an eight-year old boy with a benign osteoblastoma of the C3 vertebra. The clinical, radiologicaL, gross and histopathological findings concerning the tumor are sufficiently characteristic and exclusive to justify the diagnosis of "osteoblastoma". Key Words: Osteoblastoma, scintigraphy, spine, surgery. INTRODUCTION Osteoblastoma is arare, vaseular, and progressively expansive, benign tumor of the bone, and has a 34.41 % predileetion for the spine (7,8,15). Sinee the lesion is histologieally indistinguishable from osteoid osteoma, a differential diagnosis was ma de on the basis of natural history, lesion size and radiological images (3,7,8,15,16). The majority of the cases is under the age of 30, with a peak in the second deeade of life (8,15). In this report, it is aimed to draw attention to the applieation of vertebral angiography in primary eervieal spine neoplasni.s, for aseertaining the existenee or absenee of any vaseular eomplieations CASE REPORT An eight-year old ehild with a history of ne ek pain over a six month period was admitted to the Neurosurgieal Department of Medical Faeu1ty of Osmangazi University in Deeember 1994. He was 88 Özet: Servikal üçüncü omurgada benigin osteoblastomu olan sekiz yasinda bir erkek hasta sunulmaktadir. Bu tümörün klinik, radyolojik, makroskopik ve mikroskopik patolojik bulgulari 'osteoblastoma' tanisini kesinlikle koyduracak kadar kendine özgüdür. Anahtar sözcükler: Cerrahi, omurga, osteoblastoma, sintigrafi suffering insidious pa in during the day whieh intensified at least twiee during the night. Salieylates provided some relief from the pain. The neurologieal examination show ed no abnormality; however, a eervieal postero-anterior plain radiogram revealed a well- cireumseribed ovallesion on the pedides of the C2, C3, and C4 vertebrae on the right (Figure 1). Further, although the tumor itself was not visible in the other projeetions, a narrowing second intervertebral foramen appeared on the right oblique film, and the lateral radiogram revealed loss of eervieal eurvature. A whole-body seintigram performed three hours af ter an intravenous injection of 10 mCi Te-99 m methylene diphosphonate (MDP) revealed a well-demareated, hyperaetive round lesion on the third eervical vertebral body (Figure 2). A eomputerized tomography (CT) sean of the eervieal spine showed a patehy, ossified nidus, surounded by a bone shell, situated on all the

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Turkish Neurosurgery 6: 88 - 92, 1996 Durmaz: Osteob/astoma of tlze Cervica/ Spiiie

Osteo blastoma

Histopathological

Ofand

The Cervical

RadiologicalSpine.

Correlation

Servikal Omurga Osteoblastomu.Bulgu

RadyolojikIliskisi

ve Histopatolojik

RAMAZAN DURMAZ, HAKAN BOZOGLU, SARE KABUKÇUOGLU,

ERKAN VARDARELI, ESREF TEL

Osmangazi University Faculty of Medicine, Oepartments of Neurosurgery (RO,HB,ED,Pathology (SK), and Nuclear Medicine (EV),Eskisehir, Turkey

Abstract: This is the case report of an eight-year old boywith a benign osteoblastoma of the C3 vertebra. Theclinical, radiologicaL, gross and histopathological findingsconcerning the tumor are sufficiently characteristic andexclusive to justify the diagnosis of "osteoblastoma".Key Words: Osteoblastoma, scintigraphy, spine, surgery.

INTRODUCTION

Osteoblastoma is arare, vaseular, andprogressively expansive, benign tumor of the bone,and has a 34.41 % predileetion for the spine (7,8,15).Sinee the lesion is histologieally indistinguishablefrom osteoid osteoma, a differential diagnosis wasma de on the basis of natural history, lesion size andradiological images (3,7,8,15,16). The majority of thecases is under the age of 30, with a peak in the seconddeeade of life (8,15). In this report, it is aimed to drawattention to the applieation of vertebral angiographyin primary eervieal spine neoplasni.s, for aseertainingthe existenee or absenee of any vaseulareomplieations

CASE REPORT

An eight-year old ehild with a history of neekpain over a six month period was admitted to theNeurosurgieal Department of Medical Faeu1ty ofOsmangazi University in Deeember 1994. He was

88

Özet: Servikal üçüncü omurgada benigin osteoblastomuolan sekiz yasinda bir erkek hasta sunulmaktadir. Butümörün klinik, radyolojik, makroskopik ve mikroskopikpatolojik bulgulari 'osteoblastoma' tanisini kesinliklekoyduracak kadar kendine özgüdür.Anahtar sözcükler: Cerrahi, omurga, osteoblastoma, sintigrafi

suffering insidious pa in during the day whiehintensified at least twiee during the night. Salieylatesprovided some relief from the pain. The neurologiealexamination show ed no abnormality; however, aeervieal postero-anterior plain radiogram revealeda well- cireumseribed ovallesion on the pedides ofthe C2, C3, and C4 vertebrae on the right (Figure 1).Further, although the tumor itself was not visible inthe other projeetions, a narrowing secondintervertebral foramen appeared on the right obliquefilm, and the lateral radiogram revealed loss ofeervieal eurvature.

A whole-body seintigram performed threehours af ter an intravenous injection of 10 mCiTe-99 m methylene diphosphonate (MDP) revealeda well-demareated, hyperaetive round lesion on thethird eervical vertebral body (Figure 2).

A eomputerized tomography (CT) sean of theeervieal spine showed a patehy, ossified nidus,surounded by a bone shell, situated on all the

Turkish Neurosurgery 6: 88 - 92, 1996 Durmaz: Osteob/astoma of the Cervica/ Spine

transverse processes and one third of the lamina ofC3 vertebra with cannectian to C2 and C3 vertebrae.

A soft component of the mass was visible betweenthe posterior border of nidus and lamina. Areactivebone formatian at the lamina and the tip of thetransverse process of the C3 vertebra was alsaobserved (Figure 3).

b

Figure 3: CT scan showing a) the ossificated nidus ofthe lesion on the lateral part of C3 vertebra,and b) the new bone formatian at the laminaand the tip transverse process (arrows). Thesoft mass between lamina and nidus is alsaobserved.

Figure 1:Posterior- anterior radiogram of the cervical spineshows the radiolucent area surrounded by a slightsderotic rim on the right pedide of C2, C3 andC4 vertebrae.

Figure 2: Bone scintigraphy showing an intense focal uptakeof radionudide by lesion on the C3 vertebrae.

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Tiirkish Neiirosiirgery 6: 88 - 92, 1996

As the tumor destroyed the right foramentransversarium, a vertebral angiogram wasperformed. This showed that the mass had causedvertebral artery displacement, with a segmentalnarrowing at the tumor level (Figure 4).

Figure 4: Right vertebral angiogram shows that the tumorcauses vertebral artery displacement anteriorlywith a segmental narrowing at the tumor leveL.

To explore the vertebral artery in the uppercervical region, a far lateral approach with thepatient in sitting position, as defined by Spetzlerand Graham, was found to be the most appropriate(24). On removing laminae of Cl, C2, and C3vertebra on the affected side, a soft, yellow-grey­reddish pathological tissue of a friable, granularconsisteney was observed, most distinctivelyunder the lamina of the C3 vertebra. Curettage ofthis entire soft mass exposed a hard nidus,approximately 2 cm in diameter, which was thencompletely removed. During surgery vertebral arterywas seen to have been displaced anteriorly under thishard component of tumor and so was carefullyseparated from it.

A subsequent histopathological examinationof the tumor confirmed it to be osteoblastoma

(Figure 5). A CT sean and wholebody scintigramwere performed one month after operation, andno residual tumor was detected. During the oneyear of follow-up, the patient remained in goodhealth.

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Diirmaz: Osteob/astoma of the Ceruica/ Spiiie

Figure 5: Histopathological examination of the lesionshowing the following: 1. Well separated,irregularly serrated osteoid and woven bonetrabeculae. 2. One to three cell layers of plumposteoblasts in dos e apposition to woven bonetrabeculae. 3. Numerous osteodasts sprinkled onthe surface of the trabeculae. 4. Stroma composedof loose spindle cells and prominent capillaries(H&E, X 200).

DISCUSSION

Osteoblastoma of the spine arises mostcommonly from the posterior elements (15,16,19,22).In this case, however, the tumor involved the postero­lateral aspect of the vertebrae, and had causedvertebral artery displacement, with a segmentalnarrowing, without causing any clinical symptom.Vertebral artery involvement generally occurs inmalignant neoplasms, as well as in massiye benigntumors, such as osteoblastoma, aneurysmal bonecysts and giant cell tumors of the cervical spine(1,2,4,6,23). Bohlman et aL.noted a complication dueto vertebral artery distraction during removal of theneoplasm in the cervical spine , and suggested that incases where the tumor is located laterally, the extentof vertebral artery involvement should be defined byarteriogram (2).This is ofbenefit to surgical planning,as well as to preserving the artery during surgery. Incases with vertebral artery involvement the antero­lateral or postero-lateral routes are used (2,23);in thisease , a far lateral approach was found appropriate,not only to remove the laminae on the affected side,but also in order to gain a better view on the transverseprocess es and pedic1es.

Histologieally, osteogenie sareoma may bemisdiagnosed as an osteoblastoma (14). In some easesof recurrent osteoblastoma sarcomatous changes are

Turkish Neiirosurgery 6: 88 - 92, 1996

found that are probably due to initial misdiagnosisof a low-grade maIignant lesion (9).The features thatwere helpful in distinguishing this particular casefrom osteosarcoma were 1) absence of anaplasia, 2)focal osteoblastic rimming, 3) absence of cartilageproduction, and 4) sharply circumscribed edge ofthe lesion from the host lameller bone, and the lackof permeative pattem (7). On the other hand, severalcases of osteoblastoma with same features have

shown an aggressive behaviour to justify the clinicalterm "aggressive" as opposed to "maIignant" (10).Osteoblastoma must also be differentiated from

fibrous dysplasia; in the latter, new bone growth isnot usually rimmed by a prominent, single-rowosteoblast (17). In our case there was a rim ofosteoblasts 1-3 cells in thickness.

Although osteoblastoma in the thoracal andlumbar spine is frequently associated with painfulscoIiosis (1,11,19,22), neck pain may be the only signin a cervical lesion (5).Of patients with osteoblastomain the spine, including the subject of this report, 13% were reported to have noctumal pain (16).

On plain radiogram, osteoblastoma is generallyfound to be osteolytic, although some cases areosteosclerotic and some show a combination of two.A well-circumscribed radiolucent area surrounded

by a slight sclerotic rim is also generally observed(3,12,15,16,21). The fact that this image appearedclearly on posteroanterior film in this case, suggeststhat radiograms should be taken at four projections.

Bone scintigraphy is recommended where thereis a suspicion of osteoblastoma or osteoid osteoma,even if these are not visible on the plain radiograms(13,18,20). No false negatiye results have beenreported 0,19,22). Bone scintigraphy was appIiedintraoperatively and postoperatively, for detectionof any residual tumor tissue 03,25). In this case, theteehnique was used not only for supporting thediagnosis but also for evaluating the extent of thetumor removaL.

According to Mohan et aL.,it is possible to makea differential diagnosis between osteoblastoma andosteoid osteoma by CT sean; osteoblastoma isdistinguished by an expanding lesion, with evidenceof new bone formation and varying degrees ofealcification (18). Our case conforms with previousreports with respect to scanty ossifieation of matrix,surrounded by a shell of bone and a soft componentof lesion that are only visible by CT scan (12,18).

Durmaz: Osteob/astoma of the Cervica/ Spiiie

Osteoblastoma can be completely eured by en­bloc resection (8,9,15). In conclusion, early detectionof osteoblastoma in the spine is essential for theprevention of neural or vascular complications, andfor preserving the stability of the spinal eolumn.

Correspondence: Dr. Ramazan DurmazVisnelik Mh. Ali Fuat Güven Cd.Önçag Sitesi B-Blok 0:2026020, Eskisehir, TurkeyTelephone: (222) 239 29 79

REFERENCES

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2. Bohlman HH, Sachs BL, Carter JR, Riley L, RobinsonRA: Primary neoplasms of the cervical spine: Diagnosisand treatment of twenty-three patients. J Bone JointSurg 68-A:483-94,1986

3. Dias L de S, Frost HM: Osteoblastoma of the cervicalspine: A reviewand report of eight new cases. ClinOrthop 91:141-51,1973

4. Goel A, Bhayani R, Nagpal RO: Massive benignosteoblastoma of the eliyus and atlas. Br J Neurosurg8:483-86,1994

5. Grop D, Loehr J: Osteoblastoma of the cervical spine:Case report. Arch Orthop Trauma Surg 108:179-81,1989

6. Hay MC, Paterson D, Taylor TKF: Aneurysmal bonecysts of the spine. J Bone Joint Surg 60-B:406-11,1978

7. Huvos AG: Bone Tumors, 2nd edition, Philadelphia;WB Saunders, 1991:67-83

8. Jackson RP, Reckling FW, Mantz FA: Osteoid osteomaand osteoblastoma: Similar histologic lesions withdifferent natural histories. Clin Orthop 128:303-13,1977

9. Jackson RP: Recurrent osteoblastoma: A review. ClinOrthop 131:229-33,1978

10. Kenan S, Floman Y, Robin GC, Laufer A: Aggressiveosteoblastoma: A case report and review of theliterature. Clin Orthop 195:294-98,1985

11. Kirwan EO'G, Hutton PAN, Pozo JL, Ransford AO:Osteoid osteoma and benign osteoblastoma of thespine: Clinical presentation and treatment. J Bone JointSurg 66-A:21-26,1984

12. Kroon HM, Schurmans J: Osteoblastoma: Clinical andradiologic findings in 98 new cases. Radiology 175:783­90,1990

13. Mandeii GA, Hareke HT: Scintigraphy of spinaldisorders in adolescents: Review. Skeletal Radiol22:393-401,1993

14. Marsh HO, Choi C-B: Primary osteogenic sareoma ofthe cervical spine originaiiy mistaken for benignosteoblastoma: A case report. J Bone Joint Surg 52­A:1467-71,1970

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Turkish Neurosurgery 6: 88 - 92, 1996

15. Marsh BW, Bonfiglio M, Brady LP, Enneking WF:Benign Osteoblastoma: Range of manifestations. J BoneJoint Surg 57-A:1-9,1975

16. Meleod RA, Dahiin DC, Beabout JW: The spectrum ofosteoblastoma. AJR 126:321-35,1976

17. Mirra JM, Picci P, Gold RH: Bone Tumors, 2nd edition,Philadelphia; Lea & Febiger, 1989: 389-432

18. Mohan V, Sabri T, Markiund T, Sayed M, Gupta RP:Clinicoradiological diagnosis of benign osteoblastomaof the spine in children: Current problem cases. ArchOrthop Trauma Surg 110:260-64,1991

19. Myles ST, Macrae ME: Benign osteoblastoma of thespine in childhood. J Neurosurg 68:884-88,1988

20. Papanicolaou N, Treves S: Bone scintigraphy in thepreoperative evaIuation of osteoid osteoma andosteoblastoma of the spine. Ann Radiol Paris 27:104­10,1984

Durmaz: Osteoblastoma of the Cervical Spi/ie

21. Pochaczevsky R, Yen YM, Sherman RS: The roentgenappearance ofbenign osteoblastoma. Radiology 75:429­37,1960

22. Pettine KA, Klassen RA: Osteoid osteoma andosteoblastoma of the spine. J Bone Joint Surg 68-A:354­61,1986

23. Sen C, Eisenberg M, Casten AM, Sundaresan N,Catalano PJ: Management of the vertebral artery inexcision of extradural tumors of the cervical spine:Technique and application. Neurosurgery 36:106­15,1995

24. Spetzler RF, Grahm TW: The far lateral approach tothe inferior eliyus and the upper cervical region. BNIQ 6:35-38,1990

25. Sty J, Simons G: Intraoperative 99m technetium boneimaging in the treatment ofbenign osteoblastic tumorsoClin Orthop 165:223-27,1982.

Internet Web Site

Turkish Neurosurgical Society

http://www.ankara.edu.tr/r-.Jtnd

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