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  Table of Contents    
CASE REPORT  
Year : 2013  |  Volume : 56  |  Issue : 2  |  Page : 169-171
Aggressive osteoblastoma in mastoid process of temporal bone with facial palsy


1 Department of Pathology, Sanjay Gandhi Institute of Medical Science, Lucknow, India
2 Department of Neurosurgery, Sanjay Gandhi Institute of Medical Science, Lucknow, India

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Date of Web Publication23-Sep-2013
 

   Abstract 

Osteoblastoma is an uncommon primary bone tumor with a predilection for posterior elements of spine. Its occurrence in temporal bone and middle ear is extremely rare. Clinical symptoms are non-specific and cranial nerve involvement is uncommon. The cytomorphological features of osteoblastoma are not very well defined and the experience is limited to only few reports. We report an interesting and rare case of aggressive osteoblastoma, with progressive hearing loss and facial palsy, involving the mastoid process of temporal bone and middle ear along with the description of cyto-morphological features.

Keywords: Aggressive osteoblastoma, facial palsy, mastoid, temporal bone

How to cite this article:
Jain M, Rana C, Rajkumar. Aggressive osteoblastoma in mastoid process of temporal bone with facial palsy. Indian J Pathol Microbiol 2013;56:169-71

How to cite this URL:
Jain M, Rana C, Rajkumar. Aggressive osteoblastoma in mastoid process of temporal bone with facial palsy. Indian J Pathol Microbiol [serial online] 2013 [cited 2019 Nov 17];56:169-71. Available from: http://www.ijpmonline.org/text.asp?2013/56/2/169/118684



   Introduction Top


Osteoblastoma is a rare neoplasm of bone comprising of about 1% of all bone tumors. [1] Involvement of calverium is rare and those arising from mastoid process of temporal bone and involving the middle ear are extremely rare. [2] Diagnosis of osteoblastoma by fine needle aspiration (FNA) is rare as in most patients excision biopsy is performed. [3] To our knowledge only 14 cases of osteoblastoma of mastoid region have been reported so far with only six reported cases of facial palsy in osteoblastoma. [3] We are reporting an interesting case of 32-year-old female who presented with rapidly increasing painful mastoid swelling and progressive hearing loss along with facial palsy. We also describe the cyto-morphological features and diagnostic problem encountered during reporting of this entity.


   Case Report Top


A 32-year-old female presented with a rapidly increasing swelling in left mastoid region for 6 months. During initial 2 months there was only pain in mastoid region which was followed by development of swelling. She also complained of progressive hearing loss in the right side along with swaying of the body while walking with tendency to fall to the left side. On physical examination her higher motor functions were normal but there was grade II facial palsy on the left side, left sensorineural hearing loss, and left cerebellar signs. Right ear and facial nerve were intact. On examination of mastoid region there was a hard posterior auricular swelling which was extending toward the occipital and parietal region [Figure 1]a. Magnetic resonance imaging of head with contrast revealed a large temporal lesion, which was hypo intense on T1 and heterogeneously contrast enhancing on T2 with compression of the ipsilateral cerebellar hemisphere [Figure 1]b. No osteolytic changes were appreciated. The lesion was involving the lesser as well as greater wing of sphenoid and also eroding the middle ear and cavity. FNA from mastoid swelling was performed. 3D reconstructed images were also created [Figure 1]c. Cytological smears were moderately cellular and showed presence of atypical cells in groups and rows along with single scattered population in a hemorrhagic background. These atypical cells have eccentric anisomorphic nuclei, dispersed chromatin, conspicuous nucleoli, and abundant amount of cytoplasm. Osteoclastic giant cells were also seen [Figure 1]d and e. Hence on FNA cytology the lesion was diagnosed as positive for atypical cells with a suggestion of osteoblastoma, however, histopathological examination was advised for confirmation. Left reteromastoid suboccipital RMSO craniotomy was done through trans-labyrinthine transcochlear approach for complete excision of left parieto-mastoid bony tumor. Per-operatively a 5 × 5 cm bony hard swelling with a soft tissue part was identified in the left mastoid. Specimen was sent for histopathological examination which revealed a tumor composed of woven bone trabeculae and spicules lined by osteoblasts along with presence of many osteoclastic giant cells. These osteoblasts were plump with a perinuclear clearing and prominent nucleoli [Figure 1]f. The intertrabeculae stroma showed numerous extravasated red blood cells, capillaries, and spindle cells. Final diagnosis on the basis of histopathology was given as epithelioid osteoblastoma. Post-operative scan revealed a complete removal of tumor with no signs of cerebrospinal fluid CSF leak, hydrocephalus, edema or any infarction [Figure 2]. At the time of discharge the patient was ambulatory, conscious, well oriented with a healthy wound.
Figure 1: (a) Post-eriorauricular swelling extending to parieto-occipital region. (b) Coronal contrast enhanced magnetic resonance imaging revealing a heterogeneously enhancing mass in temporal region compressing ispilateral cerebellum. (c) Pre-operative 3D reconstructed image of the mass. (d) Cytomorphology [May Graunwald Giemsa (MGG], ×400): Well differentiated osteoblasts lying in rows and clusters with eccentric nuclei, abundant basophilic cytoplasm and conspicuous nucleoli. (e) osteclastic giant cell (MGG, ×400). (f) Histology of epithelioid osteoblastoma (H and E, ×400): Bony trabeculae lined by plump osteoblasts with more cytoplasm, perinuclear clearing and prominent nucleoli along with presence of many multinucleated osteoclastic giant cells

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Figure 2: Post-operative complete removal of tumor. (a) Magnetic resonance imaging. (b) 3D reconstructed image

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   Discussion Top


Osteoblastoma was first described by Jaffe in 1956, [4] is a rare primary tumor of the bone, representing 0.8-1% of all bone tumors. [1] Any area of the skeleton may be affected by osteoblastoma, but the most common sites of occurrence are the vertebral column and the long tubular bones of the lower extremities. Skull is involved in about 14-20% cases, however, involvement of mastoid region and middle ear is extremely rare. [5] To the best of our knowledge only 14 cases of osteoblastoma of mastoid region have been reported so far. [2] It usually occurs in adolescents and young adults with a male predominance.

Clinical symptoms are non-specific and comprise of insidious onset of pain not responding to anti-inflammatory drugs. When it arises from temporal bone and middle it may cause progressive hearing loss. Facial nerve compression can occur in this setting but it is very rare. [6] There are only six reported cases of osteoblastoma with facial palsy. [7] Diagnosis is usually made on excision biopsy and FNA cytology is rarely performed, thereby limiting the experience on cytomorphological description. [4]

Aggressive osteoblastoma were first described by Dorfman and Weiss. [8] They used the term for osteblastoma where the lining osteoblasts were plump with more cytoplasm, perinuclear clearing and prominent nucleoli. However, they did not mention whether all the tumor cells have to appear epithelioid. Later the term "aggressive osteoblastoma" was used, if the tumor cells had epithelioid appearance throughout. Now this entity is considered as a borderline malignancy between conventional osteoblastoma and osteosarcoma. Aggressive osteoblastoma are locally more aggressive with a higher tendency for recurrence. The present case was also locally aggressive with complete destruction of mastoid region, involvement of middle ear and destruction of internal ear along with compression on cerebellum and extension towards occipital and parietal bone. In such setting clinically there is always a suspicion of osteosarcoma. Sheets of cells with osteoid production and permeation of pre-existing bone are two features that favor diagnosis of osteosarcoma.

Osteoid osteoma is also similar to osteoblastoma but clinically it is characterized by pain that is relieved by salicylate therapy and does not have a soft tissue mass. Histologically no differentiation can be made, so that lesions <1 cm are considered as osteoid osteoma and those >1 cm as osteoblastoma. [6]

Osteoblastoma has the potential for local invasion, aggressive growth and recurrence hence total surgical excision in the treatment of choice. Radiotherapy and chemotherapy may be useful in surgically unresectable cases, however, radiation induced malignant transformation of benign osteoblastoma has been described. [9] To conclude, the present case is an interesting case of aggressive osteoblastoma of mastoid region with middle ear involvement and facial palsy at time of presentation. Hence, osteoblastoma should be one differential in rapidly progressing swelling in posterior auricular region presenting with facial palsy.

 
   References Top

1.Low Y, Foo CL, Seow WT. Childhood temporal bone osteoblastoma: A case report. J Pediatr Surg 2000;35:1127-9.  Back to cited text no. 1
[PUBMED]    
2.Njim L, Dhouibi A, Touil N, Lahmar I, Hadhri R, Moussa A, et al. Benign osteoblastoma in an unusual mastoid location. Eur Ann Otorhinolaryngol Head Neck Dis 2010;127:183-5.  Back to cited text no. 2
    
3.Rhode MG, Lucas DR, Krueger CH, Pu RT. Fine-needle aspiration of spinal osteoblastoma in a patient with lymphangiomatosis. Diagn Cytopathol 2006;34:295-7.  Back to cited text no. 3
[PUBMED]    
4.Jaffe HL. Benign osteoblastoma. Bull Hosp Jt Dis 1956;17:141-51.  Back to cited text no. 4
    
5.Pérez-Mora RM, Sanchez-Carrion S, Barea FL, Garcia-Raya P, Lassaletta L. Osteoblastoma of the temporal bone. Skull Base 2009;19:345-8.  Back to cited text no. 5
    
6.Khashaba A, De Donato G, Vassallo G, Landolfi M, Russo A, Taibah A, et al. Benign osteoblastoma of the mastoid part of the temporal bone: Case report. J Laryngol Otol 1995;109:565-8.  Back to cited text no. 6
    
7.Naclerio RM, Jenkins HA, Herzog JA. Osteoblastoma of the temporal bone presenting as facial paralysis. Int J Pediatr Otorhinolaryngol 1985;8:253-62.  Back to cited text no. 7
[PUBMED]    
8.Dorfman HD, Weiss SW. Borderline osteoblastic tumors: Problems in the differential diagnosis of aggressive osteoblastoma and low-grade osteosarcoma. Semin Diagn Pathol 1984;1:215-34.  Back to cited text no. 8
[PUBMED]    
9.Potter C, Conner GH, Sharkey FE. Benign osteoblastoma of the temporal bone. Am J Otol 1983;4:318-22.  Back to cited text no. 9
[PUBMED]    

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Correspondence Address:
Manoj Jain
Department of Pathology, Sanjay Gandhi Post Graduate Institute of Medical Sciences. Lucknow - 226 014, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.118684

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