Indian Journal of Pathology and Microbiology
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Year : 2012  |  Volume : 55  |  Issue : 2  |  Page : 230-232
Primary plemorphic adenoma of bone: Report of a case and literature review

1 Department of Pathology, Taizhou People's Hospital, Taizhou, Jiangsu Provience, China
2 Department of Oncology, Fudan University Shanghai Cancer center, Shanghai, China
3 Department of Pathology, Shanghai Medical College, Fudan University, China

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Date of Web Publication3-Jul-2012


Plemorphic adenoma mainly occurs in the salivary glands. A similar tumor arising in the bone is extremely rare, and only three cases have been reported so far. Here, we present an additional case of primary plemorphic adenoma of bone; describe its clinical, pathological and radiological features; and compare them with those of the reported cases.

Keywords: Diagnosis, differential diagnosis, plemorphic adenoma

How to cite this article:
Yu H, Liu X, Li H, Shi D, Wang C. Primary plemorphic adenoma of bone: Report of a case and literature review. Indian J Pathol Microbiol 2012;55:230-2

How to cite this URL:
Yu H, Liu X, Li H, Shi D, Wang C. Primary plemorphic adenoma of bone: Report of a case and literature review. Indian J Pathol Microbiol [serial online] 2012 [cited 2022 Jan 28];55:230-2. Available from: https://www.ijpmonline.org/text.asp?2012/55/2/230/97890

   Introduction Top

Plemorphic adenoma is a common salivary gland neoplasm, and is less frequently seen in other parts of the body. The neoplasm primarily arising in bone is extremely rare. Here, we present one case, which occurs in the thigh.

   Case Report Top

The patient, a 35-year-old man, sought medical observation because of deep pain over his left knee for 2 months. Physical examination detected a swelling, tender mass over the distal end of the left thigh. Magnetic resonance imaging (MRI) revealed a multilobulated expansile lytic lesion with size 6 cm × 5 cm involving the metaphysis and epiphysis with local cortical destruction at the distal end of the left femur. There was no area of calcification in the lesion. Periosteal interaction and true extension into soft tissue were not detected [Figure 1]. General physical examination through the body and computed tomographic (CT) scans of the chest, abdomen-pelvis and head and neck were done. No other mass was found but the left femur. Laboratory tests demonstrated no abnormalities. Curettage of the lesion was carried out. The patient was not treated with chemotherapy or radiation therapy after surgery. At 16 months after surgery, he was free of disease and is undergoing follow-up.
Figure 1: Magnetic resonance images of a multilobulated mass in the left femur with an approximately sharp periphery

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Grossly, the removed specimen was fragmental with a total size of 4.0 cm × 2.5 cm × 1.0 cm. The cut surface was homogeneous and white, with a focal glistening appearance and a few fragmental bones. Histologically, the lesion showed a multilobulated arrangement composed mainly of epithelia and cartilaginous matrix. The epithelial cells were round, polygonal, plasmacytoid or short spindle and arranged in nests, sheets or single cells set in the hyalinized matrix. Local keratinization indicated epithelial squamous differentiation. The nuclei were uniformly bland with small nucleoli and a low mitotic frequency. Cartilaginous matrix was blue and hypocellular, interweaved with the epithelial component. Locally, the host bone was surrounded by the tumor tissue, indicating a local infiltrating growth pattern [Figure 2].
Figure 2: Histopathologic features of the present case. The lesion shows blue chondroid matrix with a local invasion of host bone (hematoxylin and eosin, ×100)

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Immunohistochemically, the squamous differentiating element was positive for cytokeratin (AE1/AE3), 34betaE12 and p63; the plasmacytoid and short spindle components were positive for vimentin, p63 and S-100 protein in addition to AE1/AE3 and 34betaE12; and the cartilaginous area was positive for vimentin and S-100 protein [Figure 3] and [Figure 4]. α-SMA and desmin were negative in all components. Based on basis of pathologic, immunohistochemical features and the clinical findings, the tumor on the left femur was diagnosed as primary plemorphic adenoma.
Figure 3: Immunochemical staining of the present case. Neoplasm cells are positive for AE1/AE3 two-step method (IHC, ×200)

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Figure 4: Immunochemical staining of the present case. Neoplasm cells are positive for S-100 protein two-step method (IHC, ×200)

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Plemorphic adenoma is the most common salivary gland neoplasm, and is less frequently seen in other parts of the body. To our knowledge, only three case of plemorphic adenoma primarily arising in bone have been reported. The first case was reported by Hirsch and Helwig [1] in 1961. The reported patient had two lesions: one was located anteriorly in the plantar aspect of the left foot and the other in the tibia of the left leg. The lesion in the foot was excised and the other was only biopsied. The patient was in good condition at 60 months after diagnosis. The two lesions were thought to be independent primary tumors rather than a metastasis of the plantar lesion to the tibia, or vice versa. In 2001, de Pinieux et al. [2] reported on a 44-year-old woman with a tumor whose morphologic and immunophenotypic features of the biopsy specimens were consistent with a plemorphic adenoma. No other lesion in her body was found by clinical and radiographic examinations. At 12 months after the lesion had been resected, a local recurrence appeared. An en bloc resection of the recurrence mass with allograft reconstruction was done. Throughout the 4 years after the second resection, the patient was asymptomatic and staging studies continued to show no evidence of mass. In 2001, McGough et al. [3] reported one more case, a 42-year-old woman who had the tumor in the proximal part of the tibia. Resection of the lesion was performed. Two years after resection, local recurrence occurred. The resection of the recurrence lesion was performed. One year after the second resection, radiographs of the tibia showed no signs of local recurrence. A computed tomography (CT) scan of the chest, however, revealed multiple nodules in the right lower lung field. A CT-guided biopsy of one nodule in the lung was performed, and morphology of the lesion still showed a diagnosis of plemorphic adenoma with a "benign appearance" identical to that of the tibial lesion, which suggested metastasis.

Most of the extraskeletal plemorphic adenomas are believed to be benign, but a small subset exhibit aggressive behavior, especially in the cases with an infiltrative margin. [4] Because of the infrequence of plemorphic adenoma of bone, its behavior is hardly known. We think that primary plemorphic adenoma of bone may have individual behaviors. As mentioned above, primary plemorphic adenoma of bone with a benign appearance could present local recurrence, even distant metastasis. We suggest that a long-term follow-up management should be carried out for the patient with primary plemorphic adenoma of bone, even if when the lesion shows a "benign appearance." Identification of the exact biobehavioral criteria for this disease entity must be determined on the basis of more patient cases. In the present case, at 16 months after surgery, he was free of disease and is undergoing follow-up.

   References Top

1.Hirsch P, Helwig EB. Chondroid syringoma, mixed tumour of skin, salivary gland type. Arch Dermatol 1961;84:835-47.  Back to cited text no. 1
2.de Pinieux G, Beabout JW, Unni KK, Sim FH. Primary mixed tumour of bone. Skeletal Radiol 2001;30:534-6.  Back to cited text no. 2
3.McGough RL, Wang LJ, Gnepp DR, Terek RM. Metastatic mixed tumor arising in bone. A case report and review of the literature. J Bone Joint Surg AM 2001;83-A:1396-402.  Back to cited text no. 3
4.Kilpatrick SE, Hitchcock MG, Kraus MD, Calonje E, Fletcher CD. Mixed tumours and myoepitheliomas of soft tissue. A clinicopathologic clinicopathologic study of 19 cases with a unifying concept. Am J Surg Pathol 1997;21:13-22.  Back to cited text no. 4

Correspondence Address:
Chaofu Wang
Department of Pathology, Cancer Hospital of Fudan University, 27 Dong An Road, Shanghai, 200 032
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0377-4929.97890

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  [Figure 1], [Figure 2], [Figure 3], [Figure 4]


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