Cementoma of the calcaneus: a case report
© The Author(s). 2017
Received: 7 November 2016
Accepted: 28 February 2017
Published: 14 March 2017
The cementoma is a common disease of the dental root apex, which generally occurs in the maxilla and the mandible, but the cementoma occurring in the long bone is rare. Moreover, the incidence of cementoma in the calcaneus is extremely infrequent.
The present study reports an unusual case of a 19-year-old girl, who complained of pain in the left heel. Subsequent radiographs and computed tomography (CT) were used in the diagnosis. The imaging features of the lesion included a radiopaque matrix and radiolucent tissue, particularly an arc-shaped fat band. An excisional biopsy was performed. Histopathological examination confirmed the diagnosis of cementoma in the calcaneus. After the operation, the patient was followed up without recurrence.
Imaging examination plays an important role in the differential diagnosis of cementoma of the calcaneus.
KeywordsCementoma Calcaneus Radiography Computerized tomography
Extragnathic cementoma of long bone as a benign bone neoplasm is very rare, which was reported to mainly locate in the lower limb in the literature [1–4]. In 1969, Friedman and Goldman first reported cementoma of long bones . To 2005, there were 10 cases reports about this tumor of long bones in total .
We report an extragnathic cementoma of the calcaneus in a 19-year-old woman, who presented with intermittent pain at the left heel half a year ago. The imaging features including radiography and computerized tomography (CT) are described in our study. To the best of our knowledge, no report has previously introduced this tumor occurring in the calcaneus. Written informed consent was obtained from the patient.
According to the World Health Organization (WHO), the classification of odontogenic tumor keeps changing [6, 7]. Cementoma, as a common benign odontogenic tumor, generally occurs in the maxilla and the mandible . Cementoma occurring in the long bone is the rare bone lesion [1, 8]. There is a controversy in the cementoma located in the long bones, due to the reason that many authors do not accept the existence of true cementum outside the gnathic region. Mirra et al.  thought cementoma of long bone is merely a form of simple bone cysts. In 1985, Adler  found there was calcified cementum-like material accompanied by few fibroblasts in the solitary bone cysts. They thought cementoma was originally bone cysts which calcified steadily with age . This tumor in the long bone has been reported in three separate types of lesions , including a simple bone cyst with associated cementum-like particles in the cyst lining, calcified cementum such as is present in our patient, cementifying fibroma. To date, there are only 10 reported cases of cementomas occurring in the extragnathic long bones [2, 4, 5]. In addition, Kolar et al.  first reported cementoma of the metacarpus occurring in the short tubular bone. So far the case of the calcaneus has not been reported in the literature. The present case which belongs to extragnathic irregular bones is the first report of cementoma occurring in the calcaneus.
In the English literature, the radiographic appearances in the cementomas of the long bones were quite similar . The lesions were mostly located in the center of the medullary cavity, and mainly diaphyseal-metaphysis . Most tumors appeared as well-defined, expansive, radiolucent lesions with radiopaque matrix . The pathologic specimen consists of cementum corpuscle as is present in the cementomas of the jaw bones .
This new observation of this rare entity found in our patient differs from others by its localization (calcaneus medullary, not long bone). The radiographic and microscopic appearance of the cementoma found in the present patient was consistent with the findings reported by previous authors. On radiograph, the lesion presented a cavity surrounded by an incomplete sclerotic rim and a radiopaque mass was present within it. On CT, it was cortical expansive, and had a well defined sclerotic border, peripheral radiolucency, and an amorphous eccentric focus of matrix calcification. These radiographic features resembled those of their maxillary counterparts and other previously reported lesions of the long bones containing cementum or cementum-like material [1, 9, 10]. One noteworthy feature of the lesion from the imaging studies should be mentioned that is the peripheral radiolucency seen on CT images around the calcified matrix. Some reports suggested that the peripheral radiolucency tissue was in a transitional stage , or the absence of calcified spherules within the fibrous tissue on the periphery of the lesion . However, it is a pity that we could not find any literature to support this manifestation of radiolucency of tumor tissue with an arc-shaped fat band. Therefore, we present this rare case as an effort to further characterize the lesion and to facilitate the diagnosis of similar cases.
Radiologically, cementoma of extragnathic bone has been classified into three stages [12–14]: osteolytic stage with cystic low density, cementum formation stage with mass shadow in the lesion and mature stage. Characteristic mature stage is also called inactive stage which has high clinical value, such as our case.
The radiographic differential diagnosis of cementoma of the calcaneus includes enchondroma , osteoblastoma , bone infarction , intra-osseous lipoma  and calcifying solitary bone cyst . Enchondroma usually grows in the center of the medullary cavity of calcaneus with an irregular bone protuberance with a wide base and contains characteristic chondroid-like calcifications without adjacent sclerotic rim [4, 15]. However cementoma of the calcaneus displays a well-defined, sclerotic rim surrounding the lesion. Osteoblastoma shows a clear thin shell-shaped calcifications surrounding the lesion with low density bone expansion and destruction and soft tissue infiltration . Bone infarction usually displays map-shaped lesion in the center of the calcaneus . The lytic areas are permeated by focal or diffuse medullary osteosclerosis but not affected the cortex. Intra-osseous lipoma  shows the lesion divided by fibrous septa and an almost uniform density of fat. CT examination is a helpful method to rule it out. Generally, it is difficult to distinguish between diagnosis of calcifying solitary bone cyst and cementoma , especially for the cementum of osteolytic stage. Calcifying solitary bone cyst is a changed solitary bone cyst, and associated with symptoms such as swelling, deformation and pathological fracture . Calcifying solitary bone cyst displays the lesion, a fluid filled structure, with cortical thinning or destruction of the calcaneus with small or multiple foci calcifications. In cementoma, however, the calcification is an amorphous central or eccentric cementum-like matrix.
Limitations of the study
Since this study was a retrospective study, no MR examination was done and intraoperative photos were not acquired.
We first present a rare case of cementoma of the calcaneus. When compared with dental cementoma, cementoma of the extragnathic bone is rare, mainly occurring in the long bone. The radiologic imaging features of this lesion, including the radiopague deposit and radiolucent tissue with an arc-shaped fat band should be concerned by the radiologists. When cementum corpuscle was detected, the diagnosis of cementoma should be considered. We report this case and hope to provide informative data that confirm that the cementoma of the extragnathic bone exists as a distinct entity.
World Health Organization
We thank for Kai Sheng who gave the help for pathological diagnosis.
Availability of data and materials
This is a case report of a single patient. To protect privacy and respect confidentially, no raw data have been made available in any public repository. The original operation reports, imaging studies, and outpatient clinic record are retained as per normal procedure within the medical records of our institution.
ZZ* is in charge of the main idea and is the guarantor of integrity of the entire clinical study; XL# and ZP# are in charge of the study concepts, design, manuscript preparation and editing; ML and AL, are in charge of the language polishing and the grammar revision; WC is in charge of the collection of the study data. All authors read and approved the final manuscript.
The authors declare that they have no competing interests.
Consent for publication
We confirm that the patient has given his written consent for the case report to be published.
Ethics approval and consent to participate
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