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British Journal of Oral and Maxillofacial Surgery 51 (2013) 996–997
Journal oracle
Denosumab osteonecrosis of the mandible: a new entity? A case report. Pichardo SEC, Kuypers SCC, Richard van Merkesteyn JP. J CranioMaxillofac Surg 2013;41(4):e65–9
In the treatment of osteoporosis, and bone metastases from prostate and breast cancer bisphosphonates play a major role. Not all patients respond well to bisphosphonate treatment. Since a few years adverse effects of these drugs have been reported. A new drug, denosumab, a fully human monoclonal antibody to RANKL, has recently been developed. This case reports a 74-year-old male patient with a medical history of diabetes mellitus, angina pectoris, coronary bypasses, hypertension, and prostate cancer with multiple metastases to lymph nodes, bone and lungs. The prostate cancer was treated according to the protocol. But he was never treated with bisphosphonates. Instead he was included in a phase III randomized double blind multicenter trial, testing the efficacy of denosumab compared to zoledronic acid in the treatment of bone metastases of hormone resistant prostate cancer. Only 7 months after start of denosumab infectious symptoms developed, followed by infestation of the mandible. Despite surgical treatment fistula and exposed bone remained. This case illustrates that use of denosumab can lead to 0266-4356/$ – see front matter
http://dx.doi.org/10.1016/j.bjoms.2013.08.006
a type of osteonecrosis resembling bisphosphonate related osteonecrosis of the jaws. Floor-of-mouth dermoid cysts: report of 3 variants and a suggested change in terminology. Gordon PE, Faquin WC, Lahey E, Kaban LB. J Oral Maxillofac Surg 2013;71(6):1034–41 Dermoid cyst is a frequently used descriptive term, but its definition changes according to the clinician (e.g. dermatologists, neurologists, gynecologists, oral and maxillofacial surgeons, and plastic surgeons). It is sometimes used synonymously with teratoma. In oral and maxillofacial surgery, it is applied to describe congenital floor-of-mouth cysts of 3 histologic types: epidermoid, dermoid, and teratoid. This terminology is confusing and has led to some ambiguity in the literature. The purpose of this report is to document 3 cases illustrating the utility of a more specific term, congenital germline fusion cyst of the floor of the mouth. Patients who presented with floorof-mouth swelling were evaluated by history, clinical examination, and imaging studies (magnetic resonance imaging and computed tomography). Three cases (1 epidermoid variant, 1 dermoid variant, and 1 teratoid variant) are documented to illustrate the new terminology. Congenital germline fusion cyst is more reflective of the embryologic
origins of the lesion than dermoid cyst and is inclusive of all 3 histologic variants. Dermoid cysts are commonly found in the skin overlying the anterior fontanelle, bregma (junction of the coronal and sagittal sutures), upper lateral region of the forehead, lateral upper eyelid, and submental region. However, they can occur anywhere on the scalp, face, spinal axis, and other body sites. The etiopathogenesis is thought to be an abnormal infolding of the ectodermal layer (hence, the other often used term, inclusion cyst) along normal embryonic lines of fusion of the facial processes or within the neural axis. In the craniofacial region, dermoid cysts develop during fusion of the first and second pharyngeal arches between the third and fourth weeks of gestation. Grossly, they contain a thick sebum-like material; histologically, skin appendages are often present within the cyst wall. Floor-of-mouth dermoids are rare. In 1937, New and Erich3 published a series of 1495 patients with dermoid cysts, and only 7% (105) were found in the head and neck. Of these 105 lesions, 49.5% were confined to the periorbital region and 23.3% were found in the floor of the mouth. Meyer classified congenital floorof-mouth cysts into 3 variants by histologic criteria: (1) epidermoid (epithelial-lined cyst without adnexal structures); (2) dermoid
Journal oracle / British Journal of Oral and Maxillofacial Surgery 51 (2013) 996–997
(epithelial-lined cyst with dermal appendages, such as hair follicles, sebaceous glands, or sweat glands, in the underlying connective tissue); and (3) teratoid (epithelial-lined cysts containing epithelial and nonepithelial elements, such as bone, muscle, respiratory, and gastrointestinal tissue). Teratoma is defined as a germ cell tumor containing elements of all 3 germ cell layers: ectoderm, mesoderm, and endoderm. Teratomas may be benign or
malignant and often have cystic components. The term dermoid cyst also has been used interchangeably with teratoma, leading to even more inconsistency in the literature. To be more precise and clear, the authors suggest substituting congenital germline fusion cyst for dermoid cyst as a general term and adding the specific variant to complete the description. The authors classify these cysts as congenital because, based on embryology, they are present at birth, although they may
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not be clinically evident until later in life. This is analogous to a deep vascular malformation, which is present at birth but might not become evident until years later. The term fusion is used because the cysts develop during fusion of the first and second pharyngeal arches between the third and fourth weeks of gestation. In this report, the authors describe 3 patients with floorof-mouth congenital germline fusion cysts, 1 with each variant, to illustrate and clarify their differences.