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首页> 外文期刊>Dental and Medical Problems >Tetrad presentation of non-syndromic odontogenic keratocyst: An uphill diagnostic and therapeutic challenge
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Tetrad presentation of non-syndromic odontogenic keratocyst: An uphill diagnostic and therapeutic challenge

机译：非综合征牙源性角化囊肿的四联表现：艰难的诊断和治疗挑战

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Odontogenic keratocyst (OKC), in the last decade sceptically referred to as keratocystic odontogenic tumor (KCOT), is known for its subclinical extensive growth potential and significant rate of recurrences. Odontogenic keratocyst, being the third most common cystic lesion (10–20%) of the maxillofacial region, is often recognized as a sporadic lesion and is well-documented in the literature. Multiple presentation of these cysts over a lifetime is relatively uncommon and is usually seen in conjunction with nevoid basal cell carcinoma syndrome (NBCC), orofacial digital syndrome, Noonan syndrome, Ehlers–Danlos syndrome, Simpson–Golabi–Behmel syndrome, or other syndromes. The ‘two-hit’ hypothesis postulated by Knudson best explains this anomaly, wherein multiple OKCs associated with the syndromes arise as a consequence of the allelic loss in the patched (PTCH) gene, mapped to the long arm of chromosome 9q22.3-q31. A partial expression of the gene may result in multiple OKCs (5%) without any related syndromes. Though concurrent occurrence of non-syndromic multiple OKCs is a rare phenomenon, a handful of cases have been documented over the past few years. Adding to this, we report a case of multiple OKCs occurring synchronously and bilaterally in all 4 quadrants in non-syndromic, otherwise healthy persons, which could indicate a shift in trend.
机译：牙源性角化囊肿（OKC）在过去十年中被通俗地称为角化囊性牙源性肿瘤（KCOT），以其亚临床广泛的生长潜力和显着的复发率而闻名。牙源性角膜囊肿是上颌面区域中第三常见的囊性病变（10％至20％），通常被认为是散发性病变，并且在文献中有充分记载。这些囊肿在一生中多次出现是相对罕见的，通常与无基底细胞癌综合征（NBCC），口面数字综合征，Noonan综合征，Ehlers-Danlos综合征，Simpson-Golabi-Behmel综合征或其他综合征同时出现。克努森（Knudson）提出的“两次打击”假说最能解释这种异常，其中，与该综合征相关的多个OKC是由于修补的（PTCH）基因中的等位基因缺失而导致的，定位到9q22.3-q31染色体长臂。基因的部分表达可能导致多个OKC（5％），而没有任何相关的综合征。尽管并发出现多个非症状性OKC的现象很少见，但在过去几年中已记录了少数案例。除此之外，我们报告了一例非综合症患者（否则为健康人）在所有四个象限中同时发生和同时发生多个OKC的情况，这可能表明趋势发生了变化。

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