新生儿先天性表皮样囊肿和牛痘共存1例。报告一个独特的案例。

IF 1 Q3 DENTISTRY, ORAL SURGERY & MEDICINE eJournal of Oral Maxillofacial Research Pub Date : 2023-01-01 DOI:10.5037/jomr.2023.14105
Erofili Papadopoulou, Efstathios Pettas, Lampros Gkoutzanis, Konstantinos Katoumas, Maria Georgaki, Emmanouil Vardas, Evangelia Piperi, Nikolaos G Nikitakis
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摘要

背景:涉及口腔底的先天性囊性肿胀包括各种病变,如发育性囊肿(如皮样囊肿和表皮样囊肿)、毛囊、血管畸形等。然而,这些条件的共存,可能有因果关系,是罕见的。本病例报告的目的是提出一个罕见的先天性表皮样囊肿合并粘液潴留囊肿的新生儿病例。方法:2019年10月,一名6个月大的女婴被转至希腊雅典的一家口腔医学诊所,以评估她出生后儿科医生首次注意到的口腔底部肿胀。临床表现为左侧下颌下管口附近有一个淡黄色的“珍珠”结节,后向左侧口腔底弥漫性蓝色囊性肿胀过渡。临时诊断为皮样囊肿和/或瘘管,在全身麻醉下进行手术切除。结果:在组织病理学上,在前部观察到一个明确的、充满角蛋白的囊性腔,内衬有正角化的层状鳞状上皮,而在后部和附近,可以看到一个扩张的唾管,内衬有圆柱形、立方体或假层状上皮。最终诊断的表皮样囊肿密切相关的粘液潴留囊肿(ranula)的下颌骨导管呈现。结论:口腔底同时存在两种囊性病变,分别为表皮样囊肿和粘液潴留囊肿,这是一种罕见的情况,其发病机制令人费解,尤其是在新生儿中。
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Co-existence of Congenital Epidermoid Cyst and Ranula in a Newborn. Report of a Unique Case.

Background: Congenital cystic swellings involving the floor of the mouth include various lesions such as developmental cysts (e.g., dermoid and epidermoid cysts), ranulas, vascular malformations etc. However, coexistence of such conditions, possibly with a cause-and-effect- relationship, is rare. The purpose of this case report is to present a rare case of a congenital epidermoid cyst associated with a mucous retention cyst in a newborn.

Methods: A 6-month-old female infant was referred to an Oral Medicine Clinic in Athens, Greece on October 2019 for evaluation of a swelling at the floor of the mouth, first noticed by her paediatrician just after birth. Clinically, a yellowish "pearly" nodule in close association with the orifice of the left submandibular duct, posteriorly transitioning to a diffuse bluish cystic swelling of the left floor of the mouth was observed. With a provisional diagnosis of a dermoid cyst and/or ranula, a surgical excision was performed under general anaesthesia.

Results: Histopathologically, a well-defined, keratin-filled, cystic cavity lined by orthokeratinized stratified squamous epithelium was observed in the anterior aspect while posteriorly and in close proximity, a dilated salivary duct lined by cylindrical, cuboidal or pseudostratified epithelium was noted. A final diagnosis of an epidermoid cyst intimately associated with a mucus retention cyst (ranula) of the submandibular duct was rendered.

Conclusions: The coexistence of two cystic lesions in the floor of the mouth with features of epidermoid and mucous retention cyst, respectively, is rare and its pathogenesis intriguing, especially in a newborn.

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