Congenital heterotopic gastrointestinal cyst of the oral cavity in a neonate: Case report and review of literature

Department of Otolaryngology, Texas Children's Hospital, Baylor College of Medicine, St., Houston 77030-2399, USA.
International Journal of Pediatric Otorhinolaryngology (Impact Factor: 1.19). 07/1996; 36(1):69-77. DOI: 10.1016/0165-5876(96)01331-6
Source: PubMed


Heterotopic gastrointestinal cysts of the oral cavity are rare benign lesions which may mimic both benign and malignant neoplasms. These cysts are usually discovered during infancy, but may not appear until well into adulthood. This lesion involves the tongue and floor of the mouth in 97% of cases and has a male predilection. The cyst may range in size from less than 1 cm to 9 cm, with most lesions being 1-3 cm in size. About 30% of affected individuals have symptoms related to difficulties with feeding, swallowing and respiration. The epithelial lining is quite variable, but all cysts have an enteric lining. The histogenesis is related to entrapment of undifferentiated, noncommitted endoderm within the oral cavity during the 3rd-4th week of fetal life. The purpose of this paper is to report a congenital heterotopic gastrointestinal cyst of the oral cavity presenting in a neonate. The clinical and histopathologic features of this cyst are reviewed, as well as the histogenesis of this lesion.

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    • "Review of reports of lingual cysts involving both gastric and respiratory epithelia suggests a male predilection (Table 1) which has also been observed to varying degrees in reviews of oral cysts in general [2] [4] [30]. Most reported cysts present during the first year of life and localize to the anterior tongue and/or the floor of the mouth. "
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    ABSTRACT: Foregut cysts are uncommon, mucosa-lined congenital lesions that may occur anywhere along the gastrointestinal or respiratory tract and typically present within the first year of life. Although infrequent, these cysts may generate feeding or respiratory difficulties depending on the size and location of the lesion. Foregut cysts of the oral cavity are rarely seen and of those cases localized to the tongue are even more uncommon. We describe a 4-month-old girl with a foregut cyst involving the floor of mouth and anterior tongue. Subsequent histologic analysis demonstrated a cyst lined with both gastric and respiratory epithelia. This case represents an extremely rare finding of both gastric and respiratory epithelia lined within a single cystic structure in the tongue. Although a very rare finding, a foregut cyst should be on the differential diagnosis of any lesion involving the floor of mouth or tongue in an infant or child.
    Case Reports in Medicine 08/2015; 2015(10):278376. DOI:10.1155/2015/278376
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    • "Several theories have been proposed, but they cannot explain all types of heterotopic tissues.7 According to the most recent theory, the cysts arise from islands of endoderm that originate from the lining of the primitive stomodeum and become entrapped at the embryonic age of 4-5 weeks when the entire gastrointestinal endoderm remains undifferentiated.12 Another theory is that the cysts develop from salivary retention cysts, where the salivary gland tissue is dedifferentiated and subsequently differentiated into gastrointestinal tissue.13 "
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    ABSTRACT: Heterotopic gastrointestinal cysts are rarely found in the oral cavity. Most of these cysts are lined with gastric mucosa and involve the tongue. There have been no reported heterotopic intestinal cysts of the submandibular gland that are completely lined with colonic mucosa. An 8-year-old girl presented with an enlarging swelling in the left submandibular area, and a 4-cm unilocular cyst was fully excised. The cyst was completely lined with colonic mucosa that was surrounded by smooth muscle layer, and the lining cells were positive for CDX-2, an intestinal marker, indicating a high degree of differentiation. The pathogenesis remains unclear, but it may be related to the misplacement of embryonic rests within the oral cavity during early fetal development. Although heterotopic intestinal cysts rarely occur in the submandibular gland, they should be considered in the differential diagnosis of facial swellings in the pediatric population.
    The Korean Journal of Pathology 06/2013; 47(3):279-83. DOI:10.4132/KoreanJPathol.2013.47.3.279 · 0.17 Impact Factor
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    • "Under the appropriate influences these pleuripotent cells can then differentiate into both gastrointestinal and respiratory structures in their new location. Other putative explanations have been described elsewhere [1] [2]. In the majority of reported cases the patients have been under the age of 2, supporting the hypothesis that the cysts are congenital. "
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    ABSTRACT: The presence of normal alimentary tissue in an abnormal location is referred to as a heterotopic gastrointestinal cyst or duplication cyst. These are thought to be congenital and rarely occur in the neck. More often found in the oral cavity, they usually present as an asymptomatic swelling and are treated with surgical excision. We describe the rare presentation and diagnosis of an 8-month-old girl with a congenital heterotopic gastrointestinal cyst in the neck.
    International Journal of Pediatric Otorhinolaryngology Extra 09/2007; 2(3):154-157. DOI:10.1016/j.pedex.2007.04.005
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