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Congenital heterotopic gastrointestinal cyst of the oral cavity: report of rare case
ARTICLE IN PRESS
YBJOM-4755; No. of Pages 2
Available online at www.sciencedirect.com
British Journal of Oral and Maxillofacial Surgery xxx (2015) xxx–xxx
Letter to the Editor Congenital heterotopic gastrointestinal cyst of the oral cavity: report of rare case Sir, Gastrointestinal heterotopic cyst is a rare benign lesion diagnosed mainly in young boys, but it can also occur in adults.1 It is possible that ectopic islands of endodermal cells infiltrate the primitive stomodeum during the fourth week of embryonic development, and cause multidirectional differentiation.2 Heterotopic gastric mucosa can appear almost anywhere in the gastrointestinal tract, but mostly in the mediastinum, retroperitoneum, and oral cavity, where it can be found on the tongue, floor of the mouth, and lips.3 On histological examination, the cyst shows gastric or intestinal mucosa covered by columnar ciliated epithelium, with content that is solid or cystic. Differential diagnosis consists of dermoid cysts, thyroglossal duct cysts, vascular malformations, lipomas, and salivary gland diseases.4 Resection is the preferred treatment, as relapse is uncommon. A 3-month-old boy was seen at the Servicio Autónomo Hospital Universitario de Maracaibo, Venezuela, with a congenital lump underneath his tongue, which caused dysphagia and labial incompetence. The lesion was normochromic, soft, and moved on palpation. Computed tomography (CT) of the head and neck showed a fluid-filled nodule under the ventral surface of the tongue, with no prolapse through the mylohyoid muscle, or danger to the airway. Despite thyroid function tests being within normal limits, thyroid ultrasound was done to rule out glandular involvement of the nodule. The patient was operated on under general anaesthesia. We aspirated the intraluminal cyst completely, then instilled intralesional alginate, and did a blunt excision of the lesion (Fig. 1). We achieved haemostasis with polyglactin 910 sutures (Vicryl 3-0) and sent the specimen for histological examination. Microscopic examination showed that the lesion was lined with hyperplasic squamous stratified keratinised epithelium, and cube-shaped and ciliated columnar pseudostratified epithelium with caliciform cells. In deeper layers we found intestinal colon-like glandular tissue within fibrous connective stroma, which are associated with moderate mononuclear inflammatory infiltrate, and blood vessels of variable calibre (Fig. 2).
Fig. 1. Excision of the lesion.
Fig. 2. Photomicrograph of the sample showing hyperplasic squamous stratified keratinised epithelium, cube-shaped and ciliated columnar, pseudostratified epithelium, and caliciform cells (haematoxylin and eosin, original magnification x10).
http://dx.doi.org/10.1016/j.bjoms.2016.01.014 0266-4356/© 2016 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
Please cite this article in press as: Solano N, et al. Congenital heterotopic gastrointestinal cyst of the oral cavity: report of rare case. Br J Oral Maxillofac Surg (2015), http://dx.doi.org/10.1016/j.bjoms.2016.01.014
ARTICLE IN PRESS
YBJOM-4755; No. of Pages 2
2
Letter to the Editor / British Journal of Oral and Maxillofacial Surgery xxx (2015) xxx–xxx
Magnetic resonance imaging (MRI), CT, or ultrasonography are usually recommended5 so we used the latter two to assess the location of the lesion, its relation to structures nearby, and its approximate size, and consistency. We then considered that it might be a thyroglossal duct cyst, because of its midline position and relation with the tongue, but rejected this after ultrasound examination of the thyroid. Differential diagnosis included tumours of the salivary gland, vascular, or adipose tissue. At last, histological examination confirmed the diagnosis of gastrointestinal heterotopic cyst. We recommend excision using the alginate instillation technique, which simplifies both recognition of the cystic capsule from the surrounding tissues and blunt divulsion, and reduces the risk of leaving remnants that could cause further relapse. CT and ultrasonography are useful imaging tools before biopsy examination. Finally, we recommend long-term follow up of these lesions to prevent relapse during adulthood.
Conflict of interest We have no conflicts of interest.
Ethics statement/confirmation of patient’s permission This research complies with the World Medical Association Declaration of Helsinki on medical protocols and ethics. As the images of the patient were essential to this paper, the patient’s mother gave written consent.
Acknowledgements
References 1. Ohbayashi Y, Miyake M, Nagahata S. Gastrointestinal cyst of the tongue: a possible duplication cyst of foregut origin. J Oral Maxillofac Surg 1997;55:626–30. 2. Gorlin RJ, Jirasek JE. Oral cyst containing gastric or intestinal mucosa. An unusual embryological accident or heterotopia. Arch Otolaryngol 1970;91:594–7. 3. Popkharitov AI, Guluvoba MV, Dandov AD, et al. Heterotopic gastrointestinal cyst mimicking chronic cholecystitis: a case report. J Med Case Rep 2008;2:173. 4. Drennen KC, Myers EN. Heterotopic gastrointestinal mucosa of the oral cavity. Otolaryngol Head Neck Surg 1998;118:99–101. 5. Wetmore RF, Barlett SP, Papsin B, et al. Heterotopic gastric mucosa of the oral cavity: a rare entity. Int J Pediatr Otorhinolaryngol 2002;66: 139–42.
Nicolás Solano a Andrés Gómez-Delgado b,∗ Luis Sarmiento c Ligia Pérez d a Oral and Maxillofacial Department; Plastic, Reconstructive and Maxillofacial Surgery Department; Oncology Department, Hospital Universitario de Maracaibo, Venezuela b Oral and Maxillofacial Surgery Residency Program, Universidad El Bosque, Bogotá, Colombia c Oral Surgery Program, Dental School, Universidad del Zulia, Maracaibo, Venezuela d Dental School, Universidad del Zulia, Maracaibo, Venezuela ∗ Corresponding author at: Universidad El Bosque, Departmento de Cirugía Oral y Maxilofacial, Carrera 7 B Bis No. 132-11. Código Postal 110121. Bogotá, Colombia. Tel.: +57 6489000. E-mail address: [email protected] (A. Gómez-Delgado)
To the Paediatric Surgeon Leyla Fereira, and the Oral Surgery Residents Juan Pablo García, María José Perozo, Edgar Graterol and Mariana Meza.
Please cite this article in press as: Solano N, et al. Congenital heterotopic gastrointestinal cyst of the oral cavity: report of rare case. Br J Oral Maxillofac Surg (2015), http://dx.doi.org/10.1016/j.bjoms.2016.01.014
YBJOM-4755; No. of Pages 2
Available online at www.sciencedirect.com
British Journal of Oral and Maxillofacial Surgery xxx (2015) xxx–xxx
Letter to the Editor Congenital heterotopic gastrointestinal cyst of the oral cavity: report of rare case Sir, Gastrointestinal heterotopic cyst is a rare benign lesion diagnosed mainly in young boys, but it can also occur in adults.1 It is possible that ectopic islands of endodermal cells infiltrate the primitive stomodeum during the fourth week of embryonic development, and cause multidirectional differentiation.2 Heterotopic gastric mucosa can appear almost anywhere in the gastrointestinal tract, but mostly in the mediastinum, retroperitoneum, and oral cavity, where it can be found on the tongue, floor of the mouth, and lips.3 On histological examination, the cyst shows gastric or intestinal mucosa covered by columnar ciliated epithelium, with content that is solid or cystic. Differential diagnosis consists of dermoid cysts, thyroglossal duct cysts, vascular malformations, lipomas, and salivary gland diseases.4 Resection is the preferred treatment, as relapse is uncommon. A 3-month-old boy was seen at the Servicio Autónomo Hospital Universitario de Maracaibo, Venezuela, with a congenital lump underneath his tongue, which caused dysphagia and labial incompetence. The lesion was normochromic, soft, and moved on palpation. Computed tomography (CT) of the head and neck showed a fluid-filled nodule under the ventral surface of the tongue, with no prolapse through the mylohyoid muscle, or danger to the airway. Despite thyroid function tests being within normal limits, thyroid ultrasound was done to rule out glandular involvement of the nodule. The patient was operated on under general anaesthesia. We aspirated the intraluminal cyst completely, then instilled intralesional alginate, and did a blunt excision of the lesion (Fig. 1). We achieved haemostasis with polyglactin 910 sutures (Vicryl 3-0) and sent the specimen for histological examination. Microscopic examination showed that the lesion was lined with hyperplasic squamous stratified keratinised epithelium, and cube-shaped and ciliated columnar pseudostratified epithelium with caliciform cells. In deeper layers we found intestinal colon-like glandular tissue within fibrous connective stroma, which are associated with moderate mononuclear inflammatory infiltrate, and blood vessels of variable calibre (Fig. 2).
Fig. 1. Excision of the lesion.
Fig. 2. Photomicrograph of the sample showing hyperplasic squamous stratified keratinised epithelium, cube-shaped and ciliated columnar, pseudostratified epithelium, and caliciform cells (haematoxylin and eosin, original magnification x10).
http://dx.doi.org/10.1016/j.bjoms.2016.01.014 0266-4356/© 2016 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
Please cite this article in press as: Solano N, et al. Congenital heterotopic gastrointestinal cyst of the oral cavity: report of rare case. Br J Oral Maxillofac Surg (2015), http://dx.doi.org/10.1016/j.bjoms.2016.01.014
ARTICLE IN PRESS
YBJOM-4755; No. of Pages 2
2
Letter to the Editor / British Journal of Oral and Maxillofacial Surgery xxx (2015) xxx–xxx
Magnetic resonance imaging (MRI), CT, or ultrasonography are usually recommended5 so we used the latter two to assess the location of the lesion, its relation to structures nearby, and its approximate size, and consistency. We then considered that it might be a thyroglossal duct cyst, because of its midline position and relation with the tongue, but rejected this after ultrasound examination of the thyroid. Differential diagnosis included tumours of the salivary gland, vascular, or adipose tissue. At last, histological examination confirmed the diagnosis of gastrointestinal heterotopic cyst. We recommend excision using the alginate instillation technique, which simplifies both recognition of the cystic capsule from the surrounding tissues and blunt divulsion, and reduces the risk of leaving remnants that could cause further relapse. CT and ultrasonography are useful imaging tools before biopsy examination. Finally, we recommend long-term follow up of these lesions to prevent relapse during adulthood.
Conflict of interest We have no conflicts of interest.
Ethics statement/confirmation of patient’s permission This research complies with the World Medical Association Declaration of Helsinki on medical protocols and ethics. As the images of the patient were essential to this paper, the patient’s mother gave written consent.
Acknowledgements
References 1. Ohbayashi Y, Miyake M, Nagahata S. Gastrointestinal cyst of the tongue: a possible duplication cyst of foregut origin. J Oral Maxillofac Surg 1997;55:626–30. 2. Gorlin RJ, Jirasek JE. Oral cyst containing gastric or intestinal mucosa. An unusual embryological accident or heterotopia. Arch Otolaryngol 1970;91:594–7. 3. Popkharitov AI, Guluvoba MV, Dandov AD, et al. Heterotopic gastrointestinal cyst mimicking chronic cholecystitis: a case report. J Med Case Rep 2008;2:173. 4. Drennen KC, Myers EN. Heterotopic gastrointestinal mucosa of the oral cavity. Otolaryngol Head Neck Surg 1998;118:99–101. 5. Wetmore RF, Barlett SP, Papsin B, et al. Heterotopic gastric mucosa of the oral cavity: a rare entity. Int J Pediatr Otorhinolaryngol 2002;66: 139–42.
Nicolás Solano a Andrés Gómez-Delgado b,∗ Luis Sarmiento c Ligia Pérez d a Oral and Maxillofacial Department; Plastic, Reconstructive and Maxillofacial Surgery Department; Oncology Department, Hospital Universitario de Maracaibo, Venezuela b Oral and Maxillofacial Surgery Residency Program, Universidad El Bosque, Bogotá, Colombia c Oral Surgery Program, Dental School, Universidad del Zulia, Maracaibo, Venezuela d Dental School, Universidad del Zulia, Maracaibo, Venezuela ∗ Corresponding author at: Universidad El Bosque, Departmento de Cirugía Oral y Maxilofacial, Carrera 7 B Bis No. 132-11. Código Postal 110121. Bogotá, Colombia. Tel.: +57 6489000. E-mail address: [email protected] (A. Gómez-Delgado)
To the Paediatric Surgeon Leyla Fereira, and the Oral Surgery Residents Juan Pablo García, María José Perozo, Edgar Graterol and Mariana Meza.
Please cite this article in press as: Solano N, et al. Congenital heterotopic gastrointestinal cyst of the oral cavity: report of rare case. Br J Oral Maxillofac Surg (2015), http://dx.doi.org/10.1016/j.bjoms.2016.01.014