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CASE REPORTS
Duplication Cyst of Tongue
IV Meisheri, Mayur Maheshwari, Jayul Kamdar
Duplication cyst of tongue is a very rare surgical entity. Only a few cases have been reported in literature. Duplication have reported from tongue to anus, small intestine being the most common site for duplication. Tongue duplication has been associated with heterotropic mucosa within it. Tongue duplication cysts present a curious entity as far as diagnosis is concerned. Duplication cysts of tongue have the potential for air - way obstruction and may cause respiratory distress. Complete excision is the treatment of choice.
INTRODUCTION
Enteric duplications have been described in whole of the Gastrointestinal (GI) tract.1 Mostly they are reported in small intestine. Oral duplications are one of the rare GI duplications. Various reports show tongue duplications in relation to some portion of tongue.2.3 A variety of heterotropic mucosa have been reported in association with tongue duplication including gastric, colonic and respiratory mucosa.2.4.5 We had a baby who presented with duplication cyst of tongue in relation to hard palate and was treated with complete excision of duplication, which is the treatment of choice.
CASE REPROT
A neonate presented on day one of his life with a mass arising from hard palate measuring approx. 3 x 2 x 1 cms. It was firm in consistency with smooth surface. There was no associated anomaly. Baby had normal respiration. Clinically as diagnosis was doubtful, a CT scan was done. On CT scan its origin from hard palate was confirmed and no communication anywhere. Baby was unable to suck and hence was kept on Ryle’s tube feeding. Baby was taken up for surgery on an elective basis. Complete excision of mass was done with primary closure of mucosal defect. Postoperative period was uneventful. Histopathology report showed it as duplication cyst of tongue with presence of circumvalate and folate papillae and smooth muscle. There was no heterotropic mucosa in it. Baby is fine at one-year follow up.
DISCUSSION
Alimentary tract duplications have been reported from tongue to anus, small intestine is the most common site and that too in ileum.6.7 Synchronous duplications are seen in upto 15% of cases. Thoracic and thoraco-abdominal duplications may be associated with vertebral anomalies such as bifid or fused spines, hemivertebra, and myelomeningocele in which case term neurenteric cyst is more appropriate. Tubular hindgut duplications may be associated with genitourinary and genital defects.8 Some duplications are silent while others are symptomatic. Neoplastic changes have been reported in some duplication.9
Oral cavity is a very rare site for duplication. Oral cysts containing heterotropic mucosa has been referred as duplication cyst, Choristomatic cyst or heterotropic cyst. Duplication cyst is characterized by a coat of muscle, attachment to some part of GI tract, mucosal lining similar to some part of GI tract.10 On the contrary, choristomatic cyst or heterotropic cyst need not have a muscle coat.11
There are many theories regarding the pathogenesis of lingual duplications. One theory postulates that they arise from lining of primitive stomodium which become entrapped in the mouth during 4th or 5th week of embryonic development, a time at which entire GI endoderm is undifferentiated. This endoderm is sheltered from normal embryonic induction and may differentiate in a variety of mucosa.12 Various theories regarding pathogenesis of enteric duplications such as development from epithelial inclusions trapped during fusion of primordial tissues, persistence of epithelial buds within the wall of bowel, from nests of trapped endothelial cells or from incomplete coalescence of lacunae that form between epithelial cells of solid core of developing gut does not explain fully lingual cysts as the tongue does not develop in the same way as hollow viscera.5
Tongue duplications have been reported to be having gastric, colonic and respiratory mucosa. Awonters, Reychler reported 2 cases with heterotropic mucosa.4 Mirchandani et al reported a congenital oral duplication cyst on ventral surface of tongue associated with both GI and respiratory epithelium.2 Colonic mucosa also has been reported by Lipsett et al.5
Oral duplication cysts may occur at various sites such as ventral aspect of tongue, dorsum of tongue, anterior two-third of tongue. Willner et al reported an enteric duplication in anterior two-third of tongue.3 Chen et al reported two enteric sublingual duplications.13 Duplication cyst of tongue has to be differentiated from dermoid cyst, haemangioma,lingual thyroid remnant, ranula and cystic hygroma.
Oral duplication cysts carry a potential for air-way obstruction and difficult intubation. Cyst can be aspirated to provide access for intubation, otherwise tracheostomy is required. Although Laser ablation has been described, complete excision remains the treatment of choice.13
Our patient presented with a mass of solid nature attached to hard palate, showed no heterotropic mucosa, was not associated with any other anomaly. Except for inability to suck, baby was asymptomatic. Baby was kept on Ryle’s tube feeding initially and later complete excision of duplication was done. Baby is asymptomatic on follow-up at one year.
REFERENCES
1. Gross RE. The surgery of infancy and childhood, WB Saunders, Philadelphia. 1953; 221-25.
2. Mirchandani R, Sciubba J, Gloster ES. Congenital oral cyst with heterotopic gastrointestinal and respiratory mucosa. Arch Pathol Lab Med 1989; 113 : 1301-2.
3. Willner A, Feghali J, Bassila M. An enteric duplication cyst occurring in anterior two-thirds of the tongue. Int J Pediatr - Otorhinolaryngol 1991; 21: 169-177.
4. Awonters P, Reychler H. Enteric duplication in the oral cavity. Int J Oral Maxilofac - Surg 1991; 20 : 12-14.
5. Lipsset J, Sparnon AL, Byard RW. Embryogenesis of enterocystomas - enteric duplication cysts of tongue. Oral - Surg - Oral - Med - Oral - Pathol 1993; 75 : 623-30.
6. Iyer CP, Mahour GH. Duplication of alimentary tract in infancy and children. J Pediatr Surg 1995; 30 : 1267-70.
7. Bond SJ, Graff DB. Gastrointestinal duplication, in O Neill LA, Rowe MI, et al (Eds) Mosby yearbook, 1998; 1257-67.
8. Ravitch MM. Hindgut duplication - Doubling of colon and genital urinary tracts. Ann Surg 1953; 137 : 588-601.
9. Orr MM, Edwards AJ. Neoplastic changes in duplications of alimentary tract. BJ Surg 1971; 62 : 269-74.
10. Ladd WE, Gross RE. Surgical treatment of duplications of alimentary tract : Enterogenous cyst, enteric cyst or ileum duplex. Surg Gynecol Obst 1940; 70 : 295-307.
11. Eppley BL, Bell MJ, Sclaroff A. Simultaneous occurrence of dermoid and heterotopic intestinal cysts in the floor of mouth of a new - born. J Oral Maxillofac Surg 1985; 43 : 880-81.
12. Daley TD, Wysocki GP, Lovas GL. Heterotropic gastric cyst of the oral cavity: Head and Neck Surgery 1984; 7 : 168-71.
13. Chen MK, Gross E, Lobe TE. Perinatal management of enteric duplication cysts of tongue. Am J Perinat 1997; 14 : 161-63.
HIGH-NORMAL BLOOD PRESURE - MORE “HIGH” THAN “NORMAL”
On the basis of evidence from multiple clinical trials, it is recommended that antihypertensive therapy be instituted for patients with confirmed hypertension - that is when the systolic blood pressure exceeds 140 mm Hg or the diastolic blood pressure exceeds 90 mm Hg. In fact, it is recognized that cardiovascular risk increases linearly at blood-pressure levels lower than those that usually trigger the use of antihypertensive therapy - specifically at a systolic pressure of 130 to 135 mm Hg and a diastolic pressure of 80 to 85 mm Hg.
The authors found that the participants with high-normal blood pressure (systolic pressure of 130 to 139 mm Hg., diastolic pressure of 85 to 89 mm Hg., or both) had higher rates of cardiovascular events than those with optimal blood pressure (defined as systolic pressure of less than 120 mm Hg and diastolic pressure of less than 80 mm Hg). These findings lend further credence to the theory that high-normal blood pressure must be categorized differently from normal or optimal blood pressure.
N Engl J Med, November, 2001; 345 : 1337.
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