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Peptic ulceration in a lingual sinus
Peptic Ulceration ByD.H.
in a Lingual Sinus
Parikh, S.K. Ibrahim, and R.C. Cook Liverpool, England
l Heterotopic gastric mucosa is reported in a sinus present since birth in the midline of the anterior two thirds of the
tongue. Current theories concerning the presence of heterotopic mucosa in the oral cavity are presented and discussed. Copyright
B 1991 by W.B. Saunders Company
INDEX WORDS:
Gastric mucosa, heterotopic,
tongue.
H
ETEROTOPIC gastric mucosa is commonly found in various parts of the alimentary tract, most commonly in Meckel’s diverticulum, duodenum, gallbladder, ileum, appendix,’ and rectum. Less commonly reported sites are the lungs, larynx, esophagus,’ pancreas, urinary bladder, and placenta. Gastric mucosa has also been found in the oral cavity,3 in the submandibular salivary gland, and very rarely in the anterior, lateral, and posterior parts of the tongue.4-b Histologically proven gastric mucosa was first described in 1927 by Toyama as a tongue mass.’ Since then, different types of tongue lesions containing gastric mucosa have been reported, either in a cyst’ or as a mass. We have not found any reports of a midline sinus with such heterotopia, and so present the case of a 3-year-old boy who bled from a sinus in the midline of the anterior two thirds of the tongue. The sinus contained ulcerated gastric mucosa (Fig 1).
Fig 2. Lower power micrograph of lining of the sinus showing both gastrointestinal mucosa and stratified squamous epithelium.
CASE REPORT A 3-year-old boy presented because of recurrent episodes of bleeding from a sinus in the midline of the anterior two thirds of his tongue. The sinus had been noticed by his mother soon after birth. The l-cm-deep sinus was excised. The tongue healed quickly, and he has remained well and asymptomatic since. Histologically the sinus was lined by the body type of gastric mucosa (including glands containing parietal and chief cells) mucous glands, large intestinal type crypts, and stratified squamous epithelium (Figs 2 and 3). There was an area of ulceration with granulation tissue from where we assume he had bled.
SINUS Fig 1. tongue.
A lingual sinus in the midline of the anterior two thirds of the
JournalofPediafricSurgery,
Vol26, No 1 (January), 1991: pp gg-100
From the Departments of Paediatric Surgery and HistopatholoD, Alder Hey Children’s Hospital, Liverpool, England. Address reprint requests to D.H. Parikh, MB, MS, FRCS, Depaltment of Paediatric Surgery Alder Hey Children k Hospital, Eaton Rd. Liverpool L12 2AP, England. Copyright o 1991 by W.B. Saunders Company 0022-3468/9112601-0027$03.OOlO 99
PARIKH, IBRAHIM, AND COOK
some locally abnormal inductive stimulus.” Daley et al” suggested that entrapment between the mesoderma1 masses of the developing tongue would separate sequestrated undifferentiated endodermal cells from the normal local inductive mechanism. In the reported cases these heterotopic mucosal rests show a high degree of differentiation and organization, and incomplete removal of these lesions leads to the recurrence of the symptoms. No report of heterotopic gastric mucosa in a midline lingual sinus has been traced in the literature. There are reports of midline or lateral cysts or masses in the tongue, interfering with sucking in infancy, and speech,‘z3’3 swallowing, and articulation in the older patients.5.14.15Recurrent hemorrhage into a cyst has also been described.6 An accurate preoperative diagnosis is unlikely, the lesion being removed because of its symptoms and the true nature being shown only on histology. REFERENCES
Fig 3. High-power view of Fig 2 showing body type gastric glands (arrow), intestinal crypts, and stratified squamous epithelium with a mononuclear cell infiltrate.
DISCUSSION
Different theories have been put forward to explain the presence of the gastric mucosa in the tongue. Endoderm of the gastric anlage, which lies in the neck region in the fourth and fifth week embryo, may become sequestrated in the developing tongue, trapped in the midline between the lateral lingual swellings when they fuse over the tuberculum impar. If these sequestrations were trapped below the developing tongue, the resulting lesion would lie in the floor of the mouth.3 The theory fails to explain lesions laterally in the tongue, or those that contain small intestinal and colonic mucosa.’ Primitive endoderm might escape the influence of normal local induction and then be influenced by
1. Aubrey DA: Gastric heterotopia in the vermiform appendix. Arch Surg 101:628-629,197O 2. Jabbari M, Goresky C, Lough J, et al: The inlet patch: Heterotopic gastric mucosa in the upper oesophagus. Gastroenterology 89:352-356,1985 3. Gorlin RJ, Jirasek JE: Oral cysts containing gastric or intestinal mucosa: Unusual embryonic accident or heterotopia. J Oral Surg 28:9-11, 1970 4. Willis RA: Some unusual developmental heterotopias. Br Med J 3:267-272, 1968 5. Gruskin P, Landolfe FR: Heterotopic gastric mucosa of the tongue. Arch Path01 94:184-186, 1972 6. Melato M, Ferlito A: Heterotopic gastric mucosa of the tongue and oesophagus. Otorhinolaryngol37:244-254,1975 7. Toyama MS: Occurrences of stomach mucosa at base of tongue. J Kyoto Med Univ 1:13-17,1927 8. Lister J, Zachary RB: Cystic duplications in the tongue. J Pediatr Surg 3:491-493, 1968 9. Jankus A: Heterotopic large bowel cyst in the tongue. J Path01 97:137-138, 1969 10. Picard EJ, Picard JJ, Jorissen J, et al: Heterotopic gastric mucosa in the epiglottis and rectum. Dig Dis 23:217-221, 1978 11. Daley TD, Wysocki GP, Lovas GL, et al: Heterotopic gastric cyst of the oral cavity. Head Neck Surg 7:168-171,1984 12. Duncun GW, Daniel RA Jr: Cystic tumour of the tongue. Arch Surg 44:164-169, 1942 13. Bury HPR: A cyst of the tongue containing gastric epithehum. J Path01 Bacterial 83:560-561,1962 14. Gorlin RJ, Kalnins V, Izant RJ Jr: Occurrence of heterotopic gastric mucosa in the tongue. J Pediatr 64:604-606, 1964 15. Wurster CF, Ossoff RH, Rao MS, et al: Heterotopic gastric mucosa of the tongue. Otol Head Neck Surg 93:92-95,1985
in a Lingual Sinus
Parikh, S.K. Ibrahim, and R.C. Cook Liverpool, England
l Heterotopic gastric mucosa is reported in a sinus present since birth in the midline of the anterior two thirds of the
tongue. Current theories concerning the presence of heterotopic mucosa in the oral cavity are presented and discussed. Copyright
B 1991 by W.B. Saunders Company
INDEX WORDS:
Gastric mucosa, heterotopic,
tongue.
H
ETEROTOPIC gastric mucosa is commonly found in various parts of the alimentary tract, most commonly in Meckel’s diverticulum, duodenum, gallbladder, ileum, appendix,’ and rectum. Less commonly reported sites are the lungs, larynx, esophagus,’ pancreas, urinary bladder, and placenta. Gastric mucosa has also been found in the oral cavity,3 in the submandibular salivary gland, and very rarely in the anterior, lateral, and posterior parts of the tongue.4-b Histologically proven gastric mucosa was first described in 1927 by Toyama as a tongue mass.’ Since then, different types of tongue lesions containing gastric mucosa have been reported, either in a cyst’ or as a mass. We have not found any reports of a midline sinus with such heterotopia, and so present the case of a 3-year-old boy who bled from a sinus in the midline of the anterior two thirds of the tongue. The sinus contained ulcerated gastric mucosa (Fig 1).
Fig 2. Lower power micrograph of lining of the sinus showing both gastrointestinal mucosa and stratified squamous epithelium.
CASE REPORT A 3-year-old boy presented because of recurrent episodes of bleeding from a sinus in the midline of the anterior two thirds of his tongue. The sinus had been noticed by his mother soon after birth. The l-cm-deep sinus was excised. The tongue healed quickly, and he has remained well and asymptomatic since. Histologically the sinus was lined by the body type of gastric mucosa (including glands containing parietal and chief cells) mucous glands, large intestinal type crypts, and stratified squamous epithelium (Figs 2 and 3). There was an area of ulceration with granulation tissue from where we assume he had bled.
SINUS Fig 1. tongue.
A lingual sinus in the midline of the anterior two thirds of the
JournalofPediafricSurgery,
Vol26, No 1 (January), 1991: pp gg-100
From the Departments of Paediatric Surgery and HistopatholoD, Alder Hey Children’s Hospital, Liverpool, England. Address reprint requests to D.H. Parikh, MB, MS, FRCS, Depaltment of Paediatric Surgery Alder Hey Children k Hospital, Eaton Rd. Liverpool L12 2AP, England. Copyright o 1991 by W.B. Saunders Company 0022-3468/9112601-0027$03.OOlO 99
PARIKH, IBRAHIM, AND COOK
some locally abnormal inductive stimulus.” Daley et al” suggested that entrapment between the mesoderma1 masses of the developing tongue would separate sequestrated undifferentiated endodermal cells from the normal local inductive mechanism. In the reported cases these heterotopic mucosal rests show a high degree of differentiation and organization, and incomplete removal of these lesions leads to the recurrence of the symptoms. No report of heterotopic gastric mucosa in a midline lingual sinus has been traced in the literature. There are reports of midline or lateral cysts or masses in the tongue, interfering with sucking in infancy, and speech,‘z3’3 swallowing, and articulation in the older patients.5.14.15Recurrent hemorrhage into a cyst has also been described.6 An accurate preoperative diagnosis is unlikely, the lesion being removed because of its symptoms and the true nature being shown only on histology. REFERENCES
Fig 3. High-power view of Fig 2 showing body type gastric glands (arrow), intestinal crypts, and stratified squamous epithelium with a mononuclear cell infiltrate.
DISCUSSION
Different theories have been put forward to explain the presence of the gastric mucosa in the tongue. Endoderm of the gastric anlage, which lies in the neck region in the fourth and fifth week embryo, may become sequestrated in the developing tongue, trapped in the midline between the lateral lingual swellings when they fuse over the tuberculum impar. If these sequestrations were trapped below the developing tongue, the resulting lesion would lie in the floor of the mouth.3 The theory fails to explain lesions laterally in the tongue, or those that contain small intestinal and colonic mucosa.’ Primitive endoderm might escape the influence of normal local induction and then be influenced by
1. Aubrey DA: Gastric heterotopia in the vermiform appendix. Arch Surg 101:628-629,197O 2. Jabbari M, Goresky C, Lough J, et al: The inlet patch: Heterotopic gastric mucosa in the upper oesophagus. Gastroenterology 89:352-356,1985 3. Gorlin RJ, Jirasek JE: Oral cysts containing gastric or intestinal mucosa: Unusual embryonic accident or heterotopia. J Oral Surg 28:9-11, 1970 4. Willis RA: Some unusual developmental heterotopias. Br Med J 3:267-272, 1968 5. Gruskin P, Landolfe FR: Heterotopic gastric mucosa of the tongue. Arch Path01 94:184-186, 1972 6. Melato M, Ferlito A: Heterotopic gastric mucosa of the tongue and oesophagus. Otorhinolaryngol37:244-254,1975 7. Toyama MS: Occurrences of stomach mucosa at base of tongue. J Kyoto Med Univ 1:13-17,1927 8. Lister J, Zachary RB: Cystic duplications in the tongue. J Pediatr Surg 3:491-493, 1968 9. Jankus A: Heterotopic large bowel cyst in the tongue. J Path01 97:137-138, 1969 10. Picard EJ, Picard JJ, Jorissen J, et al: Heterotopic gastric mucosa in the epiglottis and rectum. Dig Dis 23:217-221, 1978 11. Daley TD, Wysocki GP, Lovas GL, et al: Heterotopic gastric cyst of the oral cavity. Head Neck Surg 7:168-171,1984 12. Duncun GW, Daniel RA Jr: Cystic tumour of the tongue. Arch Surg 44:164-169, 1942 13. Bury HPR: A cyst of the tongue containing gastric epithehum. J Path01 Bacterial 83:560-561,1962 14. Gorlin RJ, Kalnins V, Izant RJ Jr: Occurrence of heterotopic gastric mucosa in the tongue. J Pediatr 64:604-606, 1964 15. Wurster CF, Ossoff RH, Rao MS, et al: Heterotopic gastric mucosa of the tongue. Otol Head Neck Surg 93:92-95,1985