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High esophageal stricture: A complication of “inlet patch” mucosa
GASTROENTEROLOGY
1988;94:521-4
High Esophageal Stricture: A Complication of “Inlet Patch” Mucosa CHARLES PETER
STEADMAN,
PAUL
KERLIN,
TEAGUE,
and
STEPHENSON
Departments of Gastroenterology Brisbane, Queensland, Australia
and Anatomical
High esophageal stricture associated with a circumferential “inlet patch” of heterotopic gastric mucosa is reported in 2 patients. Biopsy specimens taken from the strictures demonstrated inflamed or ulcerated gastric fundal-type mucosa in both cases. Dilatation of the strictures followed by treatment with histamine Hz-antagonists produced almost complete resolution of dysphagia in both patients.
eterotopic gastric-type columnar epithelium may occur throughout the gastrointestinal tract and in (extraintestinal cysts (11. Gastric-type mucosa in the esophagus may be in continuity with the gastric epithelium [Barrett’s esophagus (a,~)] or be separated from gastric columnar epithelium by normal squamous esophageal mucosa (4). The former pathology is acquired in the setting of chronic gastroesophageal reflux (3), but discrete heterotopic gastric-type mucosa in the esophagus probably represents a congenital abnormality of foregut development (4,s). Either situation can be associated with ulceration in the areas of columnar epithelium (6-8). This report documents 2 patients who experienced high dysphagia and were found to have high esophageal fibrotic (peptic) strictures in association with isolated heterotopic gastric mucosa in the upper esophagus (“inlet patch”).
H
Case
CLINTON
Reports
Case 2 N. H., a 6%yr-old man, complained of intermittent difficulty in swallowing since 1950. A pharyngeal stricture had been diagnosed in 1977 at another institution and treated by dilatation. The cause of the stricture had not been determined. At presentation in August 1985, he complained of progressive dysphagia for solids at the level of the sternal notch. He had mild aching pain radiating to the anterior triangle of the left side of his neck. No weight loss or constitutional symptoms had occurred. Treatment
Pathology, Princess Alexandra
Hospital,
for hypertension with metoprolol (100 mg each morning) had been started before investigation. Barium swallow examination demonstrated a stricture impressing the esophagus (Figure 1) from the left side at the level of the sternal notch. There was mild ballooning of the esophagus proximal to the stricture, but no delay in flow of contrast was seen. A small sliding hiatus hernia was present but only a small amount of reflux could be demonstrated. In August 1985, rigid esophagoscopy demonstrated a stricture that appeared benign at 20 cm from the lips. Dilatation of the stricture was performed. The biopsy specimens taken from the stricture demonstrated columnar epithelium. In view of mildly symptomatic regurgitation and the unusual finding of columnar epithehum in the upper esophagus, the patient underwent further assessment with a flexible fiberoptic esopbagogastroscope. This instrument was able to traverse the stricture. At the level of the stricture (21 cm from the incisors] there was a circumferential red patch of muLosa extending -I cm distally. Beyond the “red” mucosa, normal pearly white esophageal mucosa was observed to 40 cm. Mild linear antral gastritis was seen. The duodenum was normal. No other similar red patches were seen in the esophagus or upper gastrointestinal tract. Biopsy specimens of the inlet patch demonstrated irregular groups of glands in the lamina propria that contained both parietal and chief cells, as’ well as “halo” endocrine cells (Figure 2). Gastric-type mucus cells were present but intestinal metaplasia was not demonstrated. The lamina propria showed a mild chronic inflammatory cell infiltrate. Specimens taken from below the lesion demonstrated normal squamous mucosa. Cimetidine (400 mg twice daily) was instituted with a marked reduction in dysphagia and almost complete loss of left neck pain. At follow-up assessment in May 1986 minimal difficulty in swallowing had occurred during 7 mo of maintenance cimetidine (400 mg daily). At repeat endoscopic examination the circumferential red patch of columnar epithelium at 21 cm was unchanged but no 1986, after ulceration or stricture was seen. In November
0 1988 by the American
Gastroenterological 0016.5085/861$3.50
Association
522
STEADMAN ET AL.
(~ASTKOENTEKOLOGY Vol.
~4. No. 2
biopsy specimen demonstrated a severe acute on chronic gastritis with mild inflammatory dysplasia. Heterotopic gastric mucosa of the upper esophagus, complicated by ulceration and stricture formation, was diagnosed. Ranitidine (150 mg twice daily] was administered with loss of all symptoms. A follow-up endoscopy 2 mo after presentation [June 1986) demonstrated a circumferential patch of red mucosa in the upper esophagus from 18 to 21 cm, with no evidence of active inflammation. The patient
‘igure 1. Radiograph of the uppe ‘r esophagus in patient 1 (antero. posterior view on left. Iateral view on right). There is a stricture at the level of I:he sternal notch with dilatation above.
complaints of mild dysphagia for viscous substances, endoscopy revealed a minor stricture recurrence. Cimetidine (400 mg at night) is to be continued indefinitely. Case
2
A. S., a 42-yr-old seaman, complained of difficulty in swallowing solid food for 5 yr. The obstruction was localized to the suprasternal area. No odynophagia was present and he denied symptoms of heartburn. Intermittent regurgitation of food had occurred but both weight and appetite were stable. Examination demonstrated no evidence of a cervical mass lesion or anemia. A barium swallow examination demonstrated stenosis of the esophagus at the level of the second thoracic vertebra with dilatation of the esophagus above the stenosis. Upper gastrointestinal endoscopy confirmed an erythematous and ulcerated stricture at 20 cm, obscuring the mucosa at that level. Normal esophageal mucosa was seen beyond the stricture. Mild gastritis was present. Dilatation of the stricture to 51F gauge was performed. Biopsy specimens taken from the stricture demonstrated the presence of gastric fundal-type mucosa with severe inflammation and confirmed the presence of ulceration (Figure 3). There was no evidence of malignancy. Specimens taken from the esophagus below the stricture showed normal squamous esophageal mucosa. The gastric
Figure 2. Photomicrographs of biopsy specimens patch in case 1. A. Typical tall columnar
of the inlet
gastric-type mucus cells form the surface. Inflammatory Lells, mostly lymphocytes, are prominent focally in the lamina propria. The glands contain numerous “halo” t?Ildocrine cells (arrows). H&E, x140. B. Several adjacent glands are shown. Both parietal cells ((IrroM’s) and chief cells (arrowheuds) are present. H&E, X560.
E‘ebruary 1988
Figure
‘INLET PATCH”
3. Photomicrograph of a portion of one of the esophageal biopsy fragments obtained from case 2. It consists predominantly of inflamed granulation tissue and amorphous exudate, confirming the presence of ulceration. H&E, x175.
has remained
asymptomatic
on
ranitidine
(150
mg
at
night].
Discussion Heterotopic gastric mucosa in the cervical esophagus is an unusual cause of high dysphagia. Reports of inlet patch-related dysphagia have noted both localized (9) and circumferential (10)patches of gastric mucosa, but the mechanism of dysphagia has not been clarified. Proposals have included web formation at the proximal squamocolumnar junction of the patch (10) and increased cricopharyngeal muscular tone secondary to acid production by the gastric-type mucosa (I I). Increased upper esophageal sphincter tone in response to luminal acid has been demonstrated in controlled experiments (12). Symptom production may relate to the area and distribution of columnar epithelium and to the acidproducing capability. In symptomatic cases, the histology of the heterotopic mucosa has been largely gastric body or fundic type (10,11,13,14). Acid production has been demonstrated by Congo red staining (11)and pH electrode measurement of esophageal a.cidity (13). In the latter study larger areas of heterotopic gastric mucosa produced the largest falls in local pH after pentagastrin stimulation. The endoscopk and histologic appearances indicate a peptic etiology for stricture formation in the cases reported here. The response to drug therapy also supports this hypothesis. Furthermore, the circumferential distri-
ESOPHAGEAL
STRICTIJRE
523
bution of heterotopic gastric mucosa observed may have caused a greater reduction in local pH, with a consequent increase in peptic injury. In contrast to the symptomatic cases, most people who have the inlet patch appear to be asymptomatic. An autopsy study of 1000 children (5)found 7.8% with heterotopic esophageal gastric mucosa, of which 51% was in the upper one-third. Gastric mucosa without parietal cells occurred more frequently than that with parietal cells, but no glands of the latter type were found in the lower one-third of the esophagus. A more recent endoscopic study of 420 sequential endoscopies found an inlet patch prevalence of 3.8% (13),but most were
References 1. Wolff M. Heterotopic gastric report of three new cases with
epithelium in the rectum: a a review of 87 cases of gastric
524
2.
3. 4. 5. 6. 7.
8.
9.
10.
STEADMAN
ET AL
GASTKOEN?‘E:KOI,O(;Y
heterotopia in the alimentary canal. Am J Clin Path01 1971;55:604-16. Barrett NR. Chronic peptic ulcer of the esophagus and esophagitis. Br J Surg 1950;38:175-82. Allison PR, Johnstone AS. The esophagus lined with gastric mucous membrane. Thorax 1953;8:87-101. Johns BAE. Developmental changes in the esophageal epithebum in man. J Anat 1952;86:431-42. Rector LE, Connerley ML. Aberrant mucosa in the esophagus in infants and children. Arch Pathol 1941;31:285-94. Tchertkoff V, Lee BY. Ulcerative esophagitis with heterotopic gastric mucosa. Am J Gastroenterol 1962;37:174-9. Hanson EL, Daly JF, Davis DA. Ulceration associated with an islet of columnar epithelium in the mid-esophagus: new evidence for an acquired etiology of Barrett’s syndrome. J Pediatr Surg 1970;5:370-5. Kerlin P, D’Mellow G, Van Deth A. Barrett’s esophagus: clinical, endoscopic and histologic spectrum in fifty patients. Aust NZ J Med 1986;16:198-205. Raine CH. Ectopic gastric mucosa in the upper esophagus as a cause of dysphagia. Ann Otol Rhino1 Laryngol 1983;92:656. Weaver GA. Upper esophageal web due to a ring formed by a squamocolumnar other
explanation
syndrome]. 11. Hamilton gastric
junction of the
with
ectopic
gastric
Paterson-Kelly,
mucosa
(an-
Plummer-Vinson
Dig Dis Sci 1979;24:959-63. JW, Thune
epithelium
RG. Morrisey
of the cervical
JF. Symptomatic esophagus.
ectopic
Demonstration
12.
13.
14. 15.
16.
17. 18.
Vol. 94. No. 2
of acid production with Congo red. Dig Dis Sci 1986:31:33742. Gerhardt DC, Shuck TJ, Bordeaux RA. Winship DH. Human upper esophageal sphincter. Response to volume, osmotic: and acid stimuli. Gastroenterology 1978;75:268-74. Jabbari M, Goresky CA, Lough J, et al. The inlet patch: heterotopic gastric mucosa in the upper esophagus. Gastroenterofogy 1985;89:352-6. Wang MM, Spear M, McGrew W. Heterotopic gastric mucosa of the esophagus. Sout Med J 1986;79:633-5. Emery JL, Haddadin AJ. Gastric-type epithelium in the upper esophageal pouch in children with trachea-esophageal fistula. J Pediatr Surg 1971;6:449-53. De Ridder PH. Long JC, Shah KK. Columnar epithelium of the cervical esophagus in the adult population, a potentially significant finding (abstr). Gastrointest Endosc 1985;31:128. Rattner HM, McKinley M. Heterotopic gastric mucosa of the upper esophagus (lett). Gastroenterology 1986;90:1309. Carrie A. Adenocarcinoma of the upper end of the esophagus arising from ectopic gastric epithelium. Br J Surg 1950;37:474.
Received March 30, 1987. Accepted September 21. 1987. Address requests for reprints to: P. Kerlin. M.D.. F.R.A.C.P., Director of Gastroenterology, Princess Alexandra Hospital, Ipswich Road, Brisbane, Queensland 4102, Australia. The authors thank Dr. D. Weedon for supplying histopathology data relating to case 2 and Valerie Millar for secretarial assistance.
1988;94:521-4
High Esophageal Stricture: A Complication of “Inlet Patch” Mucosa CHARLES PETER
STEADMAN,
PAUL
KERLIN,
TEAGUE,
and
STEPHENSON
Departments of Gastroenterology Brisbane, Queensland, Australia
and Anatomical
High esophageal stricture associated with a circumferential “inlet patch” of heterotopic gastric mucosa is reported in 2 patients. Biopsy specimens taken from the strictures demonstrated inflamed or ulcerated gastric fundal-type mucosa in both cases. Dilatation of the strictures followed by treatment with histamine Hz-antagonists produced almost complete resolution of dysphagia in both patients.
eterotopic gastric-type columnar epithelium may occur throughout the gastrointestinal tract and in (extraintestinal cysts (11. Gastric-type mucosa in the esophagus may be in continuity with the gastric epithelium [Barrett’s esophagus (a,~)] or be separated from gastric columnar epithelium by normal squamous esophageal mucosa (4). The former pathology is acquired in the setting of chronic gastroesophageal reflux (3), but discrete heterotopic gastric-type mucosa in the esophagus probably represents a congenital abnormality of foregut development (4,s). Either situation can be associated with ulceration in the areas of columnar epithelium (6-8). This report documents 2 patients who experienced high dysphagia and were found to have high esophageal fibrotic (peptic) strictures in association with isolated heterotopic gastric mucosa in the upper esophagus (“inlet patch”).
H
Case
CLINTON
Reports
Case 2 N. H., a 6%yr-old man, complained of intermittent difficulty in swallowing since 1950. A pharyngeal stricture had been diagnosed in 1977 at another institution and treated by dilatation. The cause of the stricture had not been determined. At presentation in August 1985, he complained of progressive dysphagia for solids at the level of the sternal notch. He had mild aching pain radiating to the anterior triangle of the left side of his neck. No weight loss or constitutional symptoms had occurred. Treatment
Pathology, Princess Alexandra
Hospital,
for hypertension with metoprolol (100 mg each morning) had been started before investigation. Barium swallow examination demonstrated a stricture impressing the esophagus (Figure 1) from the left side at the level of the sternal notch. There was mild ballooning of the esophagus proximal to the stricture, but no delay in flow of contrast was seen. A small sliding hiatus hernia was present but only a small amount of reflux could be demonstrated. In August 1985, rigid esophagoscopy demonstrated a stricture that appeared benign at 20 cm from the lips. Dilatation of the stricture was performed. The biopsy specimens taken from the stricture demonstrated columnar epithelium. In view of mildly symptomatic regurgitation and the unusual finding of columnar epithehum in the upper esophagus, the patient underwent further assessment with a flexible fiberoptic esopbagogastroscope. This instrument was able to traverse the stricture. At the level of the stricture (21 cm from the incisors] there was a circumferential red patch of muLosa extending -I cm distally. Beyond the “red” mucosa, normal pearly white esophageal mucosa was observed to 40 cm. Mild linear antral gastritis was seen. The duodenum was normal. No other similar red patches were seen in the esophagus or upper gastrointestinal tract. Biopsy specimens of the inlet patch demonstrated irregular groups of glands in the lamina propria that contained both parietal and chief cells, as’ well as “halo” endocrine cells (Figure 2). Gastric-type mucus cells were present but intestinal metaplasia was not demonstrated. The lamina propria showed a mild chronic inflammatory cell infiltrate. Specimens taken from below the lesion demonstrated normal squamous mucosa. Cimetidine (400 mg twice daily) was instituted with a marked reduction in dysphagia and almost complete loss of left neck pain. At follow-up assessment in May 1986 minimal difficulty in swallowing had occurred during 7 mo of maintenance cimetidine (400 mg daily). At repeat endoscopic examination the circumferential red patch of columnar epithelium at 21 cm was unchanged but no 1986, after ulceration or stricture was seen. In November
0 1988 by the American
Gastroenterological 0016.5085/861$3.50
Association
522
STEADMAN ET AL.
(~ASTKOENTEKOLOGY Vol.
~4. No. 2
biopsy specimen demonstrated a severe acute on chronic gastritis with mild inflammatory dysplasia. Heterotopic gastric mucosa of the upper esophagus, complicated by ulceration and stricture formation, was diagnosed. Ranitidine (150 mg twice daily] was administered with loss of all symptoms. A follow-up endoscopy 2 mo after presentation [June 1986) demonstrated a circumferential patch of red mucosa in the upper esophagus from 18 to 21 cm, with no evidence of active inflammation. The patient
‘igure 1. Radiograph of the uppe ‘r esophagus in patient 1 (antero. posterior view on left. Iateral view on right). There is a stricture at the level of I:he sternal notch with dilatation above.
complaints of mild dysphagia for viscous substances, endoscopy revealed a minor stricture recurrence. Cimetidine (400 mg at night) is to be continued indefinitely. Case
2
A. S., a 42-yr-old seaman, complained of difficulty in swallowing solid food for 5 yr. The obstruction was localized to the suprasternal area. No odynophagia was present and he denied symptoms of heartburn. Intermittent regurgitation of food had occurred but both weight and appetite were stable. Examination demonstrated no evidence of a cervical mass lesion or anemia. A barium swallow examination demonstrated stenosis of the esophagus at the level of the second thoracic vertebra with dilatation of the esophagus above the stenosis. Upper gastrointestinal endoscopy confirmed an erythematous and ulcerated stricture at 20 cm, obscuring the mucosa at that level. Normal esophageal mucosa was seen beyond the stricture. Mild gastritis was present. Dilatation of the stricture to 51F gauge was performed. Biopsy specimens taken from the stricture demonstrated the presence of gastric fundal-type mucosa with severe inflammation and confirmed the presence of ulceration (Figure 3). There was no evidence of malignancy. Specimens taken from the esophagus below the stricture showed normal squamous esophageal mucosa. The gastric
Figure 2. Photomicrographs of biopsy specimens patch in case 1. A. Typical tall columnar
of the inlet
gastric-type mucus cells form the surface. Inflammatory Lells, mostly lymphocytes, are prominent focally in the lamina propria. The glands contain numerous “halo” t?Ildocrine cells (arrows). H&E, x140. B. Several adjacent glands are shown. Both parietal cells ((IrroM’s) and chief cells (arrowheuds) are present. H&E, X560.
E‘ebruary 1988
Figure
‘INLET PATCH”
3. Photomicrograph of a portion of one of the esophageal biopsy fragments obtained from case 2. It consists predominantly of inflamed granulation tissue and amorphous exudate, confirming the presence of ulceration. H&E, x175.
has remained
asymptomatic
on
ranitidine
(150
mg
at
night].
Discussion Heterotopic gastric mucosa in the cervical esophagus is an unusual cause of high dysphagia. Reports of inlet patch-related dysphagia have noted both localized (9) and circumferential (10)patches of gastric mucosa, but the mechanism of dysphagia has not been clarified. Proposals have included web formation at the proximal squamocolumnar junction of the patch (10) and increased cricopharyngeal muscular tone secondary to acid production by the gastric-type mucosa (I I). Increased upper esophageal sphincter tone in response to luminal acid has been demonstrated in controlled experiments (12). Symptom production may relate to the area and distribution of columnar epithelium and to the acidproducing capability. In symptomatic cases, the histology of the heterotopic mucosa has been largely gastric body or fundic type (10,11,13,14). Acid production has been demonstrated by Congo red staining (11)and pH electrode measurement of esophageal a.cidity (13). In the latter study larger areas of heterotopic gastric mucosa produced the largest falls in local pH after pentagastrin stimulation. The endoscopk and histologic appearances indicate a peptic etiology for stricture formation in the cases reported here. The response to drug therapy also supports this hypothesis. Furthermore, the circumferential distri-
ESOPHAGEAL
STRICTIJRE
523
bution of heterotopic gastric mucosa observed may have caused a greater reduction in local pH, with a consequent increase in peptic injury. In contrast to the symptomatic cases, most people who have the inlet patch appear to be asymptomatic. An autopsy study of 1000 children (5)found 7.8% with heterotopic esophageal gastric mucosa, of which 51% was in the upper one-third. Gastric mucosa without parietal cells occurred more frequently than that with parietal cells, but no glands of the latter type were found in the lower one-third of the esophagus. A more recent endoscopic study of 420 sequential endoscopies found an inlet patch prevalence of 3.8% (13),but most were
References 1. Wolff M. Heterotopic gastric report of three new cases with
epithelium in the rectum: a a review of 87 cases of gastric
524
2.
3. 4. 5. 6. 7.
8.
9.
10.
STEADMAN
ET AL
GASTKOEN?‘E:KOI,O(;Y
heterotopia in the alimentary canal. Am J Clin Path01 1971;55:604-16. Barrett NR. Chronic peptic ulcer of the esophagus and esophagitis. Br J Surg 1950;38:175-82. Allison PR, Johnstone AS. The esophagus lined with gastric mucous membrane. Thorax 1953;8:87-101. Johns BAE. Developmental changes in the esophageal epithebum in man. J Anat 1952;86:431-42. Rector LE, Connerley ML. Aberrant mucosa in the esophagus in infants and children. Arch Pathol 1941;31:285-94. Tchertkoff V, Lee BY. Ulcerative esophagitis with heterotopic gastric mucosa. Am J Gastroenterol 1962;37:174-9. Hanson EL, Daly JF, Davis DA. Ulceration associated with an islet of columnar epithelium in the mid-esophagus: new evidence for an acquired etiology of Barrett’s syndrome. J Pediatr Surg 1970;5:370-5. Kerlin P, D’Mellow G, Van Deth A. Barrett’s esophagus: clinical, endoscopic and histologic spectrum in fifty patients. Aust NZ J Med 1986;16:198-205. Raine CH. Ectopic gastric mucosa in the upper esophagus as a cause of dysphagia. Ann Otol Rhino1 Laryngol 1983;92:656. Weaver GA. Upper esophageal web due to a ring formed by a squamocolumnar other
explanation
syndrome]. 11. Hamilton gastric
junction of the
with
ectopic
gastric
Paterson-Kelly,
mucosa
(an-
Plummer-Vinson
Dig Dis Sci 1979;24:959-63. JW, Thune
epithelium
RG. Morrisey
of the cervical
JF. Symptomatic esophagus.
ectopic
Demonstration
12.
13.
14. 15.
16.
17. 18.
Vol. 94. No. 2
of acid production with Congo red. Dig Dis Sci 1986:31:33742. Gerhardt DC, Shuck TJ, Bordeaux RA. Winship DH. Human upper esophageal sphincter. Response to volume, osmotic: and acid stimuli. Gastroenterology 1978;75:268-74. Jabbari M, Goresky CA, Lough J, et al. The inlet patch: heterotopic gastric mucosa in the upper esophagus. Gastroenterofogy 1985;89:352-6. Wang MM, Spear M, McGrew W. Heterotopic gastric mucosa of the esophagus. Sout Med J 1986;79:633-5. Emery JL, Haddadin AJ. Gastric-type epithelium in the upper esophageal pouch in children with trachea-esophageal fistula. J Pediatr Surg 1971;6:449-53. De Ridder PH. Long JC, Shah KK. Columnar epithelium of the cervical esophagus in the adult population, a potentially significant finding (abstr). Gastrointest Endosc 1985;31:128. Rattner HM, McKinley M. Heterotopic gastric mucosa of the upper esophagus (lett). Gastroenterology 1986;90:1309. Carrie A. Adenocarcinoma of the upper end of the esophagus arising from ectopic gastric epithelium. Br J Surg 1950;37:474.
Received March 30, 1987. Accepted September 21. 1987. Address requests for reprints to: P. Kerlin. M.D.. F.R.A.C.P., Director of Gastroenterology, Princess Alexandra Hospital, Ipswich Road, Brisbane, Queensland 4102, Australia. The authors thank Dr. D. Weedon for supplying histopathology data relating to case 2 and Valerie Millar for secretarial assistance.