Journal of Pediatric Surgery xxx (2015) xxx–xxx
Contents lists available at ScienceDirect
Journal of Pediatric Surgery journal homepage: www.elsevier.com/locate/jpedsurg
Heterotopic gastric mucosa mimicking a Meckel’s diverticulum in a young girl☆ James S. Davis a, Alicia C. Hirzel b, Maria M. Rodriguez b, Holly L. Neville c, Juan E. Sola c,⁎ a b c
Department of Surgery, University of Miami Miller School of Medicine, Miami, FL, United States Department of Pathology, University of Miami Miller School of Medicine, Miami, FL, United States Division of Pediatric Surgery, Department of Surgery, University of Miami Miller School of Medicine, Miami, FL, United States
a r t i c l e
i n f o
Article history: Received 7 November 2014 Received in revised form 29 January 2015 Accepted 30 January 2015 Available online xxxx
a b s t r a c t Heterotopic gastric mucosa (HGM) is a rare, but acknowledged source of gastrointestinal pathology in pediatric patients. Sometimes clinically confused with a Meckel’s diverticulum, HGM diagnosis is often made postoperatively by pathology. We present a case of jejunal HGM with a positive technetium pertechnetate scan in the right lower quadrant that resembled a Meckel’s diverticulum. © 2015 Elsevier Inc. All rights reserved.
Key words: Heterotopic gastric mucosa Gastrointestinal bleeding Meckel’s diverticulum
Heterotopic gastric mucosa (HGM) is a rare pathology that consists of gastric secreting rests of tissue present throughout the gastrointestinal tract. The lesion may easily be confused with alternative pathologies more commonly associated with pediatric gastrointestinal bleeding, such as Meckel’s diverticulum, polyps, clotting disorders, arteriovenous fistulas, and inflammatory bowel disease.
normal with stable hemoglobin throughout the duration of her hospital stay. Emergent upper endoscopy revealed no signs of active bleeding. A technetium-99 m pertechnetate (Tc-99 m) scan demonstrated a focus
1. Case report A 7-year-old girl with a past history of jejunal atresia, repaired at 3 days of age, presented with abdominal pain, bright red blood per rectum, and subsequent syncopal episode immediately after vomiting partially digested food. In the Emergency Department, the patient had coffee ground emesis and a hemoglobin of 8.8 g/dL (reference range: 11–16 g/dL), but had a normal blood pressure and heart rate. Upon admission to the floor, the patient experienced a large melanotic stool, followed by hemodynamic instability with a heart rate of 125 beats/min, blood pressure of 80/50 mm Hg, and hemoglobin of 6.5 g/dL. The patient was administered 430 mL of packed red blood cells, additional crystalloid resuscitation, and was transferred to the pediatric intensive care unit (PICU) for further monitoring. In the PICU, the patient had a nasogastric tube placed and was administered a continuous Octreotide drip and Ranitidine. She responded to intravenous fluid resuscitation and remained hemodynamically ☆ Sources of funding: none. ⁎ Corresponding author at: Division of Pediatric Surgery, Department of Surgery, University of Miami Miller School of Medicine, Clinical Research Building, Suite 450K, 1120 N.W. 14th Street, Miami, Florida 33136. Tel.: +1 305 243 5072. E-mail address: [email protected] (J.E. Sola).
Fig. 1. Technetium-99 m pertechnetate scan at forty minutes post-injection demonstrating increased uptake in the patient’s right lower quadrant. Normal uptake of the Tc-99 m nuclide is seen in the stomach and collecting in the bladder.
http://dx.doi.org/10.1016/j.jpedsurg.2015.01.016 0022-3468/© 2015 Elsevier Inc. All rights reserved.
Please cite this article as: Davis JS, et al, Heterotopic gastric mucosa mimicking a Meckel’s diverticulum in a young girl, J Pediatr Surg (2015), http://dx.doi.org/10.1016/j.jpedsurg.2015.01.016
2
J.S. Davis et al. / Journal of Pediatric Surgery xxx (2015) xxx–xxx
Fig. 2. Gross display of the resected jejunum demonstrating diffuse polypoid mucosa and few areas of erythema.
The surgical specimen consisted of a segment of small intestine with multiple smooth, tan sessile polyps extending along the length of the specimen and several small hemorrhagic areas (Fig. 2). Microscopically, extensive, polypoid HGM replaced most of the small intestinal epithelium in the surgical specimen (Fig. 3A and B). The aforementioned images may be compared to a photomicrograph of normal gastric mucosa showing the characteristic histology consisting of foveolar epithelium and underlying glands (Fig. 3C). However, in certain areas gastric glands could be seen directly beneath the intestinal villi. The heterotopic tissue was composed of superficial foveolar epithelium with glands containing a mixture of parietal and chief cells below. There was also expansion of the lamina propria by lymphocytes, plasma cells and eosinophils. In addition, microscopic examination confirmed hemorrhagic foci (Fig. 3D). The patient had an uneventful postoperative course with return of bowel function on post-operative day three and her diet was advanced. She was discharged on post-operative day five, seven days after admission. The patient returned to outpatient clinic on post-operative day 18 for follow-up examination that was normal. In over 1 year, she has had no further episodes of bleeding. 2. Discussion
of Tc-99 m uptake in the right lower quadrant suspicious for a Meckel’s diverticulum (Fig. 1). Additional activity coursing transversely across the abdomen was also noted. Given these findings, the patient was taken to the operating room for laparoscopic excision of a Meckel’s diverticulum. During laparoscopy, multiple dense adhesions were lysed and the terminal ileum was examined without evidence of Meckel’s diverticulum. The operation was converted to an open exploratory laparotomy and the bowel was run multiple times, but no Meckel’s diverticulum could be identified. However, 15 cm of jejunum immediately distal to the previous jejunal atresia anastomosis felt thickened and boggy. The anastomosis was revised, resecting the abnormal area in its entirety.
HGM is a rare, but recognized source of gastrointestinal problems in the pediatric population. The lesion consists of rests of gastrin-secreting tissue, and is most commonly located in the duodenum, but its presence in the jejunum, colon, and esophagus has all been described. HGM may occur in gastrointestinal duplications [1] or within the true gastrointestinal tract itself. While frequently isolated to a specific area of the bowel, HGM has occasionally been reported as disseminated [2]. Complications of HGM include bleeding, obstruction, volvulus, perforation, and even death [3]. Correspondingly, HGM has appeared infrequently in the literature. Approximately 30 cases have been reported beyond the ligament of
Fig. 3. A. Microscopic view of resected jejunum demonstrating polypoid gastric mucosa (2×). B. Higher magnification showing characteristic foveolar epithelium (10×). C. Normal gastric mucosa. D. A focus showing an area of bleeding.
Please cite this article as: Davis JS, et al, Heterotopic gastric mucosa mimicking a Meckel’s diverticulum in a young girl, J Pediatr Surg (2015), http://dx.doi.org/10.1016/j.jpedsurg.2015.01.016
J.S. Davis et al. / Journal of Pediatric Surgery xxx (2015) xxx–xxx
Treitz [4]. Beobeyi et al. provide the best overview, documenting 14 cases in the English language literature [5]. Interestingly, only 2 (14%) of those cases presented with gastrointestinal bleeding, as noted in our case. HGM is difficult to diagnose preoperatively, due to a combination of its rarity and urgent presentation. Often, it is discovered during laparotomy, and more specifically post-operatively upon pathologic examination. However, HGM exhibits strong affinity for Tc-99 m, in a manner similar to a positive Meckel’s scan as in our case (Fig. 1). McAfee et al. first reported technetium uptake by gastric mucosa in 1964 [6], and this was followed closely by the suggested application for detecting Meckel’s diverticulum. The first reported case of HGM identified by Tc-99 m scan dates to 1980 [7]. The most common cause of gastrointestinal bleeding in children is a Meckel’s diverticulum, and a positive Tc-99 m scan confirms this diagnosis. Possible ways to distinguish HGM from Meckel’s diverticulum on Tc-99 m scan include a large area of diffuse technetium uptake, rather than one concentrated area [8]. Accumulation throughout the small intestine may also indicate a diffuse HGM presentation [2]. Some have even suggested augmenting the Tc-99 m scan with intraoperative gamma counter localization [9]. This degree of localization may aid in identifying heterotopic bowel that requires resection. In our case, the Tc-99 m scan exhibited strong uptake in the right lower quadrant. Yet, no Meckel’s diverticulum was identified. It is thought that given the patient’s previous surgery, the jejunal segment of HGM adhered to the right lower quadrant. To the best of our knowledge,
3
there is no reported relationship between jejunal atresia and HGM. Pediatric surgeons should consider HGM in children with gastrointestinal bleeding, a positive Meckel’s scan, but no evidence of Meckel’s diverticulum on exploration. References [1] Kothari PR, Shanker G, Gupta A, et al. Perforation in a jejunal duplication lined by ectopic gastric mucosa. Indian J Gastroenterol 2004;23:152. [2] Heinrichs VM, Kemper MJ, Burdelski M, et al. Disseminated islands of gastric mucosa in jejunum and ileum detected by technetium-99 m-pertechnetate scintigraphy. J Nucl Med 1997;38:818–20. [3] Lambert MP, Heller DS, Bethel C. Extensive gastric heterotopia of the small intestine resulting in massive gastrointestinal bleeding, bowel perforation, and death: report of a case and review of the literature. Pediatr Dev Pathol 2000;3:277–80. [4] Jimenez JC, Emil S, Steinmetz B, et al. Recurrent gastrointestinal tract bleeding secondary to jejunal gastric heterotopia. J Pediatr Surg 2005;40:1654–7. [5] Boybeyi O, Karnak I, Güçer S, et al. Common characteristics of jejunal heterotopic gastric tissue in children: a case report with review of the literature. J Pediatr Surg 2008;43:e19–22. [6] McAfee JG, Fueger CF, Stern HS, et al. Tc-99 M pertechnetate for brain scanning. J Nucl Med 1964;5:811–27. [7] Case records of Massachusetts General Hospital. Case 16-1980: duplication of ileum with gastric mucosa and peptic ulceration with performation. N Engl J Med 1980; 302:958–62. [8] Karakatsanis KG, Chatzipavlidou V, Zafiriadou E, et al. Abdominal scanning with technetium-99 m pertechnetate localizes ectopic gastric mucosa in the jejunum: case report and review of the literature. Eur J Nucl Med 1993;20:547–50. [9] Bueno RC, Hardman JM, Shim WK. Intraoperative localization of ectopic gastric mucosa in the nonduplicated intestinal lumen with technetium 99 m pertechnetate scanning. J Pediatr Surg 2001;36:1720–1.
Please cite this article as: Davis JS, et al, Heterotopic gastric mucosa mimicking a Meckel’s diverticulum in a young girl, J Pediatr Surg (2015), http://dx.doi.org/10.1016/j.jpedsurg.2015.01.016
Contents lists available at ScienceDirect
Journal of Pediatric Surgery journal homepage: www.elsevier.com/locate/jpedsurg
Heterotopic gastric mucosa mimicking a Meckel’s diverticulum in a young girl☆ James S. Davis a, Alicia C. Hirzel b, Maria M. Rodriguez b, Holly L. Neville c, Juan E. Sola c,⁎ a b c
Department of Surgery, University of Miami Miller School of Medicine, Miami, FL, United States Department of Pathology, University of Miami Miller School of Medicine, Miami, FL, United States Division of Pediatric Surgery, Department of Surgery, University of Miami Miller School of Medicine, Miami, FL, United States
a r t i c l e
i n f o
Article history: Received 7 November 2014 Received in revised form 29 January 2015 Accepted 30 January 2015 Available online xxxx
a b s t r a c t Heterotopic gastric mucosa (HGM) is a rare, but acknowledged source of gastrointestinal pathology in pediatric patients. Sometimes clinically confused with a Meckel’s diverticulum, HGM diagnosis is often made postoperatively by pathology. We present a case of jejunal HGM with a positive technetium pertechnetate scan in the right lower quadrant that resembled a Meckel’s diverticulum. © 2015 Elsevier Inc. All rights reserved.
Key words: Heterotopic gastric mucosa Gastrointestinal bleeding Meckel’s diverticulum
Heterotopic gastric mucosa (HGM) is a rare pathology that consists of gastric secreting rests of tissue present throughout the gastrointestinal tract. The lesion may easily be confused with alternative pathologies more commonly associated with pediatric gastrointestinal bleeding, such as Meckel’s diverticulum, polyps, clotting disorders, arteriovenous fistulas, and inflammatory bowel disease.
normal with stable hemoglobin throughout the duration of her hospital stay. Emergent upper endoscopy revealed no signs of active bleeding. A technetium-99 m pertechnetate (Tc-99 m) scan demonstrated a focus
1. Case report A 7-year-old girl with a past history of jejunal atresia, repaired at 3 days of age, presented with abdominal pain, bright red blood per rectum, and subsequent syncopal episode immediately after vomiting partially digested food. In the Emergency Department, the patient had coffee ground emesis and a hemoglobin of 8.8 g/dL (reference range: 11–16 g/dL), but had a normal blood pressure and heart rate. Upon admission to the floor, the patient experienced a large melanotic stool, followed by hemodynamic instability with a heart rate of 125 beats/min, blood pressure of 80/50 mm Hg, and hemoglobin of 6.5 g/dL. The patient was administered 430 mL of packed red blood cells, additional crystalloid resuscitation, and was transferred to the pediatric intensive care unit (PICU) for further monitoring. In the PICU, the patient had a nasogastric tube placed and was administered a continuous Octreotide drip and Ranitidine. She responded to intravenous fluid resuscitation and remained hemodynamically ☆ Sources of funding: none. ⁎ Corresponding author at: Division of Pediatric Surgery, Department of Surgery, University of Miami Miller School of Medicine, Clinical Research Building, Suite 450K, 1120 N.W. 14th Street, Miami, Florida 33136. Tel.: +1 305 243 5072. E-mail address: [email protected] (J.E. Sola).
Fig. 1. Technetium-99 m pertechnetate scan at forty minutes post-injection demonstrating increased uptake in the patient’s right lower quadrant. Normal uptake of the Tc-99 m nuclide is seen in the stomach and collecting in the bladder.
http://dx.doi.org/10.1016/j.jpedsurg.2015.01.016 0022-3468/© 2015 Elsevier Inc. All rights reserved.
Please cite this article as: Davis JS, et al, Heterotopic gastric mucosa mimicking a Meckel’s diverticulum in a young girl, J Pediatr Surg (2015), http://dx.doi.org/10.1016/j.jpedsurg.2015.01.016
2
J.S. Davis et al. / Journal of Pediatric Surgery xxx (2015) xxx–xxx
Fig. 2. Gross display of the resected jejunum demonstrating diffuse polypoid mucosa and few areas of erythema.
The surgical specimen consisted of a segment of small intestine with multiple smooth, tan sessile polyps extending along the length of the specimen and several small hemorrhagic areas (Fig. 2). Microscopically, extensive, polypoid HGM replaced most of the small intestinal epithelium in the surgical specimen (Fig. 3A and B). The aforementioned images may be compared to a photomicrograph of normal gastric mucosa showing the characteristic histology consisting of foveolar epithelium and underlying glands (Fig. 3C). However, in certain areas gastric glands could be seen directly beneath the intestinal villi. The heterotopic tissue was composed of superficial foveolar epithelium with glands containing a mixture of parietal and chief cells below. There was also expansion of the lamina propria by lymphocytes, plasma cells and eosinophils. In addition, microscopic examination confirmed hemorrhagic foci (Fig. 3D). The patient had an uneventful postoperative course with return of bowel function on post-operative day three and her diet was advanced. She was discharged on post-operative day five, seven days after admission. The patient returned to outpatient clinic on post-operative day 18 for follow-up examination that was normal. In over 1 year, she has had no further episodes of bleeding. 2. Discussion
of Tc-99 m uptake in the right lower quadrant suspicious for a Meckel’s diverticulum (Fig. 1). Additional activity coursing transversely across the abdomen was also noted. Given these findings, the patient was taken to the operating room for laparoscopic excision of a Meckel’s diverticulum. During laparoscopy, multiple dense adhesions were lysed and the terminal ileum was examined without evidence of Meckel’s diverticulum. The operation was converted to an open exploratory laparotomy and the bowel was run multiple times, but no Meckel’s diverticulum could be identified. However, 15 cm of jejunum immediately distal to the previous jejunal atresia anastomosis felt thickened and boggy. The anastomosis was revised, resecting the abnormal area in its entirety.
HGM is a rare, but recognized source of gastrointestinal problems in the pediatric population. The lesion consists of rests of gastrin-secreting tissue, and is most commonly located in the duodenum, but its presence in the jejunum, colon, and esophagus has all been described. HGM may occur in gastrointestinal duplications [1] or within the true gastrointestinal tract itself. While frequently isolated to a specific area of the bowel, HGM has occasionally been reported as disseminated [2]. Complications of HGM include bleeding, obstruction, volvulus, perforation, and even death [3]. Correspondingly, HGM has appeared infrequently in the literature. Approximately 30 cases have been reported beyond the ligament of
Fig. 3. A. Microscopic view of resected jejunum demonstrating polypoid gastric mucosa (2×). B. Higher magnification showing characteristic foveolar epithelium (10×). C. Normal gastric mucosa. D. A focus showing an area of bleeding.
Please cite this article as: Davis JS, et al, Heterotopic gastric mucosa mimicking a Meckel’s diverticulum in a young girl, J Pediatr Surg (2015), http://dx.doi.org/10.1016/j.jpedsurg.2015.01.016
J.S. Davis et al. / Journal of Pediatric Surgery xxx (2015) xxx–xxx
Treitz [4]. Beobeyi et al. provide the best overview, documenting 14 cases in the English language literature [5]. Interestingly, only 2 (14%) of those cases presented with gastrointestinal bleeding, as noted in our case. HGM is difficult to diagnose preoperatively, due to a combination of its rarity and urgent presentation. Often, it is discovered during laparotomy, and more specifically post-operatively upon pathologic examination. However, HGM exhibits strong affinity for Tc-99 m, in a manner similar to a positive Meckel’s scan as in our case (Fig. 1). McAfee et al. first reported technetium uptake by gastric mucosa in 1964 [6], and this was followed closely by the suggested application for detecting Meckel’s diverticulum. The first reported case of HGM identified by Tc-99 m scan dates to 1980 [7]. The most common cause of gastrointestinal bleeding in children is a Meckel’s diverticulum, and a positive Tc-99 m scan confirms this diagnosis. Possible ways to distinguish HGM from Meckel’s diverticulum on Tc-99 m scan include a large area of diffuse technetium uptake, rather than one concentrated area [8]. Accumulation throughout the small intestine may also indicate a diffuse HGM presentation [2]. Some have even suggested augmenting the Tc-99 m scan with intraoperative gamma counter localization [9]. This degree of localization may aid in identifying heterotopic bowel that requires resection. In our case, the Tc-99 m scan exhibited strong uptake in the right lower quadrant. Yet, no Meckel’s diverticulum was identified. It is thought that given the patient’s previous surgery, the jejunal segment of HGM adhered to the right lower quadrant. To the best of our knowledge,
3
there is no reported relationship between jejunal atresia and HGM. Pediatric surgeons should consider HGM in children with gastrointestinal bleeding, a positive Meckel’s scan, but no evidence of Meckel’s diverticulum on exploration. References [1] Kothari PR, Shanker G, Gupta A, et al. Perforation in a jejunal duplication lined by ectopic gastric mucosa. Indian J Gastroenterol 2004;23:152. [2] Heinrichs VM, Kemper MJ, Burdelski M, et al. Disseminated islands of gastric mucosa in jejunum and ileum detected by technetium-99 m-pertechnetate scintigraphy. J Nucl Med 1997;38:818–20. [3] Lambert MP, Heller DS, Bethel C. Extensive gastric heterotopia of the small intestine resulting in massive gastrointestinal bleeding, bowel perforation, and death: report of a case and review of the literature. Pediatr Dev Pathol 2000;3:277–80. [4] Jimenez JC, Emil S, Steinmetz B, et al. Recurrent gastrointestinal tract bleeding secondary to jejunal gastric heterotopia. J Pediatr Surg 2005;40:1654–7. [5] Boybeyi O, Karnak I, Güçer S, et al. Common characteristics of jejunal heterotopic gastric tissue in children: a case report with review of the literature. J Pediatr Surg 2008;43:e19–22. [6] McAfee JG, Fueger CF, Stern HS, et al. Tc-99 M pertechnetate for brain scanning. J Nucl Med 1964;5:811–27. [7] Case records of Massachusetts General Hospital. Case 16-1980: duplication of ileum with gastric mucosa and peptic ulceration with performation. N Engl J Med 1980; 302:958–62. [8] Karakatsanis KG, Chatzipavlidou V, Zafiriadou E, et al. Abdominal scanning with technetium-99 m pertechnetate localizes ectopic gastric mucosa in the jejunum: case report and review of the literature. Eur J Nucl Med 1993;20:547–50. [9] Bueno RC, Hardman JM, Shim WK. Intraoperative localization of ectopic gastric mucosa in the nonduplicated intestinal lumen with technetium 99 m pertechnetate scanning. J Pediatr Surg 2001;36:1720–1.
Please cite this article as: Davis JS, et al, Heterotopic gastric mucosa mimicking a Meckel’s diverticulum in a young girl, J Pediatr Surg (2015), http://dx.doi.org/10.1016/j.jpedsurg.2015.01.016
