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Journal of Pediatric Surgery (2010) 45, E9–E12

Foveolar cell hyperplasia at the pyloric canal: an unusualcause of gastric outlet obstruction in 3 young childrenLihua Zhao a, Xianqing Jin a,⁎, Jin Zhub, Xiaoqing Li a, Rui Liang a, Zhenghua Guoa

aDepartment of Pediatric Surgery, Affiliated Children's Hospital, Chongqing Medical University, Chongqing, ChinabDepartment of Pathology, Affiliated Children's Hospital, Chongqing Medical University, Chongqing, China

Received 6 February 2010; revised 28 April 2010; accepted 28 April 2010

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Key words:Young child;Pylorus;Gastric outlet obstruction;Foveolar cell hyperplasia;Diagnosis;Treatment

Abstract Foveolar cell hyperplasia (FCH) in the pylorus is rare in children. Between January 1993 andDecember 2008, 3 children with pyloric canal FCH presented with gastric outlet obstruction and/oranemia and underwent operation because a clear diagnosis could not be established with conservativemanagement. The median age of the children was 18 months. The onset of FCH at the pyloric canal isproblematic, and its diagnosis is difficult to make in pediatric patients. Exploratory laparotomy is areasonable method of establishing the diagnosis and managing the condition.© 2010 Elsevier Inc. All rights reserved.

Foveolar hyperplasia was defined by Robert et al [1] as propose a decision-making protocol for suspected cases

the presence of abnormal branching, twisting, and at least a2-fold elongation of gastric foveolae and pits. Foveolarhyperplasia is not uncommon in the stomach, and mostcases are seen in the gastric corpus or antrum [2]. However,the incidence of gastric foveolar cell hyperplasia (FCH)without the concomitant presence of infantile hypertrophicpyloric stenosis (IHPS) is low. Foveolar cell hyperplasia inthe pyloric canal can induce gastric outlet obstruction and/orhemorrhage; therefore, close attention should be paid tochildren with deteriorating symptoms [3-8]. Previously,standardized methods, such as endoscopy, ultrasound (US),or contrast radiology, were often either unavailable orunsuccessful in diagnosing pyloric canal FCH in youngchildren such as noted in our patients. We report the clinicalcharacteristics, diagnosis, and treatment of FCH and

⁎ Corresponding author. Department of Surgery and Laboratory ofncology, Affiliated Children's Hospital, Chongqing Medical University,uzhong District, Chongqing 86 400014, China. Tel.: +86 23 63638834;x: +86 23 63638824.E-mail address: xianqingjin2010@163.com (X. Jin).

022-3468/$ – see front matter © 2010 Elsevier Inc. All rights reserved.oi:10.1016/j.jpedsurg.2010.04.021

affecting the pylorus.

1. Case reports

Relevant characteristics of the cases are summarized inTable 1.

1.1. Case 1

A 13-month-old boy weighing 8.5 kg was brought to ourclinic because of intermittent, nonbilious vomiting contain-ing coffee grounds, progressive anemia, and weight loss.Laboratory tests showed a red blood cell count of 2.83 × 1012

cells/L, and the hemoglobin level was 72 g/dL. The coffee-ground vomitus and stool specimen were positive for occultblood. Esophagogastroduodenoscopy demonstrated esopha-gitis, gastric outlet obstruction, and gastric retention (Fig. 1).Abdominal B mode sonography showed pyloric stenosis andgland hyperplasia without pyloric muscle hypertrophy. 99TC

Table 1 Clinical characteristics of the cases

Features Age Sex Duration ofsymptoms

Blood hemoglobinlevel (reference value:120-160 g/L)

Size of thepolypoid masses

Location of thepolypoid masses

Histologic condition ofthe biopsy specimens

Case 1 13 mo Male 15 d 72 g/dL 1.5 × 1.5 × 1 cm At the inner posteriorwall of pyloric canal

FCH

Case 2 26 mo Male 40 d 125 g/dL 2.0 × 1.0 × 0.5 cm At the pyloric orifice FCHCase 3 15 mo Male 4 d 88 g/dL 1.0 × 1.0 × 0.8 cm At the anterior wall

of pyloric canalFCH

E10 L. Zhao et al.

scanning was negative for the presence of a Meckel′sdiverticulum. Contrast radiology was not performed becauseof the bleeding. A clear diagnosis could not be obtained, andexploratory laparotomy was performed. A 1.5 × 1.5 × 1.0-cmpedunculated polypoid mass with an ulcerated mucosalsurface and bleeding pedicle was seen at the inner posteriorwall of the pyloric canal. Surgical resection of the mass wasperformed using a longitudinal incision of the pyloric canalthat was closed transversely as a classic pyloroplasty. Biopsyspecimens were obtained and stained with H&E, thehistopathologic features of which were consistent withFCH-specific diagnostic histologic criteria [1]. Therefore,the final diagnosis was FCH. No significant postoperativecomplications occurred. There was no evidence of recur-rence after 10 years of follow-up.

1.2. Case 2

This 26-month-old boy weighing 10.5 kg, referred to ourhospital because of a common cold, had been troubled byintermittent gastric vomiting for 40 days. Basic laboratorystudies revealed a platelet count of 538 × 109 cells/L and aserum chloride level of 78.4 mmol/L. Ultrasound revealedgastric retention but no pyloric hypertrophy or dilation in theduodenum (Fig. 2). Contrast radiography and esophagogas-troduodenoscopy revealed esophagitis and complete gastricoutlet obstruction. Exploratory laparotomy was performed,and a 2.0 × 1.0 × 0.5-cm pedunculated mass with a smoothsurface was seen at the pyloric orifice and completely

Fig. 1 Gastroduodenoscopy images from case 1 indicated signs of esolens could not get through the pylorus (B, arrow), and much undigested fowas erythematous (D, arrow).

obstructed the pyloric canal. Surgical resection wasperformed. Histopathologic examination showed that thespecimen exhibited FCH without dysplasia or malignancy(Fig. 3). The patient has been observed for 9 years with nofurther anemia or vomiting observed.

1.3. Case 3

This 15-month-old boy weighing 8.5 kg was admittedbecause of diarrhea and persistent vomiting with coffee-ground gastric content. Laboratory data were normal expectfor the hemoglobin level (Table 1). An upper gastrointestinalcontrast study demonstrated gastric dilatation, pyloric stenosisand gastroesophageal reflux. Ultrasound and esophagogas-troduodenoscopy noted duodenogastroesophagoreflux andileus. Exploratory laparotomy was performed and demon-strated that the pyloric canal mucosa was thickened; a 1.0 ×1.0 × 0.8-cm pedunculated mass with superficial erosions wasseen at the anterior wall of pyloric canal, causing partialobstruction. Surgical resection was performed. Histologicexamination showed FCH. The postoperative course wasuneventful, and the patient is well after 9 years of follow-up.

2. Discussion

Gastric outlet obstruction because of FCH is a rarecondition in children. The foveolae are pits in the gastric

phagus with erosion, hyperemia and edema (A, arrow). The cameraod was deposited (C, arrow). The sinus ventriculi mucous membrane

Fig. 2 Ultrasound examination images from case 2 revealed obvious gastric content retention. It was difficult to get through the pylorusbecause the gastric contents seen on the dynamic scan, and the pyloric canal intraluminal architecture were unclear, although the pylorus wasclear and had no thickening or stenosis (arrow).

E11Foveolar cell hyperplasia at the pyloric canal

mucosa that are normally longest in the antrum. Foveolar cellhyperplasia is because of deepening of the foveolae andhyperplasia of the foveolar cells with increased mucinproduction [9]. The etiology of FCH in young children isunclear at this time. Katz et al [10] reported that FCH isgenerally idiopathic with no specific primary cause identi-fied, as was seen in our cases [4]. However, others havereported that it may be related to prostaglandin therapy,severe upper airway obstruction, or Helicobacter pyloriinfection [7,11]. Yasunaga et al [12] reported that, analogousto H pylori-induced foveolar hyperplasia, chronic inflam-mation may induce interleukin production, leading to thedevelopment of foveolar hyperplasia in some cases. The

Fig. 3 Photomicrograph of a biopsy specimen from case 2demonstrated elongated and twisted gastric foveolae and pits withno inflammation and expansion of the lamina propria (H&E,original magnification ×40).

presence of H pylori infection was not detected in ourpatients although it is an important factor in the pathogenesisof FCH in adults. Our patients had not received any specialmedications, had been previously healthy, their clinicalhistories were short, and the onset of symptoms wereimmediate. The intraluminal pyloric masses in our caseswere polypoid in nature, and the remaining bowel wasessentially normal. These findings suggest there were nospecific predisposing events in our cases.

Foveolar cell hyperplasia in the pyloric canal can causegastric outlet obstruction, dyspepsia, and/or anemia andshould be diagnosed as soon as possible. Nevertheless,achieving a diagnosis of FCH in our patients was difficult.Although some investigators reported that pyloric canalpolyps could be diagnosed by endoscopy, US, and/orcontrast radiology [3,5-7], in our patients, because of theobstruction of the pyloric canal by the polypoid mass, neitherthe endoscope nor barium contrast could traverse through thepyloric canal (Fig. 1). The excessive gas in the dilatedstomach made it hard to find the mass clearly by US. It wasdifficult for us to establish a definitive diagnosis in ourpatients, and therefore, exploratory laparotomy was per-formed. All 3 patients were found to have a mass that partlyor completely obstructed the pyloric canal, which resulted inintermittent or progressive gastric outlet obstruction, anemia,and malnutrition.

The pathologic characteristics of FCH in our patientswere clearly different from patients with IHPS because thegastric outlet obstruction was not because of hypertrophy ofthe muscular layers of the pylorus (shown in Fig. 2).However, it has been reported that foveolar hyperplasia has acausal role in postoperative persistent gastric outlet obstruc-tion after pyloromyotomy for IHPS [13-15]. Foveolar cellhyperplasia in our patients occurred in a group of older

E12 L. Zhao et al.

children and is likely a different disease entity and not theremnant of resolved IHPS. Of interest is the syndromedescribed in our patients has many parallels with childhoodMenetrier's disease. Childhood Menetrier's disease is a raredisease of unknown etiology characterized by protein-losinghypertrophic gastropathy and hypoproteinemia. Neverthe-less, both the focus of Menetrier's on foveolar hyperplasia inthe fundic region of the stomach and the clinical character-istics of our patients did not support the diagnosis ofMenetrier's [16].

Different modes of therapy, such as cessation ofprostaglandin infusion, transpyloric feeding, and surgerywith removal of the pylorus (a somewhat radical measure),have been used in management of previously reported casesof gastric outlet obstruction associated with gastric masses.Operative management in the current cases involved removalof the polypoid masses and pyloroplasty. All of our patientsresponded well to a longitudinal incision of the pylorus withclassic pyloroplasty. In these cases, endoscopic resection wasnot possible because of the inability of the endoscope to passthrough the pyloric canal. Symptoms completely disap-peared after the excision of masses, and the children haveremained well on long-term follow-up.

3. Conclusions

Foveolar cell hyperplasia of the pylorus is a rare disease inyoung children and is associated with gastric outletobstruction, the onset of which is severe and problematic.Symptoms included vomiting and hemorrhage that at thetime (10 years ago) could not be relieved by standardtreatment. Foveolar cell hyperplasia should be consideredwhen a young child (≤3 years) presents with features ofgastric outlet obstruction and/or anemia. Exploratory lapa-rotomy may be an effective way to diagnose and manage thiscondition when endoscopy, US, or other less invasivemethods are unsuccessful. The importance of early recogni-tion and prompt intervention should be stressed.

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