Journal of Indian Association of Pediatric Surgeons
Journal of Indian Association of Pediatric Surgeons
                                                   Official journal of the Indian Association of Pediatric Surgeons                           
Year : 2022  |  Volume : 27  |  Issue : 1  |  Page : 38--41

Primary acquired cicatrizing gastric outlet obstruction in children


Manish Pathak1, Rahul Saxena2, Hardik Patel1, Arvind Sinha1,  
1 Department of Pediatric Surgery, All India Institute of Medical Sciences, Jodhpur, Rajasthan, India
2 Department of General Surgery, RNT Medical College, Udaipur, Rajasthan, India

Correspondence Address:
Dr. Manish Pathak
Department of Pediatric Surgery, All India Institute of Medical Sciences, Jodhpur - 342 005, Rajasthan
India

Abstract

Background: Gastric outlet obstruction (GOO) is a very rare condition in children, when infantile hypertrophic pyloric stenosis is excluded as a cause. Five cases of pediatric GOO were successfully managed at our institute. Materials and Methods: We retrospectively evaluated children with idiopathic GOO from 2009 to 2016. Medical records were reviewed for demographic data including age, sex, presenting symptoms, diagnostic investigations, treatment methods, complications, and long-term follow-up. Results: During 7 year period (2009–2016), 5 cases of GOO admitted to our hospital with a history of persistent vomiting. The vomiting used to occur 12–18 h after meal and vomitus contained foul-smelling undigested meal. There was no history of any caustic ingestion. Their age ranged from 2 to 10 years, with a median age of 6 years. Out of 5 cases, 2 were females and the rest were males. The diagnosis of idiopathic GOO was confirmed by history, clinical examination, contrast study, endoscopy, and endoscopic mucosal biopsy. The remarkable finding was the cicatrization causing stricture of the pyloric region in all cases. Out of 5 cases; we have done Heineke-Mikulicz pyloroplasty in 1 case, V-Y advancement antropyloroplasty in 2 cases and Kimura's Diamond-shaped Gastroduodenostomy in 2 cases. The length of the strictured segment determined the type of surgical procedure. All cases did well postoperatively with no mortality. Conclusions: Length of the stricture determines the procedure to be done. In long and narrow stricture V-Y advancement antropyloroplasty and in small stricture Kimura's diamond-shaped Gastroduodenostomy are better procedures to be done. Heineke-Mikulicz pyloroplasty should be avoided as it is difficult to suture transversely after vertical incision because of the presence of fibrosis in the strictured segment.



How to cite this article:
Pathak M, Saxena R, Patel H, Sinha A. Primary acquired cicatrizing gastric outlet obstruction in children.J Indian Assoc Pediatr Surg 2022;27:38-41


How to cite this URL:
Pathak M, Saxena R, Patel H, Sinha A. Primary acquired cicatrizing gastric outlet obstruction in children. J Indian Assoc Pediatr Surg [serial online] 2022 [cited 2022 May 16 ];27:38-41
Available from: https://www.jiaps.com/text.asp?2022/27/1/38/335561


Full Text



 Introduction



After excluding infantile hypertrophic pyloric stenosis, gastric outlet obstruction (GOO) is rare condition in children; causes include caustic ingestion, peptic ulceration, tumors, chronic granulomatous condition, eosinophilic gastroenteritis, and idiopathic.[1],[2],[3],[4] Out of which idiopathic or primary acquired GOO is extremely rare condition. Patients present with persistent vomiting, early satiety, postprandial fullness, and rapid loss of weight. Barium study, ultrasonography, endoscopy, and biopsy need to exclude other causes. Helicobactor pylori (H. pylori) infection is not common in children but in adolescents, Urease test and Giemsa staining can be useful to exclude H. pylori as a cause. Primary acquired GOO with normal pylorus (Jodhpur disease) was first described by Sharma et al.[2] Heineke-Mikulicz pyloroplasty is the procedure of choice in such condition with the excellent outcome because the pylorus is essentially normal without any cicatrization. In our 5 cases of primary acquired GOO, all patients had cicatrization at the pylorus with variable lengths of strictured pylorus. This entity of primary acquired GOO with cicatrization is extremely rare and only one case has been described in the literature before.[5] The purpose of this study is to describe this rare entity of primary acquired GOO with cicatrization and formulate the management strategy for it.

 Materials and Methods



We retrospectively evaluated children with idiopathic GOO from 2009 to 2016. Medical records were reviewed for demographic data including age, sex, presenting symptoms, diagnostic investigations, treatment methods, complications, and long-term follow-up.

 Results



During 7 year period (2009–2016), 5 cases of GOO were admitted to our hospital with a history of persistent vomiting. According to the Modified Kuppuswamy socioeconomic scale, 2 families belonged to lower middle class, 3 to upper lower class and one to lower socioeconomic class. The vomiting appeared 12–18 h after meal and vomitus contained foul-smelling undigested meal. There was no history of any caustic ingestion or ingestion of any indigenous herb or self-medication. The median age was 6 years (range-2–10 years). Out of 5 cases, 2 were females and the rest were males. The median duration of follow was 6 years (4–8 years).

All patients presented with epigastric distension after feeds, nonbilious vomiting, and loss of weight. The diagnosis was confirmed by history, clinical examination, contrast study, endoscopy, and endoscopic biopsy. Serum gastrin level was normal in all patients. Rapid urease test also found to be negative in all cases which excluded H. pylori as the cause. Upper gastrointestinal (GI) contrast studies showed normal esophagus, dilated stomach, and stenosed pylorus with hold up of contrast in the stomach and no/little passage of contrast across pylorus [Figure 1]. Upper GI endoscopy reveals pyloric stenosis with dilated stomach and normal esophagus. Endoscopy-guided gastric mucosal biopsy was unremarkable for any tumor, peptic ulceration, eosinophilic gastroenteritis, or any evidence of Helicobacter pylori (using Giemsa stain). Thus diagnosis was made as idiopathic GOO in all 5 cases.{Figure 1}

The initial trial of medical treatment using proton pump inhibitors failed to show any improvement. Hence, elective surgery was performed in all five cases. In first case we did Heineke-Mikulicz pyloroplasty. However, we noted that it is difficult to suture transversely after vertical incision due to the presence of cicatrization in the strictured segment. In two cases, strictured segment was small so we performed Kimura's Diamond-shaped Gastroduodenostomy. In the other 2 cases, patients' general condition was poor so we did feeding jejunostomy, following which nutritional status improved. In these cases, stricture was long and narrow so we did V-Y advancement antropyloroplasty as definitive surgery after 6 weeks of feeding jejunostomy.

All patients were on regular follow-up showing weight gain and improvement of nutritional status. All patients did well postoperatively with no mortality. Postoperative contrast studies showed the passage of contrast across pylorus and opacification of distal bowel [Figure 2].{Figure 2}

 Discussion



Acquired GOO is very rare condition in children, causes include caustic ingestion, peptic ulceration, tumors, chronic granulomatous condition, eosinophilic gastroenteritis, and idiopathic.[1],[2],[3],[4],[5]

Patients with GOO present with nonbilious vomiting, early satiety, and postprandial abdominal distension. Rapid loss of weight is also a common presentation in children.

Upper GI contrast studies remain a useful diagnostic tool in patients, which usually show dilated stomach, strictured pylorus with no/little passage of contrast across pylorus.[6] Early fiberoptic endoscopy is recommended as the diagnostic method of choice, which reveals the degree of pylorus narrowing, dilated stomach and any associated esophageal or gastric abnormality.[7] Moreover, endoscopy helps to take biopsy which is required to rule out other causes of GOO such as tumors, peptic ulcer, eosinophilic gastroenteritis, or any evidence of Helicobacter pylori by using Giemsa staining.

Idiopathic acquired GOO is also called primary acquired GOO. Primary acquired GOO in infancy and childhood was first described in India in 1997 by Sharma et al. and he labeled it as “Jodhpur disease.”[2],[3]

Diagnosis of Jodhpur disease is aided by ultrasonography that shows normal pyloric canal with no pyloric muscle hypertrophy, while upper GI contrast study shows a large stomach with increased gastric emptying time. Gastroscopy reveals no intra-luminal pathology with normal gastric mucosal rugosities in a large-size stomach with narrow pyloric canal. Histopathologically, there is normal cellular pattern of all coats without inflammatory, fibro-proliferative, or neoplastic cells with a normal number of ganglion cells.[3] The procedure of choice is Heineke-Mikulicz pyloroplasty with excellent outcome. In contrast to Jodhpur disease, cicatrization was present in all our cases. Ratan et al. also reported a case of acquired cicatrizing nonspecific inflammation-causing GOO in infancy.[5] Our series is the largest series reporting such an entity. We consider it a variant of primary acquired GOO in infancy and childhood and should be labeled as primary acquired cicatrizing GOO in infancy and childhood. It is an acquired condition of idiopathic etiology with the presence of cicatrization at the pylorus. It is a diagnosis of exclusion where secondary causes like acid-peptic disease, eosinophilic gastroenteritis, neoplasm, and chemical injury have been ruled out.

We propose a modification to the existing classification of GOO proposed by Sharma et al. with subdivision of primary acquired GOO into the noncicatrizing and cicatrizing types [Table 1].{Table 1}

Heineke-Mikulicz pyloroplasty is not a good procedure in such cases due to the presence of cicatrization and fibrosis in the affected segment. In the presence of cicatrization either diamond gatrodudenostomy based on Kimura's technique or Y-V advancement pylorolasty should be preferred depending on the length of the affected segment.[8] If the strictured segment is small, Kimura's gastro-duodenostomy is feasible. However, in the presence of long strictured segment, the Y-V advancement pyloroplasty should be preferred.

Other techniques have also been described for the management of GOO such as endoscopic balloon dilatation[9] and/or intralesional steroid injection,[10] partial gastric resection, pyloroplasty,[11],[12] and gastroenterostomy.[13] A recent review of literature suggests that endoscopic balloon dilatation with or without intra-lesional steroid injection proves to be useful in a few cases, but the long-term results are still awaited.[11],[12] Gastrojejunostomy is a safer alternative in the presence of extensive perigastric adhesion, unhealthy duodenum, and poor general condition.[13],[14]

If patient's nutritional status is poor, the definitive procedure should be delayed. In such cases feeding jejunostomy is preferred to improve patient's general condition initially followed by definitive procedure about 6 weeks later.

Although these patients are doing well during the medium-term follow-up ranging from 4 to 8 years, the long-term outcome of these surgical procedures that lead to disruption of the pyloantral mechanism are yet to be seen. We do not have any existing recommendations regarding the duration and nature of follow-up. Theoretically, these patients may develop gastric metaplasia of the duodenum due to the chronic exposure of the duodenum to the acidic gastric chime following disruption of the pyloric mechanism. Hence, we intend to do upper GI endoscopy in all our patients, 10 years after the surgery.

 Conclusions



Primary acquired cicatrizing GOO is rare entity in children. Upper GI contrast study suggests and endoscopy remains the investigation of choice for diagnosis. Length of stricture determines the procedure to be done. In long and narrow stricture V-Y advancement antropyloroplasty and in small stricture Kimura's diamond-shaped Gastroduodenostomy are better procedures to be done. Heineke-Mikulicz pyloroplasty should be avoided as it is difficult to suture transversely after vertical incision because of the presence of fibrosis in strictured segments.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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