Severe Dumping Syndrome in a 6-year-old Girl with Congenital Microgastria Treated by Hunt-Lawrence Pouch

Filisetti, Claudia; Maestri, Luciano; Meroni, Milena; Marinoni, Federica; Riccipetitoni, Giovanna

doi:10.1055/s-0037-1601305

Cited by 9 publications

(12 citation statements)

References 15 publications

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“…Noteworthy is the fact that in only four patients (4/51; 8%), microgastria was present as an isolated anomaly. 1,16,38,39 The most common associated anomalies are defects of the spleen and the limbs.…”

Section: Resultsmentioning

confidence: 99%

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Treatment of Congenital Microgastria

et al. 2020

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Introduction Congenital microgastria is an extremely rare birth defect. The aim of this study was to present an overview of existing literature on the treatment of microgastria. Materials and Methods The term “microgastria” was used in a PubMed and Medline search. Since merely case reports were found, only a narrative synthesis with limited statistical analysis can be given. Data of different treatment modalities were collected and divided into two groups: conservative or less invasive treatment (C/LT, i.e., modified diet or a gastrostomy/jejunostomy) and extensive gastric surgery (EGS, i.e., Hunt–Lawrence pouch or total esophageal gastric dissociation). Clinical outcome parameters (nutrition, growth pattern, and mortality) were compared. Results Out of 73 articles published from 1973 to 2019, 38 articles describing 51 cases were included. In four patients, microgastria was an isolated anomaly (8%). Type of treatment was described in only 46 patients, 19 were treated by C/LT. Mortality was 9/19 (47%) in the C/LT group versus 4/27 (15%) in the EGS group (chi-square = 5.829, p = 0.016, Fisher = 0.022). There was a negative correlation between the invasiveness of the treatment and both mortality (r = −0.356, p = 0.015) and comorbidity (r = −0.506, p <0.001). Patients in the C/LT group had significantly more comorbidity than in the EGS group (mean = 4.32 vs. 2.26, p = 0.001). There was a positive correlation between comorbidity and mortality (r = 0.400, p = 0.006). Median follow-up was 42 months (range: 1–240). Type and way of nutrition were poorly described. In at least 9 of the 33 surviving patients, oral feeding was reported as normal, of whom 8 belonged to the EGS group. In all patients, growth could be acknowledged, but in comparison to peers, final body length was less. There was no difference in final body length between the two treatment groups. Conclusion In patients with congenital microgastria, only minimal differences in clinical outcome in terms of type of nutrition and body growth were found when C/LT was compared with treatment by EGS. Mortality was significantly higher in the first group as well as the amount of comorbidities.

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Section: Resultsmentioning

confidence: 99%

“…4 Since then, this procedure has been applied in roughly 18 cases with variable success. 1,4,5,7,8,10,13,14,16,20,32,38,39 Kawaguchi et al introduced the use of the TEGD. 26 Up until now, this procedure has been performed five times, with good outcome being reported on gastroesophageal reflux symptoms.…”

Section: Discussionmentioning

confidence: 99%

Treatment of Congenital Microgastria

et al. 2020

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“…then, this procedure has been applied in roughly 18 cases with variable success. 1,4,5,7,8,10,13,14,16,20,32,38,39 Kawaguchi et al introduced the use of the TEGD. 26 Up until now, this procedure has been performed five times, with good outcome being reported on gastroesophageal reflux symptoms.…”

Section: Discussionmentioning

confidence: 99%

Untitled

2021

Eur J Pediatr Surg

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Introduction Congenital microgastria is an extremely rare birth defect. The aim of this study was to present an overview of existing literature on the treatment of microgastria. Materials and Methods The term "microgastria" was used in a PubMed and Medline search. Since merely case reports were found, only a narrative synthesis with limited statistical analysis can be given. Data of different treatment modalities were collected and divided into two groups: conservative or less invasive treatment (C/LT, i.e., modified diet or a gastrostomy/jejunostomy) and extensive gastric surgery (EGS, i.e., Hunt-Lawrence pouch or total esophageal gastric dissociation). Clinical outcome parameters (nutrition, growth pattern, and mortality) were compared. Results Out of 73 articles published from 1973 to 2019, 38 articles describing 51 cases were included. In four patients, microgastria was an isolated anomaly (8%). Type of treatment was described in only 46 patients, 19 were treated by C/LT. Mortality was 9/19 (47%) in the C/LT group versus 4/27 (15%) in the EGS group (chi-square ¼ 5.829, p ¼ 0.016, Fisher ¼ 0.022). There was a negative correlation between the invasiveness of the treatment and both mortality (r ¼ À0.356, p ¼ 0.015) and comorbidity (r ¼ À0.506, p <0.001). Patients in the C/LT group had significantly more comorbidity than in the EGS group (mean ¼ 4.32 vs. 2.26, p ¼ 0.001). There was a positive correlation between comorbidity and mortality (r ¼ 0.400, p ¼ 0.006). Median follow-up was 42 months (range: 1-240). Type and way of nutrition were poorly described. In at least 9 of the 33 surviving patients, oral feeding was reported as normal, of whom 8 belonged to the EGS group. In all patients, growth could be acknowledged, but in comparison to peers, final body length was less. There was no difference in final body length between the two treatment groups. Conclusion In patients with congenital microgastria, only minimal differences in clinical outcome in terms of type of nutrition and body growth were found when C/LT was compared with treatment by EGS. Mortality was significantly higher in the first group as well as the amount of comorbidities.

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“…A group in Japan used CGM in infants after fundoplication and found wide sugar fluctuation in two out of four patients 5. There have been case reports of late dumping syndrome in patients who underwent oesophageal surgery/gastrostomy/gastrojejunostomy, where the tip had migrated further into the duodenum 6–9. It is being increasingly recognised that dumping syndrome may be more prevalent than previously recognised and the diagnosis is often missed.…”

Section: Discussionmentioning

confidence: 99%

Late dumping syndrome in an infant on feeding jejunostomy

Jain

Gupta²,

Jat

et al. 2019

BMJ Case Rep

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This article presents the case of a 3-month-old male child, who while on bolus jejunostomy tube feeds, developed recurrent episodes of hypoglycaemia. This infant had presented with failure to thrive with moderate gastroesophageal reflux necessitating a feeding jejunostomy. The infant was started on bolus feeds through the jejunostomy tube but developed recurrent episodes of hypoglycaemia. On evaluation, these episodes were hyperinsulinaemic and the baby was subsequently diagnosed with a late dumping syndrome. On changing the feeds to a continuous infusion and by eliminating added sugar from the feeds, the glucose fluctuations resolved. Dumping syndrome is a well-known complication in adults undergoing gastric surgeries. In the paediatric age group, dumping syndrome has been reported rarely, most commonly as a complication of Nissen fundoplication.

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Severe Dumping Syndrome in a 6-year-old Girl with Congenital Microgastria Treated by Hunt-Lawrence Pouch

Abstract: Microgastria is a rare congenital condition often associated with other anomalies. In the present report we describe the case of a 6-year-old girl with isolated CM who presented with dumping syndrome successfully treated by a Hunt-Lawrence pouch.

Cited by 9 publications

References 15 publications

Treatment of Congenital Microgastria

Treatment of Congenital Microgastria

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Late dumping syndrome in an infant on feeding jejunostomy

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