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Jodhpur disease revisited: a rare cause of severe protein energy malnutrition.

Aggarwal M, Sood V, Kumar A, Saurabh K - Ann Gastroenterol (2012)

Bottom Line: A 3.5-year-old grossly cachectic female child presenting with recurrent vomiting, fever, abdominal distention, abdominal pain and severe weight loss was evaluated for the cause of severe protein energy malnutrition.Investigation revealed a massively dilated stomach with delayed gastric emptying and normal pylorus.To conclude, this entity should always be included in the differential diagnosis of gastric outlet obstruction with severe malnutrition especially in older children.

View Article: PubMed Central - PubMed

Affiliation: Department of Pediatrics, VMMC and Safdarjang Hospital, New Delhi, India.

ABSTRACT
A 3.5-year-old grossly cachectic female child presenting with recurrent vomiting, fever, abdominal distention, abdominal pain and severe weight loss was evaluated for the cause of severe protein energy malnutrition. Investigation revealed a massively dilated stomach with delayed gastric emptying and normal pylorus. On exploratory laparotomy, diagnosis of primary acquired gastric outlet obstruction (Jodhpur disease) was confirmed and she underwent pyloroplasty with uneventful post-operative period. To conclude, this entity should always be included in the differential diagnosis of gastric outlet obstruction with severe malnutrition especially in older children.

No MeSH data available.


Related in: MedlinePlus

Perioperative picture with normal smooth muscle thickness, slight increase in thickness of mucosal and submucosal layer, no intraluminal or extraluminal cause identified
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Figure 2: Perioperative picture with normal smooth muscle thickness, slight increase in thickness of mucosal and submucosal layer, no intraluminal or extraluminal cause identified

Mentions: On investigation, her hemogram and chest X-ray were normal, but serum electrolytes were abnormal (hyponatremia with hypokalemia). HIV serology, PPD test and gastric aspirates (n=3) for tuberculosis were negative. Stool examination was negative for ova, cysts or opportunistic pathogens. X-ray abdomen showed no air-fluid level but only gaseous distention of stomach and intestine. Ultrasound of abdomen showed distended stomach with normal pyloric canal. Barium meal showed grossly distended stomach with slow passage of barium from the antero-pyloric region distally (Fig. 1). Suspecting gastric outlet obstruction, exploratory laparotomy was done that revealed hugely distended stomach with normal smooth muscle thickness but no intra-luminal valve, ring or diaphragm or extra-luminal compression was seen (Fig. 2). Heineke-Mikulicz pyloroplasty was done. Pyloric biopsy showed sub-mucosal edema and mild congestion and no inflammatory cells were seen. The diagnosis of primary acquired gastric outlet obstruction during infancy and childhood (Jodhpur disease) with severe malnutrition was made. The child recovered well postoperatively, became asymptomatic gradually and gained 2.5 kg in one month post surgery.


Jodhpur disease revisited: a rare cause of severe protein energy malnutrition.

Aggarwal M, Sood V, Kumar A, Saurabh K - Ann Gastroenterol (2012)

Perioperative picture with normal smooth muscle thickness, slight increase in thickness of mucosal and submucosal layer, no intraluminal or extraluminal cause identified
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC3959372&req=5

Figure 2: Perioperative picture with normal smooth muscle thickness, slight increase in thickness of mucosal and submucosal layer, no intraluminal or extraluminal cause identified
Mentions: On investigation, her hemogram and chest X-ray were normal, but serum electrolytes were abnormal (hyponatremia with hypokalemia). HIV serology, PPD test and gastric aspirates (n=3) for tuberculosis were negative. Stool examination was negative for ova, cysts or opportunistic pathogens. X-ray abdomen showed no air-fluid level but only gaseous distention of stomach and intestine. Ultrasound of abdomen showed distended stomach with normal pyloric canal. Barium meal showed grossly distended stomach with slow passage of barium from the antero-pyloric region distally (Fig. 1). Suspecting gastric outlet obstruction, exploratory laparotomy was done that revealed hugely distended stomach with normal smooth muscle thickness but no intra-luminal valve, ring or diaphragm or extra-luminal compression was seen (Fig. 2). Heineke-Mikulicz pyloroplasty was done. Pyloric biopsy showed sub-mucosal edema and mild congestion and no inflammatory cells were seen. The diagnosis of primary acquired gastric outlet obstruction during infancy and childhood (Jodhpur disease) with severe malnutrition was made. The child recovered well postoperatively, became asymptomatic gradually and gained 2.5 kg in one month post surgery.

Bottom Line: A 3.5-year-old grossly cachectic female child presenting with recurrent vomiting, fever, abdominal distention, abdominal pain and severe weight loss was evaluated for the cause of severe protein energy malnutrition.Investigation revealed a massively dilated stomach with delayed gastric emptying and normal pylorus.To conclude, this entity should always be included in the differential diagnosis of gastric outlet obstruction with severe malnutrition especially in older children.

View Article: PubMed Central - PubMed

Affiliation: Department of Pediatrics, VMMC and Safdarjang Hospital, New Delhi, India.

ABSTRACT
A 3.5-year-old grossly cachectic female child presenting with recurrent vomiting, fever, abdominal distention, abdominal pain and severe weight loss was evaluated for the cause of severe protein energy malnutrition. Investigation revealed a massively dilated stomach with delayed gastric emptying and normal pylorus. On exploratory laparotomy, diagnosis of primary acquired gastric outlet obstruction (Jodhpur disease) was confirmed and she underwent pyloroplasty with uneventful post-operative period. To conclude, this entity should always be included in the differential diagnosis of gastric outlet obstruction with severe malnutrition especially in older children.

No MeSH data available.


Related in: MedlinePlus