J Ped Surg Case Reports 1 (2013) 47e49

Contents lists available at SciVerse ScienceDirect

Journal of Pediatric Surgery CASE REPORTS

journal homepage: www.jpscasereports.com

Gastric : A rare association with hyperamylasemia

Ramnik Patel*, Bharat More, C.K. Sinha, Ashok Rajimwale, G.K. Ninan

Department of Paediatric Surgery, Directorate of Children’s Services, University Hospitals of Leicester NHS Trust, Leicester Royal Infirmary, Infirmary Square, Leicester LE1 5WW, UK article info abstract

Article history: We wish to report two rare and potentially fatal cases of acute gastric volvulus in children associated Received 23 January 2013 with hyperamylasemia. Plain films, CT scan, contrast studies and upper gastrointestinal were Received in revised form helpful in confirming the diagnosis. They underwent emergency laparotomy with devolvulus and 18 February 2013 temporary gastrostomy in the first case and posterior gastropexy in association with plication of even- Accepted 19 February 2013 trated left hemi-diaphragm with good recovery. Ó 2013 Elsevier Inc. All rights reserved.

Key words: Gastric volvulus Hyperamylasemia Diaphragmatic anomalies

Gastric volvulus is a rare surgical emergency in children. Its admission she became tender in the epigastrium. The amylase level association with hyperamylasemia without the evidence of was raised at 520 IU. The white cell count remained elevated at is a new observation in our cases. It is vital to know this 31.4 109/L with a CRP of 193 mg/L. Breath sounds were decreased association and differentiate it from , as managing at the left base and a chest radiograph demonstrated a raised left a child with gastric volvulus conservatively could be catastrophic. hemi-diaphragm. Laboratory tests are usually not diagnostic for gastric volvulus; At this stage, a CT scan was performed which revealed normal however, elevated serum alkaline phosphatase and hyper- pancreas but a distended, fluid filled with twisted splenic amylasemia have been reported [1]. There has been a report of pure vessels interposed between fundus and body. The picture was hyperamylasemia in adult case of gastric volvulus leading to strongly suggestive of left posterior eventration with associated a missed diagnosis of pancreatitis [2]. gastric volvulus (Fig. 1). Subsequent upper gastrointestinal contrast study confirmed organo-axial volvulus of the stomach. At laparotomy, a gastrotomy was necessary to decompress the 1. Case 1 stomach and 500 mL of fluid and debris were drained. A temporary Stamm gastrostomy was fashioned to anchor the stomach to the An 11-year-old girl presented with a 24-h history of non-bilious anterior abdominal wall. Mild diaphragmatic eventration was noted and left iliac fossa pain. There was no history of , on the left hand side, which did not warrant any repair. Post- or urinary symptoms. On examination she was dehy- operative recovery was complicated by a left pleural effusion on day drated and tender in the left iliac fossa and left loin. White cell 2. This resolved in next three days and she was discharged home. At count was raised with neutrophilia. The C-reactive protein (CRP) 2-year follow-up she is thriving well and healthy. was 123 mg/L, urea 9.3 mmol/L and creatinine 80 mmol/L. Initial management consisted of intravenous fluid resuscitation and analgesia. An abdominal ultrasound scan showed a moderate 2. Case 2 amount of free fluid in the pelvis. The and left ovary were not visualized but other organs were normal. Two days after initial A 4-year-old girl was transferred from a district general hospital with a 24 h history of increasing abdominal distension, colicky central , persistent non-bilious vomiting and fever * Corresponding author. Pediatric Urology, Great Ormond’s Street Children’s for 24 h. Hospital, University College London Hospitals NHS Foundation Trust, London, UK. On examination she was dehydrated. The abdomen was Tel.: þ44 7956896641; fax: þ44 1162893395. E-mail addresses: [email protected], [email protected], distended, tender and bowel sounds were decreased. She was [email protected] (R. Patel). resuscitated and nasogastric tube passed.

2213-5766/$ e see front matter Ó 2013 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.epsc.2013.02.009 48 R. Patel et al. / J Ped Surg Case Reports 1 (2013) 47e49

gastropexy with plication of the left hemi-diaphragm. Her post- operative course was complicated by collapse of the right upper and left lower lobes and a left pleural effusion, from which she recov- ered well. She remained well on follow-up at three years.

3. Discussion

Gastric volvulus was first described by Berti in 1866 [3,4].It could have acute, chronic or acute on chronic presentation. There are three types of gastric volvulus described depending on the axis of rotation:organo-axial around the longitudinal axis, mesenterico- axial perpendicular to the longitudinal axis and combined Gastric volvulus can be primary due to abnormalities of the gastric liga- ments or secondary to associated gastric functional dysmotility or diaphragmatic defects such as congenital diaphragmatic , eventration or paralysis of diaphragm. In both of our cases, gastric volvulus was found in conjunction with eventration of the dia- Fig. 1. CT scan showing gastric volvulus with distended stomach with a central twist. phragm. Only a handful of similar cases have been reported in literature [5e7]. In adults, gastric volvulus presents with classic triad of symp- toms of unproductive , localized epigastric distension and Blood tests showed normal electrolytes, urea 12.6 mmol/L, white inability to pass a nasogastric tube. In contrast to this, our patients cell count 18.2 109/L, neutrophils 14.6 109/L and raised amylase presented with productive retching (non-bilious vomiting), of 412 IU. A chest X-ray showed gaseous distension of the stomach abdominal pain or colic, failure to pass nasogastric tube was evident with a right upper lobe collapse. An abdominal X-ray revealed in second case but first case was initially evaluated by pediatric a huge gastric bubble with raised left hemi-diaphragm and minimal nephrology and teams due to chronic and unusual gas in the small bowel (Fig. 2). Abdominal ultrasound showed left lower abdominal colic and raised inflammatory markers and a grossly distended stomach. A barium swallow was performed to amylase leading to CT scan as diagnostic modality. rule out gastric outlet obstruction and demonstrated a grossly Due to raised serum amylase levels, acute pancreatitis was dilated stomach with stasis. An esophagogastroscopy revealed initially suspected in both of our patients. However, pancreatitis a hugely dilated hemorrhagic stomach. was ruled out on further imaging. A raised serum amylase has been At laparotomy, a mesenterico-axial gastric volvulus with punc- reported in conjunction with organ pathology other than acute tate hemorrhagic spots over stomach was seen in conjunction with pancreatitis [8]. The association of a raised amylase with gastric eventration of the left hemi-diaphragm. She underwent posterior volvulus in the absence of pancreatitis has only been reported once previously in adults [2]. The high amylase associated with gastric volvulus may be of salivary origin, and result from absorption of salivary amylase through an ischemic and abnormally permeable gastric mucosa [2]. Alternatively, it may represent an early pancreatitis, resulting from pancreatic duct reflux due to obstruc- tion of the proximal . Acute gastric volvulus is a surgical emergency and requires surgical intervention after appropriate resuscitation. The stomach should be decompressed with nasogastric tube. If this is not possible, gastrostomy may be needed to decompress the stomach as it happened in case 1. Operative treatment includes decompression of the stomach, reduction of the volvulus, gas- tropexy or gastrostomy and correction of underlying predispos- ing cause. There are reports of successful application of laparoscopic techniques for gastric volvulus [9,10].Anantireflux procedure may be required during primary surgery or subse- quently, as these children tend to have gastroesophageal reflux disease. Pediatric gastric volvulus is a serious surgical complication with associated very high morbidity and mortality and a plea has been made for early detection and prevention [11]. The association of raised amylase is mainly secondary to gastric but a case has been reported in which all three lesions of gastric volvulus, and acute pancreatitis coexisted [12].

4. Conclusion

Acute gastric volvulus with associated hyperamylasemia is an unusual emergency and may lead to misdiagnosis. In contrast to the Fig. 2. Plain abdominal supine film with massive distension of stomach and gasless management of acute pancreatitis, the treatment of gastric volvulus distally. is surgical. A delay in diagnosis could be catastrophic and may lead R. Patel et al. / J Ped Surg Case Reports 1 (2013) 47e49 49 to gastric gangrene. A possibility of gastric volvulus should always [5] Park WH, Choi S-O, Suh S-J. Pediatric gastric volvulusdexperience with 7 e be kept in mind while treating a child with acute abdomen and cases. J Korean Med Sc 1992;7:258 63. [6] Manikoth P, Nair P, Zachariah N, Sajwani M. Neonatal acute gastric volvulus. having raised amylase level. Our cases bring to notice a vital issue Arch Dis Child 2004;89:388e9. and powerful reminder of appropriate management of acute [7] Oh A, Gulati G, Sherman ML, Golub R, Kutin N. Bilateral eventration of the abdomen in pediatric age group. diaphragm with perforated gastric volvulus in an adolescent. J Pediatr Surg 2000;35:1824e6. [8] Salt WB, Shenker S. Amylase: its clinical significance: a review of the literature. Medicine 1976;55:269e89. References [9] Darani A, Mendoza-Sagaon M, Reinberg O. Gastric volvulus in children. J Pediatr Surg 2005;40:855e8. [1] Godshall D, Mossallam U, Rosenbaum R. Gastric volvulus: case report and [10] Singal AK, Patel R, Jain S, Gavhane J, Kadam NN. Laparoscopic management of review of the literature. J Emerg Med 1999;17:837e40. neonatal gastric volvulus: a case report and review of the literature. Eur J [2] Williams L, Lansdown MR, Larvin M, Ward DC. Gastric volvulus: a rare cause of Pediatr Surg 2009;19:191e3. hyperamylasaemia. Br J Clin Pract 1990;44:708e9. [11] Gerstle JT, Chiu P, Emil S. Gastric volvulus in children: lessons learned from [3] Miller DL, Pasquale MD, Seneca RP, Hodin E. Gastric volvulus in the pediatric delayed diagnoses. Semin Pediatr Surg 2009;18:98e103. population. Arch Surg 1991;126:1146e9. [12] Oliver MJ, Wilson ARM, Kapila L. Acute pancreatitis and gastric volvulus [4] Cameron AE, Howard ER. Gastric volvulus in childhood. J Pediatr Surg 1987; occurring in a congenital diaphragmatic hernia. J Pediatr Surg 1990;25: 22:944e7. 1240e1.