Chestnut-phytobezoar causing small bowel obstruction in a child with Dravet syndrome

Chestnut-phytobezoar causing small bowel obstruction in a child with Dravet syndrome

Journal of Pediatric Surgery Case Reports 45 (2019) 101209 Contents lists available at ScienceDirect Journal of Pediatric Surgery Case Reports journ...

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Journal of Pediatric Surgery Case Reports 45 (2019) 101209

Contents lists available at ScienceDirect

Journal of Pediatric Surgery Case Reports journal homepage: www.elsevier.com/locate/epsc

Chestnut-phytobezoar causing small bowel obstruction in a child with Dravet syndrome

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Hanna Schmidta, Tobias Wowraa, Benedikt Wintera, Alexandre Serrab,∗ a b

Department of Pediatric and Adolescent Medicine, Ulm University, Eythstraße 24, 89075, Ulm, Germany Division of Pediatric Surgery, Department of Surgery, Ulm University, Eythstraße 24, 89075, Ulm, Germany

A R T I C LE I N FO

A B S T R A C T

Keywords: Phytobezoar Dravet syndrome Mechanical ileus Chestnut

An 11-year old girl with Dravet syndrome presented with recurrent vomiting and abdominal tenderness and distension. Subsequent investigations including ultrasound and gastrointestinal fluoroscopy series showed signs of small bowel obstruction within the right hypogastric abdominal region. Laparotomy revealed three chestnutphytobezoars in the distal jejunum as cause of intestinal obstruction. This is the first reported case about a mechanical ileus related to a chestnut-phytobezoars in a pediatric patient.

Mechanical ileus due to phytobezoars has been described in adult patients and several risk factors predisposing to this condition have been identified. Considering this condition and prompt diagnosis is crucial for preventing potential lethal complications. This report presents an extremely rare case of phytobezoar and subsequent mechanical ileus in a pediatric patient with cognitive impairment, lacking most of the so far described risk factor in the adult population. With this case report, we aim to raise awareness for possible phytobezoar formation subsequent to foreign body ingestion in pediatric patients with underlying mental disorders and syndromes with cognitive impairment presenting with unclear gastrointestinal symptoms. 1. Case report 1.1. Initial presentation An 11-year old girl with Dravet syndrome was seen in our emergency department with a two-day history of recurrent vomiting. The parents also reported that the day before vomiting had started the patient had watery stools, refused oral food and liquid intake and progressively reduced her daily activities. Also, two days prior to admission the patient had a seizure due to the refusal of anti-epileptic medication. This patient was diagnosed early in life with Dravet syndrome. Therein an SCN1A mutation in a gene encoding for a voltage-gated type I sodium channel is responsible. Variants of Dravet syndrome can result in severe developmental retardation, behavioral disorder with hyperkinetic and autistic traits and refractory epilepsy. The current anti-



epileptic therapy consisted of valproate, bromide, vagus nerve stimulation and sedative medication with risperidone and melatonin. There was no history of prior abdominal surgery. Upon admission, the patient was stable but in reduced general condition, vital signs were normal. The physical examination showed clinical signs of dehydration, painful abdominal distension and slightly high-pitched bowel sounds. 1.2. Investigations Laboratory work-up consisted of blood tests including full blood count, liver and kidney function tests, electrolytes and c-reactive protein (CrP), which were all within normal ranges except slightly raised hemoglobin (Hb, 16.5 g/dl) and urea (8.4 mmol/) levels, consistent with dehydration. Accordingly, urine probes also revealed a mild ketonuria. Imaging diagnostic initially consisted of an ultrasound examination of the abdomen (Philips iu22 with linear probe L9-3, Philips Healthcare, Bothell, USA) which revealed non-propulsive peristalsis and a keybord sign, as well as dilated loops of small intestine suggesting with small bowel obstruction. In the right lower quadrant a thin but well circumscribed hyperechoic structure of 2,4 cm in diameter with a complete dorsal acoustic shadow suggested a foreign body proximal to the ileocecal valve (Fig. 1). Additionally, there was a large amount of ascites. The appendix and parenchymatous organs of the upper abdomen were unremarkable. Further imaging included a plain abdominal X-ray, which showed distended loops of small bowel and several air-fluid-levels projecting to

Corresponding author. E-mail address: [email protected] (A. Serra).

https://doi.org/10.1016/j.epsc.2019.101209 Received 28 March 2019; Accepted 7 April 2019 Available online 08 April 2019 2213-5766/ © 2019 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/BY-NC-ND/4.0/).

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Fig. 1. Ultrasound imaging shows hyperechoic structure of 2,4 cm in diameter with dorsal acoustic shadow in the right lower abdomen.

Fig. 3. Upper gastrointestinal fluoroscopy series: Contrast agent terminates in the right lower abdomen, indicating obstruction of intestinal passage.

Approximately at the ileal junction there was a complete obstruction of the intestinal lumen due to three hard objects that could not be moved further distally into the empty, normal ileum. A small Jejunotomy (3,5 cm) revealed three chestnut Phytobezoars, which were completely removed (Fig. 4). The chestnuts were not adherent to the wall but impacted in the lumen of the small intestine. Further inspection showed no signs of vascular injury and the jejunotomy was closed in 2 layers with PDS sutures perpendicular to the incision. There was no residual stricture of the small intestine after closure. The post-operative clinical course was normal until the 4th PO day, when the patient developed acutely signs of intestinal obstruction associated with sepsis symptoms and signs of intraabdominal abscess in ultrasound. Relaparotomy revealed intraabdominal abscess causing a partial dehiscence of the jejunotomy as well as a perforation of 1 × 1 cm. Due to malperfusion of the jejunal wall, resection of a 15 cm segment had to be performed. After exhaustive irrigation of the abdominal cavity both intra- and postoperatively there was no recurrence of the abscess. The anastomosis and surgical wounds healed primarily under comprehensive antibiotic, antimycotic and parenteral nutrition therapy. The patient was discharged after 19 days since the second surgery with full oral feedings and in good clinical conditions.

Fig. 2. Plain abdominal x-ray shows air-fluid levels.

the epigastric and right hypogastric abdomen. No free abdominal air was detected (Fig. 2). To further investigate the intestinal obstruction, a gastrointestinal fluoroscopy series with contrast agent was carried out. Retrograde filling of the colon showed contrast up to the ileocecal valve. The upper abdominal series showed an abrupt termination of the small bowel filling in the right hypogastric abdominal region (Fig. 3).

2. Discussion We report for the first time the case of a chestnut phytobezoar with

1.3. Treatment and course The patient was managed conservatively with intravenous fluid replacement and laxative measures due to the putative diagnosis of severe constipation. Symptoms were further deteriorating under conservative therapy and the child developed acute signs of a high intestinal obstruction, namely projectile bilious vomiting progressing to fecal vomiting, abdominal distension and high-pitched peristalsis. The patient was taken to the operating theater and primarily stabilized by the anesthesia team, including central venous and arterial accesses and extended fluid therapy, after which the surgical therapy could be initiated. This consisted in a midline infra-umbilical laparotomy with immediate drainage of 500 ml of serous ascites. The initial 1/3 of the jejunum was extremely dilated with gas and liquid stools.

Fig. 4. Two Chestnut-Phytobezoars retrieved from the terminal Jejunum through an enterotomy. 2

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control their access to indigestible parts of plants.

mechanical ileus in a child. Bezoars are trapped indigestible masses in the gastrointestinal system and are classified according to their material of origin. Phytobezoars are the most common type consisting of indigested plant constituents as fibers or indigested vegetables [1]. Less common are trichobezoars (ingested hair), lactobezoars (milk derivatives) or pharmacobezoars (medication vehicles), [1,2,13]. Several case reports have demonstrated phytobezoars as an infrequent cause of mechanical intestinal obstruction [3–5]. Of these, a small bowel obstruction following ingestion of chestnuts has been reported in a 63-year old woman with mental retardation [6]. In adult patients, previous abdominal surgery, high fiber diet and systemic diseases associated with impaired intestinal motility have been described as crucial predisposing conditions for phytobezoar formation and subsequent intestinal obstruction [1,7]. Moreover, mental impairment and psychiatric co-morbidity have been associated with phytobezoars [2,6]. Among children, an intestinal obstruction due to phytobezoars is extremely rare and rarely reported, with the exception of a phytobezoar formation in Meckel's diverticulum [8,9]. Ingestion of foreign bodies is a common phenomenon in pediatric populations, though it is predominantly suspected in toddlers [10]. In older children, foreign body ingestion is rare, but should be considered in patients with psychiatric co-morbidity or mental retardation. Mental retardation and behavioral disorder are common features of Dravet syndrome, however, complications due to ingestion of indigestible material have never been described in these patients. Imaging techniques for investigation of intestinal obstruction may include ultrasound, plain abdominal X-ray and CT-scans. According to a study of Ripollès et al., abdominal CT is the most sensitive imaging modality, detecting up to a 100% of bezoars in a series of 17 patients, whereas ultrasound images were suggestive of a bezoar causing bowel obstruction in only 88% [12]. However, because of the intense radiation exposure, ultrasound remains commonly the primary imaging investigation tool in pediatric patients. In our case, ultrasound was highly suggestive of small bowel obstruction and the presence of a foreign body within the right hypogastric region, which was further confirmed by abdominal fluoroscopy and eventually by laparotomy. The clinical presentation of mechanical intestinal obstruction due to phytobezoars does not differ from other causes of mechanical ileus, therefore rendering the differential diagnosis difficult. One study reported that in 76% of patients with small bowel obstruction due to phytobezoars, the diagnosis was only ascertained at the time of laparotomy [11]. In conclusion, this short case-report study serves to alert the community to severe and potential fatal complications of a phytobezoar in children suffering from mental retardation.

Patient consent Signed informed consent was obtained from the patients' parents. Funding This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. Authorship All authors attest that they meet the current ICMJE criteria for authorship. Conflict of interest The following authors have no financial disclosures: HS, TW, BW, AS. Acknowledgements We would like to express our thanks to the Department of Radiology of the Ulm University for providing the imaging material. Appendix A. Supplementary data Supplementary data to this article can be found online at https:// doi.org/10.1016/j.epsc.2019.101209. References [1] Dikicier E, Altintoprak A, Ozkan OV, Yagmurkaya o UMY. Intestinal obstruction due to phytobezoars: an update. World J Clin Cases 2015;3(8):721–6. [2] Iwamuro M, Okada H, Matsueda K, Inaba T, Kusumoto C, Imagawa A. Review of the diagnosis and management of gastrointestinal bezoars. World J Gastrointest Endosc 2015;7(4):336–45. [3] Ezzat RF, Rashid SA, Rashid AT, Abdullah KM, Ahmed SM. Small intestinal obstruction due to phytobezoar: a case report. J Med Case Rep 2009 Dec 2;3:9312. [4] Serrano KD1, Tupesis JP. Small bowel obstruction from potato and broccoli phytobezoar mimicking mesenteric ischemia. J Emerg Med 2013 Jan;44(1):79–81. [5] Kia M1, Aghili SM2, Aghili R3. Intestinal obstruction caused by phytobezoars. West J Emerg Med 2014 Jul;15(4):385–6. [6] Satake R, Chinda D, Shimoyama T, Satake M, Oota R. Repeated small bowel obstruction caused by chestnut ingestion without the formation of phytobezoars. Intern Med 2016 und;55(12):1565–8. [7] Bedioui H, Daghfous A, Ayadi M, Noomen R, Chebbi F, Rebai W, Makni A, Fteriche F, Ksantini R, Ammous A, Jouini M, Kacem M, Bensafta Z. A report of 15 cases of small-bowel obstruction secondary to phytobezoars: predisposing factors and diagnostic difficult. 2008. [8] Tashjian DB1, Moriarty KP. Laparoscopy for treating a small bowel obstruction due to a Meckel's diverticulum. JSLS 2003;7(3):253–5. [9] Duman L, Savas C, Ceyhan L. An unusual cause of intestinal obstruction in an infant: phytobezoar within a Meckel diverticulum. J Pediatr Surg 2011 Aug;46(8):1678–9. [10] Diaconescu S, Gimiga N, Sarbu I, Stefanescu G, Olaru C, Ioniuc I, Ciongradi I, Burlea M. Foreign bodies ingestion in children: experience of 61 cases in a pediatric gastroenterology unit from Romania. Gastroenterol Res Pract 2016;2016:1982567. [11] Ho TW1, Koh DC. Small-bowel obstruction secondary to bezoar impaction: a diagnostic dilemma. World J Surg 2007 May;31(5):1072–8. [12] Ripollés T, García-Aguayo J, Martínez MJ, Gil P. Gastrointestinal bezoars: sonographic and CT characteristics. AJR Am J Roentgenol 2001 Jul;177(1):65–9. [13] Dikicier E, Altintoprak A, Ozkan OV, Yagmurkaya oUMY. Intestinal obstruction due to phytobezoars: an update. World J Clin Cases 2015;3(8):721–6.

3. Conclusions Phytobezoar formation subsequent to foreign body ingestion should also be suspected in pediatric patients apart from the toddlers' age, presenting with underlying mental disorders and syndromes with cognitive impairment presenting with unclear gastrointestinal symptoms. Ultrasound can be highly suggestive of intestinal obstruction by a foreign body and should be the imaging method of choice in children of any age for this purpose. Parents and caretakers of patients at risk are advised to strictly

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