Case Report Malrotation with midgut volvulus associated with perforated ileal duplication Anand Pandey, Shailendra Pal Singh, Vipin Gupta, Jigyasa Pandey1, Pradeep Sachan Departments of Surgery, Uttar Pradesh Rural Institute of Medical Sciences and Research, 1Department of Radiodiagnosis, Uttar Pradesh Rural Institute of Medical Sciences and Research, Saifai, Etawah, Uttar Pradesh, India Address for correspondence: Dr. Anand Pandey, Department of Surgery, Uttar Pradesh Rural Institute of Medical Sciences and Research, Saifai, Etawah - 206 301, Uttar Pradesh, India. E-mail: [email protected]
ABSTRACT Duplication of the alimentary tract is an important surgical condition. It may occur anywhere in the gastrointestinal tract. An important complication of this entity is perforation of the normal or abnormal gut. Malrotation with midgut volvulus can be a surgical emergency. We present a patient, who presented as malrotation with midgut volvulus associated with perforated ileal duplication. The patient was successfully managed.
Access this article online Website: www.jiaps.com DOI: 10.4103/0971-9261.121123 Quick Response Code:
KEY WORDS: Alimentary tract duplication, malrotation, midgut volvulus, malrotation and duplication
INTRODUCTION Duplication of the alimentary tract is an important surgical condition, which includes a wide variety of mass lesions. [1] It may occur anywhere in the gastrointestinal tract. An important complication of this entity is perforation of the normal or abnormal gut. We encountered a patient who presented with perforation of the duplication, which was associated with malrotation with midgut volvulus. Being an extremely uncommon surgical emergency, it is being presented with a brief review of the relevant literature.
CASE REPORT A 4-year-old male child was referred to us for complaint of pain in abdomen. On enquiry, there was intermittent history of bilious vomiting since infancy. There was no previous history as regard to abdominal distension. However, there
was occasional history of abdominal pain, which was relieved by medication. On examination, his abdomen was distended, but it was non-tender. The weight of the patient was 9 kg. Thus he had failed to thrive. His ultrasound (USG) of abdomen suggested some fluid filled cystic spaces. A computerized tomography (CT) scan of abdomen was advised for confirmation of the diagnosis. On next day, just after CT abdomen, the patient suddenly developed severe abdominal pain. X-ray abdomen showed pneumoperitoneum. Clinically the patient looked septic. His pulse rate was 130/min with a feeble pulse. An urgent laparotomy was planned. After initial stabilization, the patient was explored under general anesthesia. On exploration, we noticed malrotation of the gut, which was associated with two turns of midgut volvulus. The midgut did not have any evidence of ischemia or gangrene. It was complete malrotation having cecum in left hypochondrium and narrow mesentery. Ladd’s bands were also present. The peritoneal cavity was grossly contaminated with fecal
Cite this article as: Pandey A, Singh SP, Gupta V, Pandey J, Sachan P. Malrotation with midgut volvulus associated with perforated ileal duplication. J Indian Assoc Pediatr Surg 2013;18:155-7. Source of Support: Nil, Conflict of Interest: None declared.
Journal of Indian Association of Pediatric Surgeons / Oct-Dec 2013 / Vol 18 / Issue 4
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Pandey, et al.: Malrotation with perforated duplication
fluid. The perforation site was about one foot proximal to ileocecal region. The perforation was at the mesenteric side, and it was associated with a soft mesenteric mass communicating with the normal bowel. The mass was about 10 cm in length. The perforation was just at the junction of the mesenteric mass and the normal bowel [Figure 1]. After performing the Ladd’s procedure, attention was placed towards the perforation. On attempting to resect the mesenteric mass, we noticed two lumens in the bowel distal to the perforation site and single lumen proximal to that [Figures 2 and 3]. The duplication appeared to be having all the layers of the bowel wall. When an attempt to ascertain the length of the duplication was made by inserting two Ryle’s tube through the lumens, it was reaching up to ileocecal region. As the general condition of the patient was poor, complete resection of the bowel was not done, and a double barrel (triple barrel, to be more precise) was fashioned.
Figure 1: Intraoperative photo showing perforated duplication. Mosquito forceps and one thumb forceps are in the distal duplicated lumens. The other thumb forceps is in the perforated duplication
In the postoperative period, there was wound infection with superficial wound dehiscence. It was managed conservatively. The patient was discharged after 3 weeks with proper follow-up advise. During the follow-up, we had closed the ileostomy. As the duplication was reaching up to cecum, we had performed ileoascending anastomosis. The patient is fine. Figure 2: Intraoperative photo showing duplicated lumen
DISCUSSION Congenital anomalies of the gastrointestinal tract are a significant cause of morbidity in children. These abnormalities include developmental obstructive defects of the small intestine, anomalies of rotation and fixation, intestinal duplications, etc.[2] Duplications of the alimentary tract are rare congenital malformations and may occur anywhere in the intestinal tract. Intestinal duplication cyst has been rarely found with intestinal malrotation.[1,3] In our patient, the history of bilious vomiting was suspicious of small bowel anomaly. The initial suspicion, on the basis of USG, was of some cystic mass lesion compressing the upper small bowel. The exact diagnosis, however, was confirmed only at laparotomy. The CT plates, reviewed later on, also showed twisting of mesenteric vessels suggestive of malrotation. The exact cause of perforation was not clear. Probably the perforation of the duplication was not spontaneous; it may be because of the volvulus and resultant ischemia of the wall. As malrotation with midgut volvulus is always a surgical emergency, and perforated duplication is also a surgical emergency, a combination of the two entities is definitely a surgical emergency, which needs 156
Figure 3: Line diagram showing duplication and normal bowel. (a) Duplication cyst, (b) junction of duplication and normal bowel, and it was the site of perforation, (c) normal ileum, (d) duplicated ileum reaching up to cecum, and (e) cecum and probable site of duplication termination
immediate intervention. The ideal treatment would have been a Ladd’s procedure, along with resection of the complete duplication. This was, however, not possible due to the poor general condition of the patient and large length of the duplication, which may had led to extensive resection of the bowel. Hence, ileostomy was made.
Journal of Indian Association of Pediatric Surgeons / Oct-Dec 2013 / Vol 18 / Issue 4
Pandey, et al.: Malrotation with perforated duplication
The association of duplication of small bowel and malrotation, though described in the literature, is a rare one. Our English literature review revealed less than 25 patients.[2-8] None of the patients had a presentation of acute abdomen due to perforated duplication. To conclude, this being one of a very rare combination of two pediatric surgical emergencies; needs proper surgical and postoperative management. The management needs to be tailored as per the condition of the patient. The exact preoperative diagnosis may be difficult, but a high degree of suspicion is warranted.
REFERENCES 1. Lund DP. Alimentary tract duplication. In: Coran AG, Krummel TM, Laberge J, Shamberger RC, Caldamone AA, editors. Pediatric Suregry, 7th ed. Philadelphia, PA: Elsevier Saunders; 2012. p. 1155-64.
2. Berrocal T, Lamas M, Gutieerrez J, Torres I, Prieto C, del Hoyo ML. Congenital anomalies of the small intestine, colon, and rectum. Radiographics 1999;19:1219-36. 3. Somuncu S, Cakmak M, Caglayan E, Unal B. Intestinal duplication cyst associated with intestinal malrotation anomaly: Report of a case. Acta Chir Belg 2006;106:611-2. 4. Choi SO, Park WH, Kim SP. Enteric duplications in children — an analysis of 6 cases. J Korean Med Sci 1993;8:482-7. 5. Balen EM, Hernandez-Lizoain JL, Pardo F, Longo JM, Cienfuegos JA, Alzina V. Giant jejunoileal duplication: Prenatal diagnosis and complete excision without intestinal resection. J Pediatr Surg 1993;28:1586-8. 6. Menon P, Rao KL, Vaiphei K. Isolated enteric duplication cysts. J Pediatr Surg 2004;39:e5-7. 7. Koh CC, Wang NL, Lee HC, Duh YC. Infected congenital splenic cyst associated with duodenal duplication cyst and malrotationA case report. J Pediatr Surg 2007;42:e21-2. 8. Olajide AR, Yisau AA, Abdulraseed NA, Kashim IO, Olaniyi AJ, Morohunfade AO. Gastrointestinal duplications: Experience in seven children and a review of the literature. Saudi J Gastroenterol 2010;16:105-9.
Journal of Indian Association of Pediatric Surgeons / Oct-Dec 2013 / Vol 18 / Issue 4
157
Copyright of Journal of Indian Association of Pediatric Surgeons is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
ABSTRACT Duplication of the alimentary tract is an important surgical condition. It may occur anywhere in the gastrointestinal tract. An important complication of this entity is perforation of the normal or abnormal gut. Malrotation with midgut volvulus can be a surgical emergency. We present a patient, who presented as malrotation with midgut volvulus associated with perforated ileal duplication. The patient was successfully managed.
Access this article online Website: www.jiaps.com DOI: 10.4103/0971-9261.121123 Quick Response Code:
KEY WORDS: Alimentary tract duplication, malrotation, midgut volvulus, malrotation and duplication
INTRODUCTION Duplication of the alimentary tract is an important surgical condition, which includes a wide variety of mass lesions. [1] It may occur anywhere in the gastrointestinal tract. An important complication of this entity is perforation of the normal or abnormal gut. We encountered a patient who presented with perforation of the duplication, which was associated with malrotation with midgut volvulus. Being an extremely uncommon surgical emergency, it is being presented with a brief review of the relevant literature.
CASE REPORT A 4-year-old male child was referred to us for complaint of pain in abdomen. On enquiry, there was intermittent history of bilious vomiting since infancy. There was no previous history as regard to abdominal distension. However, there
was occasional history of abdominal pain, which was relieved by medication. On examination, his abdomen was distended, but it was non-tender. The weight of the patient was 9 kg. Thus he had failed to thrive. His ultrasound (USG) of abdomen suggested some fluid filled cystic spaces. A computerized tomography (CT) scan of abdomen was advised for confirmation of the diagnosis. On next day, just after CT abdomen, the patient suddenly developed severe abdominal pain. X-ray abdomen showed pneumoperitoneum. Clinically the patient looked septic. His pulse rate was 130/min with a feeble pulse. An urgent laparotomy was planned. After initial stabilization, the patient was explored under general anesthesia. On exploration, we noticed malrotation of the gut, which was associated with two turns of midgut volvulus. The midgut did not have any evidence of ischemia or gangrene. It was complete malrotation having cecum in left hypochondrium and narrow mesentery. Ladd’s bands were also present. The peritoneal cavity was grossly contaminated with fecal
Cite this article as: Pandey A, Singh SP, Gupta V, Pandey J, Sachan P. Malrotation with midgut volvulus associated with perforated ileal duplication. J Indian Assoc Pediatr Surg 2013;18:155-7. Source of Support: Nil, Conflict of Interest: None declared.
Journal of Indian Association of Pediatric Surgeons / Oct-Dec 2013 / Vol 18 / Issue 4
155
Pandey, et al.: Malrotation with perforated duplication
fluid. The perforation site was about one foot proximal to ileocecal region. The perforation was at the mesenteric side, and it was associated with a soft mesenteric mass communicating with the normal bowel. The mass was about 10 cm in length. The perforation was just at the junction of the mesenteric mass and the normal bowel [Figure 1]. After performing the Ladd’s procedure, attention was placed towards the perforation. On attempting to resect the mesenteric mass, we noticed two lumens in the bowel distal to the perforation site and single lumen proximal to that [Figures 2 and 3]. The duplication appeared to be having all the layers of the bowel wall. When an attempt to ascertain the length of the duplication was made by inserting two Ryle’s tube through the lumens, it was reaching up to ileocecal region. As the general condition of the patient was poor, complete resection of the bowel was not done, and a double barrel (triple barrel, to be more precise) was fashioned.
Figure 1: Intraoperative photo showing perforated duplication. Mosquito forceps and one thumb forceps are in the distal duplicated lumens. The other thumb forceps is in the perforated duplication
In the postoperative period, there was wound infection with superficial wound dehiscence. It was managed conservatively. The patient was discharged after 3 weeks with proper follow-up advise. During the follow-up, we had closed the ileostomy. As the duplication was reaching up to cecum, we had performed ileoascending anastomosis. The patient is fine. Figure 2: Intraoperative photo showing duplicated lumen
DISCUSSION Congenital anomalies of the gastrointestinal tract are a significant cause of morbidity in children. These abnormalities include developmental obstructive defects of the small intestine, anomalies of rotation and fixation, intestinal duplications, etc.[2] Duplications of the alimentary tract are rare congenital malformations and may occur anywhere in the intestinal tract. Intestinal duplication cyst has been rarely found with intestinal malrotation.[1,3] In our patient, the history of bilious vomiting was suspicious of small bowel anomaly. The initial suspicion, on the basis of USG, was of some cystic mass lesion compressing the upper small bowel. The exact diagnosis, however, was confirmed only at laparotomy. The CT plates, reviewed later on, also showed twisting of mesenteric vessels suggestive of malrotation. The exact cause of perforation was not clear. Probably the perforation of the duplication was not spontaneous; it may be because of the volvulus and resultant ischemia of the wall. As malrotation with midgut volvulus is always a surgical emergency, and perforated duplication is also a surgical emergency, a combination of the two entities is definitely a surgical emergency, which needs 156
Figure 3: Line diagram showing duplication and normal bowel. (a) Duplication cyst, (b) junction of duplication and normal bowel, and it was the site of perforation, (c) normal ileum, (d) duplicated ileum reaching up to cecum, and (e) cecum and probable site of duplication termination
immediate intervention. The ideal treatment would have been a Ladd’s procedure, along with resection of the complete duplication. This was, however, not possible due to the poor general condition of the patient and large length of the duplication, which may had led to extensive resection of the bowel. Hence, ileostomy was made.
Journal of Indian Association of Pediatric Surgeons / Oct-Dec 2013 / Vol 18 / Issue 4
Pandey, et al.: Malrotation with perforated duplication
The association of duplication of small bowel and malrotation, though described in the literature, is a rare one. Our English literature review revealed less than 25 patients.[2-8] None of the patients had a presentation of acute abdomen due to perforated duplication. To conclude, this being one of a very rare combination of two pediatric surgical emergencies; needs proper surgical and postoperative management. The management needs to be tailored as per the condition of the patient. The exact preoperative diagnosis may be difficult, but a high degree of suspicion is warranted.
REFERENCES 1. Lund DP. Alimentary tract duplication. In: Coran AG, Krummel TM, Laberge J, Shamberger RC, Caldamone AA, editors. Pediatric Suregry, 7th ed. Philadelphia, PA: Elsevier Saunders; 2012. p. 1155-64.
2. Berrocal T, Lamas M, Gutieerrez J, Torres I, Prieto C, del Hoyo ML. Congenital anomalies of the small intestine, colon, and rectum. Radiographics 1999;19:1219-36. 3. Somuncu S, Cakmak M, Caglayan E, Unal B. Intestinal duplication cyst associated with intestinal malrotation anomaly: Report of a case. Acta Chir Belg 2006;106:611-2. 4. Choi SO, Park WH, Kim SP. Enteric duplications in children — an analysis of 6 cases. J Korean Med Sci 1993;8:482-7. 5. Balen EM, Hernandez-Lizoain JL, Pardo F, Longo JM, Cienfuegos JA, Alzina V. Giant jejunoileal duplication: Prenatal diagnosis and complete excision without intestinal resection. J Pediatr Surg 1993;28:1586-8. 6. Menon P, Rao KL, Vaiphei K. Isolated enteric duplication cysts. J Pediatr Surg 2004;39:e5-7. 7. Koh CC, Wang NL, Lee HC, Duh YC. Infected congenital splenic cyst associated with duodenal duplication cyst and malrotationA case report. J Pediatr Surg 2007;42:e21-2. 8. Olajide AR, Yisau AA, Abdulraseed NA, Kashim IO, Olaniyi AJ, Morohunfade AO. Gastrointestinal duplications: Experience in seven children and a review of the literature. Saudi J Gastroenterol 2010;16:105-9.
Journal of Indian Association of Pediatric Surgeons / Oct-Dec 2013 / Vol 18 / Issue 4
157
Copyright of Journal of Indian Association of Pediatric Surgeons is the property of Medknow Publications & Media Pvt. Ltd. and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
