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　Case Report Kitasato Med J 2015; 45: 90-93　

Received 23 May 2014, accepted 14 January 2015Correspondence to: Eiichiro Watanabe, Department of Surgery, Kitasato University School of Medicine1-15-1 Kitasato, Minami-ku, Sagamihara, Kanagawa 252-0374, JapanE-mail: eiichiryoma@hotmail.com

The first case report of reduced port surgery forpreoperatively diagnosed intestinal malrotation in an adult

Eiichiro Watanabe, Takatoshi Nakamura, Kiyoshi Tanaka, Hirohisa Miura,Atsuko Tsutsui, Noriko Takeda, Masahiko Watanabe

Department of Surgery, Kitasato University School of Medicine

A 31-year-old Japanese man with chronic abdominal pain underwent laparoscopic surgery using botha single port and 2.4-mm needle forceps for a preoperatively diagnosed intestinal malrotation. Abdominalfindings revealed a twisted mesenteric pedicle; it also revealed that the right-side colon was not fusedto the retroperitoneum and that both the transverse colon and the mesocolon were adhered to thesecond portion of the duodenum. After a Ladd procedure following resolution of the volvulus, thesecond portion of the duodenum was fixed to the retroperitoneum with four absorbable sutures in orderto prevent recurrence. The operation was completed laparoscopically without additional ports. Reducedport surgery was recently developed in order to establish not only minimally invasive surgery but alsobetter cosmetic results. Intestinal malrotation in adults is a rare disease and this is the first case reportof reduced port surgery for the management of preoperatively diagnosed intestinal malrotation.

Key words: intestinal malrotation, reduced port surgery, adult

Introduction

ntestinal malrotation is generally diagnosed very earlyin life. Most cases are discovered in the first month of

life, with 90% of all cases presenting within the firstyear.1 The incidences in adults are rare, estimated to beapproximately 0.2% to 1.0% of the population, but a lackof autopsy studies in this area makes the estimation ofthe adulthood prevalence difficult.1-3 In the present report,we describe a case of adult intestinal malrotation withchronic abdominal pain completely treated with reducedport surgery (RPS), in which fewer ports are used than inconventional laparoscopic procedures and in which needleinstruments are also adopted.4 RPS was developed inorder to establish not only minimally invasive surgerybut also better cosmetic results and has been appliedbroadly in various surgical fields.5,6 To our knowledge,this is the first report of RPS adopting both a single portand needle forceps for the management of preoperativelydiagnosed intestinal malrotation.

Case report

A 31-year-old Japanese man with an unremarkablemedical history consulted a doctor at a nearby medical

clinic with a complaint of chronic abdominal pain. Hewas referred to our department for further evaluation ofintestinal malrotation detected on abdominal computedtomography (CT). The CT in our department revealed atwisted mesenteric pedicle and dilatation of the distalsuperior mesenteric vein (Figure 1). Moreover, an uppergastrointestinal barium series showed a corkscrew sign,which was considered a typical image of intestinal

I

Figure 1. Abdominal computed tomography revealed a twistedmesenteric pedicle with dilatation of the distal superior mesentericvein (→).

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malrotation involved with midgut volvulus (Figure 2).The patient initially chose to forego surgical interventionbecause his pain had decreased and because of timeconstraints, and remained under observation at theoutpatient clinic with appropriate informed consent.However, over a period of 4 years, his abdominal paingradually increased, which made him decide to undergoan operation.

The operation was performed with the SILS port(Covidien, Mansfield, USA) in the umbilicus and a 2.4-mm Endo Relief needle forceps (Hope Denshi, Chiba)placed just above the pubic bone. Abdominal findingsrevealed an incomplete-rotation type of intestinalmalrotation involved with the midgut volvulus, that theright-side colon was not fused to the retroperitoneum,and that both the transverse colon and the mesocolon

Table 1. Intestinal development and various types of anomalies due to its interruption

EmbryonicStage stage Event Anomalies

(week)

・90° counterclockwise rotation of the midgutI 5−10 Omphalocele

・The midgut replacent into the abdominal cavity

・180° counterclockwise rotation of the midgut inside Malrotation (rotation)　the abdominal cavity ・Non-rotation (<90°)

II 11 ・The duodenal C loop and the small bowel become ・Incomplete rotation　attached to the posterior abdomen 　(90°−180°)・Establishment of the normal anatomy of the colon ・Reverse-rotation

・The cecum descendsIII 11− ・Both the ascending and descending colon attach to the Mobile cecum, etc.

　posterior abdomen

Figure 4. After the Ladd procedure, the second portion of theduodenum was fixed to the retroperitoneum with four absorbablesutures (　) in order to prevent recurrence.▲

Figure 3. Dilatation of the distal superior mesenteric vein (○)and the small intestine (＊) due to a twisted mesenteric pediclewere observed. The right side of the colon was not attached to theretroperitoneum.

Figure 2. Counterclockwise rotation of the mesentericpedicle (　: the corkscrew sign)

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▲

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Watanabe, et al.

were adhered to the second portion of the duodenum(Figure 3). A twisted mesenteric pedicle was resolvedmanually by pulling the small intestine distally from theileocecal to the proximal side using laparoscopic surgicalforceps. After the Ladd procedure (in whichappendectomy was not performed), the second portionof the duodenum was fixed to the retroperitoneum withfour absorbable sutures (Figure 4). The operation wascompleted laparoscopically without additional ports.

The patient was discharged on the seventh post-operative day without any complications. No abdominaldiscomfort has been reported in the 2 years since surgicalintervention.

Discussion

An understanding of intestinal development is essentialto the surgical management of intestinal malrotation,because the symptoms are often deceptive, the findingsat operation are confusing, and various types may beencountered. Healthy embryonic intestinal rotation ofthe midgut involves three stages. Stage I occurs betweenthe fifth and tenth gestational weeks. It is a 90°counterclockwise rotation of the midgut and itsreplacement into the abdominal cavity. Stage II occursat the eleventh gestational week and involves a 180°counterclockwise rotation inside the abdominal cavity,so that the duodenal C loop and the small bowel becomeattached to the posterior abdominal wall via its mesentery,following the establishment of the normal anatomy ofthe colon. In stage III, the cecum descends, and both theascending and descending colon attach to the posteriorabdomen.7-9 Interruption of any stage results in varioustypes of intestinal malrotation. Stage I anomalies includeomphalocele caused by failure of the gut to return to theabdomen. Stage II anomalies include non-rotation,incomplete rotation, and the reversed-rotation type ofintestinal malrotation. Stage III anomalies include anunattached duodenum, a mobile cecum, and an unattachedsmall bowel mesentery (Table 1).8

Typical symptoms in neonates with intestinalmalrotation are abdominal distension and biliousvomiting, whereas in adults, symptoms are mostly oneof three types: acute obstructive symptoms and signs ofimpending abdominal catastrophe, chronic abdominalcomplaints that include both pain and intermittentobstruction, and atypical symptoms of commonabdominal diseases.1,7,10 Therefore, both the definitivepreoperative diagnosis of intestinal malrotation and thedecision to operate in adult patients pose challenges dueto the variety of symptoms.

The widely accepted treatment for intestinalmalrotation involved with midgut volvulus is the Laddprocedure, which includes lysis of all abdominal bandsand adhesions, straightening of the duodenum such thatit descends directly into the right lower quadrant,widening of the small bowel mesentery by tearing itsserosal leaves, and appendectomy after replacement ofthe volvulus.11 In addition, in order to prevent laterrecurrence, additional procedures, such as fixation of theintestine to the peritoneal wall, are performed in somecases.1,7,12-14

For surgeons with significant experience in minimallyinvasive surgery, both diagnostic laparoscopy anddefinitive laparoscopic repair are viable options for thetreatment of patients with chronic symptoms of intestinalmalrotation.15,16 In addition, RPS, which was introducedas a mixed use of surgical techniques, such as single portsurgery (SPS) and needlescopic surgery, has recentlybecome popular in various surgical fields and adoptedfor various diseases.4-6,17 To date, there has been aninsufficient number of RPS cases to support the safety ofRPS, and neither RPS nor SPS is recommended as a safeand reliable option in the Japanese Society for Cancer ofthe Colon and Rectum guidelines.18 However, RPS wasdeveloped to establish not only minimally invasivesurgery but also better cosmetic results, and surgical costsare also decreased because of the use of reusable needleforceps. Therefore, RPS may become the first surgicaloption for various diseases, including intestinalmalrotation, if it is feasible.

On the other hand, RPS for intestinal malrotation hassome disadvantages. First, resolution of the volvulusbecomes more difficult, because RPS requires moresophisticated skills than standard laparoscopic surgerydue to a limited working space and the smaller numberof forceps. Second, less adhesion after RPS leads torecurrence more often than with conventional techniques,including standard laparoscopic surgery; however, to itsadvantage, it also leads to a smaller ratio of adhesiveintestinal obstruction. Third, laparoscopic surgery,including RPS, is not feasible for urgent situations suchas strangulated ileus.

In our experience, both the Ladd procedure andfixation of the duodenum to the retroperitoneum couldbe performed safely with RPS in an adult intestinalmalrotation in which the clinical condition was not severe.The twisted mesenteric pedicle was resolved naturallyby the laparoscopic method previously described (ShoheiT, et al. first introduced this technique at the 44th annualmeeting of the Japanese Society of Pediatric Surgeons inTokyo), in which the volvulus was resolved manually by

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pulling the small intestine distally from the distal side ofthe intestine with laparoscopic forceps such that rotationof the midgut volvulus was more than 360°. In addition,for the purpose of preventing recurrence, the secondportion of the duodenum could be fixed to theretroperitoneum with four absorbable sutures.

To our knowledge, this is the first report adoptingboth a single port and needle forceps for the managementof intestinal malrotation. RPS for the management ofintestinal malrotation in adults is a safe and reliableprocedure if it can be performed under good visualizationand if surgeons have definite understanding of its uniquecharacteristics. More RPS cases are warranted todemonstrate its safety, good cosmetics, and minimalinvasiveness, in order for it to be established as a radicalsurgery.

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Reduced port surgery for intestinal malrotation