Laparoscopic cholecystectomy in situs inversus totalis: Feasibility and review of literature

Abstract

INTRODUCTION

Situs inversus totalis is a rare anomaly characterized by transposition of organs to the opposite site of the body. Laparoscopic cholecystectomy in those patients is technically more demanding and needs reorientation of visual-motor skills to left upper quadrant.

PRESENTATION OF CASE

Herein, we report a 10 year old boy presented with left hypochondrium and epigastric pain 2 months duration. The patient had not been diagnosed as situs inversus totalis before. The patient exhibit a left sided “Murphy's sign”. Diagnosis of situs inversus totalis was confirmed with ultrasound, computerized tomography (CT) and magnetic resonant image (MRI) with presence of multiple gall bladder stones with no intra or extrabiliary duct dilatation. The patient underwent laparoscopic cholecystectomy for cholelithiasis.

DISCUSSION

Feasibility and technical difficulty in diagnosis and treatment of such case pose challenge problem due to the contra lateral disposition of the viscera. Difficulty is encountered in skelatonizing the structures in Calot's triangle, which consume extra time than normally located gall bladder. A summary of additional 50 similar cases reported up to date in the medical literature is also presented.

CONCLUSION

Laparoscopic cholecystectomy is feasible and should be done in situs inversus totalis by experienced laparoscopic surgeon, as changes in anatomical disposition of organ not only influence the localization of symptoms and signs arising from a diseased organ but also imposes special demands on the diagnosis and surgical skills of the surgeon.

1. Introduction

Situs inversus totalis (SIT) is a very uncommon entity. It was first reported by Fabricius in 1600.¹ The incidence is thought to be in the range of 1:10 000 to 1:20 000.²

Transposition of the organs may also affect thoracic organs, besides abdominal organs. It can be associated with Kartegener triad (bronchietasis, sinusitis, and situs inversus) and cardiac anomalies. There is no evidence for increased incidence of cholelithiasis in SIT.³

Since Mouret first performed it in 1987, laparoscopic cholecystectomy (LC) has become the standard operative procedure for cholelithiasis.⁴

Several cases have been reported in patients with situs inversus totalis. Laparoscopic cholecystectomy in these patients is technically more demanding and needs reorientation of visual-motor skills to the left upper quadrant.⁵

Here in we present a case report of laparoscopic cholecystectomy in situs inversus totalis with its difficulty and review of literature of reported such cases

2. Case report

A 10 year old boy presented with left hypochondrium and epigastric pain 2 months duration. The pain was colicky in nature, prominent after meals. He was afebrile, not jaundiced. The patient had not been diagnosed as situs inversus totalis before. Apex beat was found in the right hemithorax at 5th intercostals space on physical examination. Electrocardiogram (ECG) revealed inverted P waves in I, aVL, and aVR which were suggestive of dextrocardia. The abdominal examination was unremarkable except the patient exhibit a left sided “Murphy's sign”. Chest radiography revealed situs inversus, with no evidence of bronchietasis (Fig. 1).

Fig. 1.

Chest radiography showing dextrocardia.

Further evaluation with ultrasound, computerized tomography (CT) and magnetic resonant image (MRI) confirmed situs inversus totalis and presence of multiple gall bladder stones with no intra or extrabiliary duct dilatation were observed (Figs. 2 and 3).

Fig. 2.

Computed tomography (CT) revealed the liver in the left side; spleen and stomach are in the right side of the abdomen.

Fig. 3.

Magnetic resonance image (MRI) revealed a left side liver and gall bladder.

Laboratory investigation revealed normal liver functions, normal coagulation profile excluding the hematological causes of gall stone in such age. After providing consent from the parents of the boy, the patient underwent laparoscopic cholecystectomy.

3. Technique

Laparoscopic cholecystectomy was performed with the 4-trocar Technique, according to the American variable. The operative team and laparoscopic devices were located in the theater as a mirror image configuration of normal laparoscopic cholecystectomy. The pneumoperitoneum (CO₂) was created by insertion of a veress needle through the subumblical area with a pressure of 12 mmHg. Two 10-mm trocars were inserted into the abdominal cavity, one in the position of the veress needle for laparoscope and the other one in the subxiphoid location. A 5-mm trocar was inserted at left mid-clavicular line and second 5-mm trocar was inserted in left anterior axillary line under the view of laparoscope.

At laparoscopic abdominal exploration the entirely of the abdominal contents were indeed reversed, at the Calot's triangle the cyst artery was found to lateral side on the right of cystic duct with two branches: an anterior cystic branch running to the cystic duct and Hartmann's pouch and a posterior cystic branch running to the inferior surface of the gall bladder (Fig. 4). Fundus of the gall bladder was grasped and retracted by the assistant using a clinch, which was inserted through the 5-mm trocar in the left anterior axillary line. Traction of the Hartmann's pouch was performed by left hand of the surgeon using a grasper inserted through the trocar located at left midclavicular line. Dissection of the Calot's triangle is one of the major problems for a right-handed surgeon in case of situs inversus totalis abnormality. Dissection of Calot's triangle was carried out with a posterior approach by using a forceps that was inserted through the trocar located in the midline. Meticulous dissection ensured complete freeing and definition of the course of both cystic duct and cystic artery, the duct was left to cystic artery (Fig. 5). Dissection was performed above the plane of Rouviene's sulcuc to avoid any injury. Both cystic duct and cystic arteries were clipped (double proximal, single distal 10-mm titanium clips) and then divided by hook scissors. After division of all peritoneal reflection on either side, the gall bladder was retrogradally separated from the liver bed by using electrocautry. It was then extracted through the 10-mm umbilical port. Fascial closure of large ports was sutured with absorbable material to prevent herniation.

Fig. 4.

Calot's triangle of the gall bladder in situs inversus totalis.

Fig. 5.

Dissection of Calot's triangle of the gall bladder in situs inversus totalis.

The postoperative period was uneventful, and the patient was discharged on the first day of postoperative period.

4. Discussion

Since Campos and Sipes⁶ described the first case of laparoscopic cholecystectomy in a patient with situs inversus, this uncommon malformation has been challenging and amazing many surgeons. Due to the contralateral disposition of the viscera, the diagnosis and surgical approach of these patients may be more difficult than that of orthotropic patients.

Situs inversus does not predispose one to gallbladder disease, but it leads to diagnostic confusion.⁷ Most patients presented with left-sided upper abdominal pain. However, about 10% of patients with left-sided cholelithiasis present with right-sided abdominal pain.⁷ This phenomenon has been observed for both visceral biliary pain and somatic pain in cases of cholecystitis and suggest that the central nervous system may not share in general transposition.⁸

A high index of suspicion in hence the key to avoiding mishaps in patients with situs inversus presenting with an acute abdomen. Apart from the confusion related to the side of the pain, the spectrum of clinical presentation related to complication of left-sided cholelithiasis is similar to that occurring in right-handed gallbladder.⁷

An apical beat in the right fifth intercostals space, reserved side of the liver dullness, and the right testicle hanging lower than the left occasionally suggest situs inversus.⁷ Ultrasonography, abdominal CT, chest scan, and MRI will confirm the presence of visceral transposition. Several reports in the literature emphasize the feasibility of the safe laparoscopic cholecystectomy in this challenging situation.^9,10 However, the procedure often requires more time to rearrange the equipments in the operating room and axtratime to recognize the mirror image anatomy.¹¹

The anatomical variation and, mainly, the controlateral disposition of the biliary tree demand an accurate dissection and exposure of the biliary structures to avoid iatrogenic injures.

Technical difficulties merit consideration, the mirror image reversibility of the abdominal viscera requires the surgeon to stand on the right side with video monitor above the patient left shoulder. Two 10-mm ports are placed in the epigastric and subumbilical positions. Two 5-mm ports are placed in the left midclavicular and left anterior axillary line. The lens should be 30° has been found to be superior in delineating Calot's triangle.

Technical aspect of laparoscopic cholecystectomy in patients with situs inversus totalis is privilege left-handed surgeons. The dissection of the biliary tree can be carried out with either the right or left hand,¹² however, for right-handed surgeons using the unskilled and non dominant left hand; the manipulation may be cumbersome and not precise. This is most apparent during clip application where both precision and power are required. However, in this case using the right hand has technical difficulties of either having to cross the hands or hyperflex the trunk and strike the anesthesia screen with left elbow.⁵

The skeletonzing of the structure of Calot's triangle consumes extra time and is more difficult than in patient with a normally located gallbladder, some have overcome such difficulties by standing between 2 abducted lower limbs.^1,13

The problem of crossing the hands to retract Hartmann's pouch while dissecting Calot's triangle has been overcome by some by allowing the first assistant to retract Hartmann's pouch while the primary surgeon dissect Calot's triangle using his right hand via epigastric port without hindrance.¹¹

The dissection should be carried out from lateral side as in this study by dissecting on lateral (left side) of the Hartmann's pouch and stick closely to the gall bladder wall until the cystic duct skelatonized from above downward (from the Hartmann's pouch to the cystic duct). The general agreement is that the procedure will be longer than in patient with normally located gallbladder, the need to redirect the visual-motor skills of the surgeon and the cameraman to the left upper quadrant along with the difficulty in skeletonizing Calot's triangle in responsible for longest portion of the extraoperative time.¹

Apart from mirror image transposition, patients with situs inversus usually do not have associated extrahepatic biliary, venous, and arterial anomalies.^7,8,14,15 Hence, it appears that the surgeon should not be discouraged from performing laparoscopic cholecystectomy for situs inversus on the ground of unexpected associated biliary tract anomalies. In one report, the common hepatic artery originating from the superior mesenteric artery variant that is known to occur in 17% of persons with normal anatomy.¹⁵ However, in patients with situs inversus partialis, there is an increased possibility of associated biliary tract and vascular anomalies and such patients may need intraoperative cholangiography and a low threshold for conversion to open surgery.¹ However, other still feel it is safer to perform laparoscopic cholecystectomy in those patients.¹⁶

There are also other extrabdominal anomalies, especially cardiac ones that are more frequent in patients with situs inversus. The incidence of situs inversus partialis is however much lower with only 3 cases reported among the patients who underwent laparoscopic cholecystectomy.¹

Since Campos and Sipes⁶ there were additional 50 cases of laparoscopic cholecystectomy in patients with situs inversus totalis have been reported up to date in review of the world literature (Table 1).^{1–3,5–7,9–13,17–53}

Table 1.

Summary of (50) patients with situs inversus totalis treated by laparoscopic cholecystectomy.

Serial no. of cases in each series	Series	Year of publication	Diagnosis
1	Campos and Sipes6	1991	CC
2	Takei et al.7	1992	Biliary colic
3	Lipschutz et al.17	1992	Cholangitis/CBD calculi
4	Goh18	1992	Empyema
5	Drover et al.19	1992	CC
6	Huang et al.20	1992	CC
7	Schiffino et al.9	1993	CC
8	Mc Dermott and Caushaj21	1994	Cholangitis/CBD calculi
9	Elhomsy et al.22	1996	AC
10	Malatani23	1996	AC
11	Crosher et al.3	1996	Biliary colic
12	D’Agata and Boncompagni24	1997	CC
13	Habib et al.25	1998	CC
14/15	Demetriades et al.26	1999	AC/CC
16	Djohan et al.27	2000	CC/appendectomy
17	Wong28	2001	CC/CBD calculi
18	Dorthi et al.29	2001	CC
19	Nursal et al.30	2001	CC
20/21	Yaghan et al.1	2001	CC/CC
22	Al Jumaily and Hoche2	2001	CC
23	Singh and Dhir31	2002	CC
24	Trongue et al.32	2002	CC
25	Polychronidis et al.33	2002	CC
26	Oms and Badia12	2003	AC
27	Jesudason et al.13	2004	CC
28	Kang and Han34	2004	CC/CBD calculi
29	Docimo et al.35	2004	CC
30	Antal et al.36	2004	CC
31	Pitiakoudis et al.37	2005	CC
32	McKayand Blake11	2005	AC
33	Kamitani et al.38	2005	CC
34	Puglisi et al.39	2006	CC
35	Bedioui et al.10	2006	CC
36	Aydin et al.40	2006	CC
37	Machado and Chopra5	2006	CC
38	Kumar and Fusai41	2007	CC
39	Fernandes et al.42	2008	CC
40	Hamdi and Abu hamdan43	2008	AC
41	Pavlidis et al.44	2008	AC
42	Taskin et al.45	2009	CC/gastric banding
43	Masood et al.46	2009	CC
44	Pereira-Graterol et al.47	2009	CC
45	Romano et al.48	2009	Biliary colic
46	Pataki et al.49	2010	CC
47	Hall et al.50	2010	CC
48	Gonzalez Valverde et al.51	2010	CC
49	Sanduc and Toma52	2010	CC
50	Han et al.53	2011	CC

AC = acute cholecystitis; CC = chronic cholecystitis; CBD = common bile duct.

Among those, 7 patients had acute cholecysitis,3 had biliary colic,1 had empyema, 2 had cholangitis,4 associated with common bile duct calculi and endoscopic retrograde choloangiopancreatography was performed in those 4 patients (2 among them had associated cholangitis), one associated with adjustable gastric banding, one associated with appendectomy and 33 had chronic cholecystitis.^7,17,21

Successful laparoscopic exploration of common bile duct for choledocholithiasis in a patient with situs inversus totalis has been reported recently.³⁴

The convention four ports laparoscopic cholecystectomy was used in this case report, but there is a report of feasibility of laparoscopic cholecystectomy with three ports only.⁴⁴ More recently a single incision multiport laparoscopic cholecystectomy in situs inversus totalis have been reported.⁵³

No major complications were reported, and almost all patients left the hospital within 48 h after surgery and did not require any specific postoperative care. To the best of our knowledge, no conversions to open surgery have been reported. The logical assumption is that the rate of conversion to open cholecystectomy in situs inversus should be higher in situs inversus patients than in patients with normally located gallbladder. This is not actually true as we believe that this is attributable to the fact that extra precaution is taken while carrying out laparoscopic cholecystectomy in this challenging situation.

In conclusion, laparoscopic cholecystectomy is feasible and should be done in situs inversus totalis patient by experienced laparoscopic surgeon, as changes in anatomical disposition of organ not only influence the localization of symptoms and signs arising from a diseases organ but also imposes special demands on the diagnosis and surgical skills of the surgeon.

Conflict of interest

No conflict of interest to declare.

Funding

None.

Ethical approval

This article was approved by Ethical Committee of the Department of Hepatobiliary Surgery, National Liver Institute, Menophyia University.

Author contributions

Ibrahim Abdelkader Salama: study design, writing, final revision and surgical team. Mohammed Hussein Abdullah: data collections, review of the literature and anesthesia team. Mohammed Houseni: data analysis, review of the literature and radiology team.