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Cirujano General 2021; 43 (1): 5-8 www.medigraphic.com/cirujanogeneral
www.medigraphic.org.mx
General
Cirujano
January-March 2021
Vol. 43, no. 1 / p. 5-8
Original article
* Gastrointestinal
Endoscopy Service/
Hospital General de
Zona No. 35, Instituto
Mexicano del Seguro
Social. Mexico.
‡ Department of Medical
Sciences/Universidad
Autónoma de Ciudad
Juárez. Mexico.
Received: 08/14/2019
Accepted: 08/03/2021
Primary choledocholithiasis in total situs inversus
Coledocolitiasis primaria en situs inversus total
Sergio Morales-Polanco,* Oscar I Ortiz-Ruvalcaba,* Juan de Dios Díaz-Rosales*,‡
Keywords:
Choledocholithiasis,
situs inversus,
endoscopic retrograde
cholangiopancreatography,
endoscopy.
Palabras clave:
Coledocolitiasis, situs
inversus,
colangiopancreatografía
retrógrada
endoscópica,
endoscopia.
ABSTRACT
Situs inversus totalis is a rare congenital entity characterized
by right-to-left transposition of the viscera of the thorax
and abdomen. We present the case of a 58-year-old
female patient with a history of cholecystectomy 18 years
ago, when a diagnosis of situs inversus was made, who
presented to the emergency department with obstructive
jaundice. With the surgical history and prior knowledge
of her condition, an imaging approach and successful
endoscopic treatment was performed. Cholelithiasis and
situs inversus are a rare combination of entities; this
binomial reminds us that in medicine there are no absolute
concepts.
RESUMEN
El situs inversus totalis es una rara entidad congénita
caracterizada por la transposición de derecha a izquierda
de las vísceras del tórax y abdomen. Se presenta el caso
de paciente femenino de 58 años con antecedente de co-
lecistectomía hace 18 años (donde se realizó diagnóstico
de situs inversus). Acudió a urgencias con un cuadro de
ictericia obstructiva. Con el antecedente quirúrgico y el
conocimiento previo de su condición, se realizó un abordaje
por imagen y un tratamiento endoscópico satisfactorio. La
colelitiasis y el situs inversus son una rara combinación
de entidades; este binomio nos recuerda que en medicina
no existen conceptos absolutos.
How to cite: Morales-Polanco S, Ortiz-Ruvalcaba OI, Díaz-Rosales JD. Primary choledocholithiasis in total situs inversus.
Cir Gen. 2021; 43(1): 5-8.
INTRODUCTION
Situs inversus totalis is considered a non-
pathologic congenital entity characterized
by right-to-left transposition of the totality of
the viscera of the thorax and abdomen.1 It has
a frequency of 1:5,000-10,000 live births.2
This rare condition often puts physicians in
clinical dilemmas in the face of a common
emergency.
Cholelithiasis is a public health problem and
could be considered as part of a metabolic and
degenerative problem.3 Choledocholithiasis
is a frequent complication of cholelithiasis
and generates more costs and other possible
complications in its treatment.
We present the case of a patient with
situs inversus and primary choledocholithiasis
who was successfully treated by endoscopic
retrograde cholangiography (ERCP).
PRESENTATION OF THE CASE
A 58-year-old female patient was admitted
to the emergency department for jaundice
and left upper quadrant pain of three days’
evolution. She had been suffering from colicky
pain for three months, which worsened after
consuming (atty food and improved after taking
antispasmodic drugs. On physical examination
her temperature we 37.9 oC, respiratory rate 18
per minute, heart rate 88 beats per minute, and
blood pressure 145/97 mmHg. Laboratory test
results on admission showed leukocytosis of 13
× 109 with neutrophilia of 78.3%, a serum total
bilirubin (TB) of 10.3 mg/dl, a direct bilirubin
(DB) of 7.3 mg/dl and indirect bilirubin (IB) of
2.7 mg/dl; a serum aspartate aminotransferase
(AST) of 189 IU/l, alanine aminotransferase
(ALT) of 98 IU/l, alkaline phosphatase (ALP)
of 350 IU/l, a gamma glutamyl transpeptidase
Morales-Polanco S et al. Choledocholithiasis & situs inversus
6
Cirujano General 2021; 43 (1): 5-8 www.medigraphic.com/cirujanogeneral
www.medigraphic.org.mx
(GGT) of 630 IU/l, and serum amylase of 148
IU/l and lipase of 47 IU/l.
The patient had a history of laparotomy
for acute abdomen 18 years ago with the
finding of situs inversus and cholecystitis,
for which she underwent cholecystectomy
without apparent complications. With the
surgical history, the findings described
by the patient and the time of evolution,
obstructive jaundice was diagnosed and
primary choledocholithiasis was suspected
due to the time of evolution from the
previous procedure to her current condition,
so imaging studies were requested. An
abdominal CT scan confirmed the diagnosis
of situs inversus, dilatation of the bile duct
and a single 15 mm bile duct stone in the
common bile duct (Figure 1).
The patient underwent endoscopic
retrograde cholangiography (ERCP), under
general anesthesia in prone position and
with the endoscopist on the right side of the
table. The duodenoscope was introduced up
to the stomach, and a 180o counterclockwise
rotation was performed and introduced into
the pylorus. With the duodenoscope in the
second portion of the duodenum and in a
long loop (due to the difficulty for correct
positioning) a careful visualization was
performed, and a native papilla was found
and cannulated with a one o’clock direction
(clockwise) towards the bile duct. In this case,
this “reverse” direction allowed a correct
cannulation procedure. A cholangiography
was performed, which showed dilatation of
the common bile duct and filling defects, so
a sphincterotomy, up to the transverse fold,
and dilatation of the sphincter of Oddi were
performed. Three sweeps were done with
an extraction balloon catheter, obtaining
abundant biliary detritus (Figure 2) and a
correct emptying of the contrast medium at
the end of ERCP procedure. No evidence of
other organic cause of jaundice was found,
so she was discharged the same day of the
procedure. During subsequent evaluations for
one whole year, she remained asymptomatic
and with these lab results at her last follow-
up: TB 1.2 mg/dl, DB 0.6 mg/dl, IB 0.6 mg/
dl, AST 46 UI/l, and ALT 56 UI/l.
DISCUSSION
Situs inversus is a rare condition that involves
diagnostic and therapeutic difficulties in
common diseases. Although this condition
is not per se a risk factor for cholelithiasis, it
does increase the difficulty and risks while
performing invasive procedures, such as ERCP
,
because the anatomy must be mirrored.4
There is no consensus on how to perform
ERCP in these cases, nor is there a gold standard
Figure 1: Axial and coronal CT scan confirming the diagnosis of situs inversus totalis and showing dilatation of the
bile duct and a 15 mm lithiasis.
7
Morales-Polanco S et al. Choledocholithiasis & situs inversus
Cirujano General 2021; 43 (1): 5-8 www.medigraphic.com/cirujanogeneral
www.medigraphic.org.mx
technique for treating choledocholithiasis in
situs inversus. The ERCP will be performed
by an endoscopist or surgeon who feels most
confident or as conditions allow the procedure
at that time.
Although it is a rare entity, the adult
patient may be aware of his/her condition
and give it the required importance in case
another common disease, such as appendicitis,
cholecystitis, diverticulitis, for example, occurs.5
The icteric patient with situs inversus
should undergo the same diagnostic and
therapeutic approach as a patient with normal
anatomy. Imaging studies such as abdominal
ultrasound and CT scan will make the diagnosis
in most cases. However, if prior knowledge
of the biliary anatomy is required, a magnetic
resonance cholangiography imaging will
give detailed information to plan the most
convenient therapeutic approach.6
Although there is no consensus on the
therapeutic approach, ERCP is the initial
treatment most authors claim and, therefore,
it may be considered it as the treatment of
choice. Although some authors consider
that the position of the endoscopist on
the right side of the table is not necessary
for performing ERCP in situs inversus,7 the
true is that there is no consensus, so it is
advisable to perform the procedure as the
endoscopist or surgeon feels more confident
with the technique or the conditions at that
moment allow it. This practically justifies all
the maneuvers performed to achieve the
objective, meaning extracting the biliary
stone or cleaning the biliary tract with the
greatest safety. We must emphasize that, due
to the technical difficulty, an unsatisfactory
endoscopic procedure should not be
considered as a failure,8 and in that case the
patient must be approached surgically.9-11
In conclusion, cholelithiasis and situs
inversus are a rare combination of entities, and
this binomial reminds us that in medicine there
are no absolute concepts.
REFERENCES
 1. 	Hu Y, Zeng H, Pan XL, Lv NH, Liu ZJ, Hu Y.
Therapeutic endoscopic retrograde endoscopic
cholangiopancreatography in a patient with situs
inversus viscerum. World J Gastroenterol. 2015; 21:
5744-5748.
 2. 	Fiocca F, Donatelli G, Ceci V, Cereatti F, Romagnoli F,
Simonelli L, et al. ERCP in total situs viscerum inversus.
Case Rep Gastroenterol. 2008; 2: 116-120.
 3. 	Díaz-Rosales JD, Alcocer-Moreno JA, Enríquez-
Domínguez L. Metabolic syndrome and complicated
cholecystitis in adult women. Arch Med. 2016; 16:
304-311.
 4. 	Hu L, Chai Y, Yang X, Wu Z, Sun H, Wang Z.
Duodenoscope combined with laparoscopy in
treatment of biliary stones for a patient with situs
inversus totalis: a case report. Medicine (Baltimore).
2019; 98: e14272.
 5. 	Rosen H, Petrosyan M, Mason RJ. Cholecystitis in situs
inversus totalis. Radiol Case Rep. 2008; 3 (4): 226.
 6. 	Eddes EH, Koster K. MRCP in situs inversus. Dig Surg.
2002; 19: 2.
 7. 	Sheikh I, Heard R, Tombazzi C. Technical factors
related to endoscopic retrograde retrograde
Figure 2: Cholangiography showing bile duct dilatation and balloon probe sweep.
Morales-Polanco S et al. Choledocholithiasis & situs inversus
8
Cirujano General 2021; 43 (1): 5-8 www.medigraphic.com/cirujanogeneral
www.medigraphic.org.mx
cholangiopancreatography in patients with situs inversus.
Gastroenterol Hepatol (NY). 2014; 10: 277-278.
 8. 	Morales-Rodríguez JF, Corina Cotillo E, Moreno-Loaiza
O. Surgical treatment of choledocholithiasis in a patient
with situs inversus totalis: a case report and literature
review. Medwave. 2017; 17 (06): e7002.
 9. 	Alzahrani HA, Yamani NM. Gallbladder agenesis with
a primary choledochal stone in a patient with situs
inversus totalis. Am J Case Rep. 2014; 15: 185-188.
10. 	Takalkar YP
, Koranne MS, Vashisst KS, Khedekar PG,
Garale MN, et al. Laparoscopic cholecystectomy with
choledochoduodenostomy inapatientwith situs inversus
totalis. J Minim Access Surg. 2018; 14: 241-243.
11. 	Moyon MA, Rojas CL, Moyon FX, Aguayo WG,
Molina GA, et al. Acute cholecystitis and residual
choledocholithiasis in situs inversus patient, successful
approach and ERCP a case report from Ecuador. Ann
Med Surg (Lond). 2020; 54: 101-105.
Ethical considerations and responsibility:
Data privacy. In accordance with the protocols
established at the authors’ place of work, the
authors declare that they have followed the
protocols on patient data privacy and preserved
their anonymity. The informed consent of the
patient referred to in the article is in the possession
of the main author.
Funding: No financial support was received for
this work.
Disclosure: The authors declare that there is no
conflict of interest in carrying out this work.
Correspondence:
Sergio Morales-Polanco
E-mail: sergiomp90@hotmail.com

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Primary choledocolithiasis in total situs inverses

  • 1. Cirujano General 2021; 43 (1): 5-8 www.medigraphic.com/cirujanogeneral www.medigraphic.org.mx General Cirujano January-March 2021 Vol. 43, no. 1 / p. 5-8 Original article * Gastrointestinal Endoscopy Service/ Hospital General de Zona No. 35, Instituto Mexicano del Seguro Social. Mexico. ‡ Department of Medical Sciences/Universidad Autónoma de Ciudad Juárez. Mexico. Received: 08/14/2019 Accepted: 08/03/2021 Primary choledocholithiasis in total situs inversus Coledocolitiasis primaria en situs inversus total Sergio Morales-Polanco,* Oscar I Ortiz-Ruvalcaba,* Juan de Dios Díaz-Rosales*,‡ Keywords: Choledocholithiasis, situs inversus, endoscopic retrograde cholangiopancreatography, endoscopy. Palabras clave: Coledocolitiasis, situs inversus, colangiopancreatografía retrógrada endoscópica, endoscopia. ABSTRACT Situs inversus totalis is a rare congenital entity characterized by right-to-left transposition of the viscera of the thorax and abdomen. We present the case of a 58-year-old female patient with a history of cholecystectomy 18 years ago, when a diagnosis of situs inversus was made, who presented to the emergency department with obstructive jaundice. With the surgical history and prior knowledge of her condition, an imaging approach and successful endoscopic treatment was performed. Cholelithiasis and situs inversus are a rare combination of entities; this binomial reminds us that in medicine there are no absolute concepts. RESUMEN El situs inversus totalis es una rara entidad congénita caracterizada por la transposición de derecha a izquierda de las vísceras del tórax y abdomen. Se presenta el caso de paciente femenino de 58 años con antecedente de co- lecistectomía hace 18 años (donde se realizó diagnóstico de situs inversus). Acudió a urgencias con un cuadro de ictericia obstructiva. Con el antecedente quirúrgico y el conocimiento previo de su condición, se realizó un abordaje por imagen y un tratamiento endoscópico satisfactorio. La colelitiasis y el situs inversus son una rara combinación de entidades; este binomio nos recuerda que en medicina no existen conceptos absolutos. How to cite: Morales-Polanco S, Ortiz-Ruvalcaba OI, Díaz-Rosales JD. Primary choledocholithiasis in total situs inversus. Cir Gen. 2021; 43(1): 5-8. INTRODUCTION Situs inversus totalis is considered a non- pathologic congenital entity characterized by right-to-left transposition of the totality of the viscera of the thorax and abdomen.1 It has a frequency of 1:5,000-10,000 live births.2 This rare condition often puts physicians in clinical dilemmas in the face of a common emergency. Cholelithiasis is a public health problem and could be considered as part of a metabolic and degenerative problem.3 Choledocholithiasis is a frequent complication of cholelithiasis and generates more costs and other possible complications in its treatment. We present the case of a patient with situs inversus and primary choledocholithiasis who was successfully treated by endoscopic retrograde cholangiography (ERCP). PRESENTATION OF THE CASE A 58-year-old female patient was admitted to the emergency department for jaundice and left upper quadrant pain of three days’ evolution. She had been suffering from colicky pain for three months, which worsened after consuming (atty food and improved after taking antispasmodic drugs. On physical examination her temperature we 37.9 oC, respiratory rate 18 per minute, heart rate 88 beats per minute, and blood pressure 145/97 mmHg. Laboratory test results on admission showed leukocytosis of 13 × 109 with neutrophilia of 78.3%, a serum total bilirubin (TB) of 10.3 mg/dl, a direct bilirubin (DB) of 7.3 mg/dl and indirect bilirubin (IB) of 2.7 mg/dl; a serum aspartate aminotransferase (AST) of 189 IU/l, alanine aminotransferase (ALT) of 98 IU/l, alkaline phosphatase (ALP) of 350 IU/l, a gamma glutamyl transpeptidase
  • 2. Morales-Polanco S et al. Choledocholithiasis & situs inversus 6 Cirujano General 2021; 43 (1): 5-8 www.medigraphic.com/cirujanogeneral www.medigraphic.org.mx (GGT) of 630 IU/l, and serum amylase of 148 IU/l and lipase of 47 IU/l. The patient had a history of laparotomy for acute abdomen 18 years ago with the finding of situs inversus and cholecystitis, for which she underwent cholecystectomy without apparent complications. With the surgical history, the findings described by the patient and the time of evolution, obstructive jaundice was diagnosed and primary choledocholithiasis was suspected due to the time of evolution from the previous procedure to her current condition, so imaging studies were requested. An abdominal CT scan confirmed the diagnosis of situs inversus, dilatation of the bile duct and a single 15 mm bile duct stone in the common bile duct (Figure 1). The patient underwent endoscopic retrograde cholangiography (ERCP), under general anesthesia in prone position and with the endoscopist on the right side of the table. The duodenoscope was introduced up to the stomach, and a 180o counterclockwise rotation was performed and introduced into the pylorus. With the duodenoscope in the second portion of the duodenum and in a long loop (due to the difficulty for correct positioning) a careful visualization was performed, and a native papilla was found and cannulated with a one o’clock direction (clockwise) towards the bile duct. In this case, this “reverse” direction allowed a correct cannulation procedure. A cholangiography was performed, which showed dilatation of the common bile duct and filling defects, so a sphincterotomy, up to the transverse fold, and dilatation of the sphincter of Oddi were performed. Three sweeps were done with an extraction balloon catheter, obtaining abundant biliary detritus (Figure 2) and a correct emptying of the contrast medium at the end of ERCP procedure. No evidence of other organic cause of jaundice was found, so she was discharged the same day of the procedure. During subsequent evaluations for one whole year, she remained asymptomatic and with these lab results at her last follow- up: TB 1.2 mg/dl, DB 0.6 mg/dl, IB 0.6 mg/ dl, AST 46 UI/l, and ALT 56 UI/l. DISCUSSION Situs inversus is a rare condition that involves diagnostic and therapeutic difficulties in common diseases. Although this condition is not per se a risk factor for cholelithiasis, it does increase the difficulty and risks while performing invasive procedures, such as ERCP , because the anatomy must be mirrored.4 There is no consensus on how to perform ERCP in these cases, nor is there a gold standard Figure 1: Axial and coronal CT scan confirming the diagnosis of situs inversus totalis and showing dilatation of the bile duct and a 15 mm lithiasis.
  • 3. 7 Morales-Polanco S et al. Choledocholithiasis & situs inversus Cirujano General 2021; 43 (1): 5-8 www.medigraphic.com/cirujanogeneral www.medigraphic.org.mx technique for treating choledocholithiasis in situs inversus. The ERCP will be performed by an endoscopist or surgeon who feels most confident or as conditions allow the procedure at that time. Although it is a rare entity, the adult patient may be aware of his/her condition and give it the required importance in case another common disease, such as appendicitis, cholecystitis, diverticulitis, for example, occurs.5 The icteric patient with situs inversus should undergo the same diagnostic and therapeutic approach as a patient with normal anatomy. Imaging studies such as abdominal ultrasound and CT scan will make the diagnosis in most cases. However, if prior knowledge of the biliary anatomy is required, a magnetic resonance cholangiography imaging will give detailed information to plan the most convenient therapeutic approach.6 Although there is no consensus on the therapeutic approach, ERCP is the initial treatment most authors claim and, therefore, it may be considered it as the treatment of choice. Although some authors consider that the position of the endoscopist on the right side of the table is not necessary for performing ERCP in situs inversus,7 the true is that there is no consensus, so it is advisable to perform the procedure as the endoscopist or surgeon feels more confident with the technique or the conditions at that moment allow it. This practically justifies all the maneuvers performed to achieve the objective, meaning extracting the biliary stone or cleaning the biliary tract with the greatest safety. We must emphasize that, due to the technical difficulty, an unsatisfactory endoscopic procedure should not be considered as a failure,8 and in that case the patient must be approached surgically.9-11 In conclusion, cholelithiasis and situs inversus are a rare combination of entities, and this binomial reminds us that in medicine there are no absolute concepts. REFERENCES  1. Hu Y, Zeng H, Pan XL, Lv NH, Liu ZJ, Hu Y. Therapeutic endoscopic retrograde endoscopic cholangiopancreatography in a patient with situs inversus viscerum. World J Gastroenterol. 2015; 21: 5744-5748.  2. Fiocca F, Donatelli G, Ceci V, Cereatti F, Romagnoli F, Simonelli L, et al. ERCP in total situs viscerum inversus. Case Rep Gastroenterol. 2008; 2: 116-120.  3. Díaz-Rosales JD, Alcocer-Moreno JA, Enríquez- Domínguez L. Metabolic syndrome and complicated cholecystitis in adult women. Arch Med. 2016; 16: 304-311.  4. Hu L, Chai Y, Yang X, Wu Z, Sun H, Wang Z. Duodenoscope combined with laparoscopy in treatment of biliary stones for a patient with situs inversus totalis: a case report. Medicine (Baltimore). 2019; 98: e14272.  5. Rosen H, Petrosyan M, Mason RJ. Cholecystitis in situs inversus totalis. Radiol Case Rep. 2008; 3 (4): 226.  6. Eddes EH, Koster K. MRCP in situs inversus. Dig Surg. 2002; 19: 2.  7. Sheikh I, Heard R, Tombazzi C. Technical factors related to endoscopic retrograde retrograde Figure 2: Cholangiography showing bile duct dilatation and balloon probe sweep.
  • 4. Morales-Polanco S et al. Choledocholithiasis & situs inversus 8 Cirujano General 2021; 43 (1): 5-8 www.medigraphic.com/cirujanogeneral www.medigraphic.org.mx cholangiopancreatography in patients with situs inversus. Gastroenterol Hepatol (NY). 2014; 10: 277-278.  8. Morales-Rodríguez JF, Corina Cotillo E, Moreno-Loaiza O. Surgical treatment of choledocholithiasis in a patient with situs inversus totalis: a case report and literature review. Medwave. 2017; 17 (06): e7002.  9. Alzahrani HA, Yamani NM. Gallbladder agenesis with a primary choledochal stone in a patient with situs inversus totalis. Am J Case Rep. 2014; 15: 185-188. 10. Takalkar YP , Koranne MS, Vashisst KS, Khedekar PG, Garale MN, et al. Laparoscopic cholecystectomy with choledochoduodenostomy inapatientwith situs inversus totalis. J Minim Access Surg. 2018; 14: 241-243. 11. Moyon MA, Rojas CL, Moyon FX, Aguayo WG, Molina GA, et al. Acute cholecystitis and residual choledocholithiasis in situs inversus patient, successful approach and ERCP a case report from Ecuador. Ann Med Surg (Lond). 2020; 54: 101-105. Ethical considerations and responsibility: Data privacy. In accordance with the protocols established at the authors’ place of work, the authors declare that they have followed the protocols on patient data privacy and preserved their anonymity. The informed consent of the patient referred to in the article is in the possession of the main author. Funding: No financial support was received for this work. Disclosure: The authors declare that there is no conflict of interest in carrying out this work. Correspondence: Sergio Morales-Polanco E-mail: sergiomp90@hotmail.com