Type I and II sphincter of Oddi dysfunction: a case-control study

Cirujano General 2023; 45 (1): 14-20 www.medigraphic.com/cirujanogeneral
General
Cirujano
January-March 2023
Vol. 45, no. 1 / p. 14-20
Original article
* General Surgeon
and Gastrointestinal
Endoscopist. General
Hospital of Zone No. 35
of the Mexican Institute
of Social Security
(IMSS). Mexico.
‡ Gastroenterologist
and Gastrointestinal
Endoscopist. Regional
General Hospital No.
66 of IMSS. Mexico.
§ Undergraduate
Medical Intern.
Institute of Biomedical
Sciences, Autonomous
University of Ciudad
Juarez, Ciudad Juarez,
Chihuahua, Mexico.
¶ Third-year Resident
of General Surgery.
Regional General
Hospital No. 66 of
IMSS. Mexico.
Received: 11/13/2022
Accepted: 02/22/2023
Type I and II sphincter of Oddi
dysfunction: a case-control study
Disfunción del esfínter de Oddi tipo I y II: estudio de casos y controles
Juan de Dios Díaz-Rosales,* Sergio Morales-Polanco,‡
Dante Deras-Ramos,§ Goretti Yáñez-Muñoz¶
Keywords:
choledocholithiasis,
sphincter of Oddi
dysfunction,
dyskinesia, stenosis.
Palabras clave:
coledocolitiasis,
disfunción del esfínter
de Oddi, discinesia,
estenosis.
ABSTRACT
Introduction: the sphincter of Oddi is a valvular complex
that regulates bile flow and pancreatic secretion. The
sphincter of Oddi dysfunction is divided into stenosis
(type I) or dyskinesia (type II). This study aims to
describe this pathology’s scenario, compare it with
cases of choledocholithiasis, and demonstrate if there
are differences or similarities. Material and methods:
a case-control study was performed where patients sent
to gastrointestinal endoscopy with a diagnosis of benign
biliary tract obstruction were analyzed between the
period from January 2019 to December 2021. Results:
there was no statistically significant difference between
the characteristics of patients with sphincter of Oddi
dysfunction and proven choledocholithiasis. Verifying the
statistic revealed differences in cannulation strategies or
post-endoscopic retrograde cholangiography pancreatitis
was also impossible. Conclusions: type I and type II
sphincter of Oddi dysfunction should be considered
as the same entity and treated with the same therapy
(endoscopic retrograde cholangiopancreatography with
sphincterotomy). Choledocholithiasis and sphincter of
Oddi dysfunction behave as similar pathological spectra,
since the clinical features involved do not show relevant
statistical differences.
RESUMEN
Introducción: el esfínter de Oddi es un complejo valvular
que regulariza el flujo biliar y la secreción pancreática.
La disfunción del esfínter de Oddi se divide en estenosis
(tipo I) o discinesia (tipo II). El objetivo de este estudio es
describir el escenario de esta patología, hacer una compa-
rativa con casos de coledocolitiasis y demostrar si existen
diferencias o similitudes. Material y métodos: se realizó un
estudio de casos y controles donde se analizaron pacientes
enviadas a endoscopia gastrointestinal con diagnóstico de
obstrucción benigna de la vía biliar entre el periodo de
enero de 2019 a diciembre de 2021. Resultados: entre las
características de las pacientes con disfunción del esfínter
de Oddi y coledocolitiasis comprobada no hubo diferencia
estadísticamente significativa. Tampoco fue posible verifi-
car diferencias estadísticamente reveladoras en las estrate-
gias de canulación ni en la pancreatitis postcolangiografía
retrógrada endoscópica. Conclusiones: la disfunción del
esfínter de Oddi tipo I y II deberá considerarse como
una misma entidad, tratarse con una misma terapéutica
(colangiopancreatografía retrógrada endoscópica con
esfinterotomía). La coledocolitiasis y la disfunción del
esfínter de Oddi se comportan como espectros patológicos
similares, ya que las características clínicas implicadas no
muestran diferencias estadísticas relevantes.
How to cite: Díaz-Rosales JD, Morales-Polanco S, Deras-Ramos D, Yáñez-Muñoz G. Type I and II sphincter of Oddi
dysfunction: a case-control study. Cir Gen. 2023; 45 (1): 14-20. https://dx.doi.org/10.35366/110698
doi: 10.35366/110698
INTRODUCTION
The sphincter of Oddi is a valvular complex
composed of smooth muscle that regulates
bile flow and pancreatic secretion into the
duodenal lumen. It has a resting pressure of
15 mmHg. It comprises a biliary sphincter and
a pancreatic sphincter joined at their distal
portion to form the ampullary sphincter at the
level of the second duodenal portion.1
S p h i n c t e r o f O d d i d y s f u n c t i o n
(SOD) is a diagnosis of exclusion and
encompasses a variety of disorders that
result in inappropriate function (stenosis or
dyskinesia) of this valve.2 This dysfunction
is associated with abdominal pain (although

15
Díaz-Rosales JD et al. Sphincter of Oddi type I and II dysfunction
a non-painful variant exists),3 elevation of
liver and pancreatic enzymes, bile duct and
pancreatic duct dilatation, and may also
be associated with pancreatitis.4 Although
both genders can be affected, it is more
common in women aged 20-50 years.5 The
prevalence of this disease in patients with
biliary pain after cholecystectomy is 14%.6
In both variants of SOD (stenotic and
dyskinetic), it is suggested that its etiology is
similar, and trauma is necessary (probably
from a litho smaller than 5 mm), which, when
passing through the sphincter of Oddi, causes
inflammation and the consequent formation of
a fibrotic ring by scarring (in half of the cases)
leading to the SOD syndrome.4
This entity has been controversial since its
first description, initially stratified according
to the Milwaukee classification (Table 1) and
later modified by Rome IV scale (Table 2).
This study aims to describe the scenario in
a second-level center facing this pathology,
compare it with cases of choledocholithiasis,
and demonstrate if there are differences or
similarities.
MATERIAL AND METHODS
A case-control study was conducted where
female patients referred to the gastrointestinal
endoscopy service diagnosed with benign bile
ductobstruction(diagnosisofcholedocholithiasis
referral) were studied from January 2019 to
December 2021. Patients were selected for the
case group who met the criteria for SOD: biliary
pain, altered LFTs (liver function tests) and bile
duct dilatation, absence of choledocholithiasis
and structural alteration in the bile duct (Rome
IV criteria), who had a history of uncomplicated
cholecystectomy (open or laparoscopic). As
Table 1: Milwaukee classification for sphincter of Oddi dysfunction.
Type I Type II Type III
Biliary pain + + +
Altered LFTs + ±* –
Bile duct dilatation + ±* –
Delayed biliary drainage + ±* –
LFTs = liver function tests with alanine aminotransferase (ALT) and aspartate aminotransferase (AST) twice the
average on two or more occasions.
Bile duct dilatation ≥ 12 mm on ultrasound or > 10 mm on cholangiography.
Delayed biliary drainage = drainage of contrast medium delayed for > 45 minutes after ERCP.
* One or two positive factors.
Tabla 2: Criterios diagnósticos para disfunción de esfínter de Oddi de tipo biliar según Roma IV.
Must include:
1. Biliary-type pain
2. Elevated liver enzymes or dilated bile duct (> 6 mm)
3. Absence of choledocholithiasis or other structural alterations of the biliary tract
Support criteria:
1. Normal amylase/lipase
2. Abnormal sphincter of Oddi manometry
3. Abnormal hepatobiliary scintigraphy
Suspected sphincter of Oddi dysfunction: biliary type pain + at least one associated objective finding.
Episodic functional abdominal pain: biliary-type pain without any other alteration.

16
support criteria, patients with amylase/lipase
within normal parameters at the time of the
study were included. For the control group,
60 female patients with endoscopic retrograde
cholangiopancreatography (ERCP)-confirmed
diagnoses of choledocholithiasis whose
resolution occurred at the endoscopic event
were selected. In both groups, an inclusion
criterion was that they had not undergone
previous ERCP
.
The following were evaluated: age, weight,
height, body mass index (BMI), age over 55
years, previous pancreatitis, bilirubin and
their differential, alanine aminotransferase,
aspartate aminotransferase, extrahepatic
bile duct (EHBD) size, difficult cannulation,
cannulation attempts, precutting, and post-
ERCP pancreatitis.
Two groups were pooled, the group
with SOD versus choledocholithiasis, and
the established variables were analyzed.
Values were expressed as absolute values and
percentages for categorical variables. They were
compared with the χ2 test or Fisher’s exact test.
In contrast, quantitative variables are expressed
as averages, ± standard deviation, and were
compared with Student’s t-test (for variables
of normal behavior) or Mann-Whitney U test
(for non-normal behavior variables). A value
of less than 0.05 was considered statistically
significant. The analyses were performed with
SPSS Statistics, Version 25.0 (Armonk, NY: IBM
Corp).
RESULTS
Twenty-two cases with a diagnosis of SOD
were studied, and 60 control patients with
choledocholithiasis were included. Patients
who met the criteria for suspected DEO
were referred with a diagnosis of suspected
choledocholithiasis. Four patients (18.2%)
had a previous diagnosis of pancreatitis. The
papilla of native characteristics was found in
all 22 patients. Cholangiography evidenced
increased caliber without filling defects in
all patients; a pencil-point termination and
adequate drainage of contrast medium were
observed after sphincterotomy. Ten patients
(45.4%) were cannulated with difficulty
criteria with an average of 3.8 attempts.
Precut papillotomy was used to cannulate in
four patients (18.2%), and in all 22 patients
(100%), cholangiography and sphincterotomy
were performed as treatment of the presumed
diagnosis of SOD. Extrahepatic bile duct
sweeping was performed in 17 patients
(77.3%) as part of bile duct securing. Three
patients (13.6%) had post-ERCP pancreatitis
as a complication (in one of these patients was
severe), but there was no mortality.
In the characteristics of patients with SOD
and proven choledocholithiasis, there was
no statistically significant difference in any
of the morphological or laboratory variables
(Table 3).
Nor could it prove statistically significant
differences in cannulation strategies or post-
ERCP pancreatitis (Table 4).
DISCUSSION
SOD is a broad functional disorder involving
a valve with inappropriate spasm or relaxation
and stenosis. It has an estimated prevalence
of 1.5% in the general population; however,
it appears underestimated due to the lack
of biochemical markers for its identification.
Manometric studies reveal that up to 10% of
biliary tract interventions involve the papilla,
even with no lithosclerosis lesions.7 In a study
carried out in the Hospital Juárez de México,
a prevalence (probable diagnosis) of DEO of
16.5% was observed,8 while in the Hospital
Central Militar, the prevalence was 18.9% (52
cases in 269 CPREs),9 while in another study
published by our group a prevalence of 20%
was reported.10 In patients with chronic or
idiopathic pancreatitis, the prevalence of SOD
can reach 59 and 72%, respectively.
In our study, the mean age of patients
with SOD was lower than in another study
performed in Japan (50.5 versus 62 years);
however, our study was performed only in
women, whereas in the study mentioned
above, women accounted for 69.4% of the
participants.11 In that same study, previous
pancreatitis was observed in 22%, whereas in
our analysis, the history of previous pancreatitis
was 18.2%. Regarding the caliber of the SBV,
in the Japanese study, it was 12.2 mm, while
in our study, it was 9 mm.

17
The Milwaukee classification was first used
in SOD; however, this classification could lead to
confusion, so the Rome IV criteria (which avoid
using manometry) are now used to diagnose.12
According to Rome IV, type I SOD no longer
exists and should be classified as benign
papillary stenosis (mechanical obstruction), not
a functional disorder. In contrast, type III SOD
is considered a functional entity that appears
to be unrelated to the sphincter of Oddi per
se.3 Thus, type II SOD (according to Rome IV)
is currently classified as the true SOD.13 It will
take some time to separate benign papillary
stenosis from functional disorders (Rome IV).
This diagnosis will be permanently linked to
SOD and will probably continue to be referred
to as type I SOD (even if manometry is not used
to make the diagnosis).
Table 3: Patient features.
SOD
(N = 22)
Choledocholithiasis
(N = 60) р
Age [years]* 50.5 ± 16.4 46.6 ± 16.2 0.3‡
Weight [kg]* 76.2 ± 17.4 76.7 ± 15.9 0.9‡
Height [m]* 1.58 ± 0.8 1.62 ± 0.8 0.08§
BMI [kg/m²]* 29.9 ± 5.05 28.9 ± 5.7 0.5‡
> 55 years, n (%) 9 (40.9) 11 (27.5) 0.3¶
Pancreatitis prior to ERCP, n (%) 4 (18.2) 5 (12.5) 0.7¶
TB [mg/dl] 3.5 ± 1.9 3.6 ± 2.5 0.8‡
DB [mg/dl] 2.2 ± 1.2 2.1 ± 1.6 0.9‡
IB [mg/dl] 1.3 ± 0.7 1.5 ± 1.01 0.4‡
ALT 264 ± 215.5 250.8 ± 192.6 0.8§
AST 228 ± 271 204.1 ± 192.6 0.8§
EBD size [mm] 9 ± 3.7 11.2 ± 5.2 0.6§
SOD = sphincter of Oddi dysfunction. BMI = body mass index. ERCP = endoscopic retrograde
cholangiopancreatography. TB = total bilirubin. DB = direct bilirubin. IB = indirect bilirubin. ALT = alanine
aminotransferase. AST = aspartate aminotransferase. EBD = extrahepatic bile duct.
* Data are mean ± standard deviation. ‡ Student’s t-test. § Mann-Whitney U. ¶ Pearson’s χ2.
Source: IMSS electronic file HGZ No. 35.
Table 4: Sphincter of Oddi cannulation.
Variable
SOD
(N = 22)
n (%)
Choledocholithiasis
(N = 40)
n (%) р
Cannulation (difficult) 10 (45.5) 14 (35) 0.4‡
Cannulation attempts* 3.8 ± 2.5 2.9 ± 2.01 0.18§
Precut 4 (18.2) 12 (30) 0.35¶
Post-ERCP pancreatitis 3 (13.6) 5 (12.5) 0.6¶
* Data are mean ± standard deviation. ‡ Pearson’s χ2. § Mann-Whitney U test. ¶ Fisher’s exact test.
SOD = sphincter of Oddi dysfunction.
Source: IMSS electronic file HGZ No. 35.

18
It is questionable whether SOD is a primary
pathologic process or a consequence of a
traumatic alteration of the sphincter of Oddi.
The surgical history suggests that it is the second
option, so a patient with recurrent symptoms
after cholecystectomy (due to cholelithiasis)
may be a case of secondary benign papillary
stenosis or type I SOD. Differentiating the
purely dysfunctional process (dyskinesia) from
the stenotic process is very complex. Since the
treatment is similar, it can be stated that patients
with a history of cholecystectomy could suffer
from stenosis-type dysfunction. In contrast,
those without a history of cholecystectomy and
evidence of gallbladder or common bile duct
lithiasis could be considered fully functional
(dyskinesia).
Diagnosis is complex, and overlooking
it leads to complications such as recurrent
biliary symptoms, elevated liver enzymes, and
even pancreatitis.14 There will be controversy
regarding the pain of SOD because the
characteristic is biliary type, which is related
to food (there are authors who refer that pain
in SOD is not related to food), usually lasting
from 30 minutes to a few hours and resolves
spontaneously. The diagnostic suspicion starts
with the pain clinic and laboratories, including
bilirubin, liver enzymes, amylase, and lipase.
Alkaline phosphatase may provide a clue
for diagnosis without increased bilirubin or
pancreatic enzymes.7 It is imperative to rule
out the presence of choledocholithiasis or
other biliopancreatic or ampulla of Vater
alterations, which could condition the
picture.6
Differentiating it from choledocholithiasis
was not possible in this study; there were
no characteristics with statistically significant
differences, so in our environment, ERCP has
a current role in the diagnosis and treatment of
this entity, even without a previous diagnostic
suspicion.
The gold standard for diagnosis is the
sphincter of Oddi manometry, whose
pressure > 40 mmHg (three standard
deviations above average) makes the
diagnosis. Patients with benign papillary
stenosis (SOD type I) may have normal
manometry up to 15-35%,15 while patients
with dyskinesia dysfunction (SOD type II)
may have normal manometry up to 45%.15
This suggests that the pure increase in the
sphincter of Oddi pressure is insufficient to
cause the disease’s symptoms.15
ERCP with sphincterotomy is the
management in patients with type I (stricture)
and type II (dyskinesia) SOD with a short-
term success rate greater than 90%;11 while
other series report a success rate of 60 to 94%
in patients whose diagnosis was not based
on manometry.16 The recurrence rate after
sphincterotomy treatment is 32% within six
months; however, this recurrence is related to
the presence and development of functional
dyspepsia.11
The rates of post-ERCP pancreatitis
in SOD range from 0 to 30% (if ERCP is
accompanied by manometry), so performing
manometry seems to be a risk factor for
this complication.2 In this study, the rate
of post-ERCP pancreatitis was 13.6% lower
compared to a Japanese study, where the
rate of post-ERCP pancreatitis was 36%;11
this is a very high rate even for those patients
undergoing sphincterotomy. Mortality from
adverse events after ERCP is 0.08%.17 In our
study, all cases underwent sphincterotomy
as treatment with a rate of post-ERCP
pancreatitis acceptably like controls with
choledocholithiasis (13.6 versus 12.5%, p =
0.6). ERCP with sphincterotomy is indicated
in patients with SOD who meet the criteria of
biliary-type pain, altered liver function tests,
and bile duct dilatation4 with a greater than
90% success rate in patients.18
It is recommended that during ERCP for
type I and type II SOD, indomethacin 100 mg
or diclofenac 75 mg rectally before or after
the procedure are administered,19 and place
a pancreatic stent 5 Fr and 4 cm in case of
unintentional cannulation of the pancreatic
duct,16 and that epinephrine be sprayed on
the papilla after the procedure.17
In our region (Ciudad Juarez), there is no
access to the sphincter of Oddi manometry
studies, and this scenario is constant in most
of the country. Moreover, this procedure
is less and less used due to the added risk
of pancreatitis that it entails. Other study
methods include scintigraphy with a lower
sensitivity than manometry (which shows

19
delayed emptying).20 In public and second-
level medical centers, there is also no access
to imaging studies of the biliary tract, such
as magnetic resonance cholangiography, so
ERCP is still valid as a diagnostic study? The
diagnosis of SOD in most hospitals is based
on the Rome IV criteria or post-ERCP findings
(in post cholecystectomy patients). It is stated
that 10% of patients may have a complete
diagnostic workup, and this percentage
needs to be revised.19 In these cases, how
prudent is treating them even without
complementary studies? In all the cases in
this study, the diagnosis was made post-ERCP
,
and to make this diagnosis, the presence of
choledocholithiasis or an ampullary tumor had
to be excluded.4
This study has weaknesses: it is a
retrospective study in a single hospital center,
the lack of follow-up of patients to observe the
resolution of their symptoms, and the long-term
response rate after sphincterotomy.
CONCLUSIONS
Both type I (stenosis) and type II (dyskinesia)
SOD should be considered as the same entity
that is treated with the same therapy (ERCP
with sphincterotomy). Type III SOD will be
reassigned to a functional entity in its entirety,
the treatment of which will be purely medical.
Ideal medicine is far from our reality, and
international guidelines only sometimes fit the
national scenario. Considering and treating
type I and type II dysfunction with the only
thing we have (sphincterotomy) may be risky,
but it is still justified. Choledocholithiasis and
SOD show that their clinical characteristics
involved do not present relevant statistical
differences.
REFERENCES
1. Bosch A, Peña LR. The sphincter of Oddi. Dig Dis Sci.
2007; 52: 1211-1218.
2. Peñaloza-Ramírez A, Coral-Argoty E, Castro-Ridríguez
M, Álvarez-Gil J, Aponte-Ordóñez P
. Sphincter of Oddi
dysfunction: a case report. Rev Colomb Gastroenterol.
2021; 36: 52-58.
3. Miyatani H, Mashima H, Sekine M, Matsumoto S.
Clinical features and management of painless biliary
type sphincter of Oddi dysfunction. J Int Med Res.
2019; 47: 2940-2950.
4. Boivineau G, Gonzalez JM, Gasmi M, Vitton V, Barthet
M. Sphincter of Oddi dysfunction. J Visceral Surg.
2022; 159: S16-S21.
5. Afghani E, Lo SK, Covington PS, Cash BD, Pandol
SJ. Sphincter of Oddi function and risk factors for
dysfunction. Front Nutr. 2017; 4: 1.
6. Vida-Perez L. Sphincter of Oddi dysfunction and
functional biliary disorders. RAPD online. 2017; 40:
233-242.
7. Castillo CR, Sandoval VE, Gonzalez HMA, Rosique
MJR, Wilson GG. Primary stenosing papillitis. Tab
Health. 2001; 7: 404-407.
8. Padrón AG, Pérez VE, Chávez GMA, Manrique
MA, García RE, Ortíz GR, et al. Endoscopic
cholangiopancreatography at Hospital Juárez de
México: three-year review (1997-1999). Endoscopy.
2000; 11: 161-167.
9. Reyes DA, Arana PG, Hernández CE. Therapeutic
endoscopic retrograde cholangiopancreatography
(ERCP): five-year experience at the Hospital Central
Militar. Endoscopy. 1998; 9: 41-46.
10. Ortíz-Ruvalcaba OI, Aguirre-Piria A, Díaz-Rosales JD,
Mena-Arias G, Morales-Polanco S, Guerrero-Pérez L.
Analysis of concordance between clinical diagnosis
and post-CPRE diagnosis in women with suspected
obstructive biliary tract pathology. Endoscopy. 2019;
31: 142-147.
11. Miyatani H, Mashima H, Sekine M, Matsumoto S.
Clinical course of biliary-type sphincter of Oddi
dysfunction: endoscopic sphincterotomy and
functional dyspepsia as affecting factors. Ther Adv
Gastroent Endosc. 2019; 12: 1-10.
12. Baig KR, Wilcox CM. Translational and clinical
perspectives on sphincter of Oddi dysfunction. Clin
Exp Gastroenterol. 2016; 9: 191-195.
13. Clark CJ. An update of biliary dyskinesia. Surg Clin N
Am. 2019; 99: 203-214.
14. Lariño NJ, Iglesias GJ, Domínguez MJE. Recurrent and
chronic pancreatitis. Medicine. 2012; 11: 465-472.
15. Wilcox CM. Sphincter of Oddi dysfunction type III:
new studies suggest new approaches are needed.
World J Gastroenterol. 2015; 21: 5755-5761.
16. García-Cano LJ, Viñuelas CM, Murillo MC, Muñiz
MM, del Moral M, Valiente GL, et al. ERCP treatment
of postcholecystectomy sphincter of Oddi dysfunction
without prior manometry. Endoscopy. 2017; 49: 1129-
1194.
17. Kin T, Katanuma A. Incidence of adverse
event associated with endoscopic retrograde
cholangiography in the current era: pursuit of a
lower incidence of post endoscopic retrograde
cholangiopancreatography pancreatitis. Dig Endosc.
2022; 34: 1205-1206.
18. Ren LK, Cai ZY, Ran X, Yang NH, Li XZ, Liu H, et al.
Evaluating the efficacy of endoscopic sphincterotomy
on biliary-type sphincter of Oddi dysfunction: a
retrospective clinical trial. World J Clin Cases. 2021;
9: 9835-9846.
19. Hollenbach M, Hoffmeister A. Adverse
events in endoscopic retrograde retrograde
cholangiopancreaticography (ERCP): focus on post-
ERCP-pancreatitis. United European Gastroenterol J.
2022; 10: 10-11.

20
20. Hyun JJ, Richard A, Kozarek RA. Sphincter of Oddi
dysfunction: sphincter of Oddi dysfunction or
discordance? What is the state of the art in 2018? Curr
Opin Gastroenterol. 2018; 34: 282-287.
Ethical considerations and responsibility: data
privacy. According to the protocols established
in our work center, we declare that we have
followed the protocols on patient data privacy
and preserved their anonymity.
Funding: no financial support was received for
the preparation of this work.
Disclosure: none of the authors have a conflict
of interest in the conduct of this study.
Correspondence:
Juan de Dios Díaz-Rosales, MD
E-mail: jdedios.diaz@uacj.mx

Type I and II sphincter of Oddi dysfunction: a case-control study

More Related Content

Similar to Type I and II sphincter of Oddi dysfunction: a case-control study (20)

More from Juan de Dios Díaz Rosales (20)

Recently uploaded (20)

Type I and II sphincter of Oddi dysfunction: a case-control study