Acute pancreatitis after thoracic duct ligation for iatrogenic chylothorax. A case report
Bédat et al. BMC Surgery
Acute pancreatitis after thoracic duct ligation for iatrogenic chylothorax. A case report
Benoît Bédat 0
Cosimo Riccardo Scarpa 0
Samira Mercedes Sadowski 0
Frédéric Triponez 0
Wolfram Karenovics 0
0 Thoracic and Endocrine Surgery, University Hospitals of Geneva , 1211 Geneva , Switzerland
Background: To report the association between thoracic duct ligation and acute pancreatitis. The association between sudden stop of lymphatic flow and pancreatitis has been established in experimental models. Case presentation: A 57-year-old woman operated for thymoma presented a iatrogenic chylothorax. After thoracic duct ligation, she presented an acute pancreatitis which resolved after conservative treatment. The chylothorax disappeared within 4 days of thoracic duct ligation. Conclusions: This is the first report of acute pancreatitis following thoracic duct ligation. The pancreas and digestive tract should be assessed in symptomatic patients after thoracic duct ligation.
Case report; Thoracic surgery; Thoracic duct ligation; Acute pancreatitis; Iatrogenic chylothorax; Thymoma
Chylothorax is a rare disease defined as a leakage of
chyle into the pleural space, and can be classified as
traumatic or non traumatic. Esophageal surgery is the
major cause of traumatic iatrogenic chylothorax. Other
iatrogenic causes include lymph node dissection, lung
resection and mediastinal tumor resection [1, 2]. A large
leak flow of chyle may cause dehydration, nutrient loss
and immunodeficiency. However, there is no consensus
in the management of chylothorax. Depending on
etiology, duration and flow output, therapy may either
be conservative or surgical. Low-output chyle flow
(<1000 mL/day) can generally be managed
conservatively. Failure of conservative treatment on the other
hand, typically in situations of high-output chyle flow
(>1000 mL/day), requires intervention, such as thoracic
duct ligation. This procedure has a high success rate of
up to 95% . Thoracic duct ligation has shown to have
a 38% rate of comorbidities linked to procedure, such as
atrial fibrillation and need of prolonged ventilation .
To our knowledge, there have been no abdominal
complications reported or associated with ligation of the
thoracic duct. In experimental models, some studies
demonstrated that the sudden stop of lymphatic flow
may induce intestinal and pancreatic edema with the
presence of an inflammatory infiltrate [4, 5].
We report herein the first case of an abdominal
complication associated with thoracic duct ligation, such as
A 57-year-old woman with a history of back pain underwent
a thoracic CT-scan in 2016 with the incidental discovery of
an anterior mediastinal mass. The mass had a size of
4x3cm, was round and was well delimited, compatible with
a teratoma or a thymoma. The patient had no symptom or
clinical manifestations of myasthenia gravis, with a
Quantitative Myasthenic Gravis Score grade 0. A thymomectomy
was performed using a right single-port video-assisted
thoracoscopic surgery (VATS). A chest drain was left after the
procedure. Histopathology confirmed a thymoma, type B1,
Masaoka-Koga stage 1. Two days after surgery, the patient
developed a high output chylothorax (>1000 mL/day),
without any signs of infection or inflammation in the blood
tests. We introduced a low-fat diet for 6 days and then she
was fasting for two more days. However, the chyle
continued to flow with an output of 750 mL/day without other
complications. No somatostatin or analog was introduced.
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A lymphangio-magnetic resonance imaging (MRI) didn’t
show aberrant thoracic duct anatomy nor chyle-leak (Fig. 1).
At day 10, the patient was taken to the operating room for a
right VATS. The thoracic duct was visualized and the
thoracic duct was clipped just above the diaphragm (Fig. 2).
Two days later, the chylothorax reappeared and the patient
developed increasing pain in the left hemi-abdomen, with
sign of peritonitis and abdominal distention. Her blood test
showed absence of leucocytosis, a C-reactive protein (CRP)
of 230 mg/L, and normal lipid, electrolytes, hepatic and
pancreatic levels (see Additional file 1). A CT-scan
demonstrated pancreatic edema and a peri-pancreatic infiltration
that extended to the bilateral kidney fasciae, compatible
with an acute pancreatitis (Fig. 3). Etiologies of acute
pancreatitis such as gallstone migration, alcohol,
medications, hypotension during the perioperative period, lipidic
and IgG4 disease were excluded (see Additional file 1).
There was no hypotension during the perioperative
periode. After medical supportive management the abdominal
pain resolved within 2 days and CRP decreased. The
chylothorax was treated with restricted low-fat diet, resolved
4 days after the thoracic duct ligation, and the chest tube
was removed 16 days after the initial thymomectomy. Six
months later, the patient is healthy and has had no
recurrence of chylothorax.
This case presentation was conducted in accordance
with the CARE guidelines and methodology.
The occurrence of chylothorax after thymomectomy is a
rare complication and usually associated with extensive
dissection . In this case, the resection was localized,
without radical thymectomy.
For chyle leak of traumatic iatrogenic etiology, such as
mediastinal tumor resection, management is debated
and to date no consensus exists. Initial conservative
treatment involving chest drainage and a low-fat diet
except for the medium-chain triglycerides or fasting with
Fig. 1 Axial MRI T2 showing the thoracic duct (red arrow)
Fig. 3 Arterial phase contrast CT showing an acute pancreatitis with
edema around the pancreas (red arrows)
parenteral nutrition has shown excellent outcome.
Octreotide or somatostatin therapy can be efficient for
the medical approach . However, the level of evidence
about the efficacy of these treatments remains low.
Furthermore, octreotide is a class II medication associated
with acute pancreatitis, and its use should be cautious
. For patients who fail conservative management,
surgical repair with ligation using VATS is shown to be
highly successful and is considered a safe procedure .
The difficulty lies in the visualization of the thoracic
duct injury during the surgery and its anatomic
variations. A lymphangiography, lymphoscintigraphy and
lymphangio-MRI may help localize the chyle-leak. In this
case, the leak site was not found on lympangio-MRI nor
during surgery. An intraoperatively administration of oral
cream via a stomach probe can be more easily identify the
duct and other accessory lymphatic channels . In this
situation, the upstream clipping of the thoracic duct just
above the diaphragm appears favorable for better result
than a mass ligation of the tissue in the presumed course.
The consequences of sudden disturbed lymph drainage
of the abdominal viscera, which constitutes 80% of the
lymphatic flow in the thoracic duct, are not known. It is
assumed that with time collateral lymphatic vessels
overcome the thoracic duct obstruction. Reports in literature
show that thoracic duct ligation is safe and has no
known associated abdominal or lymphatic
complications, except two cases of leg edema associated with
thoracic duct embolization . However, the role of
mesenteric lymph drainage in acute illness such as
pancreatitis, burns and hemorrhagic shock is well
established in experimental models . The impact of
thoracic duct ligation on the pancreas is known since 1958
by Papp et al. and was more investigated by Müller et al.
in 1988 with thoracic duct ligation in rats [4, 12]. Their
results demonstrated a long-lasting pancreatic edema. A
more recent study showed that thoracic duct ligation in
rats with acute hemorrhagic necrotizing pancreatitis
reduced lung injury by a decreased neutrophil infiltration
and TNF-alpha release, but increased pancreas injury
. In the intestine and the pancreas, the
myeloperoxidase activity, a marker of neutrophils infiltration, was
increased without any change of serum amylase and
diamine oxidase level. In another study, lymphatic
obstruction in dogs caused an intestinal mucosal atrophy
similar to malabsorption syndrome .
In our case, common etiologies of acute pancreatitis
were excluded, including hypotension and medications.
Auto-immune-like pancreatitis was previously described
in a patient with myasthenia gravis and autoantibodies
. However, our patient had no symptom or clinical
manifestations of myasthenia gravis with a Quantitative
Myasthenic Gravis Score grade 0. Therefore an immune
etiology seemed unlikely. The diagnosis of pancreatitis is
only based on the CT-scan. Otherwise, acute pancreatitis
with normoamylasemia and lipasemia is not uncommon,
and is a known entity . Another explanation could
be a pancreatic edema caused by a congestive lymphatic
vessels with similar symptoms. The comparison of the
experimental studies with our case is clearly limited by
their experimental design. However, the impact of a
sudden lymphatic obstruction on the pancreas and digestive
tract can be easily understood.
The recurrence of chylothorax 2 days after surgery
evokes aberrant collateral thoracic duct, which has not
been seen on MRI nor during surgery. Probably this
duct was small with a low chyle flow that could explain
a rapid healing after a conservative treatment and a
persistant disturbed lymph drainage of the abdominal
viscera with abdominal pain.
In conclusion, this is the first report of a patient
developing acute pancreatitis after thoracic duct clipping.
Although our knowledge relies on experimental models,
edema of the abdominal viscera should be assessed in
symptomatic patients with a CT-scan and a pancreatic
Additional file 1: Description of data : Blood test results 1 day after the
onset of the abdominal pain. WBC, white blood count; CRP, C-reactive
protein; AST, aspartate aminotransferase; ALT, alanine aminotransferase;
γGT, gamma-glutamyl transpeptidase; AP, alkaline phosphatase; IgG,
immunoglobulin G. (DOCX 14 kb)
BB, CRS and WK designed the report; WK performed the surgery; BB, CRS
and SMS collected the data; BB, FT and WK analyzed the data; BB and CRS
drafted the article; SMS, FT and WK revised the paper and gave the final
approval of the definitive version of the article. All authors read and
approved the final manuscript.
Ethics approval and consent to participate
Name of the ethics committee that approved the study: Commission
cantonale d’éthique de la recherche, Genève, Switzerland.
The patient gave consent to participate.
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