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WJCC https://www.wjgnet.com 10614 October 16, 2022 Volume 10 Issue 29
World Journal of
Clinical Cases
W J C C
Submit a Manuscript: https://www.f6publishing.com World J Clin Cases 2022 October 16; 10(29): 10614-10621
DOI: 10.12998/wjcc.v10.i29.10614 ISSN 2307-8960 (online)
CASE REPORT
Extracorporeal membrane oxygenation in curing a young man after
modified Fontan operation: A case report
He-Bing Guo, Jian-Bo Tan, Yong-Chao Cui, Hao-Feng Xiong, Chuan-Sheng Li, Yu-Feng Liu, Yao Sun, Lin Pu,
Pan Xiang, Ming Zhang, Jing-Jing Hao, Ning-Ning Yin, Xiao-Tong Hou, Jing-Yuan Liu
Specialty type: Critical care
medicine
Provenance and peer review:
Unsolicited article; Externally peer
reviewed.
Peer-review model: Single blind
Peer-review report’s scientific
quality classification
Grade A (Excellent): A
Grade B (Very good): 0
Grade C (Good): C, C
Grade D (Fair): 0
Grade E (Poor): 0
P-Reviewer: Apiratwarakul K,
Thailand; Cabezuelo AS, Spain;
Sharma D, India
Received: April 14, 2022
Peer-review started: April 14, 2022
First decision: July 11, 2022
Revised: July 14, 2022
Accepted: August 30, 2022
Article in press: August 30, 2022
Published online: October 16, 2022
He-Bing Guo, Jian-Bo Tan, Hao-Feng Xiong, Chuan-Sheng Li, Yu-Feng Liu, Yao Sun, Lin Pu, Pan
Xiang, Ming Zhang, Jing-Jing Hao, Ning-Ning Yin, Jing-Yuan Liu, Department of Critical Care
Medicine, Beijing Ditan Hospital, Capital Medical University, Beijing 100015, China
Yong-Chao Cui, Xiao-Tong Hou, Center for Cardiac Intensive Care, Beijing Anzhen Hospital,
Capital Medical University, Beijing 100029, China
Corresponding author: Jing-Yuan Liu, MD, Associate Professor, Doctor, Department of Critical
Care Medicine, Beijing Ditan Hospital, Capital Medical University, No. 8 Jingshun East Street,
Chaoyang District, Beijing 100015, China. dtyyicu@ccmu.edu.cn
Abstract
BACKGROUND
The Fontan operation is the only treatment option to change the anatomy of the
heart and help improve patients’ hemodynamics. After successful operation,
patients typically recover the ability to engage in general physical activity. As a
better ventilatory strategy, extracorporeal membrane oxygenation (ECMO)
provides gas exchange via an extracorporeal circuit, and is increasingly being used
to improve respiratory and circulatory function. After the modified Fontan
operation, circulation is different from that of patients who are not subjected to
the procedure. This paper describe a successful case using ECMO in curing
influenza A infection in a young man, who was diagnosed with Tausing-Bing
syndrome and underwent Fontan operation 13 years ago. The special cardiac
structure and circulatory characteristics are explored in this case.
CASE SUMMARY
We report a successful case using ECMO in curing influenza A infection in a 23-
year-old man, who was diagnosed with Tausing-Bing syndrome and underwent
Fontan operation 13 years ago. The man was admitted to the intensive care unit
with severe acute respiratory distress syndrome as a result of influenza A
infection. He was initially treated by veno-venous (VV) ECMO, which was switch-
ed to veno-venous-arterial ECMO (VVA ECMO) 5 d later. As circulation and
respiratory function gradually improved, the VVA ECMO equipment was
removed on May 1, 2018. The patient was successfully withdrawn from artificial
ventilation on May 28, 2018 and then discharged from hospital on May 30, 2018.
CONCLUSION
Guo HB et al. ECMO in a patient after Fontan operation
WJCC https://www.wjgnet.com 10615 October 16, 2022 Volume 10 Issue 29
After the modified Fontan operation, circulation is different compared with that of patients who
are not subjected to the procedure. There are certainly many differences between them when they
receive the treatment of ECMO. Due to the special cardiac structure and circulatory characteristics,
an individualized liquid management strategy is necessary and it might be better for them to
choose an active circulation support earlier.
Key Words: Acute respiratory distress syndrome; Extracorporeal membrane oxygenation; Modified Fontan
operation; Tausing-Bing syndrome; Case report
©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
Core Tip: After the modified Fontan operation, circulation is different from that of patients who are not
subjected to the procedure. In this article, we describe a 23-year-old man, with a history of modified
Fontan operation for Tausing-Bing syndrome, who was admitted to the intensive care unit with severe
acute respiratory distress syndrome as a result of influenza A infection. The man was initially treated by
veno-venous extracorporeal membrane oxygenation (ECMO), which was switched to veno-venous-arterial
ECMO 5 d later. As circulation and respiratory function gradually improved, the veno-venous-arterial
ECMO equipment was successfully removed. Then, the man was discharged from hospital successfully.
This case highlights that an individualized liquid management strategy is necessary and it might be better
for such patients to choose an active circulation support earlier.
Citation: Guo HB, Tan JB, Cui YC, Xiong HF, Li CS, Liu YF, Sun Y, Pu L, Xiang P, Zhang M, Hao JJ, Yin NN,
Hou XT, Liu JY. Extracorporeal membrane oxygenation in curing a young man after modified Fontan operation: A
case report. World J Clin Cases 2022; 10(29): 10614-10621
URL: https://www.wjgnet.com/2307-8960/full/v10/i29/10614.htm
DOI: https://dx.doi.org/10.12998/wjcc.v10.i29.10614
INTRODUCTION
The Fontan operation is the only treatment option to change the anatomy of the heart and help improve
patients’ hemodynamics[1]. The patient’s circulation after the Fontan surgery is different from that of a
normal patient. The mortality of patients diagnosed with acute respiratory distress syndrome (ARDS)
ranges from 17.3% to 41.4% among critically ill patients with H1N1 infection[2,3], and many patients
need the help of extracorporeal membrane oxygenation (ECMO). Whereas, due to the special circulation
after the Fontan surgery, supporting Fontan patients on ECMO carries high morbidity and mortality[4].
Few articles have described the special circulation about cases who receive ECMO after the modified
Fontan operation. We herein report such a case.
CASE PRESENTATION
Chief complaints
A 23-year-old male patient presented to the general intensive care unit (ICU) of Beijing Ditan Hospital
Affiliated to Capital Medical University on April 5, 2018 mainly due to fever for 5 d, with 39.1 °C as the
highest temperature.
History of present illness
Upon history-taking, the patient revealed that he got a fever with 39.1 °C as the highest temperature on
April 1, 2018. He received treatment at a local health clinic, where he was prescribed with cephalosporin
antibiotics on April 2, 2018. Then, the patient saw a doctor at Beijing Huairou District Hospital. A chest
computed tomography (CT) scan showed large consolidation of the lower lobe of the right lung and
superior lobe of the left lung on April 4, 2018. Arterial blood gas analysis revealed: pH 7.476, PCO2 22
mmHg, PO2 54.1 mmHg, and HCO3- 20.4 mmol/L. The fraction of inspiration O2 (FiO2) at the time of
taking arterial blood samples was 21%. The antigen of influenza A was positive. Then, he was
transferred to the general ICU of Beijing Ditan Hospital Affiliated Capital Medical University for further
treatment on April 5, 2018.
Guo HB et al. ECMO in a patient after Fontan operation
WJCC https://www.wjgnet.com 10616 October 16, 2022 Volume 10 Issue 29
History of past illness
At birth, the patient was diagnosed with atrial septal defect, ventricular septal defect, pulmonary artery
stenosis, and right ventricular double outlet, also known as Tausing-Bing syndrome. He received the
modified Fontan operation 13 years ago when he was 10 years old. In the surgery, the pulmonary orifice
was sutured, and the tricuspid valve was sewn closed, then the left auricle was connected with the
pulmonary artery to ensure stable cardiopulmonary circulation during the operation. He recovered well
and was able to perform general physical activity easily by himself after the operation. Three years ago,
echocardiography at the regular visit revealed a double outlet in the right ventricle. Moreover, it was
also observed that he showed right atrial enlargement and aortic valve regurgitation. The ejection
fraction of the young man was 50%.
Personal and family history
The patient had no previous or family history of similar illnesses.
Physical examination
The physical examination revealed the following: Temperature 38.4 °C, blood pressure 86/54 mmHg,
respiration rate 40 times/min, pulse rate 110 times/min, and SPO2 80%. The breath sounds of both lungs
were thick and moist rales can be easily heard. Arrhythmia and dropped-beat pulse were found in the
physical examination. Due to respiratory failure and septic shock, his skin was wet, cold, and bluish.
Laboratory examinations
Laboratory tests at the time of admission to the ICU were as follows: White blood cell count: 11.89 × 109
/L (reference range, 4-10 × 109/L); neutrophil percentage: 84.94% (reference range, 50%-70%);
hemoglobin: 173.10 g/L (reference range, 110-150 g/L); hematocrit: 48.70% (reference range, 35%-45%);
platelet count: 178.00 × 109/L (reference range, 100-300 × 109/L); sodium: 128.5 mmol/L (reference
range, 137-147 mmol/L); creatinine 279.6 µmol/L (reference range, 41-73 µmol/L); procalcitonin: 4.51
ng/mL (reference range, < 0.05 ng/mL); C-reactive protein: 212.9 mg/L (reference range, 0-5 ng/mL);
alanine aminotransferase 48.6 U/L (reference range, 7-40 U/L); aspartate aminotransferase: 100.4 U/L
(reference range, 13-35 U/L); total bilirubin: 26.2 µmol/L (reference range, 0-18.8 µmol/L); direct
bilirubin: 21.4 µmol/L (reference range, 0-6.8 µmol/L).
Imaging examinations
The chest X-ray of the patient on admission is shown in Figure 1A. The chest X-ray of the patient at the
first day of his receiving the veno-venous (VV) ECMO therapy is shown in Figure 1B.
MULTIDISCIPLINARY EXPERT CONSULTATION
Enhanced chest CT was performed on April 9, 2018 (Figure 2A and B), which revealed postoperative
changes of the heart and bilateral pneumonia changes. A small amount of bilateral pleural effusion was
also detected. The direction of blood flow is shown in Figure 2C. The chest X-ray of the patient at the
second day and ninth day of his receiving the veno-venous-arterial (VVA ECMO) therapy is,
respectively, shown in Figure 2D and E. The chest X-ray before the patient finished the VVA ECMO
therapy is shown in Figure 2F. The chest X-ray and lung CT images on May 24, 2018 are shown in
Figure 2G and H, respectively.
FINAL DIAGNOSIS
The patient was mainly diagnosed with type A influenza. Other diagnoses were pulmonary infection,
severe respiratory failure, acute kidney injury, acute hepatic injury, electrolyte disturbance, and atrial
fibrillation.
TREATMENT
Upon admission into the ICU, the patient’s APACHE II and Sequential Organ Failure Assessment scores
were, respectively, 22 and 10. A Venturi mask was used for supporting respiratory function, and the
oxygen flow volume was 15 L/min. The saturation of pulse oxygen of the patient was 80%. Meanwhile,
he had difficulty in breathing and breathed 40 breaths per minute. The arterial blood gas analysis before
mechanical ventilation indicated the following: pH 7.45, PCO2 30 mmHg, and PO2 48.2 mmHg. Due to
severe ARDS, the young patient was intubated on April 5, 2018, and the ventilator mode was
intermittent positive pressure ventilation. Other parameters were as follows: FiO2 100%, tidal volume
Guo HB et al. ECMO in a patient after Fontan operation
WJCC https://www.wjgnet.com 10617 October 16, 2022 Volume 10 Issue 29
Figure 1 Chest X-rays of the patient at admission and the first day of receiving veno-venous extracorporeal membrane oxygenation
therapy. A: Chest X-ray of the patient at admission to the intensive care unit; B: Chest X-ray of the patient at the first day of receiving veno-venous extracorporeal
membrane oxygenation therapy.
Figure 2 Follow-up images at two months after veno-venous extracorporeal membrane oxygenation therapy. A: Computed tomography (CT)
images of the lung on April 9, 2018; B: Chest enhanced CT images on April 9, 2018; a: The right atrium; b: The left atrium; c: The left ventricle; d: The right ventricle;
e: The thoracic aorta; f: The ascending aorta; g: The aortic arch; h: The pulmonary vein; i: The pulmonary artery; C: Diagram of Fontan circulation; D: Chest X-ray of
the patient at the second day of receiving veno-venous extracorporeal membrane oxygenation (VVA ECMO) therapy; E: Chest X-ray of the patient at the ninth day of
receiving VV-A ECMO therapy; F: Chest X-ray before the patient finished VV-A ECMO therapy; G: Chest X-ray on May 24, 2018; and H: Lung CT images on May 24,
2018.
560 mL, respiratory frequency 20 times/min, positive end-expiratory pressure = 10 cmH2O, and peak
airway pressure 23 cm H2O. Moxifloxacin hydrochloride and paramivir were used to combat infection
when the patient was admitted to the ICU. Anti-infective drugs were switched to cefoperazone-
sulbactam sodium and vancomycin hydrochloride on April 9, 2018. To control a fungal infection,
voriconazole was used on April 9, 2018.
Guo HB et al. ECMO in a patient after Fontan operation
WJCC https://www.wjgnet.com 10618 October 16, 2022 Volume 10 Issue 29
Due to the special heart structure after the modified Fontan operation, the central venous pressure
(CVP) of the young man was 40 mmHg on admission. As a result of septic shock, noradrenaline was
used to raise blood pressure. For severe AKI, hyperkalemia, and metabolic acidosis, continuous renal
replacement therapy was initiated when the patient was admitted to the ICU. Even if the protective lung
ventilation strategy and ventilation in prone position were properly conducted after mechanical
ventilation, his respiratory failure was persistent and did not significantly improve. The arterial blood
gas analysis revealed the following: pH 7.193, PCO2 48 mmHg, PO2 52 mmHg, base excess -10 mmol/L,
and lactate 1.34 mmol/L. VV ECMO was applied to correct the respiratory failure on April 6, 2018. The
two vein indwelling catheters were established, respectively, in the left femoral and right internal
jugular veins. The initial parameters of VV ECMO were as follows: Rotation speed 3100 turns/min,
blood flow volume 4.3 L/min, oxygen flow volume 4.5 L/min, and FiO2 100%.
As an attempt to ameliorate severe heart failure and cardiogenic shock, the VV ECMO procedure was
replaced by VVA ECMO on April 11, 2018. The patient’s right femoral artery was punctured and
intubated as the infusion tube, and combined deep venous catheters inserted in the left femoral vein and
right internal jugular vein were used as the drainage tube. The initial parameters of VVA ECMO were as
follows: Rotation speed 3800 turns/min, blood flow volume 4 L/min, oxygen flow volume 4 L/min,
and FiO2 100%. We tried to use negative liquid equilibrium to improve the left heart failure at the early
stage of VVA ECMO. The negative fluid balance at the first and second day was, respectively, 272 and
345 mL. As a result, the CVP of the young man decreased to 28 mmHg and his circulation tended to
deteriorate. In order to maintain his circulation, the dose of noradrenaline had to adjust from 0.7 to 1.4
ug/kg/min. Then we tried to change the liquid management strategy. The cumulative positive balance
was 10000 mL in the following 7 d. His CVP gradually increased to 35 mmHg and the dose of
noradrenaline was gradually tapered until stopped on April 20, 2018. The oxygenator and circulation
line of ECMO were replaced as the equipment had achieved its design life on April 23, 2018. As
ventilator weaning was difficult in a short period for the patient, tracheotomy was operated on April 27,
2018. As circulation and respiratory function gradually improved, VVA ECMO equipment was removed
on May 1, 2018.
OUTCOME AND FOLLOW-UP
The patient was successfully withdrawn from artificial ventilation on May 28, 2018 and then discharged
from hospital on May 30, 2018. We followed him at his home on October 25, 2021, and he can take care
of himself in daily life and engage in light manual labor.
DISCUSSION
As research reported in 2005, double outlet right ventricle (DORV) occurs in 0.09 cases per 1000 live
births. As a rare congenital heart disease, the Tausing-Bing anomaly is the third most common type of
DORV[5]. So far, the Fontan operation was the only treatment option to change the anatomy of the heart
and help improve patients’ hemodynamics[1]. After successful operation, patients typically recover the
ability to engage in general physical activity. H1N1 influenza has a higher case fatality among younger
patients and the potential for fulminant ARDS[6]. The mortality of patients diagnosed with ARDS
ranges from 17.3% to 41.4% among critically ill patients with H1N1 infection[2,3]. As a better ventilatory
strategy, as well as an alternative mode of respiratory support, ECMO provides gas exchange via an
extracorporeal circuit, and is increasingly being used to improve respiratory and circulatory function
[7]. ECMO is usually used to help patients get through postoperative difficulties such as heart failure
and hemodynamically unstable and refractory arrhythmias. As the morbidity and mortality associated
with ECMO are relatively high, the survival of children with heart disease that need ECMO support is
only 33%-60%[8].
By analyzing the medical history and imaging manifestations, we concluded the direction of blood
flow in this case (Figure 2C): Right atrium, pulmonary artery, pulmonary vein, left atrium, left ventricle,
right ventricle, aorta, and right atrium. By analyzing data from the Extracorporeal Life Support
Organization, we found that only 35% of cardiac failure patients subjected to Fontan operation survived
to hospital discharge[4]. By analyzing the medical history and imaging results of the patient, there were
mainly three factors causing the severe respiratory failure. First, due to the special physiological
structure after the operation of Fontan, a single ventricle was more vulnerable to suffering severe left
heart failure compared with a normal heart. Furthermore, pulmonary edema caused by the acute left
ventricular failure was one of the reasons for respiratory failure. Second, the pulmonary infection
caused by H1N1 influenza affected his respiratory function. Thus, severe ARDS might be the second
cause of severe respiratory failure. Third, pulmonary arterial hypertension might aggravate systemic
circulation congestion and lead to left ventricular preload insufficiency.
Guo HB et al. ECMO in a patient after Fontan operation
WJCC https://www.wjgnet.com 10619 October 16, 2022 Volume 10 Issue 29
The patient’s circulation after the Fontan surgery was different than that of a normal patient.
Therefore, supporting Fontan patients on ECMO carries high morbidity and mortality[4]. After the
Fontan operation, pulmonary and systemic circulation are mainly sustained by the single ventricle. A
study found that patients for whom the Fontan operation was not successful usually suffered anatomic
obstruction to flow, pulmonary vascular remodeling, atrioventricular valve dysfunction, univentricular
diastolic dysfunction and chronic underfilling, and/or univentricular systolic dysfunction[9]. There are
three stages of failure in a Fontan patient, each of which is associated with certain underlying etiologies
[10]. Early Fontan failure is often marked by anatomic obstruction. Most patients usually have an early
acute onset of failure, prior to end organ injury[11]. Patients with middle and late phase Fontan failure
usually exhibit signs of end organ damage. Late phase failure patients present protein losing
enteropathy, plastic bronchitis, cirrhosis, or renal failure in the process of medical treatment[12]. In this
case, after the Fontan operation, the right atrium of the patient was directly linked with the pulmonary
artery. The CVP of the patient at admission to the ICU was 40 mmHg, therefore he was diagnosed with
pulmonary hypertension. As his circulation depended on the single ventricle, severe sepsis accelerated
the process of heart failure. At the beginning of treatment, it was hard to improve the systemic and
pulmonary circulation congestion.
Relevant research noted that about 30% of Fontan patients suffered from heart failure in 20 years[13,
14]. Patients with neurologic complications, surgical bleeding, and renal failure were inclined to have a
higher mortality during the course of ECMO, indicating that ECMO complications may limit survival
outcomes for these patients[4]. The patient initially received VV ECMO treatment for severe respiratory
failure on April 6, 2018. Even though the man’s respiratory function significantly improved with the
help of VV ECMO, shock persisted and did not effectively relieve. By analyzing the pathophysiological
characteristics of his heart anatomical structure, we concluded that severe left heart failure might
explain the refractory shock. Then, in response to severe heart failure and cardiogenic shock, the VV
ECMO was converted to VVA ECMO. The shock was significantly improved with the help of VVA
ECMO. Vasoactive drugs were disused in 1 d after the mode of the machine was switched.
We tried to use negative liquid equilibrium to improve the left heart failure at the early stages of VVA
ECMO, but failed. The negative liquid equilibrium was smoothly conducted 3 d after the mode of
ECMO was changed. As pulmonary pressure of the case was high, the right ventricular ejection
depended on pressure differences and needed a higher volume. With the improvement of cardiac
function and oxygen, heart and respiratory failures were effectively improved. Meanwhile, pulmonary
arterial hypertension declined to some extent, making negative liquid equilibrium a feasible option. In
the case, the proper CVP might be 35 mmHg. We had to search a suitable liquid state to balance both the
respiratory and circulatory systems. Therefore, an individualized liquid management strategy was
necessary.
With the help of VVA ECMO, heart failure and shock were gradually improved. After about 20 d of
circulation support, the patient successfully had the VVA ECMO equipment removed. Due to the
special cardiac structure and circulatory characteristics, it might be better for him to choose an active
circulation support earlier. Other modes such as VAV ECMO might be another choice, especially for
those who have a risk of different hypoxia, in which cardiac recovery precedes lung recovery[15]. For
this case, VAV ECMO would provide the right atrium with oxygen-rich blood and improve coronary
oxygen supply, which might be better for heart recovery.
Different types of antibiotics were used throughout the course of the disease. The structural
abnormality of the heart and congestive heart failure increased pulmonary edema and aggravated
pulmonary infection. Meanwhile, longer duration of ECMO increased the risk of bloodstream infection.
Although the outcome in our research was favorable, it is important to note that the case took a longer
ECMO course to achieve lung recovery[16]. Most previous studies had demonstrated the use of ECMO
in patients with the Fontan operation for cardiac support, but this case illustrated its value as a bridge to
lung recovery in acute respiratory failure due to pH1N1 infection.
CONCLUSION
The circulation after the modified Fontan operation is different from that of patients who does not
undergo the operation. There are certainly many differences between them when they receive the
treatment of ECMO. Due to the special cardiac structure and circulatory characteristics, it might be
better for them to choose an active circulation support earlier.
FOOTNOTES
Author contributions: Liu JY and Hou XT designed the research study and provided research ideas; Guo Hb and Tan
JB were major contributors in writing the manuscript; Cui YC, Xiong HF, Li CS, Liu YF, Sun Y, Pu L, Xiang P, Zhang
M, Hao JJ, and Yin NN read the literature and collected the patient’s medical records; all authors read and approved
the final manuscript.
Guo HB et al. ECMO in a patient after Fontan operation
WJCC https://www.wjgnet.com 10620 October 16, 2022 Volume 10 Issue 29
Supported by the Capital Foundation of Medical Development, No. 2018-1-2171; and the Seedling Plan from the
Beijing Ditan Hospital, Capital Medical University, No. DTYM201802.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report
and any accompanying images.
Conflict-of-interest statement: All the authors report no relevant conflicts of interest for this article.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was
prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by
external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-
NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license
their derivative works on different terms, provided the original work is properly cited and the use is non-
commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Country/Territory of origin: China
ORCID number: He-Bing Guo 0000-0002-7353-7795; Jian-Bo Tan 0000-0002-0694-2555; Yong-Chao Cui 0000-0001-8968-
8961; Hao-Feng Xiong 0000-0002-9282-8694; Chuan-Sheng Li 0000-0003-3538-7295; Yu-Feng Liu 0000-0003-1710-0270;
Yao Sun 0000-0001-8169-9404; Lin Pu 0000-0002-5659-2848; Pan Xiang 0000-0001-8302-9770; Ming Zhang 0000-0003-3092-
918X; Jing-Jing Hao 0000-0001-9347-1351; Ning-Ning Yin 0000-0003-3359-2447; Xiao-Tong Hou 0000-0002-7047-0492;
Jing-Yuan Liu 0000-0002-0592-9789.
S-Editor: Xing YX
L-Editor: Wang TQ
P-Editor: Xing YX
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