We present the case of a 51-year-old male admitted to the department of thoracic surgery for respiratory distress and suspicion of right pneumothorax. Chest and abdomen CT scan showed an abundance of cysts located in the peritoneum, liver, mediastinum and both lungs, with findings suggestive for systemic infection with Echinococcus granulosus, as well as extensive bilateral pulmonary emphysema and right total pneumothorax. Respiratory distress was managed by right pleural drainage without significant improvement. Right pulmonary cysts were managed by capitonnage through lateral thoracotomy. In order to resolve the left pulmonary cysts, we decided to use extracorporeal membrane oxygenation instead of one lung ventilation due to the patient’s worsening condition. Veno-venous extracorporeal membrane oxygenation made possible the closure of two large cysts by capitonnage, while five smaller cysts were removed by wedge resection. The procedure was well tolerated by the patient, and on the fourth day following the procedure, extracorporeal membrane oxygenation was ceased and the patient started breathing independently.
The pulmonary infection with Echinococcus granulosus (EG) continues to be endemic in Romania, given its large rural population involved in sheep herding, with an inci-dence of 1:10,000 and a mortality of 2-4%(1). Although in most cases the treatment is straightforward and consists in systemic chemotherapy (albendazole) and the surgical removal of the cysts membrane through various techniques depending on the involved organ, in rare cases presenting simultaneous multivisceral echinococcosis we have to con-sider extreme measures.
Extracorporeal membrane oxygenation (ECMO) as heart-lung bypass support is now routinely used in Intensive Care Units (ICU) for the management of pulmonary infec-tions, severe acute respiratory distress syndrome (ARDS) or end-stage lung diseases requiring lung transplantation(2), but evidence supporting the utility of ECMO in pulmonary infections with Echinococcus granulosus is close to non-existent.
We present the case of a 51-year-old male admitted to our clinic for respiratory distress and the suspicion of right pneumothorax. The patient had lived all his life in Argeș county and worked as a shepherd at the time of presenta-tion, he was a smoker (60 packs-year), he consumed large quantities of alcohol and up to this moment his medical history was unremarkable.
The patient was examined in our emergency room, at which time he presented dyspnea, dry cough, enlarged liver and peripheral oxygen saturation of 88%, which corrected to 92% under oxygen 3 liters/minute. The initial chest X-ray showed right-sided pneumothorax and multiple opacities of different sizes, located on the left pulmonary field. Following a difficult anamnesis, we discovered that the patient had vomited a clear liquid a day before and coughed up white fragments of soft tissue.
Detailed full body CT scan showed an abundance of cysts located in the peritoneum, liver, mediastinum and both lungs, some intact, while others without content, find-ings which were suggestive for a systemic infection with Echinococcus granulosus in the context of the patient’s symp-tomatology and anamnesis. Extensive left pulmonary emphysema and right total pneumothorax were also visible during the scan (Figure 1).
The patient’s condition quickly deteriorated, with hypoxia present, which forced us to perform right pleural drainage with a 28 Fr catheter in order to resolve the res-piratory distress. Despite chest drainage, the patient’s con-dition remained unstable due to the poor state of the pulmonary parenchyma (important bilateral emphysema) and massive air leakage.
Following the discussion of the case by a medical council comprising of pulmonologists, thoracic surgeons, general surgeons, anesthesiologists and infectious phyisicians, a conclusion was reached that the treatment alone with albendazole and antibiotics will prove ineffective and restoring compromised pulmonary function by removing the cyst and suturing the air leaks were critical.
The first step was to remove the right pulmonary cysts and resolve the persistent air leaks in an emphysematous lung by muscle sparing right lateral thoracotomy. The pro-cedure was successful in resolving the cysts, but the patient could not withstand one lung ventilation (OLV) during the operation and forced us to alternate periods of apnea with periods of ventilation, which made suturing the air leaks a challenge. Pulmonary lobectomy was not an option given the patients critical state. Following surgery, the patient continued to present air leakage and still required mechani-cal ventilation (Figure 2).
In order to resolve the cysts on the left side in a patient who could not withstand another procedure under OLV due to the poor state of the pulmonary paren-chyma, we decided to attempt using ECMO as bypass for the pulmonary function.
With the patient positioned in dorsal recumbent posi-tion, a central venous line and an arterial line in place, we opted for veno-venous ECMO by inserting a 21 Fr arterial cannula in the right internal jugular vein (IJV) for the flow and a 23 Fr venous cannula in the right femoral vein (FV) for recovery. The cannulas were insert-ed without ultrasound guidance for a maximum length of 30 cm in the IJV, in order to reach the right atrium, and 60 cm in the FV to position below the hepatic veins. Given the patient’s weight of 65 kg and his height of 175 cm, we decided on a flow of 3000 mL/min, which was monitored for 10 minutes before ceasing mechanical ventilation (Figure 3).
Ten minutes later, the patient was repositioned in lateral recumbent position, the mechanical ventilation was ceased and left muscle sparing lateral thoracotomy,
in the fifth intercostal, was performed. Upon entry in the pleural cavity, the lung was manually deflated before careful inspection. After removing a part of the thick visceral pleura, two large cysts of the inferior left lobe were discovered and closed by capitonnage after removal of the membranes (Figure 4). Five cysts of the superior left lobe were also discovered and removed by wedge resection, given their small size and disposition. The procedure lasting 155 minutes was well tolerated by the patient and the ECMO flow was unaffected by the lateral decubitus position.
After surgery, the patient remained sedated, intu-bated, but not ventilated, and under ECMO support for three days. From the third day, the patient was con-scious, able to breath on his own, with minor air leakage present bilaterally, and ECMO support was turned down to a minimal 200 mL/min flow every four hours for peri-ods lasting 10-15 minutes in order to ease the cessation from ECMO. On the fourth day following the procedure, ECMO was ceased definitely and the patient was able to breath on his own (Figure 5).
Although lung transplantation remains the main indi-cation for ECMO in thoracic surgery, recently its use has also been reported for the surgery of tracheal tumors, tracheal or bronchial fistulas and pulmonary resections in trauma patients(3).
In pneumology, the main indication for ECMO remains the treatment of ARDS, especially in infections with influ-enza or in pneumococcal pneumonia, but we haven’t found any reports recommending ECMO for treating the com-plications of echinococcosis(4).
We did find a case in which ECMO was used to treat ARDS in a patient with a ruptured pulmonary cyst and sepsis(5), but our case is the first operated successfully under ECMO during which multiple wedge resections were performed and cavities were repaired by capitonnage.
Our rationale for using ECMO was to be able to perform the procedure in a patient under respiratory distress, not-withstanding OLV, while at the same time refrain from using mechanical ventilation, which was partially responsible for the persistent air leaks, and to allow for natural healing of the smaller pulmonary fistulas. Currently, after the patient’s pulmonary function has stabilized, ECMO is ceased gradu-ally, while mechanical ventilation is reintroduced. This was not an option for us since mechanical ventilation promoted air leakage, so we were forced to cease ECMO at a later stage.
Still, several questions were raised before and after the procedure, such as: does ECMO increase the risk of liver cysts rupture and parasite dissemination, does the use of ECMO contribute to faster closing of the air leaks, and is ECMO indicated in multivisceral advanced disease, with poor outcome, given its high costs?
Although fellow practitioners could argue that this case is an “end-stage” in the sense that the disease is bor-derline untreatable and the patient presented important bilateral emphysema, we cannot disconsider the patient’s right to medical treatment and it is the authors’ opinion that ECMO was the patient’s only option for surgical treatment.
We acknowledge the assistance provided by fellow thoracic consultant Mrs. Olga Dănăilă and thoracic surgery trainee Mrs. Elena Vasiliu during both surgical procedures and after-wards. We acknowledge the assistance provided by Mr. Mihai Ene and we wish to thank him for his guidance and expertise in setting up and running our ECMO device.
- Piccoli, L. et al. Molecular characterization of Echinococcus granulosus in south-eastern Romania: evidence of G1–G3 and G6–G10 complexes in humans. Clinical Microbiology and Infection. 2013; 19(6):578-582.
- Makdisi G, Wang I. Extra Corporeal Membrane Oxygenation (ECMO) review of a lifesaving technology. Journal of Thoracic Disease. 2015; 7(7):E166-E176. doi:10.3978/j.issn.2072-1439.2015.07.17.
- Rinieri P, Peillon C, Bessou JP, et al. National review of use of extracorporeal membrane oxygenation as respiratory support in thoracic surgery excluding lung transplantation. European Journal of Cardiothoracic Surgery. 2015; 47:87-94.
- Alves C, Chen JT, Patel N, Abrams D, Figueiredo P, Santos L, et al. Extracorporeal membrane oxygenation for refractory acute respiratory distress syndrome in severe malaria. Malaria Journal. 2013; 12:306.
- Sören LB. et al. Extracorporeal membrane oxygenation (ECMO) as salvage treatment for pulmonary Echinococcus granulosus infection with acute cyst rupture. International Journal of Infectious Diseases. 2017; 64:47-49.