Hydatid disease is considered a remarkable health problem worldwide, mostly endemic in developing and underdeveloped countries such as the Middle East and Africa. It is caused by the zoonotic parasite Echinococcus granulosus [1] presenting as cysts most commonly in the liver (50–80%) and lungs (10–40%) [2]. Several complications of hydatid disease of the liver may develop, one of which is formation of a hepato-pleural fistula. Hepato-pleural fistula is considered one of the reported complications and includes the formation of a fistula between the liver parenchyma and the pleural space [3]. Surgery remains the mainstay of treatment of hydatid disease, especially in established cases of hepato-pleural fistula [4]. It has been traditionally done through a trans-thoracic approach in addition to an abdominal access. However, it was associated with high morbidity and mortality especially in developing countries that have modest expertise in this field [1]. Recently, less invasive surgical approaches have been advocated for management of hepato-pleural fistula [5]. Here, we report successful transabdominal transdiaphragmatic laparoscopic approach to manage liver hydatid cyst with hepato-pleural fistula yielding satisfactory outcomes.

We report a case of a 51-year-old female housewife who was referred from an outside physician to our clinic at the American University of Beirut Medical Center for intermittent episodes of dyspnea and nonproductive cough of two months duration. The patient reported no fever, chills, night sweats, weight loss, chest pain, palpitations, or hemoptysis. She also denied any abdominal pain, change in bowel habits, dysphagia, vomiting, jaundice, or tea colored urine/clay colored stools. The patient was also known to have hypothyroidism and diabetes mellitus type II and was a former smoker. She did not have any past surgical history. To mention, this patient was an inhabitant of a rural area close to a farm in Lebanon.

On presentation, the patient was awake, alert, and oriented to person, place, and time. Physical exam revealed stable vital signs with decreased breathing sounds on the right lower lung field with no wheezing. Her abdomen was soft, non-distended with no rebound tenderness or any peritoneal signs, and murphy sign was negative. Baseline investigation was unremarkable with normal complete blood count levels except for mild eosinophilia (5%), normal basic metabolic panel, liver function tests, and coagulation profile. A plain antero-posterior chest radiogram (CXR) was performed showing blunting at the right costophrenic angle (Figure 1). Further evaluation with a computed tomography (CT) scan of the chest and abdomen/pelvis was done. Computed tomography revealed 2 large cystic lesions (measuring 7 × 8 cm and 10 × 7.5 cm) in the right lobe of the liver, communicating, and containing several irregularly shaped daughter cysts within the mother cyst, with no calcifications. Communication with the pleural space was noted with a right lower lobe consolidation. A suspicion of hydatid cyst was confirmed by a hydatid indirect hemagglutination test with a value >1:32768.

The patient was started on albendazole and was scheduled for laparoscopic evacuation of liver hydatid cyst with transabdominal transdiaphragmatic laparoscopic approach of the hepato-pleural fistula.

Intraoperatively, general abdominal laparoscopy was performed through 10 mm umbilical port. Respectively, a 5 mm trocar was inserted in the epigastrium, 12 mm trocar in the mid right sub-costal area (retrieval bag and scope if needed) and a 5 mm trocar in the lateral right subcostal area (Figure 2). During inspection, two large hydatid peri-cysts were identified arising from the hepatic dome adhering to the supra-hepatic diaphragm. The cyst contents were managed laparoscopically via closed circuit of tubing system by aspirating 100 cc of clear intra-cystic fluid and replaced by similar volume of hypertonic saline as a scolicidal agent. Using electrocautery, a small opening was done in the wall of the cyst that allowed introduction of the suction where the cyst was aspirated till dryness and repeatedly irrigated with hypertonic saline (Figure 3). The peri-cysts were divided completely from the diaphragmatic surfaces. This allowed management of the hepatic hydatid by excising the fibrotic component of the peri-cysts, retrieving the membranes and thoroughly inspecting the potential existence of bile leak or cysto-biliary fistula (Figure 4). Then, the intra-thoracic component of the hydatid was managed via the diaphragmatic communication through the same abdominal approach, where the membranes were retrieved, and cyst cavity was irrigated and washed. There was definite pleural communication and erosion to the lower surface of the lower right pulmonary lobe with tiny air leak but no communication to major bronchioles as it was identified by air water tests. And an intra-abdominal drain was inserted to drain both the chest and hepatic cavities (Figure 5). There was no need for right chest tube insertion, since the lower surface of the diaphragm was inseparable from the lung due to the inflammatory process.

Postoperatively, the patient was doing well. Bilious drainage was noted on postop day 2, and a biliary leak was suspected. Endoscopic retrograde cholangiopancreatography (ERCP) with sphincterotomy was performed to decrease biliary pressure, and a 10 Fr × 10 cm plastic stent was inserted into the biliary tree. Drain output was followed up. At postoperative day 8, the patient was doing well, and she was discharged home on albendazole, and the drain was removed.

Hydatid disease is endemic in agricultural and animal raising societies especially in the Middle East. This disease carries significant burden on public health and economy [6]. For all cysts that are larger than 15 cm, those that are complicated or those that are not suitable for interventional radiology (IR), surgical approach is the mainstay treatment [6].

One of the major complications of the hydatid cyst of the liver is its rupture into the pleura and bronchial tree. This happens either due to the pressure from the expanding hydatid cyst leading to erosion into the diaphragm or from the destructive effect of a superimposed infection [7]. The most prominent sign is bilioptysis. However, often patients may have nonspecific respiratory symptoms for weeks or months, as the case of our patient. In the past years, different invasive surgical approaches have been used for hydatid cysts with a pneumo-biliary fistula. They comprise methods of gaining access either through the abdomen or through the lung, or both [8],[9],[10]. Transthoracic approach is the surgical method of choice via posterolateral thoracotomy; however, sometimes thoraco-laparotomy or thoracophreno-laparotomy is performed. On the other hand, reports of minimally invasive approaches have been published like performing ERCP with micro-coils and ERCP with fibrin sealing [11],[12].

Indeed, advancement of laparoscopic surgery including robotic surgeries provides a technical basis for minimally invasive surgery for hydatid cysts with less morbidity, rapid recovery, and a shorter hospital stay compared to conventional open procedures [13],[14]. There are no reported case reports in the literature for the management of hepato-pleural fistula through a laparoscopic approach [15],[16]. Here, we introduce a successful transabdominal laparoscopic approach for the management of hepato-pleural fistula.

It is crucial to highlight that the understanding of the pathophysiology of the disease allows for the feasibility of our presented surgical approach. This is due to the fact that when the hydatid cyst in liver erodes through the diaphragm reaching the lung and its pleura, it forms a fibrotic tract. This allows surgeons to access the lung through the abdomen, passing through the fibrotic tract to reach the lung component of the hydatid cyst. This technique offers several advantages. First, there is no need for posterolateral thoracotomy which means that the approach avoids the high risk of post-operative complications related to thoracic surgery [17]. Second, the use of laparoscopy without the need for laparotomy or thoracotomy will allow reduced length of incision and minimal abdominal adhesions. Moreover, during the surgery, the magnified view of laparoscopy and the use of trocars will reduce the complications such as bleeding and bile leakage, and it will maximize the preservation of normal liver tissue. Furthermore, patients can resume diet earlier after surgery and have a shorter hospital stay [13].

This innovative surgical approach toward hepatopulmonary fistula falls in favor of the surgeon and the patient because it turns a complicated cumbersome surgery into a simple and favorable one. It also paves the way for minimally invasive surgeries to manage complicated hydatid disease with a lower mortality and morbidity, faster recovery, and shorter hospital stay.