Discussion:
GPF is a rare, pathological communication between the stomach and the pleural cavity 1. It was first described by Markowitz and Herter in 1960 as a complication of hiatal hernia rupture3. Since then, it has been reported secondary to trauma, peptic ulcer disease, malignancy (lymphoma and gastric tumors), and due to the spread of infection from adjacent structures such as subdiaphragmatic abscess, empyema, or invasive fungal infection in immunocompromised patients. Recently, with the emerging of obesity related bariatric surgeries, GPF has been documented as a complication of such surgeries in adult patients4. One of those surgeries is SG which has been utilized in pediatric patients to a lesser extent 2. Our patient had SG which was unfortunately complicated by GPF.
GPF has a latency in presentation, presenting months to years after the SG 1. The symptoms range from chronic cough to recurrent respiratory infections, both of which were seen in our case1. Other respiratory symptoms include chest pain and hemoptysis 5. Patients might exhibit gastrointestinal symptoms such as hematemesis or abdominal pain 16. GPF can be complicated by sepsis, hydropneumothorax, or tension pneumothorax. Chest tube drainage containing food particles or bile has also been reported 4.
Diagnosis of GPF requires a high index of suspicion and thorough investigations 4. Initial workup includes CXR, which may demonstrate pneumothorax, pleural effusion, or hydropneumothorax4. Upper GI series, preferably with water‐soluble contrast, might show contrast extravasation to the pleural cavity6. CT with oral contrast may reveal contrast leakage into the pleural space or delineate the fistula tract4. Moreover, upper GI endoscopy may identify the site of the fistula and can be utilized for therapeutic purposes7. Occasionally, GPF is diagnosed intraoperatively8.
There are no standardized treatment protocols for GPF7. Initial steps of management consist of treating the GPF complications and providing an alternative to oral feeding (parenteral nutrition or jejunostomy feeding tube)7,5. Meanwhile, the closure of the fistula can be done endoscopically or surgically. Various endoscopic interventions have been used including endoscopic clips<sup>9</sup>, stents, suturing, and fistula plug application 7,9,10. Our patient was managed conservatively and the fistula was closed endoscopically with an OTSC.
OTSC is a relatively new endoscopic intervention that has been used for chronic fistulas with variable success rate, ranging from 30% to 100%, and a low complication rate. Other indications for OTSC include gastrointestinal hemorrhage and perforation 11. Our patient failed surgical management, (i.e. trial of fistula closure laparoscopically). Other surgical options include partial gastric resection, diaphragmatic repair, and washout and drainage of the chest4. Roux-en-Y esophagojejunostomy for chronic GPF has also been used for chronic fistula 5.
In conclusion, pediatric GPF has been reported due to various causes (table1). Our patient is the first report of pediatric GPF secondary to SG. SG is not without its complications and it must be reserved for morbidly obese patients who fail conservative management. In our case, endoscopic intervention with an OTSC had better results when compared to the invasive surgical closure.
References:
1. Alghanim Fahad , Alkhaibary Ali AA and AA. Had Be j. Case Rep Surg. 2018;2018.
2. Alqahtani A, Alamri H, Elahmedi M, Mohammed R. Laparoscopic sleeve gastrectomy in adult and pediatric obese patients: A comparative study. Surg Endosc. 2012;26(11):3094–3100. doi:10.1007/s00464-012-2345-x
3. Markowitz AM, Herter FP. Gastro-pleural fistula as a complication of esophageal hiatal hernia. Ann Surg. 1960;152:129–134. doi:10.1097/00000658-196007000-00018
4. Armstrong RL, Heyse PB. Gastropleural fistula in metastatic ovarian cancer. J Surg Case Rep. 2014;2014(5):rju033–rju033. doi:10.1093/jscr/rju033
5. Garcia-Quintero P, Hernandez-Murcia C, Romero R, Derosimo J, Gonzalez A. Gastropleural fistula after bariatric surgery: a report of two cases. J Robot Surg. 2015;9(2):163–166. doi:10.1007/s11701-015-0505-4
6. Tzeng JJ, Lai KH, Lo GH, Hsu JH, Mok KT. Gastropleural fistula caused by incarcerated diaphragmatic herniation of the stomach. Gastrointest Endosc . 2001;53(3):382–384. doi:10.1067/mge.2001.111563
7. Ghanem OM, Abu Dayyeh BK, Kellogg TA. Management of Gastropleural Fistula after Revisional Bariatric Surgery: A Hybrid Laparoendoscopic Approach. Obes Surg. 2017;27(10):2773–2777. doi:10.1007/s11695-017-2836-x
8. Lakshminarayanan B, Morgan RD, Platt K, Lakhoo K. A leak too far - Gastro-pleural fistula mimicking recurrence of repaired congenital diaphragmatic hernia following fundoplication. J Radiol Case Rep. 2013;7(9):33–38. doi:10.3941/jrcr.v7i9.1505
9. Mendoza Ladd A, Al-Bayati I, Shah P, Haber G. Endoscopic closure of a gastropleural fistula. Endoscopy. 2015;47:E131–E132. doi:10.1055/s-0034-1391357
10. Andrawes S, El Douaihy Y. Using the endoscopic overstitching device and fully covered esophageal stents for closure of a gastropleural fistula and repair of a deformed gastric sleeve. VideoGIE. 2017;2(5):98–99. http://dx.doi.org/10.1016/j.vgie.2017.02.003. doi:10.1016/j.vgie.2017.02.003
11. Weiland T, Rohrer S, Schmidt A, Wedi E, Bauerfeind P, Caca K, Khashab MA, Hochberger J, Baur F, Gottwald T, et al. Efficacy of the OTSC System in the treatment of GI bleeding and wall defects: a PMCF meta-analysis. Minim Invasive Ther Allied Technol. 2019. doi:10.1080/13645706.2019.1590418
12. Prasertsan P, Anuntaseree W, Ruangnapa K, Saelim K. Gastropleural fistula masquerading as chylothorax in a child with lymphoma. BMJ Case Rep. 2019;12(7):10–13. doi:10.1136/bcr-2018-228987
13. Wuthisuthimethawee P, Sangkhathat S, Ruegklinag C, Patrapinyokul S, Laoprasopwathana K. Gastropleural fistula following a splenectomy for splenic abscess: A case report. J Med Assoc Thai. 2008;91(8):1291–1295.
14. Arun S, Lodha R, Sharma R, Agarwala S, Kabra SK. Gastropleural fistula as a complication of empyema thoracis. Indian J Pediatr. 2007;74(3):301–303. doi:10.1007/s12098-007-0051-9
15. Malik S, Giacoia GP. Candida Tropicalis Empyema Associated with Acquired Gastropleural Fistula in a Newborn Infant. Am J Perinatol. 1989;6(3):347–348. doi:10.1055/s-2007-999611