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Recurrent Hydrothorax and Surgical Diaphragmatic Repair: Report of 2 Cases and Review of the Literature

Christine Argento, A. MD*; Kim, Anthony MD; Knauert-Brown, Melissa MD, PhD*; Boffa, Daniel MD; Siegel, Mark D. MD*; Jafari, Behrouz MD, PhD*; Puchalski, Jonathan T. MD, MEd*,‡

Journal of Bronchology & Interventional Pulmonology: April 2014 - Volume 21 - Issue 2 - p 150–153
doi: 10.1097/LBR.0000000000000047
Case Reports

Background: Pleural effusions may result from intra-abdominal processes and sometimes present with dramatic clinical consequences. We present 2 cases of recurrent hydrothorax requiring surgical repair of diaphragmatic defects and describe when surgery may be the best treatment modality.

Patient 1: A 63-year-old man with end-stage renal disease requiring peritoneal dialysis presented with dyspnea on exertion that progressed to cardiac arrest. He was found to have a tension hydrothorax that was initially stabilized with thoracentesis and tube thoracostomy. He eventually underwent surgical repair of fenestrations with complete resolution of his effusion.

Patient 2: A 52-year-old man with recurrent hydrothorax in the context of hepatitis C cirrhosis and hepatocellular carcinoma following radiofrequency ablation to his liver had recurrent admissions with dyspnea and a large pleural effusion. When medical therapy failed, he underwent surgical repair of a large diaphragmatic defect.

Conclusions: Hydrothorax related to peritoneal dialysis or cirrhosis may cause life-threatening scenarios in which medical management may stabilize the patient. Ultimately, surgical corrections of diaphragmatic defects may be necessary for definitive management in selected patients. Although these scenarios are rare, clinicians should be aware of these possibilities as early collaboration between medical and surgical services is essential for optimal patient care.

Divisions of *Pulmonary, Critical Care and Sleep Medicine

Thoracic Surgery

Thoracic Interventional Program, Yale University School of Medicine, New Haven, CT

Disclosure: There is no conflict of interest or other disclosures.

Reprints: A. Christine Argento, MD, Emory University, 550 Peachtree St, NE, 6th Floor, Atlanta, GA 30308 (e-mail:

Received May 21, 2013

Accepted January 20, 2014

Pleural effusions may result from intra-abdominal processes and sometimes present with dramatic clinical consequences. Hydrothorax is a rare but well-recognized complication of peritoneal dialysis (PD) that was described in 1967 by Edwards and Unger.1 Hepatic hydrothorax may occur as a result of cirrhosis. Both are characterized by recurrent transudative effusions and are commonly managed medically. We present 2 cases of recurrent hydrothorax requiring surgical repair of diaphragmatic defects and suggest when surgery may be the most appropriate treatment modality.

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Case 1: Peritoneal Dialysis–associated Tension Hydrothorax

A 63-year-old man with end-stage renal disease presented to the emergency department with dyspnea. During evaluation, he experienced a cardiac arrest believed to be related to tension physiology from a right-sided hydrothorax (Fig. 1A). Two liters of transudative pleural fluid were removed by emergent thoracentesis, providing immediate symptomatic relief, resolution of mediastinal shift and hemodynamic instability. Rapid reaccumulation of fluid led to chest tube placement with temporary relief. Resumption of PD led to an immediate increase in chest tube output. The fluid collected from the chest tube revealed a glucose level of 743 mg/dL (concurrent plasma glucose level 105 mg/dL, dialysate glucose level 1010 mg/dL), suggesting a pleuroperitoneal leak. The patient was transitioned to hemodialysis and the pleural effusion resolved. To allow resumption of PD due to symptomatic hypotension with hemodialysis, thoracoscopic exploration was performed. Two diaphragmatic fenestrations were identified and repaired (Figs. 1B, C). In addition, talc pleurodesis was performed. PD was resumed 6 weeks following the procedure without further reaccumulation of fluid in the pleural space.



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Case 2: A Large Diaphragmatic Defect With Recurrent Hepatic Hydrothorax

A 52-year-old man was evaluated for recurrent hydrothorax secondary to hepatitis C cirrhosis and hepatocellular carcinoma. He had undergone attempts at resection 3 years earlier and during the operation, radiofrequency ablation was performed on the left lobe of the liver. Ablation of the lesion on the right was unsuccessful because of the subcapsular location and technical limitations of heat generation in an area at risk for liver capsule violation.

Over a 2-month period, he underwent repeated right-sided thoracentesis, a chest tube with talc pleurodesis, and a tunneled pleural catheter, all without relief of his severe, recurrent dyspnea and effusion. Because of these failures, a surgical approach was attempted. During the surgery, a 10×5 cm diaphragmatic defect was discovered and sutured (Fig. 2). Pleurodesis was simultaneously performed. The patient had significant improvement at 1 month follow-up but ultimately died from his malignancy.



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Treatment for PD-associated Hydrothorax

PD was performed for a uremic patient in 1923 by George Ganter but it was not until the 1960s that this form of renal replacement therapy was integrated into routine medical care for chronic renal failure.2 Common adverse events associated with PD include peritonitis, leakage of abdominal fluid, and catheter malfunction.3 Hydrothorax is an uncommon complication that occurs in approximately 2% of patients.4 Very few cases of tension hydrothorax related to PD are reported in the literature.5,6 Hydrothorax from PD typically occurs within a few weeks following therapy initiation but has a reported time of onset ranging from 1 day to 8 years.4 Mechanisms leading to formation of hydrothorax include congenital or acquired diaphragmatic defects. Acquired defects are hypothesized to result from high intra-abdominal pressures generated by PD or impaired lymphatic drainage.1

Diaphragmatic defects related to renal disease may be evaluated by lactate, methylene blue administration, or technetium studies. Stereoisomeric forms of lactate can be evaluated in the pleural fluid aspirate. L-lactate is endogenously synthesized whereas D-lactate is used as a buffer in dialysis solutions. The presence of D-lactate in the pleural fluid therefore confirms the diagnosis of hydrothorax secondary to dialysate leak.7 Methylene blue can be administered into the dialysate, 90 minutes after which a thoracentesis to look for pleuroperitoneal leak can be performed. Similarly, Tc-99m-macroaggregated human albumin can be added to the dialysate and followed by peritoneal scintigraphy for migration into the pleural cavity.8

Direct visualization of the diaphragm by video-assisted thoracoscopic surgery (VATS) was first introduced for this patient population in 1996. This approach allows visualization of the entire parietal pleura and diaphragmatic surface with concurrent therapy for management of the pleuroperitoneal leak, including diaphragm repair and pleurodesis.7

Once a communication is confirmed, switching from PD to hemodialysis is usually effective. It has been shown that temporary discontinuation of PD for 2 to 6 weeks alone is associated with a 53% success rate of resuming long-term PD.4,7,9 Chemical pleurodesis by tube thoracostomy can be performed with sclerosing agents such as talc, tetracycline, fibrin glue, autologous blood, and a hemolytic streptococcal preparation OK-432. There is disagreement in the literature as to whether PD should be discontinued or simply interrupted for pleurodesis. Pleurodesis has a reported 58% success rate in allowing resumption of PD.9 VATS may offer therapy by suturing defects, with or without Teflon or tissue patches and pleurodesis. On the basis of several case series, the success rate of VATS is better than thoracostomy-attempted pleurodesis and is in excess of 90%.7

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Surgical repair in the setting of hepatic hydrothorax is typically preceded by maximizing medical therapy (diuretics, therapeutic thoracentesis). A transjugular intrahepatic portosystemic shunt is effective in 70% to 80% of patients. Liver transplant is the definitive surgical approach for hepatic hydrothorax, although direct surgical repair of diaphragmatic defects is occasionally possible. As described previously, sutures or other sealants may be used. Milanez de Campos and colleagues treated 18 patients with refractory hepatic hydrothorax. Five patients had diaphragmatic defects that were sutured. Of these, 3 were successful. One patient worsened and another died of pneumonia 18 days after surgery. Talc alone was successful in 43.7% of patients. However, the 3-month morbidity (57%) and mortality (39%) were high.10 Mouroux et al11 successfully treated 6 patients with visible diaphragmatic defects with 7- to 36-month follow-up. Luh and Chen12 reported a 67% success rate of thoracoscopic interventions beyond 3 months in a 12 patient case series. Huang et al13 reported that all 10 of their patients had sustained resolution of hepatic hydrothorax after a mean of 7.7 months status after pleural flap or onlay mesh reinforcement. The largest series of patients reporting treatment with VATS is from Japan, whereby 29 patients were studied. The success rate was 72% overall but was 89% when a diaphragmatic defect was observed and only 38% when no defect could be isolated.14 A large study of VATS for recurrent hydrothorax has not been conducted and, as such, decisions for surgery in this patient population are individualized.

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Hydrothorax related to PD or cirrhosis may cause life-threatening scenarios. In our first case, tension physiology was evident. In the second, a massive diaphragmatic defect was discovered intraoperatively after less invasive approaches failed. Although both tension physiology and massive diaphragmatic defects are rare, they are important entities to recognize as minimally invasive strategies may not suffice and surgery may ultimately be required.

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hepatic hydrothorax; pleuroperitoneal leak; continuous ambulatory peritoneal dialysis (CAPD)

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