Cholethorax: An unusual delayed complication of recurrent cholangitis

Cholethorax: An unusual delayed complication of recurrent cholangitis

420 Letters to the Editor / Med Clin (Barc). 2016;146(9):418–421 References 1. Kloos RT, Eng C, Evans DB, Francis GL, Gagel RT, Gharib H, et al. Med...

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Letters to the Editor / Med Clin (Barc). 2016;146(9):418–421

References 1. Kloos RT, Eng C, Evans DB, Francis GL, Gagel RT, Gharib H, et al. Medullary thyroid cancer: management guidelines of the American Thyroid Association. Thyroid. 2009;19:565–612. 2. Schlumberger M, Carlomagno F, Baudin E, Bidart JM, Santoro M. New therapeutic approaches to treat medullary thyroid carcinoma. Nat Clin Pract Endocrinol Metab. 2008;4:22–32. 3. Barbosa SL, Rodien P, Leboulleux S, Niccoli-Sire P, Kraimps JL, Caron P, et al. Ectopic adrenocorticotropic hormone-syndrome in medullary carcinoma of the thyroid: a retrospective analysis and review of the literature. Thyroid. 2005;15:618–23. 4. Isidori AM, Kaltsas GA, Pozza C, Frajese V, Newell-Price J, Reznek RH, et al. The ectopic adrenocorticotropin syndrome: clinical features, diagnosis, management, and long-term follow-up. J Clin Endocrinol Metab. 2006;91:371–7. 5. Ilias I, Torpy DJ, Pacak K, Mullen N, Wesley RA, Nieman LK. Cushing’s syndrome due to ectopic corticotropin secretion: Twenty years’ experience at the National Institutes of Health. J Clin Endocrinol Metab. 2005;90:4955–62. 6. Nieman LK, Biller BMK, Findling JW, Newell-Price J, Savage MO, Stewart PM, et al. The diagnosis of Cushing’s syndrome: an Endocrine Society clinical practice guideline. J Clin Endocrinol Metab. 2008;93:1526–40.

Cholethorax: An unusual delayed complication of recurrent cholangitis夽 Biliotórax: una complicación tardía poco frecuente de la colangitis recurrente Dear Editor, Cholethorax is a very rare entity and is defined as a bilious pleural effusion. It is a rare complication of biliary and liver diseases, and can occur spontaneously in some hepatobiliary infections or, more frequently, secondary to some type of surgical manipulation or invasive percutaneous procedure.1,2 There are also forms of cholethorax without fistulas or macroscopic defects allowing bile flow from abdomen to pleural space.3 We report a case of cholethorax in a 92-year-old patient with a history of multiple biliary colic and recurrent calculous cholecystitis and cholangitis, which eventually required a laparoscopic cholecystectomy, performed 2 years earlier. A year after cholecystectomy, the patient reported a new episode of cholangitis secondary to a residual choledocholithiasis, which required a papillotomy performed with endoscopic retrograde cholangiopancreatography (ERCP). The patient was admitted to our center with symptoms of fever and pain in the right flank. The blood test data showed infection (neutrophilic leukocytosis, increased C-reactive protein), cholestasis (elevated bilirubin) and liver damage (elevated liver enzymes). Abdominal ultrasound showed aerobilia (without bile duct dilatation) and a hypoechoic lesion in segment 8 of the liver followed by a small right pleural effusion. A CT scan of chest and abdomen confirmed an abscess in the posterior segments of the right hepatic lobe communicated with the pleural space through the bare area of the liver and right hemidiaphragm. Given these findings it was decided to perform percutaneous drainage under radiological control of the loculated right pleural effusion, obtaining greenish fluid material. The pleural fluid analysis showed inflammatory cells and a high level of total bilirubin and higher than the

夽 Please cite this article as: Gorospe Sarasúa L, Ayala-Carbonero AM, FernándezMéndez MÁ, González-García A. Biliotórax: una complicación tardía poco frecuente de la colangitis recurrente. Med Clin (Barc). 2016;146:420–421.

7. Salgado LR, Fragoso MC, Knoepfelmacher M, Machado MC, Domenice S, Pereira MA, et al. Ectopic ACTH syndrome: our experience with 25 cases. Eur J Endocrinol. 2006;155:725–33. 8. Aniszewski JP, Young WF Jr, Thompson GB, Grant CS, van Heerden JA. Cushing syndrome due to ectopic adrenocorticotropic hormone secretion. World J Surg. 2001;25:934–40.

Jessica Ares ∗ , Lucía Díaz-Naya, Alicia Martín-Nieto, Joaquín Pertierra

Departamento de Endocrinología y Nutrición, Hospital Universitario Central de Asturias, Oviedo, Asturias, Spain ∗ Corresponding author. E-mail address: [email protected] (J. Ares).

level of serum bilirubin. The patient experienced a rapid clinical and radiological improvement after pleural drainage and administration of intravenous antibiotic treatment. Most of cholethorax have been described in the past associated with hepatic hydatid cysts, but today most cases are secondary to procedural percutaneous complications.4 These procedures include percutaneous transhepatic biliary drainage, percutaneous cholecystectomies, or radiofrequency of focal liver lesions. In our case, we believe that repeated clinical history of complicated biliary symptomatology, and especially cholangitis secondary to a residual choledocholithiasis (requiring sphincterotomy with ERCP) one year after cholecystectomy, predisposed the patient to an ascending cholangitis and development of a liver abscess. The appearance of deferred liver abscesses has been described as a late complication in patients with cholangitis or after cholecystectomy or ERCP.5 In our case, this liver abscess was communicated with the pleural space through the bare area of the liver (an area at the posterosuperior surface of the liver without peritoneal cover) and a small defect in the right hemidiaphragm, verified by imaging techniques. Small posterior diaphragmatic defects discovered in adults can be congenital or, more commonly, acquired. The latter are usually secondary to high energy blunt trauma or excessive strain of the diaphragm during procedures such as laparoscopy or in patients with tense ascites.6 To our knowledge, there are no cases reported in the scientific literature of spontaneous biliopleural communication (without percutaneous pleural biopsy) as a late complication of recurrent cholangitis.

References 1. Caporale A, Giuliani A, Teneriello F, della Casa U, Aurello P, Iaricci P, et al. Hydatid hepatothoracic fistulas. A report of 30 cases. Ital J Surg Sci. 1987;17: 327–33. 2. Lee MT, Hsi SC, Hu P, Liu KY. Biliopleural fistula: a rare complication of percutaneous transhepatic gallbladder drainage. World J Gastroenterol. 2007;13:3268–70. 3. Córdoba López A, Monterrubio Villar J, Bueno Álvarez-Arenas I. Biliothorax unrelated to fistula: a rare complication in gallbladder disease. Arch Bronconeumol. 2008;44:396–7. 4. De Meester X, Vanbeckevoort D, Aerts R, van Steenbergen W. Biliopleural fistula as a late complication of percutaneous transhepatic cholangioscopy. Endoscopy. 2005;37:183.

Letters to the Editor / Med Clin (Barc). 2016;146(9):418–421

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5. Van Brunt PH, Lanzafame RJ. Subhepatic inflammatory mass after laparoscopic cholecystectomy. A delayed complication of spilled gallstones. Arch Surg. 1994;129:882–3. 6. Boyce S, Burgul R, Pepin F, Shearer C. Late presentation of a diaphragmatic hernia following laparoscopic gastric banding. Obes Surg. 2008;18:1502–4.

a Servicio de Radiodiagnóstico, Hospital Universitario Ramón y Cajal, Madrid, Spain b Servicio de Medicina Interna, Hospital Universitario Ramón y Cajal, Madrid, Spain

Luis Gorospe Sarasúa a,∗ , Ana María Ayala-Carbonero a , María Ángeles Fernández-Méndez a , Andrés González-García b

∗ Corresponding author. E-mail address: [email protected] (L. Gorospe Sarasúa).