Scientific Letter
Cholecystocutaneous Fistula as a First Sign of Presentation of a Gallbladder Adenocarcinoma
Fístula colecistocutánea como forma de presentación del adenocarcinoma de vesícula biliar
Paúl Ugalde Serrano
, Lorena Solar García, Alberto Miyar de León, Ignacio González-Pinto Arrillaga, Juan González González
Corresponding author
Servicio de Cirugía General y del Aparato Digestivo, Hospital Universitario Central de Asturias, Oviedo, Spain
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We present the case of a cholecysto-cutaneous fistula as a form of presentation of gallbladder adenocarcinoma.</p><p id="par0010" class="elsevierStylePara elsevierViewall">The patient was an 83-year-old male with no prior history except for a previous episode of acute cholecystitis 2 years earlier, which had been treated conservatively with antibiotics and fluid therapy at another hospital. He came to the Emergency Department of our hospital complaining of abdominal pain in the right upper quadrant associated with the oozing of hematic-purulent content through an orifice in the abdominal wall located in the right hypochondrium. Abdominal examination revealed a mass in this region. The patient presented important anemia in the blood work-up (Hb 6<span class="elsevierStyleHsp" style=""></span>g/dl), although he was hemodynamically stable. Abdominal CT demonstrated a collection measuring 98<span class="elsevierStyleHsp" style=""></span>mm<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>78<span class="elsevierStyleHsp" style=""></span>mm<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>90<span class="elsevierStyleHsp" style=""></span>mm that was located predominantly in the extraperitoneal region, with hematic content in its interior, extending from the gallbladder bed to the subcutaneous tissue that fistulized through the skin (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>). A right subcostal laparotomy was performed; a large hematoma situated in the gallbladder bed was evacuated, the gallbladder and the fistulous tract from the gallbladder fundus to the abdominal wall were removed. The cutaneous fistula orifice was excised with the associated cholecystectomy. The pathology study reported moderately differentiated papillary adenocarcinoma, with infiltration of the cystic duct and abdominal wall (T3N0M0). The patient was discharged after an uneventful postoperative recovery.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Neoplasms of the bile duct are uncommon and are associated with a poor prognosis. Gallbladder carcinoma (GBC) represents 3% of malignant tumors and is fifth in the order of frequency within the group of gastrointestinal malignant neoplasms. The annual incidence of GBC ranges between 2 and 13/100 000 inhabitants. The most common age of presentation is between 65 and 75. In 95% of cases, the most frequent histologic type is adenocarcinoma, and there seems to be a clear association with cholelithiasis, porcelain gallbladder, repeated gallbladder infections, bile duct anomalies and benign gallbladder tumors, among others. Gallstones are the main associated factor; some studies have found that the risk for developing GBC in patients with gallstones is 1%–3%.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,4</span></a> As for the pathogenesis of this entity, chronic inflammation due to several stimuli has been implicated, and it is thought that the adenoma–carcinoma sequence is present in many cases of GBC.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Early-stage GBC presents with non-specific symptoms that are similar to cholelithiasis, while in advanced cancer more significant symptoms can appear, such as weight loss, jaundice, anorexia, ascites and palpable mass in the right hypochondrium, associated with a poorer prognosis and tumor irresectability. Because of this presentation, less than 50% of GBC cases are diagnosed preoperatively; many are incidental findings after cholecystectomies due to cholecystitis or biliary colic.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,5</span></a> In these cases and depending on the pTNM, re-laparotomy is often necessary for radical cholecystectomy (resection of segments <span class="elsevierStyleSmallCaps">iv</span>b and <span class="elsevierStyleSmallCaps">v</span> as well as hilar lymphadenectomy).<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The mortality of this disease is related to the degree of locoregional tumor dissemination. Most GBC cases are diagnosed in late stages, with a 5-year survival rate of only 5%.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,6,7</span></a> Currently, some groups have reported 5-year survival rates between 61%–80% and 30%–45% in stages T2 and T3, respectively, after extensive radical cholecystectomy, suggesting that adequate surgical management with R0 resections can improve the results in patients with GBC.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">In this patient in particular, a second procedure for radical surgery was ruled out given the patient's age, general state and advanced disease.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Despite not being a common form of presentation, isolated cases have been described of spontaneous cholecysto-cutaneous fistula due to gallbladder carcinoma. Spontaneous cholecysto-cutaneous fistula is a rare surgical entity that was first described by Thilesus in 1670. It is becoming a less and less common disease due to the early diagnosis and surgical management of biliary lithiasis, and 226 cases have been published to date.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> This disease presents fundamentally as a complication of a lithiasic inflammatory process, and corresponds with the spontaneous evolution of untreated gallbladder empyema, although there have been cases described of fistulas secondary to acalculous cholecystitis or gallbladder carcinoma.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> The gallbladder perforation generally occurs in the fundus and, once this happens, the gallbladder is able to freely drain into the abdominal cavity or adhere to neighboring structures, causing internal fistulas or, less frequently, toward the abdominal wall as external fistulas. The presentation of the fistula may be evident by observing the discharge of bile or calculi to the abdominal wall. In more difficult situations, there may be drainage of pus, leading one to consider pathologies such as infected epidermal cyst, tuberculoma, pyogenic granuloma, metastatic carcinoma or chronic costal osteomyelitis within the differential diagnosis.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,5,8</span></a></p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Ugalde Serrano P, Solar García L, Miyar de León A, González-Pinto Arrillaga I, González González J. Fístula colecistocutánea como forma de presentación del adenocarcinoma de vesícula biliar. Cir Esp. 2013;91:396–397.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 956 "Ancho" => 900 "Tamanyo" => 133459 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Abdominal computed tomography, sagittal slice.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 659 "Ancho" => 1000 "Tamanyo" => 90931 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Abdominal computed tomography, cross-sectional slice.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:8 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Spontaneous cholecystocutaneous fistula" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "R. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 3 | 0 | 3 |
| 2024 October | 13 | 2 | 15 |
| 2024 September | 18 | 3 | 21 |
| 2024 August | 33 | 7 | 40 |
| 2024 July | 26 | 5 | 31 |
| 2024 June | 19 | 5 | 24 |
| 2024 May | 21 | 6 | 27 |
| 2024 April | 17 | 5 | 22 |
| 2024 March | 21 | 1 | 22 |
| 2024 February | 30 | 4 | 34 |
| 2024 January | 41 | 2 | 43 |
| 2023 December | 39 | 7 | 46 |
| 2023 November | 40 | 4 | 44 |
| 2023 October | 58 | 8 | 66 |
| 2023 September | 26 | 5 | 31 |
| 2023 August | 13 | 2 | 15 |
| 2023 July | 13 | 5 | 18 |
| 2023 June | 26 | 2 | 28 |
| 2023 May | 30 | 3 | 33 |
| 2023 April | 29 | 0 | 29 |
| 2023 March | 42 | 7 | 49 |
| 2023 February | 38 | 6 | 44 |
| 2023 January | 26 | 3 | 29 |
| 2022 December | 18 | 6 | 24 |
| 2022 November | 34 | 9 | 43 |
| 2022 October | 36 | 4 | 40 |
| 2022 September | 29 | 10 | 39 |
| 2022 August | 31 | 14 | 45 |
| 2022 July | 21 | 7 | 28 |
| 2022 June | 26 | 8 | 34 |
| 2022 May | 22 | 4 | 26 |
| 2022 April | 29 | 7 | 36 |
| 2022 March | 31 | 14 | 45 |
| 2022 February | 19 | 9 | 28 |
| 2022 January | 25 | 9 | 34 |
| 2021 December | 26 | 8 | 34 |
| 2021 November | 11 | 8 | 19 |
| 2021 October | 49 | 11 | 60 |
| 2021 September | 19 | 11 | 30 |
| 2021 August | 33 | 5 | 38 |
| 2021 July | 24 | 7 | 31 |
| 2021 June | 30 | 7 | 37 |
| 2021 May | 51 | 9 | 60 |
| 2021 April | 57 | 4 | 61 |
| 2021 March | 45 | 5 | 50 |
| 2021 February | 7 | 2 | 9 |
| 2021 January | 16 | 10 | 26 |
| 2020 December | 19 | 6 | 25 |
| 2020 November | 12 | 13 | 25 |
| 2020 October | 8 | 3 | 11 |
| 2020 September | 16 | 6 | 22 |
| 2020 August | 11 | 14 | 25 |
| 2020 July | 21 | 6 | 27 |
| 2020 June | 21 | 6 | 27 |
| 2020 May | 20 | 14 | 34 |
| 2020 April | 12 | 7 | 19 |
| 2020 March | 13 | 6 | 19 |
| 2020 February | 5 | 7 | 12 |
| 2020 January | 10 | 6 | 16 |
| 2019 December | 12 | 11 | 23 |
| 2019 November | 14 | 7 | 21 |
| 2019 October | 10 | 5 | 15 |
| 2019 September | 9 | 9 | 18 |
| 2019 August | 10 | 5 | 15 |
| 2019 July | 14 | 13 | 27 |
| 2019 June | 29 | 18 | 47 |
| 2019 May | 88 | 33 | 121 |
| 2019 April | 43 | 9 | 52 |
| 2019 March | 12 | 2 | 14 |
| 2019 February | 7 | 6 | 13 |
| 2019 January | 7 | 6 | 13 |
| 2018 December | 2 | 2 | 4 |
| 2018 November | 3 | 2 | 5 |
| 2018 October | 11 | 5 | 16 |
| 2018 September | 6 | 0 | 6 |
| 2018 August | 1 | 0 | 1 |
| 2018 July | 10 | 4 | 14 |
| 2018 June | 2 | 1 | 3 |
| 2018 May | 5 | 4 | 9 |
| 2018 April | 12 | 0 | 12 |
| 2018 March | 8 | 0 | 8 |
| 2018 February | 12 | 4 | 16 |
| 2018 January | 7 | 3 | 10 |
| 2017 December | 7 | 4 | 11 |
| 2017 November | 2 | 3 | 5 |
| 2017 October | 12 | 2 | 14 |
| 2017 September | 13 | 0 | 13 |
| 2017 August | 10 | 7 | 17 |
| 2017 July | 9 | 0 | 9 |
| 2017 June | 13 | 10 | 23 |
| 2017 May | 10 | 4 | 14 |
| 2017 April | 11 | 4 | 15 |
| 2017 March | 10 | 2 | 12 |
| 2017 February | 15 | 7 | 22 |
| 2017 January | 13 | 2 | 15 |
| 2016 December | 20 | 9 | 29 |
| 2016 November | 17 | 6 | 23 |
| 2016 October | 26 | 7 | 33 |
| 2016 September | 26 | 2 | 28 |
| 2016 August | 28 | 5 | 33 |
| 2016 July | 23 | 1 | 24 |
| 2016 June | 23 | 6 | 29 |
| 2016 May | 27 | 19 | 46 |
| 2016 April | 24 | 16 | 40 |
| 2016 March | 16 | 17 | 33 |
| 2016 February | 17 | 4 | 21 |
| 2016 January | 18 | 11 | 29 |
| 2015 December | 19 | 7 | 26 |
| 2015 November | 24 | 10 | 34 |
| 2015 October | 21 | 8 | 29 |
| 2015 September | 22 | 7 | 29 |
| 2015 August | 20 | 4 | 24 |
| 2015 July | 9 | 6 | 15 |
| 2015 June | 4 | 3 | 7 |
| 2015 May | 16 | 0 | 16 |
| 2015 April | 21 | 2 | 23 |
| 2015 March | 21 | 3 | 24 |
| 2015 February | 13 | 4 | 17 |
| 2015 January | 15 | 4 | 19 |
| 2014 December | 41 | 8 | 49 |
| 2014 November | 25 | 5 | 30 |
| 2014 October | 13 | 3 | 16 |
| 2014 September | 13 | 3 | 16 |
| 2014 August | 13 | 3 | 16 |
| 2014 July | 25 | 5 | 30 |
| 2014 June | 11 | 1 | 12 |
| 2014 May | 13 | 3 | 16 |
| 2014 April | 6 | 1 | 7 |
| 2014 March | 7 | 3 | 10 |
| 2014 February | 5 | 1 | 6 |
| 2014 January | 15 | 5 | 20 |
| 2013 December | 6 | 2 | 8 |
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