Scientific letter
Peripancreatic lymphangioma: When fine needle aspiration changes the diagnosis—Case report and literary review
Linfangioma peripancreático: cuando la aspiración con aguja fina cambia el diagnóstico. Reporte de caso y revisión de la literatura
Inês Pestana
, Ana Caldeira, Mara Costa, Rui Sousa, Eduardo Pereira, António Banhudo
Corresponding author
Department of Gastroenterology, Hospital Amato Lusitano ULSCB, Castelo Branco, Portugal
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Reporte de caso y revisión de la literatura" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2548 "Ancho" => 2508 "Tamanyo" => 372255 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Peripancreatic cystic lesion (peripancreatic lymphangioma); (B) EUS-FNA of cystic lesion; (C) Milky fluid obtained from EUS-FNA.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Lymphangiomas are rare, slow-growing, benign lesions of vascular origin with lymphatic differentiation.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1,2</span></a> They uncommonly involve the abdomen and rarely the pancreas.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1–4</span></a> Pancreatic lymphangiomas were first described by Koch in 1913 and are extremely rare, accounting for only 1% of abdominal lymphangiomas and less than 0.2% of all pancreatic lesions.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1–3,5</span></a> Lymphangiomas are considered to be of pancreatic origin if they occur adjacent to the pancreas, within the pancreatic parenchyma, or connected to the pancreas via a pedicle.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1,3</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">A 44-year-old white male was referred to a Gastroenterology consultation after an abdominal computed tomography (CT) showed a 22<span class="elsevierStyleHsp" style=""></span>mm hypodense nodular image located between the inferior vena cava (IVC) and the second part of the duodenum externally to the splenomesaic confluent, whose density suggested a liquid/cystic nature. The physical examination and laboratory test results were unremarkable. An endoscopic ultrasound (EUS) was requested and showed a 23<span class="elsevierStyleHsp" style=""></span>mm cystic lesion with a well-defined and regular wall located between the IVC and the cephalic portion of the pancreas; the lesion was next to the pancreatic parenchyma, raising the hypothesis of a pancreatic cystic lesion. Fine needle aspiration (FNA) biopsy revealed a milky fluid with a yellowish color that might correspond to lymph, suggesting the possibility of a lymphangioma (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). Cytological and biochemical exam of the fluid revealed 18,000 nucleated cells/μL with a lymphocyte predominance and a background of proteinaceous material; an elevated triglyceride value (2953.3<span class="elsevierStyleHsp" style=""></span>mg/dL); amylase (351<span class="elsevierStyleHsp" style=""></span>U/L) and carcinoembryonic antigen (CEA) (8.84<span class="elsevierStyleHsp" style=""></span>ng/mL) within normal limits. All these aspects support the diagnosis of peripancreatic lymphangioma. As the patient was asymptomatic, a conservative approach with imaging surveillance was chosen. The patient will be re-evaluated in six months.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Lymphangiomas result from lymphangiectasia, which may be associated with congenital malformations, inflammatory processes, radiotherapy, surgical intervention or previous abdominal trauma.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1–3</span></a> These lesions have a female preponderance (unlike our case), have been reported in all age groups and have a variable size (from 3 to 20<span class="elsevierStyleHsp" style=""></span>cm).<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1,3</span></a> They are often asymptomatic and discovered as an incidental finding.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">2,5</span></a> However, the clinical presentation depends on the size, location, mass effect and complications. Common non-specific clinical manifestations include abdominal pain or mass, nausea, vomiting and weight loss, due to the pressure effect.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1,2</span></a> Owing to their rarity, this lesion is difficult to diagnose pre-operatively.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">2–4</span></a> Most of the imaging studies are non-specific.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">2,3</span></a> EUS can allow a preoperative diagnosis, particularly because of EUS-FNA of the cystic fluid and its consequent cytological and biochemical studies.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">2,4,5</span></a> Lymphangiomas can be filled with serous, serohematic, or lymphatic fluid.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">1</span></a> Milky white fluid with a triglyceride level greater than 5000<span class="elsevierStyleHsp" style=""></span>mg/dL is diagnostic of lymphangioma. Amylase and CEA should be measured to rule out epithelial lesions and pseudocysts. Cytological examination shows a high population of small mature lymphocytes.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">2,3</span></a> There have been no reports of malignant transformation.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">5</span></a> As so, when there is a definitive diagnosis, the patient is asymptomatic and the lesion is not locally invasive, a conservative approach with imaging surveillance can be undertaken, as was the choice in our case.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1,2,4,5</span></a> Although lymphangiomas are benign neoplasms, they can be locally invasive and may involve adjacent organs. Complete surgical excision is the curative treatment, providing excellent prognosis and very low incidence of recurrence.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">1–4</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">It is very important to consider this entity as a differential diagnosis of cystic pancreatic lesions, in order to avoid unnecessary or aggressive treatments.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial disclosures</span><p id="par0025" class="elsevierStylePara elsevierViewall">The authors of this manuscript do not have any financial disclosures to report.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Conflict of interest</span><p id="par0030" class="elsevierStylePara elsevierViewall">The authors of this manuscript do not have any conflicts of interest to report.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Financial disclosures" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Conflict of interest" ] 2 => array:2 [ "identificador" => "xack747912" "titulo" => "Acknowledgments" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:1 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 2548 "Ancho" => 2508 "Tamanyo" => 372255 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Peripancreatic cystic lesion (peripancreatic lymphangioma); (B) EUS-FNA of cystic lesion; (C) Milky fluid obtained from EUS-FNA.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0030" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pancreatic lymphangioma" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "J. Karajgikar" 1 => "S. Deshmukh" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1097/RCT.0000000000000818" "Revista" => array:6 [ "tituloSerie" => "J Comput Assist Tomogr" "fecha" => "2019" "volumen" => "43" "paginaInicial" => "242" "paginaFinal" => "244" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/30371621" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0035" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pancreatic lymphangioma: an unusual cause of abdominal lump" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "R. Ray" 1 => "T.D. Baruah" 2 => "H.S. Mahobia" 3 => "A. 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Alcaraz" 2 => "M.A. López" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.17235/reed.2020.7671/2020" "Revista" => array:5 [ "tituloSerie" => "Rev Esp Enferm Dig" "fecha" => "2021" "volumen" => "113" "paginaInicial" => "682" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/33393336" "web" => "Medline" ] ] ] ] ] ] ] ] 4 => array:3 [ "identificador" => "bib0050" "etiqueta" => "5" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Cystic lymphangioma of the pancreas: a hard diagnostic challenge between pancreatic cystic lesions-review of recent literature" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "F. Viscosi" 1 => "F. Fleres" 2 => "C. Mazzeo" 3 => "I. Vulcano" 4 => "E. 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