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Cervical Lymphangioma in Adults: Case Report and Current Treatment

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Cervical Lymphangioma in Adults: Case Report and Current Treatment case reports 2018; 4(1) https://doi.org/10.15446/cr.v4n1.67067 CERVICAL LYMPHANGIOMA IN ADULTS: CASE REPORT AND CURRENT TREATMENT Keywords: Adult; Lymphangioma, Cystic; Sclerotherapy; Picibanil. Palabras clave: Adulto; Linfangioma quístico; Escleroterapia; Picibanil. Enrique Cadena-Piñeros Head and Neck Unit - Instituto Nacional de Cancerología - Bogotá D.C. - Colombia. Department of Surgery - Universidad Nacional de Colombia - Faculty of Medicine - Bogotá D. C. - Colombia. Otolaryngology Unit - Clínica de Marly - Bogotá D.C. - Colombia. Head and Neck Surgery Unit - Clínica de Marly - Bogotá D.C. - Colombia Andrés Rojas Gutiérrez Head and Neck Unit - Instituto Nacional de Cancerología - Bogotá D.C. - Colombia. Corresponding author: Enrique Cadena. Head and Neck Unit - Instituto Nacional de Cancerología - Bogotá D.C. - Colombia. E-mail [email protected]. Recibido: 15/08/2017 Aceptado: 18/01/2018 Case reports Vol. 4 No. 1: 61-8 62 RESUMEN static nodes, branchial cysts, thyroglossal cysts and lymphangiomas, among others. Clinically Introducción: Los tumores quísticos del cue- speaking, lymphangiomas are slow-growing soft llo son inusuales en los adultos. Sin embargo, masses located in different spaces of the neck. se pueden encontrar metástasis a ganglios, quistes branquiales, quistes tiroglosos, linfan- Case report: This paper reports the case of giomas, entre otros. Clínicamente, estos últimos a 36-year-old woman presenting with lymph- son masas blandas de crecimiento lento que se angioma, who consulted due to a right lateral localizan en diferentes espacios del cuello. mass in the neck of 20 days of evolution with- out associated systemic symptoms. Although Reporte de caso: Se reporta un caso de lin- relapse is frequent, the patient was success- fangioma en una mujer de 36 años, quien con- fully treated with surgery, without evidence of sultó por masa lateral derecha del cuello con recurrence at 12 months of follow-up. evolución de 20 días sin síntomas sistémicos asociados. A pesar de que la recaída es fre- Discussion: When cystic tumors of the neck cuente, la paciente fue tratada con cirugía exi- occur in children, surgical urgencies may arise tosa sin evidencia de recidiva durante 12 me- due to obstruction of the airway. However, ses de seguimiento. lymphangioma in adults only produce contour deformity and rarely require urgent interven- Discusión: Cuando se presentan en niños, tion, which allows for conservative manage- los tumores quísticos del cuello se pueden ment such as observation, repeated drainage convertir en urgencias quirúrgicas debido a or sclerotherapy that can be done using OK- obstrucción de la vía aérea; no obstante, en 432 (Picibanil). Nevertheless, surgery remains los adultos solo producen deformidad de a good treatment option, but some complica- contorno y rara vez requieren una intervención tions may occur. apremiante, lo que permite conductas conser- vadoras como la observación, el drenaje repe- Conclusion: Different treatment options were tido o la escleroterapia. Esta última puede ha- reviewed, which led to conclude that surgical cerse con el OK-432 (Picibanil); sin embargo, resection of lymphangiomas continues to be a la cirugía es una buena opción de tratamiento good treatment for this complex neck lesion. sin estar exenta de complicaciones. Conclusión: Se realizó revisión de las dife- INTRODUCTION rentes opciones de tratamiento y se concluyó que la resección quirúrgica de los linfagiomas Cystic lesions of the neck are rare and difficult continúa siendo la opción más adecuada para to interpret for clinicians, since they can be be- el manejo de esta compleja lesión del cuello. nign or malignant pathologies. The most com- mon location is the posterior triangle, where ABSTRACT inflammatory, metastatic adenopathies or lymphoproliferative diseases can occur. In ad- Introduction: Cystic tumors of the neck are dition, congenital malformations such as bran- rare in adults. Some of them include meta- chial cysts, hemangiomas and lymphangiomas cervical lymphangioma in adults may be observed in this area. (1) Branchial cleft a review of the subject is presented with a 63 cysts and hemangiomas are easily diagnosed description of the different treatment options. in children under 2 years of age due to their clinical characteristics. These lesions are ob- CASE PRESENTATION served as a mass that usually does not involve inflammatory changes and presents a slow and A 36 year-old female patient, Caucasian, from painless growth or lymphangiomas, which are the city, consulted due to a right lateral mass rare after the third decade of life. (2-5) in the neck of 20 days of evolution without as- The current trend to treat lymphangiomas sociated systemic symptoms, dysphonia, dys- is different; conservative interventions such phagia or pain. No personal or family history of as sclerotherapy, now performed with new importance in relation to the current disease substances, have relegated surgery to the and no history of neck trauma or infection of background due to the high rate of relapse the head and neck area were described. On or persistence. (4,5) Considering that lymph- physical examination, a right lateral soft mass angiomas are rare in adults (4) and that the of 6cm was found, which was tender on pal- patient treated was a 36-year-old woman who pation and with displacement and lateralization underwent a complete surgical resection, of the sternocleidomastoid muscle (Figure 1). Figure 1. Deformity of the right contour of the neck. A) Anterior aspect of the neck; B) Lateral aspect of the neck. Source: Own elaboration based on the data obtained in the study. A contrast study of the neck using computed angioma (cystic hygroma). Due to the lack of tomography (CT) showed a cystic lesion with diagnostic clarity, a surgery was performed thin walls of 55x21x43mm, which compressed 15 days later. During the procedure, a medial the internal jugular vein, occupied the right cystic mass was found in the sternocleido- III and IV levels and was clearly delimited by mastoid muscle adhered to the internal jugular vascular and muscular structures (Figure 2). vein and to the phrenic, hypoglossal, spinal Fine needle aspiration (FNA) helped to and vagus nerves. Finally, the lesion was diagnose cyst content with lymphoid tissue completely resected along with the entire lining, which could be lymphocele or lymph- capsule (Figure 3). Case reports Vol. 4 No. 1: 61-8 64 Figure 2. Contrast computed tomography of the neck. A) Thin-walled cyst medial to the sternocleidomastoid muscle; B) Cystic lesion occupying the right III and IV levels. Source: Own elaboration based on the data obtained in the study. Figure 3. Intraoperative appearance. A) Transverse incision of the neck. A deep lesion of the sternocleidomastoid muscle was observed along with junctions to the internal jugular vein and accessory nerve. B) Lymphangioma was dissected from the internal jugular vein. Source: Own elaboration based on the data obtained in the study. Figure 4. Surgical specimen: transillumination revealed a thin-walled cyst. Source: Own elaboration based on the data obtained in the study. cervical lymphangioma in adults Transillumination of the surgical specimen Lymphangiomas correspond to 25% of all 65 revealed a thin-walled, cystic-like mass with vascular tumors in children and adolescents; citrine material content, which correlated with 70% of cases are found in the head and the the diagnosis of preoperative FNA (Figure 4). neck. In 2006, Thompson (11) proposed The final pathology reported a cystic classifying them into cystic (cystic hygroma), mass of 6.9x3.2x3cm, with smooth external capillary or cavernous, but there are different surface and a clear liquid content. Additionally, histological classifications: the smooth inner surface was accompanied Colbert et al. (12) classified them into by an adipose tissue of 2.5x1.8x1cm with macrocysts, formerly known as cystic hy- another tissue of 1.5x0.6x0.5cm. The final gromas, and in microcysts, which may occur diagnosis of the pathology was lymphangioma simultaneously in a lesion. and five reactive lymph nodes, negative for Bhayya et al. (13) classified them in tumor infiltration. The patient did not present lymphangioma simplex, composed of small any complication during the postoperative thin-walled lymphatics; cavernous lymphan- period, nor motor deficit related to the dis- gioma, comprised of dilated lymphatic vessels sected structures, nor evidence of relapse or with surrounding adventitia, cystic lymph- persistence of the lesion after 12 months of angioma, consisting of a large lesion, with follow-up. No complaints were made by the macroscopic lymphatic spaces surrounded by patient, who reported excellent functional and fibrovascular tissue and smooth muscle, and cosmetic results. benign lymphangioendothelioma, lymphatic channels that seem to be dissected through DISCUSSION dense collagen bundles. In 1995, de Serres et al. (14) proposed a Lymphangiomas, also known as cystic hygro- classification according to anatomical location: mas, are congenital malformations that are Stage I, unilateral infrahyoid lesion; Stage II, usually diagnosed at birth and can cause air- unilateral suprahyoid lesion; Stage III, unilat- way obstruction, thus constituting a medical eral suprahyoid and infrahyoid lesion; Stage emergency. (6) These lesions are considered IV, bilateral suprahyoid lesion, and Stage V, vascular anomalies of the lymphatic system bilateral suprahyoid and infrahyoid lesion. and are classified as
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