English

Abstract

Cystic lymphangioma is a lymphatic malformation which usually occurs at the time of birth or in the first few months of life. Cystic lymphangioma presenting in adulthood is less common. We report a case of a 21 yr. old adult male who presented with a rapidly progressive cystic swelling in the right submandibular region for the past 5 months. Further investigations were suggestive of lymphangioma. Surgical excision was challenging and resulted in complete cure without any morbidity, there was no evidence of recurrence at 3 years of follow up. We review the literature along with the case report of cystic lymphangioma presenting in adults. INTRODUCTION: 'Hygroma' in Greek means water containing tumor. Cystic Hygroma or Cystic lymphangioma is a congenital malformation of the lymphatic system. 50% of these malformations appear at birth and about 80% to 90% manifest by 2year of life 1 . The initial presentation in adults is less common, only about 100 cases have been reported in literature 2 . Lymphangioma can occur anywhere in the body but most common site is the head and neck region 3. No sex preponderance or side predilection has been reported 1. Infection in the respiratory tract or the skin or trauma in the head and neck region is associated with the rapid growth or engorgement with lymph or blood resulting in rapid increase in size. Cervicofacial lymphangioma has been reported to spontaneously regress in size in upto 15% of patients. Malignant change of lymphangioma has not been reported. CASE REPORT: 21 year old male presented to the OPD with history of a rapidly progressive swelling in the right submandibular region for the past 5 months. He noticed that the swelling was bigger on lying down. There was no history of trauma, fever, pain, tooth ache or difficulty in breathing or swallowing. On examination a 5 x 4.5 cm, soft, cystic, non-tender, fluctuant, compressible diffuse swelling (fig.1) was seen in the right submandibular region and upper part of neck extending superiorly to ramus of mandible, inferiorly to thyroid notch, medially just crossing the midline and laterally deep to sternocleidomastoid. Swelling was not reducible did not move on deglutition or protrusion of tongue. There was no cough impulse and transillumination was negative. There was no visible swelling in the floor of mouth and on bidigital palpation submandibular gland was palpable along with the swelling. On auscultation there was no bruit. All the baseline investigations were normal. FNAC from the swelling was inconclusive. CT neck with contrast revealed multiloculated cyst with smooth septa, which enhanced uniformly after contrast injection. (fig.2) Excision of the mass under general anesthesia was performed through a horizontal skin crease incision placed just 3 cm below the ramus of mandible. The finger like multiloculated cyst (fig.3) was seen extending deep to the mylohyoid muscle up to the floor of mouth which was fully