Cystic hygroma in a young adult: A case report and recent management
We are reporting a case of a 27-year-old young female who presented with right side neck swelling without any associated obstructive symptoms and any other grave signs and symptoms. She noticed a gradual increase in the size of the swelling within a period of 2 years. After investigation and surgical excision, the swelling was diagnosed as cystic hygroma. The root cause of the development of cervical lymphangioma is the congenital malformation of the developing lymphatic system. Cystic hygroma is benign in nature and the cause in adults is still unclear. The most common site of origin is in head and neck region, and cystic hygroma accounts for 75% of lymphatic malformations. The most common presentation of cystic hygroma is painless swelling with ill-defined lesion, most commonly located at the posterior triangle of the neck. The common age group is between birth and 2 years of age, with very rare presentation in adults. Hence, it is necessary to rule out all differential diagnosis of cervical lymphangioma, which is presented with cystic neck swelling. Complete surgical excision is the recommended standard treatment.
Gow L, Gulati R, Khan A, et al., 2011, Adult-onset cystic hygroma: A case report and review of management. Grand Rounds, 11: 5–11. https://doi.org/10.1102/1470-5206.2011.0002
Suk S, Sheridan M, Saenger JS, 1997, Adult lymphangioma: A case report. Ear Nose Throat J, 76(12): 881–883.
Bloom DC, Perkins JA, Manning SC, 2004, Management of lymphatic malformations. Curr Opin Otolaryngol Head Neck Surg, 12(6): 500–4. https://doi.org/10.1097/01.moo.0000143971.19992.2d
Naidu SI, McCalla MR, 2004, Lymphatic malformations of the head and neck in adults: A case report and review of the literature. Ann Otol Rhinol Laryngol., 113(3 Pt 1): 218–222. https://doi.org/10.1177/000348940411300309
Schefter RP, Olsen KD, Gaffey TA, 1985, Cervical lymphangioma in the adult. Otolaryngol Head Neck Surg, 93(1): 65–69. https://doi.org/10.1177/019459988509300113
Antoniades K, Kiziridou A, Psimopoulou M, 2000, Traumatic cervical cystic hygroma. Int J Oral Maxillofacial Surg, 29(1): 47–48.
Brea-Álvarez B, Roldan-Fidalgo A, 2015, Quistes en el triángulo cervical posterior en adultos [Cysts in the posterior triangle of the neck in adults]. Acta Otorrinolaringol Esp, 66(2): 106–110. https://doi.org/10.1016/j.otorri.2014.02.013
Curran AJ, Malik N, McShane D, et al., 1996, Surgical management of lymphangiomas in adults. J Laryngol Otol, 110(6): 586–589. https://doi.org/10.1017/s0022215100134334
Farmand M, Kuttenberger JJ, 1996, A new therapeutic concept for the treatment of cystic hygroma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod, 81(4): 389–395. https://doi.org/10.1016/s1079-2104(96)80013-8
Filston HC, 1994, Hemangiomas, cystic hygromas, and teratomas of the head and neck. Semin Pediatr Surg, 3(3): 147–159.
Kennedy TL, Whitaker M, Pellitteri P, Wood WE, 2001, Cystic hygroma/lymphangioma: a rational approach to management. Laryngoscope, 111(11): 1929–1937.
Rajendran R, Sivapathasundaram B, 2009, Shafer’s Textbook of Oral Pathology. 6th ed. New Delhi, India: Elsevier.
Teresa MO, Rickert SM, Diallo AM, et al., 2013, Lymphatic malformations of the airway. Otolaryngol Head Neck Surg, 149(1): 156–160. https://doi.org/10.1177/0194599813485065
Bossert T, Gummert JF, Mohr FW, 2002, Giant cystic lymphangioma of the mediastinum. Eur J Cardiothorac Surg, 21(2): 340. https://doi.org/10.1016/s1010-7940(01)01096-x
Nakazato Y, Ohno Y, Nakataa Y, et al., 1995, Cystic lymphangioma of themediastinum. AmericanHeart J, 129(2): 406–409.
Sarin YK, 2000, Cystic hygroma. Indian Pediatrics, 37(10): 1139–1140.
Shields TW, 2002, General Thoracic Surgery. Tumours of Lymph Vessel Origin. 5th ed. Philadelphia, PA, USA: Lippincott Williams and Wilkins.
Chen YN, Chen CP, Lin CJ, et al., 2017, Prenatal ultrasound evaluation and outcome of pregnancy with fetal cystic hygromas and lymphangiomas. J Med Ultrasound. 25(1): 12–15.
Moustatos GH, Baffes TG, 1963, Cervical masses in infants and children. Pediatrics, 32: 251–256.
Ricciardelli EJ, Richardson MA, 1991, Cervicofacial cystic hygroma: Patterns of recurrence and management of the difficult case. Arch Otolaryngol Head Neck Surg, 117(5): 546–553. https://doi.org/10.1001/archotol.1991.01870170092021
Colbert SD, Seager L, Haider F, et al., 2013, Lymphatic malformations of the head and neck-current concepts in management. Br J O Maxillofac Surg, 51(2): 98–102. http://doi.org/10.1016/j.bjoms.2011.12.016
de Serres LM, Sie KC, Richardson MA, 1995, Lymphatic malformations of the head and neck. A proposal for staging. Arch Otolaryngol Head Neck Surg, 121(5): 577–582. http://doi.org/10.1001/archotol.1995.01890050065012
Fageeh N, Manoukian J, Tewfik T, et al., 1997, Management of head and neck lymphatic malformations in children. J Otolaryngol, 26(4): 253–258.
Miceli A, Stewart KM, 2017, Lymphangioma. Treasure Island, FL: StatPearls Publishing. Available from: https://goo.gl/7iBg5s
Ibrahim AH, Kandeel A, Bazeed MF, 2009, Successful non-surgical management of a huge life threatening cervico mediastinal cystic hygroma case report and review of the literature. J Pediatr Surg Specialt, 3: 48–50.
Mahajan JK, Bharathi V, Chowdhary SK, et al., 2004, Bleomycin as intralesional sclerosant for cystic hygromas. J Indian Assoc Pediatr Surg, 9: 3–7.
Okada A, Kubota A, Fukuzawa M, et al., 1992, Injection of bleomycin as a primary therapy of cystic lymphangioma. J Pediatr Surg, 27: 440–443. http://doi.org/10.1016/0022-3468(92)90331-z
Orford J, Barker A, Thonell S, et al., 1995, Bleomycin therapy for cystic hygroma. J Pediatr Surg, 30(9): 1282–1287.
Zhong PQ, Xhi FX, Li R, et al., 1998, Long-term results of intratumorous bleomucin-A5 injection for head and neck lymphangioma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod, 86(2): 139–144. http://doi.org/10.1016/s1079-2104(98)90115-9
Selz PA, Arjmand EM, 1998, Laryngeal lymphangioma: A case report of an uncommon entity. Otolaryngol Head Neck Surg, 118(3 Pt 1): 382–384. http://doi.org/10.1016/s0194-5998(98)70320-1
Sobol SE, Manoukian JJ, 2001, Acute airway obstruction from a laryngeal lymphangioma in a child. Int J Pediatr Otorhynolaryngol, 58(3): 255–257. http://doi.org/10.1016/s0165-5876(01)00433-5
Shumaker PR, Dela Rosa KM, Krakowski AC. 2013, Treatment of lymphangioma circumscriptum using fractional carbon dioxide laser ablation. Pediatr Dermatol. 30(5): 584–586. http://doi.org/10.1111/pde.12165
Ogita S, Tsuto T, Tokiwa K, et al., 1987, Intracystic injection of OK-432: A new sclerosing therapy for cystic hygroma in children. Br J Surg, 74(8): 690–691. http://doi.org/10.1002/bjs.1800740812
Ohta N, Fusake S, Watanabe T, et al., 2010, Effects and mechanism of OK-432 therapy in various neck cystic lesions. Acta Otolaryngol, 130(11): 1287–1292. http://doi.org/10.3109/00016489.2010.483480