DOI: http://dx.doi.org/10.18203/issn.2454-5929.ijohns20221397

A huge high rising cervico-facial lymphangioma in an adolescent male: from pterygoid to hyoid

Sharmistha Chakravarty, Nitin M. Nagarkar, Nidhin S. B., Abhishek A. P.

Abstract


Cervicofacial lymphangioma is a benign swelling of the neck and is commonly seen in paediatric population. Common sites of involvement are the submandibular space and the posterior triangle and rarely involves the face. We present a rare case of a huge cervico-facial lymphangioma in a 14 year old male where the swelling presented one year back and had progressively increased in size extending from the medial pterygoids and pterygomandibular space above to myelohyoid muscle below. Intraoperatively, it was closely abutting the submandibular gland mimicking salivary gland retention cyst and the facial artery was traversing through the mass. Complete surgical excision of the mass was performed along with removal of the ipsilateral submandibular gland. Salivary gland mucocele and cervicofacial lymphangioma were the two main differential diagnosis in our case. Intraoperative findings of macrocystic spaces with septations pointed towards a lymphangioma. Post operative histopathology confirmed it to be lymphangioma lined with endothelial cells. We concluded from this case that cervicofacial swellings in higher age groups should have lymphangiomas as differential diagnosis. As the swelling ascended up to the skull base, careful radiological assessment, meticulous surgical plan and cranio-caudal exposure from the zygomatic arch above to the hyoid below helped in complete surgical excision of the mass.  


Keywords


Lymphangioma, Lymphatic malformations, Submandibular space, Masticator space, Submandibular gland

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References


Zadvinskis DP, Benson MT, Kerr HH, Mancuso AA, Cacciarelli AA, Madrazo BL et al. Congenital malformations of the cervicothoracic lymphatic system: embryology and pathogenesis. Radiographics. 1992 ;12(6):1175-89.

Gow L, Gulati R, Khan A, Miahaimeed F. Adult onset cystic hygroma: A case report and review of management. Grand Rounds. 2011;11:5-11.

Agha RA, Borrelli MR, Farwana R, Koshy K, Fowler AJ, Orgill DP. SCARE Group. The SCARE 2018 statement: Updating consensus Surgical CAseREport (SCARE) guidelines. Int J Surg. 2018;60:132-6.

Agha RA, Franchi T, Sohrabi C. The SCARE 2020 Guideline: Updating Consensus Surgical CAseREport (SCARE) Guidelines. International Journal of Surgery (London, England). 2020;84:226-30.

El Sayed M, Touny M, Ibrahim N, Kasb I, Al-Azzawi Z. A rare case of cystic hygroma in neck and extending into thoracic cavity. Int J Surg Case Rep. 2020;76:174-7.

Coffin CM, Dehner LP. Vascular tumors in children and adolescents: a clinicopathologic study of 228 tumors in 222 patients. PatholAnnu. 1993;28(1):97-120.

Grasso DL, Pelizzo G, Zocconi E, Schleef J. Lymphangiomas of the head and neck in children. Acta Otorhinolaryngol Ital. 2008;28(1):17-20.

Brumfield CG, Wenstrom KD, Davis RO, Owen J, Cosper P. Second-trimester cystic hygroma: prognosis of septated and nonseptated lesions. Obstet Gynecol. 1996;88(6):979-82.

Potdar PD, Deshpande S, Chaugule S. Development and Molecular Characterization of Cystic Hygroma Cell Line to be Used as an in Vitro Model System to Study the Progression of Hygroma in Young Children. Pediatrics Research International Journal. 2013;2013:879081.

Preeti A, Rajiv K. Cystic hygroma: cytological and radiological co-relation. J Clin Diagn Res. 2011;5(5):1008-10.

Bahl S, Shah V, Anchlia S, Vyas S. Adult-onset cystic hygroma: a case reportof rare entity. Indian J Dent. 2016;7(1):51-4.

April MM, Rebeiz EE, Friedman EM, Healy GB, Shapshay SM. Laser therapy for lymphatic malformations of the upper aerodigestive tract. An evolving experience. Arch Otolaryngol Head Neck Surg. 1992;118(2):205-8.

Balakrishnan A, Bailey CM. Lymphangioma of the tongue. A review of pathogenesis, treatment and the use of surface laser photocoagulation. J Laryngol Otol. 1991;105(11):924-9.

Mikhail M, Kennedy R, Cramer B, Smith T. Sclerosing of recurrent lymphangioma using OK-432. J Pediatr Surg. 1995;30(8):1159-60.