International Journal of Clinical Pediatric Dentistry

Register      Login

VOLUME 15 , ISSUE 6 ( November-December, 2022 ) > List of Articles

CASE REPORT

Embryological Basis of Cystic Hygroma: A Case Report

Shefali Yadav, Nikita Gulati, Devi Charan Shetty, Saurabh Juneja

Keywords : Amorphous, Cystic hygroma, Lymphoid aggregates, Malformation

Citation Information : Yadav S, Gulati N, Shetty DC, Juneja S. Embryological Basis of Cystic Hygroma: A Case Report. Int J Clin Pediatr Dent 2022; 15 (6):774-778.

DOI: 10.5005/jp-journals-10005-2472

License: CC BY-NC 4.0

Published Online: 14-02-2023

Copyright Statement:  Copyright © 2022; The Author(s).


Abstract

Aim: We intend to present a cystic hygroma (CH) case in a 2-year-old baby in the anterior cervical triangle, which is a rare site, as the most common site for the occurrence of CH is the supraclavicular fossa of the posterior cervical triangle. Background: Among developmental abnormalities in the lymphoid system, CH are usually seen in the posterior neck. Lymphatic malformations are generally exhibited either at birth or before the age of 2 years. Lymphatic channels are attenuated endothelium-lined spaces devoid of any cells and smooth muscle layer. Also, morphologically distinguishing normal lymphatic channels from venules or capillaries is a challenge. Case description: A 2-year-old female patient reported having a chief complaint of swelling in the left submandibular region for 4 days. The patient underwent surgery for CH 18 days after birth. Swelling was rubbery in texture and firm in consistency. Conclusion: A D2-40 immunoexpression was an identifying clue for normal lymphatics in comparison to morphology. Henceforth, this can be concluded that such tumors depict at least partial differentiation of endothelial cells lining lymphatic spaces. Clinical significance: The present article helps in illuminating the role of D2-40 in the diagnosis of lymphatic malformations, such as CH, and also highlights the embryological basis of the pathogenetic mechanism of this rare disease, which potentiates the role of various treatment modalities in pediatric cases for management considerations.


PDF Share
  1. Bonet-Coloma C, Minguez-Martinez I, Aloy Prosper M, et al. Clinical characteristics, treatment, and evolution in 14 cases of pediatric orofacial lymphangioma. J Oral Maxillpfac Surg 2011;69(6):e96–e99. DOI: 10.1016/j.joms.2010.07.029
  2. Kayhan KB, Keskin Y, Kesimli MC, et al. Lymphangioma of the tongue: report of four cases with dental aspects. Kulak Burun Bogaz Ihtis Derg 2014;24(3):172–176. DOI: 10.5606/kbbihtisas.2014.00236
  3. Tumors and Malformations of Lymphatic Vessels. In: Goldblum JR, Folpe AL and Weiss SW. Enzinger and Weiss's Soft tissue tumors. 6th edi. Philadelphia, PA: Saunders, Elsevier Inc; 2014. p. 733–748.
  4. Adams MT, Saltzman B, Perkins JA. Head and neck lymphatic malformation treatment: a systematic review. Otolaryngol Head Neck Surg 2012;147(4):627–639. DOI: 10.1177/0194599812453552
  5. Damaskos C, Garmpis N, Manousi M, et al. Cystic hygroma of the neck: single center experience and literature review. Eur Rev Med Pharmacol Sci 2017;21(21):4918–4923.
  6. Beech AN, Farrier JN. An interesting association of cystic hygroma of the neck and Lymphangioma causing a paediatric swollen tongue. Case Rep Paediatr 2016;2016:7930945. DOI: 10.1155/2016/7930945
  7. Levy AT, Berghella V, Al-Kouatly HB. Outcome of 45,X fetuses with cystic hygroma: a systematic review. Am J Med Genet Part A 2020;185(1):26–32. DOI: 10.1002/ajmg.a.61902
  8. Machin G: Hydrops, cystic hygroma, hydrothorax, pericardial effusions, and fetal ascites, in Gilbert-Barness E (ed): Potter's Pathology of the Fetus and Infant. St. Louis, MO, Mosby; 1997: pp 163–181.
  9. Kolay SK, Parwani R, Wanjari S, et al. Oral lymphangiomas - clinical and histopathological relations: an immunohistochemically analyzed case series of varied clinical presentations. J Oral Maxillofac Pathol 2018;22(Suppl 1):S108–S111. DOI: 10.4103/jomfp.JOMFP_157_17
  10. Fakunaga M. Expression of D2-40 in lymphatic endothelium of normal tissues and in vascular tumours. Histopathology 2005;46(4):396–402. DOI: 10.1111/j.1365-2559.2005.02098.x
PDF Share
PDF Share

© Jaypee Brothers Medical Publishers (P) LTD.