Clinical and Experimental Pediatrics

The Korean Pediatric Society (대한소아청소년과학회)
권호 표지
DOI QR코드

DOI QR Code

Macroglossia secondary to lymphangioma of the deep neck space: Report of two cases

심 경부 림프관종에 의한 거설증 2례

  • Cho, Han-Gil (Departments of Pediatrics, College of Medicine, Chonnam National University) ;
  • Kim, Soo-Young (Departments of Pediatrics, College of Medicine, Chonnam National University) ;
  • Song, Eun-Song (Departments of Pediatrics, College of Medicine, Chonnam National University) ;
  • Lee, Joon-Kyoo (Otolaryngology-Head and Neck Surgery, College of Medicine, Chonnam National University) ;
  • Choi, Young-Youn (Departments of Pediatrics, College of Medicine, Chonnam National University)
  • 조한길 (전남대학교 의과대학 소아과학교실) ;
  • 김수영 (전남대학교 의과대학 소아과학교실) ;
  • 송은송 (전남대학교 의과대학 소아과학교실) ;
  • 이준규 (전남대학교 의과대학 이비인후과학교실) ;
  • 최영륜 (전남대학교 의과대학 소아과학교실)
  • Received : 2009.09.15
  • Accepted : 2009.10.19
  • Published : 2010.01.15
Download PDF
⟨ Previous Next ⟩

Abstract

Lymphangioma is a rare, benign, and hamartomatous tumor of the lymphatic vessels that shows a marked predilection for the head and neck region. When this tumor occurs on the tongue or mouth floor or in the deep neck space, blockage of the efferent lymphatic vessels can result in secondary macroglossia. We report here two patients who showed unusual macroglossia from birth. Initially, there was no noticeable cervical or mandibular swelling. However, mandibular swellings were noted during follow-up examinations, which led to MRI scans on the two infant patients at 5 months and 5 weeks of age, respectively. Subsequently, both patients were diagnosed with lymphangioma or lymphangiohemangioma in the deep neck space.

림프관종은 드물게 발생하는 림프혈관의 양성 과오종으로, 주로 두경부에 호발하는데, 혀나 구저부 또는 심 경부에 발생할 경우 원심성 림프관 흐름의 방해로 이차적 거설증이 발생할 수 있다. 저자들은 출생 시에는 경부 또는 하악 부종 없이 거설증만 보였던 두 명의 환아에서 각각 생후 5개월과 5주에 하악 부종이 보여 MRI로 심 경부 림프관종을 확인한 2례를 경험하였기에 보고하는 바이다.

Keywords

References

  1. Brightman VJ. Diseases of the tongue. In : Lynch HA, editor. Burket's oral medicine. 9th ed. Philadelphia : Lippincott-Raven, 1994:251-7
  2. Stewart RE, Boggs W. Pathology of soft tissues and jaws. In : Stewart RE, Barber TK, Troutman KC, Wei SHY, editors. Pediatric dentistry. St. Louis : CV Mosby Co, 1982:184
  3. Shafer WG, Hine MK, Levy BM. Developmental disturbances of the tongue. A textbook of oral pathology. 4th ed. Philadelphia : WB Saunders Co, 1983:24-5
  4. White MA. Lymphangioma of the tongue: report of a case. ASDC J Dent Child 1987;54:280-2
  5. Rice JP, Carson SH. A case report of lingual lymphangioma presenting as recurrent massive tongue enlargement. Clin Pediatr (Phila)1985;24:47-50 https://doi.org/10.1177/000992288502400111
  6. Lobitz B, Lang T. Lymphangioma of the tongue. Pediatr Emerg Care 1995;11:183-5 https://doi.org/10.1097/00006565-199506000-00012
  7. Deitmer T. Therapy of cystic lymphangioma in childhood. Report of 4 cases with manifestations in the area of the head-neck. Laryngorhinootologie 1996;75:166-70 https://doi.org/10.1055/s-2007-997556
  8. Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and maxillofacial pathology. 1st ed. Philadelphia : WB Saunders Co, 1995:395-7
  9. Regezi JA, Scjubba JJ. Oral Pathology. Clinical pathologic correlations. 3rd ed. Philadelphia : WB Saunders Co, 1999:196-7
  10. Paladini D, Morra T, Guida F, Lamberti A, Martinelli P. Prenatal diagnosis and perniatal management of a lingual lymphangioma. Ultrasound Obstet Gynecol 1998;11:141-3 https://doi.org/10.1046/j.1469-0705.1998.11020141.x
  11. Cable BB, Mair EA. Radiofrequency ablation of lymphagiomatous macroglossia. Laryngoscope 2001;111:1859-61 https://doi.org/10.1097/00005537-200110000-00035
  12. Lalwani AK, Engel TL. Teratoma of the tongue: a case report and review of the literature. Int J Pediatr Otorhinolaryngol 1992;24:261-8 https://doi.org/10.1016/0165-5876(92)90024-J
  13. Seashore JH, Gardiner LJ, Ariyan S. Management of giant cystic hygromas in infants. Am J Surg 1985;149:459-65 https://doi.org/10.1016/S0002-9610(85)80040-4
  14. Regezi JA, Sciubba JJ. Oral pathology. Clinical pathologic correlations. 2nd ed. Philadelphia : WB Saunders Co, 1993: 214-6
  15. Kennedy TL. Cystic hygroma-lymphangioma: a rare and still unclear entity. Laryngoscope 1989;99(10 Pt 2 Suppl 49):1-10
  16. Heether J, Whalen T, Doolin E. Follow-up of complex unresectable lymphangiomas. Am Surg 1994;60:840-1
  17. Hartl DM, Roger G, Denoyelle F, Nicollas R, Triglia JM, Garabedian EN. Extensive lymphangioma presenting with upper airway obstruction. Arch Otolaryngol Head Neck Surg 2000;126:1378-82
  18. Padwa BL, Hayward PG, Ferraro NF, Mulliken JB. Cervicofacial lymphatic malformation: clinical course, surgical intervention and pathogenesis of skeletal hypertrophy. Plast Reconstr Surg 1995;95:951-60 https://doi.org/10.1097/00006534-199505000-00001
  19. Leboulanger N, Roger G, Caze A, Enjolras O, Denoyelle F, Garabedian EN. Utility of radiofrequency ablation for haemorrhagic lingual lymphangioma. Int J Pediatr Otorhinolaryngol 2008;72:953-8 https://doi.org/10.1016/j.ijporl.2008.03.013

Cited by

  1. The effects of macroglossia on speech: A case study vol.26, pp.1, 2010, https://doi.org/10.3109/02699206.2011.588370