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Hwang, Lee, Lim, Lee, and Seo: Surgical Treatment of Difficult Cervicofacial Lymphangioma in Children
Original Article

Journal of the Korean Association of Pediatric Surgeons 2015; 21(2): 17-23.

Published online: 22 December 2015

DOI: https://doi.org/10.13029/jkaps.2015.21.2.17

Surgical Treatment of Difficult Cervicofacial Lymphangioma in Children

Deokbi Hwang1, Sanghoon Lee1, So Young Lim2, Suk-Koo Lee1, Jeong-Meen Seo1

1Division of Pediatric Surgery, Department of Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea.

2Department of Plastic Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea.

Correspondence: Jeong-Meen Seo. Department of Surgery, Samsung Medical Center, Sungkyunkwan University School of Medicine, 81 Irwon-ro, Gangnam-gu, Seoul 06351, Korea. Tel: +82-2-3410-0282, Fax: +82-2-3410-0040, jm0815.seo@samsung.com

Received 28 July 2015       Revised 21 September 2015       Accepted 12 October 2015

Copyright © 2015 by the Korean Association of Pediatric Surgeons

(open-access):

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract

Purpose

Cervical lymphangiomas are rare lymphovascular malformations arising in the neck, which form huge fluid-containing cysts. Treatment of the malformation consists of surgery and sclerotherapy. However, the optimal approach is still controversial. Here, we describe a series of cervical lymphangiomas which have been treated with surgical approaches.

Methods

We retrospectively investigated the medical records of 82 patients who had been diagnosed with cervicofacial lymphangioma from 2001 to 2012 in our center. A closed suction drainage with negative pressure was placed on the operative lesion following excision to prevent reaccumulation of lymphatic fluid and the drainage tube was removed after injecting OK-432 through the tube.

Results

Twelve patients underwent surgical excision of cervical lymphangioma. The median patient age was 3 months at the time of the operation. The patients have been followed-up over a period of 34 months. When lesions were located near vital organs such as the trachea or carotid artery or did not respond to repetitive OK-432 injections, surgical treatment might bring good outcomes. However, swallowing difficulty, lip palsy, or dyslalia due to adjacent nerve damage temporarily appeared as postoperative complications. Five children had tracheostomy due to tracheal or subglottic stenosis and 2 patients had gastrostomy due to aspiration while they eat after surgery.

Conclusion

Surgery for cervicofacial lymphangioma should be conducted carefully in selective cases. A well thought-out surgical plan with a multidisciplinary surgical team approach and placement of closed suction drainage tube after surgery and adjuvant OK-432 sclerotherapy through drainage tube seem to be helpful for good outcome.

Keywords: Cervical lymphangioma, Lymphatic malformation, OK-432 sclerotherapy, Excision, Suction drainage

Figures and Tables

Fig. 1
(A) Patient No. 2 was diagnosed with cervical lymphangioma prenatally by fetal MRI. (B) The patient was about to have a surgery 18 days after birth through EXIT procedure. (C) This picture was taken 1 year and 8 months after surgery. EXIT, ex utero intrapartum treatment.
jkaps-21-17-g001
Fig. 2
(A) Patient No. 4 was transferred to our hospital after OK-432 sclerotherapy. (B) This patient had a surgery about lymphangioma involving from right temporal area to neck. (C) This photograph was taken 9 months after surgery and a subsequent sclerotherapy.
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Fig. 3
(A) Patient No. 6 visited our hospital after having gotten several times of sclerotherapies using OK-432 and ethanol. (B) This photograph was taken 3 years after surgery.
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Fig. 4
(A) MRI shows macrocystic lymphatic malformations which are involving from right neck to axilla before OK-432 sclerotherapy (patient No. 9). (B) Intralesional hemorrhage happened one month after sclerotherapy.
jkaps-21-17-g004
Fig. 5
(A) Sagittal T2-weighted MRI shows cervical lymphangioma which are infiltrating tongue base (patient No. 11). (B) Decreased lymphatic malformation after surgery.
jkaps-21-17-g005
Table 1
Preoperative Characteristics of 12 Patients with Cervical Lymphangioma and perioperative Details
jkaps-21-17-i001

POD, postoperative day; EXIT, ex utero intrapartum treatment; RFA, radiofrequency ablation.

a)Median 300 min, range 59-559 min. b)Mean 19 days, range 7-46 days.

Table 2
Distribution of Our Patients according to the Anatomic Location of Lymphangiomas
jkaps-21-17-i002

Notes

CONFLICTS OF INTEREST No potential conflict of interest relevant to this article was reported.

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