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     LETTER TO THE EDITOR

Complete Resolution of Cystic Hygroma with Single Session of Intralesional Bleomycin


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Authors:  Muhammad Sharif,* Isam Elamin Elsiddig, Fadi Atwan

Institute: Department of Pediatric Surgery, King Fahad Hospital Albaha, KSA.

Address for Correspondence: Dr. Muhammad Sharif, Department of Pediatric Surgery, King Fahad Hospital Albaha, KSA.

*Email: docsharif@yahoo.com

* Corresponding Author

 


ISSN:2226-0439

Journal of Neonatal Surgery

Volume 1(3), Jul-Sep 2012


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Submitted On: 08-06-2012

Accepted On: 11-06-2012

 Published on: 01-07-2012

Local ID: jns-1-61

J Neonat Surg 2012; 1(3): 44

© Sharif et al, 2012

Conflict of Interest: None declared

Source of Support: Nil

 


How to cite:

Sharif M, Elsiddig IE, Atwan F. Complete resolution of cystic hygroma with single session of intralesional bleomycin. J Neonat Surg 2012; 1: 44.

 


 

DEAR SIR

Intralesional sclerotherapy as a primary modality of management for cystic hygroma is successfully described in literature. It has many benefits over surgical approach; recurrence being the concern of as much as 20% of patients in which apparent complete excision has been performed. Bleomycin is one of sclerosing agents used as intralesional therapy in cystic hygroma. Complete response usually occurs in multiple sessions of sclerotherapy. Rarely, complete resolution occurs with single session of bleomycin sclerotherapy [1-7]. We share our experience of managing a case of cystic hygroma of neck that completely resolved with single session of bleomycin sclerotherapy.

A 5-day-old male neonate presented with swelling in left side of neck since birth. Swelling was painless and multi-cystic. Transillumination of the swelling was positive. Clinical diagnosis was cystic hygroma of the neck. Ultrasound and CT scan were performed to see the extent of the cystic hygroma. He was kept on follow-up for 1 month when he was again admitted. Aspiration with wide bore needle was done under G/A, followed by intralesional bleomycin sclerotherapy in three quadrant (dose 0.3 mg/kg). Post sclerotherapy course was uneventful except initial increase in size and serous discharge from swelling for 15 days. Later on, all symptoms relieved and at 3 months follow-up, the cystic hygroma had completely resolved leaving behind only a small pigmented area (Fig. 1,2).

 

Figure 1: Cystic hygroma before sclerotherapy

 

Figure 2: At three months follow-up.

 

Cystic hygroma is a macrocystic type of  lymphatic malformation which is benign and asymptomatic lesion in most of the cases, however, various types of complications can arise in it i.e. recurrent bouts of infection in the lesion, respiratory distress, dysphagia, hemorrhage inside cystic hygroma, sudden increase in the size of lesion, lymph discharging sinus and disfigurement etc. The respiratory distress can be of severe nature necessitating a tracheostomy due to complete or significant laryngeal or tracheal compressions. Intralesional sclerotherapy is an effective treatment modality for the management of cystic hygroma. Surgical excision of the complex cystic hygroma of neck, involving deep structures, is difficult and demanding a meticulous technique and care to avoid per-operative complications. Moreover, recurrence and lymphatic sinus are other problems associated with surgery. Intralesional sclerotherapy with bleomycin or OK-432 is an effective alternate to surgery. With sclerotherapy, complete resolution occurs in about 60% of cases. The sclerotherapy has to be performed multiple times before complete resolution [1-7]. In our case, however, complete resolution occurred with single session of intralesional bleomycin sclerotherapy which is very rare and unusual. We cannot conclude specific reason for complete resolution in our case based on a single case. Proper studies should be designed to identify factors that may help in complete resolution with less sessions of sclerotherapy.


REFERENCES

  1. Mirza B, Ijaz L, Saleem M, Sharif M, Sheikh A. Cystic hygroma: An overview. J Cutan Aesthet Surg. 2010;3:139-44.

  2. Okada A, Kubota A, Fukuzawa M, Imura K, Kamata S. Injection of bleomycin as a primary therapy of cystic lymphangioma. J Pediatr Surg. 1992;27:440-3.

  3. Ibrahim AH, Kandeel A, Bazeed MF. Successful non surgical management of a huge life threatening cervicomediastinal cystic hygroma case report and review of the literature. J Pediatr Surg Specialt. 2009;3:48-50.

  4. Mirza B, Ijaz L, Iqbal S, Mustafa G, Saleem M, Sheikh A. Cystic hygroma of unusual sites: Report of two cases. Afr J Paediatr Surg. 2011;8:85-8.

  5. Ogita S, Tsuto T, Nakamura K, Deguchi E, Tokiwa K, Iwai N. OK-432 therapy for lymphangioma in children: Why and how does it work? J Pediatr Surg. 1996;31:477-80.

  6. Mirza B, Ijaz L, Saleem M, Sheikh A. Different modalities used to treat concurrent lymphangioma of chest wall and scrotum. J Cutan Aesthet Surg. 2010;3:189-90.

  7. Rattan KN, Kajal P, Kadian YS, Gupta R. Haemorrhage in a scrotal lymphangioma in a child: A rarity. Afr J Paediatr Surg .2009;6:110-1.


This is an Open Access article distributed under the terms of the Creative Commons Attribution unported License (http://creativecommons.org/licenses/by/3.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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