Literature DB >> 29026809

Spontaneously Resolved Macrocystic Lymphatic Malformations: Predictive Variables and Outcomes.

Michael J Phang1, Douglas J Courtemanche1,2, Marija Bucevska1, Claudia Malic3, Jugpal S Arneja1,2.   

Abstract

INTRODUCTION: Lymphatic malformations are benign, low-flow vascular malformations that typically present at or near birth. Due to morbidity associated with operative treatment, nonoperative treatment with injection of sclerosant has become the mainstay of therapy. Over the past 15 years, several patients at our centre with macrocystic (>2 cm cyst size) lymphatic malformations have seen their lesions resolve spontaneously while awaiting treatment. In this study, we review features of these patients that may contribute to spontaneous resolution.
METHOD: A retrospective chart review was conducted from our Vascular Anomalies Clinic database (1999-2014) of all macrocystic lymphatic malformations; characteristics of patients with spontaneous resolution were reviewed.
RESULTS: Of 61 patients with macrocystic lymphatic malformations, 7 cases (11.5%; 4 females, 3 males) resolved spontaneously. Median age at malformation appearance was 2 years (range: 0-6.5 years), with median age at resolution of 4 years (range: 10 months-7 years). Median time from appearance to resolution was 24 months (range: 3-43 months), with a median follow-up time of 4 years (range: 1-15 years). All but 1 case was associated with local or upper respiratory tract infection antecedent to resolution. Six of the 7 lesions were located in the neck.
CONCLUSION: Among the cases reviewed, there was a common theme of upper respiratory tract or local infection antecedent to spontaneous lesion resolution. Compared to the literature, our proportion of malformations presenting after birth and the proportion of malformations presenting in the neck region were higher than those of other series. Although side effects associated with treatment are generally mild and/or rare, risks related to sclerotherapy and the accompanying requirement for general anesthesia in pediatric populations nevertheless exist. As the median time from lesion appearance to resolution was 24 months, it may be reasonable to observe these malformations for up to 24 months before proceeding with treatment if the lesion does not impair function and disfigurement is not considerable, particularly if the lesion presents after birth and/or is located in the neck region.

Entities:  

Keywords:  macrocystic lymphatic malformations; treatment

Year:  2017        PMID: 29026809      PMCID: PMC5626187          DOI: 10.1177/2292550317693815

Source DB:  PubMed          Journal:  Plast Surg (Oakv)        ISSN: 2292-5503            Impact factor:   0.947


  18 in total

1.  Lymphatic malformations: clinical course and management in 64 cases.

Authors:  Marcia Hogeling; Susan Adams; Jenaleen Law; Orli Wargon
Journal:  Australas J Dermatol       Date:  2011-06-09       Impact factor: 2.875

2.  Spontaneous resolution of lymphatic and venous malformations.

Authors:  C Xavier; S Emil
Journal:  Eur J Pediatr Surg       Date:  2010-01-22       Impact factor: 2.191

3.  Involution of a cystic hygroma of the face following local infection.

Authors:  Bárbara Nader Vasconcelos; Marcela Duarte Villela Benez; Aline Lopes Bressan; Elisa Fontenelle de Oliveira
Journal:  An Bras Dermatol       Date:  2011 Jan-Feb       Impact factor: 1.896

4.  Cystic hygroma of the head and neck--a long-term follow-up of 44 cases.

Authors:  B Charabi; P Bretlau; M Bille; M Holmelund
Journal:  Acta Otolaryngol Suppl       Date:  2000

Review 5.  Vascular malformations.

Authors:  Jugpal S Arneja; Arun K Gosain
Journal:  Plast Reconstr Surg       Date:  2008-04       Impact factor: 4.730

Review 6.  Vascular anomalies: hemangiomas and beyond--part 2, Slow-flow lesions.

Authors:  Gerald G Behr; Craig M Johnson
Journal:  AJR Am J Roentgenol       Date:  2013-02       Impact factor: 3.959

7.  Cystic hygroma of the head and neck. A review of 37 cases.

Authors:  P J Emery; C M Bailey; J N Evans
Journal:  J Laryngol Otol       Date:  1984-06       Impact factor: 1.469

8.  Lymphangioma. An otolaryngologic perspective.

Authors:  M E Brock; R J Smith; S E Parey; D L Mobley
Journal:  Int J Pediatr Otorhinolaryngol       Date:  1987-12       Impact factor: 1.675

9.  Efficacy and safety of OK-432 immunotherapy of lymphatic malformations.

Authors:  Mark C Smith; M Bridget Zimmerman; Diane K Burke; Nancy M Bauman; Yutaka Sato; Richard J H Smith
Journal:  Laryngoscope       Date:  2009-01       Impact factor: 3.325

Review 10.  Treatment of lymphatic malformations with OK-432 (Picibanil): review of the literature.

Authors:  Michelle T Poldervaart; Corstiaan C Breugem; Lucienne Speleman; Suzanne Pasmans
Journal:  J Craniofac Surg       Date:  2009-07       Impact factor: 1.046

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  2 in total

1.  Neonatal venolymphatic malformation with spontaneous parotid duct rupture: first case report.

Authors:  Darshanika C T Gamage; Bernard Deepal Wanniarachchi Jayamanne; Syed Faizan Quasim; Kosmos Kailidis; Anas Olabi
Journal:  Oxf Med Case Reports       Date:  2022-06-23

2.  Clinical and ultrasound characteristics of pediatric lateral neck masses.

Authors:  Nemanja Rankovic; Jovana Todorovic; Radoje Simic
Journal:  PLoS One       Date:  2021-05-12       Impact factor: 3.240

  2 in total

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