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Sclerosing agents in the management of lymphatic malformations in children: A systematic review
Sairvan Fernandes1, Phillip Yeung1, Manraj Heran2
1University of British Columbia, Vancouver, B.C., Canada.
Insights
Sclerotherapy effectively treats pediatric lymphatic malformations (LMs), with macrocystic types showing higher success rates. Complication rates are low, but microcystic LMs have modest outcomes, warranting further research.
Area of Science:
- Pediatric Surgery
- Vascular Anomalies
- Minimally Invasive Therapies
Background:
- Lymphatic malformations (LMs) are congenital vascular anomalies.
- Sclerotherapy is a common treatment modality for LMs.
- Various sclerosants and techniques exist, with limited comparative data in pediatric populations.
Purpose of the Study:
- To review the efficacy and safety of sclerotherapy agents for pediatric lymphatic malformations.
- To analyze resolution rates and complication profiles of different sclerosants.
Main Methods:
- PRISMA-guided systematic review of multiple databases.
- Inclusion of studies on pediatric patients (0-18 years) treated with injection sclerotherapy for LMs.
- Data extraction on sclerosant agent, dose, site, complications, and resolution rates.
Main Results:
- Eighty-eighty-six patients from 48 studies were analyzed, with an overall success rate of 89%.
- Macrocystic LMs demonstrated higher resolution rates (89%) compared to mixed (71%) and microcystic (34%) variants.
- OK-432, bleomycin, and doxycycline showed varying complete regression rates for head/neck LMs, with OK-432 associated with more major complications.
Conclusions:
- Sclerotherapy is a low-complication treatment for pediatric LMs.
- Macrocystic LMs respond favorably, while microcystic LMs have modest outcomes.
- Agent use varies globally; further prospective studies are needed to optimize treatment.
Purpose:
Sclerotherapy is frequently employed in treating lymphatic malformations (LMs), and multiple agents, practitioners and strategies exist. This review investigates the reported efficacy and safety of sclerosants in the pediatric population.
Methods:
Adhering to PRISMA guidelines, multiple databases were queried without linguistic or temporal restriction. Inclusion criteria were patients aged 0-18 exclusively receiving injection sclerotherapy for the treatment of LMs with follow-up data. Data abstracted included agent, dose, anatomic site and key outcome measures including complications (major/minor) and resolution rates (>95% reduction in volume). Critical appraisal was undertaken using the MINORS tool.
Results:
Forty-eight studies met the inclusion criteria with a mean MINORS score of 0.65 ± 0.08. Included studies yielded 886 patients, across nearly 30 years. The overall observed rate of success was 89%, with variable follow-up across publications (6 weeks - 10 years). Most reported LMs were macrocystic (82%) and had a higher resolution rate than mixed/microcytic variants (89%, 71%, 34%, p<0.01) For head/neck LMs, rates of complete regression for OK-432, bleomycin, and doxycycline were 67% ± 27% (n = 26), 91% ± 53% (n = 34) and 85% ± 16% (n = 52) respectively. Major complications were most commonly reported with OK-432, including airway compromise or subsequent operation.
Conclusions:
In pediatric patients treated for LM by sclerotherapy, complication rates were low. Macrocystic lesions respond well but success rates were modest at best for microcystic disease. Differences in agent utilization were noted between high and low resourced contexts; despite its lack of federal approval, OK-432 was the most reported agent. Further prospective research is warranted. LOE: 3a.
