Top

European Archives of Oto-Rhino-Laryngology

Open Access 07-05-2024 | Sclerotherapy | Review Article

Surgery versus sclerotherapy versus combined therapy in head and neck lymphatic malformations in the pediatric population: systematic review and meta-analysis

Authors: Jurriën L. A. Embrechts, Steven Hiddinga, Joseph C. Bot, Jan-Jaap Hendrickx, Rik van Eekelen, Johannes C. F. Ket, C. René Leemans, Remco de Bree

Published in: European Archives of Oto-Rhino-Laryngology

Abstract

Purpose

To systematically review current literature on the treatment of lymphatic malformations (LMs) of the head and neck to guide treatment strategy.

Methods and materials

A systematic review and meta-analysis of literature until 16 November 2021 was performed on treatments of LMs in the head and neck.

Results

Out of 9044 articles, 54 studies were eligible for inclusion with 26 studies providing detailed participant data. A total number of 1573 patients with a mean age of 21.22 months were analysed. Comparative meta-analysis did not reveal significant differences two proportions of volume reduction (≥ 50% and 100%) between sclerotherapy and surgical treatment. Regression demonstrated that positive predictors for volume reduction were surgery 17 (95% CI 0.26–34; p = 0.047) and treatment of macrocystic lesions 19 (95% CI 5.5–32; p = 0.006). Treatment of mixed lesions also demonstrated a trend towards achieving a greater volume reduction (p = 0.052). A higher de Serres stage of the lesion had a negative effect on the amount of volume reduction − 3.7 (95% CI − 7.0 to − 0.35; p = 0.030).

Conclusion

This comprehensive meta-analysis demonstrated no significant difference in volume reduction between various treatment modalities at study level. However, individual patient data indicated that surgery and larger cyst types are associated with a significant higher percentage of volume reduction, whereas a higher de Serres stage negatively impacted the amount of volume reduction. These findings can be used for patient counseling and treatment planning based on cyst type and de Serres stage. However volume reduction constitutes just one objective within a more complex treatment spectrum.

Available only for authorised users

National Organisation for Rare Disorders. Lymphatic Malformations [internet]. 2022 [updated 2021; cited 2022 May 5]. Available from https://rarediseases.org/rare-diseases/lymphatic-malformations

Hogeling M, Adams S, Law J, Wargon O (2011) Lymphatic malformations: clinical course and management in 64 cases. Austral J Dermatol 52:186–190. https://doi.org/10.1111/j.1440-0960.2011.00777.xCrossRef

Wassef M, Blei F, Adams D et al (2015) Vascular anomalies classification: recommendations from the international society for the study of vascular anomalies. Pediatrics 136:E203–E214. https://doi.org/10.1542/peds.2014-3673CrossRefPubMed

Cystic hygroma. Lancet. 1990;335(8688):511. https://doi.org/10.1016/0140-6736(90)90743-O

Malic CC, Guilfoyle R, Courtemanche RJM et al (2017) Lymphatic malformation architecture: implications for treatment with OK-432. J Craniofac Surg 28:1721–1724. https://doi.org/10.1097/SCS.0000000000003789CrossRefPubMed

Zhou Q, Zheng JW, Mai HM et al (2011) Treatment guidelines of lymphatic malformations of the head and neck. Oral Oncol 47:1105–1109. https://doi.org/10.1016/j.oraloncology.2011.08.001CrossRefPubMed

Miceli A, Stewart KM (2022) Lymphangioma. In: StatPearls. StatPearls Publishing, Treasure Island

Jin L, Chen J, Li X (2017) Surgical excision with bleomycin irrigation: a better primary treatment choice for pediatric submandibular lymphatic malformations. J Oral Maxillofac Surg 75:437.e1-437.e7. https://doi.org/10.1016/j.joms.2016.09.041CrossRefPubMed

10.

Bhattacharyya NC, Yadav K, Mitra SK, Pathak IC (1981) Lymphangiomas in children. Aust N Z J Surg 51:296–300. https://doi.org/10.1111/j.1445-2197.1981.tb05961.xCrossRefPubMed

11.

Saijo M, Munro IR, Mancer K (1975) Lymphangioma. A long-term follow-up study. Plast Reconstr Surg 56:642–651. https://doi.org/10.1097/00006534-197512000-00005CrossRefPubMed

12.

Emery PJ, Bailey CM, Evans JN (1984) Cystic hygroma of the head and neck. A review of 37 cases. J Laryngol Otol 98:613–619CrossRefPubMed

13.

Balakrishnan K, Menezes MD, Chen BS et al (2014) Primary surgery vs primary sclerotherapy for head and neck lymphatic malformations. JAMA Otolaryngol Head Neck Surg 140:41–45. https://doi.org/10.1001/jamaoto.2013.5849CrossRefPubMed

14.

Shiels WE 2nd, Kang DR, Murakami JW et al (2009) Percutaneous treatment of lymphatic malformations. Otolaryngol Head Neck Surg 141:219–224. https://doi.org/10.1016/j.otohns.2009.04.001CrossRefPubMed

15.

de Serres LM, Sie KC, Richardson MA (1995) Lymphatic malformations of the head and neck. A proposal for staging. Arch Otolaryngol Head Neck Surg 121:577–582. https://doi.org/10.1001/archotol.1995.01890050065012CrossRefPubMed

16.

Ueno S, Fujino A, Morikawa Y et al (2019) Indications for tracheostomy in children with head and neck lymphatic malformation: analysis of a nationwide survey in Japan. Surg Today 49:410–419. https://doi.org/10.1007/s00595-018-1755-3CrossRefPubMed

17.

Sjogren PP, Arnold RW, Skirko JR, Grimmer JF (2017) Anatomic distribution of cervicofacial lymphatic malformations based on lymph node groups. Int J Pediatr Otorhinolaryngol 97:72–75. https://doi.org/10.1016/j.ijporl.2017.02.030CrossRefPubMed

18.

Mulliken JB, Burrows PE, Fishman SJ, Mulliken JB (2013) Mulliken and Young’s vascular anomalies: Hemangiomas and malformations, 2nd edn. Oxford University Press, OxfordCrossRef

19.

Broomhead IW (1964) Cystic hygroma of the head and neck. Br J Plast Surg 17:225–244CrossRefPubMed

20.

Smith RJH (2004) Lymphatic malformations. Lymph Res Biol 2:25–31. https://doi.org/10.1089/1539685041690436CrossRef

21.

Nehra D, Jacobson L, Barnes P et al (2008) Doxycycline sclerotherapy as primary treatment of head and neck lymphatic malformations in children. J Pediatr Surg 43:451–460. https://doi.org/10.1016/j.jpedsurg.2007.10.009CrossRefPubMed

22.

Perkins JA, Manning SC, Tempero RM et al (2010) Lymphatic malformations: review of current treatment. Otolaryngol Head Neck Surg 142:795-8031.e1. https://doi.org/10.1016/j.otohns.2010.02.026CrossRefPubMed

23.

Wang S, Zhang J, Ge W et al (2017) Efficacy and safety of oral sildenafil in treatment of pediatric head and neck lymphatic malformations. Acta Otolaryngol 137:674–678. https://doi.org/10.1080/00016489.2016.1272135CrossRefPubMed

24.

De Maria L, De Sanctis P, Balakrishnan K et al (2020) Sclerotherapy for lymphatic malformations of head and neck: systematic review and meta-analysis. J Vasc Surg Venous Lymphat Disord 8:154–164. https://doi.org/10.1016/j.jvsv.2019.09.007CrossRefPubMed

25.

Cheng J, Bastidas N (2016) Considerations for management of head and neck lymphatic malformations in children. J Craniofac Surg 27:908–912. https://doi.org/10.1097/SCS.0000000000002621CrossRefPubMed

26.

Adams MT, Saltzman BS, Perkins JA (2011) Treatment of lymphatic malformations: a systematic review. Otolaryngol Head Neck Surg 145:249. https://doi.org/10.1177/0194599811415823a374CrossRef

27.

Wang Y, Tang W, Li X (2020) Safety and efficacy of surgery combined with bleomycin irrigation for complex cervical-facial lymphatic malformations of children. Int J Pediatr Otorhinolaryngol 128:109724. https://doi.org/10.1016/j.ijporl.2019.109724CrossRefPubMed

28.

Ghaffarpour N, Burgos CM, Wester T (2018) Surgical excision is the treatment of choice for cervical lymphatic malformations with mediastinal expansion. J Pediatr Surg 53:1820–1824. https://doi.org/10.1016/j.jpedsurg.2017.10.048CrossRefPubMed

29.

Dindo D, Demartines N, Clavien P-A (2004) Classification of surgical complications: a new proposal with evaluation in a cohort of 6336 patients and results of a survey. Ann Surg 240:205–213. https://doi.org/10.1097/01.sla.0000133083.54934.aeCrossRefPubMedPubMedCentral

30.

Balduzzi S, Rücker G, Schwarzer G (2019) How to perform a meta-analysis with R: a practical tutorial. Evid Based Mental Health 22:153–160. https://doi.org/10.1136/ebmental-2019-300117CrossRef

31.

Giguère CM, Bauman NM, Sato Y et al (2002) Treatment of lymphangiomas with OK-432 (Picibanil) sclerotherapy: a prospective multi-institutional trial. Arch Otolaryngol Head Neck Surg 128:1137–1144. https://doi.org/10.1001/archotol.128.10.1137CrossRefPubMed

32.

Smith MC, Zimmerman MB, Burke DK et al (2009) Efficacy and safety of OK-432 immunotherapy of lymphatic malformations. Laryngoscope 119:107–115CrossRefPubMed

33.

Greinwald JH Jr, Burke DK, Sato Y et al (1999) Treatment of lymphangiomas in children: an update of Picibanil (OK-432) sclerotherapy. Otolaryngol Head Neck Surg 121:381–387CrossRefPubMed

34.

Kim DW (2014) OK-432 sclerotherapy of lymphatic malformation in the head and neck: Factors related to outcome. Pediatr Radiol 44:857–862. https://doi.org/10.1007/s00247-014-2889-0CrossRefPubMed

35.

Bonilla-Velez J, Moore BP, Cleves MA et al (2020) Surgical resection of macrocystic lymphatic malformations of the head and neck: Short and long-term outcomes. Int J Pediatr Otorhinolaryngol 134:110013. https://doi.org/10.1016/j.ijporl.2020.110013CrossRefPubMed

36.

Cahill AM (2011) Vascular malformations in children. Cardiovasc Intervent Radiol 34:471–474. https://doi.org/10.1007/s00270-011-0216-9CrossRef

37.

Bajaj Y, Hewitt R, Ifeacho S, Hartley BEJ (2011) Surgical excision as primary treatment modality for extensive cervicofacial lymphatic malformations in children. Int J Pediatr Otorhinolaryngol 75:673–677. https://doi.org/10.1016/j.ijporl.2011.02.009CrossRefPubMed

38.

Ghaffarpour N, Petrini B, Svensson LA et al (2015) Patients with lymphatic malformations who receive the immunostimulant OK-432 experience excellent long-term outcomes. Acta Paediatr Int J Paediatr 104:1169–1173CrossRef

39.

Chen WL, Liao JK, Zhou B et al (2020) Comprehensive treatment of massive macroglossia due to venous and lymphatic malformations. Int J Oral Maxillofac Surg 49:874–881. https://doi.org/10.1016/j.ijom.2020.01.005CrossRefPubMed

40.

Aluffi Valletti P, Brucoli M, Boffano P et al (2020) A single-center experience in the management of head and neck lymphangiomas. Oral Maxillofac Surg 24:109–115. https://doi.org/10.1007/s10006-020-00832-zCrossRefPubMed

41.

Ghaffarpour N, Claesson G, Wester T, Boman KK (2019) Long-term health-related quality of life in children with lymphatic malformations treated with sclerotherapy generally matched age-appropriate standardised population norms. Acta Paediatr (Oslo, Norway: 1992) 108:1499–1506. https://doi.org/10.1111/apa.14700CrossRef

42.

Horbach SER, Rongen APM, O TM et al (2018) Outcome measurement for vascular malformations of the head and neck. Otolaryngol Clin North Am 51:111–117. https://doi.org/10.1016/j.otc.2017.09.014CrossRefPubMed

43.

Horbach SER, van de Ven JS, Nieuwkerk PT et al (2018) Patient-reported outcomes of bleomycin sclerotherapy for low-flow vascular malformations and predictors of improvement. Cardiovasc Intervent Radiol 41:1494–1504. https://doi.org/10.1007/s00270-018-1999-8CrossRefPubMedPubMedCentral

44.

Jamal N, Ahmed S, Miller T et al (2012) Doxycycline sclerotherapy for pediatric head and neck macrocystic lymphatic malformations: a case series and review of the literature. Int J Pediatr Otorhinolaryngol 76:1127–1131. https://doi.org/10.1016/j.ijporl.2012.04.015CrossRefPubMed

45.

Rozman Z, Thambidorai CR, Zaleha AM et al (2011) Lymphangioma: is intralesional bleomycin sclerotherapy effective? Biomed Imaging Intervent J 7:1–8. https://doi.org/10.2349/biij.7.3.e18CrossRef

46.

Tu JH, Do HM, Patel V et al (2017) Sclerotherapy for lymphatic malformations of the head and neck in the pediatric population. J NeuroIntervent Surg 9:1023–1026. https://doi.org/10.1136/neurintsurg-2016-012660CrossRef

Title: Surgery versus sclerotherapy versus combined therapy in head and neck lymphatic malformations in the pediatric population: systematic review and meta-analysis
Authors: Jurriën L. A. Embrechts
Steven Hiddinga
Joseph C. Bot
Jan-Jaap Hendrickx
Rik van Eekelen
Johannes C. F. Ket
C. René Leemans
Remco de Bree
Publication date: 07-05-2024
Publisher: Springer Berlin Heidelberg
Keyword: Sclerotherapy
Published in: European Archives of Oto-Rhino-Laryngology
Print ISSN: 0937-4477
Electronic ISSN: 1434-4726
DOI: https://doi.org/10.1007/s00405-024-08661-6

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Surgery versus sclerotherapy versus combined therapy in head and neck lymphatic malformations in the pediatric population: systematic review and meta-analysis

Abstract

Purpose

Methods and materials

Results

Conclusion

Keynote webinar | Spotlight on medication adherence

Springer Medicine

Abstract

Purpose

Methods and materials

Results

Conclusion

Please log in to get access to this content