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13.07.2020 | Original Article

Lymphatic malformations compromising the upper airway in children: ultrasound-guided intralesional focal sclerotherapy with bleomycin targeting culprit lesions

verfasst von: Hidehito Usui, Masato Shinkai, Norihiko Kitagawa, Kyoko Mochizuki, Issei Kawakita, Yukihiro Tsuzuki, Shota Shinohara, Yuma Yagi, Munetaka Masuda

Erschienen in: Pediatric Surgery International | Ausgabe 9/2020

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Abstract

Purpose

Lymphatic malformations (LMs) compromising the upper airway is a life-threatening and intractable disease. Here, we establish a novel method to perform intralesional focal sclerotherapy targeting the culprit for airway stenosis.

Methods

Between July 2015 and February 2020, 11 patients with airway-compromising LMs were enrolled. To yield maximal effects on the compromised airway with minimal adverse effects, ultrasound-guided intralesional bleomycin sclerotherapy assisted by balloon was performed, aimed at the most responsible lesion around the airway. A retrospective analysis was performed.

Results

Ten patients presented with respiratory symptoms, eight of whom required airway support. The last asymptomatic patient showed airway compression on magnetic resonance imaging. The dose of bleomycin injected ranged from 1.3–9 mg per patient per course. A median of one course was required for withdrawal from airway support, and the median time was 15 days. A median of two courses was required to eliminate the lesion adjacent to the airway, which would have potential risk of airway stenosis. No complications were observed.

Conclusions

Our intralesional focal sclerotherapy technique with bleomycin targeting the culprit lesion is dose-sparing, safe, and effective in achieving rapid shrinkage of LMs compromising the upper airway in children, thereby avoiding tracheostomy.

Kennedy TL, Whitaker M, Pellitteri P et al (2001) Cystic hygroma/lymphangioma: a rational approach to management. Laryngoscope 111:1929–1937. https://doi.org/10.1097/00005537-200111000-00011 CrossRefPubMed

Florez-Vargas A, Vargas SO, Debelenko LV et al (2008) Comparative analysis of D2–40 and LYVE-1 immunostaining in lymphatic malformations. Lymphology 41:103–110PubMed

Bruder E, Perez-Atayde AR, Jundt G et al (2009) Vascular lesions of bone in children, adolescents, and young adults. A clinicopathologic reappraisal and application of the ISSVA classification. Virchows Arch 454:161–179. https://doi.org/10.1007/s00428-008-0709-3 CrossRefPubMed

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.2014-3673 CrossRefPubMed

Tm O, Rickert SM, Diallo AM et al (2013) Lymphatic malformations of the airway. Otolaryngol Head Neck Surg 149:156–160. https://doi.org/10.1177/0194599813485065 CrossRef

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/s00595-018-1755-3 CrossRefPubMed

Oomen KP, Paramasivam S, Waner M et al (2016) Endoscopic transmucosal direct puncture sclerotherapy for management of airway vascular malformations. Laryngoscope 126:205–211. https://doi.org/10.1002/lary.25284 CrossRefPubMed

Gaffuri M, Torretta S, Iofrida E et al (2019) Multidisciplinary management of congenital giant head and neck masses: our experience and review of the literature. J Pediatr Surg 54:733–739. https://doi.org/10.1016/j.jpedsurg.2018.09.018 CrossRefPubMed

Legiehn GM, Heran MK (2008) Venous malformations: classification, development, diagnosis, and interventional radiologic management. Radiol Clin North Am 46:545–597. https://doi.org/10.1016/j.rcl.2008.02.008 CrossRefPubMed

10.

Colletti G, Valassina D, Bertossi D et al (2014) Contemporary management of vascular malformations. J Oral Maxillofac Surg 72:510–528. https://doi.org/10.1016/j.joms.2013.08.008 CrossRefPubMed

11.

Defnet AM, Bagrodia N, Hernandez SL et al (2016) Pediatric lymphatic malformations: evolving understanding and therapeutic options. Pediatr Surg Int 32:425–433CrossRef

12.

Schoinohoriti OK, Theologie-Lygidakis N, Tzerbos F et al (2012) Lymphatic malformations in children and adolescents. J Craniofac Surg 23:1744–1747CrossRef

13.

Bajaj Y, Hewitt R, Ifeacho S et al (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.2011.02.009 CrossRefPubMed

14.

Berg EE, Sobol SE, Jacobs I (2013) Laryngeal obstruction by cervical and endolaryngeal lymphatic malformations in children: proposed staging system and review of treatment. Ann Otol Rhinol Laryngol 122:575–581. https://doi.org/10.1177/000348941312200907 CrossRefPubMed

15.

Adams MT, Saltzman B, Perkins JA (2012) Head and neck lymphatic malformation treatment: a systematic review. Otolaryngol Head Neck Surg 147:627–639. https://doi.org/10.1177/0194599812453552 CrossRefPubMed

16.

Balakrishnan K, Edwards TC, Perkins JA (2012) Functional and symptom impacts of pediatric head and neck lymphatic malformations: developing a patient-derived instrument. Otolaryngol Head Neck Surg 147:925–931. https://doi.org/10.1177/0194599812450838 CrossRefPubMed

17.

Manning SC, Perkins J (2013) Lymphatic malformations. Curr Opin Otolaryngol Head Neck Surg 21:571–575. https://doi.org/10.1097/moo.0000000000000007 CrossRefPubMed

18.

Simon BM, Fowler SM, Handler SD (1983) Communication development in young children with long-term tracheostomies: preliminary report. Int J Pediatr Otorhinolaryngol 6:37–50. https://doi.org/10.1016/S0165-5876(83)80102-5 CrossRefPubMed

19.

DeMauro SB, D'Agostino JA, Bann C et al (2014) Developmental outcomes of very preterm infants with tracheostomies. J Pediatr 164:1203–1310. https://doi.org/10.1016/j.jpeds.2013.12.014 CrossRef

20.

Kamijo A, Hatsushika K, Kanemaru S et al (2013) Five adult laryngeal venous malformation cases treated effectively with sclerotherapy. Laryngoscope 123:2766–2769. https://doi.org/10.1002/lary.24095 CrossRefPubMed

21.

Horbach SE, Lokhorst MM, Saeed P et al (2016) Sclerotherapy for low-flow vascular malformations of the head and neck: a systematic review of sclerosing agents. J Plast Reconstr Aesthet Surg 69:295–304. https://doi.org/10.1016/j.bjps.2015.10.045 CrossRefPubMed

22.

Perkins JA, Manning SC, Tempero RM et al (2010) Lymphatic malformations: review of current treatment. Otolaryngol Head Neck Surg 142:795–803. https://doi.org/10.1016/j.otohns.2010.02.026 CrossRefPubMed

23.

Mulligan PR, Prajapati HJ, Martin LG et al (2014) Vascular anomalies: classification, imaging characteristics and implications for interventional radiology treatment approaches. Br J Radiol 87:20130392. https://doi.org/10.1259/bjr.20130392 CrossRefPubMedPubMedCentral

24.

Zhong PQ, Zhi FX, Li R et al (1998) Long-term results of intratumorous bleomycin-A5 injection for head and neck lymphangioma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 86:139–144. https://doi.org/10.1016/s1079-2104(98)90115-9 CrossRefPubMed

25.

Mathur NN, Rana I, Bothra R et al (2005) Bleomycin sclerotherapy in congenital lymphatic and vascular malformations of head and neck. Int J Pediatr Otorhinolaryngol 69:75–80. https://doi.org/10.1016/j.ijporl.2004.08.008 CrossRefPubMed

26.

Bai Y, Jia J, Huang XX et al (2009) Sclerotherapy of microcystic lymphatic malformations in oral and facial regions. J Oral Maxillofac Surg 67:251–256. https://doi.org/10.1016/j.joms.2008.06.046 CrossRefPubMed

27.

Chaudry G, Guevara CJ, Rialon KL et al (2014) Safety and efficacy of bleomycin sclerotherapy for microcystic lymphatic malformation. Cardiovasc Intervent Radiol 37:1476–1481. https://doi.org/10.1007/s00270-014-0932-z CrossRefPubMed

28.

Horbach SE, Rigter IM, Smitt JH et al (2016) Intralesional bleomycin injections for vascular malformations: a systematic review and meta-analysis. Plast Reconstr Surg 137:244–256CrossRef

29.

Fevurly RD, Fishman SJ (2012) Vascular anomalies in pediatrics. Surg Clin North Am 92:769–800. https://doi.org/10.1016/j.suc.2012.03.016 CrossRefPubMed

30.

James MP, Collier PM, Aherne W et al (1993) Histologic, pharmacologic, and immunocytochemical effects of injection of bleomycin into viral warts. J Am Acad Dermatol 28:933–937. https://doi.org/10.1016/0190-9622(93)70133-e CrossRefPubMed

31.

Blum RH, Carter SK, Agre K (1973) A clinical review of bleomycin–a new antineoplastic agent. Cancer 31:903–914. https://doi.org/10.1002/1097-0142(197304)31:4%3C903:aid-cncr2820310422%3E3.0.co;2-n CrossRefPubMed

Titel: Lymphatic malformations compromising the upper airway in children: ultrasound-guided intralesional focal sclerotherapy with bleomycin targeting culprit lesions
verfasst von: Hidehito Usui
Masato Shinkai
Norihiko Kitagawa
Kyoko Mochizuki
Issei Kawakita
Yukihiro Tsuzuki
Shota Shinohara
Yuma Yagi
Munetaka Masuda
Publikationsdatum: 13.07.2020
Verlag: Springer Berlin Heidelberg
Erschienen in: Pediatric Surgery International / Ausgabe 9/2020
Print ISSN: 0179-0358
Elektronische ISSN: 1437-9813
DOI: https://doi.org/10.1007/s00383-020-04715-y

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Lymphatic malformations compromising the upper airway in children: ultrasound-guided intralesional focal sclerotherapy with bleomycin targeting culprit lesions