Malformación linfática retroperitoneal como causa de dolor abdominal en paciente pediátrico. Reporte de caso
Retroperitoneal lymphatic malformation as a cause of abdominal pain in a pediatric patient. Case report
DOI:
https://doi.org/10.15446/cr.v10n2.107809Keywords:
Malformación Linfática, Retroperitoneal, Dolor Abdominal, Niño, Escleroterapia (es)Lymphatic Malformation, Retroperitoneal, Abdominal Pain, Child, Sclerotherapy (en)
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Introducción. Las malformaciones linfáticas abdominales (MLA) son anomalías vasculares congénitas poco frecuentes en pediatría que se producen por alteraciones del desarrollo de los vasos linfáticos. Debido a que la literatura sobre su presentación retroperitoneal en pediatría es escasa, es importante dar a conocer alternativas de manejo como la escleroterapia, ya que su resección completa es complicada debido a su ubicación anatómica y su alta tasa de recidiva.
Presentación del caso. Niño de 6 años, quien fue llevado al servicio de urgencias de una institución de tercer nivel de atención de Tunja (Colombia) por dolor abdominal y fiebre. Tres días previos a la consulta, el paciente sufrió trauma toracoabdominal cerrado por caída desde su propia altura. Al ingreso se tomó ecografía abdominal que mostró una colección líquida abdominal de 800mL. Debido al antecedente de trauma, taquicardia y dolor abdominal, se realizó laparotomía exploratoria en la que se halló una masa retroperitoneal derecha. Se realizó una tomografía computarizada (TC) abdominal con contraste en la cual se observó malformación linfática (macroquística) retroperitoneal de 152x87x119mm aislada. Por sus características, se indicó manejo médico con drenajes percutáneos guiados por TC e infiltraciones intralesionales con bleomicina (0.5UI/kg), con posterior resolución de los síntomas y regresión del 98% del volumen de la lesión.
Conclusiones. El tratamiento de las MLA depende de su estructura interna, su localización y las lesiones asociadas. Aunque el abordaje quirúrgico fue considerado la terapia de elección, actualmente la escleroterapia se ha convertido en una opción segura y eficaz que previene complicaciones y trata los síntomas, aun en ubicaciones inusuales como en el caso reportado.
Abstract
Introduction: Abdominal lymphatic malformations (ALM) are rare congenital vascular anomalies in pediatrics that result from alterations in the development of the lymphatic vessels. Since literature on retroperitoneal presentation in pediatrics is scarce, it is essential to provide information on treatment alternatives such as sclerotherapy, since complete resection is challenging because of its anatomical location and high recurrence rate.
Case presentation: A 6-year-old boy was taken to the emergency department of a tertiary care center in Tunja (Colombia) due to abdominal pain and fever. Three days prior to his visit, the patient suffered a blunt thoracoabdominal trauma due to a fall from his own height. On admission, abdominal ultrasound was performed, showing an 800mL abdominal fluid collection. Given the history of trauma, tachycardia, and abdominal pain, an exploratory laparotomy was performed, during which a retroperitoneal mass was found on the right side. A contrast-enhanced abdominal computed tomography (CT) scan was performed, finding an isolated retroperitoneal lymphatic malformation (macrocystic) measuring 152x87x119mm. Due to its characteristics, medical treatment was ordered with CT-guided percutaneous drainage and intralesional infiltrations with bleomycin (0.5UI/kg). Subsequently, symptoms subsided and 98% of the lesion volume disappeared.
Conclusion: The treatment of the ALMs depends on their architecture, location, and associated lesions. Even though surgical treatment was previously considered the therapy of choice, sclerotherapy has now emerged as a safe and effective option that prevents complications and resolves symptoms, even in unusual locations, as in the presented case.
References
References
1. Lal A, Gupta P, Singhal M, Sinha SK, Lal S, Rana S, et al. Abdominal lymphatic malformation: Spectrum of imaging findings. Indian J Radiol Imaging. 2016;26(4):423-8. https://doi.org/n9j6.
2. Kulungowski AM, Patel M. Lymphatic malformations. Semin Pediatr Surg. 2020;29(5):150971. https://doi.org/gq3q8p.
3. Amodeo I, Cavallaro G, Raffaeli G, Colombo L, Fumagalli M, Cavalli R, et al. Abdominal cystic lymphangioma in a term newborn: A case report and update of new treatments. Medicine (Baltimore). 2017;96(8):e5984. https://doi.org/f9rsfh.
4. De Bast Y, Hossey D, Boon L, Duttmann R, Theunis A, Chambers S, et al. Intra-abdominal lymphatic malformation. Acta Gastroenterol Belg. 2006;69(2):209-12.
5. Brungardt JG, Cooke-Barber J, Tiao G, Hammill AM, Ricci K, Patel MN, et al. Intra-abdominal Lymphatic Malformations: A Case Series. Authorea. 2022. https://doi.org/n9kc.
6. Torrealba I, de Barbieri F. Linfangioma abdominal. Caso clínico. Rev. Chil. Pediatr. 2012;83(1):68-72. https://doi.org/n9kd.
7. Elbaaly H, Piché N, Rypens F, Kleiber N, Lapierre C, Dubois J. Intra-abdominal lymphatic malformation management in light of the updated International Society for the Study of Vascular Anomalies classification. Pediatr Radiol. 2021;51(5):760-72. https://doi.org/n9kg.
8. Gimero-Aranguez M, Colomar-Palmer P, González-Mediero I, Ollero-Caprani JM. Aspectos clínicos y morfológicos de los linfangiomas infantiles: revisión de 145 casos. An Esp Pediatr. 1996;45(1):25-8.
9. Elluru RG, Balakrishnan K, Padua HM. Lymphatic malformations: diagnosis and management. Semin Pediatr Surg. 2014;23(4):178-85. https://doi.org/f6j3km.
10. Zamora AK, Barry WE, Nowicki D, Ourshalimian S, Navid F, Miller JM, et al. A multidisciplinary approach to management of abdominal lymphatic malformations. J Pediatr Surg. 2021;56(8):1425-9. https://doi.org/g54dzn.
11. Gunadi, Kashogi G, Prasetya D, Fauzi AR, Daryanto E, Dwihantoro A. Pediatric patients with mesenteric cystic lymphangioma: A case series. Int J Surg Case Rep. 2019;64:89-93. https://doi.org/n9km.
12. de Lagausie P, Bonnard A, Berrebi D, Lepretre O, Statopoulos L, Delarue A, et al. Abdominal lymphangiomas in children: interest of the laparoscopic approach. Surg Endosc. 2007;21(7):1153-7. https://doi.org/dvk48x.
13. Dubois J, Thomas-Chaussé F, Soulez G. Common (Cystic) Lymphatic Malformations: Current Knowledge and Management. Tech Vasc Interv Radiol. 2019;22(4):100631. https://doi.org/gq3q82.
14. Restrepo R. Multimodality imaging of vascular anomalies. Pediatr Radiol. 2013;43(Suppl 1):S141-54. https://doi.org/n9kn.
15. Martínez-Montalvo CM, Muñoz-Delgado DY, Jiménez-Sánchez HC, Siado-Guerrero SA, Esguerra DC, Ordóñez DA. Quiste mesentérico gigante: reporte de caso. Rev Colomb Gastroenterol. 2021;36(2):257-62. https://doi.org/n9kp.
16. Yan J, Xie C, Chen Y. Surgical Treatment of Mesenteric Lymphatic Malformations in Children: An Observational Cohort study. J Pediatr Surg. 2023;58(9):1762-9. https://doi.org/n9kq.
17. Tripathy PK, Jena PK, Pattnaik K. Management outcomes of mesenteric cysts in paediatric age group. Afr J Paediatr Surg. 2022;19(1):32-5. https://doi.org/n9kr.
18. Yilmaz H, Yilmaz Ö, Çamlidağ I, Belet Ü, Akan H. Single center experience with intralesional bleomycin sclerotherapy for lymphatic malformations. Jpn J Radiol. 2017;35(10):590-6. https://doi.org/n9kx.
19. Thorburn C, Price D. Expectant management of pediatric lymphatic malformations: A 30-year chart review. J Pediatr Surg. 2022;57(5):883-7. https://doi.org/n9kz.
20. Van Damme A, Seront E, Dekeuleneer V, Boon LM, Vikkula M. New and Emerging Targeted Therapies for Vascular Malformations. Am J Clin Dermatol. 2020;21(5):657-68. https://doi.org/n9k2.
21. Cuervo JL, Galli E, Eisele G, Johannes E, Fainboim A, Tonini S, et al. Malformaciones linfáticas: Tratamiento percutáneo con bleomicina. Arch. Argent. Pediatr. 2011;109(5):417-22. https://doi.org/fxvgw6.
22. Rosales-Parra ND, Acero-Murillo CF, García-Aristizabal MP, Romero-Espitia WD. Malformaciones linfáticas abdominales en una población pediátrica: experiencia en un centro de referencia de Medellín, Colombia. Rev Colomb Cir. 2022;37:245-50. https://doi.org/n9k5.
23. Echeagaray-Sánchez HL, Del Bosque-Méndez JE, Soto-Becerril OA, Hernández-Abarca E, Gómez-De la Cruz CA. Escleroterapia con bleomicina en el tratamiento de malformación linfática microquística. An Orl Mex. 2019;64(4):229-33.
24. Horbach SE, Lokhorst MM, Saeed P, de Goüyon Matignon de Pontouraude CM, Rothová A, van der Horst CM. Sclerotherapy for low-flow vascular malformations of the head and neck: A systematic review of sclerosing agents. J Plast Reconstr Aesthet Surg. 2016;69(3):295-304. https://doi.org/f8czkb.
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