Autopsy and Case Reports
https://autopsyandcasereports.org/article/doi/10.4322/acr.2017.009
Autopsy and Case Reports
Article / Clinical Case Report

Isolated fetal lymphatic malformation of the thigh: prenatal diagnosis and follow-up  

Huseyin Durukan, İlay Gözükara, Murside Cevikoglu, Talat Umut Kutlu Dilek

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Abstract

Fetal lymphatic malformation can be found in different parts of the fetal body. It occurs most frequently in the nuchal and axillary region and less frequently in the abdomen or inguinal areas. Lymphatic malformation has been associated with fetal aneuploidy, hydrops fetalis, structural malformations, and intrauterine fetal death. A 31-year-old gravida 3, para 2 woman was admitted to our hospital at 22 weeks of gestation (confirmed by ultrasonographic examination). The fetus was alive, and had a mass derived from the left inguinal region extending to the anterior left leg with fluid-filled cavities about 3–5 cm in size. There was no evidence of intra-abdominal extension of the mass. Amniocentesis was performed. Fetal magnetic resonance imaging revealed a left inguinal cystic mass, which extended to the left thigh. Antenatal follow-up was uneventful. The mother gave birth at term with a cesarean section. Postnatal clinical examination and imaging examination confirmed the diagnosis of lymphatic malformation. Fetal lymphatic malformation carries a high risk of aneuploidy and fetal malformations. Patients diagnosed with lymphatic malformation in antenatal follow-up should be assessed in terms of coexistent anomalies. Fetal karyotyping should be done and the fetus should be monitored for fetal hydrops.

Keywords

Fetal Thigh; Lymphatic Malformation; Prenatal Diagnosis.

References

1. Wassef M, Blei F, Adams D, et al. Vascular Anomalies classification: recommendations from the International Society for the Study of Vascular Anomalies. Pediatrics. 2015;136(1):e203-14. PMid:26055853. http://dx.doi.org/10.1542/peds.2014-3673.

2. Schild RL, Orhan Y, Meyberg H, Braunschweig T, Kno¨pfle G, Gembruch U. Three-dimensional ultrasound of a massive fetal lymphangioma involving the lower extremity. Ultrasound Obstet Gynecol. 2003;22(5):547- 51. PMid:14618671. http://dx.doi.org/10.1002/uog.915.

3. Temizkan O, Abike F, Ayvaci H, Demirag E, Görücü Y, Isık E. Fetal axillary cystic hygroma:a case report and review. 2011 Licensee PAGEPress. Italy Rare Tumors. 2011;3(4):e39. http://dx.doi.org/10.4081/rt.2011.e39.

4. Masood SN, Masood MF. Case report of fetal axillothoraco-abdominal cystic hygroma. Arch Gynecol Obstet. 2010;281(1):111-5. PMid:19360432. http://dx.doi.org/10.1007/s00404-009-1068-2.

5. Rasidaki M, Sifakis S, Vardaki E, Koumantakis E. Prenatal diagnosis of a fetal chest wall cystic lymphatic malformation using ultrasonography and MRI: a case report with literature review. Fetal Diagn Ther. 2005;20(6):504-7. PMid:16260884. http://dx.doi.org/10.1159/000088039.

6. Gedikbasi A, Gul A, Sargin A, Ceylan Y. Cystic hygroma and lymphatic malformation: associated findings, perinatal outcome and prognostic factors in live-born infants. Arch Gynecol Obstet. 2007;276(5):491-8. PMid:17429667. http://dx.doi.org/10.1007/s00404-007-0364-y.

7. Descamps P, Jourdain O, Paillet C, et al. Etiology, prognosis and management of nuchal cystic hygroma:

2. new cases and literature review. Eur J Obstet Gynecol Reprod Biol 1997;71(1)3-10.

8. Bagrodia N, Defnet AM, Kandel JJ. Management of lymphatic malformations in children. Curr Opin Pediatr. 2015;27(3):356-63. PMid:25888145. http://dx.doi.org/10.1097/MOP.0000000000000209.

9. Atalar MH, Cetin A, Kelkit S, Buyukayhan D. Giant fetal axillo-thoracic cystic hygroma associated with ipsilateral foot anomalies. Pediatr Int. 2006;48(6):634-7. PMid:17168988. http://dx.doi.org/10.1111/j.1442-200X.2006.02263.x.

10. Ozeki M, Kanda K, Kawamoto N, et al. Propranolol as an alternative treatment option for paediatric lymphatic malformation. Tohoku J Exp Med. 2013;229(1):61-6. PMid:23257321. http://dx.doi.org/10.1620/tjem.229.61.

11. Luo Y, Liu L, Rogers D, et al. Rapamycin inhibits lymphatic endothelial cell tube formation by downregulating vascular endothelial growth factor receptor 3 protein expression. Neoplasia. 2012;14(3):228-37. PMid:22496622. http://dx.doi.org/10.1593/neo.111570.

12. Okazaki T, Iwatani S, Yanai T, et al. Treatment of lymphatic malformation in children: our experience of 128 cases. J Pediatr Surg. 2007;42(2):386-9. PMid:17270554. http://dx.doi.org/10.1016/j.jpedsurg.2006.10.012.

13. Grabb WC, Dingman RO, Oneal RM, Dempsey PD. Facial hamartomas in children: neurofibroma, lymphatic malformation, and haemangioma. Plast Reconstr Surg. 1980;66(4):509-2715. PMid:7208665. http://dx.doi.org/10.1097/00006534-198010000-00003.

14. Deshpande P, Twining P, O’Neill D. Prenatal diagnosis of fetal abdominal lymphatic malformation by ultrasonography. Ultrasound Obstet Gynecol. 2001;17(5):445-8. PMid:11380973. http://dx.doi.org/10.1046/j.1469-0705.2001.00367.x.

15. Rha SE, Byun JY, Kim HH, et al. Prenatal sonographic and MR imaging findings of extensive fetal lymphatic malformation: a case report. Korean J Radiol. 2003;4(4):260-3. PMid:14726645. http://dx.doi.org/10.3348/kjr.2003.4.4.260.


Publication date:
03/31/2017

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