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by the American Institute of Ultrasound in Medicine J Ultrasound Med 22:419-423 • 0278-4297
Prenatal Diagnosis of a Large Axillary Cystic Lymphangioma by Three-dimensional Ultrasonography and Magnetic Resonance ImagingDepartments of Maternity (R.R., J.-P.A., A.-G.G., Y.D., M.D.) and Pediatric Radiology (I.S., P.S.), Hôpital Necker-Enfants Malades, Assistance Publique–Hôpitaux de Paris (AP-HP), Université Paris V, Paris, France. Address correspondence and reprint requests to Rodrigo Ruano, MD, Maternité, Hôpital Necker-Enfants Malades, 149 rue de Sèvres, 75743 Paris Cedex 15. France.
Abbreviations: MRI, magnetic resonance imaging • 3DUS, three-dimensional ultrasonography • 2DUS, two-dimensional ultrasonography
Cystic lymphangiomas are benign tumors of the lymphatic system, appearing as uniseptate or multiseptate cystic masses, often located in the area of the neck (75%) or the axilla (20%).1 Postnatal outcome depends mainly on the size and location of the lesion.2 An increasing number of such congenital abnormalities are detected on routine conventional prenatal ultrasonography.2–4 The absence of blood flow on color Doppler mapping is characteristic of lymphangiomas as opposed to hemangiomas.5 So far, prenatal evaluation for the prognosis of fetal lymphangiomas has been based solely on two-dimensional ultrasonography (2DUS).3,4 Magnetic resonance imaging (MRI) may help in assessing the extent of a lesion, as suggested in a recent case report of a fetal intra-abdominal lymphangioma.6 We report a case of prenatal assessment of axillary lymphangioma by threedimensional ultrasonography (3DUS), and we analyze the information provided by this technique in comparison with MRI and 2DUS.
A 30-year-old woman, gravida 1, para 0, was referred at 27 weeks to our Fetal Medicine Unit after prenatal diagnosis of a lymphangioma. Prenatal diagnosis was based on ultrasonographic imaging of a large subcutaneous cystic mass measuring 73 x 53 x 25 and 101 x 64 x 30 mm at 22 and 26 weeks’ gestation, respectively. Amniocentesis revealed a normal 46,XX karyotype. After she had been counseled on the anticipated pediatric outcome and postnatal treatment, the patient elected to continue the pregnancy.
At the time of referral, conventional 2DUS confirmed the presence of a large multilocular, septate cystic mass located on the right side of the fetal trunk, measuring 100 x 70 x 31 mm (Fig. 1
To assess the connection of the mass with adjacent structures more precisely, fetal MRI was performed at 30 weeks with fast spin echo T2-weighted sequences (Signa 1.5 T; GE Medical Systems, Waukesha, WI) in the 3 planes. The lesion was cystic and therefore gave a high signal on the T2-weighted images. It was located on the right side of the fetal trunk extending from the axilla to the pelvis. There was no evidence of involvement of the right arm, and the lesion did not seem to extend across the thoracic wall (Fig. 2  ).
At 31 weeks’ gestation, the patient consented to undergo 3DUS examination (Voluson 730; Kretztechnik, AG, Zipf, Austria; 4- and 8-MHz transducer) after being informed that the procedure would be undertaken for research purposes and that the results would not be disclosed to the medical staff in charge of perinatal management. The operator performing 3DUS (R.R.) was blinded to MRI data.
After multiplanar images of the lymphangioma were acquired and analyzed, the reconstructed volume image was rotated by 30° on the 3 orthogonal views and traced manually to estimate tumor volume, which was found to be 100.5 mL. It was also possible to delineate the shape of fetal skin covering the lesion. We were able to generate rendered images confirming that the axilla and the arm were not involved, the upper limit of the mass ending a few millimeters below the apex of the armpit (Fig. 3
A follow-up 2DUS scan performed at 36 weeks’ gestation showed a small increase in the size of the cystic mass (120 x 93 x 40 mm). Elective cesarean delivery was performed at 38 weeks. A female neonate was delivered, weighing 2945 g with Apgar scores of 9 at 1 minute and 10 at 5 minutes.
Neonatal physical examination confirmed the presence of a large, soft subcutaneous lesion, which extended from the iliac bone to the lower part of the neck (Fig. 6
The case we report suggests that both 3DUS and MRI could be of interest for the assessment of the extension of fetal tumors. Cystic lymphangiomas are thought to result from obstruction of lymphatic vessels leading to the jugular venous system. The sizes of these tumors vary from a few millimeters to several centimeters.7 Postnatal outcome is mainly related to the connections of the lesion with surrounding organs.2 Large lymphangiomas may have excellent prognoses, provided that connections with surrounding structures allow for complete surgical resection. In contrast, multifocal lesions are difficult to treat, and cases complicated by pleural effusion, ascites, or hydrops may be lethal.2,7 Lesions located on the posterior aspect of the fetal neck, which are often considered hygroma colli, are associated with a higher risk of chromosomal abnormalities.7 Prenatal diagnosis of a large lymphangioma can be achieved easily by conventional 2DUS on the basis of the finding of a multilocular or septate cystic mass. Two-dimensional ultrasonography can assess the size of the lesion, leading to elective cesarean delivery when a large mass is expected to cause dystocia. Two-dimensional ultrasonography is also effective in identifying predominantly cystic lesions that may benefit from prenatal needle aspiration to reduce their volume before delivery.3,8 However, it may be difficult to accurately evaluate the limits of the tumor and its connections with surrounding organs. Kaminopetros et al6 reported a case of an extensive intra-abdominal lymphangioma that was followed by MRI prenatally. They pointed out that MRI had accurately shown the extent and character of the lesion. In the present case, the axillary cystic lymphangioma was easily diagnosed on the basis of conventional 2DUS, but its extent and limits could not be ascertained by this technique alone. This prompted us to record fetal MRI and 3DUS scans. Both imaging techniques established the absence of involvement of the right axilla, arm, and thoracic wall. It is not possible to compare the diagnostic value of MRI and 3DUS on the basis of a single case report; however, in this case, similar information was derived from the 2 techniques. Further studies are needed to establish the diagnostic value of 3DUS. These studies should include cases with microcystic lesions, which could be more easily detected on MRI than on ultrasonography. Nevertheless, 3DUS might have the advantage over MRI of being more widely available, thus enabling, for instance, serial volume measurements that could be of interest in monitoring tumor growth.
Received November 26, 2002, from the Departments of Maternity (R.R., J.-P.A., A.-G.G., Y.D., M.D.) and Pediatric Radiology (I.S., P.S.), Hôpital Necker-Enfants Malades, Assistance Publique–Hôpitaux de Paris (AP-HP), Université Paris V, Paris, France. Revision requested December 2, 2002. Revised manuscript accepted for publication December 18, 2002. We thank Arnaud Dujardin and Dominique Esnoult (Kretztechnik, Velisy, France) for technical support.
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