This retrospective study explored the applications of prenatal ultrasound in the diagnosis of fetal cutaneous hemangioma. We also reviewed the structural complications and pregnancy outcomes in these cases.
Previous studies reported that most cutaneous hemangiomas were in the head and neck,20,21 and less often in the trunk, extremities, and mucosal surface.19 In the present study, cutaneous hemangiomas were found in the head and neck (55%), lower limbs (25%), trunk (10%), and upper limbs. This differed from previous reports,3,19 in which the incidence in the middle trunk was higher than in the lower extremities. Of note, all 5 cases in which the pregnancy was terminated had cutaneous hemangiomas in the head and neck. Hemangiomas in these locations suggest a poor outcome.
The main prenatal ultrasonic characteristics of cutaneous hemangiomas were thickening of the subcutaneous soft tissue, with solid or solid-cystic mixed masses. The internal echo could be hypoechoic, iso-echoic, or hyperechoic. Color Doppler ultrasound could detect multiple strip color blood flow signals, which were important toward diagnosing hemangioma. The PSV and RIs of the cutaneous hemangioma blood supply varied significantly and had little diagnostic value. A hemangioma of large size was usually associated with advanced gestational age and a fast growth rate.
Cutaneous hemangiomas are easily misdiagnosed as teratoma, placental hemangioma, meningocele, or meningoencephalocele, and in the present study this occurred in 5 cases. Teratomas are mostly located in the sacrococcygeal region, head, and neck, with lipid stratification and calcified hyperechoic mass. Placental hemangiomas are mostly located on the fetal surface of the placenta, close to the entrance of the umbilical cord placenta and protruding into the amniotic cavity. The internal echo and blood supply are similar to that of fetal cutaneous hemangiomas. The relationship between the mass and surrounding tissues and organs should be monitored to determine its origin, in order to differentiate between placental and cutaneous hemangioma. Meningocele or meningoencephalocele is the interruption of the continuity of the strong echo light band of the skull, with a bulging mass at the skull defect. The meninges, brain tissue, or both bulge outward through the defect to form the mass. In Fig. 6, it can be clearly observed that the fetal skull had good continuity. The mass was not connected with the intracranial tissue.
There were no complications among cases in which the maximum diameter of the hemangioma was < 5 cm or volume < 40 cm3. The complication rates in cases in which the maximum diameter was ≥ 5 cm or volume ≥ 40 cm3 were 66.7% and 100%, respectively. Most complications were cardiac-related (87.5%), mainly indicated by an increased cardiothoracic area ratio and atrioventricular valve regurgitation. Tricuspid regurgitation could range from mild to severe in the third trimester. This suggested that ultrasound should be used to rule out structural abnormalities in fetuses when the cutaneous hemangioma has a maximum diameter ≥ 5 cm or mass volume ≥ 40 cm3.
One newborn developed KMP after the birth. KMP can be life-threatening, with significant thrombocytopenia, decreased fibrinogen level, increased fibrin degradation products, and accelerated tumor growth. It is a rare consumptive coagulation illness that occurs with certain vascular tumors.3,4,9,20 Successful management can resolve KMP and make angioma regress.4 The only case of postnatal death in this study involved a newborn with congenital clustered hemangioma or kaposiform hemangioendothelioma (KHE) related to KMP. It is necessary to rule out cluster hemangiomas and KHE in cases with KMP.
Polyhydramnios can occur in up to 40% of fetuses with prenatal diagnosis of large neck masses.28 When there is polyhydramnios, the possibility of esophagus and airway obstruction should be highly suspected. Real-time ultrasound can evaluate these functions in the fetus. Ultrasound is the first choice and highly accurate for prenatal examination of fetal cutaneous hemangiomas, while MRI can be used as a supplementary method. Prenatal ultrasound and MRI may also help delivery planning and immediate postnatal care.31
The high cardiac output caused by fetal cutaneous hemangioma can lead to fetal hydrops. Fetuses with fetal hydrops are more likely to have fetal distress in utero, which is easily complicated with KMP. Fetuses with hemangioma complicated with arteriovenous fistula and high cardiac output should rule out postnatal pulmonary hypertension. In Patient 4 and 5 in the present study, severe pulmonary hypertension occurred after the birth. After failing to respond to propranolol, they received artery ligation and achieved satisfactory outcomes. Therefore, clinical monitoring of growth in the mid and late pregnancy is very important.
In the present study, chromosome examination was performed in 6 patients before delivery, with no obvious abnormalities found. This result was similar to a previous study reported by Zheng et al.9
If the newborn survives, most congenital hemangiomas regress rapidly during the first year of life.22,23 Some of them may remain stable or partially regress.2 In the present study, the hemangiomas of newborns who were not give surgery or interventional treatments regressed to varying degrees during the follow-ups of up to 6 years. Regarding the treatment of cutaneous hemangioma, the conventional methods include oral glucocorticoids, cryotherapy, laser, local injection of sclerosing agent, and surgical resections. The outcome is uncertain and side effects can be significant. Currently, oral propranolol is the first-line treatment for hemangioma. It should be initiated as early as possible to minimize any potential side effects.32,35,36 For large and complex hemangiomas, a variety of combined treatments can be considered.