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Mædica - a Journal of Clinical Medicine CASE REPORTS 334 Maedica A Journal of Clinical Medicine, Volume 11 No.4 2016 MAEDICA – a Journal of Clinical Medicine 2016; 11(4):334-340 Ultrasound Prenatal Diagnosis and Emergency Interventional Radiologic Therapy of Galen Aneurysmal Malformation in a Newborn Roxana Elena BOHILTEA a,b , Natalia TURCAN a,b , Cristian MIHALEA c,d , Bogdan DOROBAT b , Eliza Elena CINTEZA a,e , Adriana DAN b , Magdalena MIHAI b , Adela DIMITRIADE b , Cristian BOROS b , Mircea DUMITRU b , Ionut GOBEJ f , Octavian MUNTEANU a,b , Monica Mihaela CIRSTOIU a,b a “Carol Davila” University of Medicine and Pharmacy, Bucharest, Romania b University Emergency Hospital, Bucharest, Romania c Department of Interventional Neuroradiology, NEURI, Hopital Bicetre, APHP, Paris Sud Universite´ - Paris, France d Department of Neurosurgery, University of Medicine and Pharmacy “Victor Babes,” Timisoara, Romania e “Maria Sklodowska Curie” Emergency Children’s Hospital, Bucharest, Romania f Colentina Clinic Hospital, Bucharest, Romania Address for correspondence: Eliza Cinteza Postal address: “Maria Sklodowska Curie” Emergency Children’s Hospital, Constantin Brancoveanu Blv, nr 20, sect 4, Bucharest, Romania E-mail; [email protected], Bucharest, Romania Article received on the 08 rd of December 2016. Article accepted on the 12 rd of December 2016. ABSTRACT Located under the cerebral hemispheres and draining the anterior and central regions of the brain into the sinus of the posterior cerebral fossa, the vein of Galen aneurysmal malformation is considered to be a rare cause of hydrocephaly. The presence of this condition in the neonatal period typically includes intractable heart failure and a poor prognosis. We report a case of aneurysm of the vein of Galen diagnosed prenatally at 28 weeks of gestation, with the delivery at term by caesarean section of a female infant. Sonographically, the vein of Galen appeared in the mid-sagittal plane, large, supratentorial, non-pulsatile; on color Doppler, the structure filled with bright color, reflecting a turbulent venous flow. A low grade of ventriculomegaly was present during the evolution of pregnancy; regarding the cardiovascular function, an intrauterine right cardiac insufficiency overlapped a tricuspid regurgitation and right atrial dilatation. A multidisciplinary committee decided a neonatal embolization of the aneurysm as an emergency requirement due to increased pulmonary hypertension developed in the next 24 hours after birth. After the embolization of the two main drainage vessels, the cardiac dysfunction persists. Two days later the evolution became unfavorable, leading to the necessity of the second embolization, which resulted in a 48 hours’ coma and death, due to a cerebral hemorrhage secondary to thrombosis and fissure of the embolized aneurysm. The prognosis for the neonate with malformation of the Galen vein depends upon the severity of the cardiovascular status. Embolization represents actually the treatment of choice with the best results of these cases, but the mortality remains as high as 50 percent even in the most specialized centers of the world. As far as we know this is the only case of Galen aneurysmal malformation in Romania which beneficiated of embolization by interventional treatment in neonatal period. Keywords: Galen aneurysm, interventional neuroradiology, embolization

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Page 1: Ultrasound Prenatal Diagnosis and Emergency …16)_No4_pg334-340.pdfUltrasound Prenatal Diagnosis and Emergency Interventional Radiologic Therapy of Galen Aneurysmal Malformation in

Mædica - a Journal of Clinical Medicine

CASE REPORTS

334 Maedica

A Journal of Clinical Medicine, Volume 11 No.4 2016

MAEDICA – a Journal of Clinical Medicine2016; 11(4):334-340

Ultrasound Prenatal Diagnosis and

Emergency Interventional Radiologic

Therapy of Galen Aneurysmal

Malformation in a NewbornRoxana Elena BOHILTEAa,b, Natalia TURCANa,b, Cristian MIHALEAc,d, Bogdan DOROBATb, Eliza Elena CINTEZAa,e, Adriana DANb, Magdalena MIHAIb, Adela DIMITRIADEb, Cristian BOROSb, Mircea DUMITRUb, Ionut GOBEJf, Octavian MUNTEANUa,b, Monica Mihaela CIRSTOIUa,b

a “Carol Davila” University of Medicine and Pharmacy, Bucharest, Romania

b University Emergency Hospital, Bucharest, Romania

c Department of Interventional Neuroradiology, NEURI, Hopital Bicetre, APHP, Paris Sud Universite´ - Paris, France

d Department of Neurosurgery, University of Medicine and Pharmacy “Victor Babes,” Timisoara, Romania

e “Maria Sklodowska Curie” Emergency Children’s Hospital, Bucharest, Romania

f Colentina Clinic Hospital, Bucharest, Romania

Address for correspondence:Eliza CintezaPostal address: “Maria Sklodowska Curie” Emergency Children’s Hospital, Constantin Brancoveanu Blv, nr 20, sect 4, Bucharest, RomaniaE-mail; [email protected], Bucharest, Romania

Article received on the 08rd of December 2016. Article accepted on the 12rd of December 2016.

ABSTRACT

Located under the cerebral hemispheres and draining the anterior and central regions of the brain into the sinus of the posterior cerebral fossa, the vein of Galen aneurysmal malformation is considered to be a rare cause of hydrocephaly. The presence of this condition in the neonatal period typically includes intractable heart failure and a poor prognosis. We report a case of aneurysm of the vein of Galen diagnosed prenatally at 28 weeks of gestation, with the delivery at term by caesarean section of a female infant. Sonographically, the vein of Galen appeared in the mid-sagittal plane, large, supratentorial, non-pulsatile; on color Doppler, the structure filled with bright color, reflecting a turbulent venous flow. A low grade of ventriculomegaly was present during the evolution of pregnancy; regarding the cardiovascular function, an intrauterine right cardiac insufficiency overlapped a tricuspid regurgitation and right atrial dilatation. A multidisciplinary committee decided a neonatal embolization of the aneurysm as an emergency requirement due to increased pulmonary hypertension developed in the next 24 hours after birth.

After the embolization of the two main drainage vessels, the cardiac dysfunction persists. Two days later the evolution became unfavorable, leading to the necessity of the second embolization, which resulted in a 48 hours’ coma and death, due to a cerebral hemorrhage secondary to thrombosis and fissure of the embolized aneurysm. The prognosis for the neonate with malformation of the Galen vein depends upon the severity of the cardiovascular status. Embolization represents actually the treatment of choice with the best results of these cases, but the mortality remains as high as 50 percent even in the most specialized centers of the world. As far as we know this is the only case of Galen aneurysmal malformation in Romania which beneficiated of embolization by interventional treatment in neonatal period.

Keywords: Galen aneurysm, interventional neuroradiology, embolization

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ULTRASOUND PRENATAL DIAGNOSIS AND EMERGENCY INTERVENTIONAL RADIOLOGIC THERAPY OF GALEN ANEURYSMAL MALFORMATION IN A NEWBORN

335Maedica A Journal of Clinical Medicine, Volume 11 No.4 2016

INTRODUCTION

The aneurysm of the vein of Galen lines among intracranial arteriovenous mal-formations, with an incidence of 1% of all fetal arteriovenous vascular abnor-malities. The association with the persis-

tence of the embryonic median prosencephalic vein of Murkowski is not uncommon (1). This is a rare choroidal arteriovenous malformation that begins to develop in the early embryonic stages and has a declared mortality rate of about 50% (2). The symptomatology is rarely present before third trimester of pregnancy, therefore the diagno-sis before 24 weeks of gestation is unusual. Very rare cases early diagnosed are mentioned in litera-ture: one case reported be Lee et al. (3) of vein of Galen aneurysm was diagnosed at 23 weeks of gestation and another case of detection of an an-eurysm of the vein of Galen following signs of car-diac overload was diagnosed in a 22-week old fetus (4). Increased morbidity and mortality relat-ed to this condition is importantly correlated to the associated fetal or neonatal impaired heart function. Severe heart failure is developed early in the fetal life and is produced by a large systemic shunt. This causes cardiomegaly, heart failure and pulmonary hypertension (5). Clinical manifesta-tions of Galen aneurysmal malformation depend on the volume of arteriovenous shunting. Two forms are distinguished: the severe form with con-gestive heart failure with a rapid in utero progres-sion and a high mortality, and a mild childhood form (6). According to Litvak et al (7), Galen aneu-rysmal malformation can be subdivided into a true form (if arteriovenous shunting is occurring directly into the vein of Galen) and secondary form (arteriovenous shunt is located near or far from the vein of Galen). The signs of cardiac over-load or fetal hydrops may be first detected on a routine ultrasound detailed fetal examination and implicitly lead to detection of various vascular

malformations. On ultrasound, an intracerebral cystic structure or a hypoechoic intracranial struc-ture, usually, represents aneurysmal malforma-tion. Doppler technique elucidates the diagnosis by identifying the structure as a vascular malfor-mation. In 1983, Hirsch et al, declared that the vein of Galen aneurysm is the first malformation where the application of Doppler ultrasound has confirmed the prenatal diagnosis (8). The poor prognosis of vein of Galen aneurysmal malforma-tions is due to the quick development of multisys-tem organ failure, the hypoperfusion of intact brain parenchyma, the “steal” phenomenon, es-pecially when the brain injury is already present.

There are two main methods of treatment suggested: direct surgical removal and endovas-cular embolization. Since the introduction of the endovascular method the mortality decreased importantly and varies from 4% to 40% (9). By endovascular embolization the arteriovenous shunt is occluded and the pathological arteries and aneurysmal veins are disconnected from the circulation. q

CASE REPORT

We present the case of a female newborn, extracted through caesarian section at 38

weeks of gestation, for intrauterine signs of car-diac failure due to Galen aneurysmal malforma-tion.

A 21-year-old woman, gravida 1, para1, was admitted in our department at 28 weeks of ges-tation for a routine pregnancy control and a third trimester ultrasound. The fetus had been diag-nosed on ultrasound (US) with a cerebral arteri-al-venous malformation, confirmed by Doppler 3D ultrasound technique (Figure 1a-c, 2) and magnetic resonance imaging (MRI) (Figure 3-4). Sonographically, the vein of Galen appeared in the mid-sagittal plane, anechoic, large, ovalar shaped, supratentorial, non-pulsatile, continued to the posterior by the dilated straight sinus; on

FIGURE 1 a-c. Ultrasound 3D HD live. Flow image of aneurysm of Galen

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color Doppler, the structure filled with bright color, reflecting a turbulent venous flow. A par-ticular “8 shape” aspect of Willis polygon has been noticed (Figure 2). A mild ventriculomegaly was present during the evolution of pregnancy; regarding the cardiovascular function, an intra-uterine right cardiac insufficiency overlapped a tricuspid regurgitation and right atrial dilatation. The pregnancy had an uneventful course until 38 weeks of gestation when in context of a labor debut the newborn was extracted through cae-sarean section. The mother had a quick recovery without any post operatory complications. At birth baby had: weight 3050 g, length 50 cm, head circumference 32.5 cm, chest circumfer-ence 31 cm, Apgar score of 7 at 1 minute and 8 at 5 minutes. Clinical examination after birth re-vealed difficult postnatal adaptation, with respi-ratory distress syndrome: tachypnea, expiratory grunting, oxygen-dependency, signs of cardiac

failure (edema, tachycardia, loud sound II, car-diac murmur, positive Harzer sign, cardiomegaly, hepatomegaly), and murmur at anterior fonta-nel, mild hypotonic.

Chest X-ray revealed an increased cardiotho-racic index and globally decreased pulmonary transparency. Head US showed, in coronal plane, a large anechoic circular image in the midline, interventricular, with a loud Doppler signal, turbulent flow, suggesting a vascular anomaly. Echocardiography evaluation con-firmed the enlargement of heart chambers, espe-cially on the right side, important tricuspid regur-gitation, severe pulmonary hypertension (systolic pressure in main pulmonary artery up to 108 mmHg), and large patent ductus arteriosus, with bidirectional shunting.

Immediately after birth, the newborn was ad-mitted in the neonatal intensive care unit (NICU), placed in an incubator, received hood oxygen therapy (FiO

2 40%) for 24h, cardiac inotropic

support with Milrinone, diuretic therapy with Fu-rosemide, prophylactic antibiotic therapy (Ampi-cillin and Gentamycin), endovenous infusion of electrolytes, parenteral nutrition. Due to unfa-vorable course of the respiratory status, with in-creased oxygen need, the baby was intubated at 24 hours of age and put on mechanical ventila-tion. Because of the persistent increased pulmo-nary pressure, above the systemic mean arterial pressure, the shunting through the patent ductus

FIGURE 2. Ultrasound Color Doppler atypical image of Willis polygon

FIGURE 3. MRI image in sagital plane of aneurysmal dilatation and straight sinus dilatation

FIGURE 4. MRI image in transverse plane of aneurysmal dilatation and straight sinus dilatation

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arteriosus became unidirectional, right to left, which was considered beneficial for decreasing the pulmonary pressure, therefore treatment with Prostin (prostaglandine E1) for maintaining the ductus was initiated.

Because in this case, as in most of the arterial-venous malformations, the cause of the cardiac

failure is the increased flow to the heart (in-creased preload), the only efficient treatment should be aimed to close by embolization the anomalous vessels. Although generally recom-mended age for intervention is 3 to 6 months, in this case the intervention was decided to be done in emergency, due to the gravity of the car-diac disease. The embolization procedure was made in two steps, first at 3 days of life, second

FIGURE 5. Injection in left vertebral artery (fi rst intervention)

FIGURE 6. Final control after embolization of a few branches with coils and glue (Glubran + Lipiodol)

FIGURE 7. Injection in right ICA (second intervention)

FIGURE 8. Ultraselective catetherization with the microcatheter (very tortuous anatomy)

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ULTRASOUND PRENATAL DIAGNOSIS AND EMERGENCY INTERVENTIONAL RADIOLOGIC THERAPY OF GALEN ANEURYSMAL MALFORMATION IN A NEWBORN

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at 7 days of life, by catheterization of femoral artery, under general anesthesia, in the angiogra-phy laboratory of the Emergency University Hos-pital Bucharest. The purpose of the endovascu-lar treatment was to close at least 60-70 % of the direct arterial shunts involved mainly from the posterior circulation (left and right branches of posterior cerebral arteries and left and right su-perior cerebellar arteries were involved) of this mural type VGAM (Figure 5). During the first procedure, our neuro-interventional team suc-ceed to close 45-50% from the total flow (Figure 6) by obstructing two direct shunts, one from right superior cerebellar artery using 2 detach-able coils and a mixture of 80% Glubran and 20% Lipiodol, and the second one from left pos-terior cerebral artery with a mixture of 60% Glu-bran and 40% Lipiodol. The most challenging technical aspect was to close the first shunt due to the huge aspiration effect and to avoid the pulmonary artery direct embolization; we were obliged to use coils and also a higher Glubran concentration. The second endovascular treat-ment was imposed by clinical presentation and had taken place just 7 days after the first one, even the best option, if it should be possible from cardiac point of view, is to have at least 3 months interval (Figure 7). The micro-catheteri-sation was this time more difficult due to impor-tant tortuosity of the selected two branches (Fig-ure 8) one from right internal carotid artery and

one from posterior circulation. In the end of the procedure almost 80% of the flow was reduced (Figure 9) and no contrast extravasation, respec-tive active bleeding was observed during this procedure. At the trans fontanel echography done 2 hours post procedure a high suspicion of intra-ventricular haemorrhage was evocate and finally confirmed by CT scan.

Our opinion is that this delayed rupture was initiated due to acute thrombosis of the vein of Galen aneurysm itself, an important quantity of proteolytic enzymes being realised and leading to acute wall rupture. This mechanism is well knotweed and described in the delayed giant in-tracerebral aneurysms rupture post endovascular treatment and represents a terrifying complica-tion in our field.

In the next 3 hours an external ventricular drainage was put in emergency by neurosurgery team in the hope that this intra-ventricular haemorrhage could be tolerated.

The second intervention was considered nec-essary because of the unsatisfactory response of the cardiac function after the first embolization (systolic pulmonary pressure measured echocar-diographically had become 60mmHg, but with signs of decreased biventricular systolic func-tion). Indeed, after the second procedure a sig-nificant improvement in cardiorespiratory state of the baby was noticed: the murmur at the an-terior fontanel disappeared, the heart rate de-creased, Harzer sign disappeared, the heart size decreased on chest X ray and echocardiography, systolic pressure in main pulmonary artery de-creased to 39 mmHg with normalized biventric-ular systolic function.

Despite improvement in hemodynamics, the clinical course of the baby was unfavorable from neurological point of view: decreased tone, re-activity, reflexes, evolving to coma, due to a ce-rebral hemorrhage (intraventricular and periven-tricular) with secondary hydrocephaly. There were debates regarding the opportunity of anti-coagulant therapy (thrombosis of the cerebral sinuses is often a complication of embolization), but since the newborn had prolonged coagula-tion time, the treatment was not initiated. Exter-nal ventricular drainage was instituted which evacuated approximately 50 ml of cerebrospinal fluid, with hematic content, during 10 days. De-spite complex therapeutic measures the baby died at 17 days of life. q

FIGURE 9. Control after embolization (fi nal aspect)

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DISCUSSION AND CONCLUSION

Along with the continuous medical develop-ment the pathologies related to arteriove-

nous malformation, including vein of Galen an-eurysm was repetitively described and upgraded. Formed by the confluence of the two internal cerebral veins among to the basal vein of Rosen-thal, vein of Galen aneurysm in 1989 was classi-fied into three different types: choroidal, mural and dilatation of vein of Galen secondary to in-creased blood inflow as a consequence of the arteriovenous malformation (9). Only the last type is considered to be actually the vein of Ga-len aneurysm. The first two types are character-ized by the dilatation of the embryonal median vein of prosencephalon. Our case can be includ-ed in the third type; the aneurysm in this case had an important size due to the blood drainage supplied by the arteriovenous malformation.

Regarding the manifestations of this condi-tion, they depend on the age of patient and on the proportion of the indirect signs (cardiomega-ly, pulmonary hypertension, fetal hydrops) when diagnosed antenatally. The proportion of the an-tenatal diagnosis is declared to be about 40% (10). Imaging methods as angiography, ultraso-nography, computed tomography (CT) and mag-netic resonance imaging are essential for a clear and certificated diagnosis. In the neonatal period the aneurysm of the vein of Galen is diagnosed with the help of pulsed Doppler ultrasonography and color flow mapping. In our ultrasound case, a dilated sagittal sinus was observed and a mid-line supratentorial cystic structure which on col-ors Doppler presented a turbulent vascular flow, respectively a pronounced multicolored mosaic pattern; in this conditions the diagnosis was clar-ified at 28 weeks of gestation. In the neonatal period, clinical manifestations may be represent-ed by macrocephaly, loud intracranial bruit and dilatation of orbital veins, seizures, hydrocepha-lus (caused by the obstruction of the aqueduct of Sylvius by the dilated aneurysm) and subarach-noid hemorrhage (10). In our cases the signs of cardiac failure were developed after 32 weeks of gestation, after the arteriovenous malformation was diagnosed; in that moment, a mild tricuspid insufficiency was present among to a mild right cardiomegaly. In childhood, the patient may present with headache, cognitive dysfunction or a focal neurological deficit caused by the “steal”

phenomenon, with the diversion of the blood to the aneurysm and poor circulation into the pa-renchyma, resulting brain infarcts and periven-tricular white matter lesions (11).

One of the greatest complications that could appear in a gigantic dilatated vein of Galen is the thrombosis of the aneurysm; two main patho-physiological ways are involved in the spontane-ous thrombosis of vein of Galen: slow blood flow and impediment to the venous outflow (12). This complication is encountered rarely, latest case was described by Konovalov in 2002 (13) and is confirmed by CT and MRI when varying degree of calcification are seen.

The aneurysm of the vein of Galen is consid-ered to arise between 6 and 11 weeks of gesta-tion as a result to an insult to the cerebral vascu-lature. This appears to be more frequent on male fetuses (14). In our case, a female infant was born. Clinically the symptomatology was domi-nated by signs of heart failure. While in the most cases medical stabilization of heart failure suf-fices while preparing for interventional proce-dure, in our case it was impossible.

The treatment of choice is represented by vascular embolization. There were reported good after-procedural results and good neuro-logical outcome (15). Acute cardiac failure, such as in our case necessitates an emergency mode intervention, that is preferred to be performed early in order to prevent irreversible cardiac fail-ure and brain injuries. The success of the embo-lization is dominated by the necessity of know-ing accurately the vascular anatomy of the vein of Galen aneurysm. The MRI is preferred pre-interventional due to the fact that it can “freeze” fetal movements (16).

The aneurysm of the vein of Galen is a rare and severe arteriovenous malformation, with an overall rate of survival of 58% (17). It is consid-ered that above the major impact on the cere-bral parameters and heart function, the only measurement that can predict a better or poorer diagnosis is the volume of the aneurysm. As far as we know this is the only case of Galen aneu-rysmal malformation in Romania which benefi-ciated of embolization by interventional treat-ment in neonatal period. q

Conflict of interests: none declared.Financial support: none declared

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ULTRASOUND PRENATAL DIAGNOSIS AND EMERGENCY INTERVENTIONAL RADIOLOGIC THERAPY OF GALEN ANEURYSMAL MALFORMATION IN A NEWBORN

340 Maedica A Journal of Clinical Medicine, Volume 11 No.4 2016

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