Prenatal Diagnosis of a Huge Foetal Immature Sacrococcygeal Teratoma

Page 1 of 6

Case report

Prenatal diagnosis of a huge foetal immature sacrococcygeal teratoma: our experience of a rare case and review Obstetrics & Gynecology of the literature M Varras1*, G Diakakis1, I Monselas1, CH Akrivis2

Abstract 6 - Introduction - increased blood flow into the tumour.- behaviour . Sacrococcygeal terato Pulsed Doppler showed the resist mas arise from the Hensen’s nodule Sacrococcygeal teratomas are rare- ance index of flow velocity wave- located on the anterior surface of the germ cell tumours associated with forms on the tumoural arteries to be- sacrum or coccyx11,12 and may be cystic, high perinatal and postnatal mor 0.51. Amnioreduction of 740 cc amni multilocular or cystic, and solid and tality and morbidity. The purpose of otic fluid was performed under ultra variable in size . our study was to report a case of a sonographic examination. Caesarean- The morbidity and mortality of foetal huge immature sacrococcygeal section was performed at 33 weeks’ the sacrococcygeal teratomas are low- teratoma diagnosed prenatally and- gestation due to profuse polyhydram when they are diagnosed prenatally8,9 managed successfully in the early nios via an upper vertical incision in and the surgical management is per Caseneonatal report period with surgical resec the uterus. After the stabilization of formed during the neonatal period . tion of the tumour. the newborn, tumour resection was- The prognosis is usually poor when successfully performed on the first they are diagnosed prior to 30 weeks’ A foetal mass with solid and× cystic day after delivery. Grossly, in the sur gestation and are predominantly solid components in the sacral region of- gical specimen the tumour measured with high vascularisation due to high- the foetus measuring 42 34 mm Conclusion18 cm in its maximum diameter and output cardiac failure, development- was detected during the foetal anat weighted 1 500 g. - of hydrops fetalis, premature delivery omy ultrasound scan, in a 28-year-- and intrauterine foetal demise. Arteri old Greek woman, gravida 2, para 1. Prenatal diagnosis and ultrasono- ovenous shunting through the tumour The foetal karyotyping showed nor graphic follow-up are needed for the causes the high-output cardiac failure. mal number of chromosomes 13, good prognosis of sacrococcygeal ter- The causes for the prematurity are 18 and 21 (46 ΧY). Polyhydramnios atomas. Prenatal intervention should usually the uterine distension from the was seen, at 31 weeks of gestation,- be considered when the foetus devel neoplasm and the polyhydramnios,- and the patient was admitted to our ops hydrops for foetal salvage or in while the intrauterine foetal demise hospital owing to a risk of prema cases with placentomegaly to avoid is secondary to the vascular steal syn ture labour. Ultrasound examination- Introductionthe maternal risk of mirror syndrome. drome and the increased metabolic7–9,13–15 showed the sacrococcygeal mass to demands of a huge neoplasm -. be enlarged with maximum diam In addition, the foetus is 16 at risk of- eter of 20 cm. There were no signs- Teratomas are the most common chronic foetal anaemia due to haem- of hydrops as demonstrated by the perinatal tumours developed from orrhage within the tumour . The suc absence of ascites, pleural or peri- multipotent stem cells, are usually- cessful management of sacrococcy cardial effusion or placentomegaly.- extragonadal and contain tissues of- geal teratomas depends on the mode Colour Doppler ultrasound exami ectodermal, endodermal and meso- of delivery 7,9,13 and the classification of nation showed extensive vasculari dermal origin, foreign to the1–3 ana the tumour . Delivery of the foetus- sation within the mass, suggesting tomic site, in which they arise . Ter through a caesarean section prevents atomas account for 25%–52% of all dystocia, tumour rupture14,15 and pro congenital tumours and have a yearly- fuse tumoural bleeding . The risk incidence of one in 3,4–720 000 to one in of sacrococcygeal teratoma rupture is *Corresponding author Email: [email protected] 40 000 live births . Sacrococcy increased during the ablative tumour 1 geal teratomas have an incidence8–10 of resection and particularly when the Third Department of Obstetrics and - Gynecology, “Elena Venizelou” General 35 000–40 000 live births and have- diameters of the neoplasm exceed 2 Maternity Hospital, Athens, Greece usually a female predominance . 10 cm. The middle sacral artery usu- Ioannina,Department Greece of Obstetrics and Gynecology They are classified as mature, imma ally derives the most blood supply “G. Chatzikosta” General State Hospital, ture or malignant based on their of these neoplasms. The progno histopathologic appearance and sis is influenced by the presence of Licensee OA Publishing London 2013. Creative Commons Attribution License (CC-BY)

Varras M, Diakakis G, Monselas I, Akrivis CH. Prenatal diagnosis of a huge foetal immature sacrococ-

For citation purposes: none declared. interests: none declared. Conflict of interests: Competing the final manuscript. as well read and approved design, and preparation of the manuscript, the conception, to All authors contributed rules of disclosure. ethical Ethics (AME) for Medical the Association All authors abide by cygeal teratoma: our experience of a rare case and review of the literature. OA Case Reports 2013 Jan 31;2(1):10. Page 2 of 6

Case report

immature elements in the neoplasm 4.5 mm (normal upper rate 6.00 mm). sacrococcygeal mass to be enlarged; and there is a benign behaviour in The spine appeared normal and the the lesion was with solid and cystic- cases with 2,15 only neuroectodermal morphology of the cerebellum as well. components and maximum diameter components . It is estimated that The estimated foetal weight was 368 g of 20 cm (Figures 1 and 2). Polyhy at delivery17,18 4%–20% have malignant by ultrasounds. - dramios was found. There were no- tissue . Severe clinical consequen- No other abnormalities were- signs of hydrops as demonstrated by ces and frequent recurrences are detected. A diagnosis of sacrococcy the absence of ascites, pleural or per observed when the neoplasms are dis geal teratoma was made. Foetal kary- icardial effusion or placentomegaly.- covered after the age of two months otyping was recommended. - Middle cerebral artery peak systolic- or the surgeons fail to perform However, during the first trimes velocity was normal. M-mode ultra proper coccygectomy or when they ter ultrasound scan for nuchal trans sound examination showed that nei- contain foci of yolk sac tumour, 2,19–23even- lucency thickness measurement at ther heart ventricle was dilated and- an optimal surgical24 resection . 12 weeks of gestation, the neoplasm the fractional shortening of both ven Gilcrease et al. suggested that micro- was not detected. The foetal crown– tricles were found normal. The infe scopic foci of yolk sac tumour can be- rump length was 62 mm and the- rior vena cava was not enlarged and identified in low-grade immature ter nuchal translucency thickness was the preload index of the inferior vena- casesatomas2 as well and there is a possibil 1.4 mm. No signs of cardiac dysfunc cava was normal. The heart rate was ity for malignant recurrences in these- tion, such as tricuspid regurgitation- 150 beats per minute. Colour Dop . In severe cases, sacrococcygeal- and/or absent or reverse flow in the pler ultrasound examination showed teratomas with hydrops are associ ductus venosus during atrial contrac extensive vascularisation within the ated with maternal risk for develop tion were found in the +foetus during mass, suggesting increased blood ment of mirror syndrome, which is the first trimester ultrasound5 scan. - a severe form of preeclampsia and25 is- A repeat scan at 24 weeks was associated with placentomegaly . - performed for amniocentesis and foe We report a case of a huge sacro tal karyotyping and for checking the coccygeal teratoma diagnosed pre development of the foetus. The foetal natally at 21 weeks’ gestation by biometry showed: BPD: 70.73 mm, two-dimensional ultrasound scan,- HC: 235.56 mm, ΑC: 202.15 mm, with foetal delivery via caesarean FL: 48.14 mm. The amount of amniotic section at 33 weeks because of pro fluid was normal. The estimated foetal fuse polyhydramnios and successful weight was 807 g by ultrasounds. The tumour resection management on foetal heart rate was 167 beats per Casethe first report day after caesarean section. 82minute. × ×The neoplasm contained both Figure 1: solid and cystic elements and measured 63 75 mm. The normal number Axial view of the foetus In a 28-year-old Greek+2 woman, gravida- of chromosomes 13, 18 and 21 was demonstrated the sacrococcygeal 2, para 1, during the foetal anatomy found (46 ΧY). The mutation DF508 for teratoma (31-week gestational age). ultrasound scan at 21 weeks of gesta cystic fibrosis was not detected. tion, a foetal mass in the sacral region Polyhydramnios was seen, at was× detected containing both solid 31 weeks of gestation, and the and cystic elements and measuring patient was admitted to our hospital- 42 34 mm. The neoplasm had incre owing to a risk of premature labour. ased vascularity. Foetal biometry was The patient stated that she had regu- normal for gestational age: biparietal- lar follow-up in a medical centre of head diameter (BPD): 50.00 mm, head- another city throughout the previ circumference (HC): 187.00 mm, cis ous gestational weeks. The foetal terna magma: 4.8 mm, transverse cer biometry showed: BPD: 91.40 mm, ebellar diameter: 21 mm, abdominal HC: 304.00 mm, ΑC: 299.00 mm,+ Figure 2: circumference (ΑC): 160.00 mm, femur FL: 64.20 mm. The ultrasonographic5 - length (FL): 32 mm, humeral length: age of the foetus corresponded to 33 Midline sagittal view of 33.00 mm, HC/AC: 1.17. The amount weeks. The foetus was with cephalic the foetus showed the sacrococcy of amniotic fluid was normal and presentation. The estimated foetal geal teratoma, consisting of cystic the umbilical cord had three vessels. weight was 2314 g by ultrasounds. and solid components (31-week gestational age). Foetal nuchalLicensee fold was OA measuredPublishing to LondonUltrasound 2013. examination Creative Commons showed the Attribution License (CC-BY)

Varras M, Diakakis G, Monselas I, Akrivis CH. Prenatal diagnosis of a huge foetal immature sacrococ-

- - flow into the tumour (Figure 3). Some days later, the risk of continu resection of the tumour on the first Pulsed Doppler showed the resist- ing with the pregnancy was explained day after delivery at the Department- ance index (RI) of flow velocity and, following counselling, a caesarean- of Paediatric Surgery. Postoperatively, waveforms on the tumoural arter delivery was performed at 33 weeks the baby did well. At the time of writ ies to be 0.51 (Figure 4). The patient of gestation through a midline verti ing he was over nine months of age was hospitalized and monitored with cal incision in the abdominal wall and- Pathologyand continued to do well. serial non-stress tests. Corticosteroid upper vertical incision in the uterus as Macroscopic examination therapy was initiated for pulmonary- well (Figure 5). A male infant weigh maturation and indomethacin for ing 4 000 g was delivered with Apgar- × 16 × preventive tocolysis. No cervical dila score of 5–6 at one minute and 7–8 Grossly, the surgical specimen of soft tation was found. Three days later- at five minutes (Figure 6). The new tissues had dimensions 20 13 cm, amnioreduction of 740 cc amniotic- born underwent emergency surgical weight 1 500 g and consisted of fibro- fluid was performed under the ultra adipose tissue and part of striated- sonographic examination. Consulta sionsmuscle 18 fibres × (Figure× 7). A relatively tion by paediatricians and paediatric circumscribed neoplasm with dimen- surgeons was given to the parent and 14 12 cm, which× was it was concluded that postpartum partially covered by a fusiform epi surgery should be performed. dermal piece measuring 17 13 cm, occupied the surgical specimen. The tumoural sections showed partially a cystic conformation× 8 × consisting of a cystic area underneath the skin with dimensions 12 3 cm and some smaller cystic areas with maximum- diameters between 0.8 and 2.00 cm, which were full of mucous materi als. Solid neoplasmatic areas were Figure 5: seen between × the × cystic areas; the larger solid neoplasmatic area had Delivery via caesarean Figure 3: - dimensions 5 4 3.5 cm and was section was performed in the foetus - in continuity with the cystic cavity, with the huge sacrococcygeal tumour. - Colour Doppler ultra which had maximum diameter of sonography showed extensive vascu 12 cm. They had yellowish/whit larity of the immature sacrococcygeal ish hue and elastic consistency with teratoma suggesting increased blood places of important haemorrhagic flow into the tumour. infiltration and necrosis. At the periphery of the tumour by one pole,

Figure 6:

The premature infant was- Figure 4: born at 33 weeks of gestational age.- The appearance of the huge sacrococ Pulsed Doppler ultrasono cygeal tumour was shown in the new- Figure 7: graphy showed the resistance index- born lying in the right lateral decubitus (RI) of flow velocity on the tumoural position immediately after the caesar 20 × 16 × The surgical specimen arteries to be 0.51 (31-week gesta ean section. The combined weight of after the initial resection measured tional age). the tumour and baby was 4 000 g. 13 cm; its weight was 1 500 g. Licensee OA Publishing London 2013. Creative Commons Attribution License (CC-BY)

Varras M, Diakakis G, Monselas I, Akrivis CH. Prenatal diagnosis of a huge foetal immature sacrococ-

- opposing to the epidermal covering, type or mucosal of endocervical type an encapsulation of portions of- there was recognised× a continuous agglomerations of seromucinous glan large venous and arterial vascular fractured part of cartilage and coccyx dules. Characteristics were: (i) the- branches and growth of islets of foe Microscopicalbone measuring examination 1.5 0.7 cm. presence of multiple individual islets tal liver parenchyma and glia in the of foetal hepatic parenchyma in vari- wall of the middle venous vases of ous stages of development and foetal the area of the co-excised cartilage- Histological type of the epidermal pancreatic tissue and foetal gastroin tissue were found. The operative piece, subcutaneous adipose tissue testinal structures in various stages margins showed neoplastic expan and striated muscular fibres with- of development; (ii) the emergence of sion to the fibro-adipose and dense localisation of extragonadal tumour multiple bi-layer clear-cell glandules fibrous connective tissue. There was from germ cells. Teratoma consist with sub-nuclear or extra-nuclear no tumoural infiltration in the two ing of immature tissues of embryonic vacuoles, focally contiguous with high tiny nodes with maximum diameter type and partially mature tissues of cellularity aggregations of asteroid/- of 0.4 and 0.5 cm respectively, which adult type, with representation of the- roundish cells without marked nuc were found within the co-excised three blastic layers with prevalence lear atypia× and low mitotic activ Immunohistochemistryfibro-adipose tissue. of the ectoderm (50%), notable pres ity (3–7 mitotic divisions/10 optic - ence of the endoderm (30%) and the fields 40); (iii) few cystic cavities- - mesoderm (20%). and communicating cavernous spaces The immunohistochemical exami The ectoderm was represented- with clear-celled investment or invest nation showed expression of kera by prevalence of cerebral substance ment from highly eosinophilic cells; tin 8.18 in the epithelial structures under the form of mature neuro (iv) aggregations of follicles of thyroid and expression of GFAP in the glia. glia and neurons, oligodendroglial gland in growth phase. - Also, expression of synaptophysin in cells, ependymal tubules and many The mesoderm was represented the neurons and expression of EMA cavities invested by ependyma and from the meninge, the smooth mus in the investment tubules and the chorioid plexus. Characteristic was cle fasciae in developmental phase lumen of the glandular structures the noticeable presence (20% of the- and organising in smooth muscle including clear-cell glandules were total volume) of immature neuro- layer, the striated muscle fibres of found. In addition focal expression of epithelium with mild to severe cellu embryonal type, the multiple sites Glypican 3 in the clear-cell glandules- larity, which was composed of small of immature mesenchyma, the areas was detected. Expression of AFP was and average size cells with scarce of embryonal and mature adipose not observed in the epithelial struc cytoplasm, thin-walled nucleus tissue, the islets of immature and tures including the few clear-cell without evident nucleoli, absence mature hyaline cartilage, the islets- glandules. The expected expression of notable nuclear atypia with × high of neo-formed coarse bone tissue of AFP and Glypican 3 in the islets mitotic activity (15–50 mitotic and the osteoid with mild hyperpla of foetal liver was observed as well. divisions per 10 optic fields 40)- sia of the osteoblasts. Multiple foci of The cell proliferation marker Ki-67/ and with formation of nodules, extramedullary haematopoiesis and MIB-1 was detected heterogeneously tubules and focally papules. In addi important haemorrhagic infiltration in the epithelial structures (5%) and tion, recognised were sites and- in some areas, and ischaemic type in the immature foci of the immature cavities with investment of mela necrosis (20%) and focal deposits of Discussionneuroepithelium (15%–20%). nochrostic epithelium, neuronal fas calcium salts were also observed. ciae and agglomerations of ganglia Extension of infiltrative type was - cells and cavities with epidermal observed in the consecutive striated In our case, the tumour was an investment, presence of chorion in muscle fibres and in the co-excised extragonadal teratoma of the sacro developmental phase and adnexal portion of cartilage and bone tissue- coccygeal area of the immature type particles and spheres of squamous and beyond, in the form of cerebral- with a maximum diameter of 18 cm non-keratinized epithelium. - substance and cavities lined by chori and maturity level 3. In the tumour The endoderm was represented oid plexus or clear cells. Also, expan- there was representation of all the- with multiple cavities invested by res sion of the tumour was observed in- three blastic layers with prevalence- piratory type epithelium, other coated the form of cavities coated with res of ectoderm elements, mostly neu with cylindrical or transitional type- piratory epithelium type or muci roectoderm, important representa epithelium with squamous metaplasia,- nous gastric type or colonic cells up tion of the endoderm and visible cavities invested with gastric epithe to the reticular dermis of the skin representation of the mesoderm. lium and colonic mucosa, other cavi and without expansion to the skin. Infiltrative type extension of the ties lined with serosal paramesonefric neoplasm in the adjacent striated Licensee OA Publishing LondonAdditionally, 2013. Creative within theCommons tumour, Attribution License (CC-BY)

Varras M, Diakakis G, Monselas I, Akrivis CH. Prenatal diagnosis of a huge foetal immature sacrococ-

muscle fibres and co-excised portion and surgical trauma to hypogastric In our case the foetal karyotyping was of cartilage/coccyx bone tissue and nerves during tumour resection- normal. No other foetal abnormalities- also beyond, in the adipose tissue cause damage to the innervation of were found in our patient; the only was observed. The surgical margins the lower urinary11,14,27–31 tract in sacrococ complication was the polyhydram showed extension of the tumour in cygeal teratoma . In addition,- nios from the 31st week to delivery the fibro-adipose tissue. - neuropathic bladder, vesicoureteric- by caesarean section. There were no- The gradation of immaturity of reflux, incontinence, recurrent uri ultrasonographic findings of foetal the extragonadal immature tera- nary tract infections and impo hyprops or abnormal foetal echocar tomas in the paediatric population tence are possible complications diographic findings. In patients within accordance to the criteria refer after resection10,29,32,33 of sacrococcygeal- sacrococcygeal teratomas greater- ring to the ovarian teratomas does teratomas . than 5 cm, caesarean section is rec not reflect the biological behaviour The prenatal diagnosis of the sac- ommended usually with upper verti18,26 of the tumour, which is determined- rococcygeal teratomas 12 can be easily cal uterine incision to avoid dystocia by the presence or absence of yolk made with conventional two-dimen or trauma during normal delivery . sac tumour elements. The immu sional ultrasound scan . In the first- In specific surgical centres,in foetalutero nohistochemical detection of AFP- trimester these neoplasms usually interventions have been performed and Glypican 3 is expected in islets- escape the ultrasonographic 12,13 diagno with some success including - of foetal liver, as in our case, with sis because of their small size . The open foetal surgery, radiofrequency out being a yolk sac tumour com advantages of the three-dimensional tumoural ablation, laser vessel abla- ponent. Detection of Glypican 3 in ultrasound scan are the definition of- tion and alcohol sclerosis in an clear-cell glandules with parallel the total volume of the neoplasm, its attempt to increase survival in foe EMA expression and absence of AFP- extension to the 12 pelvis and the pos tuses with hydrops, while25 the mothers expression does not document a sible skeletal involvement for a better Conclusionare still in good health . yolk sac tumour diagnosis of intes surgical excision . Rapidly growing- tinal type. Elements of other germ huge tumours with predominantly- cell tumours, including the yolk sac solid components are often recog Sacrococcygeal teratomas are rare tumour, were not recognised in the nised in cases with an immature his germ cell tumours associated with tumour of our case. tology as in our case or malignant high perinatal and postnatal mortality Recurrences after resections of histology. Also, in these cases a high- and morbidity. Prenatal intervention sacrococcygeal teratomas have been- nisedvascularisation during the colour should be considered when the foetus described and half of the recurrences Doppler17,34,35 examination is usually recog develops hydrops for foetal salvage or are malignant. As the tumour is che- . In our case, RI of the blood in cases with placentomegaly to avoid mosensitive, follow-up with serum- flow was 0.51 and it is in accordance Consentthe maternal risk of mirror syndrome. AFP levels and physical examina with the other literature reports, in tion for at least three years7,18,26 is recom which the blood flow RI is lower in the - mended postoperatively . The AFP immature or malignant than in mature Written informed consent was- levels are high in foetuses and return- sacrococcygeal teratomas< but not as obtained from the patient for publi to normal at the ninth postpartum low as in adult women with17,34,35 ovarian- cation of this case report and accom month. Therefore, any rapid postop cancer (cut-off levels 0.40) . The panying images. A copy of the written erative increase in AFP7,18,26 levels suggests differential diagnosis at mid-gesta consent is available for review by the tumoural recurrence . In our case, tion of a sacral12 cystic mass includes ReferencesEditor-in-Chief of this journal. there was no evidence of recurrence- meningomyelocele and urogenital until the ninth month of the follow-up anomalies . Although chromosomal- period despite the diagnosis of imma- anomalies have been found in cases- 1. Schwartz S, Raffel LJ, Sun CC, Waters E.- ture sacrococcygeal teratoma. with sacrococcygeal teratomas, foe An unusual mosaic karyotype detected Urologic complications of the con tal karyotyping is not usually rec through prenatal diagnosis with dupli - cation of 1q and 19p and associated genital sacrococcygeal teratomas ommended because tumoural cells teratoma development., Teratology., 1992, have been described such as uri can desquamate into the amniotic Oct;46(4):399–404., - nary retention due to bladder outlet- fluid and in case with normal foetal- 2. Sergi C Ehemann V Beedgen B compression of tumour followed by- chromosomes, foetal karyotyping by Linderkamp O Otto HF. Huge fetal sac secondary renal deterioration, olig amniocentesis may result in mosai- rococcygeal teratoma with a completely 1999 ohydramnios and pulmonary hypo cism. Therefore, in these cases, the12 formed eye and intratumoral DNA ploidy plasia. Compression or infiltration foetal karyotyping should be supple heterogeneity. Pediatr Dev Pathol. Jan–Feb;2(1):50–7. of the sacralLicensee nerves OA by Publishingthe tumour Londonmented 2013. with Creativefoetal blood Commons sampling Attribution. License (CC-BY)

Varras M, Diakakis G, Monselas I, Akrivis CH. Prenatal diagnosis of a huge foetal immature sacrococ-

Case report

, , -

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Varras M, Diakakis G, Monselas I, Akrivis CH. Prenatal diagnosis of a huge foetal immature sacrococ-