Original Article

Acta Radiologica
2018, Vol. 59(2) 236–246
! The Foundation Acta
Sacrococcygeal teratomas in newborns: a Radiologica 2017
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DOI: 10.1177/0284185117710680
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Hee Mang Yoon1, Sun-ju Byeon2, Jae-Yeon Hwang3,

Jeong Rye Kim1, Ah Young Jung1, Jin Seong Lee1,
Hye-Kyung Yoon4 and Young Ah Cho1

Abstract
Sacrococcygeal teratomas are the most common solid tumor in newborn infants. The diagnosis is not difficult in many
cases; however, there should be additional information on imaging studies in order to manage those infants properly.
Details include histology, morphologic classification, complications such as rupture, bleeding, and mass effects on the
adjacent structures. Although imaging features cannot accurately predict the histologic subtypes of the tumors, thorough
evaluation of the imaging features can help distinguish malignant tumors from benign tumors. In this article,
pathogenesis, histological characteristics, clinical considerations, and morphologic characteristics will be discussed.

Keywords
Sacrococcygeal teratoma, germ cell tumor, neonate, congenital, computed tomography, magnetic resonance imaging
Date received: 24 December 2016; accepted: 29 March 2017

Introduction article, we discuss the pathogenesis, pathologic fea-tures,

imaging features, complications due to mass effects of the
Extragonadal germ cell tumors (GCTs) are defined as tumor, and clinical management of SCTs.
GCTs arising outside the testes or the ovaries.
Extragonadal GCTs typically develop in midline loca- Pathogenesis
tions and involvement sites vary with age. In adults, the
anterior mediastinum, retroperitoneum, and the pineal and SCTs develop at the base of the coccyx and are thought to
suprasellar regions are most commonly involved. In be derived from Hensen’s node (primitive knot), a
infants and young children, the most common site is the rounded and elevated area at the cranial end of the
sacrococcygeal region, which accounts for 40–70% of primitive streak (9–11) (Fig. 1). The primitive streak
teratomas (1). Sacrococcygeal teratomas (SCTs) are the
most common GCT in neonates occurring in
approximately 1 in 27,000 live births (2) with a female 1Department of Radiology and Research Institute of Radiology,
prevalence in a 3:1 to 4:1 ratio (3,4). At birth, the great University of Ulsan College of Medicine, Asan Medical Center,
majority of SCTs are benign (2). Benign SCTs can Seoul, Republic of Korea
2Department of Pathology, University of Ulsan College of
manifest malignant degeneration and malignant trans-
formation can occur in children with advanced age. Medicine, Asan Medical Center, Seoul, Republic of Korea
3Department of Radiology, Pusan National University Children’s
Approximately 70% of SCTs are malignant at the patient Hospital, Yangsan-Si, Republic of Korea
age of nine months (3,5–7). Furthermore, although SCTs 4Department of Radiology, Kangwon National University
are diagnosed as benign tumors, they can recur as Hospital, Chuncheon-Si, Republic of Korea
malignant SCTs after surgical resection (5,7,8). Therefore,
prompt surgical resection is required and imaging studies Corresponding author:
play an important role in confirm-ing the diagnosis, Young Ah Cho, Department of Radiology and Research Institute
of Radiology, University of Ulsan College of Medicine, Asan
demonstrating intra-abdominal extension and effects on Medical Center, 88 Olympic-ro 43-gil, Songpa-gu, Seoul 138-736,
adjacent structures. In this Republic of Korea. Email: ped.yacho@gmail.com
Yoon et al. 237

Fig. 1. Embryology in development of the caudal region. Drawing of a 16-day embryo (left) shows the primitive streak, which is a
longitudinal ridge of ectodermal cells at the caudal end of the bilaminar embryonic disc. Drawing of a 20-day embryo (middle)
demonstrates that the primitive streak moves caudally and undergoes regression. Drawing of a four-week embryo (right) shows
a regressed primitive streak and Hensen’s node located ventral to the developing sacrum and coccyx.

is a longitudinal ridge of ectodermal cells at the caudal somatic sites. These tissues include functional glandular
end of the bilaminar embryonic disc. This structure structures such as pancreatic Langerhans cells or seba-
appears at the beginning of the third embryonic week and ceous glands and fully developed bones, hair, and teeth.
is formed by proliferation of the ectodermal cells, which Immature teratomas have immature embryonic elem-ents
move towards the midline of the embryo. The primitive or incompletely differentiated tissue foci. A primi-tive
streak consists of totipotent cells, which are able to neuroectodermal feature is commonly present. Mature and
transform into any type of cells. The primitive streak immature teratomas are considered as benign tumors.
forms the intra-embryonic mesoderm, septum SCTs that contain malignant elements are considered to be
transversum, and notochord. This structure determines the malignant tumors. The most common malignant elements
future craniocaudal axis of the embryo and demar-cates are yolk sac components. Other malignant elements, such
the embryo into left and right halves. After for-mation of as embryonal carcin-oma and primitive neuroectodermal
the intra-embryonic mesoderm, the primitive streak tumors, can be present. It is possible that microfoci of
normally regresses and completely disappears by the end malignant elem-ents may escape being sectioned on
of the fourth week or becomes an insignificant structure in pathologic examin-ation. Therefore, very extensive
the sacrococcygeal region of the embryo. If totipotent cells sectioning of the SCT is necessary to avoid missing the
of the primitive streak remain after the fourth week, these presence of malignant components. The majority of SCTs
cells give rise to a SCT (10,12). The degree and type of are benign (5,6,15– 20). Histologically mature SCT
differentiation of these totipotent cells decide the specific accounts for about 50– 60% and immature SCT accounts
type of tumor (13). Primordial germ cells, which are for about 18–34% of sacrococcygeal GCTs (1,5,8,18,21).
totipotent cells found in the gona-dal ridge of a late Malignant SCTs account for approximately 12–14% of
embryo, remaining in a primitive undifferentiated state sacrococcygeal GCTs (8,18). There is a grading system
lead to embryonal carcinoma. If differentiation proceeds for SCTs as follows: grade 0 ¼ tumors contain only
beyond the embryonal carcin-oma stage into a pathway of mature tissue; grade 1 ¼ tumors contain rare foci of
embryonic structure pro-duction, mature or immature immature tissues; grade 2 ¼ tumors contain moderate
teratoma may occur. Differentiation along an extra- amounts of imma-ture tissues; grade 3 ¼ tumors contain
embryonic pathway leads to yolk sac tumor or large amounts of immature tissue with or without
choriocarcinoma (13). malignant yolk sac elements (22). However, this grading
system is not cor-related with prognosis, unlike that of the
ovarian tera-toma. The overall recurrence rate of SCTs is
Histologic classification in the range of 11–16.4% (23–25). The recurrence rates of
By definition, teratomas are composed of more than one mature teratomas, immature teratomas, and malignant
germ cell layers (ectoderm, mesoderm, and endo-derm) teratomas are 0–26%, 12–55%, and 0–36%, respect-ively
(10). SCTs contain a variety of tissues from more than one (23–25). Both mature and immature teratomas can
germ cell layer. SCTs are categorized into three undergo malignant transformation (24,26) and malignant
histopathologic types: mature teratomas; imma-ture teratomas are also prone to benign recur-rence (Fig. 2).
teratomas; and malignant teratomas (14). Mature SCTs
consist of well-differentiated tissues from various
238 Acta Radiologica 59(2)

Fig. 2. Malignant recurrence of a mature teratoma as an endo-

dermal sinus tumor in a female neonate. Initial sagittal T2W
imaging (left) shows a presacral mass (type IV) with cystic (arrow)
and fat (arrowhead) components within the tumor. The mass was
confirmed as a mature teratoma after surgical resection. Sagittal-
enhanced CT image (right) obtained two years after mass exci-
Fig. 3. Immature SCT in a male neonate. Clinical
sion shows a large heterogeneous enhancing mass (asterisk) in
photograph depicts a large tail mass with swelling of the
overlying skin, scrotum, and both thighs. The anal
the presacral area. Adjacent sacral bone destruction (arrows) and
opening, which is anteriorly displaced, is present through
epidural extension (arrow head) are shown.
the tumor (arrow). The patient suffered from constipation
even though the tumor was success-fully resected.
Clinical considerations
SCTs are currently detected by prenatal screening ultra- components and assessed to monitor tumor relapse during
sound (US). When the tumor is too large, dystocia or the follow-up periods.
difficulty in delivery can occur. After birth, the tumor is SCTs are generally diagnosed at birth or in infancy.
present as a skin-covered tail mass. If the tumor is quite Serum AFP level is normally elevated in this period
large, the skin covering the mass can be ulcerated and because AFP is produced by the yolk sac, fetal liver, and
necrotized. The anus can be displaced anteriorly (Fig. 3). fetal gastrointestinal tract (36). Therefore, inter-pretation
Rectal examination to look for the internal component of an elevated AFP level at birth or in infancy requires
should be considered. Patients with SCTs can be careful attention. Normally, elevated serum AFP level
asymptomatic or have signs of obstruction of the rectum decreases and reaches adult level by eight months after
or colon, such as constipation or frequent stools, or birth. Serial follow-up levels should be measured to
obstruction of the bladder neck (27). properly interpret this tumor marker (37).
The reported incidence of congenital malformations
associated with SCTs is in the range of 5–43% (27–30). Morphologic classification
The majority of these are anorectal and genital malfor-
mations. Other various anomalies have been reported SCTs arise from the base of the coccyx and may con-
including spinal dysraphism, sacral agenesis, hip dis- tinuously grow in the posterior direction forming an
location, meningocele, Currarino’s triad (anorectal external protrusion, or in the anterior direction dissect-ing
malformation, sacrococcygeal osseous defect, presacral and distorting surrounding structures such as the rectum,
mass), and vertebral anomalies (10,27,31,32). vagina, and bladder (5,10). Thus, the tumor may have
Malignant SCTs may have elevated tumor markers internal and external components. Based on this
(3,19,33). The most commonly produced tumor marker is morphologic characteristic, the American Academy of
alpha fetoprotein (AFP) because yolk sac compo-nents are Pediatrics Surgery Section Survey suggested a clas-
the most common malignant elements (3,34,35). Other sification system, which categorizes SCTs into four types
malignant elements can produce beta human chorionic (Fig. 4) (5): type I SCTs are predominantly exter-nal with
gonadotropin (beta-hCG). Therefore, serum AFP and a minimal presacral component; type II SCTs are present
beta-hCG levels should be evaluated at the initial externally but have a significant intrapelvic component;
diagnostic work-up to look for malignant type III SCTs have an apparent but small
Yoon et al. 239

Fig. 4. Morphologic classification of SCTs according to the American Academy of Pediatrics Surgery Section Survey. Type I is primarily
external in location. Type II has equal amounts of internal and external components with a dumbbell shape. Type III has a small external
component and is primarily located within the abdomen and pelvis. Type IV is entirely internal without a visible external component.

Fig. 5. Mature SCT in a male neonate. (a, b) Sagittal T2W imaging (a) and gadolinium-enhanced fat saturated T1W
imaging (b) show a multiseptated cystic mass (type II) with mild enhancement along the septa and cystic wall. (c)
Photomicrograph (hematoxylin and eosin [H&E] stain, 200) shows most of the tumor consists of mature epithelial tissues
(arrow), mesenchymal tissues (arrowhead), and mature neural tissues (asterisk).
240 Acta Radiologica 59(2)

Fig. 6. Mature SCT in a female neonate. (a) Sagittal T2W imaging demonstrates predominantly fatty mass with mostly
external location (type I). There are T2 hyperintense cystic components (arrows) with solid components (arrowheads). (b)
On photo-micrograph (H&E stain, 0.5), the tumor is mainly composed of mature mesenchymal tissues and there are
abundant fat tissues with intervening fibrous septa and mature chondroid tissue (asterisk).

external component with a significant pelvic mass for postnatal imaging of SCTs; however, it is not
extending into the abdominal cavity; type IV SCTs generally used due to exposure to ionizing radiation.
occupy the presacral area with no external presenta-tion.
The most common type of SCT is type I, followed by Imaging pattern-based approach
types II, III, and IV (8). Type IV SCTs are detected later
for diagnosis of SCT
than types I, II, and III because they have no external
components. Malignant elements are more fre-quently Sacrococcygeal teratomas consist of variable tissues from
observed in types IV SCTs and are lowest in SCTs with a all three germ cell layers. Therefore, imaging find-ings of
large external component (1,5,21). In order to have SCTs are heterogeneous and variable. These tumors may
accurate communication with surgeons, radi-ologists have variable amounts of cystic and solid components.
should appropriately use the morphologic clas-sification Not infrequently, they may show a com-bination of both
of SCTs when describing the type of mass, because cystic and solid components. Although imaging findings
complete resection of the mass is the corner-stone for of SCTs cannot predict the histologic subtypes of the
management of this tumor. tumors, benign SCTs are more likely to have cystic
components, calcification, and prominent fat tissue than
malignant SCTs. The presence of hemorrhage and/or
Imaging techniques necrosis within the mass is more likely to be suggestive of
Prenatally, most SCTs are diagnosed in utero by US malignant SCTs.
screening. If a SCT is suspected on prenatal US screening, Cystic components of SCTs are usually seen in benign
in utero fetal magnetic resonance imaging (MRI) is tumors (Fig. 5). Mild peripheral rim enhance-ment along
recommended where available. In utero fetal MRI can the cystic wall may present after gadolinium contrast
depict accurate tumor size and characteris-tics including material injection. Contents of the cystic com-ponent can
intraabdominal extent and effect on adjacent organs (38– be variable due to various degrees of fat, hemorrhage, or
40). This additional information provided by in utero fetal other materials. Therefore, imaging characteristic of cystic
MRI can be helpful in prenatal counseling as well as components can be altered according to the internal
preoperative surgical planning. In case of in utero fetal contents.
MRI was not per-formed, abdominopelvic MRI should be Calcification is frequently seen in benign SCTs but it
obtained after birth in order to evaluate tumor can be present in case of malignant form (10,13,41).
characteristics and relation with adjacent other structures. Radiographs can depict calcifications, but CT is the most
US is also frequently performed as a first imaging sensitive imaging modality to demonstrate calcifi-cations.
modality due to easy accessibility and lack of radiation. However, considering the ionizing radiation hazard of CT
However, US has intrinsic disadvantage of operator scans in neonates, CT is not routinely used to find
dependency, and MRI is preferred imaging modality. calcifications in SCTs.
Computed tomography (CT) can be another option SCTs with considerable fat components are usually
benign (13) (Fig. 6). Distinguishing fat components in
Yoon et al. 241

Fig. 7. Malignant SCTs in a female neonate. (a) Prenatal Doppler US image at 28 weeks and five days demonstrates a large,
predominantly solid sacrococcygeal mass with internal vascularity. (b) Clinical photograph shows massive bleeding and rupture
of the mass. Hypovolemic shock with disseminated intravascular coagulation (DIC) was developed in this patient due to
bleeding. After surgical resection, the mass was proven as a yolk sac tumor. (c) Axial contrast-enhanced CT image obtained
after surgery shows lobular contour with capsular retraction and areas of low attenuation in the liver, suggesting submassive
hepatic necrosis after prolonged DIC and hypovolemic shock. (d, e) The tumor was composed of 70% of immature teratomas (d)
and 30% of yolk sac tumor (e) with immunopositivity for alpha fetoprotein and spalt like transcription factor 4 (not shown).

SCTs from the normal subcutaneous fat tissue on ultra- components. On MRI, solid components may show iso-or
sound (US) images alone is not easy. CT and MR scans hyposignal intensity on T1-weighted (T1W) imaging and
are the best choice for demonstrating fat components, but iso- to high signal intensity on T2-weighted (T2W)
MRI is superior to CT considering the ionizing radiation imaging. Solid portions show heterogeneous enhance-
hazards of a CT scan. ment on CT or MRI after intravenous contrast material
Masses with predominantly solid components are injection.
highly suggestive of malignant SCTs (Fig. 7). Solid A significant amount of hemorrhage and/or necrosis is
components are seen as hyperechoic areas by US. CT or highly suggestive of malignant SCTs (Fig. 7). Therefore,
MRI plays an important role in demonstrating solid complex heterogeneous areas appearing in
242 Acta Radiologica 59(2)

Fig. 8. Immature SCTs in a female neonate. (a) Coronal T2W imaging shows a complex cystic and solid coccygeal mass.
(b) There is small amount of hyperintense fluid on T1W imaging, representing hemorrhage. (c) On gadolinium-enhanced
coronal T1W imaging, mild enhancement in the solid component and along the cystic wall is noted. (d) Photograph of the
cut surface of the mass depicts a solid mass with multiple small cystic portions. (e) Immature neuroepithelial components
are frequently observed on the microscopic finding, which is appropriate for immature teratoma, grade III.

the mass should be carefully evaluated. Sacral bony combination of various tissue components in an indi-
destruction, invasion of surrounding structures, or vidual case, even a malignant SCT can have a large cystic
metastatic disease are also important clues indicating component, calcification, or fat tissue. Radiologists should
malignancy. be aware that prediction of histo-logic characteristics
Differential diagnosis between mature and immature based on their appearance on ima-ging studies is limited,
teratomas based on imaging features is limited. Immature and the most important role of imaging is making a
teratomas are more likely to be large in volume and show diagnosis of a SCT and providing the necessary
complex cystic and solid masses (Fig. 8). Because SCTs information for surgical planning. CT and MRI are
can generally have a variable excellent modalities for depicting tumor
Yoon et al. 243

Fig. 9. Mature SCT in a female neonate. (a) Sagittal T2W imaging shows a large multicystic sacrococcygeal mass with internal
and external components. In order to release the urinary obstruction, a Foley catheter was inserted (arrow) in the urinary
bladder. (b) Coronal T2W imaging depicts fluid signal intensity mixed with intermediate signal intensity in the dilated vagina
(arrow), indicating mucocolpos. (c) Bilateral hydronephrosis (arrows) was noted on an axial T2W imaging. Asterisk indicates the
superior portion of the mass. (d) Dislocated bilateral hip joints (arrows) are seen on an axial T2W imaging.

extent and those two modalities are complementary to one distension, hypoplasia of pelvic muscles, mucocolpos or
another. However, MRI is superior to CT imaging in soft hip dislocation, may be present on imaging studies
tissue characterization. (10,27,31,32) (Fig. 9). These complications are fre-quently
accompanied by SCTs with significant internal
components. Large type II, III, or IV SCTs show anter-ior
Complications
displacement of the anus and hypoplastic pelvic floor
Due to high output cardiac failure resulting from muscles, and these conditions may result in func-tional
arteriovenous shunting within the tumor and/or anemia problems such as fecal incontinence or constipa-tion. If
due to intratumoral hemorrhage, fetal hydrops can occur internal components of SCTs obstruct the urinary tract
(42). Fetal hydrops is known as a prognostic factor for during fetal development, obstructive hydronephrosis and
fetuses with SCTs, thus, prenatal careful monitoring is urinary bladder outlet obstruction can develop. These
mandatory (42). Hemorrhage or rupture may occur with conditions may cause neuropathic bladder, ureter
traumatic delivery because the SCT is a highly vascular obstruction, and vesicoureteral reflux (43). Occasionally, a
tumor (Fig. 7) (10,13). large SCT may cause abnormal position of the fetus in
SCTs are usually large enough to have a significant utero; therefore, hip dislocation may occur. These
mass effect on adjacent structures, and the mass effect conditions may be associated with potential long-term
begins during fetal development. Various co-morbid-ities sequelae despite successful surgical resection of the
due to mass effects such as obstructive hydrone-phrosis, primary tumor (44,45). Therefore, radi-ologists should
anterior displacement of the anorectum, bowel carefully evaluate the urogenital system,
244 Acta Radiologica 59(2)

Fig. 10. A case of a SCT treated with prenatal radiofrequency ablation (RFA). Prenatal US at 22 weeks and five days
(left) shows a large sacrococcygeal solid and cystic mass. After cystic fluid aspiration with RFA at 26 weeks (right), the
cystic portion of the mass disappeared and size of the mass was reduced.

Fig. 11. A case of an immature teratoma treated with preoperative embolization. (a) IV contrast-enhanced coronal CT
scan dem-onstrates a type II SCT with tortuously enlarged tumor vasculature (arrows). (b) Abdominal aortography in the
lateral projection shows feeding arteries from the bilateral internal iliac arteries (c) After embolization of feeders using gel
foam, polyvinyl alcohol, and coils, decreased arterial flow was seen through the feeders.

gastrointestinal system (especially the anorectum), and hip After birth, early and complete surgical resection is key
and interpret subsidiary imaging findings besides the to management of mature and immature SCTs, regardless
tumor itself. of the size of tumor or the patient’s age (5,25,34,35,53–
55). Incomplete resection is a major cause for recurrence
(24,35); therefore, the coccyx should be resected with the
Clinical management
tumor. Complete excision is adequate for management of
Thanks to advances in prenatal detection of masses and mature and immature teratomas. Although immature
improvement in prenatal intervention, in utero open fetal histology is known as a risk factor for recurrence (24),
surgery is a treatment option for some high-risk SCT adjuvant chemotherapy has no benefit in those patients
fetuses. Minimally invasive in utero approaches have been (35,54). Thus, observa-tion with complete resection is
recently introduced to treat fetuses with hydrops. recommended in patients with immature teratomas (34).
Approaches include laser (46,47) or radiofre-quency Platinum-based chemotherapy is the most important
ablation techniques (48,49), urinary bladder drainage for component after surgical resection for treatment of
obstructive uropathy (50,51), and aspir-ation of cystic patients with malignant SCTs. Chemotherapy has been
components (52) (Fig. 10). Minimally invasive techniques used for patients with advanced disease before surgical
focus on inducing tumor necrosis, which allow the resection to reduce the tumor volume (35,56,57). Large,
surgeon to perform a lower-segment uterine incision hyper-vascular SCTs with arteriovenous shunting may
instead of a classic incision during Cesarean delivery. cause heart failure, hemolysis, rupture, or bleeding
(27,58).
Yoon et al. 245

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to prevent this challenging situation (Fig. 11). Thus, Study of the factors associated with recurrence in children
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Declaration of Conflicting Interests 76:754–759.
17. Okada T, Sasaki F, Cho K, et al. Management and out-come
The author(s) declared no potential conflicts of interest with
in prenatally diagnosed sacrococcygeal teratomas. Pediatr
respect to the research, authorship, and/or publication of this
Int 2008;50:576–580.
article.
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