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,

Extragonadal germ cell tumors (GCTs) are defined as
GCTs arising outside the testes or the ovaries.
Extragonadal GCTs typically develop in midline loca-
tions and involvement sites vary with age. In adults, the
anterior mediastinum, retroperitoneum, and the pineal and
suprasellar regions are most commonly involved. In
infants and young children, the most common site is the
sacrococcygeal region, which accounts for 40–70% of
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
prevalence in a 3:1 to 4:1 ratio (3,4). At birth, the great
majority of SCTs are benign (2). Benign SCTs can
manifest malignant degeneration and malignant trans-
formation can occur in children with advanced age.
Approximately 70% of SCTs are malignant at the patient
age of nine months (3,5–7). Furthermore, although SCTs
are diagnosed as benign tumors, they can recur as
malignant SCTs after surgical resection (5,7,8). Therefore,
prompt surgical resection is required and imaging studies
play an important role in confirm-ing the diagnosis,
demonstrating intra-abdominal extension and effects on
adjacent structures. In this
imaging features, complications due to mass effects of the
tumor, and clinical management of SCTs.
Pathogenesis
SCTs develop at the base of the coccyx and are thought to
be derived from Hensen’s node (primitive knot), a
rounded and elevated area at the cranial end of the
primitive streak (9–11) (Fig. 1). The primitive streak
1Department of Radiology and Research Institute of Radiology,
University of Ulsan College of Medicine, Asan Medical Center,
Seoul, Republic of Korea
2Department of Pathology, University of Ulsan College of
Medicine, Asan Medical Center, Seoul, Republic of Korea
3Department of Radiology, Pusan National University Children’s
Hospital, Yangsan-Si, Republic of Korea
4Department of Radiology, Kangwon National University
Hospital, Chuncheon-Si, Republic of Korea
Corresponding author:
Young Ah Cho, Department of Radiology and Research Institute
of Radiology, University of Ulsan College of Medicine, Asan
Medical Center, 88 Olympic-ro 43-gil, Songpa-gu, Seoul 138-736,
Republic of Korea. Email: ped.yacho@gmail.com
Yoon et al.
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
end of the bilaminar embryonic disc. This structure
appears at the beginning of the third embryonic week and
is formed by proliferation of the ectodermal cells, which
move towards the midline of the embryo. The primitive
streak consists of totipotent cells, which are able to
transform into any type of cells. The primitive streak
forms the intra-embryonic mesoderm, septum
transversum, and notochord. This structure determines the
future craniocaudal axis of the embryo and demar-cates
the embryo into left and right halves. After for-mation of
the intra-embryonic mesoderm, the primitive streak
normally regresses and completely disappears by the end
of the fourth week or becomes an insignificant structure in
the sacrococcygeal region of the embryo. If totipotent cells
of the primitive streak remain after the fourth week, these
cells give rise to a SCT (10,12). The degree and type of
differentiation of these totipotent cells decide the specific
type of tumor (13). Primordial germ cells, which are
totipotent cells found in the gona-dal ridge of a late
embryo, remaining in a primitive undifferentiated state
lead to embryonal carcinoma. If differentiation proceeds
beyond the embryonal carcin-oma stage into a pathway of
embryonic structure pro-duction, mature or immature
teratoma may occur. Differentiation along an extra-
embryonic pathway leads to yolk sac tumor or
choriocarcinoma (13).
Histologic classification
By definition, teratomas are composed of more than one
germ cell layers (ectoderm, mesoderm, and endo-derm)
(10). SCTs contain a variety of tissues from more than one
germ cell layer. SCTs are categorized into three
histopathologic types: mature teratomas; imma-ture
teratomas; and malignant teratomas (14). Mature SCTs
consist of well-differentiated tissues from various
237
somatic sites. These tissues include functional glandular
structures such as pancreatic Langerhans cells or seba-
ceous glands and fully developed bones, hair, and teeth.
Immature teratomas have immature embryonic elem-ents
or incompletely differentiated tissue foci. A primi-tive
neuroectodermal feature is commonly present. Mature and
immature teratomas are considered as benign tumors.
SCTs that contain malignant elements are considered to be
malignant tumors. The most common malignant elements
are yolk sac components. Other malignant elements, such
as embryonal carcin-oma and primitive neuroectodermal
tumors, can be present. It is possible that microfoci of
malignant elem-ents may escape being sectioned on
pathologic examin-ation. Therefore, very extensive
sectioning of the SCT is necessary to avoid missing the
presence of malignant components. The majority of SCTs
are benign (5,6,15– 20). Histologically mature SCT
accounts for about 50– 60% and immature SCT accounts
for about 18–34% of sacrococcygeal GCTs (1,5,8,18,21).
Malignant SCTs account for approximately 12–14% of
sacrococcygeal GCTs (8,18). There is a grading system
for SCTs as follows: grade 0 ¼ tumors contain only
mature tissue; grade 1 ¼ tumors contain rare foci of
immature tissues; grade 2 ¼ tumors contain moderate
amounts of imma-ture tissues; grade 3 ¼ tumors contain
large amounts of immature tissue with or without
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
in the range of 11–16.4% (23–25). The recurrence rates of
mature teratomas, immature teratomas, and malignant
teratomas are 0–26%, 12–55%, and 0–36%, respect-ively
(23–25). Both mature and immature teratomas can
undergo malignant transformation (24,26) and malignant
teratomas are also prone to benign recur-rence (Fig. 2).
238
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-
sion shows a large heterogeneous enhancing mass (asterisk) in
the presacral area. Adjacent sacral bone destruction (arrows) and
epidural extension (arrow head) are shown.
Clinical considerations
SCTs are currently detected by prenatal screening ultra-
sound (US). When the tumor is too large, dystocia or
difficulty in delivery can occur. After birth, the tumor is
present as a skin-covered tail mass. If the tumor is quite
large, the skin covering the mass can be ulcerated and
necrotized. The anus can be displaced anteriorly (Fig. 3).
Rectal examination to look for the internal component
should be considered. Patients with SCTs can be
asymptomatic or have signs of obstruction of the rectum
or colon, such as constipation or frequent stools, or
obstruction of the bladder neck (27).
The reported incidence of congenital malformations
associated with SCTs is in the range of 5–43% (27–30).
The majority of these are anorectal and genital malfor-
mations. Other various anomalies have been reported
including spinal dysraphism, sacral agenesis, hip dis-
location, meningocele, Currarino’s triad (anorectal
malformation, sacrococcygeal osseous defect, presacral
mass), and vertebral anomalies (10,27,31,32).
Malignant SCTs may have elevated tumor markers
(3,19,33). The most commonly produced tumor marker is
alpha fetoprotein (AFP) because yolk sac compo-nents are
the most common malignant elements (3,34,35). Other
malignant elements can produce beta human chorionic
gonadotropin (beta-hCG). Therefore, serum AFP and
beta-hCG levels should be evaluated at the initial
diagnostic work-up to look for malignant
Acta Radiologica 59(2)
Fig. 3. Immature SCT in a male neonate. Clinical
photograph depicts a large tail mass with swelling of the
overlying skin, scrotum, and both thighs. The anal
opening, which is anteriorly displaced, is present through
the tumor (arrow). The patient suffered from constipation
even though the tumor was success-fully resected.
components and assessed to monitor tumor relapse during
the follow-up periods.
SCTs are generally diagnosed at birth or in infancy.
Serum AFP level is normally elevated in this period
because AFP is produced by the yolk sac, fetal liver, and
fetal gastrointestinal tract (36). Therefore, inter-pretation
of an elevated AFP level at birth or in infancy requires
careful attention. Normally, elevated serum AFP level
decreases and reaches adult level by eight months after
birth. Serial follow-up levels should be measured to
properly interpret this tumor marker (37).
Morphologic classification
SCTs arise from the base of the coccyx and may con-
tinuously grow in the posterior direction forming an
external protrusion, or in the anterior direction dissect-ing
and distorting surrounding structures such as the rectum,
vagina, and bladder (5,10). Thus, the tumor may have
internal and external components. Based on this
morphologic characteristic, the American Academy of
Pediatrics Surgery Section Survey suggested a clas-
sification system, which categorizes SCTs into four types
(Fig. 4) (5): type I SCTs are predominantly exter-nal with
a minimal presacral component; type II SCTs are present
externally but have a significant intrapelvic component;
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
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
extending into the abdominal cavity; type IV SCTs
occupy the presacral area with no external presenta-tion.
The most common type of SCT is type I, followed by
types II, III, and IV (8). Type IV SCTs are detected later
than types I, II, and III because they have no external
components. Malignant elements are more fre-quently
observed in types IV SCTs and are lowest in SCTs with a
large external component (1,5,21). In order to have
accurate communication with surgeons, radi-ologists
should appropriately use the morphologic clas-sification
of SCTs when describing the type of mass, because
complete resection of the mass is the corner-stone for
management of this tumor.
Imaging techniques
Prenatally, most SCTs are diagnosed in utero by US
screening. If a SCT is suspected on prenatal US screening,
in utero fetal magnetic resonance imaging (MRI) is
recommended where available. In utero fetal MRI can
depict accurate tumor size and characteris-tics including
intraabdominal extent and effect on adjacent organs (38–
40). This additional information provided by in utero fetal
MRI can be helpful in prenatal counseling as well as
preoperative surgical planning. In case of in utero fetal
MRI was not per-formed, abdominopelvic MRI should be
obtained after birth in order to evaluate tumor
characteristics and relation with adjacent other structures.
US is also frequently performed as a first imaging
modality due to easy accessibility and lack of radiation.
However, US has intrinsic disadvantage of operator
dependency, and MRI is preferred imaging modality.
Computed tomography (CT) can be another option
Acta Radiologica 59(2)
for postnatal imaging of SCTs; however, it is not
generally used due to exposure to ionizing radiation.
Imaging pattern-based approach
for diagnosis of SCT
Sacrococcygeal teratomas consist of variable tissues from
all three germ cell layers. Therefore, imaging find-ings of
SCTs are heterogeneous and variable. These tumors may
have variable amounts of cystic and solid components.
Not infrequently, they may show a com-bination of both
cystic and solid components. Although imaging findings
of SCTs cannot predict the histologic subtypes of the
tumors, benign SCTs are more likely to have cystic
components, calcification, and prominent fat tissue than
malignant SCTs. The presence of hemorrhage and/or
necrosis within the mass is more likely to be suggestive of
malignant SCTs.
Cystic components of SCTs are usually seen in benign
tumors (Fig. 5). Mild peripheral rim enhance-ment along
the cystic wall may present after gadolinium contrast
material injection. Contents of the cystic com-ponent can
be variable due to various degrees of fat, hemorrhage, or
other materials. Therefore, imaging characteristic of cystic
components can be altered according to the internal
contents.
Calcification is frequently seen in benign SCTs but it
can be present in case of malignant form (10,13,41).
Radiographs can depict calcifications, but CT is the most
sensitive imaging modality to demonstrate calcifi-cations.
However, considering the ionizing radiation hazard of CT
scans in neonates, CT is not routinely used to find
calcifications in SCTs.
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-
sound (US) images alone is not easy. CT and MR scans
are the best choice for demonstrating fat components, but
MRI is superior to CT considering the ionizing radiation
hazards of a CT scan.
Masses with predominantly solid components are
highly suggestive of malignant SCTs (Fig. 7). Solid
components are seen as hyperechoic areas by US. CT or
MRI plays an important role in demonstrating solid
components. On MRI, solid components may show iso-or
hyposignal intensity on T1-weighted (T1W) imaging and
iso- to high signal intensity on T2-weighted (T2W)
imaging. Solid portions show heterogeneous enhance-
ment on CT or MRI after intravenous contrast material
injection.
A significant amount of hemorrhage and/or necrosis is
highly suggestive of malignant SCTs (Fig. 7). Therefore,
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
destruction, invasion of surrounding structures, or
metastatic disease are also important clues indicating
malignancy.
Differential diagnosis between mature and immature
teratomas based on imaging features is limited. Immature
teratomas are more likely to be large in volume and show
complex cystic and solid masses (Fig. 8). Because SCTs
can generally have a variable
combination of various tissue components in an indi-
vidual case, even a malignant SCT can have a large cystic
component, calcification, or fat tissue. Radiologists should
be aware that prediction of histo-logic characteristics
based on their appearance on ima-ging studies is limited,
and the most important role of imaging is making a
diagnosis of a SCT and providing the necessary
information for surgical planning. CT and MRI are
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
another. However, MRI is superior to CT imaging in soft
tissue characterization.
Complications
Due to high output cardiac failure resulting from
arteriovenous shunting within the tumor and/or anemia
due to intratumoral hemorrhage, fetal hydrops can occur
(42). Fetal hydrops is known as a prognostic factor for
fetuses with SCTs, thus, prenatal careful monitoring is
mandatory (42). Hemorrhage or rupture may occur with
traumatic delivery because the SCT is a highly vascular
tumor (Fig. 7) (10,13).
SCTs are usually large enough to have a significant
mass effect on adjacent structures, and the mass effect
begins during fetal development. Various co-morbid-ities
due to mass effects such as obstructive hydrone-phrosis,
anterior displacement of the anorectum, bowel
distension, hypoplasia of pelvic muscles, mucocolpos or
hip dislocation, may be present on imaging studies
(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
displacement of the anus and hypoplastic pelvic floor
muscles, and these conditions may result in func-tional
problems such as fecal incontinence or constipa-tion. If
internal components of SCTs obstruct the urinary tract
during fetal development, obstructive hydronephrosis and
urinary bladder outlet obstruction can develop. These
conditions may cause neuropathic bladder, ureter
obstruction, and vesicoureteral reflux (43). Occasionally, a
large SCT may cause abnormal position of the fetus in
utero; therefore, hip dislocation may occur. These
conditions may be associated with potential long-term
sequelae despite successful surgical resection of the
primary tumor (44,45). Therefore, radi-ologists should
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
and interpret subsidiary imaging findings besides the
tumor itself.
Clinical management
Thanks to advances in prenatal detection of masses and
improvement in prenatal intervention, in utero open fetal
surgery is a treatment option for some high-risk SCT
fetuses. Minimally invasive in utero approaches have been
recently introduced to treat fetuses with hydrops.
Approaches include laser (46,47) or radiofre-quency
ablation techniques (48,49), urinary bladder drainage for
obstructive uropathy (50,51), and aspir-ation of cystic
components (52) (Fig. 10). Minimally invasive techniques
focus on inducing tumor necrosis, which allow the
surgeon to perform a lower-segment uterine incision
instead of a classic incision during Cesarean delivery.
After birth, early and complete surgical resection is key
to management of mature and immature SCTs, regardless
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
tumor. Complete excision is adequate for management of
mature and immature teratomas. Although immature
histology is known as a risk factor for recurrence (24),
adjuvant chemotherapy has no benefit in those patients
(35,54). Thus, observa-tion with complete resection is
recommended in patients with immature teratomas (34).
Platinum-based chemotherapy is the most important
component after surgical resection for treatment of
patients with malignant SCTs. Chemotherapy has been
used for patients with advanced disease before surgical
resection to reduce the tumor volume (35,56,57). Large,
hyper-vascular SCTs with arteriovenous shunting may
cause heart failure, hemolysis, rupture, or bleeding
(27,58).
Yoon et al.
Obstruction of the tumor-feeding arteries might be helpful
to prevent this challenging situation (Fig. 11). Thus,
preoperative embolization of the feeding arteries can
reduce blood loss during operations and enable faster and
safer tumor resection (58,59).
Conclusions
SCTs are the most common solid tumor in newborn
infants. Three histopathologic types exist: mature
teratomas; immature teratomas; and malignant tera-tomas.
Morphologic classification plays an important role in
predicting outcomes and for preoperative sur-gical
planning. 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. Occasionally,
complications such as rupture, bleeding, or co-morbidities
including urogenital and anorectal obstruction, hypoplasia
of pelvic muscle, or hip dislocation, may occur and those
imaging fea-tures should be interpreted in the radiologic
report. Surgical resection is the cornerstone of treatment.
When a tumor is too big for a baby to be delivered safely,
prenatal minimal invasive treatment could be one
treatment option.
Declaration of Conflicting Interests
The author(s) declared no potential conflicts of interest with
respect to the research, authorship, and/or publication of this
article.
Funding
The author(s) received no financial support for the research,
authorship, and/or publication of this article.
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