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Adult sacrococcygeal teratoma with coccygectomy: A case report with a review of the literature

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Luis Daniel Diaz-Aguilar at University of California, San Diego
Luis Daniel Diaz-Aguilar
Sergei Terterov
Sergei Terterov
Sergei Terterov
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Rudi Scharnweber
Rudi Scharnweber
Rudi Scharnweber
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Catherine Merna
Catherine Merna
Catherine Merna
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Abstract

Background Saccrococcygeal teratomas (SCT) are derived from embryonic germ cell layers. They frequently present at the base of the coccyx within the pelvis. While these tumors are common in children, they are exceedingly rare in adults. In adults, a majority of these tumors are intrapelvic and associated with a low risk of malignant transformation. Therefore, this contributes to a good prognosis following resection of mostly benign lesions. Case Description An adult female with chronic pelvic pain presented with a sacral teratoma. She failed conservative treatment and underwent a coccygectomy with an en-bloc excision of the tumor. Microscopic histological analysis showed no evidence of immature or malignant elements, confirming the diagnosis of a mature, benign, cystic SCT. Conclusions Mature SCTs in adults are rare malignant lesions. In this case, the patient was cured following primary surgical excision requiring en-bloc coccygectomy.
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SNI: Spine
Case Report
Adult sacrococcygeal teratoma with coccygectomy: A case report
with a review of the literature
Daniel Diaz‑Aguilar, Sergei Terterov, Rudi Scharnweber, Catherine Merna, Stephanie Wang,
Shayan Rahman1
Department of Neurosurgery, David Geffen School of Medicine at UCLA, 1Department of Neurosurgery, Kaiser Permanente Medical Center, Los Angeles,
California, USA
E‑mail: *Daniel Diaz‑Aguilar ‑ Ldiazaguilar@mednet.ucla.edu; Sergei Terterov ‑ steterov@mednet.ucla.edu; Rudi Scharnweber ‑ rscharnweber@mednet.ucla.edu;
Catherine Merna ‑ catmerna@gmail.com; Stephanie Wang ‑ StephanieWang@mednet.ucla.edu; Shayan Rahman ‑ Shayan.X.Rahman@kp.org
*Corresponding author
Received: 20 June 17 Accepted: 15 August 17 Published: 24 October 17
Abstract
Background: Saccrococcygeal teratomas (SCT) are derived from embryonic germ
cell layers. They frequently present at the base of the coccyx within the pelvis.
While these tumors are common in children, they are exceedingly rare in adults. In
adults, a majority of these tumors are intrapelvic and associated with a low risk of
malignant transformation. Therefore, this contributes to a good prognosis following
resection of mostly benign lesions.
Case Description: An adult female with chronic pelvic pain presented with a sacral
teratoma. She failed conservative treatment and underwent a coccygectomy with
an en‑bloc excision of the tumor. Microscopic histological analysis showed no
evidence of immature or malignant elements, conrming the diagnosis of a mature,
benign, cystic SCT.
Conclusions: Mature SCTs in adults are rare malignant lesions. In this case,
the patient was cured following primary surgical excision requiring en‑bloc
coccygectomy.
Key Words: Adult, mature teratoma, sacral coccygectomy
INTRODUCTION
Sacrococcygeal teratomas (SCT) are multipotential
cell tumors most commonly seen in neonates
but only rarely in adults. SCTs carry a female
preponderance of 4:1, and a prevalence of 1:30,000
births.[2] A vast majority of SCT are benign and have
a low potential for malignancy. Although surgical
resection carries an excellent prognosis, there is a
high 37% postoperative recurrence rate for these
lesions.[2,3] Here, we present the case of an adult
female with chronic pelvic pain and an SCT, who was
successfully treated with an en‑bloc coccygectomy
resulting in successful gross total removal of her
sacral teratoma.
How to cite this article: Diaz-Aguilar D, Terterov S, Scharnweber R, Merna C,
Wang S, Rahman S. Adult sacrococcygeal teratoma with coccygectomy: A case report
with a review of the literature. Surg Neurol Int 2017;8:260.
http://surgicalneurologyint.com/Adult-sacrococcygeal-teratoma-with-
coccygectomy:-A-case-report-with-a-review-of-the-literature/
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DOI:
10.4103/sni.sni_224_17
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Surgical Neurology International 2017, 8:260 http://www.surgicalneurologyint.com/content/8/1/260
CASE DESCRIPTION
Clinical presentation
A 23‑year‑old female presented with pelvic pain. Magnetic
resonance imaging(MRI) of the pelvis revealed a complex
cystic structure anterior to the coccyx [Figure 1].
The lesion, measuring approximately 3 × 2 × 4 cm,
contained both cystic and proteinaceous components
consistent with the diagnosis of a pre‑sacral intraspinal
teratoma; notably, it did not enhance with contrast.
The differential diagnosis included meningocele and
chordoma[Figure2]. Afine needle biopsy anterior to the
coccyx revealed scattered histiocytes and keratinocytes
consistent with the diagnosis of an Altman type IV SCT.
She failed conservative measures to treat her intractable
pelvic pain, and ultimately underwent successful surgical
intervention at the age of 26.
Surgery
The patient underwent an en‑bloc posterior
coccygectomy.Following exposure of the coccyx and lower
part of the sacrum, the rectum was carefully dissected
away from the tumor. An osteotome cut through the
coccyx6 cm above the tip [Figure3]. The final tumor
specimen was 1.9×2.5×1.6 cm. Gross analysis revealed
an irregular tumor with cystic nodules attached to a
firm portion of the coccyx[Figure4]. The microscopic
analysis confirmed a benign, mature sacrococcygeal cystic
teratoma. The patient was discharged home without any
complications. One year later, her she had no tumor
recurrence or residual symptoms/signs.
DISCUSSION
Incidence and symptoms of sacrococcygeal
teratoma
SCT are the most common fetal neoplasm. They
account for 50% of teratomas in children but are rarely
seen in adults.[3] The clinical presentation in adults
includes bowel dysfunctions, urinary incontinence, lower
back pain, and/or venous engorgement of the lower
limbs.
Figure 4: Histology of the mass demonstrating various types of
mature tissue elements including lobules of smooth muscle, adipose
tissue, stratied squamous epithelium (a, H and E, ×4; b, H and E, ×20)
respiratory type ciliated epithelium (c, H and E, ×40; and mucous
glandular tissue adjacent to stratified squamous epithelium
(d, H and E, ×10)
d
c
b
a
Figure 3: A solid cystic tumor (a, gross) containing yellow
seromucoid uid (b, gross)
b
a
Figure 2: Coronal CT (a) and 3D reconstruction (b) of the coccyx
status post resection of previously identied teratoma in the
precoccygeal region
b
a
Figure 1: Axial T1‑weighted (a), T2‑weighted fat‑saturated (b),
sagittal T1‑weighted post‑gadolinium fat‑saturated (c), coronal
T1‑weighted (d), and T2 fat‑saturated (e) images demonstrating
a complex precoccygeal cystic structure with multiple septations
and uid content of different signals and uid levels
d
c
b
a
e
Surgical Neurology International 2017, 8:260 http://www.surgicalneurologyint.com/content/8/1/260
Sacrococcygeal teratoma characteristics in adults
SCTs are divided into Types I–IV (criteria proposed by
Altman et al.) [Table 1]. Most adult SCTs are typeIII
and typeIV; they are cystic and less liable to demonstrate
malignant transformation(incidence 1–2%) compared to
those seen in children and infants. Adult SCT are also
primarily intrapelvic with no obvious physical cutaneous
or dermatologic presence, and are often recognized
following unresolved symptoms associated with a tumor
mass compressing adjacent organs(e.g.,vagina, uterus, or
rectum).[4,5]
Histopathology and use of markers
Mature SCT can be distinguished from immature
teratomas by the presence of epithelial structures
along with well‑distinguished cartilage and muscular
tissue. Immature teratomas contain primitive
combinations of germ layers with occasional mature
elements.[1] The final diagnosis of mature vs. immature
teratoma is dependent on histopathological examination.
Several biochemical markers, including β‑human chorionic
gonadotropin (β‑hCG) and α‑fetoprotien (AFP), are
used for initial screenings or the detection of tumor
recurrences. However, their application is less applicable
to mature teratomas as they often secrete fewer of these
markers than immature or malignant teratomas.[2]
Predominance
Computed tomography and MRI are both vital
preoperative diagnostic studies for the visualization of a
SCT.[2] Our patient’s studies revealed a lobulated solid
and cystic mass in the presacral area anterior to the
coccyx without calcifications.[5,7] These tumors frequently
contain multiple cystic or solid components with
necrotic areas that are highly suspicious for malignant
transformation; however, the reported lesion was
benign.[6] This SCT was curable with surgical excision/
coccygectomy.
CONCLUSION
SCT, rarely observed in adults, are frequently benign,
slowly growing intrapelvic masses best seen on MR
studies. Mature benign SCT are highly susceptible to
complete and early excision, without the additional need
for chemotherapy or radiation.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
REFERENCES
1. Al-Essa A-A, Malik TA, Baghdadi MK, Tayeb El AA. Adult sacrococcygeal
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2. Allsopp G, Sgouros S, Barber P, Walsh AR. Spinal teratoma: Is there a
place for adjuvant treatment? Two cases and a review of the literature. Br
J Neurosurg 2000;14:482-8.
3. Altman RP, Randolph JG, Lilly JR. Sacrococcygeal teratoma: American Academy
of Pediatrics Surgical Section Survey-1973. J Pediatr Surg 1974;9:389-98.
4. Audet IM, Goldhahn RT, Dent TL. Adult sacrococcygeal teratomas. Am Surg
2000;66:61-5.
5. Bruneton JN, Diard F, Drouillard JP, Sabatier JC, Tavernier JF. Primary
retroperitoneal teratoma in adults: Presentation of two cases and review
of the literature. Radiology 1980;134:613-6.
6. Miles RM, Stewart GS. Sacrococcygeal teratomas in adult. Ann Surg
1974;179:676-83.
7. Panageas E. General diagnosis case of the day. Primary retroperitoneal
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Table 1: Classification of sacrococcygeal teratomas[3]
Altman type Location Incidence (%) Malignant (%)
I Entirely outside pelvis 46.5 8
II Mostly outside pelvis 34.5 21
III Mostly inside pelvis 8.75 34
IV Entirely inside pelvis 12.75 38
... Sacrococcygeal teratoma is a tumour that develops in the sacrococcygeal area that incorporate tissue from many primitive germ layers. [6] Its cause remains unknown [7] The classification of SCT according to AAPSS American Academy of pediatrics surgical section (AAPSS) in 1973 described as follows. ...
... The clinical manifestation of SCT is determined by its location and extent. In Sacro-coccygeal teratoma early detection and management are important because it has higher risk of malignancy transformation which increases with age [6]. Sacro-coccygeal teratoma can cause critical disturbance in defecation function of infant although this disease is extremely rare benign tumour. ...
... Pain in the sacrococcygeal area and a palpable presacral lump were reported by patient. [6]. The differential diagnoses based on radiological features include: meningocele, rectal duplication cyst, lipoma, and liposarcoma [11]. ...
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Introduction: The Sacro-coccygeal teratoma is rare tumour that appears near the distal end of the tailbone in new-borns (coccyx). It's the most common congenital tumour, and it can appear at any time throughout pregnancy. It occurs in about 1/35000 to 1/40000 of all live births. Female new-borns are more likely to have this birth defect than male ones. Sacro-coccygeal teratoma can cause critical disturbance in defecation function of infant although this disease is extremely rare benign tumour. Appropriate early recognition can prevent progression to this devastating outcome. In this case report, we present a Sacro-coccygeal teratoma originating from gluteal region. The case is used to highlight important nursing management, clinical consideration and how to prevent further complications. We describe a Case of Sacro-coccygeal teratoma in 5 year girl child presenting with increased gluteal swelling of 15 days duration. In addition she had difficulty in passing stool and urine and also difficulty in sitting.
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... There's no known cause for teratomas yet [2]. Children frequently get these tumors, which can also be detected prenatally [3], but they are extremely uncommon in adulthood [4] with only a few cases documented in the literature [5]. Adult incidence ranges from 1 in 40 000 to 1 in 63 000, with a 3:1 female-to-male ratio [2,6,7]. ...
... SCTs are the most common type of fetal tumors, and they account for about half of all teratomas in children [4]. SCTs have an unclear etiology; however, they are hypothesized to arise from the migration of totipotent stem cells to the coccygeal region [2]. ...
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Sacrococcygeal teratoma (SCT) in adults is very rare with only a few cases documented in the literature, adult prevalence varies between 1 in 40 000 and 1 in 63 000. Most SCTs are located either mainly extra-pelvic (types I and II), which are more commonly seen in neonates; however, mainly intra-pelvic tumors (types III and IV) are more typical in adulthood. Extra-pelvic teratomas are extremely rare in adults. When SCT manifests in an adult, it appears as a slow-growing tumor without symptoms and usually manifests after becoming large enough to cause compression symptoms. SCT has a 1-2% probability of malignant transformation. Herein, we report a 20-year-old female, who was diagnosed with lower back swelling since childhood that increased in size over the last 2 months; imaging revealed an extra-pelvic mass. This case describes an atypical scenario for SCT, which was successfully managed with surgery. The histopathology report confirmed the diagnosis.
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... Most adults can be asymptomatic or present with pressure symptoms [6,9,10]. Symptoms of mass effect can result in constipation, pain in sacrococcygeal region, bladder dysfunction, venous engorgement of lower limbs and neurological symptoms [1,3,9,11]. Intra-pelvic masses are common in adults, in contrast to neonates where more than 90% present as extra pelvic masses [1,3,4,9]. ...
... MRI has superior specificity and accuracy than CT to visualize the soft-tissue extent in SCT [4]. Elevation of serum tumor markers such as alpha-feto protein (AFP), carcinoembryonic antigens (CEA), human chorionic gonadotropin (HCG) and lactate dehydrogenase (LDH) are usually suggestive of malignant transformation [1,4,7,11]. ...
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Background Sacrococcygeal teratomas are tumors originating from pluripotent embryonic germ cell layers located in the fetal coccyx. These tumors are highly vascular if they undergo malignant transformation. Typically, they are found in infants and children and occasionally can be diagnosed prenatally. Adult cases are very rare, and represent tumors present since birth with delayed detection. Case presentation We describe a case of a giant sacrococcygeal teratoma in a 25 years old female college student presenting with right gluteal swelling of 4 months’ duration. In addition to the huge disfiguring mass on the perineal area, she also had lower abdominal pain, urinary complaints, and difficulty with ambulation. Discussion Pre-operative impression was of a sacrococcygeal mass and histopathology following complete surgical excision revealed a sacrococcygeal teratoma. She recovered well after surgery with no radiologic evidence of recurrence at six months. Conclusion Although rare, sacrococcygeal teratoma should be considered as a differential diagnosis for female adults presenting with perineal and/or pelvic masses. Complete surgical excision remains the mainstay of treatment.
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... • Sacrococcygeal teratoma is the most common congenital tumour with an incidence of 1:35,000-40,000 live births [17], a prevalence of 1:30,000 live births [18] and a femaleto-male ratio of 4:1 [19]; • Mediastinal teratoma with reported frequency percentages ranging from 2.6 to 4 [20,21] and represents approximately 60-70% of mediastinal germ cell tumours [22] and lower incidence in children compared to adults with a ratio of approximately 1:5 [23]; • Gastric teratoma is very rare and only 102 cases have been described in the literature [24]; • Retroperitoneal teratoma is rare and accounts for 5% of all childhood teratomas [3] and is even rarer in adults [25]; • Intracranial teratoma is rare and accounts for a greater than 2-4% of intracranial tumours in children [26]; • Cervical teratoma is very rare and accounts for only 1.5-5% of all teratomas [27]; ...
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... Symptoms of the mass effect can lead to constipation, pain in the sacrococcygeal region, bladder dysfunction, venous engorgement of the lower extremities, and neurological symptoms [6] [7] [8] [9]. ...
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... The age of 8 female and 2 male patients ranged from 20 to 77 years (mean 51:1 ± 20:6). Diagnoses included carcinoid tumor [11], paracoccygeal teratoma [12,13], epithelial cyst [14], mature teratoma [15], sacrococcygeal chordoma [16], sacral giant cell tumor [17], rectal carcinoma metastasis [18], and benign dermoid cyst [19]. Coccygectomy was the first-line therapy in 8 patients following failure of conservative management or was performed after failure of antibiotics or chemotherapy ( Table 1). ...
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