AMERICAN JOURNAL OF PERINATOLOGY/VOLUME 9, NUMBER 5/6
Sept/Nov 1992
ULTRASOUND DIAGNOSIS OF IMMATURE CERVICAL TERATOMA: A CASE REPORT Charles J. Dunn, M.D., Dan L. Nguyen, M.D., and Joe C. Leonard, M.D.
ABSTRACT
Cervical teratomas are rare tumors of the neonatal period. They make up only about 5.5% of all neonatal teratomas, which are reported to occur in approximately 1:20,000 to 1:40,000 live births.1 The classic findings of teratomas, cystic and solid components with calcifications, appear to hold true even in the neonatal period and are accurately defined by ultrasound.2 Despite the usefulness of ultrasound, only two articles could be found in the recent radiology literature that address this subject.1-3 CASE REPORT
A live term female infant was born to a 20-year-old gravida 2, para 1, white woman. The pregnancy was reported to be uneventful, with only one prenatal visit, which occurred in the late third trimester. The infant was born by vertex vaginal delivery with Apgar scores of 8 ( — 2 color) and 9 (—1 color). She was noted to be in mild respiratory distress with some stridor and to have a large firm midline neck mass. She was immediately transferred to this institution for further evaluation. Initial ultrasound evaluation was performed shortly after admission and showed a primarily solid, bilobed mass with a few scattered small cystic areas (Fig. 1A). There were several small hyperechoic densities with acoustical shadowing, which were believed to represent calcifications (Fig. IB). The carotid vessels were noted to be displaced posteriorly and laterally by the mass (Fig. 1A). An initial diagnosis of cervical teratoma was made. A plain film of the neck confirmed the presence of a mass with calcificatiorfs. A contrast computed tomography scan confirmed displacement of the carotid vessels and showed posterior displacement and lateral compression of the trachea (Fig. 1C). A Technetium-99m pertechnetate scan showed thyroid tissue draped over the posterior superior aspect of the mass (Fig. ID).
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We report a case of cervical teratoma in a term female infant bom to a 20-year-old white woman. There are rare tumors in neonates, occurring in approximately 1 in 20,000 to 1 in 40,000 live births. These tumors are accurately defined by ultrasound. Although benign, cervical teratomas cause respiratory compromise. In this case the tumor was removed surgically with no recurrence at the 10-month follow-up.
The patient was taken to surgery where a 7 x 4.5 x 3 cm multilobulated, well-encapsulated mass plus one anterior and two posterior enlarged firm lymph nodes were removed. The tumor mass showed primarily immature neural tissue with glandular, cartilage nous, and bony components. The lymph nodes showed primarily primitive neural tissue. The diagnosis of immature teratoma with 3 of 3 metastatic nodes was made. The patient did well both during and after surgery and was discharged home on the 6th postoperative day. Follow-up magnetic resonance imaging of the neck showed no evidence of recurrent tumor at 5 and 10 months after discharge. DISCUSSION Due to the respiratory compromise caused by cervical teratomas and the otherwise benign nature of these tumors,4 immediate and complete surgical excision is the treatment of choice,35 and the need for prompt and accurate preoperative diagnosis is imperative. A mortality rate of 89% is reported without surgery compared with 15% with prompt surgery.4 Ultrasound imaging, because of its ability to characterize the varied composition of teratomas, as well as the relationship of the mass to the great vessels of the neck, makes this a primary imaging modality for diagnosis and treatment planning. The flexibility and portable nature of ultrasound makes it ideal for imaging acutely ill or restless infants. Despite this, there have been very few articles in the recent radiologic literature on this topic. Of particular interest with these tumors is their benign nature, despite apparent aggressive behavior. Mochizuki et al,4 in addressing their benign nature, mentions onlyfivereported cases of cervical teratomas with invasion or metastases to regional lymph nodes or distant organs in children under 2 years of age. At least two of these five
Oklahoma Medical Center, Department of Radiological Sciences, Oklahoma City, Oklahoma Reprint requests: Dr. Dunn, Department of Radiological Sciences, Oklahoma Medical Center, Box 26307, Oklahoma City, OK 73126 Copyright © 1992 by Thieme Medical Publishers, Inc., 381 Park Avenue South, New York, NY 10016. All rights reserved.
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Sept/Nov 1992
Figure 1. A: Transverse ultrasound of the neck S: spine; N: anterior lymph node; open arrow: carotid artery; arrow: cyst. B: Longitudinal ultrasound of the neck, to the right of midline. Arrow: calcification. C: Axial computed tomography scan of the neck. M: mass; T: trachea; open arrow: carotid artery. D: 99mTc pertechnetate scan. Lateral view of the neck. Open arrow: thyroid.
patients were reported to be well without recurrence following complete surgical excision, suggesting that a true malignant cervical teratoma is indeed extremely rare. In the unusual case in which metastatic lymph nodes are found at the time of surgery intraoperative frozen sections can be confusing, sometimes mimicking other tumors, such as neuroblastoma. Bale,6 in his classic article on cervical teratomas, commented on the predominance of primitive neuroblastic tissue in most immature cervical teratomas. This appeared to be true both in this case and in a previously unreported case seen at this institution in 1980. The earlier case showed a large cystic component and was also well demonstrated by ultrasound. An interesting note in both of these cases was the direct continuity of the tumor with the thyroid gland, suggesting a possible thyroid origin for the tumors. The 446 definition of thyroid teratomas has been debated in the
literature;7 however, the origin of these tumors appears to have little influence on clinical treatment.4 With the widespread availability of ultrasound and the need for prompt diagnosis and intervention in infants with cervical teratomas, it is important to be familiar with the ultrasound findings of this surgically correctable tumor.
REFERENCES 1. Teal LN, Angtuaco TL, Jimenez JF: Fetal teratomas: Antenatal diagnosis and clinical management. JCU 16:329332, 1988 2. Talmi YP, Sadov R, Dulitzky F, Finkelstein Y, Zohar Y: Teratoma of the mastoid region in a newborn. J Laryngol Otol 102:1033-1035, 1988
Downloaded by: University of Pennsylvania Libraries. Copyrighted material.
AMERICAN JOURNAL OF PERINATOLOGY/VOLUME 9, NUMBER 5/6
CERVICAL TERATOMA DIAGNOSIS/Dunn, Nguyen, Leonard 54-year experience at the Children's Hospital Medical Center. Ann Surg 198:398-410, 1983 6. Bale GF: Teratoma of the neck in the region of the thyroid gland: A review of the literature and report of four cases. Am J Pathol 26:565-579, 1950 7. Silberman R, Mendelson R: Teratoma of the neck; report of two cases and review of the literature. Arch Dis Child 35: 159-170, 1960
Downloaded by: University of Pennsylvania Libraries. Copyrighted material.
3. Trecet JC, Claramunt V, Larraz J, Ruiz E, Zuzuarregui M, Ugalde FJ: Prenatal ultrasound diagnosis of fetal teratoma of the neck. JCU 12:509-511, 1984 4. Mochizuki Y, Noguchi S, Yokoyama S, Murakami N, Moriuchi A, Aisaka K, Yamashita H, Nakayama I: Cervical teratoma in a fetus and an adult. Two case reports and review of literature. Acta Pathol Jpn 36:935-943, 1986 5. Tapper D, Lack EE: Teratomas in infancy and childhood. A
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Sept/Nov 1992
ULTRASOUND DIAGNOSIS OF IMMATURE CERVICAL TERATOMA: A CASE REPORT Charles J. Dunn, M.D., Dan L. Nguyen, M.D., and Joe C. Leonard, M.D.
ABSTRACT
Cervical teratomas are rare tumors of the neonatal period. They make up only about 5.5% of all neonatal teratomas, which are reported to occur in approximately 1:20,000 to 1:40,000 live births.1 The classic findings of teratomas, cystic and solid components with calcifications, appear to hold true even in the neonatal period and are accurately defined by ultrasound.2 Despite the usefulness of ultrasound, only two articles could be found in the recent radiology literature that address this subject.1-3 CASE REPORT
A live term female infant was born to a 20-year-old gravida 2, para 1, white woman. The pregnancy was reported to be uneventful, with only one prenatal visit, which occurred in the late third trimester. The infant was born by vertex vaginal delivery with Apgar scores of 8 ( — 2 color) and 9 (—1 color). She was noted to be in mild respiratory distress with some stridor and to have a large firm midline neck mass. She was immediately transferred to this institution for further evaluation. Initial ultrasound evaluation was performed shortly after admission and showed a primarily solid, bilobed mass with a few scattered small cystic areas (Fig. 1A). There were several small hyperechoic densities with acoustical shadowing, which were believed to represent calcifications (Fig. IB). The carotid vessels were noted to be displaced posteriorly and laterally by the mass (Fig. 1A). An initial diagnosis of cervical teratoma was made. A plain film of the neck confirmed the presence of a mass with calcificatiorfs. A contrast computed tomography scan confirmed displacement of the carotid vessels and showed posterior displacement and lateral compression of the trachea (Fig. 1C). A Technetium-99m pertechnetate scan showed thyroid tissue draped over the posterior superior aspect of the mass (Fig. ID).
Downloaded by: University of Pennsylvania Libraries. Copyrighted material.
We report a case of cervical teratoma in a term female infant bom to a 20-year-old white woman. There are rare tumors in neonates, occurring in approximately 1 in 20,000 to 1 in 40,000 live births. These tumors are accurately defined by ultrasound. Although benign, cervical teratomas cause respiratory compromise. In this case the tumor was removed surgically with no recurrence at the 10-month follow-up.
The patient was taken to surgery where a 7 x 4.5 x 3 cm multilobulated, well-encapsulated mass plus one anterior and two posterior enlarged firm lymph nodes were removed. The tumor mass showed primarily immature neural tissue with glandular, cartilage nous, and bony components. The lymph nodes showed primarily primitive neural tissue. The diagnosis of immature teratoma with 3 of 3 metastatic nodes was made. The patient did well both during and after surgery and was discharged home on the 6th postoperative day. Follow-up magnetic resonance imaging of the neck showed no evidence of recurrent tumor at 5 and 10 months after discharge. DISCUSSION Due to the respiratory compromise caused by cervical teratomas and the otherwise benign nature of these tumors,4 immediate and complete surgical excision is the treatment of choice,35 and the need for prompt and accurate preoperative diagnosis is imperative. A mortality rate of 89% is reported without surgery compared with 15% with prompt surgery.4 Ultrasound imaging, because of its ability to characterize the varied composition of teratomas, as well as the relationship of the mass to the great vessels of the neck, makes this a primary imaging modality for diagnosis and treatment planning. The flexibility and portable nature of ultrasound makes it ideal for imaging acutely ill or restless infants. Despite this, there have been very few articles in the recent radiologic literature on this topic. Of particular interest with these tumors is their benign nature, despite apparent aggressive behavior. Mochizuki et al,4 in addressing their benign nature, mentions onlyfivereported cases of cervical teratomas with invasion or metastases to regional lymph nodes or distant organs in children under 2 years of age. At least two of these five
Oklahoma Medical Center, Department of Radiological Sciences, Oklahoma City, Oklahoma Reprint requests: Dr. Dunn, Department of Radiological Sciences, Oklahoma Medical Center, Box 26307, Oklahoma City, OK 73126 Copyright © 1992 by Thieme Medical Publishers, Inc., 381 Park Avenue South, New York, NY 10016. All rights reserved.
445
Sept/Nov 1992
Figure 1. A: Transverse ultrasound of the neck S: spine; N: anterior lymph node; open arrow: carotid artery; arrow: cyst. B: Longitudinal ultrasound of the neck, to the right of midline. Arrow: calcification. C: Axial computed tomography scan of the neck. M: mass; T: trachea; open arrow: carotid artery. D: 99mTc pertechnetate scan. Lateral view of the neck. Open arrow: thyroid.
patients were reported to be well without recurrence following complete surgical excision, suggesting that a true malignant cervical teratoma is indeed extremely rare. In the unusual case in which metastatic lymph nodes are found at the time of surgery intraoperative frozen sections can be confusing, sometimes mimicking other tumors, such as neuroblastoma. Bale,6 in his classic article on cervical teratomas, commented on the predominance of primitive neuroblastic tissue in most immature cervical teratomas. This appeared to be true both in this case and in a previously unreported case seen at this institution in 1980. The earlier case showed a large cystic component and was also well demonstrated by ultrasound. An interesting note in both of these cases was the direct continuity of the tumor with the thyroid gland, suggesting a possible thyroid origin for the tumors. The 446 definition of thyroid teratomas has been debated in the
literature;7 however, the origin of these tumors appears to have little influence on clinical treatment.4 With the widespread availability of ultrasound and the need for prompt diagnosis and intervention in infants with cervical teratomas, it is important to be familiar with the ultrasound findings of this surgically correctable tumor.
REFERENCES 1. Teal LN, Angtuaco TL, Jimenez JF: Fetal teratomas: Antenatal diagnosis and clinical management. JCU 16:329332, 1988 2. Talmi YP, Sadov R, Dulitzky F, Finkelstein Y, Zohar Y: Teratoma of the mastoid region in a newborn. J Laryngol Otol 102:1033-1035, 1988
Downloaded by: University of Pennsylvania Libraries. Copyrighted material.
AMERICAN JOURNAL OF PERINATOLOGY/VOLUME 9, NUMBER 5/6
CERVICAL TERATOMA DIAGNOSIS/Dunn, Nguyen, Leonard 54-year experience at the Children's Hospital Medical Center. Ann Surg 198:398-410, 1983 6. Bale GF: Teratoma of the neck in the region of the thyroid gland: A review of the literature and report of four cases. Am J Pathol 26:565-579, 1950 7. Silberman R, Mendelson R: Teratoma of the neck; report of two cases and review of the literature. Arch Dis Child 35: 159-170, 1960
Downloaded by: University of Pennsylvania Libraries. Copyrighted material.
3. Trecet JC, Claramunt V, Larraz J, Ruiz E, Zuzuarregui M, Ugalde FJ: Prenatal ultrasound diagnosis of fetal teratoma of the neck. JCU 12:509-511, 1984 4. Mochizuki Y, Noguchi S, Yokoyama S, Murakami N, Moriuchi A, Aisaka K, Yamashita H, Nakayama I: Cervical teratoma in a fetus and an adult. Two case reports and review of literature. Acta Pathol Jpn 36:935-943, 1986 5. Tapper D, Lack EE: Teratomas in infancy and childhood. A
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