Cervical Benign Teratoma: Case Report and Review of Literature

Abstract

Cervical teratomas are extremely rare germ cell tumours and it is much more common in newborn than adults, and in contrast to the paediatric cases adult teratomas have been highly malignant. Cervical teratoma incorporates lesions arising in the anterior and posterior triangles of the neck. This tumor can reach enormous size and cause airway obstruction and patients should be quickly treated. Surgery is the primary modality of treatment as malignant transformation can occur. Hereby, we present a case of benign teratoma of neck in adult which was completely misdiagnosed preoperatively due to its rare occurrence in adults.Even though cervical teratoma of adult is extremely rare, it should be considered as an important differential diagnosis in patient of midline cystic neck swelling. Preoperative radiological investigations requires high index of suspicion. Complete surgical resection is recommended. We believe that upper cervicotomy approach is a safe and effective method for the treatment of mature cervical teratoma with a few protruding into the superior mediastinum.

Supplementary Information

The online version contains supplementary material available at 10.1007/s12070-023-03748-8.

Introduction

Teratomas are abnormal growths, benign or malignant, in which tissue derived from each of the three germinal layers is recognized. Cervical Teratoma (CT)are very uncommon [1, 2]. The malignancy potential of the lesion is determined by the degree of immaturity of the tissue [2, 3]. Germ cell tumors are extra-gonadal in 5–10% of all teratoma and the most commun extra-gonadal site is mediastinum [4]. The neck regions may be affected in rare cases [4–6]. CT occurred mainly in newborn, children and they are extremely rare in adult [1, 2] with high incidence of malignancy [1, 7, 8]. CT can reach enormous size causing airway obstruction and nearby structures compression, requiring surgery [2, 3]. We report the case of a 42-year-old woman operated for a benign CT, and we review CT adults in the literature.

Case Report

A previously healthy 42-year-old woman presented with a recurrent bronchopneumpathy associated to a slowly enlarging neck tumor noticed 2 months earlier. Clinical exam showed, an elastic soft nodule,4 × 5 cm in size,in the base of the left anterior neck. The tumor was not tender and was attached to the deeper portion of the neck. No palpable cervical lymph nodes were noted. A neck X-ray demonstrated a deviation of the trachea. Computerized tomography of the chest showed a well-defined, lobulated tumor mesuring 4 cm with predominant fat density, having smaller foci of soft tissue densities and calcifications, in the left paratracheal region of the lower neck, minimally extending to the upper thorax (Fig. 1). Surgery was performed through upper cervicotomy (Fig. 2), revealing a tumor separated from the thyroid. Intra-operatively,the tumor was well encapasuled. Adhesion between the capsule and the anterior neck mass and surrounding tissues was severe. The capsule wasn’t ruptured during dissection. The tumor was completely excised(Fig. 2). It measured 6 × 5 × 2 cm, showed many cystic lumina containing butter-like material and opaque fluid. The cyst wall of the tumor is lined with squamous epithelium, with smooth muscle tissue, sebaceous gland and sweat gland tissue visible in it, and fibrous tissue in the center. Histological exam of the specimen showed features that are in keeping with mature teratoma. The patient was discharged three days after operation.

Fig. 1.

A. Chest radiograph shows left neck swelling (arrow) with right tracheal deviation B. Axial CT image showing the cervical mass with tracheal deviation to the right. Septation within the mass is indicated by the arrow. C. Coronal CT image showing relationship of the mass to the great vessels. Dark area within the mass {arrow) corresponds to the lipomatous element and ….calcifications. D. Sagittal Axial views showing a well defined cystic and solid mass in upper mediastinum extending to the neck

Fig. 2.

Intraoperative appearance of cystic lesion (a) Initial appearance of neck mass adherent to trachea (b) Dissection of the well encapsulated cervical solid lesion (c) Specimen photograph of the excised cervical teratoma

Discussion

Teratomas are embryonal neoplasms composed of tissues foreign to the anatomic site of origin with all three blastodermic layers (ectoderm, endoderm, and mesoderm) [3, 9, 10]. The mediastinum is the second most common anatomic site of teratoma [8]. The Neck is one of the least common extra-gonadal sites [1, 9, 11]. CT constitutes only 1,5–5% of all teratomas [2–4, 6, 8, 10, 12]. CT in adulthood represents 10,6% of all CT with a high incidence of malignancy [3, 10]. They pre-dominate in females (75% of the cases) [3]. In the course of our review of the literature during 70 years we were able to find 19 documented cases of benign CT in adults, as listed in Table 1.The exact cause of CT is unknown [4, 5].Mediastinal teratomas are usually asymptomatic [6, 11]. If teratoma arises in the cervical region, it increase rapidly in the growth, so we note symptoms related to compression of surrounding structures,such as,dyspnea,dysphagia,or recurrent episodes of infection [6, 11]. Differential diagnosis of CT are a papillary carcinoma of thyroid with cystic formation, a metastasis from thyroid carcinoma,cystic squamous cell carcinoma,lymphangiomas,and bronchial cysts [3, 10, 11].

Table 1.

Documented cases of cervical teratoma in adults during 70 years (since 1954)

First author (year)	Age Sex	Size of tumor	Outcome
Cavellero (1954)	24 Female	-	-
Keyness (1959)	24 Male	-	15 months ; alive
Ohara (1959)	39 Male	10*7,8 cm	7 years ;alive
Muto (1968)	25 Female	5*5*3 cm	4 years ;alive
Woods(1978)	40 Female	-	12months ; alive
Mochizuki (1986)	26 Female	3*3*2 cm	16 Months ; alive
Endo (1992)	29 Male	-	-
Sawafuji (1993)	21 Male	-	19 Months; alive
Kuhel (1996)	32 Female	4,8*4*2 cm	22 Months; alive
Abe (1997)	21 Female	7,5*4,5*2,7	7 months ;alive
KHazama(2003) (12)	27 Female	10 cm	6 months ;alive
Omranipour (2007)	36 Male	5*5*8 cm
Gaurav (2008)	19 Male	16*7*2 cm	36 months ;alive
Alimehmeti (2013) (10)	25 Female	4 cm
Siow (2015) (4)	18 Male	10*6,5 cm	1 year ; alive
Ansari (2017) (3)	24 Male	3*4 cm	-
Lee (2018)	38 Male	6,5*5, 2*2, 2 cm	5 years ;alive
Birla Roy (2020)	18 Female	6 cm	-
Liu (2020)	33 Male	4*4*5 cm	9 months ; alive
Our Patient	42 Female	4*5 cm	1 month; alive
Total	20 cases	-	-

Chest x-ray can evoke CT [11]. Ultrasonography should demonstrate relationships between masses with thyroid gland and great vessels. Neck CT scan with coverage of mediastinum is essential to evaluate intrathoracic extension and assists planning surgery [2, 6, 11]. In our patient CT scan established the continuity of mediastinal mass into the neck. Magentic resonance imaging can be also very useful in evaluating the relationship of the tumor to the great vessels and vital structures [12]. Calcified structures present in only 26% of patients with matured teratomas have been reported to present the typical shadow [12]. When a CT is encountered in an adult,the surgeon should anticipate the possibility of a tedious dissection due to adherence to the pretracheal fascia or the thyroid and the great vessels [4, 8].

Complete surgical removal of CT is the curative treatment. It allows establishing the diagnosis and preventing life threatening complications [11]. Anatomically, CT lie in the visceral space between the anterior strap muscles of the neck and the pretrachea fascia [4]. For approach of this tumor,we did not require a median sternotomy as the inferior edge was just at the level of the thoracic inlet and we were able to retract it superiorly [4]. If the mass extends to the thoracic inlet or the supraclavicular region, a manubrial or sternal osteotomy should be performed [8]. Some surgeons suggest that surgical removal of a CT may involve the removal of a portion of or the entire thyroid gland [3, 5]. Some researches conclude that radiation therapy may be used before surgery or after surgery as an adjuvant therapy [5]. Chemotherapy immediately after surgery has also been used to treat individuals with cervical teratoma [5] but adjuvant chemotherapy is indicated when malignancy is confirmed [4].

In conclusion, even though CT of adult is extremely rare, it should be considered as an important differential diagnosis in adult of cystic neck [3]. The preliminary diagnosis can be suggested on preoperative investigations [2, 4]. Complete surgical resection is recommended due to unpredictable compressive complication and the high potential of malignant transformation [3, 4].

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Abbreviations

CT

Cervical teratoma

Author Contribution

Abdennadher Mahdi: conceived the cae report, contiubuted to writing, reviewing and finalization of the manuscript.

Ben Amara Kouathar: collected clinical details.

Abdelkebir Amina: approval of the final version.

Zribi Hazem: approval of the final version.

Ben Mansour Amani: Helped in data acquisition.

Sahnoun Imen: Helped in data acquisition.

Zairi Sarra: Evaluated of the final version.

Marghli Adel: Critical of the final version.

Funding

No.

Data Availability

Yes.

Declarations

Ethics Approval and Consent to Participate

Not applicable.

Consent for Publication

Yes.

Competing Interest

No.