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Mushroom-shaped teratoma of the soft palate in a neonate: case report
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British Journal of Oral and Maxillofacial Surgery 48 (2010) e25–e26
Short communication
Mushroom-shaped teratoma of the soft palate in a neonate: case report Yin-hua Jiang a , Qin Zhou b , Jia-wei Zheng b,∗ , Yan-an Wang b a
Department of Oral and Maxillofacial Surgery, Lishui People’s Hospital and Lishui Stomatological Hospital. Lishui 323000, Zhejiang Province, China Department of Oral and Maxillofacial Surgery, Ninth People’s Hospital, College of Stomatology, Shanghai Jiao Tong University School of Medicine, Shanghai 200011, China b
Accepted 7 April 2010 Available online 11 May 2010
Abstract Teratomas of the head and neck are uncommon congenital lesions, which are rarely seen in the soft palate. We here reported a male neonate with a soft palate teratoma associated with soft palate cleft. The most common presenting symptom of oral teratoma is feeding problems. The tumour looks like a mushroom, and was successfully treated with surgery. Feeding returned to normal by removing the mass, and the patient is under follow up for selective operation of the cleft palate. This case is unique because teratoma in a mushroom shape was not reported before. © 2010 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Teratomas; Soft palate cleft; Surgery
Introduction Teratomas of the head and neck are uncommon congenital lesions that are rarely seen in the soft palate. A teratoma was defined by Weaver et al. as a tumour consisting of multiple tissues that are not indigenous to their site of origin.1 The most common sites are the sacrococcyx, anterior mediastinum, testicle, ovary, or retroperitoneum.1,2
4.5 × 3.5 × 3.0 cm in size, prevented oral feeding. It was connected to the soft palate by a peduncle 0.5 cm in diameter. In addition to the mass, he also had an incomplete cleft of the soft palate. He was operated on under general anaesthesia at the age of 6 days. The mass was completely excised and an adjacent palatal flap was transferred to repair the surgical defect. Macroscopic examination of the excised mass showed mature bone, hair follicles, and cutaneous and glandular tissues (Fig. 2a and b). Histopathological findings confirmed a well-differentiated teratoma of the soft palate.
Case report A male infant was born by caesarean section after 38 weeks’ gestation, weight 3000 g, with a pedunculated mass protruding from his mouth (Fig. 1). Physical examination showed that the mass originated from the soft palate near the midline and looked like a mushroom. The mass, which was ∗ Corresponding author. Tel.: +86 21 63138341 5271; fax: +86 21 63121780. E-mail address: [email protected] (J.-w. Zheng).
Discussion Teratomas are defined as true neoplasms composed of multiple tissues foreign to the sites from which they originate. They have a reported incidence of 1:4000 live births with around 2–9% of these in the head and neck.1 Some reports have described a slight female predilection,3 but Carney et al.3 found malignant teratoma to be more common in men (5:4).
0266-4356/$ – see front matter © 2010 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
doi:10.1016/j.bjoms.2010.04.002
e26
Y.-h. Jiang et al. / British Journal of Oral and Maxillofacial Surgery 48 (2010) e25–e26
Fig. 1. Mushroom-shaped teratoma of the soft palate protruding from the oral cavity in a neonate with a cleft palate.
nant potential contain immature tissues; benign teratomas may undergo malignant change with age. Histopathological examination showed the mass in our case to be composed of multiple mature tissues with no juvenile cells, which confirmed a benign tumour. To be a teratoma a tumour must contain multiple tissues foreign to the site at which it is located, as was the case with our patient. Purely oral teratomas rarely cause respiratory distress, unlike other head and neck tumours, particularly laryngopharyngeal teratomas. When oral teratomas grow, they tend to protrude outside the mouth rather than posteriorly towards the oropharynx. Because newborns are obligate nasal breathers, obstruction of the oral compartment is relatively less important and usually causes feeding problems.4 Patients with oral teratomas often have coexistent anomalies such as cleft palate,4 cystic hygroma,5 and other multifocal teratomas,4,6 with cleft palate being the most common.7 This is the result of mechanical obstruction of the neoplasm preventing closure of the palatal shelves. Other coexisting malformations are bifid tongues and noses.8 Most oral and maxillofacial teratomas are benign and have a good prognosis after early complete resection. However, failure to diagnose or treat teratomas of the head and neck until late adolescence or adulthood causes a risk of malignant degeneration in up to 90%.9 Recurrence is rare after early treatment.4 In our case feeding became normal after the mass had been removed, and the patient has been listed for elective operation on his cleft palate.
References
Fig. 2. (a and b) Histological examination of the teratoma showing mature bone, hair follicles, and cutaneous and glandular tissues (haematoxylin and eosin, original magnification ×20).
Teratomas can be either benign or malignant. Benign ones consist of mature tissue components, while those with malig-
1. Weaver RG, Meyerhoff WL, Gates GA. Teratomas of the head and neck. Surg Forum 1976;27:539–42. 2. Shafer WG, Hine MK, Levy BM. Textbook of Oral Pathology. 4th ed. London: WB Saunders; 1983. pp. 312–13. 3. Carney JA, ThompsonDP, Johnson CL, Lynn HB. Teratomas in children: clinical and pathologic aspects. J Pediatr Surg 1972;7: 271–82. 4. Uchida K, Urata H, Suzuki H. Teratoma of the tongue in neonates: report of a case and review of the literature. Pediatr Surg Int 1998;14: 79–81. 5. Wakhlu A, Wakhlu AK. Head and neck teratomas in children. Pediatr Surg Int 2000;16:333–7. 6. Dudgeon DL, Isaacs Jr H, Hays DM. Multiple teratomas of the head and neck. J Pediatr 1974;85:139–40. 7. Levine AB, Alvarez M, Wedgwood J, Berkowitz RL, Holzman I. Contemporary management of potentially lethal anomaly: a successful perinatal approach to epignathus. Obstet Gynecol 1990;76:962–6. 8. Vandenhaute B, Leteurtre E, Lecomte-Houcke M, et al. Epignathus teratoma: report of three cases with a review of the literature. Cleft Palate Craniofac J 2000;37:83–91. 9. Buckley NJ, Burch WM, Leight GS. Malignant teratoma in the thyroid gland of an adult: a case report and a review of the literature. Surgery 1986;100:932–7.
British Journal of Oral and Maxillofacial Surgery 48 (2010) e25–e26
Short communication
Mushroom-shaped teratoma of the soft palate in a neonate: case report Yin-hua Jiang a , Qin Zhou b , Jia-wei Zheng b,∗ , Yan-an Wang b a
Department of Oral and Maxillofacial Surgery, Lishui People’s Hospital and Lishui Stomatological Hospital. Lishui 323000, Zhejiang Province, China Department of Oral and Maxillofacial Surgery, Ninth People’s Hospital, College of Stomatology, Shanghai Jiao Tong University School of Medicine, Shanghai 200011, China b
Accepted 7 April 2010 Available online 11 May 2010
Abstract Teratomas of the head and neck are uncommon congenital lesions, which are rarely seen in the soft palate. We here reported a male neonate with a soft palate teratoma associated with soft palate cleft. The most common presenting symptom of oral teratoma is feeding problems. The tumour looks like a mushroom, and was successfully treated with surgery. Feeding returned to normal by removing the mass, and the patient is under follow up for selective operation of the cleft palate. This case is unique because teratoma in a mushroom shape was not reported before. © 2010 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved. Keywords: Teratomas; Soft palate cleft; Surgery
Introduction Teratomas of the head and neck are uncommon congenital lesions that are rarely seen in the soft palate. A teratoma was defined by Weaver et al. as a tumour consisting of multiple tissues that are not indigenous to their site of origin.1 The most common sites are the sacrococcyx, anterior mediastinum, testicle, ovary, or retroperitoneum.1,2
4.5 × 3.5 × 3.0 cm in size, prevented oral feeding. It was connected to the soft palate by a peduncle 0.5 cm in diameter. In addition to the mass, he also had an incomplete cleft of the soft palate. He was operated on under general anaesthesia at the age of 6 days. The mass was completely excised and an adjacent palatal flap was transferred to repair the surgical defect. Macroscopic examination of the excised mass showed mature bone, hair follicles, and cutaneous and glandular tissues (Fig. 2a and b). Histopathological findings confirmed a well-differentiated teratoma of the soft palate.
Case report A male infant was born by caesarean section after 38 weeks’ gestation, weight 3000 g, with a pedunculated mass protruding from his mouth (Fig. 1). Physical examination showed that the mass originated from the soft palate near the midline and looked like a mushroom. The mass, which was ∗ Corresponding author. Tel.: +86 21 63138341 5271; fax: +86 21 63121780. E-mail address: [email protected] (J.-w. Zheng).
Discussion Teratomas are defined as true neoplasms composed of multiple tissues foreign to the sites from which they originate. They have a reported incidence of 1:4000 live births with around 2–9% of these in the head and neck.1 Some reports have described a slight female predilection,3 but Carney et al.3 found malignant teratoma to be more common in men (5:4).
0266-4356/$ – see front matter © 2010 The British Association of Oral and Maxillofacial Surgeons. Published by Elsevier Ltd. All rights reserved.
doi:10.1016/j.bjoms.2010.04.002
e26
Y.-h. Jiang et al. / British Journal of Oral and Maxillofacial Surgery 48 (2010) e25–e26
Fig. 1. Mushroom-shaped teratoma of the soft palate protruding from the oral cavity in a neonate with a cleft palate.
nant potential contain immature tissues; benign teratomas may undergo malignant change with age. Histopathological examination showed the mass in our case to be composed of multiple mature tissues with no juvenile cells, which confirmed a benign tumour. To be a teratoma a tumour must contain multiple tissues foreign to the site at which it is located, as was the case with our patient. Purely oral teratomas rarely cause respiratory distress, unlike other head and neck tumours, particularly laryngopharyngeal teratomas. When oral teratomas grow, they tend to protrude outside the mouth rather than posteriorly towards the oropharynx. Because newborns are obligate nasal breathers, obstruction of the oral compartment is relatively less important and usually causes feeding problems.4 Patients with oral teratomas often have coexistent anomalies such as cleft palate,4 cystic hygroma,5 and other multifocal teratomas,4,6 with cleft palate being the most common.7 This is the result of mechanical obstruction of the neoplasm preventing closure of the palatal shelves. Other coexisting malformations are bifid tongues and noses.8 Most oral and maxillofacial teratomas are benign and have a good prognosis after early complete resection. However, failure to diagnose or treat teratomas of the head and neck until late adolescence or adulthood causes a risk of malignant degeneration in up to 90%.9 Recurrence is rare after early treatment.4 In our case feeding became normal after the mass had been removed, and the patient has been listed for elective operation on his cleft palate.
References
Fig. 2. (a and b) Histological examination of the teratoma showing mature bone, hair follicles, and cutaneous and glandular tissues (haematoxylin and eosin, original magnification ×20).
Teratomas can be either benign or malignant. Benign ones consist of mature tissue components, while those with malig-
1. Weaver RG, Meyerhoff WL, Gates GA. Teratomas of the head and neck. Surg Forum 1976;27:539–42. 2. Shafer WG, Hine MK, Levy BM. Textbook of Oral Pathology. 4th ed. London: WB Saunders; 1983. pp. 312–13. 3. Carney JA, ThompsonDP, Johnson CL, Lynn HB. Teratomas in children: clinical and pathologic aspects. J Pediatr Surg 1972;7: 271–82. 4. Uchida K, Urata H, Suzuki H. Teratoma of the tongue in neonates: report of a case and review of the literature. Pediatr Surg Int 1998;14: 79–81. 5. Wakhlu A, Wakhlu AK. Head and neck teratomas in children. Pediatr Surg Int 2000;16:333–7. 6. Dudgeon DL, Isaacs Jr H, Hays DM. Multiple teratomas of the head and neck. J Pediatr 1974;85:139–40. 7. Levine AB, Alvarez M, Wedgwood J, Berkowitz RL, Holzman I. Contemporary management of potentially lethal anomaly: a successful perinatal approach to epignathus. Obstet Gynecol 1990;76:962–6. 8. Vandenhaute B, Leteurtre E, Lecomte-Houcke M, et al. Epignathus teratoma: report of three cases with a review of the literature. Cleft Palate Craniofac J 2000;37:83–91. 9. Buckley NJ, Burch WM, Leight GS. Malignant teratoma in the thyroid gland of an adult: a case report and a review of the literature. Surgery 1986;100:932–7.