Teratoma of Palantine Tonsil in a Neonate
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《美国医学杂志》
1 Department of Pathology, Sree Siddartha Medical College, Tumkur, Karnataka, India
2 Department of Pediatrics, Sree Siddartha Medical College, Tumkur, Karnataka, India
3 Department of ENT, Sree Siddartha Medical College, Tumkur, Karnataka, India
Abstract
Among many causes of respiratory distress and feeding difficulties in the neonatal period, a tumor involving oropharyngeal region is very rare. But still rarer is the unique presentation of this case of teratoma tonsil with intermittent attacks of choking with cyanosis.
Keywords: Intermittent attacks of chocking with cyanosis; Teratoma of palatine tonsil
Arising from pluripotent cells, teratomas are neoplasms composed of wide diversity of tissues foreign to the site of origin. The origin of the term is from the Greek word ' teraton' or monster, which retains the capacity for uncoordinated growth, both tumoral and malformative.
The most common locations for tertomas in childhood includes sacrococcygeal region, gonads and mediastinum. Teratomas of the head and neck region including the basicranial and cervical forms are distinctly uncommon, accounting for less than 2% of reported cases. Since 1903, only 11 cases of teratoma tonsil have been reported table1 but none from India. This is the first case of this rare tumor in a neonate reported from India, being 12th in the series to the best of author's knowledge.
Case report
10-day-old female neonate was brought with history of intermittent attacks of choking with cyanosis lasting momentarily. The baby was asymptomatic and taking feeds well in between the attacks.
This full term baby was a product of non-consanguinous marriage, delivered vaginally in a nursing home. It had good cry at birth without any attacks of choking or cyanosis. The baby was seen by a pediatrician and after routine newborn examination, baby was discharged on 2nd post- natal day.
Parents noticed the symptoms repeatedly at home and brought the neonate to the pediatric outpatient department of our hospital. During the attack, oropharyngeal examination was done. It revealed a small fleshy mobile mass in the oropharnx. It popped up during crying and then disappeard below in the hypopharynx. There was no cleft palate. Systemic examination was normal. No other congenital anomalies were detected.
The case was referred to the ENT department of our hospital. Detailed oropharyngeal with laryngeal examination was done using Mcinthosh laryngoscope. A film, pink polypoid mass with a peduncle arising from left palatine tonsil was seen. Remainder of left tonsillar tissue was normal including right tonsil. A provisional diagnosis of Fibroma tonsil was made. Baby was admitted and elective surgery was done under general anaesthesia. The mass along with left tonsil was removed Figure1 and only the mass was sent for histopathological examination. Postoperative course was uneventful. Follow-up of the case for 2 years revealed no recurrence.
Grossly the specimen consisted of a well circumscribed oval mass with grayish-yellow to grey-brown color mass measuring 1 x 0.8 x 0.6 cm with a peduncle measuring 0.2 cm. The surface was smooth. Cut section revealed a homogeneous grayish-yellow mass with tiny lobulations in the center. Microscopically a non-keratinizing stralified squamous epithelium with crypt-like down growth beneath which adnexal structures such as sweat glands and hair follicles were seen. Adipose tissue, smooth muscle tissue, fibrous tissue and mucous gland cells with benign cytological features were also seen. A histopathological diagnosis of benign teratoma of left palatine tonsil was made.
Discussion
This teratoma with a peduncle arising from the tonsillar tissue was mobile within in the oropharynx. Because of its small size there was no difficulty in feeding. But because of its mobility it used to block glottic opening intermittently causing symptoms.
Few of the clinical causes for intermittent choking with cyanosis in a neonate are[1]:
1. Congenital esophageal atresia with T-E fistula i.e, Regular variety and also H-type of T-E fistula.
2. Choanal atresia: Bilateral complete variety presents immediately at birth. But whereas bilateral incomplete and unilateral complete varieties can present afterwards with nasal obstruction.
3. Reflex laryngo spasm secondary to gastrooesophageal reflux.
4. Pedunculated teratomas arising from nasopharyngeal and tonsillar regions.
Willis [2] described teratoma as 'a true neoplasm composed of multiple tissues foreign to the organ from which it originates'. The origin of teratoma is unknown and various hypothesis have been suggested:
(1) Teratomas may represent including twins. This explains the origin of teratomas at sites at which conjoined twins are found.
(2) Teratomas may arise by parthenogenetic development of germ cells.
(3) Teratomas may arise from 'rests' of totipotent cells left behind during embryogenesis.
Teratomas contains tissues derived from all the 3 germ cell layers i.e. ectoderm, mesoderm and endoderm. Histologically they have well differentiated cell types and may be cystic, solid or mixed.
Finally we would like to stress the importance of oropharyngeal examination in all neonates at birth and even afterwards especially presenting with intermittent symptoms of choking with cyanosis.
References
1. Morris Green. Pediatric Diagnosis; 6th edn, Indianapolis; WB Saunders Company, 2001; 44: 362-370
2. Willis RA. Pathology of Tumor, 4th edn, London; Appleton - Century-Croft; 1967
3. Angier and Lavrand. Polype dermoide de I' amydale. Ann Med Chir Inf 1903; 7: 421-424.
4. Harper J. Dermoid of tonsil. J Laryngol 1924; 39: 157
5. Rosenberg MM. Dermoid cyst of the tonsil. Arch Otolaryngol 1935; 22: 361-362
6. Lewis GK, Gottstein WK, Costanjo VA. Congenital neurogenic malformation of faucial tonsil. Arch Otolaryngol 1951; 54: 172-176
7. Correa A, Paiva L. Teratoma of tonsillar region. Rev Hosp Clin 1951; 6: 86-89
8. Baugh CD, O' Donoghue RF. Teratoma of tonsil causing respiratory obstruction in the new born. Arch Dis Child 1955; 30: 396-397
9. Seniushina OD, Hmenlik VM. Teratoma of palatine tonsil. Vestn Otorhinolaryngol 1964; 26: 95-96
10. Ferraro DT, Herperger WS, Ampbell JB. Benign Teratoma of Tonsil. Ann Otol 1967; 76: 224-226
11. Balir OM, Sanchez JE. Teratoma of tonsil. Arch Otolaryngol 1969; 89: 745-747
12. Shah LB, Vasan U, Raye JR. Teratoma of tonsil in a premature infant. Am J Dis Child 1979; 133: 79-80(Parvathidevi GK, Belagavi)
2 Department of Pediatrics, Sree Siddartha Medical College, Tumkur, Karnataka, India
3 Department of ENT, Sree Siddartha Medical College, Tumkur, Karnataka, India
Abstract
Among many causes of respiratory distress and feeding difficulties in the neonatal period, a tumor involving oropharyngeal region is very rare. But still rarer is the unique presentation of this case of teratoma tonsil with intermittent attacks of choking with cyanosis.
Keywords: Intermittent attacks of chocking with cyanosis; Teratoma of palatine tonsil
Arising from pluripotent cells, teratomas are neoplasms composed of wide diversity of tissues foreign to the site of origin. The origin of the term is from the Greek word ' teraton' or monster, which retains the capacity for uncoordinated growth, both tumoral and malformative.
The most common locations for tertomas in childhood includes sacrococcygeal region, gonads and mediastinum. Teratomas of the head and neck region including the basicranial and cervical forms are distinctly uncommon, accounting for less than 2% of reported cases. Since 1903, only 11 cases of teratoma tonsil have been reported table1 but none from India. This is the first case of this rare tumor in a neonate reported from India, being 12th in the series to the best of author's knowledge.
Case report
10-day-old female neonate was brought with history of intermittent attacks of choking with cyanosis lasting momentarily. The baby was asymptomatic and taking feeds well in between the attacks.
This full term baby was a product of non-consanguinous marriage, delivered vaginally in a nursing home. It had good cry at birth without any attacks of choking or cyanosis. The baby was seen by a pediatrician and after routine newborn examination, baby was discharged on 2nd post- natal day.
Parents noticed the symptoms repeatedly at home and brought the neonate to the pediatric outpatient department of our hospital. During the attack, oropharyngeal examination was done. It revealed a small fleshy mobile mass in the oropharnx. It popped up during crying and then disappeard below in the hypopharynx. There was no cleft palate. Systemic examination was normal. No other congenital anomalies were detected.
The case was referred to the ENT department of our hospital. Detailed oropharyngeal with laryngeal examination was done using Mcinthosh laryngoscope. A film, pink polypoid mass with a peduncle arising from left palatine tonsil was seen. Remainder of left tonsillar tissue was normal including right tonsil. A provisional diagnosis of Fibroma tonsil was made. Baby was admitted and elective surgery was done under general anaesthesia. The mass along with left tonsil was removed Figure1 and only the mass was sent for histopathological examination. Postoperative course was uneventful. Follow-up of the case for 2 years revealed no recurrence.
Grossly the specimen consisted of a well circumscribed oval mass with grayish-yellow to grey-brown color mass measuring 1 x 0.8 x 0.6 cm with a peduncle measuring 0.2 cm. The surface was smooth. Cut section revealed a homogeneous grayish-yellow mass with tiny lobulations in the center. Microscopically a non-keratinizing stralified squamous epithelium with crypt-like down growth beneath which adnexal structures such as sweat glands and hair follicles were seen. Adipose tissue, smooth muscle tissue, fibrous tissue and mucous gland cells with benign cytological features were also seen. A histopathological diagnosis of benign teratoma of left palatine tonsil was made.
Discussion
This teratoma with a peduncle arising from the tonsillar tissue was mobile within in the oropharynx. Because of its small size there was no difficulty in feeding. But because of its mobility it used to block glottic opening intermittently causing symptoms.
Few of the clinical causes for intermittent choking with cyanosis in a neonate are[1]:
1. Congenital esophageal atresia with T-E fistula i.e, Regular variety and also H-type of T-E fistula.
2. Choanal atresia: Bilateral complete variety presents immediately at birth. But whereas bilateral incomplete and unilateral complete varieties can present afterwards with nasal obstruction.
3. Reflex laryngo spasm secondary to gastrooesophageal reflux.
4. Pedunculated teratomas arising from nasopharyngeal and tonsillar regions.
Willis [2] described teratoma as 'a true neoplasm composed of multiple tissues foreign to the organ from which it originates'. The origin of teratoma is unknown and various hypothesis have been suggested:
(1) Teratomas may represent including twins. This explains the origin of teratomas at sites at which conjoined twins are found.
(2) Teratomas may arise by parthenogenetic development of germ cells.
(3) Teratomas may arise from 'rests' of totipotent cells left behind during embryogenesis.
Teratomas contains tissues derived from all the 3 germ cell layers i.e. ectoderm, mesoderm and endoderm. Histologically they have well differentiated cell types and may be cystic, solid or mixed.
Finally we would like to stress the importance of oropharyngeal examination in all neonates at birth and even afterwards especially presenting with intermittent symptoms of choking with cyanosis.
References
1. Morris Green. Pediatric Diagnosis; 6th edn, Indianapolis; WB Saunders Company, 2001; 44: 362-370
2. Willis RA. Pathology of Tumor, 4th edn, London; Appleton - Century-Croft; 1967
3. Angier and Lavrand. Polype dermoide de I' amydale. Ann Med Chir Inf 1903; 7: 421-424.
4. Harper J. Dermoid of tonsil. J Laryngol 1924; 39: 157
5. Rosenberg MM. Dermoid cyst of the tonsil. Arch Otolaryngol 1935; 22: 361-362
6. Lewis GK, Gottstein WK, Costanjo VA. Congenital neurogenic malformation of faucial tonsil. Arch Otolaryngol 1951; 54: 172-176
7. Correa A, Paiva L. Teratoma of tonsillar region. Rev Hosp Clin 1951; 6: 86-89
8. Baugh CD, O' Donoghue RF. Teratoma of tonsil causing respiratory obstruction in the new born. Arch Dis Child 1955; 30: 396-397
9. Seniushina OD, Hmenlik VM. Teratoma of palatine tonsil. Vestn Otorhinolaryngol 1964; 26: 95-96
10. Ferraro DT, Herperger WS, Ampbell JB. Benign Teratoma of Tonsil. Ann Otol 1967; 76: 224-226
11. Balir OM, Sanchez JE. Teratoma of tonsil. Arch Otolaryngol 1969; 89: 745-747
12. Shah LB, Vasan U, Raye JR. Teratoma of tonsil in a premature infant. Am J Dis Child 1979; 133: 79-80(Parvathidevi GK, Belagavi)