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Vol. 67. Issue 2.
Pages e9-e12 (March - April 2016)
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Vol. 67. Issue 2.
Pages e9-e12 (March - April 2016)
Case study
DOI: 10.1016/j.otoeng.2014.09.004
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Naso-Oropharyngeal Choristoma (Hairy Polyps) in Adults: A New Case, and Review of the Literature
Coristomas naso-orofaríngeos (pólipos pilosos) en adultos: un nuevo caso y revisión de la literatura
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Mainak Duttaa,
Corresponding author
duttamainak@yahoo.com

Corresponding author.
, Soumya Ghatakb, Soham Royc, Ramanuj Sinhaa
a Department of Otorhinolaryngology and Head-Neck Surgery, Medical College and Hospital, Kolkata, 88, College Street, Kolkata 700073, West Bengal, India
b Department of Otorhinolaryngology and Head-Neck Surgery, R. G. Kar Medical College and Hospital, Kolkata 700004, West Bengal, India
c Department of Otorhinolaryngology-Head and Neck Surgery, Children's Memorial Hermann Hospital, University of Texas Medical School at Houston, Houston, TX, USA
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Table 1. Mature Bigerminal Choristomas in Naso-Oropharynx Reported in Adults in English-Language Indexed Literature in the Last 30 Years.
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Clinical Case

A 42-year-old woman presented with progressive difficulty in swallowing, and snoring since 4 years, with right-sided nasal obstruction for 2 years. She had a persistent sense of obstruction behind her tongue; on examination, a large, smooth-walled globular mass was seen occupying the oropharynx extending up to the tongue-base and vallecula (Fig. 1). It was firm, non-tender, did not bleed on touch, and was free on all sides except superiorly, clinically resembling a choanal polyp. On diagnostic naso-endoscopy, the pedunculated mass was seen attached to the right Eustachian tube orifice and the adjacent epipharyngeal surface of soft palate, hanging from nasopharynx into the oropharynx obstructing the aerodigestive tract. Computed tomography (CT)-scan was non-contributory; the paranasal sinuses were clear, with no breach in the bones and skull-base. The lesion was excised under general anesthesia by combined naso-endoscopic/trans-oral approach. Grossly, the pear-shaped mass measured about 5cm×3.5cm×3cm, was firm, bosselated, and appeared heterogeneous in consistency. Histopathology revealed stratified squamous epithelium with epidermal appendages (hair follicles, sebaceous glands) along with well-organized, mature cartilage nests and smooth-muscle fibers with a fibro-adipose core, without any evidence of dysplasia (Fig. 2a and b). The features suggested bigerminal choristoma (hairy polyp) of the naso-oropharynx. The symptoms of the patient ameliorated following excision of the mass; she recuperated well and was disease-free on 2-year follow-up.

Figure 1.

A large fleshy mass could be seen in the oropharynx of this 42-year-old woman. It is free on all sides except superior, and seems to originate from the nasopharynx.

(0.08MB).
Figure 2.

Histopathology showed mature tissue elements of ectodermal and mesodermal origin, including (a) stratified squamous epithelium, skin adnexa (hair follicle, hollow arrow; sebaceous glands, solid arrow), and (b) fibro-adipose tissue (arrows), cartilage (hollow star) and muscle fibers (solid star). (Hematoxylin & Eosin, ×400).

(0.21MB).
Discussion

Hairy polyps are mature congenital ectodermal and mesodermal tissue-aggregates, often covered with thin hair, presenting as polypoid masses at anatomically aberrant sites (choristoma).1 They are almost exclusively seen in neonates and infants and can result in life-threatening asphyxia. Hairy polyps, however, are exceedingly rare in adults1; a PubMed/MEDLINE® search with the keywords “hairy polyp”, “choristoma”, “adult” and “naso-oropharynx” revealed only 6 such cases in the last 3 decades (Table 1).2–6 Unlike in neonates, the presentation in adults is insidious and less dramatic, clinically apparent even beyond the fifth decade in some cases,2–4 rising questions whether they should as a rule be considered as developmental malformations.

Table 1.

Mature Bigerminal Choristomas in Naso-Oropharynx Reported in Adults in English-Language Indexed Literature in the Last 30 Years.

S. No.  Year  Citation  Location/Origin  Age at presentation  Sex  Presentation 
1.  1984  Resta et al.2  Left side of Nasopharynx  71 years  Otalgia, otorrhea, hearing loss 
2.  1996  Franco et al.3  Between the palatine arches (bilateral)  58 years  Feeding difficulty, respiratory distress 
3.  1998  Cerezal et al.4  Left lateral nasopharyngeal wall  50 years  Recurrent epistaxis 
4.  2006  Green and Pearl5  Left lateral nasopharyngeal wall  24 years  Otalgia, hearing loss, feeding difficulty, respiratory distress 
5.  2013  Tariq et al.6  Reported a series of 4 patients, of which 2 were in their late teens (aged 17 and 18 years). The lesions were present in the nasopharynx in 2 cases and 1 each in lower lip and palate. Of the 4 patients, 3 were male and 1 female.
6.  2014  Dutta et al. (present case)  Right lateral nasopharyngeal wall (Eustachian tube opening)  42 years  Difficulty in swallowing, intermittent respiratory obstruction 

Hairy polyps in neonates are often associated with congenital disorders (like branchial arch anomalies)1,6; they are linked with development of the first and second pharyngeal arches owing to their endoscopy-documented anatomic associations with the Eustachian tube and tonsillar pillars.1 However, their occurrence late in the lives of previously asymptomatic adults remains unexplained.1 Also, no congenital/developmental anomaly has ever been associated with adults.1 These choristomatous lesions could occur due to delayed pluripotent cell morphogenesis where stem-cells have either escaped the local governing influences due to some inciting factors (trauma), or have been misdirected or trapped on way to their pre-destined target (“missed target hypothesis”).1 However, focal neoplasia could be an alternative explanation.1 We have carried out an analysis on the origin of hairy polyps based on an extensive literature review.1 Though the accumulated data there was based on clinical facets irrespective of age-groups and did not delve in-depth in the molecular aspects, we believe that neoplasia should be considered to explain the pathogenesis of hairy polyps in adults. Being characteristically bigerminal, they have traditionally been considered as “dermoid”. However, the consideration of hairy polyp as teratoma–a true neoplasia–is based on the changing concept of teratoma itself, whereby the classical belief of it being a trigerminal lesion is argued. Teratoma is presently considered a neoplastic mass composed of “any two germ layers”,6,7 or even as multiple tissue-aggregate non-indigenous to their anatomic location,8 making tissue composition irrelevant. Accordingly, terms like “bigerminal teratoma”9 and “benign teratoma”10 have been attributed to describe hairy polyps. Our description of hairy polyp as bigerminal choristoma, therefore, fits in with the evolving concept of teratoma in terms of composition and location. We believe that hairy polyps in adults constitute a distinct entity that has seldom, if at all, been explored before. It is to be acknowledged that their origin remains controversial with no single acceptable theory, and detailed molecular/cytogenetic analysis is required for a suitable explanation. But clinically, hairy polyps in adults seem to represent focal neoplastic proliferations, or benign teratomas, rather than mere developmental aberrations as widely believed.

Interestingly, in spite of the theoretical and speculated differences in origin, hairy polyps in adults do not differ significantly in management and prognosis from those in the neonates/infants. In both, naso-oropharyngeal hairy polyps are best examined by endoscopy. Occasional bony breach and the need to evaluate the extent might necessitate CT or magnetic resonance imaging, but none such has been reported in adults. Also, as mentioned earlier, developmental or congenital stigmata have not been associated in adults. There is a 3.5-times female preponderance and left-sided predilection in children.1 But the trend is not so clear in adults due to poor case-strength, though it seems that there is nearly an equal sex-distribution (Table 1). Likewise, although the presentation is more variable like epistaxis and ear problems (Table 1), no standard clinical picture could be derived. In adults however, hairy polyps might be confused with the commoner entities like choanal polyp (as in our patient). Combined naso-endoscopic/trans-oral excision is the treatment of choice.

Although hairy polyps are conventionally considered developmental aberrations related to the pharyngeal arches, an attempt to explain their origin in adults provides valuable insights to the present-day understanding of polygerminal lesions. This is the rationale why we intend to consider adults with hairy polyps as a distinct patient-cohort. Neoplasia should be a possibility as these lesions are being included within the “expanding domain of teratoma”,1 interpreted as neoplastic masses of “any two” germ layers non-indigenous to their site of origin. Thorough understanding of human embryology is essential to explain hairy polyps in adults, and addition of this case to the existing body of literature would help build a data-pool for the purpose.

Conflict of Interest

The authors have no conflicts of interest to declare.

References
[1]
M. Dutta, S. Roy, S. Ghatak.
Naso-oropharyngeal choristoma (hairy polyps): an overview and current update on presentation, management, origin and related controversies.
Eur Arch Otorhinolaryngol, (2014),
[2]
L. Resta, A. Santangelo, G. Lastilla.
The S. C. ‘hairy polyp’ or ‘dermoid’ of the nasopharynx (an unusual observation in older age).
J Laryngol Otol, 98 (1984), pp. 1043-1046
[3]
V. Franco, A.M. Florena, F. Lombardo, S. Restivo.
Bilateral hairy polyp of the oropharynx.
J Laryngol Otol, 110 (1996), pp. 288-290
[4]
L. Cerezal, C. Morales, F. Abascal, A. Canga, J. Gómez, M. Bustamante.
Magnetic resonance features of nasopharyngeal teratoma (hairy polyp) in adult.
Ann Otol Rhinol Laryngol, 107 (1998), pp. 987-990
[5]
V.S. Green, G.S. Pearl.
A 24-year-old woman with a nasopharyngeal mass.
Arch Pathol Lab Med, 130 (2006), pp. e33-e34
[6]
M.U. Tariq, N.U. Din, M.R. Bashir.
Hairy polyp, a clinicopathological study of four cases.
Head Neck Pathol, 7 (2013), pp. 232-235
[7]
T.J. Steinbach, A. Reischauer, I. Kunkemoller, M.G. Mense.
An oral choristoma in a foal resembling hairy polyp in humans.
Vet Pathol, 41 (2004), pp. 698-700
[8]
R.G. Weaver, W.I. Meyerhoff, G.A. Gates.
Teratomas of the head and neck.
Surg Forum, 27 (1976), pp. 539-544
[9]
A. Delides, F. Sharifi, E. Karagianni, A. Stasinopoulou, E. Helidonis.
Multifocal bigerminal mature teratomas of the head and neck.
J Laryngol Otol, 120 (2006), pp. 967-969
[10]
A. Ferlito, K.O. Devaney.
Developmental lesions of the head and neck: terminology and biologic behavior.
Ann Otol Rhinol Laryngol, 104 (1995), pp. 913-918

Please cite this article as: Dutta M, Ghatak S, Roy S, Sinha R. Naso-orofaríngea coristomas (Hairy pólipos) en adultos: un nuevo caso y revisión de la literatura. Acta Otorrinolaringol Esp. 2016;67:e9–e12.

This paper has been presented as a Poster in the Annual Meetings of The Triological Society (TRIO) at the 116th Combined Otolaryngology Spring Meetings (COSM) on 10th–14th April 2013, held at Orlando, Florida, USA. Links: http://www.researchposters.com/Posters/COSM/COSM2013/2-224.pdf and http://www.triomeetingposters.org/wp-content/uploads/2013/04/2-224.pdf.

Copyright © 2014. Elsevier España, S.L.U. and Sociedad Española de Otorrinolaringología y Cirugía de Cabeza y Cuello
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