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Frontal recess polyp in a 14-year-old child resembling an antro-choanal polyp
Journal of Taibah University Medical Sciences (2016) 11(1), 82e85
Taibah University
Journal of Taibah University Medical Sciences www.sciencedirect.com
Case Report
Frontal recess polyp in a 14-year-old child resembling an antro-choanal polyp Saumya R. Das, MS a, Satyabrata Dash, MS a, Ranjan K. Sahoo, MD b, Mahesh C. Sahu, PhD c and Rabindra N. Padhy, PhD c, * a
Department of Ear, Nose and Throat, Institute of Medical Sciences and Sum Hospital, Siksha O Anusandhan University, Bhubaneswar, Odisha, India b Department of Radio-diagnosis, Institute of Medical Sciences and Sum Hospital, Siksha O Anusandhan University, Bhubaneswar, Odisha, India c Central Research Laboratory, Institute of Medical Sciences and Sum Hospital, Siksha O Anusandhan University, Bhubaneswar, Odisha, India
Received 31 March 2015; revised 8 August 2015; accepted 10 August 2015; Available online 6 November 2015
ﺍﻟﻤﻠﺨﺺ ﻧﻘﺪﻡ ﻓﻲ ﻫﺬﻩ ﺍﻟﻮﺭﻗﺔ ﺣﺎﻟﺔ ﻟﺤﻤﻴﺔ ﺃﻧﻔﻴﺔ ﻧﺎﺩﺭﺓ ﻧﺎﺷﺌﺔ ﻣﻦ ﺟﻴﺐ ﺃﻧﻔﻲ ﺟﺒﻬﻲ ﻋﻨﺪ ﻃﻔﻠﺔ ﺑّﻴﻦ. ﻭﻛﺄﻧﻬﺎ ﻟﺤﻤﻴﺔ ﻓﻲ ﺍﻟﻔﺘﺤﺔ ﺍﻷﻧﻔﻴﺔ ﺍﻷﻣﺎﻣﻴﺔ،ﻓﻲ ﺍﻟﺮﺍﺑﻌﺔ ﻋﺸﺮﺓ ﻣﻦ ﻋﻤﺮﻫﺎ ﻭﺗﻤﺘﺪ،ﺍﻟﻤﻨﻈﺎﺭ ﺍﻷﻧﻔﻲ ﺍﻷﻣﺎﻣﻲ ﻛﺘﻠﺔ ﺳﻠﻴﻼﻧﻴﺔ ﺗﻤﻸ ﻛﺎﻣﻞ ﺍﻟﺘﺠﻮﻳﻒ ﺍﻷﻧﻔﻲ ﺍﻷﻳﺴﺮ ﺗﻢ ﺍﺳﺘﺌﺼﺎﻝ ﺍﻟﻜﺘﻠﺔ ﺑﻮﺍﺳﻄﺔ ﺍﻟﻤﻨﻈﺎﺭ ﻭﺗﺒﻴﻦ ﺃﻥ ﺃﺻﻠﻬﺎ.ﺇﻟﻰ ﻓﺘﺤﺔ ﺍﻷﻧﻒ ﺍﻷﻣﺎﻣﻴﺔ ﻭﻫﺬﺍ ﻗﻠﻴ ًﻼ ﻣﺎ ﻳﺤﺪﺙ ﻓﻲ ﺍﻟﻔﺌﺔ،ﻧﺎﺷﺊ ﻣﻦ ﺍﻟﺠﻴﺐ ﺍﻟﺠﺒﻬﻲ ﻟﻠﺘﺠﻮﻳﻒ ﺍﻷﻧﻔﻲ ﺍﻷﻳﺴﺮ .ﺍﻟﻌﻤﺮﻳﺔ ﻟﻸﻃﻔﺎﻝ ﻟﺤﻤﻴﺔ ) ﺳﻠﻴﻠﺔ ( ﺍﻟﺠﻴﺐ ﺍﻷﻧﻔﻲ ﺍﻟﺠﺒﻬﻲ؛ ﻟﺤﻤﻴﺔ ﺍﻟﻔﺘﺤﺔ ﺍﻷﻧﻔﻴﺔ:ﺍﻟﻜﻠﻤﺎﺕ ﺍﻟﻤﻔﺘﺎﺣﻴﺔ ﺍﻷﻣﺎﻣﻴﺔ؛ ﺳﻦ ﺍﻷﻃﻔﺎﻝ؛ ﻛﺘﻠﺔ ﺳﻠﻴﻠﺔ
The mass was excised endoscopically, and its origin was found to be the frontal recess of the left nasal cavity, a rare occurrence in the paediatric age group. Keywords: Antro-choanal polyp; Frontal recess polyp; Paediatric age; Polypoidal mass Ó 2015 The Authors. Production and hosting by Elsevier Ltd on behalf of Taibah University. This is an open access article under the CC BYNC-ND license (http://creativecommons.org/licenses/by-ncnd/4.0/).
Abstract A rare case of a unilateral single nasal polyp arising from a left frontal recess resembling an antro-choanal polyp in a 14-year-old girl is presented. A polypoidal mass completely occupying the left nasal cavity extending to the anterior nares was evident by anterior rhinoscopy.
* Corresponding address: Central Research Laboratory, Institute of Medical Sciences and Sum Hospital, Siksha ‘O’ Anusandhan University, K-8, Kalinga Nagar, Bhubaneswar 751003, Odisha, India. E-mail: [email protected] (R.N. Padhy) Peer review under responsibility of Taibah University.
Production and hosting by Elsevier
Introduction A nasal polyp is defined as an oedematous, pedunculated mucosa of paranasal sinus that is prolapsed into the nasal cavity. The development of a nasal polyp begins from a chronic inflammation, infection or immunologic stimulation. Nasal polyposis affects 1e4% of the population1,2 and is frequently reported in men,3,4 especially over 50 years of age,3 but rarely are children and young people affected. However, as it is associated with asthma, it is more frequent in females.4 Histologically, nasal polyposis presents with a large quantity of inflammatory cells, such as lymphocytes, mastocytes and basophiles, with the predominance of eosinophiles and non-inflammatory cells, fibroblasts and epithelial cells. Concomitantly, inflammatory mediators, such as cytokines and growth factors, are also present in the region.1,5
1658-3612 Ó 2015 The Authors. Production and hosting by Elsevier Ltd on behalf of Taibah University. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). http://dx.doi.org/10.1016/j.jtumed.2015.08.008
S.R. Das et al. Nasal polyps commonly arise around the openings of the paranasal sinuses. While allergic ethmoid polyps are a common type of nasal polyp in adults, antro-choanal polyps are frequently observed in children. Indeed, antro-choanal polyps are believed to arise from the lateral wall or floor of the maxillary antrum, and they typically grow rearward and present as a choanal mass. Although the aetiology of antrochoanal polyps is not clearly known, they are associated with chronic sinus infections or allergies. However, polyps originating from the mucosa of the nasal septum, though rare, have been reported, arising from any respiratory epithelium of the nasal cavity.6 A case of a polyp arising from the maxillary sinus and entering the nose via a wide accessory ostium has also been recorded.7 The classical first case report was of an antro-choanal polyp, termed Killian’s polyp.8 Maxillary polyps and those arising in other sinuses (e.g., spheno-choanal polyps) or the nasal septum are known to be solitary in origin, whereas ethmoid polyps are almost always in multiples. A rare occurrence of an isolated ethmoidal polyp arising from an anterior ethomodal cell has been reported.9,10 A long-standing nasal polyp can grow to be considerable in size, but a maxillary polyp of 15 5 cm in diameter has been reported.11 A case of a rare occurrence of a nasal polyp arising from the frontal recess, an unusual situation in a 14-year-old child, is presented here. Such cases have never been reported in patients of any age group, according to a systematic database search. Case report A 14-year-old girl presented to the ear, nose and throat outpatient department of a hospital complaining of a progressive left nasal obstruction and nasal discharge that had lasted 8 months. There was no history of allergies, asthma, diplopia or loss of vision. A clinical examination, full blood counts with a differential white cell count, a sweat chloride test and serum immunoglobulin counts were normal. On examination, there was no external nasal deformity or tenderness of the paranasal sinuses. Anterior rhinoscopy revealed a polypoid mass completely occupying the left nasal cavity, extending to the anterior nare (Figure 1). The nasal mass was non-tender, soft and mobile, without any bleeding on probing. A computed tomography (CT) scan of the paranasal sinuses revealed soft tissue attenuation in the left maxillary, ethmoid and frontal sinuses extending to the left nasal cavity with blockage of the left osteo-meatal unit. There was no radiological evidence of any bony erosion or intracranial extension (Figure 2). A provisional diagnosis of the antro-choanal polyp (ACP) was completed initially and the patient was prepared for endoscopic excision. The nasal mass was excised under general anaesthesia by nasal endoscopy (Figure 3). Intra-operatively, the peduncle of the polypoid nasal mass was found to be from the left frontal recess, not from the maxillary antrum (Figure 4). The agger nasi cell was identified by elevating the flap in the axilla of the middle turbinate, which was at its antero-superior insertion at the lateral nasal wall. The agger nasi cell was completely opened and removed using a Kerrison rongeur, by which the frontal recess was accessible. The polypoid mucosa from the frontal recess was removed. It was ascertained that there was
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Figure 1: Polypoid mass in the left nasal cavity extending to the anterior nare.
no involvement of the frontal, maxillary and ethmoid sinuses apart from the collection of secretions in those sinus cavities due to the blockage of the ostia. The maxillary ostium and the frontal recess area were adequately opened along with the anterior ethmoidectomy for ventilation and drainage of those sinuses. Intravenous ceftriaxone and amikacin were administered to the patient throughout her hospital stay. The postoperative period was uneventful, and the patient was discharged 5 days after surgery. Histopathological examination of the excised specimen revealed that the mass was a nasal polyp that contained respiratory epithelium, oedematous loose stroma, hyperplastic mucous glands and inflammatory infiltrates consisting of lymphocytes, plasma cells, eosinophils, neutrophils as well as mast cells. The patient was given a course of cefixime and anti-histamine tablets during the postoperative period and was prescribed alkaline nasal douching for 1 month. She had no clinical or radiological recurrence of the nasal polyp during the one-year postoperative follow-up period. Discussion The common sites of nasal polyp formation are the ethmoid and maxillary sinuses. Rarely, polyps may also arise from other paranasal sinuses. Sites in the nasal mucosa other than the paranasal sinus are rare, although polyps arising from the nasal septum have also been reported.6 While ethmoidal polyps are likely to be allergic in origin, the cause of polyp formation in other sinuses is not clearly known. A single predisposing cause cannot account for the occurrence of nasal polyps in all patients. Polyp formation and growth are activated and promoted by an integrated involvement of mucosal epithelium and inflammatory cells, which in turn may be due to both infectious and non-infectious inflammation.12 Although the relationship between nasal polyps and chronic rhinosinusitis is debated, nasal polyposis is believed to be the result of an inflammatory reaction involving the nasal mucosa and that of the paranasal sinuses. The ACPs originating from the maxillary antrum usually extend towards the choana.8 In a previous study, it was recorded that 6 out of 23 cases of antro-choanal polyps presented with total blockage of both choanae, with bilateral
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Nasal frontal recess polyp
Figure 2: CT scan showing soft tissue attenuation in the left nasal cavity and paranasal sinuses.
Figure 3: The excised nasal mass.
nasal obstruction and snoring due to the large size of the polyps occupying the entire nasal cavity extending to the choana.9 In this case the mass originated from the left frontal recess and extended towards the anterior nare of the left nasal cavity instead of the choana. Typically, polyps originating from ethmoid sinuses are multiple masses. Similar to ACPs, which are single nasal masses, in this case, a single nasal polyp was observed completely occupying the left nasal cavity. In the present case, a CT scan of the paranasal sinuses revealed soft tissue attenuation in the maxillary, frontal and ethmoid sinuses as well as in the left nasal cavity. However, intraoperatively, it was discernible that no mass was present in the maxillary, frontal or ethmoid sinuses. The present case of a single left nasal mass was indeed suspected to be an ACP, but its growth towards the anterior nare was uncommon. The underlying cause of the abnormality is basically unexplained, but chronic inflammation at the frontal recess could be the causative factor. Earlier, we reported a case of a malignant peripheral nerve sheath tumour of nose and
Figure 4: Intra-operative endoscopic pictures showing the peduncle of the nasal polyp arising from the frontal recess of the left nasal cavity.
S.R. Das et al. paranasal sinuses with an orbital extension,13 but in this case, no evidence of malignancy in the mass was detected. Acute sinusitis mimicking an antro-choanal polyp was observed in a case series; no polyp was found at surgery, but the presence of a redundant hypertrophic polypoid mucosa was confirmed.14 Furthermore, the sinus mucosa can become sufficiently oedematous and redundant, prolapsing into the nasal cavity through the sinus ostium, which has not been recognized. The sinus ostium is widened, a priory, secondary to pressure necrosis, in a manner similar to that observed with antro-choanal polyps. There would be difficulty in prognosis between sinusitis with a redundant prolapsed mucosa and an antro-choanal polyp because of similar radiographic images.14 Indeed, the typical CT appearances of nasal polyps are of smooth, convex, enhancing soft-tissue masses, and it is often difficult to differentiate the polyp from retained secretions on CT imaging. A MRI would be useful for such cases because of the difference in signal characteristics between soft tissue and fluid.15 In the present case, the possible explanation for the differences between the CT scan and the intra-operative findings could be from pressure necrosis of the maxillary ostium by the expansile polyp in the osteomeatal complex arising from the frontal recess and hypertrophied mucosa with retained secretions inside the maxillary sinus cavity. Nonetheless, papillomas are cauliflower-like tumours of the mucous membrane, which may be pedunculated or raspberry-like growths.16 And an inverted papilloma accounts for approximately 0.5e5% of nasal neoplasms and typically presents in middle-aged males. It typically arises in the lateral aspect of the nasal cavity, close to the middle turbinate.17 Clinically, papillomatosis presents with symptoms similar to other nasal diseases. It may have a gross appearance identical to the ordinary nasal polyp or may be a diffuse lesion involving broad surfaces of the nasal and sinus mucous membranes.18 In the present case, because of the paediatric age and female sex of the patient and the gross appearance of the nasal mass e a single mass with a smooth surface e a diagnosis of nasal papillomatosis was not initially considered. There are multiple endoscopic approaches to access the frontal recess, which vary according to the preference of the surgeon. Exposure and removal of the agger nasi cell using the axillary flap technique to gain access to the frontal recess is a widely accepted surgical method19 and was adopted in the present case. Complete excision of the polyp along with the establishment of drainage pathways of the paranasal sinuses was the suitable approach for addressing the problem. In conclusion, nasal polyps in children and adolescents may rarely arise from sites other than the maxillary antrum, such as the frontal recess, as was encountered in this case. As the clinical and radiologic evidence was misleading, the anticipation of unusual sites of origin was helpful for the preoperative and intraoperative approach to the paediatric patient presenting with a nasal polyp. Authors’ contributions S.R. Das examined the patient and designed the study at the pre-operative stage, prepared the patient for endoscopic excision of the nasal mass. Postoperative examination and treatment was carried out by him. S. Dash assisted in
85
surgery, post-operative care and follow ups at regular intervals for clinical examination. R.K. Sahoo made the radiological interpretation of the nasal mass. M.C. Sahu interacted with surgeons for writing the paper. He wrote the paper in consultation with R.N. Padhy, who interpreted data with S.R. Das and drafted the final manuscript. Conflict of interest The authors have no conflict of interest to declare. Acknowledgements We are grateful for the support of Dr. DK Roy, Medical Director, IMS and Sum Hospital and the facilities of Mr. Gopabandhu Kar, Managing Member, IMS and Sum Hospital. References 1. Souza BB, Serra MF, Dorgam JV, Sarreta SM, Melo VR, Anselmo-Lima W. Polipose Nasossinusal: Doenc¸a Inflamato´ria Croˆnica Evolutiva? Rev Bras Otorrinol 2003; 69(3): 318e325. 2. Puwankar R. Nasal polyposis: an update. Curr Opin Allergy Immunol 2003; 3: 1e6. 3. Matsuyama C. Polipose nasossinusal: Uma Revisa˜o Bibliogra´fica. Rev Bras Otorrinol 2000; 7(3): 82e84. 4. Collins M, Pang Y-T, Loughran S, Wilson JA. Environmental risk factors and gender in nasal polyposis. Clin Otolaryngol 2002; 27: 314e317. 5. Bateman ND, Fahy C, Woolford J. Nasal polyps still more questions than answers. J Laringol Otol 2003; 117(1): 1e6. 6. Bailey Q. Choanal polyp arising from the posterior end of the nasal septum. J Larngol Otol 1979; 93(7): 735e736. 7. Zuckerkandl E. Normale und pathologische Anatomie der Nasentholime; 1892. Vienna. 8. Killian G. The origin of choannal polyp. Lancet 1906; 2: 81e82. 9. Bozzo C, Garrel R, Meloni F, Stomeo F, Crampette L. Endoscopic treatment of antro-choanal polyps. Eur Arch Oto-RhinoLaryngol 2007; 264(2): 145e150. 10. Yanagisawa E, Lesnik David J. Isolated polyp of the ethmoid sinus. Rhinoscopic Clin, Ear, Nose Throat J 2002; 81(10): 689e690. 11. Bhat M, Vaidyanatham V. Sausage in the throat. A case of giant antro-choanal polyp. J Clin Diagno Res 2010; 4(2): 2282e2285. 12. Stiernal PL. Nasal polyps: relationship to infections and inflammation. Allergy Asthma Proc 1996; 17(5): 251e257. 13. Das SR, Dash S, Pradhan B, Sahu MC, Padhy RN. Malignant peripheral nerve sheath tumour of nose and paranasal sinuses with orbital extension. J Taibah Univ Med Sci 2015; 10(2): 238e242. 14. Nino-Murcia M, Rao VM, Mikaelian DO, Som P. Acute sinusitis mimicking antro-choanal polyp. Amer J Neur Radio 1986; 7: 513e516. 15. Szewczyk-Bieda MJ, White RD, Budak MJ, Ananthakrishnan G, Brunton JN, Sudarshan TA. A whiff of trouble: tumours of the nasal cavity and their mimics. Clin Radiol 2014; 69: 519e528. 16. Kramer R, Som M. True papilloma of the nasal cavity. Arch Otolaryngol 1935; 22(1): 22e43. 17. Savy L, Lloyd G, Lund VJ. Optimum imaging for inverted papilloma. J Laryngol Otol 2000; 114: 891e893. 18. Snyder RN, Perzin KH. Papillomatosis of nasal cavity and paranasal sinuses (inverted papilloma, squamous papilloma): a clinicopatologic study. Cancer 1972; 30(3): 668e690. 19. Wormald PJ. Surgery of the frontal recess and frontal sinus. Rhinology 2005; 43: 82e85.
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Journal of Taibah University Medical Sciences www.sciencedirect.com
Case Report
Frontal recess polyp in a 14-year-old child resembling an antro-choanal polyp Saumya R. Das, MS a, Satyabrata Dash, MS a, Ranjan K. Sahoo, MD b, Mahesh C. Sahu, PhD c and Rabindra N. Padhy, PhD c, * a
Department of Ear, Nose and Throat, Institute of Medical Sciences and Sum Hospital, Siksha O Anusandhan University, Bhubaneswar, Odisha, India b Department of Radio-diagnosis, Institute of Medical Sciences and Sum Hospital, Siksha O Anusandhan University, Bhubaneswar, Odisha, India c Central Research Laboratory, Institute of Medical Sciences and Sum Hospital, Siksha O Anusandhan University, Bhubaneswar, Odisha, India
Received 31 March 2015; revised 8 August 2015; accepted 10 August 2015; Available online 6 November 2015
ﺍﻟﻤﻠﺨﺺ ﻧﻘﺪﻡ ﻓﻲ ﻫﺬﻩ ﺍﻟﻮﺭﻗﺔ ﺣﺎﻟﺔ ﻟﺤﻤﻴﺔ ﺃﻧﻔﻴﺔ ﻧﺎﺩﺭﺓ ﻧﺎﺷﺌﺔ ﻣﻦ ﺟﻴﺐ ﺃﻧﻔﻲ ﺟﺒﻬﻲ ﻋﻨﺪ ﻃﻔﻠﺔ ﺑّﻴﻦ. ﻭﻛﺄﻧﻬﺎ ﻟﺤﻤﻴﺔ ﻓﻲ ﺍﻟﻔﺘﺤﺔ ﺍﻷﻧﻔﻴﺔ ﺍﻷﻣﺎﻣﻴﺔ،ﻓﻲ ﺍﻟﺮﺍﺑﻌﺔ ﻋﺸﺮﺓ ﻣﻦ ﻋﻤﺮﻫﺎ ﻭﺗﻤﺘﺪ،ﺍﻟﻤﻨﻈﺎﺭ ﺍﻷﻧﻔﻲ ﺍﻷﻣﺎﻣﻲ ﻛﺘﻠﺔ ﺳﻠﻴﻼﻧﻴﺔ ﺗﻤﻸ ﻛﺎﻣﻞ ﺍﻟﺘﺠﻮﻳﻒ ﺍﻷﻧﻔﻲ ﺍﻷﻳﺴﺮ ﺗﻢ ﺍﺳﺘﺌﺼﺎﻝ ﺍﻟﻜﺘﻠﺔ ﺑﻮﺍﺳﻄﺔ ﺍﻟﻤﻨﻈﺎﺭ ﻭﺗﺒﻴﻦ ﺃﻥ ﺃﺻﻠﻬﺎ.ﺇﻟﻰ ﻓﺘﺤﺔ ﺍﻷﻧﻒ ﺍﻷﻣﺎﻣﻴﺔ ﻭﻫﺬﺍ ﻗﻠﻴ ًﻼ ﻣﺎ ﻳﺤﺪﺙ ﻓﻲ ﺍﻟﻔﺌﺔ،ﻧﺎﺷﺊ ﻣﻦ ﺍﻟﺠﻴﺐ ﺍﻟﺠﺒﻬﻲ ﻟﻠﺘﺠﻮﻳﻒ ﺍﻷﻧﻔﻲ ﺍﻷﻳﺴﺮ .ﺍﻟﻌﻤﺮﻳﺔ ﻟﻸﻃﻔﺎﻝ ﻟﺤﻤﻴﺔ ) ﺳﻠﻴﻠﺔ ( ﺍﻟﺠﻴﺐ ﺍﻷﻧﻔﻲ ﺍﻟﺠﺒﻬﻲ؛ ﻟﺤﻤﻴﺔ ﺍﻟﻔﺘﺤﺔ ﺍﻷﻧﻔﻴﺔ:ﺍﻟﻜﻠﻤﺎﺕ ﺍﻟﻤﻔﺘﺎﺣﻴﺔ ﺍﻷﻣﺎﻣﻴﺔ؛ ﺳﻦ ﺍﻷﻃﻔﺎﻝ؛ ﻛﺘﻠﺔ ﺳﻠﻴﻠﺔ
The mass was excised endoscopically, and its origin was found to be the frontal recess of the left nasal cavity, a rare occurrence in the paediatric age group. Keywords: Antro-choanal polyp; Frontal recess polyp; Paediatric age; Polypoidal mass Ó 2015 The Authors. Production and hosting by Elsevier Ltd on behalf of Taibah University. This is an open access article under the CC BYNC-ND license (http://creativecommons.org/licenses/by-ncnd/4.0/).
Abstract A rare case of a unilateral single nasal polyp arising from a left frontal recess resembling an antro-choanal polyp in a 14-year-old girl is presented. A polypoidal mass completely occupying the left nasal cavity extending to the anterior nares was evident by anterior rhinoscopy.
* Corresponding address: Central Research Laboratory, Institute of Medical Sciences and Sum Hospital, Siksha ‘O’ Anusandhan University, K-8, Kalinga Nagar, Bhubaneswar 751003, Odisha, India. E-mail: [email protected] (R.N. Padhy) Peer review under responsibility of Taibah University.
Production and hosting by Elsevier
Introduction A nasal polyp is defined as an oedematous, pedunculated mucosa of paranasal sinus that is prolapsed into the nasal cavity. The development of a nasal polyp begins from a chronic inflammation, infection or immunologic stimulation. Nasal polyposis affects 1e4% of the population1,2 and is frequently reported in men,3,4 especially over 50 years of age,3 but rarely are children and young people affected. However, as it is associated with asthma, it is more frequent in females.4 Histologically, nasal polyposis presents with a large quantity of inflammatory cells, such as lymphocytes, mastocytes and basophiles, with the predominance of eosinophiles and non-inflammatory cells, fibroblasts and epithelial cells. Concomitantly, inflammatory mediators, such as cytokines and growth factors, are also present in the region.1,5
1658-3612 Ó 2015 The Authors. Production and hosting by Elsevier Ltd on behalf of Taibah University. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). http://dx.doi.org/10.1016/j.jtumed.2015.08.008
S.R. Das et al. Nasal polyps commonly arise around the openings of the paranasal sinuses. While allergic ethmoid polyps are a common type of nasal polyp in adults, antro-choanal polyps are frequently observed in children. Indeed, antro-choanal polyps are believed to arise from the lateral wall or floor of the maxillary antrum, and they typically grow rearward and present as a choanal mass. Although the aetiology of antrochoanal polyps is not clearly known, they are associated with chronic sinus infections or allergies. However, polyps originating from the mucosa of the nasal septum, though rare, have been reported, arising from any respiratory epithelium of the nasal cavity.6 A case of a polyp arising from the maxillary sinus and entering the nose via a wide accessory ostium has also been recorded.7 The classical first case report was of an antro-choanal polyp, termed Killian’s polyp.8 Maxillary polyps and those arising in other sinuses (e.g., spheno-choanal polyps) or the nasal septum are known to be solitary in origin, whereas ethmoid polyps are almost always in multiples. A rare occurrence of an isolated ethmoidal polyp arising from an anterior ethomodal cell has been reported.9,10 A long-standing nasal polyp can grow to be considerable in size, but a maxillary polyp of 15 5 cm in diameter has been reported.11 A case of a rare occurrence of a nasal polyp arising from the frontal recess, an unusual situation in a 14-year-old child, is presented here. Such cases have never been reported in patients of any age group, according to a systematic database search. Case report A 14-year-old girl presented to the ear, nose and throat outpatient department of a hospital complaining of a progressive left nasal obstruction and nasal discharge that had lasted 8 months. There was no history of allergies, asthma, diplopia or loss of vision. A clinical examination, full blood counts with a differential white cell count, a sweat chloride test and serum immunoglobulin counts were normal. On examination, there was no external nasal deformity or tenderness of the paranasal sinuses. Anterior rhinoscopy revealed a polypoid mass completely occupying the left nasal cavity, extending to the anterior nare (Figure 1). The nasal mass was non-tender, soft and mobile, without any bleeding on probing. A computed tomography (CT) scan of the paranasal sinuses revealed soft tissue attenuation in the left maxillary, ethmoid and frontal sinuses extending to the left nasal cavity with blockage of the left osteo-meatal unit. There was no radiological evidence of any bony erosion or intracranial extension (Figure 2). A provisional diagnosis of the antro-choanal polyp (ACP) was completed initially and the patient was prepared for endoscopic excision. The nasal mass was excised under general anaesthesia by nasal endoscopy (Figure 3). Intra-operatively, the peduncle of the polypoid nasal mass was found to be from the left frontal recess, not from the maxillary antrum (Figure 4). The agger nasi cell was identified by elevating the flap in the axilla of the middle turbinate, which was at its antero-superior insertion at the lateral nasal wall. The agger nasi cell was completely opened and removed using a Kerrison rongeur, by which the frontal recess was accessible. The polypoid mucosa from the frontal recess was removed. It was ascertained that there was
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Figure 1: Polypoid mass in the left nasal cavity extending to the anterior nare.
no involvement of the frontal, maxillary and ethmoid sinuses apart from the collection of secretions in those sinus cavities due to the blockage of the ostia. The maxillary ostium and the frontal recess area were adequately opened along with the anterior ethmoidectomy for ventilation and drainage of those sinuses. Intravenous ceftriaxone and amikacin were administered to the patient throughout her hospital stay. The postoperative period was uneventful, and the patient was discharged 5 days after surgery. Histopathological examination of the excised specimen revealed that the mass was a nasal polyp that contained respiratory epithelium, oedematous loose stroma, hyperplastic mucous glands and inflammatory infiltrates consisting of lymphocytes, plasma cells, eosinophils, neutrophils as well as mast cells. The patient was given a course of cefixime and anti-histamine tablets during the postoperative period and was prescribed alkaline nasal douching for 1 month. She had no clinical or radiological recurrence of the nasal polyp during the one-year postoperative follow-up period. Discussion The common sites of nasal polyp formation are the ethmoid and maxillary sinuses. Rarely, polyps may also arise from other paranasal sinuses. Sites in the nasal mucosa other than the paranasal sinus are rare, although polyps arising from the nasal septum have also been reported.6 While ethmoidal polyps are likely to be allergic in origin, the cause of polyp formation in other sinuses is not clearly known. A single predisposing cause cannot account for the occurrence of nasal polyps in all patients. Polyp formation and growth are activated and promoted by an integrated involvement of mucosal epithelium and inflammatory cells, which in turn may be due to both infectious and non-infectious inflammation.12 Although the relationship between nasal polyps and chronic rhinosinusitis is debated, nasal polyposis is believed to be the result of an inflammatory reaction involving the nasal mucosa and that of the paranasal sinuses. The ACPs originating from the maxillary antrum usually extend towards the choana.8 In a previous study, it was recorded that 6 out of 23 cases of antro-choanal polyps presented with total blockage of both choanae, with bilateral
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Nasal frontal recess polyp
Figure 2: CT scan showing soft tissue attenuation in the left nasal cavity and paranasal sinuses.
Figure 3: The excised nasal mass.
nasal obstruction and snoring due to the large size of the polyps occupying the entire nasal cavity extending to the choana.9 In this case the mass originated from the left frontal recess and extended towards the anterior nare of the left nasal cavity instead of the choana. Typically, polyps originating from ethmoid sinuses are multiple masses. Similar to ACPs, which are single nasal masses, in this case, a single nasal polyp was observed completely occupying the left nasal cavity. In the present case, a CT scan of the paranasal sinuses revealed soft tissue attenuation in the maxillary, frontal and ethmoid sinuses as well as in the left nasal cavity. However, intraoperatively, it was discernible that no mass was present in the maxillary, frontal or ethmoid sinuses. The present case of a single left nasal mass was indeed suspected to be an ACP, but its growth towards the anterior nare was uncommon. The underlying cause of the abnormality is basically unexplained, but chronic inflammation at the frontal recess could be the causative factor. Earlier, we reported a case of a malignant peripheral nerve sheath tumour of nose and
Figure 4: Intra-operative endoscopic pictures showing the peduncle of the nasal polyp arising from the frontal recess of the left nasal cavity.
S.R. Das et al. paranasal sinuses with an orbital extension,13 but in this case, no evidence of malignancy in the mass was detected. Acute sinusitis mimicking an antro-choanal polyp was observed in a case series; no polyp was found at surgery, but the presence of a redundant hypertrophic polypoid mucosa was confirmed.14 Furthermore, the sinus mucosa can become sufficiently oedematous and redundant, prolapsing into the nasal cavity through the sinus ostium, which has not been recognized. The sinus ostium is widened, a priory, secondary to pressure necrosis, in a manner similar to that observed with antro-choanal polyps. There would be difficulty in prognosis between sinusitis with a redundant prolapsed mucosa and an antro-choanal polyp because of similar radiographic images.14 Indeed, the typical CT appearances of nasal polyps are of smooth, convex, enhancing soft-tissue masses, and it is often difficult to differentiate the polyp from retained secretions on CT imaging. A MRI would be useful for such cases because of the difference in signal characteristics between soft tissue and fluid.15 In the present case, the possible explanation for the differences between the CT scan and the intra-operative findings could be from pressure necrosis of the maxillary ostium by the expansile polyp in the osteomeatal complex arising from the frontal recess and hypertrophied mucosa with retained secretions inside the maxillary sinus cavity. Nonetheless, papillomas are cauliflower-like tumours of the mucous membrane, which may be pedunculated or raspberry-like growths.16 And an inverted papilloma accounts for approximately 0.5e5% of nasal neoplasms and typically presents in middle-aged males. It typically arises in the lateral aspect of the nasal cavity, close to the middle turbinate.17 Clinically, papillomatosis presents with symptoms similar to other nasal diseases. It may have a gross appearance identical to the ordinary nasal polyp or may be a diffuse lesion involving broad surfaces of the nasal and sinus mucous membranes.18 In the present case, because of the paediatric age and female sex of the patient and the gross appearance of the nasal mass e a single mass with a smooth surface e a diagnosis of nasal papillomatosis was not initially considered. There are multiple endoscopic approaches to access the frontal recess, which vary according to the preference of the surgeon. Exposure and removal of the agger nasi cell using the axillary flap technique to gain access to the frontal recess is a widely accepted surgical method19 and was adopted in the present case. Complete excision of the polyp along with the establishment of drainage pathways of the paranasal sinuses was the suitable approach for addressing the problem. In conclusion, nasal polyps in children and adolescents may rarely arise from sites other than the maxillary antrum, such as the frontal recess, as was encountered in this case. As the clinical and radiologic evidence was misleading, the anticipation of unusual sites of origin was helpful for the preoperative and intraoperative approach to the paediatric patient presenting with a nasal polyp. Authors’ contributions S.R. Das examined the patient and designed the study at the pre-operative stage, prepared the patient for endoscopic excision of the nasal mass. Postoperative examination and treatment was carried out by him. S. Dash assisted in
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surgery, post-operative care and follow ups at regular intervals for clinical examination. R.K. Sahoo made the radiological interpretation of the nasal mass. M.C. Sahu interacted with surgeons for writing the paper. He wrote the paper in consultation with R.N. Padhy, who interpreted data with S.R. Das and drafted the final manuscript. Conflict of interest The authors have no conflict of interest to declare. Acknowledgements We are grateful for the support of Dr. DK Roy, Medical Director, IMS and Sum Hospital and the facilities of Mr. Gopabandhu Kar, Managing Member, IMS and Sum Hospital. References 1. Souza BB, Serra MF, Dorgam JV, Sarreta SM, Melo VR, Anselmo-Lima W. Polipose Nasossinusal: Doenc¸a Inflamato´ria Croˆnica Evolutiva? Rev Bras Otorrinol 2003; 69(3): 318e325. 2. Puwankar R. Nasal polyposis: an update. Curr Opin Allergy Immunol 2003; 3: 1e6. 3. Matsuyama C. Polipose nasossinusal: Uma Revisa˜o Bibliogra´fica. Rev Bras Otorrinol 2000; 7(3): 82e84. 4. Collins M, Pang Y-T, Loughran S, Wilson JA. Environmental risk factors and gender in nasal polyposis. Clin Otolaryngol 2002; 27: 314e317. 5. Bateman ND, Fahy C, Woolford J. Nasal polyps still more questions than answers. J Laringol Otol 2003; 117(1): 1e6. 6. Bailey Q. Choanal polyp arising from the posterior end of the nasal septum. J Larngol Otol 1979; 93(7): 735e736. 7. Zuckerkandl E. Normale und pathologische Anatomie der Nasentholime; 1892. Vienna. 8. Killian G. The origin of choannal polyp. Lancet 1906; 2: 81e82. 9. Bozzo C, Garrel R, Meloni F, Stomeo F, Crampette L. Endoscopic treatment of antro-choanal polyps. Eur Arch Oto-RhinoLaryngol 2007; 264(2): 145e150. 10. Yanagisawa E, Lesnik David J. Isolated polyp of the ethmoid sinus. Rhinoscopic Clin, Ear, Nose Throat J 2002; 81(10): 689e690. 11. Bhat M, Vaidyanatham V. Sausage in the throat. A case of giant antro-choanal polyp. J Clin Diagno Res 2010; 4(2): 2282e2285. 12. Stiernal PL. Nasal polyps: relationship to infections and inflammation. Allergy Asthma Proc 1996; 17(5): 251e257. 13. Das SR, Dash S, Pradhan B, Sahu MC, Padhy RN. Malignant peripheral nerve sheath tumour of nose and paranasal sinuses with orbital extension. J Taibah Univ Med Sci 2015; 10(2): 238e242. 14. Nino-Murcia M, Rao VM, Mikaelian DO, Som P. Acute sinusitis mimicking antro-choanal polyp. Amer J Neur Radio 1986; 7: 513e516. 15. Szewczyk-Bieda MJ, White RD, Budak MJ, Ananthakrishnan G, Brunton JN, Sudarshan TA. A whiff of trouble: tumours of the nasal cavity and their mimics. Clin Radiol 2014; 69: 519e528. 16. Kramer R, Som M. True papilloma of the nasal cavity. Arch Otolaryngol 1935; 22(1): 22e43. 17. Savy L, Lloyd G, Lund VJ. Optimum imaging for inverted papilloma. J Laryngol Otol 2000; 114: 891e893. 18. Snyder RN, Perzin KH. Papillomatosis of nasal cavity and paranasal sinuses (inverted papilloma, squamous papilloma): a clinicopatologic study. Cancer 1972; 30(3): 668e690. 19. Wormald PJ. Surgery of the frontal recess and frontal sinus. Rhinology 2005; 43: 82e85.