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Table of Contents
Year : 2021  |  Volume : 23  |  Issue : 1  |  Page : 1-5

Inverted papilloma in children

1 Department of Otolaryngology, Head and Neck Surgery, Qassim University, Qassim, Saudi Arabia
2 Department of Otolaryngology, Head and Neck Surgery, King Saud Hospital, Unaizah, Saudi Arabia
3 Medical Internship Unit, College of Medicine, Qassim University, Qassim, Saudi Arabia
4 Department of Family and Community Medicine, Unaizah College of Medicine, Qassim University, Qassim, Saudi Arabia

Date of Submission06-Jan-2020
Date of Decision23-Jan-2020
Date of Acceptance06-Feb-2020
Date of Web Publication08-Mar-2021

Correspondence Address:
Dr. Mazyad Alenezi
Department of Otolaryngology, Head and Neck Surgery, Qassim University, Qassim
Saudi Arabia
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/SJOH.SJOH_2_20

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Inverted papilloma (IP) is considered a rare benign sinonasal lesion, which is usually presented in adults. The presentation of IP is usually a unilateral nasal obstruction. IPs in the pediatric age group are quite rare as only a few cases were reported. This review aims to increase the awareness of IP to consider this entity in the diagnosis of unilateral nasal obstruction in children. This study was a review of 10 articles which reported the occurrence of IPs in children. Data include age, presentation, diagnosis, and treatment which were extracted and analyzed. The total number of cases was 12. The mean age of all cases was 10.5 years. The majority were male (66%). The main presenting symptom of all cases was nasal obstruction along with other nasal symptoms. The majority of the patients were treated by surgical excision of the lesion. Most of the cases 6 (50%) used endoscopic sinus surgery, and 5 (41%) of them were managed by lateral rhinotomy with or without medial maxillectomy. Follow-up showed that 7 (58%) of the cases did not document any recurrence of the disease. The reoccurrence occurred in 5 (41%) of the cases within the 1st year after the management. IPs can arise in the pediatric age group and should be considered in the differential diagnosis of unilateral nasal obstruction. It is diagnosed and treated likewise in adults.

Keywords: Children, inverted papilloma, nose, pediatric, Schneiderian papilloma

How to cite this article:
Alenezi M, Altheyab F, Alabood S, Almutairi A, Alanazy S, Al-Wutayd O. Inverted papilloma in children. Saudi J Otorhinolaryngol Head Neck Surg 2021;23:1-5

How to cite this URL:
Alenezi M, Altheyab F, Alabood S, Almutairi A, Alanazy S, Al-Wutayd O. Inverted papilloma in children. Saudi J Otorhinolaryngol Head Neck Surg [serial online] 2021 [cited 2022 Dec 4];23:1-5. Available from: https://www.sjohns.org/text.asp?2021/23/1/1/310985

  Introduction Top

Inverted papilloma (IP) is considered a rare benign sinonasal lesion, which consists of 0.5%–4% of primary nasal tumors in adults.[1] IP was originally described in 1854 and was known as “Schneiderian papilloma.”[2] It is a tumor of the nasal cavities and paranasal sinuses, with three main characteristic features that distinguish it from other sinonasal tumors: relative local aggression, high rates of recurrence, and possible association with carcinoma.[3] Moreover, Busquets et al reported in their meta-analysis a recurrence rate of 15%-20%.[4],[5] IP etiology remains unclear. Certain hypotheses have been suggested, but a direct cause has never been established. Some of the suspected factors are smoking, allergy, or certain occupational exposures.[6] Recurrence and carcinomatous potential have been previously thought to suggest a viral origin. An implication of Epstein–Barr virus has been studied but yields inconclusive results.[7]

Furthermore, human papillomavirus (HPV) has been suspected of playing a major role in the pathophysiology of IP, but the literature data remain contradictory.[8] The usual age of IP presentation is around 50–60 years of age, and it is three times more common in males.[9]

IP incidence annually is roughly 0.75–1.5/100,000 in Europe.[8] The usual presentation of IP in adults is a unilateral nasal obstruction.[8] Furthermore, other presentations include epistaxis, headache, anosmia, dysosmia, and epiphora.[8] However, in the pediatric age group, IPs are quite rare and only a few cases have been reported.

We aim in this study to identify the common age of IP presentations of symptoms and signs among the pediatric age group, how they were managed, and the recurrence rate among our cases in this review and furthermore to increase the awareness of IP in the pediatric age group.

  Methods Top

A thorough review was conducted on PubMed, Oxford Academic, and ScienceDirect, using the following search terms: “papilloma, inverted” and “children,” “pediatrics,” and “Schneiderian papilloma.” All cases involving sites other than the nose were excluded. Only articles published in English were included. Data include age, presentation, diagnosis, and treatment, which were extracted. Articles with missing data were excluded. Articles were read and analyzed, with relevant data being extracted. A flowchart of the selected articles in [Figure 1].
Figure 1: Flowchart of the strategy used for article selection

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  Results Top

A total number of 12 cases were analyzed. Male patients accounted for 8 cases (66%), and female patients were 4 (33%). The mean age of all cases was 10.5 years ranging from 6 to 15 years. All the cases had nasal obstruction as their main complaint, and some other common complaints were epistaxis (7, 58%) and rhinorrhea (5, 41%). In all of the cases (8, 66%), a computed tomography (CT) scan was performed, 6 (50%) had done plain sinus radiography, 3 (25%) had a magnetic resonance imaging (MRI), and 7 (58%) of the cases had done biopsy before definitive management. The majority of the patients were treated by surgical excision of the lesion, most of the cases, 6 (50%) used endoscopic sinus surgery (ESS), and 5 (41%) of them were managed by lateral rhinotomy with or without medial maxillectomy. One study reported the use of Caldwell-Luc procedure for removal of the lesion. Following surgery, two studies reported complications of epistaxis and lacrimal duct injury manifested as epiphora. Follow-up showed that 7 (58%) of the cases did not document any recurrence of the disease. The reoccurrence occurred in 5 (41%) of the cases within the 1st year after the management, and the disease reoccurred in different intervals following the surgery: 2 months, 3 months, 5 months, 10 months, and 1 year. From the five cases that had recurrence, three were managed by excision, one was managed by cauterization, another one was managed by sphenoethmoidectomy. A summary of all the cases is listed in [Table 1].
Table 1: Summary of the studies included in the review

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  Discussion Top

Pediatric sinonasal IP cases are rare and can result in considerable diagnostic and management challenges. This review aims to increase awareness of this disease in the pediatric age group. The etiology of this lesion is unknown, but for a long time, it has been considered to be associated with HPV because of the multifocal origin, high reoccurrence rate, and tendency to cover a large area.[10] DNA of HPV similar to HPV-11 was isolated from nasal tissue of a child diagnosed with IP.[10] Anjali suggests that intrapartum transmission plays a role in the etiology of IP.[10]

The occurrence of IPs in the pediatric age group is rare; it characteristically presents in adults at the age of 50–60 years; however, the incidence in the pediatric age group was reported.[11] Previously reported cases of IP in children occurred frequently in males at ages ranging from 5 to 15 years and arose most commonly from lateral nasal walls and usually secondarily involving the maxillary and ethmoid sinuses.[12] However, Cooter et al. reported that other nasal sinuses can be involved in the disease most frequently ethmoid, maxillary, frontal, and sphenoid sinuses, respectively.[13] Furthermore, it can extend extra nasally to the nasopharynx.[11] The presenting symptoms of IPs are nonspecific including nasal obstruction, rhinorrhea, facial pain, epistaxis, and anosmia[13],[14] but most commonly present with unilateral nasal obstruction.[11],[14]

There is no significant difference in the clinical behavior and histological features of IP in children compared to adult patients except that most of the pediatric cases were diagnosed after recurrence, and this attributed to the elimination of IP from the differential diagnosis in children.[11] IPs are diagnosed and treated in children the same as in adults.[11] Histopathologic examination is used to confirm the diagnosis of IPs either preoperatively or postoperatively after excision of the mass. There is a variety of benign lesions included in the differential diagnosis of IPs which are antrochoanal polyps, angiofibromatous polyps, fibrous histiocytomas, meningiomas, neurofibromas, neurilemmomas, ordinary “allergic” polyps, hemangiomas, pyogenic granulomas, and pleomorphic adenomas of salivary glandular origin.[15] Developmental anomalies (meningoencephaloceles) should be suspected as well in the pediatric age group.[15] These lesions behave in pediatrics similarly as in adults, and the association of malignancy was reported in children, but the mechanism of malignancy is still unknown.[10] The malignancy can occur at the same time or years later, and whether IP develops to a malignant lesion or they exist together at the same time is unknown.[10] Literature showed that most of the malignancies associated with IP were squamous cell carcinoma.[16] One study showed a 5.13% malignancy transformation rate, and all patients did not have a family history of malignancy or exposure to carcinogenic factors.[16] Wang suggested that it is related to the patients' biological characteristics.[16] The association of HPV with malignant transformation is controversial. The pathophysiology remains unclear, but HPV infection is thought to have a role in tumor genesis through the viral oncoproteins E6 and E7 which result in unregulated cell proliferation and oncogenesis.[17] There is a lack of data regarding IP-associated malignancy in the pediatric age group.

No radiologic findings are considered specific for IP;[15] however, imaging study aids in delineating the origin of the lesion and evaluating its extent.[11] In suspected recurrence, MRI is recommended as a basic examination. Recently, it was reported that MRI examination can be helpful in recognizing malignant transformation of IP. It has been known that IPs in the conventional MRI should be indicated by a convoluted cerebriform pattern (CCP), a band-like region of hyperintense and hypointense signals on T2-weighted images, or/and postcontrast-enhanced T1-weighted images. Recently, the authors suggested that the focal loss of a CCP might be indicatory for IPs concomitant with malignancy.[17] Nonenhanced and static combined with dynamic contrast-enhanced MRI could be a useful tool for differential diagnosis of malignancy in IP.[18]

IP in adults has a major difference in clinical presentation of published pediatric cases in that almost all cases were diagnosed after having a recurrence, and the recurrence in adults occurs after longer period. This was due to the fact that the diagnosis of IP was not initially considered in the diagnostic workup of the patient, thus resulting in either omission of a surgical biopsy or inadequate initial surgery or subtotal excision of the lesion leading to higher rate of recurrence, therefore, the need for a definitive second or third procedure. In a recent case report by Jayakody,[19] preoperative CT and MRI were done and intraoperative frozen sections were taken which confirmed the diagnosis before the definitive procedure, resulting in no reoccurrence within a 12-month follow-up.

One study which discussed different factors related to the recurrence rate indicates that the site from which the lesion is originating can increase the recurrence rate, especially lesions originating from the frontal sinus which has an impact on the difficulty of the removal of the lesion completely. Considering the staging of IP according to the Krouse staging system as a factor affecting the recurrence is debatable. Wang suggested that staging is an insignificant factor affecting the recurrence rate, whereas another study suggested that the recurrence rate is higher in T3 and T4.[16],[17] The most important factor in recurrence is incomplete surgical resection, and different surgical techniques do not play an important role in recurrence. However, Marta Gamrot-Wrzoł et al. suggested that the lowest recurrence rate was observed after combined treatment: endoscopic and open method.[17] A high recurrence rate was associated with nasal polyps and sinusitis, especially those who had undergone multi-time polypectomy. It is recommended to take general histopathological examination after the removal of nasal polyps to reduce the recurrence rate and misdiagnosis rate.[16] Another factor includes smoking which leads to swelling and chronic inflammatory condition of the mucosa of the nose and sinuses.[17] In our review, reoccurrence was observed in 5 patients and no factors were recognized to be associated with the recurrence. The methods used to remove reoccurred lesions include cauterization, excision by intranasal biopsy forceps, and sphenoethmoidectomy.[10],[20]

Due to the extremely rare incidence of IP in pediatrics, no trials exist that define optimal management. A standard procedure should be wide local excision with intraoperative histopathological investigation to prove tumor resection with a margin of healthy tissues which must be used to avoid the reoccurrence of the lesion.[15] Previously traditional open techniques such as the Caldwell-Luc procedure and lateral rhinotomy were used for the treatment of IPs which were overwhelmed by the evolving endoscopic approaches.[14] Endoscopic surgeries showed less reoccurrence rate (1%) compared to the conventional techniques (6%).[14] Furthermore, Kamel reported no reoccurrence rate 23 months following ESS. There is a lack of information when comparing these techniques in pediatric patients because of the infrequent occurrence in this age group.[13] Endoscopic surgery has less blood loss, no external scar, and short hospital stay when compared to conventional methods. However, when there is an extensive sinus involvement, and the mass is very large, it is better to use traditional or combined techniques.[13] Complications of endoscopic surgery for IPs have been reported between 0% and 19.6%. The most commonly reported complications are epistaxis, epiphora, temporary infraorbital hypesthesia, minimal orbital fat exposure, and periorbital ecchymosis, depending on the extent of the orbital bone removal or aggressive mucosal removal within the maxillary sinus cavity and surrounding structures (i.e., lacrimal system, infraorbital nerve, etc.). Cerebral spinal fluid leakage was reported in 6%. Most of these cases with CSF leak were either planned or in some cases there were already in existence with bony dehiscence and the IP was removed from the adjacent dura mater.[21] Our review reported complications following surgical removal: epiphora due to lacrimal duct injury which was corrected by dacryocystorhinostomy 2 years later.[22]

Study limitation

The occurrence of IPs in children is considered a rare condition; thus, very few studies were found, and most of them were old.

  Conclusions Top

IP is a benign tumor which can arise in the pediatric age group and should be considered in the differential diagnosis of unilateral nasal obstruction. It is diagnosed and treated likewise in adults. Endoscopic surgeries were preferred because they showed less reoccurrence rate compared to the conventional methods.

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Conflicts of interest

There are no conflicts of interest.

  References Top

Myers EN, Fernau JL, Johnson JT, Tabet JC, Barnes EL. Management of inverted papilloma. Laryngoscope 1990;100:481-90.  Back to cited text no. 1
Wakley Jun T. A mirror of the practice of medicine and surgery in the hospitals of London: Royal free hospital. Chronic disease of Knee-joint; amputation. Lancet 1850;55:390-1.  Back to cited text no. 2
Lisan Q, Laccourreye O, Bonfils P. Sinonasal inverted papilloma: From diagnosis to treatment. Eur Ann Otorhinolaryngol Head Neck Dis 2016;133:337-41.  Back to cited text no. 3
Busquets JM, Hwang PH. Endoscopic resection of sinonasal inverted papilloma: A meta-analysis. Otolaryngol Head Neck Surg 2006;134:476-82.  Back to cited text no. 4
Peng P, Har-El G. Management of inverted papillomas of the nose and paranasal sinuses. Am J Otolaryngol 2006;27:233-7.  Back to cited text no. 5
Buchwald C, Franzmann MB, Tos M. Sinonasal papillomas: A report of 82 cases in Copenhagen County, including a longitudinal epidemiological and clinical study. Laryngoscope 1995;105:72-9.  Back to cited text no. 6
WHO Classification of Tumours, Vol. 9; 2900. Available from: http://apps.who.int/bookorders/anglais/detart1.jsp?codlan=1& codcol=70&codcch=9. [Last accessed on 2020 Jan 02].  Back to cited text no. 7
Syrjänen K, Syrjänen S. Detection of human papillomavirus in sinonasal papillomas: Systematic review and meta-analysis. Laryngoscope 2013;123:181-92.  Back to cited text no. 8
Lawson W, Kaufman MR, Biller HF. Treatment outcomes in the management of inverted papilloma: An analysis of 160 cases. Laryngoscope 2003;113:1548-56.  Back to cited text no. 9
Papilloma IS, The OF, Tract S, Children IN. Inverted schneiderian papilloma of the sinonasal tract in children. Pediatr Pathol 1989;583-90.  Back to cited text no. 10
Rafii BY, Kuhn MA, Opher E, Hartman A, Lim JW. Pediatric sinonasal inverted papilloma : An uncommon occurrence and its proposed management. Laryngoscope 2011;121(Suppl. 4):s208.  Back to cited text no. 11
D'Angelo AJ Jr., Marlowe A, Marlowe FI, McFarland M. Inverted papilloma of the nose and paranasal sinuses in children. Ear Nose Throat J 1992;71:264-6.  Back to cited text no. 12
Cooter MS, Charlton SA, Lafreniere D, Spiro J. Endoscopic management of an inverted nasal papilloma in a child. Otolaryngol Head Neck Surg 1998;118:876-9.  Back to cited text no. 13
Pasquini E, Sciarretta V, Farneti G, Modugno GC, Ceroni AR. Inverted papilloma: Report of 89 cases. Am J Otolaryngol. 2004;25:178-185. doi:10.1016/j.amjoto.2004.01.004.  Back to cited text no. 14
Stanley RJ, Kelly JA, Matta II, Falkenberg KJ. Inverted papilloma in a 10-year-old boy. Arch Otolaryngol 1984;110:813-5.  Back to cited text no. 15
Li WX, Wei P. Factors affecting recurrence of sinonasal inverted papilloma. 2013;1349-53.  Back to cited text no. 16
Gamrot-Wrzoł M, Sowa P, Lisowska G, Ścierski W, Misiołek M. Risk Factors of Recurrence and Malignant Transformation of Sinonasal Inverted Papilloma. Yasumatsu R, editor. Biomed Res Int 2017;2017:9195163. doi.org/10.1155/2017/9195163.  Back to cited text no. 17
Xinyan W, Zhengyu Z, Xiaoli C, Jing L, Junfang X. inverted papilloma and malignant tumors in the nasal cavity. Chin Med J (Engl) 2014;127:1696-701.  Back to cited text no. 18
Jayakody N, Ward M, Wijayasingham G, Fowler D, Harries P, Salib R. A rare presentation of a paediatric sinonasal inverted papilloma. J Surg Case Rep 2018;2018:rjy321. Doi:10.1093/jscr/rjy321.  Back to cited text no. 19
Go K. Recurrent inverted papilloma of a pediatric patient: Clinico-radiological considerations. Int J Pediatr Otorhinolaryngol 2005;69:861-4.  Back to cited text no. 20
Wood JW, Casiano RR. Inverted papillomas and benign nonneoplastic lesions of the nasal cavity. Am J Rhinol Allergy 2012;26:157-63.  Back to cited text no. 21
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  [Figure 1]

  [Table 1]


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