- Email: [email protected]
Nasal septal perforation in children: Presentation, etiology, and management
Accepted Manuscript Nasal septal perforation in children: Presentation, etiology, and management David T. Chang, Alexandria L. Irace, Kosuke Kawai, Carolyn R. Rogers-Vizena, Roger Nuss, Eelam A. Adil PII:
S0165-5876(16)30430-X
DOI:
10.1016/j.ijporl.2016.12.003
Reference:
PEDOT 8338
To appear in:
International Journal of Pediatric Otorhinolaryngology
Received Date: 20 September 2016 Revised Date:
28 November 2016
Accepted Date: 1 December 2016
Please cite this article as: D.T. Chang, A.L. Irace, K. Kawai, C.R. Rogers-Vizena, R. Nuss, E.A. Adil, Nasal septal perforation in children: Presentation, etiology, and management, International Journal of Pediatric Otorhinolaryngology (2017), doi: 10.1016/j.ijporl.2016.12.003. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Nasal septal perforation in children: presentation, etiology, and management David T. Chang, MD, PhDa, Alexandria L. Irace, BAa, Kosuke Kawai, ScDa,b, Carolyn R.
a
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Rogers-Vizena, MDc, Roger Nuss, MDa,b, Eelam A. Adil, MD, MBAa,b
Department of Otolaryngology and Communication Enhancement, Boston Children’s Hospital,
300 Longwood Avenue, Boston, MA 02115, USA b
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Department of Otolaryngology, Harvard Medical School, 25 Shattuck Street, Boston, MA
02115, USA
Department of Plastic and Oral Surgery, Boston Children’s Hospital, 300 Longwood Avenue,
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c
Boston, MA 02115, USA
Presented at the American Society of Pediatric Otolaryngology Annual Meeting on May 20,
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2016 in Chicago, IL
Financial Disclosures: None
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Correspondence:
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Conflict of Interest: None
Eelam A. Adil, MD, Department of Otolaryngology and Communication Enhancement, Boston Children’s Hospital, 300 Longwood Avenue, LO-367, Boston, MA 02115. Email [email protected].
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Abstract Objective: The presentation, etiology, and treatment of nasal septal perforation have been described in the adult literature; however, reports in the pediatric population are limited. In this
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study, we review our experience with pediatric nasal septal perforations with a focus on presentation, pathogenesis, management, and outcomes of surgical repair.
Methods: A retrospective chart review was performed on pediatric patients diagnosed with
characteristics, and treatment were extracted and analyzed.
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nasal septal perforations from 1998 - 2015. Data regarding patient demographics, perforation
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Results: Twenty-seven patients met inclusion criteria. Mean age was 10.8 years (range 2 months-17 years). Nasal crusting (n=19, 73%) and epistaxis (n=15, 58%) were the most common complaints at presentation. The most common etiologies were trauma (n=9, 33%), iatrogenic sources (n=5, 19%), and neoplasm (n=3, 11%). Septal perforations were primarily located in the
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anterior septum (n= 17, 81%) and the average size was 0.9 cm (± 0.37) in diameter. Four patients were managed with a nasal septal button. Successful closure was achieved in four out of six patients (66.7%) who underwent surgical repair.
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Conclusions: In our series, septal perforations in children occurred most frequently due to digital nasal trauma, and crusting was the most common symptom. Factors to consider prior to repair
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include symptomatology, the etiology of the perforation, co-morbidities, ability to comply with post-operative care/restrictions, availability of adjacent tissue/grafts, and potential effects on nasal growth. Even with careful consideration of these factors, successful closure was limited to two-thirds of patients who were offered repair. Keywords: pediatric nasal septal perforation, cautery, nasal trauma, epistaxis
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1. Introduction Nasal septal perforation is an anatomic defect that connects the two nasal cavities through the nasal septum. This anomalous channel results in turbulent airflow patterns and damage to
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respiratory epithelia, especially when the lesion is large or located in the anterior septum [1-4]. The presentation, etiology, and treatment of nasal septal perforation have been well described in the adult literature. Common symptoms include dryness, whistling, discomfort, rhinorrhea,
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crusting, bleeding, and nasal obstruction; however, healed, well-circumscribed perforations in the posterior septum are often asymptomatic [1,5-7]. Septal perforations that remain untreated
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for long periods of time may lead to destruction of respiratory epithelia with loss of cilia, causing the mucosa to become very dry and uncomfortable [7].
Etiology plays an important role in the size, severity, and management of septal perforations. Common causes of adult nasal septal perforation include trauma, intranasal drug
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use or abuse, infection, neoplasia, connective tissue disorders, and autoimmune disorders, such as granulomatosis with polyangiitis (formerly known as Wegener’s Granulomatosis) [1,4]. It has been suggested that there is a relationship between etiology and the location of the lesion, with
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systemic disease more likely to result in a posterior perforation than anterior [1]. Ruling out significant underlying conditions is an important step in the diagnostic work-up, as septal
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perforation may be the initial presentation of some neoplastic processes such as nasal NK/T-cell lymphoma.
While symptoms, etiologies, and treatment options for septal perforation are well
described in the adult literature, reports in the pediatric population are limited to case reports and small case series. The purpose of this study is to review our cohort of pediatric nasal septal perforation patients to determine the most common presentation, etiology, and outcomes of these
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patients. Following a review of our experience, we aim to highlight some of the differences between pediatric and adult perforations.
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2. Methods
The Boston Children’s Hospital Institutional Review Board approved this retrospective study prior to acquisition of data, and its guidelines were followed. All pediatric patients under
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the age of 18 years who were diagnosed with a nasal septal perforation at our institution from 1998 to 2015 were included in this study. Patients who were over the age of 18 years at the time
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of diagnosis were excluded. We reviewed patient presentation symptoms, etiology, size and location of the perforation, and surgical repair technique and outcome, if attempted. The etiologies of perforations were divided into 10 general categories: trauma, iatrogenic, malignancy, autoimmune disease, connective tissue disease, infectious, benign lesions, nasal
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button battery, osteoradionecrosis, and unknown. Within the trauma category, digital nasal trauma (i.e. nose picking), necrosis due to septal hematoma, frequent suctioning, and prolonged nasogastric tube placement were included. Iatrogenic sources included prior nasal surgery and
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nasal cautery. Granulomatosis with polyangiitis and sarcoidosis were included in the autoimmune disease category, while systemic lupus erythematosus was included in the
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connective tissue disease group. Malignancies included lymphoma and leukemia; there were no primary nasal malignancies in our cohort. Information regarding symptoms was not available for one patient, perforation diameter was not available for five patients, and perforation location was not available for six patients. Descriptive statistics were used to describe patient’s clinical characteristics and surgical outcomes. All analyses were conducted using SAS version 9.3 (SAS Institute, Cary NC).
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3. Results A total of 27 patients met inclusion criteria. Patient demographics, presentation, and
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perforation characteristics are summarized in Table 1. There was no significant gender
difference in our series of patients, as 15 (56%) of the patients were male and 12 (44%) were female. The mean age was 10.8 years with a range of 2 months to 17 years. Nasal crusting
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(n=19, 73%) and epistaxis (n=15, 58%) were the most common presenting symptoms.
Septal perforations in our series of children occurred most frequently due to trauma,
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primarily digital nasal trauma (Figure 1). Iatrogenic sources, including nasal cautery and prior nasal surgery, were the second most common causes. Although less common, malignancy (n=3, 11%), autoimmune disorders (n=2, 7%), connective tissue disease (n=1, 4%), and infection (n=1, 4%) were also causes of septal perforation. The anterior nasal septum (n= 17, 81%) was the most
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common site. Twenty-two patients had size measurements available. The average size was 0.9 centimeter in diameter with a range of 0.3 cm to 1.5 cm. Ten out of the twenty-seven patients underwent some type of closure. Each of these
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patients is described in more detail in Table 2. The youngest to undergo repair was 2 years old (patient #5); in this case, the perforation was the result of a nasal button battery and repair was
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expedited because of concerns that he would develop a saddle nose deformity. This patient ultimately required a revision repair, which was performed through an external rhinoplasty approach. All other repairs were performed through an endonasal approach. Various techniques were employed for perforation closure. Four patients (mean age 10.5
+/- 2.1 years) had septal buttons placed. Surgical repairs for the remaining six patients (mean age 10.7 +/-6.0 years) were performed with mucoperichondrial rotational flaps, free grafts such
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as AlloDerm® (LifeCell Corporation, Branchburg, NJ), temporalis fascia, and cartilage, or a combination of these techniques. Successful closure was achieved in 4 out of 6 patients (66%).
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Mean follow-up was 2.4 years (range 1.4-4.6 years).
4. Discussion
Septal perforations in the pediatric population are rare. Reports in the literature focusing
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on children with septal perforation are sparse, outside of case reports and small case series of 3 or fewer patients. To our knowledge, this study is the first to evaluate pediatric nasal septal
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perforation using a relatively large cohort of patients. In our series, children with perforations most commonly presented with nasal crusting (73%). This is primarily the result of dry nasal mucosa secondary to turbulent nasal airflow. More than half of patients presented with epistaxis and approximately one-third complained of nasal congestion. Conversely, adults with nasal
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septal perforations most commonly present with epistaxis (58%) followed by crusting (43%) [1,5,7]. Compared to symptoms described in the adult literature, the frequency of nasal obstruction, pain, and whistling in children are similar. Additionally, the predominantly anterior
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location of septal perforation in our series of children is similar to what is reported in the literature for adult patients [5].
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In our series, trauma was the most common etiology, followed by iatrogenic causes.
These two etiologies accounted for over 50% of all cases. Septal perforations could be a consequence of trauma as simple as digital intranasal manipulation (i.e. nose picking), which we found to be the most common traumatic cause. Frequent nasal suctioning or prolonged nasogastric tube placement were other common sources of traumatic septal perforations. We suspect that mucosal injury or pressure is the inciting event in this cascade. This leads to
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inflammation and further mucosal breakdown, which then becomes a nidus for infection. Eventual breakdown of the mucoperichondrium leads to loss of blood supply to the cartilage, thereby leading to perforation. Host factors, including poor wound healing, can also contribute.
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To avoid injury, providers should avoid the nasal septum during suctioning or nasogastric tube placement. Patients should receive nasal saline daily and humidified oxygen if possible. The septum should also be assessed frequently and antibiotic ointment can be considered if there are
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signs of breakdown. In our series, all nasogastric tubes were secured to the skin using adhesive tape and no patients had a transeptal suture. There is no literature describing which technique is
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less injurious, but this could be examined in future research.
Iatrogenic sources were the second most common cause. Healthcare providers should be aware of the risk of intranasal mucosal injury leading to septal perforation during intranasal procedures such as nasal cautery. We suggest being judicious with the use of nasal cautery in
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pediatric patients with recurrent epistaxis. In our series, data regarding the method of nasal cautery is limited because it was performed at outside institutions; therefore, we cannot determine if there is a difference in perforation risk between chemical cautery (i.e. silver nitrate)
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versus monopolar or bipolar cautery techniques. In addition, we suspect bilateral cautery is more likely to result in perforation when compared to unilateral cautery; however, only one patient’s
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chart indicated that cautery had been performed bilaterally, so we are unable to provide any definitive data.
In almost 20% of cases, neoplastic and autoimmune causes were found. These less
common causes should be considered if there is no clear etiology for the septal perforation. It is important to note that those included in the malignancy category were patients with lymphoma
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and leukemia. No patients had a primary nasal malignancy. Therefore, the perforation may have been due to their systemic illness, chemotherapy, and resultant poor wound healing. The most common causes for perforations in our series are somewhat different than those
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found in adults. One retrospective study of 54 adult cases with a mean age of 54 years, showed idiopathic or unknown etiologies account for over 40% of perforations, followed by 20%
attributed to digital trauma [5]. A prospective study of 95 patients with a mean age of 41 years
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revealed nasal sprays (steroid, vasoconstrictors, or combination) were the most common cause
trauma, and 17% unknown [8].
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(28%), iatrogenic causes (nasal surgery and cautery) to account for 25% of cases, 20% from
There are nonsurgical and surgical options for management of septal perforations. Asymptomatic cases can be managed conservatively with nasal saline sprays, irrigations, emollients, and antibiotic ointments, which can prevent symptoms such as bleeding, crusting,
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dryness, and pain [7,9-12]. Surgical management can be considered in patients with refractory symptoms; however, in the pediatric population there are some special considerations that should be taken into account prior to considering surgical repair. An algorithm for the management of
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symptomatic pediatric septal perforations is presented in Figure 2. First, the etiology of the perforation should be reversed prior to considering repair. Since the most common cause in our
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series of patients was digital nasal trauma, this behavior has to discontinue prior to considering definitive repair. Second, the patient should be at an age where they can tolerate the expected post-operative care, including the possibility of nasal splints and nasal hygiene with saline/ointments. In addition, the size of the perforation and age of the child need to be considered. Availability of adjacent tissue and/or grafts may be limited in young children or those with large perforations, which could restrict repair techniques. Co-morbidities or
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medications that result in poor wound healing should also be considered, as these patients may not be ideal candidates for adjacent tissue transfer or free grafts. Finally, any potential effects on nasal growth restriction should be considered. There is no literature regarding the effects of
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pediatric septal perforation repair on future nasal growth and we do not have sufficient long-term data to make any definitive conclusions in this regard. However, the pediatric septoplasty literature indicates that unilateral or bilateral mucoperichondrial flap elevation does not
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negatively impact facial growth [14]. Cartilage separation or excision should be avoided at
growth and support zones including the perpendicular plate, which should be considered if a free
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septal cartilage graft is being considered for perforation repair.
If a child does not meet criteria for definitive septal perforation repair, then a septal button can be considered as a temporizing and sometimes permanent measure. This involves placing a prosthetic device within the perforation to mechanically obstruct the defect. In the
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pediatric population, this is often performed under general anesthesia. Risks of the button include bleeding, enlargement of the perforation, crusting, and pain. Four patients in our series achieved successful obturation of their perforations using septal buttons. In three of these patients, a
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septal button was chosen because it was felt that their perforations were symptomatic enough to warrant repair; however, they were unlikely to comply with the post-operative care or refrain
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from digital trauma. In one patient, frequent nasal suctioning was necessary and unavoidable. Therefore, a septal button was placed because of pain and epistaxis related to the perforation, but an irreversible source of continued nasal injury. Definitive surgical repair can be considered for patients who are medically stable with
refractory symptoms, a non-progressive etiology, and an ability to cooperate with post-operative instructions [1]. If there are concerns about loss of nasal support and possible saddle nose
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deformity development secondary to the size and location of the perforation, then surgical repair can also be considered. When surgical repair is desired, the goal is not only to close the defect, but also to establish normal function. An endonasal approach is feasible in most pediatric
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patients and avoids the need for a visible scar. Perforation closure was initially attempted
through an endonasal approach in all of our patients, even the very young patients who were 2 and 5 years of age. Patient #5 eventually required a revision procedure, which was performed
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through an external rhinoplasty approach. This approach was selected because he required
spreader grafts in addition to septal repair as he developed internal valve collapse secondary to
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his button battery injury.
Many different surgical repair techniques have been described, most of which involve placement of free grafts and/or local flaps [7,10,12-14]. However, there is not sufficient statistical evidence to support one type of procedure over another [10]. As a result, clinicians
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have not come to a consensus on a universal method to repair nasal septal perforations [10,12,14]. Predictive factors for successful surgical closure in primarily adult patient populations include surgeon skill, smaller size, and bilateral flap coverage [7,12,14].
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Consideration of surgical repair and technique is undoubtedly different for a child compared to an adult. Repair of a perforation for a young child can be more difficult than repair of the same
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sized perforation in an adult because of access limitations, adequate mucosal flap availability, and adherence to post-operative instructions. In our series, successful closure was achieved in 66% with combined free graft and mucosal flap repairs, as well as mucosal flap only repairs. The two failures both included graft and mucosal flap closures. We suspect that the failure in patient #9 was secondary to poor wound healing from his underlying granulomatosis with polyangiitis. In patient #10, the septal hematoma that led to his perforation probably resulted in
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poor vascularity to the adjacent tissues. Given the small number of patients who met criteria for surgical repair, meaningful comparison of surgical techniques for closure of perforations was not possible. The only pediatric case series in the literature on this topic reported results of three
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patients with symptomatic perforations who underwent auricular cartilage interposition grafts [15]. The authors reported that their smaller perforations (0.5 cm and 0.8 cm) closed fully after one procedure. The largest perforation was 1.2 cm and had to be revised twice after the initial
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surgery due to graft failure. Once additional studies on this topic are published, a systematic review and meta-analysis of different surgical techniques could be accomplished.
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Seventeen patients in our series did not undergo any type of repair. In nine patients, the caregivers chose not to pursue any type of perforation repair because they felt it was unlikely that their child could comply with post-operative restrictions. Caregivers chose not to pursue any type of repair or repair was not offered in five patients because of the severity of their child’s
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underlying systemic illness (malignancy, osteoradionecrosis, and/or systemic infection). Two patients deferred surgical repair because their perforations were minimally symptomatic. One patient was offered repair, but did not pursue it for insurance reasons.
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There are several limitations to this study. First, this is a retrospective study and data collection was limited to surgeon notes, which may not include all patient details. In particular,
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subtle details regarding decisions about management and surgical technique were not available for some patients. Second, although this study represents the largest cohort of pediatric septal perforation patients, the sample size was still relatively small and parametric analysis could not be completed; therefore, it would be valuable to have similar reports published by other providers so that we can combine data and begin to provide more definitive details regarding this entity and management outcomes. Finally, the mean follow-up period was 2.4 years (range 1.4-
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4.6 years); it is possible that some patients eventually developed a reperforation outside of the follow-up period.
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5. Conclusions
In our series, septal perforations in children occurred most frequently due to digital nasal trauma, which is different from the adult literature where idiopathic, medication, and traumatic
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sources are most common. Neoplastic or autoimmune causes were infrequent, but should be considered in pediatric patients with no other clear etiology for their septal perforation. Careful
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consideration should be given to the etiology of the perforation and cooperativeness of the
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patient prior to considering definitive pediatric septal perforation repair.
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REFERENCES [1] Lanier B, Kai G, Marple B, Wall GM, Pathophysiology and progression of nasal septal perforation, Ann Allergy Asthma Immunol. 99 (6) (2007) 473-479, quiz 480-471, 521.
nasal septal perforation, Yonsei Med J. 49 (2) (2008) 244-248.
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[2] Lee KC, Lee NH, Ban JH, Jin SM, Surgical treatment using an allograft dermal matrix for
[3] Pless D, Keck T, Wiesmiller KM, Lamche R, Aschoff AJ, Lindemann J, Numerical
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simulation of airflow patterns and air temperature distribution during inspiration in a nose model with septal perforation, Am J Rhinol. 18 (6) (2004) 357-362.
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[4] Brain D, The nasal septum, in: Kerr A (Ed.), Scott Brown's Otolaryngology, 5th ed, Butterworth & Co., London, 1987, pp. 154-179.
[5] Diamantopoulos, II, Jones NS, The investigation of nasal septal perforations and ulcers, J Laryngol Otol. 115 (7) (2001) 541-544.
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[6] Eviatar A, Myssiorek D, Repair of nasal septal perforations with tragal cartilage and perichondrium grafts, Otolaryngol Head Neck Surg. 100 (4) (1989) 300-302. [7] Kridel RW, Considerations in the etiology, treatment, and repair of septal perforations, Facial
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Plast Surg Clin North Am. 12 (4) (2004), 435-450, vi. [8] Dosen LK, Haye R, Nasal septal perforation 1981-2005: changes in etiology, gender and
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size, BMC Ear Nose Throat Disord. 7 (2007) 1. [9] Chiang MY, Shah P, Nasal septal perforation enlargement related to topical ocular steroids, Br J Clin Pharmacol. 60 (6) (2005) 664-665. [10] Goh AY, Hussain SS, Different surgical treatments for nasal septal perforation and their outcomes, J Laryngol Otol. 121 (5) (2007) 419-426.
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[11] Redleaf MI, Fyler EA, Frodel JL, Jr., Hoffman HT, Custom septal button, Otolaryngol Head Neck Surg. 108 (2) (1993) 196-198. [12] Kim SW, Rhee CS, Nasal septal perforation repair: predictive factors and systematic review
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of the literature, Curr Opin Otolaryngol Head Neck Surg. 20 (1) (2012) 58-65.
[13] al-Khabori MJ, Simple method of insertion of Xomed one piece septal button, J Laryngol Otol. 106 (4) (1992) 358-360.
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[14] Moon IJ, Kim SW, Han DH, et al, Predictive factors for the outcome of nasal septal perforation repair, Auris Nasus Larynx. 38 (1) (2011) 52-57.
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[15] Chua DY, Tan HK, Repair of nasal septal perforations using auricular conchal cartilage graft in children: report on three cases and literature review, Int J Pediatr Otorhinolaryngol.
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70 (7) (2006) 1219-24.
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Figure Legend Figure 1. Pie graph depicting the causes of pediatric nasal septal perforation. Data is presented as counts (% of total).
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Figure 2. Proposed algorithm for management of symptomatic pediatric nasal septal
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perforations.
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12
44%
10.8
(± 4.8)
Crusting
19
(73%)
Epistaxis
15
(58%)
Congestion
8
(31%)
Whistling
5
(19%)
Pain
5
(19%)
0.86
(± 0.37)
Anterior
17
(81%)
Mid-septum
3
(14%)
Posterior
1
Age (years), mean (±SD)
Diameter (cm), mean (±SD)
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Symptoms
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Location
(5%)
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Note that data regarding symptoms was available for 26, diameter for 22, and location for 21 patients.
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*
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Gender, female
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Table 1. Demographics and perforation details of 27* patients with septal perforation Characteristics N %
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Table 2. Summary table of 10 patients who underwent closure of their septal perforations Patient Age Diameter Etiology Type of Surgical Repair (years) (cm)
N/A
3.1
N/A
2.6
N/A
1.4
Complete closure
4.6
Complete closure Complete closure Complete closure
2.2
1) Mucoperichondrial rotational flap closure 2) Auricular cartilage graft
Reperforation
2.3
1) Alloderm graft 2) Same with mucoperichondrial rotational flap over graft
Reperforation
1.8
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N/A
Follow-up Duration (years) 2.2
8
1
Suction Trauma
Nasal septal button
2
10
1.4
Unknown
Nasal septal button
3
11
1.25
Nasal septal button
4
13
0.5
Digital Nasal Trauma Unknown
5
2
1.0
Nasal battery
6
5
1.0
Hemangioma
1) Repair with auricular cartilage 2) External rhinoplasty approach with temporalis fascia and auricular cartilage grafts Mucoperichondrial rotational flap closure
7
11
1.5
Unknown
Mucoperichondrial rotational flap closure
8
13
0.3
9
16
1.0
10
17
0.8
Prior nasal surgery and nasal cautery Granulomatosis with polyangiitis (Wegener’s granulomatosis) Necrosis due to septal hematoma
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Nasal septal button
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1
1.9 1.6
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Septal dermatoplasty with temporalis fascia graft
Result of Repair
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S0165-5876(16)30430-X
DOI:
10.1016/j.ijporl.2016.12.003
Reference:
PEDOT 8338
To appear in:
International Journal of Pediatric Otorhinolaryngology
Received Date: 20 September 2016 Revised Date:
28 November 2016
Accepted Date: 1 December 2016
Please cite this article as: D.T. Chang, A.L. Irace, K. Kawai, C.R. Rogers-Vizena, R. Nuss, E.A. Adil, Nasal septal perforation in children: Presentation, etiology, and management, International Journal of Pediatric Otorhinolaryngology (2017), doi: 10.1016/j.ijporl.2016.12.003. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT
Nasal septal perforation in children: presentation, etiology, and management David T. Chang, MD, PhDa, Alexandria L. Irace, BAa, Kosuke Kawai, ScDa,b, Carolyn R.
a
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Rogers-Vizena, MDc, Roger Nuss, MDa,b, Eelam A. Adil, MD, MBAa,b
Department of Otolaryngology and Communication Enhancement, Boston Children’s Hospital,
300 Longwood Avenue, Boston, MA 02115, USA b
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Department of Otolaryngology, Harvard Medical School, 25 Shattuck Street, Boston, MA
02115, USA
Department of Plastic and Oral Surgery, Boston Children’s Hospital, 300 Longwood Avenue,
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c
Boston, MA 02115, USA
Presented at the American Society of Pediatric Otolaryngology Annual Meeting on May 20,
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2016 in Chicago, IL
Financial Disclosures: None
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Correspondence:
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Conflict of Interest: None
Eelam A. Adil, MD, Department of Otolaryngology and Communication Enhancement, Boston Children’s Hospital, 300 Longwood Avenue, LO-367, Boston, MA 02115. Email [email protected].
ACCEPTED MANUSCRIPT
Abstract Objective: The presentation, etiology, and treatment of nasal septal perforation have been described in the adult literature; however, reports in the pediatric population are limited. In this
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study, we review our experience with pediatric nasal septal perforations with a focus on presentation, pathogenesis, management, and outcomes of surgical repair.
Methods: A retrospective chart review was performed on pediatric patients diagnosed with
characteristics, and treatment were extracted and analyzed.
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nasal septal perforations from 1998 - 2015. Data regarding patient demographics, perforation
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Results: Twenty-seven patients met inclusion criteria. Mean age was 10.8 years (range 2 months-17 years). Nasal crusting (n=19, 73%) and epistaxis (n=15, 58%) were the most common complaints at presentation. The most common etiologies were trauma (n=9, 33%), iatrogenic sources (n=5, 19%), and neoplasm (n=3, 11%). Septal perforations were primarily located in the
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anterior septum (n= 17, 81%) and the average size was 0.9 cm (± 0.37) in diameter. Four patients were managed with a nasal septal button. Successful closure was achieved in four out of six patients (66.7%) who underwent surgical repair.
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Conclusions: In our series, septal perforations in children occurred most frequently due to digital nasal trauma, and crusting was the most common symptom. Factors to consider prior to repair
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include symptomatology, the etiology of the perforation, co-morbidities, ability to comply with post-operative care/restrictions, availability of adjacent tissue/grafts, and potential effects on nasal growth. Even with careful consideration of these factors, successful closure was limited to two-thirds of patients who were offered repair. Keywords: pediatric nasal septal perforation, cautery, nasal trauma, epistaxis
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1. Introduction Nasal septal perforation is an anatomic defect that connects the two nasal cavities through the nasal septum. This anomalous channel results in turbulent airflow patterns and damage to
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respiratory epithelia, especially when the lesion is large or located in the anterior septum [1-4]. The presentation, etiology, and treatment of nasal septal perforation have been well described in the adult literature. Common symptoms include dryness, whistling, discomfort, rhinorrhea,
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crusting, bleeding, and nasal obstruction; however, healed, well-circumscribed perforations in the posterior septum are often asymptomatic [1,5-7]. Septal perforations that remain untreated
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for long periods of time may lead to destruction of respiratory epithelia with loss of cilia, causing the mucosa to become very dry and uncomfortable [7].
Etiology plays an important role in the size, severity, and management of septal perforations. Common causes of adult nasal septal perforation include trauma, intranasal drug
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use or abuse, infection, neoplasia, connective tissue disorders, and autoimmune disorders, such as granulomatosis with polyangiitis (formerly known as Wegener’s Granulomatosis) [1,4]. It has been suggested that there is a relationship between etiology and the location of the lesion, with
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systemic disease more likely to result in a posterior perforation than anterior [1]. Ruling out significant underlying conditions is an important step in the diagnostic work-up, as septal
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perforation may be the initial presentation of some neoplastic processes such as nasal NK/T-cell lymphoma.
While symptoms, etiologies, and treatment options for septal perforation are well
described in the adult literature, reports in the pediatric population are limited to case reports and small case series. The purpose of this study is to review our cohort of pediatric nasal septal perforation patients to determine the most common presentation, etiology, and outcomes of these
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patients. Following a review of our experience, we aim to highlight some of the differences between pediatric and adult perforations.
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2. Methods
The Boston Children’s Hospital Institutional Review Board approved this retrospective study prior to acquisition of data, and its guidelines were followed. All pediatric patients under
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the age of 18 years who were diagnosed with a nasal septal perforation at our institution from 1998 to 2015 were included in this study. Patients who were over the age of 18 years at the time
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of diagnosis were excluded. We reviewed patient presentation symptoms, etiology, size and location of the perforation, and surgical repair technique and outcome, if attempted. The etiologies of perforations were divided into 10 general categories: trauma, iatrogenic, malignancy, autoimmune disease, connective tissue disease, infectious, benign lesions, nasal
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button battery, osteoradionecrosis, and unknown. Within the trauma category, digital nasal trauma (i.e. nose picking), necrosis due to septal hematoma, frequent suctioning, and prolonged nasogastric tube placement were included. Iatrogenic sources included prior nasal surgery and
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nasal cautery. Granulomatosis with polyangiitis and sarcoidosis were included in the autoimmune disease category, while systemic lupus erythematosus was included in the
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connective tissue disease group. Malignancies included lymphoma and leukemia; there were no primary nasal malignancies in our cohort. Information regarding symptoms was not available for one patient, perforation diameter was not available for five patients, and perforation location was not available for six patients. Descriptive statistics were used to describe patient’s clinical characteristics and surgical outcomes. All analyses were conducted using SAS version 9.3 (SAS Institute, Cary NC).
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3. Results A total of 27 patients met inclusion criteria. Patient demographics, presentation, and
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perforation characteristics are summarized in Table 1. There was no significant gender
difference in our series of patients, as 15 (56%) of the patients were male and 12 (44%) were female. The mean age was 10.8 years with a range of 2 months to 17 years. Nasal crusting
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(n=19, 73%) and epistaxis (n=15, 58%) were the most common presenting symptoms.
Septal perforations in our series of children occurred most frequently due to trauma,
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primarily digital nasal trauma (Figure 1). Iatrogenic sources, including nasal cautery and prior nasal surgery, were the second most common causes. Although less common, malignancy (n=3, 11%), autoimmune disorders (n=2, 7%), connective tissue disease (n=1, 4%), and infection (n=1, 4%) were also causes of septal perforation. The anterior nasal septum (n= 17, 81%) was the most
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common site. Twenty-two patients had size measurements available. The average size was 0.9 centimeter in diameter with a range of 0.3 cm to 1.5 cm. Ten out of the twenty-seven patients underwent some type of closure. Each of these
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patients is described in more detail in Table 2. The youngest to undergo repair was 2 years old (patient #5); in this case, the perforation was the result of a nasal button battery and repair was
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expedited because of concerns that he would develop a saddle nose deformity. This patient ultimately required a revision repair, which was performed through an external rhinoplasty approach. All other repairs were performed through an endonasal approach. Various techniques were employed for perforation closure. Four patients (mean age 10.5
+/- 2.1 years) had septal buttons placed. Surgical repairs for the remaining six patients (mean age 10.7 +/-6.0 years) were performed with mucoperichondrial rotational flaps, free grafts such
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as AlloDerm® (LifeCell Corporation, Branchburg, NJ), temporalis fascia, and cartilage, or a combination of these techniques. Successful closure was achieved in 4 out of 6 patients (66%).
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Mean follow-up was 2.4 years (range 1.4-4.6 years).
4. Discussion
Septal perforations in the pediatric population are rare. Reports in the literature focusing
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on children with septal perforation are sparse, outside of case reports and small case series of 3 or fewer patients. To our knowledge, this study is the first to evaluate pediatric nasal septal
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perforation using a relatively large cohort of patients. In our series, children with perforations most commonly presented with nasal crusting (73%). This is primarily the result of dry nasal mucosa secondary to turbulent nasal airflow. More than half of patients presented with epistaxis and approximately one-third complained of nasal congestion. Conversely, adults with nasal
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septal perforations most commonly present with epistaxis (58%) followed by crusting (43%) [1,5,7]. Compared to symptoms described in the adult literature, the frequency of nasal obstruction, pain, and whistling in children are similar. Additionally, the predominantly anterior
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location of septal perforation in our series of children is similar to what is reported in the literature for adult patients [5].
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In our series, trauma was the most common etiology, followed by iatrogenic causes.
These two etiologies accounted for over 50% of all cases. Septal perforations could be a consequence of trauma as simple as digital intranasal manipulation (i.e. nose picking), which we found to be the most common traumatic cause. Frequent nasal suctioning or prolonged nasogastric tube placement were other common sources of traumatic septal perforations. We suspect that mucosal injury or pressure is the inciting event in this cascade. This leads to
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inflammation and further mucosal breakdown, which then becomes a nidus for infection. Eventual breakdown of the mucoperichondrium leads to loss of blood supply to the cartilage, thereby leading to perforation. Host factors, including poor wound healing, can also contribute.
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To avoid injury, providers should avoid the nasal septum during suctioning or nasogastric tube placement. Patients should receive nasal saline daily and humidified oxygen if possible. The septum should also be assessed frequently and antibiotic ointment can be considered if there are
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signs of breakdown. In our series, all nasogastric tubes were secured to the skin using adhesive tape and no patients had a transeptal suture. There is no literature describing which technique is
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less injurious, but this could be examined in future research.
Iatrogenic sources were the second most common cause. Healthcare providers should be aware of the risk of intranasal mucosal injury leading to septal perforation during intranasal procedures such as nasal cautery. We suggest being judicious with the use of nasal cautery in
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pediatric patients with recurrent epistaxis. In our series, data regarding the method of nasal cautery is limited because it was performed at outside institutions; therefore, we cannot determine if there is a difference in perforation risk between chemical cautery (i.e. silver nitrate)
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versus monopolar or bipolar cautery techniques. In addition, we suspect bilateral cautery is more likely to result in perforation when compared to unilateral cautery; however, only one patient’s
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chart indicated that cautery had been performed bilaterally, so we are unable to provide any definitive data.
In almost 20% of cases, neoplastic and autoimmune causes were found. These less
common causes should be considered if there is no clear etiology for the septal perforation. It is important to note that those included in the malignancy category were patients with lymphoma
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and leukemia. No patients had a primary nasal malignancy. Therefore, the perforation may have been due to their systemic illness, chemotherapy, and resultant poor wound healing. The most common causes for perforations in our series are somewhat different than those
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found in adults. One retrospective study of 54 adult cases with a mean age of 54 years, showed idiopathic or unknown etiologies account for over 40% of perforations, followed by 20%
attributed to digital trauma [5]. A prospective study of 95 patients with a mean age of 41 years
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revealed nasal sprays (steroid, vasoconstrictors, or combination) were the most common cause
trauma, and 17% unknown [8].
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(28%), iatrogenic causes (nasal surgery and cautery) to account for 25% of cases, 20% from
There are nonsurgical and surgical options for management of septal perforations. Asymptomatic cases can be managed conservatively with nasal saline sprays, irrigations, emollients, and antibiotic ointments, which can prevent symptoms such as bleeding, crusting,
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dryness, and pain [7,9-12]. Surgical management can be considered in patients with refractory symptoms; however, in the pediatric population there are some special considerations that should be taken into account prior to considering surgical repair. An algorithm for the management of
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symptomatic pediatric septal perforations is presented in Figure 2. First, the etiology of the perforation should be reversed prior to considering repair. Since the most common cause in our
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series of patients was digital nasal trauma, this behavior has to discontinue prior to considering definitive repair. Second, the patient should be at an age where they can tolerate the expected post-operative care, including the possibility of nasal splints and nasal hygiene with saline/ointments. In addition, the size of the perforation and age of the child need to be considered. Availability of adjacent tissue and/or grafts may be limited in young children or those with large perforations, which could restrict repair techniques. Co-morbidities or
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medications that result in poor wound healing should also be considered, as these patients may not be ideal candidates for adjacent tissue transfer or free grafts. Finally, any potential effects on nasal growth restriction should be considered. There is no literature regarding the effects of
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pediatric septal perforation repair on future nasal growth and we do not have sufficient long-term data to make any definitive conclusions in this regard. However, the pediatric septoplasty literature indicates that unilateral or bilateral mucoperichondrial flap elevation does not
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negatively impact facial growth [14]. Cartilage separation or excision should be avoided at
growth and support zones including the perpendicular plate, which should be considered if a free
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septal cartilage graft is being considered for perforation repair.
If a child does not meet criteria for definitive septal perforation repair, then a septal button can be considered as a temporizing and sometimes permanent measure. This involves placing a prosthetic device within the perforation to mechanically obstruct the defect. In the
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pediatric population, this is often performed under general anesthesia. Risks of the button include bleeding, enlargement of the perforation, crusting, and pain. Four patients in our series achieved successful obturation of their perforations using septal buttons. In three of these patients, a
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septal button was chosen because it was felt that their perforations were symptomatic enough to warrant repair; however, they were unlikely to comply with the post-operative care or refrain
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from digital trauma. In one patient, frequent nasal suctioning was necessary and unavoidable. Therefore, a septal button was placed because of pain and epistaxis related to the perforation, but an irreversible source of continued nasal injury. Definitive surgical repair can be considered for patients who are medically stable with
refractory symptoms, a non-progressive etiology, and an ability to cooperate with post-operative instructions [1]. If there are concerns about loss of nasal support and possible saddle nose
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deformity development secondary to the size and location of the perforation, then surgical repair can also be considered. When surgical repair is desired, the goal is not only to close the defect, but also to establish normal function. An endonasal approach is feasible in most pediatric
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patients and avoids the need for a visible scar. Perforation closure was initially attempted
through an endonasal approach in all of our patients, even the very young patients who were 2 and 5 years of age. Patient #5 eventually required a revision procedure, which was performed
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through an external rhinoplasty approach. This approach was selected because he required
spreader grafts in addition to septal repair as he developed internal valve collapse secondary to
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his button battery injury.
Many different surgical repair techniques have been described, most of which involve placement of free grafts and/or local flaps [7,10,12-14]. However, there is not sufficient statistical evidence to support one type of procedure over another [10]. As a result, clinicians
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have not come to a consensus on a universal method to repair nasal septal perforations [10,12,14]. Predictive factors for successful surgical closure in primarily adult patient populations include surgeon skill, smaller size, and bilateral flap coverage [7,12,14].
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Consideration of surgical repair and technique is undoubtedly different for a child compared to an adult. Repair of a perforation for a young child can be more difficult than repair of the same
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sized perforation in an adult because of access limitations, adequate mucosal flap availability, and adherence to post-operative instructions. In our series, successful closure was achieved in 66% with combined free graft and mucosal flap repairs, as well as mucosal flap only repairs. The two failures both included graft and mucosal flap closures. We suspect that the failure in patient #9 was secondary to poor wound healing from his underlying granulomatosis with polyangiitis. In patient #10, the septal hematoma that led to his perforation probably resulted in
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poor vascularity to the adjacent tissues. Given the small number of patients who met criteria for surgical repair, meaningful comparison of surgical techniques for closure of perforations was not possible. The only pediatric case series in the literature on this topic reported results of three
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patients with symptomatic perforations who underwent auricular cartilage interposition grafts [15]. The authors reported that their smaller perforations (0.5 cm and 0.8 cm) closed fully after one procedure. The largest perforation was 1.2 cm and had to be revised twice after the initial
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surgery due to graft failure. Once additional studies on this topic are published, a systematic review and meta-analysis of different surgical techniques could be accomplished.
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Seventeen patients in our series did not undergo any type of repair. In nine patients, the caregivers chose not to pursue any type of perforation repair because they felt it was unlikely that their child could comply with post-operative restrictions. Caregivers chose not to pursue any type of repair or repair was not offered in five patients because of the severity of their child’s
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underlying systemic illness (malignancy, osteoradionecrosis, and/or systemic infection). Two patients deferred surgical repair because their perforations were minimally symptomatic. One patient was offered repair, but did not pursue it for insurance reasons.
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There are several limitations to this study. First, this is a retrospective study and data collection was limited to surgeon notes, which may not include all patient details. In particular,
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subtle details regarding decisions about management and surgical technique were not available for some patients. Second, although this study represents the largest cohort of pediatric septal perforation patients, the sample size was still relatively small and parametric analysis could not be completed; therefore, it would be valuable to have similar reports published by other providers so that we can combine data and begin to provide more definitive details regarding this entity and management outcomes. Finally, the mean follow-up period was 2.4 years (range 1.4-
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4.6 years); it is possible that some patients eventually developed a reperforation outside of the follow-up period.
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5. Conclusions
In our series, septal perforations in children occurred most frequently due to digital nasal trauma, which is different from the adult literature where idiopathic, medication, and traumatic
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sources are most common. Neoplastic or autoimmune causes were infrequent, but should be considered in pediatric patients with no other clear etiology for their septal perforation. Careful
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consideration should be given to the etiology of the perforation and cooperativeness of the
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patient prior to considering definitive pediatric septal perforation repair.
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REFERENCES [1] Lanier B, Kai G, Marple B, Wall GM, Pathophysiology and progression of nasal septal perforation, Ann Allergy Asthma Immunol. 99 (6) (2007) 473-479, quiz 480-471, 521.
nasal septal perforation, Yonsei Med J. 49 (2) (2008) 244-248.
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[2] Lee KC, Lee NH, Ban JH, Jin SM, Surgical treatment using an allograft dermal matrix for
[3] Pless D, Keck T, Wiesmiller KM, Lamche R, Aschoff AJ, Lindemann J, Numerical
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simulation of airflow patterns and air temperature distribution during inspiration in a nose model with septal perforation, Am J Rhinol. 18 (6) (2004) 357-362.
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[4] Brain D, The nasal septum, in: Kerr A (Ed.), Scott Brown's Otolaryngology, 5th ed, Butterworth & Co., London, 1987, pp. 154-179.
[5] Diamantopoulos, II, Jones NS, The investigation of nasal septal perforations and ulcers, J Laryngol Otol. 115 (7) (2001) 541-544.
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[6] Eviatar A, Myssiorek D, Repair of nasal septal perforations with tragal cartilage and perichondrium grafts, Otolaryngol Head Neck Surg. 100 (4) (1989) 300-302. [7] Kridel RW, Considerations in the etiology, treatment, and repair of septal perforations, Facial
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Plast Surg Clin North Am. 12 (4) (2004), 435-450, vi. [8] Dosen LK, Haye R, Nasal septal perforation 1981-2005: changes in etiology, gender and
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size, BMC Ear Nose Throat Disord. 7 (2007) 1. [9] Chiang MY, Shah P, Nasal septal perforation enlargement related to topical ocular steroids, Br J Clin Pharmacol. 60 (6) (2005) 664-665. [10] Goh AY, Hussain SS, Different surgical treatments for nasal septal perforation and their outcomes, J Laryngol Otol. 121 (5) (2007) 419-426.
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[11] Redleaf MI, Fyler EA, Frodel JL, Jr., Hoffman HT, Custom septal button, Otolaryngol Head Neck Surg. 108 (2) (1993) 196-198. [12] Kim SW, Rhee CS, Nasal septal perforation repair: predictive factors and systematic review
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of the literature, Curr Opin Otolaryngol Head Neck Surg. 20 (1) (2012) 58-65.
[13] al-Khabori MJ, Simple method of insertion of Xomed one piece septal button, J Laryngol Otol. 106 (4) (1992) 358-360.
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[14] Moon IJ, Kim SW, Han DH, et al, Predictive factors for the outcome of nasal septal perforation repair, Auris Nasus Larynx. 38 (1) (2011) 52-57.
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[15] Chua DY, Tan HK, Repair of nasal septal perforations using auricular conchal cartilage graft in children: report on three cases and literature review, Int J Pediatr Otorhinolaryngol.
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70 (7) (2006) 1219-24.
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Figure Legend Figure 1. Pie graph depicting the causes of pediatric nasal septal perforation. Data is presented as counts (% of total).
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Figure 2. Proposed algorithm for management of symptomatic pediatric nasal septal
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perforations.
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12
44%
10.8
(± 4.8)
Crusting
19
(73%)
Epistaxis
15
(58%)
Congestion
8
(31%)
Whistling
5
(19%)
Pain
5
(19%)
0.86
(± 0.37)
Anterior
17
(81%)
Mid-septum
3
(14%)
Posterior
1
Age (years), mean (±SD)
Diameter (cm), mean (±SD)
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Symptoms
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Location
(5%)
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Note that data regarding symptoms was available for 26, diameter for 22, and location for 21 patients.
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*
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Gender, female
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Table 1. Demographics and perforation details of 27* patients with septal perforation Characteristics N %
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Table 2. Summary table of 10 patients who underwent closure of their septal perforations Patient Age Diameter Etiology Type of Surgical Repair (years) (cm)
N/A
3.1
N/A
2.6
N/A
1.4
Complete closure
4.6
Complete closure Complete closure Complete closure
2.2
1) Mucoperichondrial rotational flap closure 2) Auricular cartilage graft
Reperforation
2.3
1) Alloderm graft 2) Same with mucoperichondrial rotational flap over graft
Reperforation
1.8
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N/A
Follow-up Duration (years) 2.2
8
1
Suction Trauma
Nasal septal button
2
10
1.4
Unknown
Nasal septal button
3
11
1.25
Nasal septal button
4
13
0.5
Digital Nasal Trauma Unknown
5
2
1.0
Nasal battery
6
5
1.0
Hemangioma
1) Repair with auricular cartilage 2) External rhinoplasty approach with temporalis fascia and auricular cartilage grafts Mucoperichondrial rotational flap closure
7
11
1.5
Unknown
Mucoperichondrial rotational flap closure
8
13
0.3
9
16
1.0
10
17
0.8
Prior nasal surgery and nasal cautery Granulomatosis with polyangiitis (Wegener’s granulomatosis) Necrosis due to septal hematoma
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Nasal septal button
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1
1.9 1.6
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Septal dermatoplasty with temporalis fascia graft
Result of Repair
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