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| ORIGINAL ARTICLE |
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| Year : 2016 | Volume
: 13
| Issue : 1 | Page : 36-40 |
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Outcome of treatment in patients with recurrent respiratory papilomatosis in Kano: A 10 years retrospective analysis
Ajiya Abdulrazak1, Iliyasu Yunusa Shuaibu2, Abdulazeez Omeiza Ahmed1, Abdullahi Hamisu1
1 Department of ENT, Aminu Kano Teaching Hospital, Kano, Nigeria
2 Department of Surgery, ENT Unit, Ahmadu Bello University Teaching Hospital, Zaria, Nigeria
| Date of Web Publication | 12-Feb-2016 |
Correspondence Address:
Iliyasu Yunusa Shuaibu
Department of Surgery, ENT Unit, Ahmadu Bello University Teaching Hospital, Zaria
Nigeria
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/0331-8540.172148
Background/Objectives: Respiratory papillomatosis (RP), which most often affects the larynx, is a relatively rare benign disease that can, however, have an aggressive clinical course. This study aimed to review the outcome of the patients treated for recurrent RP at Aminu Kano Teaching Hospital and the factors associated with the outcome. Materials and Methods: The records of patients managed for laryngeal papillomatosis at the Department of Otorhinolaryngology, Aminu Kano Teaching Hospital, Kano, Nigeria, over a period of 10 years between January 2004 and December 2013 were reviewed. Information obtained from the case files included demographic characteristics and clinical information. The data obtained were analysed using Statistical Package for Social Science version 16. Results: A
total of 31 patients with recurrent RP were seen. Among these, 16 (51.6%) were males and 15 (48.4%) were females with sex ratio (M: F) of 1.1:1. The mean age was 11.7 years with standard deviation of ± 1.26. The main presenting symptoms were hoarseness in all patients and difficulty in breathing in 9 (29%). Patients who had the onset of symptoms between 6 and 10 years of age constituted the majority, 17 (54.8%). Only one patient had a solitary lesion on laryngoscopy, the majority had multiple papillomatous lesions, 30 (97.8%). Emergency tracheostomy was performed to relieve upper airway obstruction in 10 (32.3%). Twenty-seven of the patients constituting 87.1% had 1–3 surgical extirpations while 4 (12.9%) had surgery between 4 and 6 times. The voice outcome was mostly poor among the patients post-surgery, 27 (87.1%). Majority of the patients were lost to follow-up, 21 (67.7%), with 1 (3.2%) recorded death and 9 (29%) still on follow-up. Conclusion: There appears to be a limited success in the outcome of our patients managed for recurrent RP. However, early diagnosis and avoidance of tracheostomy where possible are factors that may improve the outcome. Keywords: Early diagnosis, respiratory papillomatosis, treatment outcome
How to cite this article:
Abdulrazak A, Shuaibu IY, Ahmed AO, Hamisu A. Outcome of treatment in patients with recurrent respiratory papilomatosis in Kano: A 10 years retrospective analysis. Niger J Basic Clin Sci 2016;13:36-40 |
How to cite this URL:
Abdulrazak A, Shuaibu IY, Ahmed AO, Hamisu A. Outcome of treatment in patients with recurrent respiratory papilomatosis in Kano: A 10 years retrospective analysis. Niger J Basic Clin Sci [serial online] 2016 [cited 2023 Mar 31];13:36-40. Available from: https://www.njbcs.net/text.asp?2016/13/1/36/172148 |
| Introduction | |  |
Respiratory papillomatosis (RP), which most often affects the larynx, is a relatively rare benign disease that can, however, have an aggressive clinical course.[1],[2] Although relatively rare, laryngeal papillomatosis (LP) is still the most common paediatric neoplasm found in the larynx.[3] The disease course can vary greatly between individuals. In many instances, spontaneous clearance occurs after only a few surgical procedures. In other cases, however, the papillomas continue to grow, spreading throughout the airway and ultimately affecting surgically inaccessible and treatable area, where eventual degeneration to squamous cell carcinoma portends the death of these patients.
Human papillomavirus (HPV) has been found in the great majority of patients with RP. More than 120 different types have already been found; although, RP is most often caused by types 6 and 11.[4]
LP is recognised as a disease of both children and adults. The incidence among children in the United States is estimated at 4.3 per 100,000 children-meaning around 2400 new cases every year.[5] For patients in Denmark, the incidence is 3.84/100,000/year. The rate among children is 3.62/100,000, whereas adult-onset cases occur at a rate of 3.94/100,000.[6] However, the prevalence rate in Sub-Saharan Africa is not readily available.
Linderberg et al. proposed a classification of the disease into juvenile and adult forms in relation to the first appearance of the disease.[7] In terms of the clinical course of the disease, a classification into 'aggressive' and 'non-aggressive' forms is more appropriate. The term 'aggressive form of the disease' is used when a total of more than 10 surgical procedures, or more than three procedures annually, are required to improve the patients conditions.[8]
The aggressive course of the disease occurs almost equally among juvenile and adult forms (17% and 19%, respectively).[5] Some of the factors found to be associated with an aggressive disease course include: Infection with HPV11, early spread of the disease to the subglottis, appearance of the disease before age three, gastroesophageal reflux, and concurrent infection with other viruses.[4],[9],[10],[11],[12]
No single modality of treatment has been shown to be effective in the eradication of LP. The current standard treatment is surgical excision with the aim of providing optimum relief of symptoms while preserving essential anatomy and function. The CO2 laser has been the most successful tool in surgical treatment for a number of years and recently a 'microdebrider' has been increasingly used. Dye lasers have also been successful in the treatment of RP because of their anxiolytic
effect on the blood vessels in the papillomas.[4],[9],[10],[11],[13] Cold-steel microlaryngeal excision is the main-stay of treatment in Sub-Saharan Africa.
The frequent recurrence of papillomas has resulted in the use of different adjuvant treatments alongside surgery. Adjuvant treatments currently used include cidofovir, indole-3-carbinol, ribavirin, mumps vaccine and photodynamic therapy. As with surgical management, viral persistence occurs following treatment with these adjuvant modalities.[9]
Several sub-Saharan African studies have looked at the clinical pattern of RP and the outcome of patients with the disease.[14],[15],[16] Furthermore, an assessment of the impediments to clinical diagnosis and management of RP revealed several factors including patients, physician, equipment, lesion as well as administrative.[17] However, continued re-assessment of the outcome after treatment and the factors associated will go a long way in improving management of the disease and aiding further research.
The aim of this study was to review the outcome of the patients treated for recurrent RP at Aminu Kano Teaching Hospital and the factors associated with the outcome.
| Materials And Methods | | |
This was a retrospective descriptive study of patients who were managed for LP at the Department of otorhinolaryngology, Aminu Kano Teaching Hospital, Kano, Nigeria, over a 10-year period between January 2004 and December 2013. Ethical approval was obtained from the Ethical Review Committee of Aminu Kano Teaching Hospital, Kano. All the patients included had clinical, radiologic and histopathological confirmation of the disease. The main modality of treatment was rigid laryngoscopy with extirpation of lesions using cupped laryngeal forceps. Excluded from the study were patients whose case records were either not found or did not have complete information.
Information obtained from the case files included demographic characteristics such as age and sex. Clinical information included main presenting symptoms, age of onset of symptoms, number of surgical interventions and interval between surgical interventions. Presence or absence of tracheostomy, voice clarity after surgery and present status of patients were also noted.
The data were entered into the spreadsheet and analysed using the Statistical Package for Social Science version 16.0 (SPSS Inc., Chicago, Illinois, USA). Quantitative data were summarised as frequencies and percentages and presented as tables. Chi-square test was used to determine P value and to test statistical significance, which was set at a P < 0.05.
| Results | | |
Within the 10-year period of review, a total of 31 patients with recurrent RP were seen. Among the patients, 16 (51.6%) were males and 15 (48.4%) were females with sex ratio (M: F) of 1.1:1. The age ranged from 2 years to 70 years with a mean age of 11.7 years (standard deviation [SD] = 1.26) [Table 1]. The age group 6–10 years constituted the largest number of patients seen, 14 (45.2%), followed by 1–5 years age group and 11–15 years age group, 6 (19.4%) both. Up to 4 (12.9%) were above the age of 20 years [Table 1]. The main presenting symptoms were hoarseness in all patients and difficulty in breathing in 9 (29%) [Table 2]. Patients who had onset of symptoms between 6 and 10 years of age constituted the majority, 17 (54.8%) followed by those whose onsets of symptoms were between 1 and 5 years of age, 11 (35.6%) [Table 2]. Only one patient had a solitary lesion on laryngoscopy, the majority had multiple papillomatous lesions, 30 (97.8%) [Table 3]. Emergency tracheostomy was performed to relieve upper airway obstruction in 10 (32.3%) of all patients [Table 3].
Twenty-seven of the patients constituting 87.1% had 1–3 surgical extirpations while 4 (12.9%) had surgery between 4 and 6 times [Table 4]. One to 6 months was the interval between surgery in 9 (47.4%) of the patients, followed by 7–12 months in 8 (42.1%) of them. Only 2 (10.5%) patients had intervals as long as 1 year and beyond [Table 4]. The voice outcome was mostly poor among the patients post-surgery, 27 (87.1%) [Table 4]. Majority of the patients were lost to follow-up, 21 (67.7%), with 1 (3.2%) recorded death and 9 (29%) still on follow-up [Table 4].
[Table 5] shows that those patients with onset of symptoms at younger age group (1–5 years) underwent increased number of surgeries (4–6) though the relationship was not statistically significant (P = 0.53). [Table 6] shows the relationship between the performance of emergency tracheostomy and the frequency of surgical extirpations. There was a statistically significant correlation between the performance of tracheostomy and increased number of surgical interventions (exact test P = 0.007). | Table 5: Cross-tabulation of number of surgeries and age of onset (n=31)
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 | Table 6: Cross-tabulation of number of surgeries and tracheostomy (n=31)
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| Discussion | | |
Recurrent RP though relatively rare, is still a subject of extensive research because of its association with significant morbidity and occasionally mortality. The outcome of its management is still of concern to physicians as both surgical, and adjuvant therapy are yet to give satisfactory results.
The study revealed a marginal male predominance of 51.6%, with a sex ratio of 1.1:1, similar to other studies in Nigeria [15],[16],[17],[18] but in contrast to some other authors in the West African subregion [14],[19] The mean age of the patients was 11.7 years (SD = 1.26), similar to findings by some authors,[14],[18] but in slight contrast to that of Nwaorgu et al. in Ibadan, Nigeria.
In agreement with many other similar studies,[14],[15],[16],[18],[19] those between the ages of 1 and 10 years constituted the majority in our study. The study in addition, revealed the 6-10 years age group been most affected as similarly reported by Nwaorgu et al.[15] In contrast, Aliyu et al. in Kaduna, Nigeria [18] reported 1–5 years old has been mostly affected.
Comparable to findings by Mgbor et al.[19] who reported 64% of the studied patients to be 15 years and below, this study revealed similar findings. As in most studies worldwide, the disease is more commonly a disease of children.
Hoarseness was the most common presenting symptom among our patients. This is similar to findings in other studies.[14],[16],[18],[19] Though dyspnoea was a less common symptom when compared to findings by Adoga et al. in Jos, Nigeria,[16] it agrees with other studies in Nigeria,[18],[19]
Multiple laryngeal polyps was the dominant intraoperative findings in this study, this compares with findings by Mgbor et al.[19] Surgical airway bypass in the form of tracheostomy was performed in a relatively lower percentage of our patients when compared to some other studies,[15],[16],[17] but agrees with similar studies.[14],[18],[19] In addition, the study revealed a significant correlation between tracheostomy and recurrence of laryngeal lesions. The increased frequency of surgical extirpations in those patients with tracheostomy could be due to disease aggression not directly from the presence of tracheostomy as suggested by Orji et al. in his study.[20] Moreover, Shapiro et al.[21] in his study found a satisfactory outcome in his tracheostomized patients, further dissociating tracheostomy from disease aggression. However, Cole et al.[22] suggested tracheostomy worsens the outcome in his own patients, though the focus of the study was tracheal spread.
The age at onset of disease in this study was predominantly between 6 and 10 years, which is also the age group most commonly affected by the disease in our study. This could suggest early presentation by our patients to a health facility and could explain the low incidence of upper airway obstruction necessitating tracheostomy among them.
An aggressive disease is defined by the number of surgical extirpations required to improve a patient's condition. This study shows that the majority of our patients reviewed have 1–3 surgical excisions. This is contrary to other similar studies in the West African subregion where the patients underwent more sessions of surgical extirpations.[14],[15] This could mean most of our patients have the non-aggressive form of the disease. However, the recurrence rate of between 6 months and below was a common occurrence in this study, in contrast to the finding by a study in Ilorin, Nigeria.[17] Moreover, our study also found no significant correlation between the age at onset of disease and the number of surgical extirpations done for the patients.
Voice clarity after surgery was generally poor in this study and though worse, compares with a similar study by Holler et al.[23] The importance of appropriate consent taking in patients cannot be overemphasised.
The high number of patients lost to follow-up in this study probably highlights the frustration of the parents/guardians or patients and underscores the importance of proper education of patients or their parents about the nature of the disease. Though many patients in this study were lost to follow-up, there was only one recorded mortality in all the cases managed.
| Conclusion | | |
There appears to be a limited success in the outcome of our patients managed for recurrent RP. However, early diagnosis and avoidance of tracheostomy where possible are factors that may improve the outcome.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
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[Table 1], [Table 2], [Table 3], [Table 4], [Table 5], [Table 6]
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