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East and Central African Journal of Surgery
Association of Surgeons of East Africa and College of Surgeons of East Central and Southern Africa
ISSN: 1024-297X EISSN: 2073-9990
Vol. 13, Num. 2, 2008, pp. 105-108
Untitled Document

East and Central African Journal of Surgery, Vol. 13, No. 2, September, 2008, pp. 105-108

Recurrent Respiratory Papillomatosis in Jos, Nigeria: Clinical Presentation, Management and Outcome

A. A. Adoga1, L. T. Nimkur1, A. S. Adoga1,

1Lecturer/Otorhinolaryngologist, Otorhinolaryngology Unit, Department of Surgery, Jos University Teaching Hospital, Pmb 2076, Jos, Plateau State, Nigeria.
Correspondence to:
Dr. Adeyi A. Adoga, Email: adeyiadoga@yahoo.com

Code Number: js08042

Background: Recurrent respiratory papillomatosis is a benign neoplastic disease of viral aetiology which can have significant morbidity or mortality and presents great challenges in it management. This study evaluates our experience with the management of this disease.
Methods: This was a 10 year retrospective review of 29 patients with histologically confirmed recurrent respiratory papillomatosis managed in the Jos University Teaching Hospital, Jos, Nigeria.
Results:
Twenty nine patients aged between 2 months and 45 years were studied. There were 21(72.4%) males and 8 (27.6%) females. The highest incidence (62.1%) was in children less than 10 years.The presenting features were hoarseness in all patients and dyspnea in 28 (96.6%) patients who had emergency tracheostomy. Twenty five (89.3%) patients were decannulated within 1 week. No patient had distal viral spread. Twenty three (79.3%) patients presented within 10 months of onset of symptoms and 6 (20.7%) patients after this period.All the patients had endoscopic surgical excision. The commonest site of lesions was the vocal cords in 16 (55.2%) patients and the anterior commissure in 13 (44.8%) patients. Recurrence occurred in 2 (6.9%) patients. No malignant transformation occurred.
Conclusion:
Conventional endoscopic surgical excision is safe and tracheostomy when needed is recommended in the management of patients with recurrent respiratory papillomatosis.

Introduction

Recurrent respiratory papillomatosis is a benign neoplastic disease characterized by warty-like lesions occurring anywhere along the aerodigestive tract and caused by the Human Papilloma virus (HPV) of which 90 subtypes has been described1. HPV types 6 and 11 are responsible for papilloma in the airway2. Two clinical forms of the disease occur, the juvenile-onset recurrent respiratory papillomatosis (JORRP), an aggressive form and the adult-onset recurrent respiratory papillomatosis (AORRP), which is less aggressive, the former being commoner3. It is a potentially devastating disease exhibiting high recurrence and can have significant morbidity presenting with hoarseness or result in mortality due to airway compromise or less commonly malignant transformation4- recurrent respiratory papillomatosis is an acknowledged risk factor for pulmonary squamous cell carcinoma5.

Surgical debulking is the foundation of treatment with endoscopic laser surgery being the current mainstay of treatment6. However, newer surgical techniques utilizing powered microdebriders are taking the place of carbon-dioxide laser ablation7. Research studies are ongoing investigating adjuvant medical therapies such as oral indole-3-carbinol8, intralesional cidofovir injections9 and lymphoblastoid interferon10 amongst others. No safe adjuvant therapy is currently available7.

Tracheostomy in the management of recurrent respiratory papillomatosis is controversial, with advocates11 and those who recommend its avoidance if possible as distal viral spread to uninvolved respiratory mucosa could occur12. This group also suggests early decannulation if tracheostomy is unavoidable. Iatrogenic airway stenosis, posterior glottic stenosis13 and glottic webs14 may also occur post-operatively. We present the clinical presentation findings, management and outcome of patients with recurrent respiratory papillomatosis treated over a 10 year period in the Jos University Teaching Hospital, Jos, Nigeria.

Patients and Methods

Following approval from the Ethical Clearance Committee of our institution, the medical records of consecutive patients presenting to our hospital with recurrent respiratory papilloma within the period August 1997 to July 2007 was analyzed for age, sex, clinical presentation, management profile and outcome of treatment. The results are presented in simple descriptive form and tables.

Results

Twenty nine patients aged between 2 months and 45 years were managed for respiratory papilloma within the study period. There were 21(72.4%) males and 8 (27.6%) females, giving a male: female ratio of 2.6:1. The highest incidence (62.1%) of this disease was seen in children less than 10 years of age (Table 1). All the patients had histological confirmation of recurrent respiratory papilloma. The presenting features were hoarseness in all the patients and dyspnea in 28 (96.6%) of the cases. Twenty three (79.3%) patients presented within 10 months of onset of symptoms and 6 (20.7%) patients after this period.

All but one patient, a 37 year old male, had emergency tracheostomy. Twenty five (89.3%) of these patients were decannulated within 1 week of insertion of tracheostomy tubes, 2 (7.1%) patients within 2 weeks and 1 (3.6%) patient within 3 weeks (Table 2). No patient had evidence of distal viral spread to uninvolved respiratory mucosa. All the patients had direct laryngoscopy and clearance with relief of symptoms. The commonest site of lesions was the vocal cords in 16 (55.2%) patients and the anterior commissure in 13 (44.8%) patients. Follow-up revealed recurrence in two patients who had one repeated episode of direct laryngoscopy and clearance each, giving a recurrence rate of 6.9%. There was no case of post-operative airway stenosis, glottic web and no malignant transformation recorded.

Discussion

Recurrent respiratory papillomatosis is a potentially devastating benign neoplastic disease of viral etiology which can have significant morbidity or result in mortality and present great challenges in its management.

Like in a previous study in Ibadan, Nigeria15 the incidence of this disease is higher in the under 10 age group. Our study still reports the vocal cords and the anterior commissure as the commonest sites of recurrent respiratory papillomatosis.We recorded a relatively low recurrence rate of 6.9% compared to other documented studies4,15. Post-operative complications such as airway stenosis13 and glottic webs14 may occur. Our study reports no case of complications following surgery and there was no malignant transformation recorded.

Though endoscopic laser surgery remains the mainstay of treatment of recurrent respiratory papillomatosis, our study shows that in the absence of such a facility e.g. in health institutions of developing countries like Nigeria, the conventional endoscopic surgical excision of this lesion can be done with minimal or no complications and that tracheostomy when needed to relieve upper respiratory obstruction is advocated in the management of these patients.

Acknowledgement

The authors are grateful to Dr. L. Chirdan of the Pediatric Surgery Unit of the Department of Surgery, Jos University Teaching Hospital for allowing the use of his patient for this study and Mr. Miri of the Medical Records Department of the Jos University Teaching Hospital for his assistance in the retrieval of patients’ case notes.

References

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  4. Kimberlin DW, Malis DJ. Juvenile onset recurrent respiratory papillomatosis: possibilities for successful antiviral therapy. Antiviral Res. 2000 Feb; 45(2): 83-93.
  5. Sakoupolos A, Kesler KA, Weisberger EC, Turpentine MW, Conces DJ Jr. Surgical management of pulmonary carcinoma secondary to recurrent respiratory papillomatosis. Ann Thorac Surg. 1995 Dec; 60(6): 1806-7.
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  8. Rosen CA, Woodsen GE, Thompson JW, Hengesteg AP, Bradlow HL. Preliminary results of the use of indole-3-carbinol for recurrent respiratory papillomatosis. Otolaryngol Head Neck Surg. 1998 Jun; 118(6): 810-5.
  9. Pransky SM, Magit AE, Kearns DB, Kang DR, Duncan NO. Intalesional cidofovir for recurrent respiratory papillomatosis in children. Arch Otolaryngol Head Neck Surg.1999 Oct; 125(10): 1143-8.
  10. Leventhal BG, Kashima HK, Mounts P, Thurmond L, Chapman S, Buckley S et al. Long-term response of recurrent respiratory papillomatosis to treatment with lymphoblastoid interferon alfa-N1. Papilloma study group. NEJM. 1991 Aug; 352(9): 613-7.
  11. Shapiro AM, Rimell FL, Shoemaker D, Pou A, Stool AE. Tracheotomy in children with juvenile-onset recurrent respiratory papillomatosis: the Children’s Hospital of Pittsburgh experience. Ann Otol Rhinol Laryngol. 1996 Jan; 105(1): 1-5.
  12. Cole RR, Myer CM 3rd, Cotton RT. Tracheotomy in children with recurrent respiratory papillomatosis. Head Neck. 1989 May-Jun; 11(3): 226-30.
  13. Perkins JA, Inglis AF Jr, Richardson MA. Iatrogenic airway stenosis with recurrent respiratory papillomatosis. Arch Otolaryngol Head Neck Surg. 1998 Mar; 124(3): 281-7.
  14. Preuss SF, Klussmann JP, Jungehulsing M, Eckel HE, Guntinas-Lichius O, Damm M.. Long term results of surgical treatment for recurrent respiratory papillomatosis. Acta Otolaryngol. 2007 Nov; 127(11): 1196-201.
  15. Nwaorgu OG, Bakari AA, Onakoya PA, Ayodele KJ. Recurrent respiratory Papillomatosis in Ibadan, Nigeria. Niger J Med. 2004 Jul-Sep; 13(3): 235-8.

© 2008 East and Central African Journal of Surgery


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