Juvenile Laryngeal Papillomatosis in Benin: Epidemiological, Diagnostic, Therapeutic and Evolutionary Aspects

Laryngeal papillomatosis is a benign squamous papillary tumor proliferation developed at the expense of the laryngeal mucosa in 90% of cases [1]. Broadly observed in children, though relatively rare it is the most common benign tumors of the larynx. Prevalence of the juvenile laryngeal papillomatosis is 0.7 to 3 [2,3]. The pathogenesis is poorly known evoking intricate theories that are caused by hormones, viral and traumatic. The latter is the only one to date that allows the isolation of the human papillomavirus (HPV) type HPV6 and HPV11. Currently, there is no cure which makes treatment difficult. The clinical course is unpredictable: evolutionary pursuit with a risk of asphyxiation and death, often enamelled healing recurrence with or without an extension to various parts of the respiratory tree. The degeneration is possible but rare and occurs most often in the adulthood [4]. The aim of this study was to describe the epidemiological profile of laryngeal papillomatosis, to identify diagnostic aspects, and to determine the evolutionary aspects after treatment.

This is a retrospective study we conducted based on patients’ medical record in ear nose throat department at Hubert Koutoukou Maga National Teaching Hospital of Cotonou from May 1995 to April 2011. The study covered patients with ages ranging between 0 and 17 year old and for whom the diagnosis of laryngeal papillomatosis was done based on anamnestic, clinical and paraclinical arguments. The examination indicated firstly a chronic dysphonia evolving for more than three weeks before then secondarily a dyspnea, and at indirect or direct laryngoscopy clusters blackberrys aspect in supraglottic, glottic or glotto-subglottic stage. We have received some patients at dyspnea stages as classified by Pineau and Chevalier Jackson (Table 1).

A rescue tracheotomy with an operation of a cannula was essential when the subject had a stage 3 dyspnoea of this classification. All patients underwent surgical treatment of papillomas with forcep peeling on bi-weekly average. The pathological examination after biopsy confirmed the diagnosis of laryngeal papillomatosis. This was followed by a painting with mitomycin site where the lesion was exuberant or recurrent. When the laryngeal was absorptive then the decannulation was made after weaning by gradual light reduction of the material in use. The parameters studied were frequency, sex, age, consultation period, reasons for consultation with or without associated signs, the location of lesions, treatment and evolution.

Table 1: Classification of Pineau and Chevalier Jackson.

Over the 16 years study period, 18 patients were consulted with a rate of 1.12 cases per year. The sample included of 10 males/18 with a frequency of 0.55 and an average age of 6.95 years (21 months-16 years). 14 children were in school or at age to be.

Table 2: Distribution of patients by endoscopic examination.

Clinically, the consultation period was 1.17 years (1 week-5 years). 1/3 consulted for dysphonia drawling, another 1/3 dysphonia association and dyspnea without signs of decompensation and the remaining one 1/3 for dyspnea stage 3. Table 2 summarizes the distribution of patients according to the results of the endoscopic review. Clamp peeling with an average of 4.6 (1-23) was treatment opted for in our series. Mitomycin was applied 1 to 4 times by brushing after surgery in 3 patients. In an emergency, a tracheotomy rescue was necessary in 12 cases and in total during the study period, this act was performed 15 times with an average of 1.25 (1-3). Pathological examination of samples has proved laryngeal papillomatosis. 04 patients underwent additional consultations in emergency way; in a case there were 2 readmissions and 1 readmission in 3 cases.

The evolution was assessed in 14 patients over an average of 14.85 months (3 months-175 months) on the quality of the voice, breathing and the occurrence of complications (Table 3). Ten (10) patients had both normal breathing and voice. We noted the impossibility of decanulation in 1 case and in 3 cases the patients had suprasternal draw supine and intermittent dysphonia after decanulation.

Table 3: Distribution of patients according to the complications. All 18 patients were peeled -12 underwent emergency tracheotomy and wore a canula.

We consulted 18 patients over 16 years with a rate of 1.12 cases per year. The sample included 10 males with a frequency of 0.55 in favour of males and an average age of 6.95 years. The consultation period was 1.17 years (1 week-5 years). 1/3 consulted for dysphonia drawling, another 1/3 dysphonia association and dyspnea without signs of decompensation and the remaining on third for dyspnea stage 3. Clamp peeling with a mean of 4.6 (1-23) was the treatment opted for in our series. Mitomycin was applied 1 to 4 times by brushing after surgery in 3 patients. In an emergency, a tracheotomy rescue was necessary in 12 cases and in total during the study period, this act was performed 15 times with an average of 1.25 (1-3). The evolution was assessed in 14 patients over an average of 14.85 months (3 months-175 months) on the quality of the voice, breathing and the occurrence of complications.

Epidemiologically, laryngeal papillomatosis is a benign and occasional disease which variable incidence is 0.2 to 0.7 cases per 100,000 population [5,6]. The number of new cases per year of the juvenile laryngeal papillomatosis is 0.7 to 3 [3] while it was 1.12 in our series. If the male predominance is for Ndiaye [3], laryngeal papillomatosis is more common in children and regardless of gender with an average age ranging from 6 to 10 years [7,8].
The diagnosis is often delayed because of the long use of consultation up to 5 years. Indeed, dysphonia first sign of laryngeal papillomatosis may be laryngitis or an asthmatics status differential diagnosis [9,10]. This is not the case in the series of Traissac [11], where 90% of cases were consulted at the stage of dysphonia. In our series, patients were consulted in 2/3 of cases at the stage of dyspnea. This is a laryngeal dyspnea faster or slower type inspiratory bradypnea with draw and wheezing.

The review to nasofibroscopy or direct laryngoscopy under A/G achieves clinical assessment by objectifying in a mobile larynx lesions pink or greyish as grains, clusters, sometimes florid, obstructive or not. The lesions extended to the 3 floors of the larynx and oropharynx in one case. As for the tracheobronchial tree, it has not been explored due to limited technical facilities in our work environment.

The pathological examination was performed in all cases to confirm the benign lesions.
Therapeutically, the delay in consultation, sometimes multistage extension of the larynx and the obstructive nature of lesions required a tracheotomy first in 12/18 cases. This gesture would be saving an aggravating factor as it would favor the extension of lesions throughout the respiratory tract [1,12,13]. Peeling was the only surgical treatment for all patients because the gesture is simple, easily accessible and inexpensive.
In evolutionarily, 14/18 patients were reviewed. Recurrences were frequent with the need to resume peelings every 2 weeks. Apart from these controls set, 4 patients were received emergency in an array of dyspnea stage 3. During the monitoring period, the average peelings was 4.6 (1-23). The complications were related to emergency tracheotomy and residence of the cannula. The facial-cervical-thoracic emphysema can be explained by tight stitches on both sides of the incision instead of tracheotomy or too small tracheostomy tube allowing fuser upstream and downstream of the air. As for tracheal mucosa, it produces in contact with the tracheostomy tube of secretions which at long term gets over infected and goes down to the lower airways. Similarly mini friction of the cannula can create irritation, then a oeso-tracheal fistula and a granuloma. The sequelae of peeling are firstly a bad voice quality probably due to a lack of confrontation of the vocal cords after fibrosis or poor healing of the laryngeal mucosa and also observed the occurrence of dysplasia one patient by the mucosa redesign.

Laryngeal papillomatosis is a common pathology of the child. It is fairly easy to diagnose but poses therapeutic difficulties. The development is toward recurrence and education disruption of the child. The functional and vital prognosis can be involved, hence an early consultation is needed.