儿童复发性呼吸道乳头状瘤病临床研究

OBJECTIVE: To study the clinical behavior of juvenile-onset recurrent respiratory papillomatosis in order to find some factors correlated to the development of this disease, and to sum up the significance and experience of CO2 laser surgery. METHOD: Sixty patients with juvenile-onset recurrent respiratory papillomatosis from September 1995 to December 1998 were retrospectively analyzed. RESULTS: The age of onset in 50 cases (83.3%) was below 4 years, and the peak-age was 2 years. The rates of recurrence were 72.0% and 45.7% (chi 2 = 4.71, P < 0.05) below and over 2 years, respectively. The rates of aggressive disease were 88.0% and 54.3% (chi 2 = 7.66, P < 0.01) below and over 2 years, respectively. The predominant sites of the disease were the vocal cords, the false vocal cords, the laryngeal ventricle, the laryngeal surface of the epiglottis and the subglottic region. Tracheostomy induced the development of tracheal papilloma, therefore should be avoided as possible. Laryngeal papilloma might be divided into four types on the basis of the growth manner and surface form corresponding to clinical behaviors. Five patients were followed-up for 1.5 years without recurrence, 18 patients had fewer recurrences following treatment, 33 patients were under treatment, and 3 patients died. Nineteen patients lost follow-up. The major complications included laryngeal and tracheal stenosis. CONCLUSION: Clinical behaviors of juvenile-onset recurrent respiratory papillomatosis were relevant to the age, growth form and tracheotomy. CO2 laser was an ideal instrument for ablation of the laryngeal papillomas with the following advantages: simple management, less bleeding, preservation of laryngeal structure and avoidance of tracheostomy.     

Treatment of recurrent respiratory papillomatosis in children with the microdebrider

The mainstay of treatment of children with recurrent respiratory papillomatosis has been CO2 laser laryngoscopy. Powered microdebriders allow precise removal of laryngeal lesions without thermal damage. The microdebrider may reduce costs by reducing operative time and limiting the need for expensive laser-related equipment. To compare the use of the microdebrider with that of the CO2 laser in children undergoing surgery for recurrent respiratory papillomatosis, we retrospectively reviewed the charts of 18 patients from 2 institutions covering a treatment period between December 1998 and October 2001. These patients were initially treated with the CO2 laser, but were switched to microdebrider resection during the review period. We recorded the number of procedures, the technique(s) of resection used, and the operative time for each procedure. We identified 18 patients, 11 male and 7 female, ages 18 months to 14 years. A total of 127 procedures were performed with the CO2 laser, and a total of 50 procedures were performed with the microdebrider. The mean number of procedures per patient was 10 (range, 2 to 17 procedures). The mean surgical time per procedure using the CO2 laser was 59.2 minutes (range, 8 to 130 minutes). The mean time per procedure with the microdebrider was 32.4 minutes (range, 12 to 47 minutes). This reduction in operative time was clinically and statistically significant (p = .0001, unpaired Student's t-test). We concluded that microdebrider resection of laryngeal papilloma in children allowed more rapid surgery with potentially reduced treatment costs. Other advantages of this technique include precise excision without thermal injury. Our report details useful refinements of technique with the microdebrider.     

Airway reconstruction in children with recurrent respiratory papillomatosis

OBJECTIVE: To determine if children with recurrent respiratory papillomatosis were at increased risk of complications due to their disease following major airway reconstruction. METHODS: We retrospectively reviewed our airway surgery database and the medical records of all children diagnosed with recurrent respiratory papillomatosis who were evaluated at Cincinnati Children's Hospital Medical Center between January 1998 and August 2003. All patients with active airway papillomas or a history of recurrent respiratory papillomatosis who underwent open airway reconstruction at our institution were included in the study. RESULTS: Seven children with recurrent respiratory papillomatosis underwent major airway reconstruction. Six children had tracheotomies and five are now decannulated following their airway reconstruction. Five children had active papillomas at the time of surgery and none had significant worsening of their papillomas following their procedures. Two patients in remission underwent airway reconstruction without recurrence of their papillomas. CONCLUSIONS: Major airway reconstruction can be safely performed in children with recurrent respiratory papillomatosis.     

Microdebrider removal of tracheal papilloma via tracheostomy in the child with an obliterated larynx

In children, recurrent respiratory papillomatosis is the most common benign neoplasm of the airway. The disease frequently involves the larynx and may spread to extralaryngeal sites. Use of a microdebrider has been suggested as a safe and low-cost technique which reduces operating time, compared with laser removal of laryngeal lesions. We describe a technique for using a microdebrider to remove tracheal papillomas when the larynx is obliterated with the disease.     

Intralesional cidofovir for recurrent respiratory papillomatosis in children.

OBJECTIVE: To assess the potential benefit of intralesional administration of cidofovir, an acyclic nucleoside phosphonate with activity against several DNA viruses, for treating severe respiratory papillomas in pediatric patients. DESIGN: Prospective case series. SETTING: Tertiary care children's hospitals. PATIENTS: Five pediatric patients with severe recurrent respiratory papillomatosis requiring laryngoscopy with carbon dioxide laser therapy more frequently than once a month to maintain airway patency. Each patient underwent between 12 and 33 laryngoscopies with laser treatment prior to being injected with cidofovir. INTERVENTION: Microsuspension laryngoscopy with intralesional injection of cidofovir (Vistide) in conjunction with mechanical debulking and carbon dioxide laser of papillomas. MAIN OUTCOME MEASURE: Papilloma stage at time of serial laryngoscopies. RESULTS: One patient was disease free and 3 patients demonstrated a dramatic response to adjuvant therapy with cidofovir at the 9-month follow-up visit after the last injection of cidofovir. One patient showed an improvement in papilloma stage that was possibly related to concurrent therapy with interferon. CONCLUSIONS: Intralesional injection of cidofovir seems to be of benefit in the treatment of severe respiratory papillomatosis in pediatric patients. Larger prospective studies with longer follow-up will be required before cidofovir can be considered an accepted means of managing this difficult disease.     

Soft-tissue complications of laser surgery for recurrent respiratory papillomatosis

Twenty-two patients with recurrent respiratory papillomatosis underwent 105 carbon dioxide (CO2) laser microlaryngoscopic and tracheobronchoscopic operations from July 1986 through February 1990. All soft-tissue complications, whether intraoperative or delayed secondary to laser surgery, were retrospectively analyzed. The intraoperative laser-related soft-tissue complication rate was zero. Two of the 22 patients acquired slight unilateral true vocal cord scar tissue and 1 patient developed a small posterior laryngeal web. The delayed soft-tissue complication rate was 13.6%, which compares favorably with published reports of 28.7% and 45%. This low complication rate has resulted from the selection of appropriate CO2 laser emission parameters and the use of the microspot micromanipulator, which help minimize lateral and/or deep thermal damage at the site of laser impact.     

Endotracheal tube obstruction: a rare complication in laser ablation of recurrent laryngeal papillomas

During the past 25 years, use of the carbon dioxide (CO2) laser has been accepted as the most appropriate surgical method for the treatment of recurrent laryngeal papillomatosis. Although CO2 laser technology and surgical techniques continue to improve, laser-related complications still occur. We describe a case of a very rare complication--to our knowledge, only the second such case reported in the literature--in which an endotracheal tube was almost completely obstructed by a piece of papillomatous tissue during CO2 laser microlaryngoscopy for the treatment of recurrent laryngeal papillomatosis.     

Pulmonary involvement in a case of juvenile-onset recurrent respiratory papillomatosis

Juvenile-onset recurrent respiratory papillomatosis is primarily a disease of children and adolescents. Although most patients experience spontaneous regression at a later age, some patients continue to be affected throughout their lives. We report the case of a 35-year-old woman with a 33-year history of juvenile-onset recurrent respiratory papillomatosis who developed pulmonary dissemination with malignant transformation. Malignant transformation is the most feared sequela of pulmonary dissemination, and it should be addressed aggressively. If treated promptly, the patient can enjoy prolonged survival. Computed tomography is superior to plain radiography for detecting and evaluating the extent of disease.     

Spontaneous respiration anesthesia for respiratory papillomatosis

Several anesthetic techniques for microlaryngeal laser surgery in children have been advocated. The objective of this study was to evaluate the operative conditions and safety of the spontaneous respiration anesthesia technique for carbon dioxide laser surgery in children with recurrent respiratory papillomatosis (RRP). The anesthetic and operative records of 60 pediatric patients with RRP who underwent microlaryngeal laser surgeries between 1991 and 1997 were reviewed. Papilloma location, severity of involvement, oxygen saturation, airway stability, need for intubation, duration of procedure, and complications were recorded. The patients underwent 744 surgical procedures. Two hundred two procedures were randomly studied (statistical power > or = 80%). The supraglottis and glottis were most commonly involved. Most of the papillomas were graded as moderate or severe disease. Complications occurred in 11 procedures. The surgical procedure was completed in all cases. No variable had predictive value regarding the outcome. Spontaneous respiration anesthesia is an efficient and relatively safe technique for microlaryngeal laser surgery in children with RRP.     

Complications of laser surgery for recurrent respiratory papillomatosis

During an 8-year period, 66 patients with recurrent respiratory papillomatosis underwent 890 laser microlaryngoscopy procedures. A retrospective analysis of immediate and delayed complications occurring with laser microlaryngoscopy in the treatment of these patients revealed that complications secondary to anesthetic technique and laser methodology were remarkably low (0.3%). Delayed complications of tissue injury (36% of pediatric patients and 17% of adult patients) were more frequent and included anterior glottic webbing, interarytenoid scarring and arytenoid fixation, vocal fold fibrosis, and generalized endolaryngeal glottic stenosis. All patients were evaluated by direct visualization and recording at the time of the operative procedure, as well as by review of intraoperative photographs. A smaller group of patients underwent analysis of voice recordings and laryngeal stroboscopy. The frequency and severity of tissue injury was higher in the patients with more severe disease who underwent multiple operative procedures.     

Complications of laser surgery for laryngeal papillomatosis.

Carbon dioxide laser surgery has become the treatment of choice for laryngeal papillomatosis. The purpose of this study was to determine the type, incidence, and severity of complications that occur with laser microlaryngoscopy for a disease that often requires multiple operations. Forty patients with laryngeal papillomatosis underwent a total of 222 carbon dioxide laser laryngoscopies over the 6 1/2-year period from June 1977 through December 1983. The results showed that 13 patients sustained a total of 23 separate complications. Intraoperative complications consisted of one episode of bilateral pneumothorax and one episode of cervical subcutaneous emphysema, both associated with the use of jet ventilation anesthesia, and one episode of a loosened tooth in a child with carious teeth. The delayed complications consisted of 10 patients with anterior laryngeal webbing, 2 patients with posterior webbing, 6 patients with laryngeal edema or fibrosis, and one episode each of prolonged dysphagia and tracheal foreign body. No airway fires occurred. Only 2 of 28 patients who had 5 or fewer laser laryngoscopies developed complications, but 11 or 12 patients undergoing 6 or more laser operations had complications. In summary, although the incidence of life threatening complications was low, the occurrence of minor complications such as small anterior glottic webs and persistent edema was relatively high, especially in those patients who required multiple laser laryngoscopies.     

Complications of treatment of recurrent laryngeal papillomatosis with the carbon dioxide laser in children.

A retrospective study of the complications of treatment with the carbon dioxide (CO2) laser of 17 patients diagnosed to be suffering from recurrent laryngeal papillomatosis is presented. No immediate complications occurred except one case of laryngospasm and failure to intubate during anaesthesia leading to hypoxic encephalopathy. Three patients were completely free from disease and complications. Another patient was free from laryngeal lesions but developed a papilloma in the right tonsillar pillar. Five other patients showed one or more multiple sites of involvement in addition to the larynx. Laryngeal scarring developed in ten patients. Six patients (35.29 per cent) developed scarring as anterior glottic webs while in two scarring (11.7 per cent) occurred as posterior glottic webs. One developed scarring in the supraglottic region. The remaining one had scarring in both the glottic and supraglottic regions. One patient developed tracheal scarring necessitating laryngo-tracheal separation. Two patients were psychologically disturbed during treatment requiring psychiatric consultation and therapy.     

Microdebrider versus CO2 laser removal of recurrent respiratory papillomas: a prospective analysis

OBJECTIVE: To compare postoperative patient discomfort, voice quality, and procedure time and cost for removal of recurrent respiratory papillomas using the microdebrider versus the CO2 laser. STUDY DESIGN: A randomized prospective study comparing children undergoing excision of recurrent respiratory papillomas by CO2 laser versus excision by microdebrider. METHODS: For the 6-month study, patients for whom removal of recurrent respiratory papillomas was indicated were randomly assigned by birth year to microdebrider or CO2 laser therapy. Disease severity was scored as the sum of ratings of 1+ (minimal), 2+ (moderate), or 3+ (severe) for involvement of 27 areas of the aerodigestive tract by direct microlaryngoscopy immediately before treatment. Parents scored patient discomfort and improvement in voice quality 24 hours after surgery, using a 5-point (0 = no pain; 4 = worst pain) and a 10-point (1 = minimal change; 10 = significant improvement) scale, respectively. RESULTS: Nineteen patients ranging in age from 2.5 to 20 years underwent 32 procedures in all. Groups did not differ significantly in age, sex, or severity of disease. For disease of equivalent severity, microdebrider treatment was associated with equivalent 24-hour-postoperative pain scores, greater improvement in voice quality, shorter procedure times, and lower overall procedure cost. CONCLUSIONS: Immediate postoperative results indicate that the microdebrider may be as safe as and, at some institutions, might be more cost-effective than the CO2 laser for removal of recurrent respiratory papillomas.     

儿童复发性呼吸道乳头状瘤病的联合治疗

目的：探讨儿童复发性呼吸道乳头状瘤病（JO-RRP）的治疗方法。方法：回顾性分析36例JO-RRP患儿的临床资料，针对病情分别采用支撑喉镜下激光切除术（27例）和喉裂开造口激光切除术（9例），对复发≥2次／年或（和）病变范围累及≥2个解剖亚区的28例患儿术后1周给予干扰素治疗。结果：全部患儿术后均无明显误咽，28例（77．8％）已拔除气管套管。并发症包括：声音嘶哑8例，喉狭窄6例，肺部感染3例。结论：支撑喉镜下激光切除术和喉裂开造口激光切除术是治疗JO-RRP的有效术式，术后联合干扰素治疗可抑制复发。     

小儿复发性呼吸道乳头状瘤治疗进展

小儿复发性呼吸道乳头状瘤是指发生在呼吸道上下的乳头状瘤病,是一种高度复发性和侵袭性的疾病。患儿可出现声音嘶哑症状,严重时有呼吸困难表现。乳头状瘤疾病常为HPV6和HPV11感染,可由于分娩时患儿经母亲产道感染乳头状瘤病毒所致。手术是治疗的主要的方法,但不能根治,往往因为多次手术给患儿和家属带来巨大的痛苦,故需要其他药物来辅助治疗。本文综述概括了目前各种治疗小儿乳头状瘤的方法。     

Recurrent respiratory papillomatosis associated with gastroesophageal reflux disease in children

The hallmark of gastroesophageal reflux disease (GERD) is an increased exposure of esophageal and laryngeal mucosa to gastric juice. This exposure can cause complications such as chronic laryngitis or chronic respiratory diseases. We report our experience in managing three pediatric patients with severe recurrent juvenile laryngeal papillomatosis (JLP) associated with GERD. All patients showed a high rate of recurrence requiring multiple laser surgeries. Systemic αinterferon therapy over a period of more than 1 year and photodynamic therapy with dihematoporphyrin produced no improvement. However, after therapy for GERD, the rate of recurrence of JLP decreased significantly. Although the course of respiratory papillomatosis is known to fluctuate, our findings suggest that gastroesophageal reflux may have a role in aggravating papillomatosis. Received: 22 September 1998 / Accepted: 12 November 1998     

A scheduled protocol for the treatment of juvenile recurrent respiratory papillomatosis with intralesional cidofovir

OBJECTIVE: To assess the efficacy of treating juvenile recurrent respiratory papillomas with intralesional cidofovir using a scheduled treatment protocol. DESIGN: Prospective case series. SETTING: Tertiary care academic medical center. PATIENTS: Of 5 pediatric patients with recurrent respiratory papillomas, 2 had severe recurrent papillomatosis requiring long-term therapy of laser ablations every 2 weeks prior to cidofovir treatments. The 3 other patients were newly diagnosed or had milder disease. INTERVENTION: Intralesional cidofovir (1 mg/kg) was administered during each scheduled visit. The first 4 treatments were at 2-week intervals (week 0, 2, 4, and 6). Subsequent treatment intervals were each increased by 1 week (treatments took place at week 9, 13, 18, 24, etc). Concomitant laser ablation was used only for bulky lesions. MAIN OUTCOME MEASURES: Papilloma stage and need for laser ablation at each scheduled visit. RESULTS: The mean follow-up time was 66 weeks. The mean (SD) papilloma stage decreased from 9.2 (5.5) at initial presentation to 3.4 (2.6) within 2 weeks of the first injection (P<.05), and continued to decrease for the remaining of the follow-up period. Papilloma stage 0 was achieved in 4 of the 5 patients. The need for laser ablation of papillomas also decreased within 4 weeks of treatment initiation (P<.05). At 9 weeks, no patient required laser therapy. One patient was removed from the protocol after 58 weeks. CONCLUSION: An intralesional treatment protocol with cidofovir and increasing intervals between scheduled treatment was successful the long-term management of juvenile respiratory papillomatosis.     

Surgical treatment and adjuvant therapies of recurrent respiratory papillomatosis

Recurrent respiratory papillomatosis or recurrent laryngeal papillomatosis is a disease of the larynx caused by human papilloma virus, characterized by verrucous epithelial lesions and usually recurring. In the literature there are several types of treatment, such as surgery to cold, laser and/or use of microdebrider, as of adjuvant therapies; all possible to decrease the permanent sequelae of the disease.ObjectiveTo review the literature regarding this disease with emphasis on surgical techniques and adjuvant therapies used today.MethodWe used the literature review, through surveys based electronic data in the public domain, to search for articles between 1992-2012, using keywords: papilloma, human pappiloma virus infection, larynx, therapeutic, papilloma virus vaccine.ResultsWe surveyed 357 articles, of which 49 were used as the basis for this review. Scientific studies indicate a reduction of relapse in most adjuvant therapeutic presented. However, the survey showed different methodologies and samples, which did not allow to compare the types of treatment and adjuvant therapies.ConclusionThe choice of surgical technique varies among studies, but there is a trend to use the microdebrider. The newer adjuvant therapies, such as cidofovir, quadrivalent vaccine against human papilloma virus and bevacizumab, require further studies.     

气管内窥镜在治疗小儿复发性气管内乳头状瘤中的应用

目的 :探讨小儿复发性喉乳头状瘤播散至气管内的治疗方法。方法 :利用气管内窥镜在全麻支撑喉镜下治疗小儿复发性气管内乳头状瘤 12例。结果 :12例中 ,3例随访 1年余未复发 ,已拔除气管套管 ;9例术后复发 ,但复发周期较既往应用气管镜手术明显延长 ,且病灶有逐渐局限、减少的趋势。结论 :应用气管内窥镜治疗气管内乳头状瘤 ,术野清楚 ,肿瘤摘除彻底 ,术后复发周期延长。