Abstract

Human papilloma virus has been implicated as the cause of juvenile-onset recurrent respiratory papillomatosis in first-born children. Thus, cesarean section is strongly recommended by obstetricians to avoid direct contact with papilloma in an infected birth canal. We report a parturient with silent hypopharyngeal papilloma, which was associated with severe clinical problems at the induction of general anesthesia. The anesthesiologist considered general anesthesia for this case because of disseminated skin warts and secondary pus across the patient's body. Although the patient's breathing was smooth before starting general anesthesia, it was difficult to maintain positive pressure ventilation despite administration of a muscle relaxant. High air-way resistance without chest wall motion was noted, despite the insertion of an oral airway. Therefore, direct laryngoscopy was immediately performed and an obstructing mass was found. This mass acted as a check valve during positive ventilation. Our report should remind clinicians that human papilloma virus infection, although benign, can be disastrous in certain circumstances, as in our case where it resulted in airway obstruction and distal spread during cesarean section. Its presence necessitated preoperative laryngoscopic evaluation and aggressive treatment.

Keywords

airway obstruction; papillomavirus infections; parturients;

1. Introduction

Human papilloma virus (HPV) is often associated with diseases of the anogenital region and upper aerodigestive tract in children and adults. The incidence varies from 13% to 46% depending on the population studied and the methods used.^1,2 It was reported that 83.6% of patients with HPV infection are ≤ 40 years old, and 38.7% were aged 21−30 years old.³⁻⁵ Approximately 29% and 2.4% of women were reported to have HPV infection in the cervix and oral cavity, respectively.³ At delivery, 68% of affected women had an oncogenic HPV-type infection in the cervix.³ Here, we report a patient with HPV infection, but with a different presentation and different consequential damage. The patient was a parturient with low-risk HPV infection who suffered from recurrent genital warts and had a silent growth of a bulky papilloma with stalk inside the pharynx, without symptoms of hoarseness, stridor or breathing difficulties, during the course of her pregnancy. During induction of general anesthesia for cesarean section, the patient was found to have near-total airway obstruction. We present this case report to remind anesthesiologists that patients with adultonset recurrent respiratory papillomatosis (RRP) who are being scheduled for cesarean section should undergo preoperative airway evaluation by an otolaryngologist.

2. Case Report

 A 27-year-old pregnant woman, gravida 2, para 0, weighing 62 kg and with a body mass index of 24.2 kg/m2, was scheduled for an emergency cesarean section to prevent juvenile-onset RRP in the newborn. Reviewing the patient’s medical history, the patient had undergone laser therapy for perineum condyloma accuminatum in the second trimester of pregnancy at a local medical institution. The post-laser course was uneventful. The patient had also received regular check-ups during the course of gestation. The warts disseminated graduallyn over the patient’s whole body, and recurrent condyloma accuminatum around the perineum and mouth worsened.

  At week 37 of pregnancy, because of rupture of the membrane with active recurrent perineum condyloma infection, the patient was transferred from the local medical institution to Chang Gung Memorial Hospital and an emergency cesarean section was recommended by the obstetrician in charge. In formed consent was obtained and the patient’s preoperative anesthetic risks were evaluated. The patient’s vital signs were stable without any coexisting pregnancy-related high-risk disease. The patient’s breathing pattern was smooth and pulse oximetric saturation was 100%. The anesthesiologist in charge considered general anesthesia because of the skin infections covering the patient’s body with secondary pus formation. Although papillomas were present around the angles of the mouth, the patient could open her mouth freely and airway assessment was Mallampati Classification Class II. Evaluation of the distance between the chin and hyoid bone suggested that the airway was not difficult. Then, 0.4 mg atropine was intravenously administered for premedication to avoid excessive saliva secretion. The patient had fasted for 8 hours before the induction of anesthesia, and nasogastric tube decompression and antacid administration were considered unnecessary.

  Before inducing general anesthesia, preoxygenation with pure oxygen was given for more than 3 minutes, during which disinfection and surgical drapes were applied by the obstetrician. General anesthesia was induced with 1.5 mg/kg ketamine (100 mg), 1 mg/kg 1% propofol (60 mg) and 1 mg/kg rocuronium (60 mg). After 90 seconds of ventilation arrest with a cricoid pressure maneuver, positive mask ventilation was started. However, high airway resistance
and static chest wall motion, even with the placement of an oral airway, were noted. Therefore, laryngoscopy was immediately performed and a sizable mass obstructing the airway was found. Since the mass could be pushed aside by the intubating white stylet, there was room for tracheal intubation. After successful intubation, cesarean delivery began and anesthesia was maintained with 0.6% isoflurane and N2O in 33% O2 (N2O:O2 = 3:1). Pulse oximetric saturation remained at 100%. A 2800-g female baby, with Apgar scores of 9 and 10 at 1 and 5 minutes, respectively, was delivered.

  After completing the cesarean section, emergency consultation with an otolaryngologist was requested to evaluate the patient’s laryngeal problem. Fiberoptic bronchoscopy examination was done to check for any dislodgment of the papilloma. The papillomatous tumor covered the arytenoids cartilage, supraglottis and tongue base, but the trachea, carina and both bronchi were clear and normal, without any papillomatic growth (Figure 1). After obtaining informed consent from the patient’s husband, the otolaryngologist performed a biopsy for histopathological confirmation and viral typing. Human immunodeficiency viral antibody (HIV Ab) screening was also done. Because an accurate diagnosis of the disease entity was not possible at the time of biopsy and the possibility of malignancy could not be excluded, the consulting otolaryngologist planned no further aggressive ablation or total excision of the tumor unless the papilloma was likely to create a difficult airway.

  Weaning from intubation was then planned. The smallest-size laser-safe endotracheal tube with an inner diameter (ID) of 4.5 mm (Laser-Flex; Mallinckrodt Inc., Hazelwood, MO, USA) was prepared at hand should re-intubation for laser ablation be necessary to secure the airway. The otolaryngologist feared that tracheostomy would increase the spread of the papilloma down to the trachea, the bronchi and the lungs. Emergency tracheostomy was considered as a last resort if the upper airway obstruction could not be resolved by any other means. The patient was extubated after completely recovering from general anesthesia and regaining spontaneous breathing. Symptoms including dysphagia, hoarseness, orthopnea, stridor and the use of accessory muscles of respiration were closely monitored, the absence of which serve as hallmarks of a patent airway. Fortunately, the patient could breathe smoothly without any sign of upper airway obstruction. She was kept in a semi-sitting position during her stay in the postoperative recovery room. A postoperative visit by the otolaryngologist was also arranged. The patient underwent laser therapy for the papillomatous growth 2 months later.

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Figure 1 Visualization of the human papillomavirus infection under rigid bronchoscopy. The papillomatous tumor can be found over the arytenoids cartilage, supraglottis and tongue base.

3. Discussion

The role of cesarean delivery to prevent juvenileonset RRP at birth is controversial. Most obstetricians would consider cesarean delivery for young women with visible condyloma who are giving birth to their first child. Although this is not the first case of a parturient with condyloma accuminatum involving the pharyngeal area, it appears to be the first report of a parturient with a silent hypopharyngeal papilloma without respiratory obstruction during spontaneous breathing but which became apparent during positive ventilation with a face mask. The otolaryngologist also performed HIV Ab to exclude serious autoimmune disease, which was negative for immune deficiencies. The reasons for this clinical presentation might be due to the following features: (1) sexual behavior, poor personal hygiene and health care; and (2) hormonal changes associated with the second trimester of pregnancy together with RRP may increase the rate of papilloma growth and result in disease recurrence after a period of remission.⁶

  HPV, the pathogen associated with perineum condyloma accuminatum, is now recognized to play a role in the pathogenesis of a subset of head and neck squamous cell carcimonas.⁷⁻¹⁰ HPVs are DNA viruses with a specific tropism for squamous epithelia, and more than 120 different HPV types have been isolated to date.^7,8 HPVs can be classified into two groups based on clinical presentation. Low-risk HPVs, such as HPV-6 and -11, induce benign hyperproliferation of the epithelium resulting in papillomas or warts.⁷ In contrast, the high-risk HPVs such as HPV-16, -18, -31, -33 and -35 are strongly associated with cervical cancer.⁷ The prototypic highrisk types, HPV-16 and -18, can transform epithelial cells derived from the genital and upper respiratory tracts.^11,12 Pathologic analysis of our case showed
benign papillomatosis, acanthosis and parakeratosis of the squamous epithelium, and the presence of koilocytotic cells. Polymerase chain reaction-based methods were used to detect and genotype the HPVs, and were negative for prototypic HPV-16, -18 and -33. Thus, we are not certain as to which of the prototypes was present in this case. However, the clinical presentations and the benign pathological report prompt us to favor low-risk HPV infection.

  In this case, we could not check the subglottic area after inserting the endotracheal tube. According to the patient’s medical history and her husband’s description, no symptoms or signs of inspiratory stridor or shortness of breath had bothered the patient during the gestational course or before inducing anesthesia for the cesarean delivery. The airway ob struction in this case was due to a floating pharyngeal papilloma with a stalk, which acted as a check valve and completely obstructed the laryngeal opening during positive ventilation. Unless the airway obstruction cannot be resolved by any other means, emergency tracheostomy should not be considered to avoid distal spread of the papilloma down to the trachea and the lungs. Modified Howard Kelly dilating forceps with a Shiley endotracheal tube with an ID of 6.0 mm was prepared for emergency tracheostomy to rapidly secure the airway. Cricothyrotomy or other surgical airway procedures may also be appropriate in such cases. The use of dexamethasone was not considered for fear of disseminating the viral infection. Racemic epinephrine was prepared in case stridor occurred.

  Weaning from intubation was then planned when the patient’s muscle power had recovered. The smallest possible laser-safe endotracheal tube with an ID of 4.5 mm (Laser-Flex; Mallinckrodt Inc.) was prepared to replace the Shiley endotracheal tube to secure the airway if emergency re-intubation was necessary for laser ablation. Reducing the size of papilloma to create a patent airway is the main goal of emergency removal. Patients with RRP will need to undergo papillomatous resection several times to avoid laryngeal trauma or other serious complications.

  The patient was finally extubated and was able to breathe spontaneously, without any signs of upper airway obstruction. She was continuously followedup at our otolaryngology department. According to Chinese traditions, the parturient should have a month of postpartum rehabilitation. The otolaryngologist scheduled a 2-month follow-up to check for any spontaneous regression. The standard treatment for RRP is surgery. The most commonly used techniques are carbon dioxide laser therapy, cold instrumentation, and the microdebrider. Meanwhile, up to 10% of patients with RRP require adjuvant medical therapy, and intralesional injection of the nucleoside analog cidofovir is a favorable treatment option for severe laryngeal papillomatosis.^13,14 Our patient was finally treated by laser therapy and continued follow-up at our otolaryngology department.

  In conclusion, we believe this report should remind clinicians that HPV can infect different sites and may manifest with different clinical presentations because of its specific tropism for squamous epithelium. Parturients with severe recurrent perineum condyloma accuminatum associated with disseminated skin infection should visit an otolaryngology clinic for detailed hypopharyngeal examination before cesarean delivery. The upper airway, particularly the pharyngeal area and the laryngeal structures, should be carefully assessed by endoscopy. The method of intubation will depend on the severity and extent of the papillomas. Awake fiberoptic bronchoscopy intubation with spontaneous breathing is the preferred technique to avoid unnecessary airway occlusion with positive mask ventilation. Although the lesions of HPV infection are often benign, they can cause severe outcomes, such as airway obstruction, distal airway spread and malignant transformation, requiring great effort to avoid such events.