Abstract
Objective
The goal of this study is to describe a unique finding of ulcerative lesions of the larynx in two pediatric patients presenting with prolonged acute laryngotracheitis and compare to previously described reports to determine the typical clinical picture, need for intervention, and management model.
Methods
We present two cases of ulcerative lesions of the larynx in immunocompetent children, one with PCR positive HSV, which presented as severe croup requiring intensive care unit admission. Literature review was completed to assess for current knowledge of this entity. Our cases are discussed in the context of previously reported cases of HSV laryngotracheitis. Descriptive analysis was completed focusing on presentation, physical exam findings, treatment, length of therapy, and outcomes.
Results
Literature review uncovered six case reports including 10 individual cases of prolonged croup with findings of HSV laryngitis and one retrospective review describing 15 cases of prolonged croup found to be caused by ulcerative laryngitis. All patients underwent direct laryngoscopy and bronchoscopy for evaluation. Analysis was completed comparing the studies to our patients with significant findings including high intubation rate of 77%, ulcerative stomatitis in 63%, and treatment with antiviral medication directed at HSV in 85% with improvement in symptoms.
Conclusion
It is important to consider HSV as a possible pathogen in cases of prolonged or atypical croup. Laryngoscopy should be used for diagnostic intervention and identification of ulcerative lesions. Stomatitis may be an indication for earlier direct inspection. Treatment with anti-viral therapy and with discontinuation or taper of steroid is suggested.
1
Introduction
Acute laryngotracheitis, or croup, is a common childhood illness, occurring in about 3%–5% of children. It is the most common form of infectious upper airway obstruction in children . Children typically present between the ages of 6 months and 3 years, with peak incidence 18–24 months . Parainfluenza, influenza, adenovirus, and respiratory syncytial virus most commonly cause laryngotracheitis . Presenting features include low grade fever, inspiratory stridor, hoarseness, and a barking cough. The presence of biphasic stridor, retractions, high respiratory rate, or oxygen desaturations indicates severe airway obstruction . Typically, symptoms peak in severity at 3 to 5 days with no specific treatments required at home. Management strategies for viral croup have included glucocorticoids, racemic epinephrine, and heliox. Acute laryngotracheitis is considered atypical if lasting more than 7 days or if it does not respond to appropriate treatments . HSV has been reported as a rare finding in immunocompetent children presenting with atypical ulcerative acute laryngotracheitis.
In the past year at the Children’s Hospital of Wisconsin (CHW), the Otolaryngology Department has been consulted on two children with atypical croup who were found to have ulcerative lesions of the larynx and ultimately treated for HSV laryngotracheitis. Approval was granted from the CHW Institutional Review Board for the study of this case series.
2
Case reports
2.1
Case 1
A 13-month-old boy was transferred to CHW with an eight day history of croup. He was evaluated on day one at an outside hospital emergency department (ED) and given one dose of steroids. Three days later he returned to the ED because he was not improving. At that time he was treated with amoxicillin for a suspected ear infection. At seven days his “barking” cough persisted and he was again brought to the hospital where he was admitted and started on IV steroids and racemic epinephrine. His respiratory status continued to decline and he was transferred to CHW and admitted to the intensive care unit (ICU). At this time he had inspiratory stridor and increased work of breathing with retractions. He was treated with heliox with improvement in stridor and retractions. Otolaryngology was consulted after he was unable to be weaned from the heliox at hospital day two. He had no history of intubation and only one previous episode of croup, which was very mild. He was treated with IV dexamethasone. Viral cultures were negative for adenovirus, influenza A, B, and parainfluenza 1, 2, 3. He remained on heliox intermittently for the first six days at CHW.
Direct laryngoscopy with rigid bronchoscopy was performed at hospital day five with findings of ulceration of the supraglottis and glottis with subglottic edema ( Fig. 1 ). He was brought back to the ICU without intubation. Acyclovir treatment was empirically started and steroid taper was initiated. Augmentin was started for concomitant bacterial infection for 10-day course. PCR from swab of oropharynx was positive for HSV-1. Infectious disease (ID) consultation then recommended a 5-day course of valacyclovir. Respiratory status improved significantly with initiation of antiviral with improvement in work of breathing and stridor. He was discharged five days after bronchoscopy, but readmitted the next day with increased stridor and retractions. He was noted to have oral thrush and diaper rash on readmission to the ICU, which was treated with nystatin. Valacyclovir and Augmentin were continued. He did not require heliox or oxygen on readmission. He continued to have inspiratory stridor and mild retractions.
He returned to the operating room (OR) 13 days after the first bronchoscopy for re-evaluation due to persistence of stridor and retractions. Healing ulcers were seen as well as 30% narrowing of subglottis. The dose of valacyclovir was increased and ID recommended an additional three-week course (total of ~ 5 weeks). His respiratory status improved to the point of not experiencing retractions or stridor at rest, only with agitation. He was deemed stable for discharge home after a total of 20 hospital days. On follow up one month later, and again 8 months later, he had no further stridor or symptoms of respiratory distress.
2.2
Case 2
A 16-month-old boy presented to the ED after two days of fevers, increased work of breathing, ulcerative stomatitis, and ulcerative lesions on his chest. Stridor was present with agitation. He was admitted to the hospital with diagnosis of croup and started on methylprednisolone and racemic epinephrine. He continued to have increased work of breathing with retractions, without desaturations, and was transferred to the ICU. There he was started on heliox and continued to receive racemic epinephrine and methylprednisolone. He had intermittent fevers. Otolaryngology was then consulted at hospital day two for evaluation due to continued increased work of breathing. Patient was taken to the OR for direct laryngoscopy and rigid bronchoscopy. He was found to have multiple oral ulcerations, glottic edema causing 95% obstruction, and ulcerations present to supraglottis and glottis ( Fig. 2 ). He had a normal appearing trachea and bronchi. 3.0 endotracheal tube was placed secondary to significant glottic edema. He was started on empiric acyclovir after operative airway evaluation with plan for 14-day course. He was also started on Unasyn for concomitant bacterial infection for 10 days. He was continued on Decadron while intubated. Extubation occurred 3 days later. HSV PCR was performed on blood and lesion, both returned negative. Also reported negative were cultures for adenovirus, parainfluenza virus 1, 2, 3, enterovirus, and CMV. Acyclovir was continued for suspected HSV infection. He was transferred back to the floor post extubation as he was in less respiratory distress, though stridor remained with agitation. Steroid taper was initiated. Patient was stable for discharge home at hospital day nine. Repeat bronchoscopy as an outpatient demonstrated no sequela of disease.
