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Acute herpes simplex laryngotracheitis: Report of two pediatric cases and review of the literature
International Journal of Pediatric Otorhinolaryngology (2007) 71, 341—345
www.elsevier.com/locate/ijporl
CASE REPORT
Acute herpes simplex laryngotracheitis: Report of two pediatric cases and review of the literature Nitin Chauhan a,c, Joan L. Robinson a,b,c, Jennifer Guillemaud a,c, Hamdy El-Hakim a,c,* a
Pediatric Otolaryngology (Division of Pediatric Surgery), The Stollery Children’s Hospital, Edmonton, Canada b The Division of Pediatric Infectious Diseases, The Stollery Children’s Hospital, Edmonton, Canada c The Department of Pediatrics, The Stollery Children’s Hospital, Edmonton, Canada Received 28 June 2006; received in revised form 12 October 2006; accepted 15 October 2006
KEYWORDS Laryngitis; Herpes simplex virus; Atypical laryngitis; Laryngotracheobronchitis; Croup
Summary Herpes simplex virus (HSV) is a rare cause of laryngotracheitis (LT) with only 22 previously reported and confirmed pediatric cases in the literature. It is often associated with immune deficiency states and presents with a severe acute upper airway obstruction commonly requiring intensive care management and artificial ventilation. We present two cases of atypical laryngotracheitis in which HSV was found to be the causative pathogen subsequent to laryngoscopy and microbiologic investigations. While the first case was a previously well 8-month-old girl, the second was a 22-month-old immunosuppressed boy. One-third of the total confirmed cases in the English literature required intensive care management eliciting the setting as a pointer to the diagnosis. Laryngoscopy is a readily available tool for rapid diagnosis and controlled securing of the airway. This step may avoid controversial and potential counter-productive use of systemic steroids in these cases. # 2006 Elsevier Ireland Ltd. All rights reserved.
1. Introduction Acute laryngotracheitis (ALT) in the pediatric population is a relatively common clinical condition * Corresponding author at: Pediatric Otolaryngology, 2C3.57 Walter MacKenzie Health Science Centre, 8440-112 St., Edmonton, Alberta, Canada T6G 2B7. Tel.: +1 780 407 8629; fax: +1 780 407 2004. E-mail address: [email protected] (H. El-Hakim).
encountered most often by primary care physicians. Typical signs and symptoms include a sore throat, hoarse voice and harsh cough [1,2]. ALT is mostly viral in etiology and the pathogens most commonly responsible are the typical causative agents of croup–—parainfluenza virus 1, 2 and 3, respiratory syncytial virus (RSV), influenza virus A and B, human adenovirus and less commonly enteroviruses and measles virus [2]. The treatment of ALT is usually symptomatic and supportive, anticipating complete
0165-5876/$ — see front matter # 2006 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2006.10.012
342 resolution within a few days. Herpes simplex virus (HSV) is an uncommonly encountered etiologic agent [3—23]. It usually occurs in immuno-deficient patients, manifesting a particularly severe and rapidly progressive clinical course [4,22]. We present two cases and present a literature review on the topic.
2. Case reports 2.1. Case 1 The Pediatric Otolaryngology service was consulted for evaluation of a previously well 8-month-old girl presenting with severe respiratory distress to the Emergency Department. At the time of presentation, she was very irritable, aphonic and diaphoretic with biphasic stridor and evidence of labored breathing. In the preceding 11 days, the patient had been admitted to hospital twice with a similar clinical picture, and had been diagnosed with viral croup. During each encounter, she responded partially to treatment with systemic dexamethasone and inhaled racemic epinephrine, and was discharged home within 24 h of admission. Given the severity of her respiratory distress on this occasion, she was examined under general anesthesia. With the child spontaneously breathing, flexible and rigid airway examination revealed severe laryngeal inflammation with white, ulcerative lesions noted on the tongue, palate, throughout the larynx and on the pharyngeal walls (Fig. 1).
Fig. 1 A rigid endoscope view of the larynx and pharynx. Demonstrates multiple herpetic vesicles with surrounding erythema.
N. Chauhan et al. Laryngeal movement was normal. Also noted was an edematous subglottis and upper trachea, while the lower trachea and main stem bronchi were normal. Swabs from the lesions were submitted for bacterial and viral culture. The patient was then intubated nasally with a 3.5 mm endotracheal tube over a flexible bronchoscope and admitted to the Pediatric Intensive Care Unit (PICU). Direct fluorescent antibody tests and cultures performed on the lesions in the posterior pharynx and epiglottis were positive for HSV type 1. Thus, the patient was started on intravenous acyclovir therapy (30 mg/kg/day q 8 h for 10 days). The patient was screened for immunodeficiencies by performing T and B lymphocyte counts, diphtheria and tetanus titres, immunoglobulin levels and HIV serology. The results of these investigations were normal. Although she failed an extubation attempt 5 days after admission, the child was successfully weaned off over the next 2 days. She was fed via nasogastric tube temporarily, but resumed oral feeds within 48 h of discharge from PICU and her voice quality improved slowly over the next few days prior to discharge.
2.2. Case 2 A 22-month-old boy was referred from Pediatric Oncology ward to the Pediatric Otolaryngology service with symptoms of severe respiratory distress. He had been recently diagnosed with a large, primitive neuroectodermal cell tumor involving the third ventricle and pineal regions. Following the third course of chemotherapy, the patient developed fever, progressive upper airway obstruction, with severe hypoxia and seizure-like activity. The attending Pediatric Otolaryngologist examined the patient under general anesthesia while spontaneously breathing. Flexible laryngo-bronchoscopy revealed an inflamed epiglottis, with a single discrete ulcer on the tip (Fig. 2). There was also an inflamed trachea, and mucopurulent secretions which were aspirated and sent for microbiological investigation. There was marked subglottic edema but vocal cord movement was deemed normal. The patient was intubated endoscopically with a size 3.5 endotracheal tube. Direct fluorescent antibody tests and cultures performed on the epiglottic lesions were positive for HSV type 1. The patient was started on intravenous acyclovir therapy (30 mg/kg/day q 8 h for 10 days). By the end of the course of acyclovir, his respiratory function had improved dramatically, his laryngitis was clinically resolving and he was successfully extubated.
Acute herpes simplex laryngotracheitis
Fig. 2 A flexible endoscope view of the epiglottis. Shown is a single vesicle on the tip of the epiglottis.
3. Discussion We present two cases of severe laryngitis in which the infective pathogen was found to be HSV. The first was in a previously well 8-month-old girl, while the second occurred in a 22-month-old boy on immunosuppressive chemotherapy. Both presented as ALT and both were non-responsive to conventional management and required Intensive Care Unit management. Laryngoscopic evaluation enabled accurate diagnosis, securing of their compromised airways, and timely initiation of appropriate anti-viral therapy. A search was conducted in PubMed for indexed articles using the search phrase ‘‘herpes simplex laryngitis.’’ The search encompassed the period from 1966 to 2005 and yielded 30 hits. Seven papers were in languages other than English and translations were obtained of their abstracts. Twenty-five of the 30 papers were relevant to this article and analyzed further. Cases were considered to be HSV laryngitis if detection of HSV from the larynx was described in a patient with clinically significant obstructive or infectious picture. There were 36 total cases, of which 22 were in children under 18 years of age. Four cases occurred in the perinatal period, presumably from transmission at the time of delivery [16,18]. The only study that addressed the incidence of HSV laryngitis was a 5-year retrospective review by Hatherill et al. conducted at a university hospital in South Africa [4]. All 148 children requiring airway intervention of a total of 263 children admitted to PICU with severe upper airway obstruction underwent diagnostic laryngoscopy. Fifteen of these 148
343 children were found to have ulcerative laryngitis consistent with herpetic lesions. However, viral cultures were only performed in six of these cases and HSV was confirmed in three of these six cases. In all cases where there was evidence of ulcerative laryngitis, steroid therapy was discontinued and intravenous acyclovir was started empirically. The authors recommend early diagnostic laryngoscopy for children with severe croup who follow an atypical course, a recommendation which we endorse based on our clinical experience. All three of the patients in Hatherill and coauthors’ study were seen in the PICU. Of the remaining 19 pediatric cases in the literature, only in 3 cases was there an explicit statement that ICU was the setting of management [16,18,21]. As such, with the inclusion of the current cases, 8 of a total 24 confirmed cases of HSV laryngitis (33%) required admission to PICU. Therefore, the index of suspicion for the diagnosis should be highest in these units. Croup, or ALT, is a relatively common viral infection of the upper airway in children, typically resolving without involvement of the otolaryngologist [24]. Croup is classified as atypical when the patient presents in very early infancy (<6 months of age), or beyond toddler stage, does not respond favorably to supportive measures, recurs or progresses to a fulminant course [25]. The two cases presented straddle one or more of the criteria for atypical croup. Furthermore, 7 of the previously described 22 pediatric cases occurred in immunocompromised hosts or those receiving prolonged doses of systemic corticosteroids [4,9,15,18,22,23]. Also regional propagation or systemic dissemination of HSV is a wellrecognized phenomenon in immunologically compromised patients including newborns, debilitated, malnourished or burned children [21]. As such the finding of oral lesions in the first child, and the general status of the second furnish basis for suspicion of the diagnosis. Corticosteroids are recommended for the treatment of ALT due to their ability to reduce laryngeal inflammation and edema, relieving airway obstruction. Controversy has arisen given their use in atypical cases. Some authors contend that the immunosuppressive effect of corticosteroids is a contributory factor for the development of herpetic laryngitis [15,22]. Others retort that this is unlikely as most patients have symptomatic laryngitis prior to the initiation of corticosteroid treatment [9]. It is possible that in both cases in the current report, the child initially had laryngitis with another virus and then developed secondary HSV laryngitis related to immunosuppression from corticosteroids (case #1) or chemotherapy (case #2). Of course, the particularly rare occurrence of HSV laryngitis lies in stark
344 contrast to the normal picture of ALT in which the efficacy of corticosteroids in stabilizing the airway and potentially preventing airway compromise requiring intubation has been clearly demonstrated. Regardless, it is apparent that treatment with corticosteroids in the setting of atypical laryngitis may delay diagnosis and initiation of appropriate therapy, resulting in prolongation of the clinical course. Other causes of atypical laryngitis including inflammatory processes, such as gastroesophageal reflux disease and alternate viral processes, such as recurrent respiratory papillomatosis represent additional conditions when steroid administration would not be desired. Conversely, there are circumstances of atypical laryngotracheitis due to anatomic causes, such as subglottic hemangioma, subglottic mucus retention cysts and subglottic stenosis (especially acquired), when steroid therapy would be beneficial. The efficacy of acyclovir or other anti-virals for HSV laryngitis has not been studied. Early treatment markedly improves the efficacy of anti-virals, but given the potential severity of HSV laryngitis, it would seem reasonable to initiate anti-virals in all cases even if the diagnosis has been delayed. In immunocompromised or neonatal hosts, use of anti-virals is mandatory to prevent dissemination of HSV [25].
4. Conclusion HSV infection should be considered for atypical laryngotracheitis, especially in children and infants that require intensive care admission. Endoscopic examination demonstrates the severity and level of the problem and allows control of the airway, in conjunction with obtaining viral cultures of tracheal secretions and material from visible lesions. The prompt institution of targeted anti-viral therapy may ameliorate acute illness and prevent viral dissemination and long-term sequelae [18].
References [1] M. Caserta, Acute laryngitis, in: G. Mandell, Douglas (Eds.), Bennett’s Principles and Practice of Infectious Diseases., fifth ed., Churchill Livingstone, Edinburgh, UK, 2000 , pp. 662—663. [2] R. Schwartz, Laryngeal and subglottic infections, in: Long: Principles and Practice of Pediatric Infectious Diseases, second ed., Churchill Livingstone, Philadelphia, 2003, pp. 211—212. [3] H.B. Einarsson, S. Jensen, L.B. Rasmussen, Pseudomembranous upper respiratory tract infection caused by herpes simplex virus in an adult, Ugeskr. Laeger 164 (December (50)) (2002) 5954—5955.
N. Chauhan et al. [4] M. Hatherill, L. Reynolds, Z. Waggie, A. Argent, Severe upper airway obstruction caused by ulcerative laryngitis, Arch. Dis. Child. 85 (October (4)) (2001) 326—329. [5] J.T. Vrabec, C.P. Molina, B. West, Herpes simplex viral laryngitis, Ann. Otol. Rhinol. Laryngol. 109 (June (6)) (2000) 611—614. [6] S. Zhang, T.L. Farmer, M.A. Frable, C.N. Powers, Adult herpetic laryngitis with concurrent candidal infection: a case report and literature review, Arch. Otolaryngol. Head Neck Surg. 126 (May (5)) (2000) 672—674. [7] U.M. Musharrafieh, G.F. Araj, N.S. Fuleihan, Viral supraglottitis in an adult: a case presentation and literature update, J. Infect. 39 (Septamber (2)) (1999) 157—160. [8] J.J. Del Toro, E. Ferrandis Pereperez, E. Serrano Badia, P. Pons Rocher, Herpetic pseudotumor laryngitis, An. Otorrinolaringol. Ibero Am. 26 (1) (1999) 39—45. [9] I. Krause, T. Schonfeld, J. Ben-Ari, I. Offer, B.Z. Garty, Prolonged croup due to herpes simplex virus infection, Eur. J. Pediatr. 157 (July (7)) (1998) 567—569. [10] E. Ferrandis Pereperez, J.J. Artazkoz del Toro, E. Serrano Badia, Herpetic laryngeal involvement simulating neoplasm, Acta Otorrinolaringol. Esp. 48 (January—February (1)) (1997) 69—71. [11] V. Bengualid, S. Keesari, V. Kandah, D. Gitler, J. Berger, Supraglottitis due to herpes simplex virus type 1 in an adult, Clin. Infect. Dis. 22 (February (2)) (1996) 382—383. [12] M.Y. Mancao, L.J. Sindel, P.H. Richardson, F.M. Silver, Herpetic croup: two case reports and a review of the literature, Acta Paediatr. 85 (January (1)) (1996) 118—120. [13] G.G. Aslanian, Acute herpetic stenosing laryngitis in children, Vestn. Otorinolaringol. (July—August (4)) (1995) 37—40. [14] V. Yeh, M.L. Hopp, N.S. Goldstein, R.D. Meyer, Herpes simplex chronic laryngitis and vocal cord lesions in a patient with acquired immunodeficiency syndrome, Ann. Otol. Rhinol. Laryngol. 103 (September (9)) (1994) 726—731. [15] A.F. Inglis Jr., Herpes simplex virus infection. A rare cause of prolonged croup, Arch. Otolaryngol. Head Neck Surg. 119 (May (5)) (1993) 551—552. [16] V.J. Vitale, L. Saiman, J. Haddad Jr., Herpes laryngitis and tracheitis causing respiratory distress in a neonate, Arch. Otolaryngol. Head Neck Surg. 119 (February (2)) (1993) 239—240. [17] M. Mesolella, B. Testa, C. Mesolella, A. Giuliano, G. Testa, Herpes of the larynx. Apropos of 3 cases, Ann. Otolaryngol. Chir. Cervicofac. 110 (6) (1993) 337—340. [18] S. Nadel, P.A. Offit, R.L. Hodinka, R.M. Gesser, L.M. Bell, Upper airway obstruction in association with perinatally acquired herpes simplex virus infection, J. Pediatr. 120 (January (1)) (1992) 127—129. [19] A.J. D’Angelo Jr., S. Zwillenberg, J.P. Olekszyk, F.I. Marlowe, J. Mobini, Adult supraglottitis due to herpes simplex virus, J. Otolaryngol. 19 (June (3)) (1990) 179—181. [20] P.N. Karnauchow, W.H. Kaul, Chronic herpetic laryngitis with oropharyngitis, Ann. Otol. Rhinol. Laryngol. 97 (May—June (3 Pt 1)) (1988) 286—288. [21] S. Bogger-Goren, Acute epiglottitis caused by herpes simplex virus, Pediatr. Infect. Dis. J. 6 (December (12)) (1987) 1133—1134. [22] J.B. Harris, R. Lusk, J.S. Wagener, R.D. Andersen, Acute viral laryngotracheitis complicated by herpes simplex virus infection, Otolaryngol. Head Neck Surg. 96 (February (2)) (1987) 190—193. [23] J.J. Larzul, B. de Labarthe, B. Fauconnier, J.P. Ledoux, A. Danrigal, One case of severe tracheobronchopathy with immune depression: herpetic disease? Poumon Coeur 34 (2) (1978) 153—156.
Acute herpes simplex laryngotracheitis [24] D.F. Wilson, Inflammatory diseases of the airway, in: C. Myer, R. Cotton, S. Shot (Eds.), The Pediatric Airway: An Interdisciplinary Approach, JB Lippincott, Philadelphia, PA, 1995 , pp. 67—100.
345 [25] T.L. Farmer, D.L. Wohl, Diagnosis of recurrent intermittent airway obstruction (‘‘recurrent croup’’) in children, Ann. Otol. Rhinol. Laryngol. 110 (July (7 Pt 1)) (2001) 600—605.
www.elsevier.com/locate/ijporl
CASE REPORT
Acute herpes simplex laryngotracheitis: Report of two pediatric cases and review of the literature Nitin Chauhan a,c, Joan L. Robinson a,b,c, Jennifer Guillemaud a,c, Hamdy El-Hakim a,c,* a
Pediatric Otolaryngology (Division of Pediatric Surgery), The Stollery Children’s Hospital, Edmonton, Canada b The Division of Pediatric Infectious Diseases, The Stollery Children’s Hospital, Edmonton, Canada c The Department of Pediatrics, The Stollery Children’s Hospital, Edmonton, Canada Received 28 June 2006; received in revised form 12 October 2006; accepted 15 October 2006
KEYWORDS Laryngitis; Herpes simplex virus; Atypical laryngitis; Laryngotracheobronchitis; Croup
Summary Herpes simplex virus (HSV) is a rare cause of laryngotracheitis (LT) with only 22 previously reported and confirmed pediatric cases in the literature. It is often associated with immune deficiency states and presents with a severe acute upper airway obstruction commonly requiring intensive care management and artificial ventilation. We present two cases of atypical laryngotracheitis in which HSV was found to be the causative pathogen subsequent to laryngoscopy and microbiologic investigations. While the first case was a previously well 8-month-old girl, the second was a 22-month-old immunosuppressed boy. One-third of the total confirmed cases in the English literature required intensive care management eliciting the setting as a pointer to the diagnosis. Laryngoscopy is a readily available tool for rapid diagnosis and controlled securing of the airway. This step may avoid controversial and potential counter-productive use of systemic steroids in these cases. # 2006 Elsevier Ireland Ltd. All rights reserved.
1. Introduction Acute laryngotracheitis (ALT) in the pediatric population is a relatively common clinical condition * Corresponding author at: Pediatric Otolaryngology, 2C3.57 Walter MacKenzie Health Science Centre, 8440-112 St., Edmonton, Alberta, Canada T6G 2B7. Tel.: +1 780 407 8629; fax: +1 780 407 2004. E-mail address: [email protected] (H. El-Hakim).
encountered most often by primary care physicians. Typical signs and symptoms include a sore throat, hoarse voice and harsh cough [1,2]. ALT is mostly viral in etiology and the pathogens most commonly responsible are the typical causative agents of croup–—parainfluenza virus 1, 2 and 3, respiratory syncytial virus (RSV), influenza virus A and B, human adenovirus and less commonly enteroviruses and measles virus [2]. The treatment of ALT is usually symptomatic and supportive, anticipating complete
0165-5876/$ — see front matter # 2006 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2006.10.012
342 resolution within a few days. Herpes simplex virus (HSV) is an uncommonly encountered etiologic agent [3—23]. It usually occurs in immuno-deficient patients, manifesting a particularly severe and rapidly progressive clinical course [4,22]. We present two cases and present a literature review on the topic.
2. Case reports 2.1. Case 1 The Pediatric Otolaryngology service was consulted for evaluation of a previously well 8-month-old girl presenting with severe respiratory distress to the Emergency Department. At the time of presentation, she was very irritable, aphonic and diaphoretic with biphasic stridor and evidence of labored breathing. In the preceding 11 days, the patient had been admitted to hospital twice with a similar clinical picture, and had been diagnosed with viral croup. During each encounter, she responded partially to treatment with systemic dexamethasone and inhaled racemic epinephrine, and was discharged home within 24 h of admission. Given the severity of her respiratory distress on this occasion, she was examined under general anesthesia. With the child spontaneously breathing, flexible and rigid airway examination revealed severe laryngeal inflammation with white, ulcerative lesions noted on the tongue, palate, throughout the larynx and on the pharyngeal walls (Fig. 1).
Fig. 1 A rigid endoscope view of the larynx and pharynx. Demonstrates multiple herpetic vesicles with surrounding erythema.
N. Chauhan et al. Laryngeal movement was normal. Also noted was an edematous subglottis and upper trachea, while the lower trachea and main stem bronchi were normal. Swabs from the lesions were submitted for bacterial and viral culture. The patient was then intubated nasally with a 3.5 mm endotracheal tube over a flexible bronchoscope and admitted to the Pediatric Intensive Care Unit (PICU). Direct fluorescent antibody tests and cultures performed on the lesions in the posterior pharynx and epiglottis were positive for HSV type 1. Thus, the patient was started on intravenous acyclovir therapy (30 mg/kg/day q 8 h for 10 days). The patient was screened for immunodeficiencies by performing T and B lymphocyte counts, diphtheria and tetanus titres, immunoglobulin levels and HIV serology. The results of these investigations were normal. Although she failed an extubation attempt 5 days after admission, the child was successfully weaned off over the next 2 days. She was fed via nasogastric tube temporarily, but resumed oral feeds within 48 h of discharge from PICU and her voice quality improved slowly over the next few days prior to discharge.
2.2. Case 2 A 22-month-old boy was referred from Pediatric Oncology ward to the Pediatric Otolaryngology service with symptoms of severe respiratory distress. He had been recently diagnosed with a large, primitive neuroectodermal cell tumor involving the third ventricle and pineal regions. Following the third course of chemotherapy, the patient developed fever, progressive upper airway obstruction, with severe hypoxia and seizure-like activity. The attending Pediatric Otolaryngologist examined the patient under general anesthesia while spontaneously breathing. Flexible laryngo-bronchoscopy revealed an inflamed epiglottis, with a single discrete ulcer on the tip (Fig. 2). There was also an inflamed trachea, and mucopurulent secretions which were aspirated and sent for microbiological investigation. There was marked subglottic edema but vocal cord movement was deemed normal. The patient was intubated endoscopically with a size 3.5 endotracheal tube. Direct fluorescent antibody tests and cultures performed on the epiglottic lesions were positive for HSV type 1. The patient was started on intravenous acyclovir therapy (30 mg/kg/day q 8 h for 10 days). By the end of the course of acyclovir, his respiratory function had improved dramatically, his laryngitis was clinically resolving and he was successfully extubated.
Acute herpes simplex laryngotracheitis
Fig. 2 A flexible endoscope view of the epiglottis. Shown is a single vesicle on the tip of the epiglottis.
3. Discussion We present two cases of severe laryngitis in which the infective pathogen was found to be HSV. The first was in a previously well 8-month-old girl, while the second occurred in a 22-month-old boy on immunosuppressive chemotherapy. Both presented as ALT and both were non-responsive to conventional management and required Intensive Care Unit management. Laryngoscopic evaluation enabled accurate diagnosis, securing of their compromised airways, and timely initiation of appropriate anti-viral therapy. A search was conducted in PubMed for indexed articles using the search phrase ‘‘herpes simplex laryngitis.’’ The search encompassed the period from 1966 to 2005 and yielded 30 hits. Seven papers were in languages other than English and translations were obtained of their abstracts. Twenty-five of the 30 papers were relevant to this article and analyzed further. Cases were considered to be HSV laryngitis if detection of HSV from the larynx was described in a patient with clinically significant obstructive or infectious picture. There were 36 total cases, of which 22 were in children under 18 years of age. Four cases occurred in the perinatal period, presumably from transmission at the time of delivery [16,18]. The only study that addressed the incidence of HSV laryngitis was a 5-year retrospective review by Hatherill et al. conducted at a university hospital in South Africa [4]. All 148 children requiring airway intervention of a total of 263 children admitted to PICU with severe upper airway obstruction underwent diagnostic laryngoscopy. Fifteen of these 148
343 children were found to have ulcerative laryngitis consistent with herpetic lesions. However, viral cultures were only performed in six of these cases and HSV was confirmed in three of these six cases. In all cases where there was evidence of ulcerative laryngitis, steroid therapy was discontinued and intravenous acyclovir was started empirically. The authors recommend early diagnostic laryngoscopy for children with severe croup who follow an atypical course, a recommendation which we endorse based on our clinical experience. All three of the patients in Hatherill and coauthors’ study were seen in the PICU. Of the remaining 19 pediatric cases in the literature, only in 3 cases was there an explicit statement that ICU was the setting of management [16,18,21]. As such, with the inclusion of the current cases, 8 of a total 24 confirmed cases of HSV laryngitis (33%) required admission to PICU. Therefore, the index of suspicion for the diagnosis should be highest in these units. Croup, or ALT, is a relatively common viral infection of the upper airway in children, typically resolving without involvement of the otolaryngologist [24]. Croup is classified as atypical when the patient presents in very early infancy (<6 months of age), or beyond toddler stage, does not respond favorably to supportive measures, recurs or progresses to a fulminant course [25]. The two cases presented straddle one or more of the criteria for atypical croup. Furthermore, 7 of the previously described 22 pediatric cases occurred in immunocompromised hosts or those receiving prolonged doses of systemic corticosteroids [4,9,15,18,22,23]. Also regional propagation or systemic dissemination of HSV is a wellrecognized phenomenon in immunologically compromised patients including newborns, debilitated, malnourished or burned children [21]. As such the finding of oral lesions in the first child, and the general status of the second furnish basis for suspicion of the diagnosis. Corticosteroids are recommended for the treatment of ALT due to their ability to reduce laryngeal inflammation and edema, relieving airway obstruction. Controversy has arisen given their use in atypical cases. Some authors contend that the immunosuppressive effect of corticosteroids is a contributory factor for the development of herpetic laryngitis [15,22]. Others retort that this is unlikely as most patients have symptomatic laryngitis prior to the initiation of corticosteroid treatment [9]. It is possible that in both cases in the current report, the child initially had laryngitis with another virus and then developed secondary HSV laryngitis related to immunosuppression from corticosteroids (case #1) or chemotherapy (case #2). Of course, the particularly rare occurrence of HSV laryngitis lies in stark
344 contrast to the normal picture of ALT in which the efficacy of corticosteroids in stabilizing the airway and potentially preventing airway compromise requiring intubation has been clearly demonstrated. Regardless, it is apparent that treatment with corticosteroids in the setting of atypical laryngitis may delay diagnosis and initiation of appropriate therapy, resulting in prolongation of the clinical course. Other causes of atypical laryngitis including inflammatory processes, such as gastroesophageal reflux disease and alternate viral processes, such as recurrent respiratory papillomatosis represent additional conditions when steroid administration would not be desired. Conversely, there are circumstances of atypical laryngotracheitis due to anatomic causes, such as subglottic hemangioma, subglottic mucus retention cysts and subglottic stenosis (especially acquired), when steroid therapy would be beneficial. The efficacy of acyclovir or other anti-virals for HSV laryngitis has not been studied. Early treatment markedly improves the efficacy of anti-virals, but given the potential severity of HSV laryngitis, it would seem reasonable to initiate anti-virals in all cases even if the diagnosis has been delayed. In immunocompromised or neonatal hosts, use of anti-virals is mandatory to prevent dissemination of HSV [25].
4. Conclusion HSV infection should be considered for atypical laryngotracheitis, especially in children and infants that require intensive care admission. Endoscopic examination demonstrates the severity and level of the problem and allows control of the airway, in conjunction with obtaining viral cultures of tracheal secretions and material from visible lesions. The prompt institution of targeted anti-viral therapy may ameliorate acute illness and prevent viral dissemination and long-term sequelae [18].
References [1] M. Caserta, Acute laryngitis, in: G. Mandell, Douglas (Eds.), Bennett’s Principles and Practice of Infectious Diseases., fifth ed., Churchill Livingstone, Edinburgh, UK, 2000 , pp. 662—663. [2] R. Schwartz, Laryngeal and subglottic infections, in: Long: Principles and Practice of Pediatric Infectious Diseases, second ed., Churchill Livingstone, Philadelphia, 2003, pp. 211—212. [3] H.B. Einarsson, S. Jensen, L.B. Rasmussen, Pseudomembranous upper respiratory tract infection caused by herpes simplex virus in an adult, Ugeskr. Laeger 164 (December (50)) (2002) 5954—5955.
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