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Isolating Candida epiglottitis
Otolaryngology–Head and Neck Surgery (2010) 142, 630-631
CASE REPORT
Isolating Candida epiglottitis Wu-Chia Lo, MD, Shiann-Yann Lee, MD, and Wei-Chung Hsu, MD, PhD, Taipei, Taiwan No sponsorships or competing interests have been disclosed for this article.
I
solated necrotizing fungal epiglottitis is rarely reported, and Candida is an uncommon cause that may develop in immunocompromised patients. This report demonstrates a treatment course more complex than those previously reported for Candida epiglottitis. Treatment using intravenous antifungal agents given for eight consecutive weeks combined with endoscopic approach for epiglottic necrotic tissue biopsy and debridement ultimately controlled the disease, which may be seen more frequently by otolaryngologists in the future.
Case Report A 54-year-old woman with adult T cell leukemia/lymphoma diagnosed six years previously and under treatment with interferon-␣ and zidovudine had a white blood cell (WBC) count of approximately 6000 cells/mm3. She presented to us with a 45-day history of progressive sore throat and odynophagia. Lateral radiography of the neck revealed a swollen epiglottis with compromised upper airway (Fig 1). Under flexible fiberoptic endoscopy, the epiglottis was shown to be distorted and thickened, with yellowish patches (Fig 2). The initial WBC count was 13,490 cells/mm3, with 52.6 percent segmented neutrophils and 2.1 percent band forms. Despite initiating intravenous co-amoxiclav (875 mg amoxicillin and 125 mg clavulanate potassium) at 3 g/day, tissue necrosis worsened. Direct laryngoscopy was arranged for tissue biopsy and culture of the epiglottis. Histopathologic examination showed many fungal spores and pseudohyphae within the tissue, and the culture revealed Candida albicans. Intravenous fluconazole (400 mg/day maintenance) was then prescribed. Unfortunately, the clinical condition did not improve and the patient underwent endoscopic excision of the necrotic epiglottis, which disclosed invasive candidiasis. Amphotericin B (0.5 mg/kg/d) was prescribed on day 14 as drug-sensitivity test showed fluconazole resistance. The symptoms deteriorated, with the appearance of white plaque extending to the glottis and tongue base. Amphotericin B was increased to 1 mg/kg/d on day 26. As there was
Figure 1 Lateral soft tissue radiography of the neck demonstrates swelling of the epiglottis (thumb sign) with decreased airway diameter.
no amelioration of the local condition, amphotericin B was replaced with caspofungin (50 mg/d maintenance) on day 44. The local condition and symptoms subsequently improved.
Discussion Candida species are opportunistic pathogens that commonly arise in immunocompromised patients. Although it is well recognized as a pathogen on the mucus membranes of the upper aero-digestive tract, isolated necrotizing epiglottitis by Candida species is very rare. There are limited case reports describing Candida epiglottitis in patients with leukemia or lymphoma, human immunodeficiency virus infection, or autoimmune disorders.1-4 The institutional review board of National Taiwan University Hospital in Taiwan approved the
Received September 2, 2009; revised September 22, 2009; accepted September 30, 2009.
0194-5998/$36.00 © 2010 American Academy of Otolaryngology–Head and Neck Surgery Foundation. All rights reserved. doi:10.1016/j.otohns.2009.09.033
Lo et al
Isolating Candida epiglottitis
631 of treatment, but both have toxicities, and possible resistant strains may be encountered after long-term use. A new antifungal agent, caspofungin, is now considered an alternative. It is as effective as amphotericin B but with a significantly superior safety profile. Because of the growing population of immunocompromised patients, the incidence of fungal infections has increased significantly over the years. Clinicians should be alert to the possibility of Candida epiglottitis in immunocompromised patients developing in the absence of oropharyngeal candidiasis, as well as the possibility that fungal infection of the epiglottis may indicate an underlying malignancy. Furthermore, if the local findings show continuing deterioration despite initial management, a shift to a more potent antifungal agent and prompt surgical intervention are necessary to minimize morbidity and prevent hematogenous dissemination.
Author Information Figure 2 Endoscopic view of the supra-glottis shows extensive white pseudo-membrane involving the distorted epiglottis.
submission of this case report (No. 200908010R) in recognition of its clinical importance. The clinical presentation of Candida epiglottitis usually includes sore throat, odynophagia, and dysphagia, which infrequently progress to upper airway compromise. The wide spectrum of initial local findings makes early diagnosis difficult. It may present with simple erythematous and edematous mucosal changes with or without ulcer, typical white plaque, or necrosis.2-4 The lesion may sometimes simulate a malignant process or even bear malignant cells,5 thereby warranting tissue biopsy to exclude neoplasm. A diagnosis of definite invasive fungal infection requires both positive culture and histopathologic examination. The pathogens of Candida epiglottitis reported in the literature are almost always Candida albicans.1-5 Treatment strategies depend on the clinical course and include oral nystatin or fluconazole, intravenous fluconazole with or without oral nystatin, intravenous amphotericin B with or without oral nystatin, and surgical excision with oral fluconazole.1-5 Surgical intervention is meant to investigate the possibility of malignancy and prevent systemic fungal infection.5 Due to the limited case numbers reported thus far, there is still no consensus as to the best form of treatment. Amphotericin B and voriconazole are the mainstays
From Department of Otolaryngology, Head and Neck Surgery, National Taiwan University Hospital, Taipei, Taiwan. Corresponding author: Wei-Chung Hsu, MD, PhD, Department of Otolaryngology, National Taiwan University Hospital, 7, Chung-Shan South Rd., Taipei, Taiwan. E-mail address: [email protected].
Author Contributions Wu-Chia Lo, writer; Shiann-Yann Lee, data collection, consultant; Wei-Chung Hsu, patient treatment, consultant.
Disclosures Competing interests: None. Sponsorships: None.
References 1. Sharma N, Berman DM, Scott GB, et al. Candida epiglottitis in an adolescent with acquired immunodeficiency syndrome. Pediatr Infect Dis J 2005;24:91–2. 2. Walsh TJ, Gray WC. Candida epiglottitis in immuno-compromised patients. Chest 1987;91:482–5. 3. Colman MF. Epiglottitis in immuno-compromised patients. Head Neck Surg 1986;8:466 – 8. 4. Cole S, Zawin M, Lundberg B, et al. Candida epiglottitis in an adult with acute non-lymphocytic leukemia. Am J Med 1987;82:662– 4. 5. Mäkitie AA, Bäck L, Aaltonen LM, et al. Fungal infection of the epiglottis simulating a clinical malignancy. Arch Otolaryngol Head Neck Surg 2003;129:124 – 6.
CASE REPORT
Isolating Candida epiglottitis Wu-Chia Lo, MD, Shiann-Yann Lee, MD, and Wei-Chung Hsu, MD, PhD, Taipei, Taiwan No sponsorships or competing interests have been disclosed for this article.
I
solated necrotizing fungal epiglottitis is rarely reported, and Candida is an uncommon cause that may develop in immunocompromised patients. This report demonstrates a treatment course more complex than those previously reported for Candida epiglottitis. Treatment using intravenous antifungal agents given for eight consecutive weeks combined with endoscopic approach for epiglottic necrotic tissue biopsy and debridement ultimately controlled the disease, which may be seen more frequently by otolaryngologists in the future.
Case Report A 54-year-old woman with adult T cell leukemia/lymphoma diagnosed six years previously and under treatment with interferon-␣ and zidovudine had a white blood cell (WBC) count of approximately 6000 cells/mm3. She presented to us with a 45-day history of progressive sore throat and odynophagia. Lateral radiography of the neck revealed a swollen epiglottis with compromised upper airway (Fig 1). Under flexible fiberoptic endoscopy, the epiglottis was shown to be distorted and thickened, with yellowish patches (Fig 2). The initial WBC count was 13,490 cells/mm3, with 52.6 percent segmented neutrophils and 2.1 percent band forms. Despite initiating intravenous co-amoxiclav (875 mg amoxicillin and 125 mg clavulanate potassium) at 3 g/day, tissue necrosis worsened. Direct laryngoscopy was arranged for tissue biopsy and culture of the epiglottis. Histopathologic examination showed many fungal spores and pseudohyphae within the tissue, and the culture revealed Candida albicans. Intravenous fluconazole (400 mg/day maintenance) was then prescribed. Unfortunately, the clinical condition did not improve and the patient underwent endoscopic excision of the necrotic epiglottis, which disclosed invasive candidiasis. Amphotericin B (0.5 mg/kg/d) was prescribed on day 14 as drug-sensitivity test showed fluconazole resistance. The symptoms deteriorated, with the appearance of white plaque extending to the glottis and tongue base. Amphotericin B was increased to 1 mg/kg/d on day 26. As there was
Figure 1 Lateral soft tissue radiography of the neck demonstrates swelling of the epiglottis (thumb sign) with decreased airway diameter.
no amelioration of the local condition, amphotericin B was replaced with caspofungin (50 mg/d maintenance) on day 44. The local condition and symptoms subsequently improved.
Discussion Candida species are opportunistic pathogens that commonly arise in immunocompromised patients. Although it is well recognized as a pathogen on the mucus membranes of the upper aero-digestive tract, isolated necrotizing epiglottitis by Candida species is very rare. There are limited case reports describing Candida epiglottitis in patients with leukemia or lymphoma, human immunodeficiency virus infection, or autoimmune disorders.1-4 The institutional review board of National Taiwan University Hospital in Taiwan approved the
Received September 2, 2009; revised September 22, 2009; accepted September 30, 2009.
0194-5998/$36.00 © 2010 American Academy of Otolaryngology–Head and Neck Surgery Foundation. All rights reserved. doi:10.1016/j.otohns.2009.09.033
Lo et al
Isolating Candida epiglottitis
631 of treatment, but both have toxicities, and possible resistant strains may be encountered after long-term use. A new antifungal agent, caspofungin, is now considered an alternative. It is as effective as amphotericin B but with a significantly superior safety profile. Because of the growing population of immunocompromised patients, the incidence of fungal infections has increased significantly over the years. Clinicians should be alert to the possibility of Candida epiglottitis in immunocompromised patients developing in the absence of oropharyngeal candidiasis, as well as the possibility that fungal infection of the epiglottis may indicate an underlying malignancy. Furthermore, if the local findings show continuing deterioration despite initial management, a shift to a more potent antifungal agent and prompt surgical intervention are necessary to minimize morbidity and prevent hematogenous dissemination.
Author Information Figure 2 Endoscopic view of the supra-glottis shows extensive white pseudo-membrane involving the distorted epiglottis.
submission of this case report (No. 200908010R) in recognition of its clinical importance. The clinical presentation of Candida epiglottitis usually includes sore throat, odynophagia, and dysphagia, which infrequently progress to upper airway compromise. The wide spectrum of initial local findings makes early diagnosis difficult. It may present with simple erythematous and edematous mucosal changes with or without ulcer, typical white plaque, or necrosis.2-4 The lesion may sometimes simulate a malignant process or even bear malignant cells,5 thereby warranting tissue biopsy to exclude neoplasm. A diagnosis of definite invasive fungal infection requires both positive culture and histopathologic examination. The pathogens of Candida epiglottitis reported in the literature are almost always Candida albicans.1-5 Treatment strategies depend on the clinical course and include oral nystatin or fluconazole, intravenous fluconazole with or without oral nystatin, intravenous amphotericin B with or without oral nystatin, and surgical excision with oral fluconazole.1-5 Surgical intervention is meant to investigate the possibility of malignancy and prevent systemic fungal infection.5 Due to the limited case numbers reported thus far, there is still no consensus as to the best form of treatment. Amphotericin B and voriconazole are the mainstays
From Department of Otolaryngology, Head and Neck Surgery, National Taiwan University Hospital, Taipei, Taiwan. Corresponding author: Wei-Chung Hsu, MD, PhD, Department of Otolaryngology, National Taiwan University Hospital, 7, Chung-Shan South Rd., Taipei, Taiwan. E-mail address: [email protected].
Author Contributions Wu-Chia Lo, writer; Shiann-Yann Lee, data collection, consultant; Wei-Chung Hsu, patient treatment, consultant.
Disclosures Competing interests: None. Sponsorships: None.
References 1. Sharma N, Berman DM, Scott GB, et al. Candida epiglottitis in an adolescent with acquired immunodeficiency syndrome. Pediatr Infect Dis J 2005;24:91–2. 2. Walsh TJ, Gray WC. Candida epiglottitis in immuno-compromised patients. Chest 1987;91:482–5. 3. Colman MF. Epiglottitis in immuno-compromised patients. Head Neck Surg 1986;8:466 – 8. 4. Cole S, Zawin M, Lundberg B, et al. Candida epiglottitis in an adult with acute non-lymphocytic leukemia. Am J Med 1987;82:662– 4. 5. Mäkitie AA, Bäck L, Aaltonen LM, et al. Fungal infection of the epiglottis simulating a clinical malignancy. Arch Otolaryngol Head Neck Surg 2003;129:124 – 6.