Actinomycosis of the larynx represents an unusual presentation for a common bacterium comprising the oral and oropharyngeal florae. There are few cases reported in the literature of laryngeal actinomycosis occurring primarily in the immunocompromised population. Here, we present a case in a 74-year-old man that occurred in the setting of neutropenia as a result of chemotherapy. Once the diagnosis was made with biopsy of the larynx, the infection was resolved after a prolonged course of penicillin-based therapy

Chheda, Neil N

Duckworth, Lizette Vila

Jaworek, Aaron J, MD

Patel, Sheylan

Patel, Vatsal

Sawhney, Raja

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Lehigh Valley Health Network LVHN Scholarly Works Department of Surgery Laryngeal Actinomycosis in an Immunocompromised Patient. Sheylan Patel Aaron J. Jaworek MD Lehigh Valley Health Network, Aaron_J.Jaworek@lvhn.org Vatsal Patel Lizette Vila Duckworth Raja Sawhney See next page for additional authors Follow this and additional works at: https://scholarlyworks.lvhn.org/surgery  Part of the Other Medical Specialties Commons, and the Surgery Commons Published In/Presented At Patel, S., Jaworek, A. J., Patel, V., Duckworth, L. V., Sawhney, R., & Chheda, N. N. (2014). Laryngeal actinomycosis in an immunocompromised patient. Journal Of Voice: Official Journal Of The Voice Foundation, 28(6), 838-840. doi:10.1016/j.jvoice.2014.02.011 This Article is brought to you for free and open access by LVHN Scholarly Works. It has been accepted for inclusion in LVHN Scholarly Works by an authorized administrator. For more information, please contact LibraryServices@lvhn.org. Authors Sheylan Patel, Aaron J. Jaworek MD, Vatsal Patel, Lizette Vila Duckworth, Raja Sawhney, and Neil N Chheda This article is available at LVHN Scholarly Works: https://scholarlyworks.lvhn.org/surgery/683 Laryngeal Actinomycosis in anImmunocompromised PatientSheylan Patel, Aaron J. Jaworek, Vatsal Patel, Lizette Vila Duckworth, Raja Sawhney, and Neil N. Chheda,Gainesville, FloridaSummary: Actinomycosis of the larynx represents an unusual presentation for a common bacterium comprising theoral and oropharyngeal florae. There are few cases reported in the literature of laryngeal actinomycosis occurring pri-marily in the immunocompromised population. Here, we present a case in a 74-year-old man that occurred in the settingof neutropenia as a result of chemotherapy. Once the diagnosis was made with biopsy of the larynx, the infection wasresolved after a prolonged course of penicillin-based therapy.Key Words: Neutropenia–Actinomycosis–Immunocompromised–Larynx.INTRODUCTIONActinomyces are gram-positive, filamentous, anaerobic bacteriathat are a normal constituent of the human oral and oropharyn-geal florae. It is estimated that nearly 100% of people are colo-nized by 2 years of age.1 Although the bacteria typically do notcause disease, individuals with a history of radiation therapy tothe neck or trauma are at increased risk of progressing to infec-tion, presumably through mucosal damage.2 However, severalcase reports have been published regarding actinomycosis in-fections of individuals who were immunocompromised mostoften because of human immunodeficiency virus (HIV) orfrom the chronic use of corticosteroids.3Diagnosis of actinomycosis is particularly challenging as it isfrequently mistaken for other infectious pathogens or even ma-lignancy, whichmay delay diagnosis and appropriate treatment.When the disease occurs, it can affect a variety of tissues; how-ever, more than 50% of the cases are oral-cervical in nature,typically affecting the soft tissues around themandible.2 Rarely,cases have been reported in which actinomycosis affects thelarynx primarily.4–17 Here, we present the case of laryngealactinomycosis that developed in an immunocompromisedpatient with chronic lymphocytic leukemia (CLL).CASE REPORTA 74-year-old man was diagnosed with CLL 12 years ago andhad been undergoing treatment with bendamustine and rituxi-mab since 2010, with his last treatment ending in November2012. In January 2013, he was admitted to the hospital forfebrile neutropenia and complaints of odynophagia anddysphagia. Computed tomography (CT) of the neck and chestwith contrast was ordered but did not demonstrate any findingsto explain the patient’s symptoms. Subsequent esophagogastro-duodenoscopy was only significant for an erythematousepiglottis. Otolaryngology was consulted, and flexible naso-pharyngolaryngoscopy did not show any mucositis, masses, orstructural abnormalities. He was started on a proton-pump in-hibitor trial for suspected laryngopharyngeal reflux. Symptomsimproved with conservative management, and the patient wasdischarged home on prophylactic ciprofloxacin and amoxi-cillin-clavulanate potassium.Two months later, the patient was readmitted for febrile neu-tropenia. Repeat CT of the neck with contrast showed a rapidlyprogressive infiltrating process (Figure 1) with associated necro-sis within the larynx. Although the location and progression ofthe radiographic findings were suspicious for tuberculosis, hisworkup including Tuberculin Skin Test was negative. The patientwas subsequently taken to the operating room for direct laryn-goscopy with biopsy. Examination under anesthesia showeddiffuse supraglottic edema and erythema with exudative lesionsalong the right true and false vocal folds. Histologic examinationrevealed markedly inflamed and ulcerated squamous andrespiratory-type mucosa (Figure 2) with numerous bacterial col-onies including Actinomyces sulfur granules (Figures 3 and 4).Acid-fast stain was negative for mycobacterial organisms, andno fungal organisms were seen on Gomori’s methenamine silver(GMS) stain. The patient was discharged home on a course ofciprofloxacin and Augmentin for 1 month as recommended byinfectious disease specialists. A thorough dental evaluation andcleaning was also recommended to reduce Actinomyces coloni-zation. A follow-up flexible laryngoscopy was unable to be per-formed; however, a telephonic interview revealed near-completeresolution of his dysphonia along with subjective improvementin dysphagia after completing the course of antibiotics.DISCUSSIONActinomycotic colonies can be present in the normal oral/oropharyngeal flora of healthy individuals; however, certainpathologic states can make an individual much more likely toprogress to infection—namely being immunocompromised.4The primary pathogen is Actinomyces israelii, although otherspecies can also cause infection.Actinomycosis can be difficult to diagnose as it is frequentlymistaken for other disease entities including cervicofacial ab-scesses and benign or malignant neoplasms. Although it is arelatively rare infection, certain ‘‘classic’’ scenarios shouldprompt the clinician to suspect actinomycosis. Infections ofthe soft tissues, such as a painless mass at the angle of theAccepted for publication February 21, 2014.Poster Presentation at Fall Voice Conference October 18–19, 2013; Atlanta, Georgia.From the Division of Laryngology, Department of Otolaryngology—Head and NeckSurgery, University of Florida Health, Gainesville, Florida.Address correspondence and reprint requests to Neil N. Chheda, Division of Laryngology,Department of Otolaryngology, PO Box 100264, Gainesville, FL 32610. E-mail: Neil.Chheda@ent.ufl.eduJournal of Voice, Vol. 28, No. 6, pp. 838-8400892-1997/$36.00 2014 The Voice Foundationhttp://dx.doi.org/10.1016/j.jvoice.2014.02.011Downloaded for library services (libraryservices@lvhn.org) at Lehigh Valley Health Network from ClinicalKey.com by Elsevier on August 31, 2022. For personal use only. No other uses without permission. Copyright ©2022. Elsevier Inc. All rights reserved.jaw, that respond to a course of empiric antibiotics and subse-quently relapses, regardless of location, should prompt the pro-vider to suspect actinomycosis. Also, infections that spreadwithout regard to fascial planes or those that create recurrent‘‘draining’’ sinus tracts should also raise suspicion for an Acti-nomyces infection.2 In this particular case report, the patient’spresentation mimicked that of a laryngeal tuberculosis infectionin the setting of immunocompromise.Biopsy is the best way to confirm the diagnosis. Histologicexamination demonstrates granulation tissue with variablenumbers of club-shaped basophilic filamentous organisms ar-ranged in a rosette pattern surrounded by neutrophils (so calledsulfur granules), which are best visualized on Gram or GMSstain. Diagnosis can also be made by culturing the sample underanaerobic conditions; however, this is less sensitive and typi-cally takes between 2 and 4 weeks for primary isolation.Because of the presence of overgrowth of other bacteria orlack of anaerobic conditions, recovery rates are often lessthan 30%. This can potentially delay or miss the diagnosis alto-gether. Perhaps, the greatest histologic mimicker of Actino-myces is Nocardia, which are also gram-positive slenderbranching filamentous bacteria. However, a modified acid-faststain can be used to differentiate Actinomyces species fromNocardia, which are partially acid fast.The immunocompromised patient may present in a variety ofways increasing the challenge of diagnosis. Involvement of thehead and neck region most frequently includes the parotid, sub-mandibular area, and the mandible with the larynx being a rarersite. Infection has been more frequently seen in thosewith a his-tory of radiotherapy. When there is involvement of the larynx,dysphonia is a frequent symptom, but this may not always bepresent. Our patient primarily endorsed dysphagia but deniedsignificant voice change and presented without neck mass ordraining tract. Chaudhry et al noted that when actinomycosiswas detected in immunocompromised patients with HIV,most patients were initially misdiagnosed.18 The severity ofthe immunosuppression supported a more aggressive andfulminant infection. This is consistent with our patient, andthe rapid progression observed on serial imaging.Actinomycosis typically responds well to a penicillin-basedtherapy. The typical treatment regimen for orocervicofacialactinomycosis is penicillin G 18–24 million units intravenousper day or 2–4 grams by mouth per day for a duration of 2–6FIGURE 1. CT neck with contrast axial cut showing right-sidedsupraglottic edema and necrosis.FIGURE 2. Inflamed granulation tissue of the larynx with densemixed inflammatory infiltrate consisting of lymphocytes, plasma cells,and eosinophils (hematoxylin and eosin; magnification at 320).FIGURE 3. Classic sulfur granule associated with Actinomyces(hematoxylin and eosin; magnification at 340).FIGURE 4. Filamentous rods within a sulfur granule ofActinomyces(magnification at 340).Sheylan Patel, et al Actinomycosis in an Immunocompromised Patient 839Downloaded for library services (libraryservices@lvhn.org) at Lehigh Valley Health Network from ClinicalKey.com by Elsevier on August 31, 2022. For personal use only. No other uses without permission. Copyright ©2022. Elsevier Inc. All rights reserved.weeks.19 If the patient is allergic to penicillin, then clindamycinor doxycycline is suitable alternatives for mild cases. For moresevere infections, rocephin or imipenem can be used. With theappropriate duration of treatment, a complete resolution of theinfection is expected. Resolution of immunosuppression willalso prevent relapse.CONCLUSIONWhen a laryngeal lesion is detected, particularly with radio-graphic signs of infiltration, the presence of malignancy mustbe evaluated. However, in the immunocompromised host,opportunistic infections, such as Actinomyces, may also be pre-sent in the larynx. Unless there is a high index of suspicion,diagnosis can be delayed because of the nonspecific nature ofsymptomatology and imaging findings. In such patients, biopsyshould be considered for both histology and microbiology ana-lyses. Full recovery may be expected with the appropriate tar-geted microbial therapy.REFERENCES1. Sarkonen N, Kononen E, Summanen P, Kanervo A, Takala A, Jousimies-Somer H. Oral colonization with Actinomyces species in infants by twoyears of age. J Dental Res. 2000;79:864–867.2. Mandell GL, Bennett JE, Dolin R. Agents of actinomycosis. Mandell,Douglas, and Bennett’s principles and practice of infectious diseases. 7thed. Edinburgh: Churchill Livingstone; 2010:3209–3219.3. Hagan ME, Klotz SA, Bartholomew W, Cherian R, McGregor D. Actino-mycosis of the trachea with acute tracheal obstruction. Clin Infect Dis.1996;6:1126–1127.4. Sari M, Yazici M, Baglam T, Inanli S, Eren F. Actinomycosis of the larynx.Acta Otolaryngol. 2007;5:550–552.5. Artesi L, Gorini E, Lecce S, Mullace M, Sbrocca M, Mevio E.Laryngeal actinomycosis. Otolaryngol Head Neck Surg. 2006;135:161–162.6. Batur CA, Ozbal AE, Basak T, Turgut S. Laryngeal actinomycosis accom-panying laryngeal carcinoma: report of two cases. Eur Arch Otorhinolar-yngol. 2006;263:783–785.7. Brandenburg JH, Finch WW, Kirkham WR. Actinomycosis of the larynxand pharynx. Otolaryngology. 1978;86:739–742.8. Nelson EG, Tybor AG. Actinomycosis of the larynx. Ear_Nose_Throat_J.1992;71:356–358.9. Tsuji DH, Fukuda H, Kawasaki Y, Kawaida M, Ohira T. Actinomycosis ofthe larynx. Auris Nasus Larynx. 1991;18:79–85.10. Melgarejo MPJ, Hellin MD, Gil VM, Ruiz MJA. Primary laryngeal actino-mycosis. Acta Otorrinolaringol Esp. 1997;48:237–238.11. Fernandez SH. Actinomycosis of the vocal cord: a case report. Malays JPathol. 1999;21:111–115.12. Koegel L Jr, Tucker HM. Postoperative actinomycotic infection of the lar-ynx. Otolaryngol Head Neck Surg. 1983;91:213–216.13. Wierzbicka M, Bartochowska A, Nowak K, Szyfter W. Laryngeal actino-mycosis— a case report and the review of the literature. Otolaryngol Pol.2013;67:308–311.14. Abed T, Ahmed J, O’shea N, Payne S, Watters GW. Primary laryngeal acti-nomycosis in an immunosuppressed woman: a case report. Ear Nose ThroatJ. 2013;92:301–303.15. Shaheen SO, Ellis FG. Actinomycosis of the larynx. J R Soc Med. 1983;76:226–228.16. Sims HS, Heywood BB. Post-transplant actinomycosis of the posteriorglottis involving both vocal processes. Otolaryngol Head Neck Surg.2007;137:967–968.17. Thomas R, Kameswaran M, Ahmed S, Khurana P, Morad N. Actinomy-cosis of the vallecula: report of a case and review of the literature. J Lar-yngol Otol. 1995;109:154–156.18. Chaudhry SI, Greenspan JS. Actinomycosis in HIV infection: a review of arare complication. Int J STD AIDS. 2000;11:349–355.19. Wong VK, Turmezei TD, Weston VC. Actinomycosis. BMJ. 2011;343:d6099.Journal of Voice, Vol. 28, No. 6, 2014840Downloaded for library services (libraryservices@lvhn.org) at Lehigh Valley Health Network from ClinicalKey.com by Elsevier on August 31, 2022. For personal use only. No other uses without permission. Copyright ©2022. Elsevier Inc. All rights reserved.

Laryngeal Actinomycosis in an Immunocompromised Patient.

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