ANSWER TO THE PHOTO QUIZ
Philip A. Mackowiak, Section Editor
A 61 year-old Female with a Prior History of
Tuberculosis Presenting with Hemoptysis
(See page 910 for the Photo Quiz.)
Figure 1. Axial (A) and coronal maximum intensity projection (B) images from a contrast-enhanced computed tomographic scan of
the chest show numerous cavities with indwelling fungal balls (white arrows), as well as bronchiectasis (black arrows).
Diagnosis: Chronic Necrotizing Pulmonary Scedosporiosis.
Heavy growth of Scedosporium apiospermum was
obtained from expectorated sputum and bronchoalveolar lavage samples. These cultures had positive
results repeatedly over the course of several months.
This, in combination with the patient’s clinical picture
and imaging findings, supports a diagnosis of chronic
necrotizing pulmonary S. apiospermum infection.
Chronic necrotizing pulmonary mycotic infection is
a relatively rare disease that is gaining appreciation in
the literature as a distinct clinical entity. It is almost
exclusively described in patients with Aspergillus
infections, referred to as chronic necrotizing pulmonary
aspergillosis (CNPA). Here we report a similar disease process caused by another filamentous fungus,
S. apiospermum.
S. apiospermum (sexual form, Pseudoallescheria boydii) is an opportunistic filamentous fungus that can be
isolated from soil and contaminated water sources. It is
found in temperate climate regions worldwide. The
other major genus member, Scedosporium prolificans, is
restricted to Australia, Spain, Portugal, and the southern
United States, as well as California [1]. On the basis of
clinical, radiographic, and histopathologic features,
Scedosporium species can be virtually indistinguishable
from Aspergillus species; however, management presents
unique challenges.
Like Aspergillus species, S. apiospermum causes
a wide spectrum of pulmonary disease ranging from
allergic bronchopulmonary mycosis to uncomplicated
fungal balls to more extensive cavitary disease with or
without angioinvasion [1]. Within this disease spectrum, our patient presented with a semi-invasive
chronic necrotizing pulmonary infection akin to
CNPA. Typical presenting symptoms include chronic
cough and intermittent hemoptysis. Systemic symptoms, such as fever and weight loss, are less common.
Risk factors include moderate immunosuppression,
such as that caused by diabetes mellitus or corticosteroid use [2–5]. This is in contrast to patients with invasive pulmonary mycoses and/or disseminated disease
who are profoundly immunosuppressed as a result of
solid-organ transplant, bone marrow transplant, or
AIDS [6–8]. Previous lung damage with underlying
cavitary disease is extremely common. In the largest
series to date of patients with CNPA, more than 90%
of patients had a prior history of mycobacterial disease [5]. In this series, the most common imaging
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Figure 2. Lactophenol cotton blue stain showing septate hyphae with large unicellular oval conidia (arrows) on elongated conidiophores.
manifestations of CNPA included cavitation (100%), parenchymal consolidation (84%), pleural thickening (81%), fungus balls (49%), and bronchiectasis (37%), all of which are
well seen on our patient’s scan (Figure 1). Other, less frequent
findings of CNPA have included bronchopleural fistula
(19%) and emphysema (14%), which were not seen in our
patient [5].
Because of the similar clinical and radiographic findings of
Scedosporium and Aspergillus species, microbiologic characterization is critical to make the diagnosis. S. apiospermum grows
rapidly on standard mycologic media, such as Sabouraud glucose agar, at 25°C. Macroscopically, colonies are initially white
and turn gray over time. Microscopic examination reveals irregularly branching septate hyphae with a single oval conidium
with truncate bases forming on conidiophores (Figure 2, arrows). This appearance can be easily distinguished from that of
the aggregate conidia of Aspergillus species, which project in
columns or chains from a unique flask-shaped vesicle at the end
of a long conidiophore.
Chronic necrotizing pulmonary scedosporiosis presents
unique management challenges. In vitro resistance to and clinical failure of amphotericin therapy have been commonly reported with scedosporiosis. Broad-spectrum azoles, such as
voriconazole and posaconazole, as well as caspofungin, show
superior in vitro activity [9]. Recent reports of consistent clinical
success with voriconazole have made this the antifungal drug of
choice [10]. Duration of treatment is not yet well defined.
Our patient was not a candidate for surgical resection because
of poor pulmonary functional capacity and the extensive nature
of her lesions. For management of her hemoptysis, selective
embolization of 4 right bronchial arteries was performed. She
began voriconazole therapy and has significantly improved,
having not had progression of her hemoptysis or required
hospitalization during the past 6 months.
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Acknowledgments
Potential conflicts of interest. D.N. has been a consultant for Pfizer,
Inc, and LifeCell Corporation. D.J.D. has been a consultant for Osiris
Therapeutics. All other authors: no conflicts.
C. M. Durand,1 D. J. Durand,2 R. Lee,3 S. C. Ray,1 and D. Neofytos1
1School of Medicine, Division of Infectious Disease, 2School of Medicine,
Department of Radiology, Johns Hopkins University, and 3Department of
Pathology, Division of Medical Microbiology, Johns Hopkins Hospital,
Baltimore, Maryland
References
1. Karoll J, Cortez KJ, Roilides E, et al. Infections caused by Scedosporium
spp. Clin Microbiol Rev 2008; 21:157–97.
2. Binder RE, Faling LJ, Pugatch RD, Mahasen C, Snider GL. Chronic
necrotizing pulmonary aspergillosis: a discrete clinical entity. Medicine
1982; 61:109–24.
3. Camuset J, Nunes H, Dombret MC, et al. Treatment of chronic pulmonary aspergillosis by voriconazole in nonimmunocompromised
patients. Chest 2007; 131:1435–51.
4. Sambatakou H, Dupont B, Lode H, Denning DW. Voriconazole
treatment for subacute invasive and chronic pulmonary aspergillosis.
Am J Med 2006; 119:527 e17–24.
5. Nam HS, Jeon K, Um SE, et al. Clinical characteristics and treatment
outcomes of chronic necrotizing pulmonary aspergillosis: a review of
43 cases. Inf J Infect Dis 2010; 14:e479–82.
6. Castiglioni B, Sutton DA, Rinaldi MG, Fung J, Kusne S. Pseudallescheria boydii (anamorph Scedosporium apiospermum) infection in
solid organ transplant recipients in a tertiary medical center and review
of the literature. Medicine 2002; 81:333–48.
7. Sarva ST, Manjunath SK, Baldwin HS, Robins DB, Freire AX. Lung
scedosporiosis in human immunodeficiency virus/acquired immunodeficiency. Am J Med Sci 2010; 339:300–3.
8. Sheu R, Bricker AO, Sahi H, Mohammed TL. Pseudoallescheria boydii
(Scedosporium species) in 3 lung transplant recipients: computed tomography findings and literature review. J Comput Assist Tomogr
2009; 33:247–52.
9. Meletiadis J, Meis JF, Mouton JW, et al. In vitro activities of new and
conventional antifungal agents against clinical Scedosporium isolates.
Antimicrob Agents Chemother 2002; 46:62–8.
10. Troke P, Aguirrebengoa K, Arteaga C, et al. Treatment of scedosporiosis with voriconazole: clinical experience of 107 patients. Antimicrob
Agents Chemother 2008; 52:1723–50.
Correspondence: Christine Durand, MD, Broadway Research Building, Ste 871, 733 N
Broadway St, Baltimore, MD 21205 ([email protected]).
Clinical Infectious Diseases 2011;52(7):957–959
Ó The Author 2011. Published by Oxford University Press on behalf of the Infectious Diseases
Society of America. All rights reserved. For Permissions, please e-mail: journals.permissions
@oup.com.
1058-4838/2011/527-0001$37.00
DOI: 10.1093/cid/cir009
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