Unusual Case of Pyopneumothorax in Tennessee(from private LLMD group)

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Unusual Case of Pyopneumothorax in Tennessee

Souad S. Youssef, MD; Vijay Ramu, MD; Felix A. Sarubbi, MD

South Med J. 2005;98(11):1139-1141. ©2005 Lippincott & Wilkins

Posted 12/16/2005

Abstract and Introduction

Abstract

Rupture of a coccidioidal pulmonary cavity with subsequent pyopneumothorax

is a rare clinical event, even in areas endemic for coccidioidomycosis. Our

encounter with a patient diagnosed with this condition in northeast Tennessee

serves notice to clinicians that coccidioidomycosis is indeed a traveling

fungal disease, and practitioners must be alert to common and uncommon

manifestations of infection associated with this fungus. A literature review

pertaining

to coccidioidal pyopneumothorax revealed that patients usually present with a

recent onset of chest pain. Serologic testing and pleural fluid culture are

highly useful, and management includes surgical intervention with or without

antifungal therapy.

Introduction

Although coccidioidomycosis is considered endemic to the southwestern region

of the United States, it has also been referred to as the traveling fungal

disease,[1] as illness caused by Coccidioides spp has been recorded in

nonendemic parts of the United States and other countries. Certain

occupations such as long-distance truck driving, tourism, and military

assignments allow individuals from various sections of the country or other

parts of the world to come into contact with this soil-dwelling fungus, even

during brief visits to endemic areas.[1] In this regard, various

authors[1-5] have encouraged practicing clinicians to be alert to the

characteristics of Coccidioides spp infection, including both the usual and

unusual disease presentations. On rare occasions, coccidioidomycosis may

present as empyema with hydropneumothorax and this can pose a diagnostic

dilemma even for clinicians practicing in endemic areas.[6] Our interaction

with a Tennessee resident who had pyopneumothorax caused by Coccidioides

immitis encouraged us to review the literature pertaining to this unusual

entity and report the case, as it represents an excellent example of this

traveling fungal disease.

Case Report

A 70-year-old retired long-distance truck driver was admitted to a

City, Tennessee, hospital complaining of the sudden onset of shortness of

breath and sharp, right-sided chest pain. He reported no fever, sweats,

anorexia, weight loss, or hemoptysis. His medical history was positive for

coronary artery disease, hypertension, chronic obstructive pulmonary

disease, rheumatoid arthritis that required prednisone therapy for several

months, and a history of positive tuberculin skin test for which he had

received appropriate treatment. Social history was positive for previous

tobacco use. He was a retired long-distance truck driver, and his work

brought him to the southwest region of the United States on several

occasions. Chest radiography at the time of admission showed apical bullae

and a small right pneumothorax. His chest pain and shortness of breath

improved, and he was discharged after a few days without undergoing further

studies or interventions. Within 1 week's time, however, the patient's

right-sided chest discomfort recurred and he was readmitted to the hospital.

Physical examination revealed dullness to percussion and diminished breath

sounds over the right mid to lower lung field. Chest radiography now showed

a right hydropneumothorax and apical cystic changes. Complete blood count

revealed a hemoglobin of 13 g/dL, white blood cell count of 15,800/mm3, and

platelet count of 249,000/mm3; the chemistry panel was within normal range.

Chest CT scan demonstrated the right hydropneumothorax, as well as other

changes consistent with emphysema and cystic changes, particularly in the

right apex. A right-side chest tube was placed, and pleural fluid showed a

white blood cell count of 345/mm3 (62% mononuclear cells and 38%

polymorphonuclear neutrophils), glucose of 0 mg/dL, protein of 3.5 g/dL and

lactate dehydrogenase of 3,772 U/mL. Routine bacterial culture revealed no

growth, stains and cultures for acid-fast bacilli were negative, and fungal

cultures grew white, fluffy colonies within a few days. Microscopic

evaluation of the fungal culture showed chains of barrel-shaped

arthroconidia suggestive of Coccidioides spp. The fungal isolate was

forwarded to the Tennessee State Laboratory and to the Centers for Disease

Control and Prevention and it was confirmed as C immitis. The patient's

complement fixation serologic test for C immitis was negative, a Spherulin

skin test was positive at 15 mm, and pleural biopsy showed necrotizing

granuloma with foreign body giant cells and inflammatory exudates. The

patient received a 4-week course of amphotericin B, followed by a lengthy

course of itraconazole. He also underwent thoracotomy with decortication and

eventually recovered.

Discussion

Human infection associated with Coccidioides spp (C immitis and Coccidioides

posadasii) is endemic in the southwestern region of the United States, where

this dimorphic fungus can be recovered from soil samples from states ranging

from California to Texas. Most infections are the result of inhaling

arthroconidia and, in the majority of cases, the infections are subclinical

or very mild.[7] Patients with symptomatic pulmonary infection may

demonstrate nonspecific unilateral lung infiltrates as well as hilar

adenopathy and pleural effusion on chest radiography, and most of these

infections resolve without complication.[2] When associated with primary

pulmonary coccidioidomycosis, pleural effusions may occur with or without

evidence of parenchymal involvement. They are often unilateral and usually

clear rapidly and completely without complication.[8] On occasion, the

pulmonary Coccidioides infection may evolve into a cavitary lesion, and

these are usually solitary, thin-walled, and located in an upper lobe.[6]

Although coccidioidal pulmonary cavitation may be asymptomatic, some

patients will have chest pain, cough, or hemoptysis, and, in certain cases,

a mycetoma will develop within the cavity.[6] A limited number of patients,

approximately 2.6% of those with cavitary coccidioidomycosis, will have

spontaneous hydropneumothorax as a consequence of rupture of a subpleural,

thin-walled cavity with associated bronchopleural fistula.[6] In this

regard, we had an opportunity to provide care for an elderly man who resided

in rural northeast Tennessee and who presented with spontaneous

hydropneumothorax (case report). Infection caused by Coccidioides spp was

not listed among the possible causes for this event, and the positive result

of the pleural fluid culture was received with surprise. On further

questioning, the patient elaborated on his previous occupation as a truck

driver and stated that his driving regularly brought him to various states

in the southwest United States. He probably encountered the organism during

one of his forays into endemic territory, and he had subsequent development

of this unusual complication some years after the primary infection.

Conceivably, his use of corticosteroid medication for arthritis could have

contributed to the disease activity.

This unique case for our rural area in northeast Tennessee stimulated a

literature review (Table) that uncovered only one other case of cavitary

coccidioidomycosis with rupture that was diagnosed outside of the usual

endemic area.[9] Of these 31 well-described reported cases,[1,2,6,9-13] 29

were diagnosed in western states, and most of these (23 cases) were reported

by Cunningham and Einstein[10] from Bakersfield, California. The majority of

patients are young men, and only 22.6% had a documented history of pulmonary

coccidioidomycosis infection. Complement fixation serology can be very

helpful (positive in 29 of 30 tested), Coccidioidin skin test results were

variable, and pleural fluid or lung specimen cultures were usually positive.

There were no deaths attributed to the pulmonary infection. Management often

included surgical intervention, in which 26 patients underwent partial or

complete lobectomy with or without decortication. Fourteen patients also

received courses of amphotericin B and/or azole therapy. Since our patient

was receiving corticosteroid therapy, he was given a course of amphotericin

B followed by itraconazole.

In summary, important lessons were learned both from our personal experience

and from our literature review concerning hydropneumothorax as a

complication of pulmonary coccidioidomycosis infection. Perhaps most

notably, we can offer firm support to the designation of coccidioidomycosis

as the traveling fungal disease.[1] Clearly, various occupations,

recreational activities, and so forth, will allow individuals from distant

geographic areas to encounter Coccidioides during visits to endemic sites

and to possibly manifest symptoms of illness on their return to nonendemic

regions. Furthermore, the signs and symptoms associated with

coccidioidomycosis hydropneumothorax are nonspecific, and chest radiography

may initially only show pneumothorax, as in our case, followed by pleural

fluid collection on subsequent studies.[10] Diagnosis usually hinges on the

recovery of Coccidioides from pleural fluid, and management commonly

involves surgical intervention (closed-chest drainage, resection and

decortication).[10] With regard to the need for antifungal therapy,

Cunningham and Einstein[10] have suggested that amphotericin B is not

routinely indicated in patients with well-controlled disease who receive

prompt surgical intervention. These authors recommended the use of

amphotericin B therapy if there is a delay in surgical intervention, when

the cavity ruptures during the acute phase of infection, when there is

otherwise active residual disease, and when the patient has certain

concomitant risk factors and medical problems such as diabetes or a

bronchopleural fistula. However, with the availability of effective and less

toxic azole antifungal agents such as fluconazole and itraconazole, we

recognize that clinicians probably would choose to offer combined medical

and surgical therapies for patients with this unusual complication of

cavitary coccidioidomycosis.

Table. Coccidioidomycosis and Spontaneous Hydropneumothorax

References

Bayer AS, Yoshikawa TT, Galpin JE, et al. Unusual syndromes of

coccidioidomycosis: diagnostic and therapeutic considerations. Medicine

1976;55:131-152.

Edelstein G, Levitt RG. Cavitary coccidioidomycosis presenting as

spontaneous pneumothorax. Am J Roentgenol 1983;141:533-534.

Snyder LS, Galgiani JN. Coccidioidomycosis: the initial pulmonary infection

and beyond. Semin Respir Crit Care Med 1997;18:235-247.

Desai SA, Minai OA, Gordon SM, et al. Coccidioidomycosis in non-endemic

areas: a case series. Respir Med 2001;95:305-309.

Crum NF, Lederman ER, Stafford CM, et al. Coccidioidomycosis: a descriptive

survey of a reemerging disease: clinical characteristics and current

controversies. Medicine 2004;83:149-175.

CE, Beard RR, Saito MT. Pathogenesis of coccidioidomycosis with

special reference to pulmonary cavitation. Ann Intern Med 1948;29:623-655.

CE, Beard RR, Whiting EG, et al. Varieties of coccidioidal infection

in relation to the epidemiology and control of the disease. Am J Public

Health 1946;36:1394-1402.

Batra P. Pulmonary coccidioidomycosis. J Thorac Imaging 1992;7:29-38.

Scully RE, Mark EJ, McNeely WF, et al. Case records of the Massachusetts

General Hospital: case 21-1994. N Engl J Med 1994;330:1516-1522.

Cunningham RT, Einstein H. Coccidioidal pulmonary cavities with rupture. J

Thorac Cardiovasc Surg 1982;84:172-177.

Haber K, Freundlich IM. Spontaneous pneumothorax with unusual

manifestations. Chest 1974;65:675-676.

Dolan MJ, Lattuada CP, Melcher GP, et al. Coccidioides immitis presenting as

a mycelial pathogen with empyema and hydropneumothorax. J Med Veterinary

Mycol 1992;30:249-255.

Hyde L, Holman DC. Coccidioidal spontaneous hydropneumothorax. Ann Intern

Med 1957;47:1234-1242.

Sidebar: Key Points

Infection due to Coccidioides can be encountered outside the usual endemic

areas and clinicians must be aware of the clinical spectrum.

Rupture of a coccidioidal pulmonary cavity is a rare event and can result in

pyopneumothorax.

Treatment of pyopneumothorax due to Coccidioides requires surgical

intervention with or without antifungal therapy.

Reprint Address

Reprint requests to Dr. Felix A. Sarubbi, Department of Medicine, H.

Quillen College of Medicine, East Tennessee State University, City,

TN 37614. Email: felix.sarubbi@...

Souad S. Youssef, MD, Vijay Ramu, MD, and Felix A. Sarubbi, MD, H.

Quillen Veterans Affairs Medical Center and H. Quillen College of

Medicine, City, TN.

Disclosure: No financial support was obtained for this paper. The authors of

this paper have no commercial or proprietary interest in any drug, device,

or equipment mentioned in the article.