Solitary Pulmonary Nodule Due to Phycomycosis (Mucormycosis)

GALE, KLEITSCH diastolic volume. After replacement of the tricuspid valve an increase in QRS voltages is apparent on several published electrocardiograms.i~8 The rising pulmonary artery pressure during late diastole was an unusual finding in our patient. Provided that a pulmonary valve is competent, the usual fall in a pulmonary arterial pressure after pulmonary valve closure must result from forward flow of blood into the pulmonary capillary and venous systems. For the pressure in the pulmonary artery to rise during diastole, both a significant resistance to this forward flow as well as a volume displacement into the pulmonary arterial system must be present. The mean pulmonary wedge pressure of 8 mrn H g would constitute resistance to forward flow and we suspect that this level may well have been lower but for the "restrictive" effect of the stretched normal pericardium. A volume displacement would be provided by the billowing of the pulmonary leaflets upwards into the lumen of the main pulmonary artery followed by the forward flow of blood from the right ventricle after the diastolic opening of the valve as the right ventricular diastolic pressure exceeded that in the pulmonary artery. Parry and Abrahamslo and Somers and co-workersl1 have observed similar pulmonary pressures in severe endomyocardial fibrosis of the right ventricle and appreciated that the pulmonary valve must open in late diastole. Because of the relatively small number of documented cases of isolated tricuspid incompetence the long-term prognosis is uncertain, but we favor the contention of Morgan and Forkefl and that of Croxson and colleagueslP that the condition is relatively benign and that long-term survival without surgery should be possible. Both these groups have reported patients with posttraumatic tricuspid insufficiency who were alive and relatively asymptomatic after more than 30 years. If we were confident that a successful valvuloplasty, as described by Croxson and c o - w o r k e r ~ ,could ~~ be performed in our patient, there might be good reason to refer her to surgery now. However, this is unpredictable and we consider that insertion of a prosthetic valve is probably not justified at present. ACKNOWLEDGMENTS: We thank Professor H. C. Seftel under whose care our patient was treated, and Dr. H. M. Salmon, Superintendent of the Johannesburg General Hospital, for permission to publish.

1 Bain RC, Edwards JE, Scheifley CH, et al: Right-sided bacterial endocarditis and endarteritis: a clinical and pathologic study. Am J Med 24:98-109, 1958 2 Ramsey RG, Gunnar RM, Tobin JR: Endocarditis in the drug addict. Am J Cardiol 25:608-618, 1970 3 Glancy DL, Marcus FI, Caudra M, et al: Isolated organic tricuspid valvular regurgitation: causes and consequences. Am J Med 46:989-996, 1969 4 Morgan JR, Forker AD: Isolated tricuspid insufficiency. Circulation 43:559-564, 1971 5 Brandenburg RO, McCoon DC, Campeau L, et al: Traumatic rupture of the chordae tendineae of the tricuspid valve: successful repair 24 years later. Am J Cardiol

18:911-915, 1966 6 Butteworth JS, Chassin MR, McGrath R, et al: Cardiac Auscultation including Audio-visual Principles. New York. Grune and Stratton, 1960, p 28 7 Shabetai R, Adolph RJ, Spencer FC: Successful replacement of the bicuspid valve 10 years after traumatic incompetence. Am J Cardiol 18:916-920, 1966 8 Salzer J, Weintraub R, Lower R, et al: Isolated tricuspid insufficiency: report of a case with valve replacement. Am J Cardiol 18:921-927, 1966 9 Ishikawa K, Berson AS, Pipberger HV: Electrocardiographic changes due to cardiac enlargement. Am Heart J 81:635-643, 1971 10 Parry EHD, Abrahams DC:The function of the heart in endomyocardial fibrosis of the right ventricle. Br Heart J 25:619-629, 1963 11 Somers K, Brenton DP, D'Arbela PG, et al: Haemodynamic features of severe endomyocardial fibrosis of right ventricle, including comparison with constrictive pericarditis. Br Heart J 30:322-332, 1968 12 Croxson MS, O'Brien KP, Lowe JB: Traumatic tricuspid regurgitation: long-term survival. Br Heart J 33:750-755, 1971

Solitary Pulmonary Nodule Due to Phycornycosis ( ~ u c o r m ~ c o s i s ) * Adelito M . Cab, M . D . , F.C.C.P." and William P. Kleitsch, M . D . , F.C.C.P.?

The case of an asymptomatic, solitary nodule appearing in the lung of a diabetic hospitalized for arteriosclerosis obliterans is presented The nodule was found to be due to Phycomycoses of the Mucoraceae strain and is the fourth case reported of pulmonary disease due to this organism to recover after treatment. It is the only instance known of thii disease appearing as a solitary nodule and emphasizes the fact that the possible etiology of such nodules is endless, and cytologic and bacteriologic identification of them remains important.

T

he need for surgical excision to establish the diagnosis of solitary pulmonary nodules has been repeatedly emphasized. This suggestion has been received with varying degrees of resistance for several reasons. Some argue that the surgical hazards in a population group in the sixth, seventh and eighth decade with a limited survival potential outweigh the risks of malignancy. Most objections to thoractomy for diagnosis of solitary nodules revolve about the malignant vs nonmalignant controversy. This has generated considerable heat and furor and has practically obscured the fact that solitary pulmonary nodules undiagnosed are still undiagnosed whether benign or malignant. Rational management of disease requires a diagnosis, cytologic and 'From the Thoracic Surmcal Service. Veterans Administration Hospital, Phoenix, Arizona. ' '"Thoracic Surgeon (now in San Diego, California). Khief, Surgical Service. Reprint requests: Dr. Kbitsch, VA Hospital, Phoenix 85012

CHEST, VOL. 62, NO. 6, DECEMBER, 1972

SOLITARY PULMONARY NODULE DUE TO PHYCOMYCOSIS

FIGURE 1. Roentgenogram taken in 1965 showing post-thoracotomy changes. bacteriologic, in order to select the proper treatment regimen. This is as important for benign disease as for malignant. The case herein reported is an illustrative one of a solitary pulmonary nodule resulting from a rare disease caused by a fungus - Phycomycetes of the Mucoraceae strain. Although the organism is generally considered a saphrophyte. it can cause a fatal disease unless the course of the pathologic process can be halted.

'

A 48-year-old Mexican-American was admitted to the hospital September 30, 1989, for the treatment of intermittent claudication with which he had suffered for four years and which had become increasingly severe. At the time of admis-

FIGURE 2. Solitary pulmonary nodule appearing in right midlung field in 1969.

CHEST, VOL. 62, NO. 6, DECEMBER, 1972

FIGURE 3. Planigram outlining the nodule. sion, he claimed inability to walk as for as one-half block. He is a diabetic with generalized arteriosclerosis and a history of myocardial infarction with residual angina pectoris. In 1953, he underwent a right lower lobectomy for bronchiectasis. On examination, pulses in the right leg were absent and were markedly diminished in the left. In the process of preoperative evaluation for peripheral vascular surgery, a solitary nodule was discovered in the upper lobe of the right lung. After review of prior roentgenograms, it was discovered that the nodule had been present for at least five months. The usual diagnostic maneuvers were unrewarding and on November 13, 1969, a resection of the anterior segment of the right upper lobe was carried out. Histopathologic study of the nodule revealed it to be an ischemic pulmonary infarct due to arterial invasion and occlusion by a Phycomycetes organism

FIGURE 4. LOWpower magnification of a histologic slide of the surgical specimen showing ischemic necrosis and the numerous hyphae of mucormycosis.

GALE, KLEITSCH vessels as found by histopathologic examination. Since the patient's eyes were somewhat prominent, a detailed neurologic and rhinologic examination was carried out with negative results. Since his original complaint was due to arterial insufficiency in the legs, careful search of the endarterectomized material was made because of the demonstrated tendency of this infection to involve blood vessels. Here again, the search was unrewarding and the only pathologic process found was arteriosclerosis. It was decided, therefore, that the pulmonary disease was indeed the primary site and that the infection was acquired by inhalation. Whether his occupation as the driver of a garbage truck was the contributing factor is conjectural since reportedly the organisms are easily cultured out of the air. Diabetes mellitus as a contributing factor in this infection has been well established. He was last seen in August, 1971, when he was treated for diabetic neuropathy. There was no evidence of further pulmonary disease.

a

FIGURE 5. A histologic view illustrating thrombosis of a vessel with invading hyphae in the thrombus.

of the Mucoraceae strain. Postoperative convalescence waq uncomplicated. In February, 1970, he was again admitted to the hospital for treatment of his peripheral vascular disease and on March 16, 1970, an endarteredomy of the distal aorta with bilateral, aorto-femoral, Dacron by-pass prosthesis and bilateral lumbar sympathectomy was accomplished. He was hospitalized again in July, 1970, for a right femoral-popliteal by-pass with a reversed saphenous vein graft which was accomplished on September 3, 1970. He withstood both procedures without difficulty and enjoyed some improvement in the circulation of his legs. After the nature of the pulmonary nodule had been ascertained, strenuous efforts were made to definitely establish the primary site of infection since it was believed that a metastatic origin of the pulmonary disease could not be excluded. especially in view of the prominent involvement of the blood

Considering the rarity of phycomycetous infections, a remarkably voluminous literature of the subject has been developed with several excellent and detailed historical reviews. Only one case presenting as a solitary pulmonary nodule has been reported previously1 and this was subsequently shown to be, in fact, a case of coccidioidomycosis. This case then is the only known instance of phycomycetosis occurring as a solitary pulmonary nodule which has been reported in the medical literature. The reasons why these ubiquitous saphrophytic organisms become pathogenic has been the cause of speculation. Certainly, they prosper best in a weak and debilitated host. Progress in therapetuics which pennits the prolonged survival of chronically ill persons, alteration of the bacteriologic population by antimicrobials, and the suppression of the immune response by the corticosteroids all play a role in permitting ordinarily harmless microorganisms to assume a lethal potential. Since 1900, 19 cases of pulmonary phycomycetosis involving the lung alone have been reported, and only three patients have s u r ~ i v e d . ~The - ~ patient here presented represents the fourth survivor of pulmonary mucormycosis. All survivors were treated by surgical excision. The fatal cases were usually terminal events in malignant or other serious conditions. It appears that if the complicating disease can be reversed or controlled, the phycomycetous infection will also be controlled. Surgical excision, when feasible, is the

Table 1 4 u l m o n u r y Anthor/Year

Age Sex

Hlakenlxrg and Verhoeff,' 1959

23

O

Dillon, Scaly and Fetter,V958'

20

O

Race

Locale

N

No Carolina

Antibiotics

No Carolina

Severe diabetes Lung abscess bilobectomy RML & RLL

No Carolina

Severe diabeh

Middle lobe syndrome, lobectomy, RML

Arizona

Mild diabetes

Solitary nodule, segmental resection, anterior RUL

Dillon and Scaly,' 1962 Gale and Kleitec~h.1971

48

d

Mex Am

Associated Factor

Pathology and Treatment Lobar necrosis, bilobectomy RML & RLL

CHEST, VOL. 62, NO. 6, DECEMBER, 1972

CONGENITAL SUPERIOR VENA CAVA ANEURYSM best form of treatment because of the associated tissue infarction caused by invasion of vessel walls and thrombotic occlusion of their lumina.

1 Murphy JD, Bornstein S: Mucormycosis of the lung. Ann Intern Med 33:442, 1950 2 Blaknberg HW, Verhoeff D: Mucormycosis of the lung. Ann Rev Resp Dis 79:357, 1959 3 Dillion ML, Sealy WC, Fetter BF: Mucormycosis of the bronchus successfully treated by lobectomy. J Thorac Cardiovasc Surg 35:464,1958 4 Dillon ML, Sealy WC: Surgical aspects of opportunistic fungus infections. Lab Invest 11:1231,1982

Physical Eurmination Her blood pressure was 110/50, pulse rate-90/min, respirations, U)/min, temperature, 100.8° F (oral). Essential features of the physical examination revealed the skin to be covered with a maculopapular rash. Cervical nodes of 1 to 1.5 cm were palpated in the anterior and posterior hiangles of the neck. No other nodes were palpable. The thyroid was normal. The chest revealed dullness to percussion over the right base and limited movement because of splinting on the same side. Diminished breath sounds and crepitant rales were present over the right lower lung field. Decreased vocal fremitus was found over the same area. The left chest was normal. The heart was normal. Abdominal palpation revealed the liver 3 cm below the right costal margin and the spleen tip was just palpable. All pulses were normal and the nuerologic examination was physiologic.

Congenital Superior Vena Cava

Course in the Hospital

Aneurysm with Complications Caused

Hemogram gave normal findings. The differential showed polymorphonuclear cells 42, atypical lymphocytes 13, lymphocytes 41, monocytes 1, and eosinophiles 3. The atypical lymphocytes were of the Downey cell type. Sedimentation rate-68 mm per one hour Westergren. Mono test-positive; heterophile titer-1:448; guinea pig kidney absorption-1:224; beef RBC-negative; SCOT-225 mIV; Alkaline phosphatase-167 mIV; LDH-400 mIV; cephalin flocculation 24 hours3 plus, 48 hours3 plus; albumin-3.5 gm; globulin-3.0 gm. Thymol turbidity, total bilirubin, fasting blood sugar, BUN, uric acid, cholesterol, calcium, phosphorus, urinalysis, febrile agglutinins, sputum culture, intermediate Mantoux, VDRL, were either within normal limits or negative. Chest x-ray fil-ns (Fig 1A and 1B) show a large anterior superior mediastinal mass, associated with a right pleural effusion. Tomography in the anteroposterior and lateral views gave no additional information. The esophagus was not displaced, and the trachea is in the midline. Barium swallow was normal. An 1311 thyroid uptake and scan was normal, as was an IVU. Liver scan revealed a normal sized liver and an enlarged spleen. A radioactive isotope angiogram suggested the mass might be vascular, but loculated fluid could not be ruled out. Needle puncture of the mass was therefore contraindicated. Repeated electrocardiograms were negative. A thoracentesis was done, but only 10 ml of a deep strawcolored fluid could be obtained. The protein content was 4.6 gm. The fluid revealed 270 WBC per cu mm. Several atypical lymphocytes of the infectious mononucleosis type were seen. Cultures were negative for bacteria and fungi. The question of surgical exploration arose. The medical service argued against this for the following reasons. Infectious mononucleosis had been proved and the patient was improving. Although ECG's were negative, myocarditis is always a distinct possibility in such cases and this, together with proved liver abnormalities, makes for increased anesthesia risk. The nature of the mass had not been proved and it was believed this should be ascertained before surgery. A two week waiting period was suggested at the end of which time, if the mass persisted, angiography would be done. The patient was transferred to the surgical service on the seventh hospital day at the request of her parents and a thoracotomy was performed the following day. Upon opening the right chest wall a large mass presented which seemed attached to the mediastinum; the mass was palpated carefully. It did not pulsate and felt semi-solid and not like a lymphoma. All lobes of the right lung revealed multiple

b y Infectious Mononucleosis Charles R. Ream, M.D., F.C.C.P.' and Anthony Giardina, M.D.O '

i

This is the fifth case report in the Englisb literature of a superior vena cava aneurysm. The case was complicated by the fact the patient had infectious mononucleo$s with a superimposed thrombophlebitis of the aneurysm caused by the infectious mononucle08is. Pulmonary emboli and infarctions resulted from the thrombus and complications of the process resulted in her death. In the four previous cases no mural thrombi were described, nor did these patients experience pulmonary emboli or pulmonary infarction.

A

20-year-old white woman college student was admitted to the hospital medical service for the &st time in April 1970. The chief complaint consisted of fever of 14 days duration, pleuritic type pain in the right chest and cough of seven days duration, and a generalized body rash for six days. The patient's illness began with fever in the range of 101103' F (oral). Malaise was experienced, and seven days before admission she developed sharp pain in the right chest stabbing in nature, aggravated by deep inspiration, and associated with a dry nonproductive cough. Early in the course of her illness she was seen by a physician who made a diagnosis of pneumonia and prescribed ampicillin. She failed to improve and six days after the antibiotic was started she developed a diffuse body rash which was maculopapular in type. Ampicillin was stopped. The patient failed to improve and was referred for admission to the medical service. Past history and system reyiew were noncontributory. The patient was athletic and participated actively in sports. No injuries had occurred. Childhood illnesses were denied, Mantoux test had always been negative, and a chest x-ray film had never been taken. 'Chairman, Department of Medicine, Medical Center, Princeton, New Jersey. "Associate Pathologist, St. Elizabeth Hospital, Elizabeth, New Jersey.

CHEST, VOL. 62, NO. 6, DECEMBER, 1972