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Nonsurgical Treatment of Histoplasma Endocarditis Involving a Bioprosthetic Valve
Gross examination of the lungs revealed cavitary lesions in the anterior segment of the right lung explaining the bronchoscopic finding of bleeding arising from the anterior segment ofthe right upper lobe. Necrotizing cavitary lesions have been described in patients with P carinii pneumonia.s" but hemoptysis was not reported in those patients. We believe that lung destruction and cavity formation are the results of host overreaction rather than destruction caused by P carinii, as there were abundant macrophage infiltrations and only a few P carinii organisms in cavitary lesions. Activated macrophages are known to produce elastase leading to lung necrosis." Although she had a ten pack-year history of smoking, prior chest roentgenograms failed to reveal right upper lobe disease. We believe she was approaching the end of her human immunodeficiency virus disease course because of two prior episodes of P carinii pneumonia, although CD4 count was not done. Although the long-term prognosis of AIDS is poor, early recognition and specific treatment of P carinii pneumonia improves the short-term outcome. We conclude that P carinii pneumonia should be considered in the differential diagnosis of hemoptysis and pulmonary infiltrates in patients with AIDS or at risk of AIDS. Clinicians, therefore, should be aware of such unusual presentations to avoid a delay in diagnosis and management.
ACKNOWLEDGMENTS: The authors wish to thank Nonna Fernandes, Stephanie Hill, and Dawn Peters for assistance with the manuscript and photographs.
Nonsurgical Treatment of Histoplasma Endocarditis Involving a Bioprosthetic Valve* David S. Konawaty, M.D.;t M. john B. Stalker, M.D.;* and hter W Munt, M.D., F.C.C.P.§
Endocardial involvement associated with disseminated histoplasmosis has been infrequently documented, especially among patients with prosthetic valves. The therapeutic approach to these patients is also not yet clearly defined. A 54-year-old man with prosthetic valve endocarditis due to histoplasmosis was successfully treated with amphotericin B. A review of the literature suggests that the optimal form of therapy is likely a combination of surgical replacement of the involved valve and high dose amphotericin B. Successful therapy with amphotericin B alone may, however, be achieved if surgery is not a viable option. (Cheat 1991; 99:253-56)
E
ndocardial involvement as a form of disseminated histoplasmosis is reported to be the third most common cause of fungal endocarditis. 1 Nevertheless, it has been infrequently documented, with only 33 published cases to date,>" ofwhich only four cases have occurred in prosthetic valves.5.11.16.17 None of the four patients survived. We describe a successfully treated case of Histoplasma endocarditis involving a bioprosthetic valve, and review the published cases of Histoplasma endocarditis to date with emphasis on both therapeutic decisions and outcomes.
REFERENCES
CASE REpORT
Murray JF, Garay SM, Hopewell PC, Mills J, Snider G, Stover DE. Pulmonarycomplicationsof the acquired immunodeficiency syndrome: an update. Am Rev Respir Dis 1987;135:504-09 2 Engelberg LA, Lerner C\v, Tapper ML. Clinical features of Pneumocystis carinii in the acquired immune deficiency syndrome. Am Rev Respir Dis 1984;130:689-94 3 Crocco JA, Rooney Jl. Fankushen DS, Di Benedetto RJ, Lyons HA. Massivehemoptysis. Arch Intern Med 1968;121:495-98 4 Conlan AA, Hurwitz SS, Krige L, Nicolaou N, Pool R. Massive hemoptysis: review of 123cases. J Thorac CardiovascSurg 1983; 85:120-24 5 Eng RHK, Bishburg E, Smith SM. Evidence for destruction of lung tissues during Pneumocystis carinii infection. Arch Intern Med 1987; 147:746-49 6 Barrio JL, Suarez M, RodriguezJL, Saldana MJ, Pitchenik AE. Pneumocystis carinii pneumonia presenting as cavitating and noncavitating solitary pulmonary nodules in patients with the acquired immunodeficiency syndrome. Am Rev Respir Dis 1986; 134:1094-96 7 Liu YC, Tomashefski JF, Tomford \v, Green H. Necrotizing Pneumocystis carinii vasculitisassociated with lung necrosis and cavitation in a patient with acquired immunodeficiency syndrome. Arch Pathol Lab Med 1989;113:494-97 8 WerbZ, Gordon S. Elastasesecretion by stimulated macrophages, characteristics and regulation. J Exp Med 1975; 142:361-67
A54-year-old white man wasadmitted in June 1987with a threeweek history oflethargy,anorexia,headaches,and chills. Pasthistory included rheumatic heart disease with mitral stenosis, necessitating valve replacement with a bovine prosthesis in 1985. Temperature was 38.8·C. Dental caries were observed. Cardiac auscultation revealed an opening snap, a 2/6 systolic murmur and 1/6 diastolic rumble, both heard at the apex. There was tenderness in the right upper quadrant without a palpable liver. The spleen tip was palpable. Questionable neck stiffness was noted. The CBC revealed a hemoglobin value of 134 gIL and leukocyte count of 4.8 x 10"1L with 36 percent band cells.The ECG showed atrial fibrillation. The chest roentgenogram was normal apart from bilateral hiIar calcifications. Lumbar puncture results were normal apart from a glucose level of 1.8 mmollL (serum 6.4) and leukocyte count of 126 x 10"1L with 57 percent polymorphs, A cr scan of the head showed only bilateral periventricular calcifications. An echocardiogram performed the day of admission revealed a normal mitral valve prosthesis with no vegetations and mild aortic regurgitation. Blood cultures drawn on admission were negative. The presumptive diagnosis was culture-negative meningitis and endocarditis which wastreated with penicillin,netilmicin,and vancomycin. The patient ·From the Department of Medicine, Divisionsof Respiratory and Critical Care Medicine and General Internal Medicine, Queen's University, Kingston, Ontario, Canada. tFellow in Respiratory Medicine. *Associate Professorof Medicine. §Professorof Medicine. CHEST I 99 I 1 I JANUARY, 1991
253
Table I-Selected Characterimc. of34lbtientB with Histoplasma Endocarditis Case
Age
Sex
13
1 213
54 47
M M
313
59
4 13 5 13
55
6"
32 64
7 13 8 13
53 56
9"
35 NA
10"
Involved Location
Outcome
Fever, lethargy Fever, malaise
None None
Death Death
M F F M M M
Mouth ulcer Fever Fever Weakness, hypotension NA Fever, mouth ulcer
Death Death Death Death Death Death
M NA M F
Aortic Aortic Tricuspid Mitral
Fever, murmur NA Mouth ulcer Fever, weight loss, leg paresis
None None None None None Stilbamidine and dihydroxystilbamadine Oral amphotericin B (TD 7.4 g) None None Amphotericin B (TO 4.8 g)
F M
Aortic Mitral Mitral Aortic Mitral NA
Fever, chills Cough, night sweats, anorexia Weakness, weight loss Fever, weight loss, meningitis Fever, chills Fever, weight loss
60
13' 14 13
39
1513 1613 17' 1813
50 59 37
45
M M M M
19'
58
M
Aortic
20'
43
M
21"
61
M
Aortic prosthesis NA
22'
46
M
23" 24"
57 57
M M
25'"
48
F
26"
37
M
27'"
55
2813
59
Antifungal Therapy
Aortic Aortic, mitral Tricuspid Mitral Aortic Tricuspid Aortic Aortic
1113 1213
27
Initial Presentation
Aortofemoral prosthetic
None None None None Amphotericin B (TO 1.73 g) Amphotericin B (TD 1.49 g)
Death Death Death Cured (2 mof/u) Death Death
Fever, fatigue, systemic emboli Fever, fatigue
None
Death Death Death Cured (21 mof/u) Death
None
Death
Embolization
Amphotericin B (TD 4 g)
Fever
Amphotericin B (TD 7.5 g)
Cured (5 yr f/u) Death
Fatigue, fever, cough Fever, murmur
Amphotericin B (TD 2.3 g) Amphotericin B (TD 2.5 g) AVR, MVR Amphotericin B (TD 2.1 g) myxoma removed None
graft
M
Mitral Aortic, mitral Left atrial myxoma Aortic prosthesis Aortic
~ight loss, mouth ulcer
22
M
Aortic
Fever, fatigue, murmur
29"
59
M
Aortic
Mouth ulcer
30" 31'"
66 54
M M
Aortic Aortic
~ight loss,
32'"
25
F
33"
46
M
Mitral prosthesis (Starr Edward) Mitral, aortic bioprosthesis Mitral, bioprosthesis
Present case
Fever, chills Fever, meningitis
Embolization mouth ulcer Fever, chills
Fever Fever, lethargy
Amphotericin AVR Amphotericin AVR Amphotericin AVR Amphotericin Amphotericin AVR Amphotericin MVRX2
B (TD 4.0 g) B (TD 2.0 g) B (TD 2.0 g) B (TD 2.7 g) B (TO 3.4 g) B (TO >3.5 g)
Amphotericin B (TD 3.78 g) MVR,AVR Amphotericin B (TD 4.0 g)
Death Cured Cured (2 mo f/u) Death Cured NA Cured (24 mof/u) NA Death Cured (16 mo f/u) Death
Death Cured (29 mof/u)
·AVR is aortic valve replacement; flu, follow-up; MVR, mitral valve replacement; NA, not available; TD, total dose.
was afebrile on day three, and subsequently became impatient, discharging himself against advice nine days later.
254
Six weeks later, he was readmitted with increasing dyspnea on exertion, a 4 kg (9Ib) weight loss, and diarrhea. Temperature was Nonsurglcal TI8lItmenl at HislopIasma EndocardIlI8 (Kanawaty, SIaJIcer, Munt)
37.T'C. Cardiovascular examination was unchanged. His splenomegaly had now increased, and hepatomegaly was also noted. RepeatCBC nowrevealeda hemoglobin valueof 104gIL, leukocyte count of 1.9 x 10"1L with 25 percent band cells, platelet count of 88 x 10"1L, and toxic granulations on blood smear. The ECG and chest roentgenogram were unchanged. Repeat lumbar puncture was normal apart from an elevated protein of .74 gIL. Ophthalmologicassessmentdisclosed bilateral, punctate, multifocal choroiditis consistent with histoplasmosis or possibly bacterial endocarditis. A few days later, bone marrow cultures on hrain heart infusion and yeast phosphate agars returned positive for Histoplasma capsulatum. Intravenous amphotericin B wasinitiated and warfarin continued. Subsequently, a repeat echocardiogram revealed a 5 mm vegetationon the mitral valveprosthesis. Cultures of blood, urine, and sputum on brain heart infusion agar were negative for fungi . Histoplasma capsulatum complement fixation titer was positive at 1:1024 with an H band on immunodiffusion. Adrenal insufficiency was diagnosed and treated with predisone and /Iudrocortisone. Immunologic assessmentrevealeda Tif. of2:1 with an absoluteT, cell count was 194x 10"1L, suggesting a severe impairment in cellular immunity. Quantitativeimmunoglobulins were normal,and lIIV antibody negative. Surgicalconsultation suggested mitral valvereplacement to be a high risk procedure due to technical difficulties with the patient's previoussurgery. The patient elected not to pursue this option and wassubsequently treated with amphotericin B to a total dose of 4 .0 g via Hickman catheter, which was subsequently removed in October 1987. Follow-up echocardiograms showed the vegetations to be unchanged. He is alive and well at present with no evidence of recurrent Histoplasma infection. DISC USSION
Successful therapy of prosthetic valve endocarditis secondary to histoplasmosis has not been reported prior to the present case . The successful outcome of our case may be related to a higher total dose of amphotericin B, but the small number of reported cases does not allow firm conclusions in this regard. It is as well possible that the 29 month follow-up to date is too short, and evidence of recurrence may become evident with time. We, however, feel this to be unlikely as all cases of recurrence have occurred within one year of therapy. H ...... I• . 17 Characteristics of the 34 reported cases of Histoplasma endocarditis to date are summarized in Table 1. The disease appears to have a predilection for middle-aged men. Structures most frequently involved are the mitral valve in seven
*
.i->
cases (23 percent), aortic valve in 16 cases (47 percent), both valves in three cases (9 percent), and the tricuspid valve in two cases (6 percent). In approximately one half of cases the diagnosis was made antemortem . Initial lumbar puncture on our patient revealed a cerehrospinal fluid neutrophilia associated with a depressed glucose. Although no organism could be identified , we feel this was consistent with meningitis due to disseminated histoplasmosis. Unfortunately, no serologic studies were performed on the cerebrospinal fluid. The clinical course, cerebrospinal fluid findings, and good response to therapy in our patient is consistent with previous experience...... The finding of adrenal insufficiency in this case is of relevance . As has been previously noted," it is frequently associated with disseminated histoplasmosis and is the most common cause of mortality that goes unrecognized. Immunologic assessment in our patient revealed significantly impaired cellular immunity. We feel the basis of this to be related to the severity of the infection rather than to a primary or associated disease process, as none could be identified, and the patient has remained otherwise well in follow-up. Repeat immunologic assessment would have been informative in this regard, but unfortunately was not performed. A review of the therapeutic modalities (Fig 1) emphasizes the importance of correctly establishing the diagnosis and initiating therapy. Of the 17 patients who did not receive therapy, all died . Of those treated with amphotericin B alone, four were presumed cured and five have died. There is no significant difference in average amphotericin dose between those who have survived (3.6 g) and those who have died (4.3 g), even excluding patient 9 who died after receiving 7.4 g orally. There was a trend towards a survival advantage with amphotericin B combined with surgical intervention, with five cures reported among the seven cases of which the outcomes are known. To summarize, disseminated histoplasmosis complicated by endocarditis, especially if a prosthetic valve is involved, has a grave prognosis if untreated. Early diagnosis and therapy is therefore essential. The optimal form of therapy is unknown; however, the current literature suggests a combination of surgical intervention and amphotericin B. If surgery is not an option, cure may be achieved from high
"cases~
No treatment lZ•3•5•11 . 13) (17)
Amphotericin 8(4,6'8,13,14.
All died
4 cured
**
Prnent
Amphoteri~ip.
surgery(9.1
p,
.1 -1)
and (8)
case) (9)
5 died
5 cured
2 died
FIGURE 1. Patient outcomes based on treatment. Numbers in brackets indicate number of patients. Asterisk includescase 8 (Table 1);double asterisk, case 29 (Table 1),outcome unspecified . CHEST I 99 I 1 I JANUARY, 1991
255
dose amphotericin B therapy alone. REFERENCES
1
2 3 4 5 6 7
8 9
10
11
12 13 14 15
16 17
18
L. Pathogenesis of infectious endocorditis. In: Braunwald E, ed. Heart disease: a textbook of cardiovascular medicine, 3rd ed. Philadelphia: WB Saunders, 1988:1093-1134 Haust MD, W10dek GK, Parker Jo. Histoplasma endocarditis. Am J Med 1962;32:46().65 Gerber HJ, Schoonmaker F\v, VazquezMD. Chronic meningitis associated with Histoplasma endocarditis. N Eng! J Med 1966; 275:74-76 Hartley RA, Remsberg JRS, Sinaly NP. Histoplasma endocarditis. Arch Int Med 1967; 119:527-31 Weaver OK, Batsakis JG, Nishiyama RH, Arbor A. Histoplasma endocarditis. Arch Surg 1968;96:19-162 Segal C, Wheeler CG, Tompsett R. Histoplasma endocarditis cured with amphotericin. N Eng! J Med 1969; 28:206-07 Matthay RA, Levin DC, Wicks AB, Ellis JH Jr. Disseminated histoplasmosis involving an aortofemoral prosthetic graft. JAMA 1976; 235:1478-79 Canlas MS, Dillon MLJr. Histoplasma capsulatum endocarditis: report of a case following heart surgery. Angiology 1977;28:45463 Olive T, Lagier A, Dumas 0, Bidart JM, Bernard PM. Histoplasmose generalisse avec localisation laryngee et endocaroite. Nouvelle Presse Med 1978; 7:2262 Rogers EW, ~yman AE, Noble RJ, Bruins SC. Left atrial myxoma infected with Histoplasma capsulatum. Am J Med 1978;64:683-90 Alexander WJ, Mowry Rw, Cobbs CG, Dismukes WE. Prosthetic valve endocarditis caused by Histoplasma capsulatum. JAMA 1979; 242:1399-1400 Waterhouse G, Burney Of, Prager RL. Histoplasma capsulatum: endocarditis requiring aortic valve replacement for aortic insufficiency. South Med J 1980;73:683-84 Blair TP, Waugh RA, Pollack M, Ashworth HE, Young NA, Anderson SE, et aI. Histoplasma capsulatum endocarditis. Am Heart J 1980;99:783-88 Goodwin RA, Shapiro JL, Thurman GH, Thurman SS, Des Prez RM. Disseminated histoplasmosis: clinical and pathologic correlations. Medicine 1980; 59:1-31 Bradsher R\v, Wickre CG, Savage AM, Harston WE, Alford RH. Histoplasma capsulatum endocarditis cured by amphotericin B combined with surgery. Chest 1980; 78:5:791-95 Gaynes Rp, Gardner P, Causey W Prosthetic valve endocarditis caused by Histoplasma capsulatum. Arch Int Med 1981; 141: 1533-37 Svirbely JR, Ayers LW, Buesching WJ. Filamentous Histoplasma cap8tJatum endocarditis involving mitral and aortic valve porcine bioprostheses. Arch Pathol Lab Med 1985; 109:273-76 Salaki JS, Louria DB, Chmel H. Fungal and yeast infections of the central nervous system: a clinical review. Medicine 1984; 63:2:108-32 ~instein
An acute aortic dissection with rupture in the descending aorta led to suffusion of blood into the soft tissue of the neck with sufficient pressure to cause not only superior vena cava obstruction, but also tracheal obstruction. (Chut 1991; 99:256-58) RA = right atrium; LA = left atrium; PA= pulmonary artery; Th = thrombus. ...ortic dissection results from a tear in the aortic intima which allows blood to enter the aortic media and may then propagate distally and on occasion, proximally. Known risk factors for aortic dissection include hypertension, congenital abnormalities of the aortic valve, coarctation of the aorta, Marfans syndrome, other connective tissue disorders and iatrogenic causes. The clinical presentation of aortic dissection can be quite variable and is often related to alteration in end-organ blood supply. The most common symptom is chest pain which is present in nearly all cases. 1 Other common presenting features include syncope, dyspnea, neurologic impairment, aortic insufficiency, and left pleural effusion. Less frequent findings include vocal cord paralysis, Horner's syndrome, hemoptysis, hematemesis, heart block and, rarely, superior vena cava (SVC) syndrome. 1 The following report describes a patient with acute aortic dissection presenting with symptoms of upper airway obstruction.
.t1
CASE REPORT
The patient was a 65-year-old caucasian man who had previously been well. On the day of admission, the patient had sudden onset
Aortic Dissection Presenting as Upper Airway Obstruction* I\1terJ Giannoccaro. M.D.; Jean-Francois Marquis, M.D.; Kwan-Leung Chan, M.D.; Virginia WaUey, M.D.; and Rosemary J Chambers, M.B. *From the University of Ottawa Heart Institute, Ottawa, Canada.
256
FIGURE 1. Patient is intubated. Measuring tape across neck reveals degree of swelling.
AortIc DIsseclion ~ng as Upper AIrway Obstruction (GianflOCC8fO et at)
ACKNOWLEDGMENTS: The authors wish to thank Nonna Fernandes, Stephanie Hill, and Dawn Peters for assistance with the manuscript and photographs.
Nonsurgical Treatment of Histoplasma Endocarditis Involving a Bioprosthetic Valve* David S. Konawaty, M.D.;t M. john B. Stalker, M.D.;* and hter W Munt, M.D., F.C.C.P.§
Endocardial involvement associated with disseminated histoplasmosis has been infrequently documented, especially among patients with prosthetic valves. The therapeutic approach to these patients is also not yet clearly defined. A 54-year-old man with prosthetic valve endocarditis due to histoplasmosis was successfully treated with amphotericin B. A review of the literature suggests that the optimal form of therapy is likely a combination of surgical replacement of the involved valve and high dose amphotericin B. Successful therapy with amphotericin B alone may, however, be achieved if surgery is not a viable option. (Cheat 1991; 99:253-56)
E
ndocardial involvement as a form of disseminated histoplasmosis is reported to be the third most common cause of fungal endocarditis. 1 Nevertheless, it has been infrequently documented, with only 33 published cases to date,>" ofwhich only four cases have occurred in prosthetic valves.5.11.16.17 None of the four patients survived. We describe a successfully treated case of Histoplasma endocarditis involving a bioprosthetic valve, and review the published cases of Histoplasma endocarditis to date with emphasis on both therapeutic decisions and outcomes.
REFERENCES
CASE REpORT
Murray JF, Garay SM, Hopewell PC, Mills J, Snider G, Stover DE. Pulmonarycomplicationsof the acquired immunodeficiency syndrome: an update. Am Rev Respir Dis 1987;135:504-09 2 Engelberg LA, Lerner C\v, Tapper ML. Clinical features of Pneumocystis carinii in the acquired immune deficiency syndrome. Am Rev Respir Dis 1984;130:689-94 3 Crocco JA, Rooney Jl. Fankushen DS, Di Benedetto RJ, Lyons HA. Massivehemoptysis. Arch Intern Med 1968;121:495-98 4 Conlan AA, Hurwitz SS, Krige L, Nicolaou N, Pool R. Massive hemoptysis: review of 123cases. J Thorac CardiovascSurg 1983; 85:120-24 5 Eng RHK, Bishburg E, Smith SM. Evidence for destruction of lung tissues during Pneumocystis carinii infection. Arch Intern Med 1987; 147:746-49 6 Barrio JL, Suarez M, RodriguezJL, Saldana MJ, Pitchenik AE. Pneumocystis carinii pneumonia presenting as cavitating and noncavitating solitary pulmonary nodules in patients with the acquired immunodeficiency syndrome. Am Rev Respir Dis 1986; 134:1094-96 7 Liu YC, Tomashefski JF, Tomford \v, Green H. Necrotizing Pneumocystis carinii vasculitisassociated with lung necrosis and cavitation in a patient with acquired immunodeficiency syndrome. Arch Pathol Lab Med 1989;113:494-97 8 WerbZ, Gordon S. Elastasesecretion by stimulated macrophages, characteristics and regulation. J Exp Med 1975; 142:361-67
A54-year-old white man wasadmitted in June 1987with a threeweek history oflethargy,anorexia,headaches,and chills. Pasthistory included rheumatic heart disease with mitral stenosis, necessitating valve replacement with a bovine prosthesis in 1985. Temperature was 38.8·C. Dental caries were observed. Cardiac auscultation revealed an opening snap, a 2/6 systolic murmur and 1/6 diastolic rumble, both heard at the apex. There was tenderness in the right upper quadrant without a palpable liver. The spleen tip was palpable. Questionable neck stiffness was noted. The CBC revealed a hemoglobin value of 134 gIL and leukocyte count of 4.8 x 10"1L with 36 percent band cells.The ECG showed atrial fibrillation. The chest roentgenogram was normal apart from bilateral hiIar calcifications. Lumbar puncture results were normal apart from a glucose level of 1.8 mmollL (serum 6.4) and leukocyte count of 126 x 10"1L with 57 percent polymorphs, A cr scan of the head showed only bilateral periventricular calcifications. An echocardiogram performed the day of admission revealed a normal mitral valve prosthesis with no vegetations and mild aortic regurgitation. Blood cultures drawn on admission were negative. The presumptive diagnosis was culture-negative meningitis and endocarditis which wastreated with penicillin,netilmicin,and vancomycin. The patient ·From the Department of Medicine, Divisionsof Respiratory and Critical Care Medicine and General Internal Medicine, Queen's University, Kingston, Ontario, Canada. tFellow in Respiratory Medicine. *Associate Professorof Medicine. §Professorof Medicine. CHEST I 99 I 1 I JANUARY, 1991
253
Table I-Selected Characterimc. of34lbtientB with Histoplasma Endocarditis Case
Age
Sex
13
1 213
54 47
M M
313
59
4 13 5 13
55
6"
32 64
7 13 8 13
53 56
9"
35 NA
10"
Involved Location
Outcome
Fever, lethargy Fever, malaise
None None
Death Death
M F F M M M
Mouth ulcer Fever Fever Weakness, hypotension NA Fever, mouth ulcer
Death Death Death Death Death Death
M NA M F
Aortic Aortic Tricuspid Mitral
Fever, murmur NA Mouth ulcer Fever, weight loss, leg paresis
None None None None None Stilbamidine and dihydroxystilbamadine Oral amphotericin B (TD 7.4 g) None None Amphotericin B (TO 4.8 g)
F M
Aortic Mitral Mitral Aortic Mitral NA
Fever, chills Cough, night sweats, anorexia Weakness, weight loss Fever, weight loss, meningitis Fever, chills Fever, weight loss
60
13' 14 13
39
1513 1613 17' 1813
50 59 37
45
M M M M
19'
58
M
Aortic
20'
43
M
21"
61
M
Aortic prosthesis NA
22'
46
M
23" 24"
57 57
M M
25'"
48
F
26"
37
M
27'"
55
2813
59
Antifungal Therapy
Aortic Aortic, mitral Tricuspid Mitral Aortic Tricuspid Aortic Aortic
1113 1213
27
Initial Presentation
Aortofemoral prosthetic
None None None None Amphotericin B (TO 1.73 g) Amphotericin B (TD 1.49 g)
Death Death Death Cured (2 mof/u) Death Death
Fever, fatigue, systemic emboli Fever, fatigue
None
Death Death Death Cured (21 mof/u) Death
None
Death
Embolization
Amphotericin B (TD 4 g)
Fever
Amphotericin B (TD 7.5 g)
Cured (5 yr f/u) Death
Fatigue, fever, cough Fever, murmur
Amphotericin B (TD 2.3 g) Amphotericin B (TD 2.5 g) AVR, MVR Amphotericin B (TD 2.1 g) myxoma removed None
graft
M
Mitral Aortic, mitral Left atrial myxoma Aortic prosthesis Aortic
~ight loss, mouth ulcer
22
M
Aortic
Fever, fatigue, murmur
29"
59
M
Aortic
Mouth ulcer
30" 31'"
66 54
M M
Aortic Aortic
~ight loss,
32'"
25
F
33"
46
M
Mitral prosthesis (Starr Edward) Mitral, aortic bioprosthesis Mitral, bioprosthesis
Present case
Fever, chills Fever, meningitis
Embolization mouth ulcer Fever, chills
Fever Fever, lethargy
Amphotericin AVR Amphotericin AVR Amphotericin AVR Amphotericin Amphotericin AVR Amphotericin MVRX2
B (TD 4.0 g) B (TD 2.0 g) B (TD 2.0 g) B (TD 2.7 g) B (TO 3.4 g) B (TO >3.5 g)
Amphotericin B (TD 3.78 g) MVR,AVR Amphotericin B (TD 4.0 g)
Death Cured Cured (2 mo f/u) Death Cured NA Cured (24 mof/u) NA Death Cured (16 mo f/u) Death
Death Cured (29 mof/u)
·AVR is aortic valve replacement; flu, follow-up; MVR, mitral valve replacement; NA, not available; TD, total dose.
was afebrile on day three, and subsequently became impatient, discharging himself against advice nine days later.
254
Six weeks later, he was readmitted with increasing dyspnea on exertion, a 4 kg (9Ib) weight loss, and diarrhea. Temperature was Nonsurglcal TI8lItmenl at HislopIasma EndocardIlI8 (Kanawaty, SIaJIcer, Munt)
37.T'C. Cardiovascular examination was unchanged. His splenomegaly had now increased, and hepatomegaly was also noted. RepeatCBC nowrevealeda hemoglobin valueof 104gIL, leukocyte count of 1.9 x 10"1L with 25 percent band cells, platelet count of 88 x 10"1L, and toxic granulations on blood smear. The ECG and chest roentgenogram were unchanged. Repeat lumbar puncture was normal apart from an elevated protein of .74 gIL. Ophthalmologicassessmentdisclosed bilateral, punctate, multifocal choroiditis consistent with histoplasmosis or possibly bacterial endocarditis. A few days later, bone marrow cultures on hrain heart infusion and yeast phosphate agars returned positive for Histoplasma capsulatum. Intravenous amphotericin B wasinitiated and warfarin continued. Subsequently, a repeat echocardiogram revealed a 5 mm vegetationon the mitral valveprosthesis. Cultures of blood, urine, and sputum on brain heart infusion agar were negative for fungi . Histoplasma capsulatum complement fixation titer was positive at 1:1024 with an H band on immunodiffusion. Adrenal insufficiency was diagnosed and treated with predisone and /Iudrocortisone. Immunologic assessmentrevealeda Tif. of2:1 with an absoluteT, cell count was 194x 10"1L, suggesting a severe impairment in cellular immunity. Quantitativeimmunoglobulins were normal,and lIIV antibody negative. Surgicalconsultation suggested mitral valvereplacement to be a high risk procedure due to technical difficulties with the patient's previoussurgery. The patient elected not to pursue this option and wassubsequently treated with amphotericin B to a total dose of 4 .0 g via Hickman catheter, which was subsequently removed in October 1987. Follow-up echocardiograms showed the vegetations to be unchanged. He is alive and well at present with no evidence of recurrent Histoplasma infection. DISC USSION
Successful therapy of prosthetic valve endocarditis secondary to histoplasmosis has not been reported prior to the present case . The successful outcome of our case may be related to a higher total dose of amphotericin B, but the small number of reported cases does not allow firm conclusions in this regard. It is as well possible that the 29 month follow-up to date is too short, and evidence of recurrence may become evident with time. We, however, feel this to be unlikely as all cases of recurrence have occurred within one year of therapy. H ...... I• . 17 Characteristics of the 34 reported cases of Histoplasma endocarditis to date are summarized in Table 1. The disease appears to have a predilection for middle-aged men. Structures most frequently involved are the mitral valve in seven
*
.i->
cases (23 percent), aortic valve in 16 cases (47 percent), both valves in three cases (9 percent), and the tricuspid valve in two cases (6 percent). In approximately one half of cases the diagnosis was made antemortem . Initial lumbar puncture on our patient revealed a cerehrospinal fluid neutrophilia associated with a depressed glucose. Although no organism could be identified , we feel this was consistent with meningitis due to disseminated histoplasmosis. Unfortunately, no serologic studies were performed on the cerebrospinal fluid. The clinical course, cerebrospinal fluid findings, and good response to therapy in our patient is consistent with previous experience...... The finding of adrenal insufficiency in this case is of relevance . As has been previously noted," it is frequently associated with disseminated histoplasmosis and is the most common cause of mortality that goes unrecognized. Immunologic assessment in our patient revealed significantly impaired cellular immunity. We feel the basis of this to be related to the severity of the infection rather than to a primary or associated disease process, as none could be identified, and the patient has remained otherwise well in follow-up. Repeat immunologic assessment would have been informative in this regard, but unfortunately was not performed. A review of the therapeutic modalities (Fig 1) emphasizes the importance of correctly establishing the diagnosis and initiating therapy. Of the 17 patients who did not receive therapy, all died . Of those treated with amphotericin B alone, four were presumed cured and five have died. There is no significant difference in average amphotericin dose between those who have survived (3.6 g) and those who have died (4.3 g), even excluding patient 9 who died after receiving 7.4 g orally. There was a trend towards a survival advantage with amphotericin B combined with surgical intervention, with five cures reported among the seven cases of which the outcomes are known. To summarize, disseminated histoplasmosis complicated by endocarditis, especially if a prosthetic valve is involved, has a grave prognosis if untreated. Early diagnosis and therapy is therefore essential. The optimal form of therapy is unknown; however, the current literature suggests a combination of surgical intervention and amphotericin B. If surgery is not an option, cure may be achieved from high
"cases~
No treatment lZ•3•5•11 . 13) (17)
Amphotericin 8(4,6'8,13,14.
All died
4 cured
**
Prnent
Amphoteri~ip.
surgery(9.1
p,
.1 -1)
and (8)
case) (9)
5 died
5 cured
2 died
FIGURE 1. Patient outcomes based on treatment. Numbers in brackets indicate number of patients. Asterisk includescase 8 (Table 1);double asterisk, case 29 (Table 1),outcome unspecified . CHEST I 99 I 1 I JANUARY, 1991
255
dose amphotericin B therapy alone. REFERENCES
1
2 3 4 5 6 7
8 9
10
11
12 13 14 15
16 17
18
L. Pathogenesis of infectious endocorditis. In: Braunwald E, ed. Heart disease: a textbook of cardiovascular medicine, 3rd ed. Philadelphia: WB Saunders, 1988:1093-1134 Haust MD, W10dek GK, Parker Jo. Histoplasma endocarditis. Am J Med 1962;32:46().65 Gerber HJ, Schoonmaker F\v, VazquezMD. Chronic meningitis associated with Histoplasma endocarditis. N Eng! J Med 1966; 275:74-76 Hartley RA, Remsberg JRS, Sinaly NP. Histoplasma endocarditis. Arch Int Med 1967; 119:527-31 Weaver OK, Batsakis JG, Nishiyama RH, Arbor A. Histoplasma endocarditis. Arch Surg 1968;96:19-162 Segal C, Wheeler CG, Tompsett R. Histoplasma endocarditis cured with amphotericin. N Eng! J Med 1969; 28:206-07 Matthay RA, Levin DC, Wicks AB, Ellis JH Jr. Disseminated histoplasmosis involving an aortofemoral prosthetic graft. JAMA 1976; 235:1478-79 Canlas MS, Dillon MLJr. Histoplasma capsulatum endocarditis: report of a case following heart surgery. Angiology 1977;28:45463 Olive T, Lagier A, Dumas 0, Bidart JM, Bernard PM. Histoplasmose generalisse avec localisation laryngee et endocaroite. Nouvelle Presse Med 1978; 7:2262 Rogers EW, ~yman AE, Noble RJ, Bruins SC. Left atrial myxoma infected with Histoplasma capsulatum. Am J Med 1978;64:683-90 Alexander WJ, Mowry Rw, Cobbs CG, Dismukes WE. Prosthetic valve endocarditis caused by Histoplasma capsulatum. JAMA 1979; 242:1399-1400 Waterhouse G, Burney Of, Prager RL. Histoplasma capsulatum: endocarditis requiring aortic valve replacement for aortic insufficiency. South Med J 1980;73:683-84 Blair TP, Waugh RA, Pollack M, Ashworth HE, Young NA, Anderson SE, et aI. Histoplasma capsulatum endocarditis. Am Heart J 1980;99:783-88 Goodwin RA, Shapiro JL, Thurman GH, Thurman SS, Des Prez RM. Disseminated histoplasmosis: clinical and pathologic correlations. Medicine 1980; 59:1-31 Bradsher R\v, Wickre CG, Savage AM, Harston WE, Alford RH. Histoplasma capsulatum endocarditis cured by amphotericin B combined with surgery. Chest 1980; 78:5:791-95 Gaynes Rp, Gardner P, Causey W Prosthetic valve endocarditis caused by Histoplasma capsulatum. Arch Int Med 1981; 141: 1533-37 Svirbely JR, Ayers LW, Buesching WJ. Filamentous Histoplasma cap8tJatum endocarditis involving mitral and aortic valve porcine bioprostheses. Arch Pathol Lab Med 1985; 109:273-76 Salaki JS, Louria DB, Chmel H. Fungal and yeast infections of the central nervous system: a clinical review. Medicine 1984; 63:2:108-32 ~instein
An acute aortic dissection with rupture in the descending aorta led to suffusion of blood into the soft tissue of the neck with sufficient pressure to cause not only superior vena cava obstruction, but also tracheal obstruction. (Chut 1991; 99:256-58) RA = right atrium; LA = left atrium; PA= pulmonary artery; Th = thrombus. ...ortic dissection results from a tear in the aortic intima which allows blood to enter the aortic media and may then propagate distally and on occasion, proximally. Known risk factors for aortic dissection include hypertension, congenital abnormalities of the aortic valve, coarctation of the aorta, Marfans syndrome, other connective tissue disorders and iatrogenic causes. The clinical presentation of aortic dissection can be quite variable and is often related to alteration in end-organ blood supply. The most common symptom is chest pain which is present in nearly all cases. 1 Other common presenting features include syncope, dyspnea, neurologic impairment, aortic insufficiency, and left pleural effusion. Less frequent findings include vocal cord paralysis, Horner's syndrome, hemoptysis, hematemesis, heart block and, rarely, superior vena cava (SVC) syndrome. 1 The following report describes a patient with acute aortic dissection presenting with symptoms of upper airway obstruction.
.t1
CASE REPORT
The patient was a 65-year-old caucasian man who had previously been well. On the day of admission, the patient had sudden onset
Aortic Dissection Presenting as Upper Airway Obstruction* I\1terJ Giannoccaro. M.D.; Jean-Francois Marquis, M.D.; Kwan-Leung Chan, M.D.; Virginia WaUey, M.D.; and Rosemary J Chambers, M.B. *From the University of Ottawa Heart Institute, Ottawa, Canada.
256
FIGURE 1. Patient is intubated. Measuring tape across neck reveals degree of swelling.
AortIc DIsseclion ~ng as Upper AIrway Obstruction (GianflOCC8fO et at)