CASE REPORTS

Summary The three cases reported are similar radiographically and confirm the uniformly dismal prognosis of bilateral eventration when symptoms are presen' at birth. The cause is probably a defect in embryogenesis, although the roles of central and peripheral diaphragmatic innervation, and of congenital infection are still unclear. REFERENCES 1. Avnet NL: Roentgenologic features of congenital bilateral anterior diaphragmatic eventration. Am J Roentgen 88:743-750,

Oct 1962 2. Beck WC: Etiologic significance of eventration of the diaphragm. Arch Surg 60:1154-1160, Jun 1950 3. Berdon WE, Baker DH, Amoury R: The role of pulmonary hypoplasia in the prognosis of newborn infants with diaphragmatic hernia and eventration. Am J Roentgen 103:413421, Jun 1968 4. Bisgard JD, Robertson GE: Congenital eventration of the diaphragm. Surgical management. Am J Surg 70:95-99, Oct 1945 5. Bovornkitti S, Kangsadal P, Sangvichien S, et al: Neurogenic muscular aplasia (eventration) of the diaphragm. Am Rev Respir Dis 82:876-880, Dec 1960 6. Bremer JL: The diaphragm and diaphragmatic hernia. AMA Arch Pathol 36:539-549, Dec 1943 7. Briggs VA, Reilly BJ, Loewig K: Lung hypoplasia and membranous diaphragm in the congenital rubella syndrome-A rare case. J Can Assoc Radiol 24:126-127, Jun 1973 8. Bulgrin JG, Holmes FH: Eventration of the diaphragm with high renal ectopia-A case report. Radiology 64:249-251, Feb 1955 9. Butsch WL, Leahy LJ: A technique for the surgical treatment of congenital eventration of the diaphragm in infancy. J Thorac Surg 20:968-973, Dec 1950 10. Carter RE, Waterston DJ, Aberdeen E: Hernia and eventration of the diaphragm in childhood. Lancet 1:656-659, 31 Mar 1962 11. Christensen P: Eventration of the diaphragm. Thorax 14: 311-319, Dec 1959 12. Crastnopol P, Hochberg LA, Kroop IG: Surgical correction of eventration of diaphragm in patient with arthrogryposis. AMA Arch Surg 70:114-119, Jan 1955 13. DeBord, RA, Giunta EJ: Congenital eventration of the diaphragm. J Thorac Surg 31:731-736, Jun 1956 14. Firestone FN, Taybi H: Bilateral diaphragmatic eventration: demonstration by pneumoperitoneography. Surgery 62:954-957, Nov 1967 15. Garbaccio C, Gyopes MT, Fonkalsrud EW: Malfunction of the intact diaphragm in infants and children. Arch Surg 105: 57-61, Jul 1972 16. Greene W, Hunt CE: Paralysis of the diaphragm (abst). J Pediatr 84:913-914, Jun 1974 17. Jewett TC Jr, Thomson NB Jr: latrogenic eventration of the diaphragm in infancy. J Thorac Cardiovasc Surg 48:861-866, Nov 1964 18. Kenigsberg K, Gwinn JL: The retained sac in repair of posterolateral diaphragmatic hernia in the newborn. Surg 57: 894-897, June 1965 19. Landon JF: Eventration of the diaphragm. J Pediatr 8:

593-599, May 1936

20. Laxdal OE, McDougall H, Mellin GW: Congenital eventration of the diaphragm. N Engl J Med 250:401-408, 11 Mar 1954 21. Lee SS: Congenital eventration of the diaphragm in infancy. Nc Med J 31:9-13, Jan 1970 22. Lundstrom CH, Allen RP: Bilateral congenital eventration of the diaphragm-Case report with roentgen manifestations. AJR 97:216-217, May 1966 23. Meckel JF: In Zwanziger HL: De Hernia Diaphragmatica: Dessertatio Inauguralis Medico-Chirurgica. 1819, Halae, p 26 24. Mellins RB, Mays AP, Gold AP, et al: Respiratory distress as the initial manifestation of Werdnig-Hoffman disease. Pediatrics 53:3340, Jan 1974 25. Michelson E: Eventration of the diaphragm. Surgery 49:410422, Mar 1961 26. Paris F, Blasco E, Canto A, et al: Diaphragmatic eventration in infants. Thorax 28:66-72, Jan 1973 27. Patra BS, Vyas PN, Upadhyay RH: Surgery in eventration of diaphragm. Indian J Chest Dis 15:226-232, Jul 1973 28. Sakula J: Congenital eventration of the right half of the diaphragm (specimen). Proc R Soc Med 33:629, Aug 1940 29. Sethi G, Reed WA: Diaphragmatic malfunction in neonates and infants. Diagnosis and treatment. J Thorac Cardiovas Surg 62: 138-143, Jul 1971 30. Shah-Mirany J, Schmitz GL, Watson RR: Eventration of the diaphragm. Arch Surg 96:844-850, May 1968

31. Smith BT: Isolated phrenic nerve palsy in the newborn. Pediatrics 49:449-451, Mar 1972 32. State D: The surgical correction of congenital eventraticn of the diaphragm in infancy. Surgery 25:461-468, Mar 1949 33. Stauffer UG, Rickham PP: Acquired eventration of the diaphragm in the newborn. J Ped Surg 7:635-640, Dec 1972 34. Stephenson RH, Hopkins WA: Volvulus of the stomach complicating eventration of the diaphragm-Report of a case. Am J Gastro-enterol 41:225-234, Mar 1964 35. Thomas TV: Congenital eventration of the diaphragm. (Collective Review). Ann Thorac Surg 10: 180-192, Aug 1970 36. Thomas TV: Nonparalytic eventration of the diaphragm. J Thorac Cardiovasc Surg 55:586-593, Apr 1968 37. Wayne ER, Burrington JD, Meyers DN, et al: Bilateral eventration of the diaphragm in a neonate with congenital cytomegalic inclusicn disease. J Ped 83:164-165, Jul 1973 38. Wells LJ: Development of human diaphragm and pleural sacs. Contrib Embryol (Nos. 23141) 35:107-134, 1954

Refer to: Hirschfeld DS, Emilson BB: Mitral valve vegetation simulating left atrial myxoma. West J Med 124:419423, May 1976

Mitral Valve Vegetation Simulating Left Atrial

Myxoma DAVID S. HIRSCHFELD, MD BIANKA B. EMILSON, Tech Eng San Francisco

(Sweden)

THE ECHOCARDIOGRAPHIC appearance of vegetations in patients with endocarditis was first described by Dillon and associates in 1973.1 Several subsequent reports have substantiated the im-

portant role of echocardiography in the diagnosis of endocarditis involving the aortic valve.2-4 Considerably less has been written on the echocardio-

graphic pattern of mitral valve vegetations. We recently saw a patient with mitral valve endocarditis in whom findings on an echocardiogram initially suggested the presence of a left atrial myxoma. The subsequent disappearance of the

abnormal echoes concomitant with effective medical treatment of the endocarditis suggests that the mass of echoes noted represented a vegetation attached to the mitral valve. From the Medical Service, San Francisco General Hospital, and the Department of Medicine, University of California, San Francisco. Submitted July 18, 1975. Reprint requests to: David S. Hirschfeld, MD, c/o Editorial Office, San Francisco General Hospital, 1001 Potrero Avenue, San Francisco, CA 94110.

THE WESTERN JOURNAL OF MEDICINE

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CASE REPORTS

Report of a Case A 30-year-old man-who had a history of the intravenous use of drugs-was admitted to San Francisco General Hospital for treatment of a swollen arm. An area of tenderness had developed over the left forearm near the site of intravenous injections four months earlier. The area became red and swollen and two weeks before admission a draining ulcer was noted; chills and weight loss did not occur. The temperature was 103°F (39.4°C), heart rate 92 beats per minute and blood pressure 136/ 86 mm of mercury. The neck veins were not dis11/22/74 EKG

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Figure 1.-Echocardiogram on day 14. A dense mass of echoes appears behind the anterior mitral leaflet shortly after onset of diastole. Note flattening of E to F slope of anterior leaflet, especially evident in second and third complexes. Veg =vegetation, AM L =anterior mitral leaflet, PML= posterior mitral leaflet, IVS= interventricular septum, Endo =endocardium. 11/22/74

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was clear and the heart tones were normal with no audible murmur. The spleen could not be palpated. There were no petechiae. Cellulitis was noted to be present over the dorsal aspect of the left forearm with a central 1 cm

ulceration and surrounding induration without fluctuation. The hemoglobin value was 10 grams per 100 ml and leukocyte count 16,000 per cu mm with 80 percent neutrophils, 15 percent lymphocytes, 4 percent monocytes and 1 percent eosinophils. On analysis of the urine, there were three to four leukocytes per high power field and no red blood cells. Serum creatinine was 1.2 mg, albumin 3.1 grams and globulin 5.1 grams per 100 ml. No abnormalities were noted on an electrocardiogram and roentgenograms of the chest. Wound debridement was carried out and penicillin therapy, 10 million units daily, given intravenously, was begun. Cultures from the wound grew Serratia marcescens, Staphylococcus aureus and enterococcus. Two blood cultures were sterile. Low grade fever persisted. A soft apical holosystolic murmur was heard on day 8. Repeat cultures of blood from the femoral vein grew enterococcus. On day 9, an abscess at the wound site was incised and drained. The patient became afebrile on day 11, and the positive blood culture was thought to be contaminated. Administration of penicillin was discontinued. On day 14, splinter hemorrhages occurred, fever recurred and the apical murmur was louder. Echocardiography was carried out (see subsequent section). Treatment was initiated with ampicillin, 3 grams every four hours given orally, and streptomycin, 500 mg given intramuscularly twice daily; repeat blood cultures grew enterococcus. Echocardiography was repeated every two weeks. The patient became afebrile on day 18. The holosystolic apical murmur persisted unchanged. No gallop or symptoms of cardiac decompensation developed. The patient was discharged on day 53.

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Figure 2.-Echocardiogram on day 14 showing sweep from mitral valve up to aortic root. Abnormal echoes (\E) are present in left atrium in early systole (compare with Figure 5B). AoAW=anterior aortic wall, AoPW=posterior aortic wall, LA=left atrium; see Figure 1 for other abbreviations.

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MAY 1976 * 124 * 5

Echocardiograms A Unirad Series 100 echocardiograph with a 2.25 megaherz transducer focused at 7.5 cm was used, interfaced with an Electronics for Medicine DR8 Strip Chart Recorder. The initial echocardiogram obtained on day 14 showed a dense mass of echoes that appeared just behind the anterior leaflet of the mitral valve shortly after the onset of diastole but was not present during systole (Figure 1 ). A sweep up to the aortic root (Fig-

CASE REPORTS

ure 2) showed abnormal echoes in the left atrium in early systole. The appearance of these atrial echoes was more in keeping with the pattern of ruptured mitral chordae than that of a prolapsing tumor (see Figure 5). Repeat echocardiograms taken over the ensuing six weeks showed gradual diminution of the abnormal block of diastolic echoes (Figure 3). I1 / 2 2/l7 4

12/ 6/74

Discussion The long history of intravenous drug use, septic abscess, multiple positive blood cultures, splinter hemorrhages and changing pathologic murmur unequivocally indicated bacterial endocarditis in this patient. The echocardiogram supported the clinical impression of mitral valve involvement, but raised the possibility of a left atrial myxoma. I/ 6f75

12/16/74

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Figure 4.-Echocardiogram from 33-year-old woman with left atrial myxoma confirmed at surgical operation. A mass of parallel echoes appears behind the anterior mitral leaflet shortly after onset of diastole. Note flattened E to F slope of anterior leaflet (see Figure 5A). Myx=myxoma; see Figure 1 for other abbreviations. THE WESTERN JOURNAL OF MEDICINE

421

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left atrial myxoma the abnormal echoes appear "detached" from the valve. They also stressed the importance of demonstrating the presence of abnormal echoes in the left atrium when making the echocardiographic diagnosis of a left atrial myxoma. In the initial echocardiogram of our patient, the abnormal mass of echoes did indeed appear detached from the mitral valve. In addition, abnormal echoes were present in the left atrium, further suggesting the possibility of a myxoma. Probably the vegetation was, in fact, prolapsing through the mitral orifice. The pattern of the echoes in the left atrium (Figure 2) is identical to that which we have seen in two patients with endocarditis and ruptured chordae to the anterior mitral leaflet (Figure 5B). There is one other description in the literature to date of a mitral valve vegetation that mimicked echocardiographically a left atrial myxoma.18 In this case, the patient had underlying rheumatic mitral stenosis.

Endocarditis associated with an atrial myxoma is known to occur,5-7 one case occurring at our hospital.7 The progressive diminution in the size and intensity of the abnormal echoes that occurred concomitantly with effective antimicrobial therapy and the absence of evidence of further embolic episodes support the contention that the block of echoes represented a vegetation and not a left atrial myxoma. There have been many descriptions of the echocardiographic appearance of left atrial myxomas.8" Pathognomonic features are generally thought to be (1) the appearance soon after the onset of diastole of a mass of parallel echoes immediately posterior to the anterior mitral leaflet (Figures 4 and 5A), (2) flattening of the diastolic (E to F) slope of the anterior mitral leaflet, frequently accompanied by an obscuring of the posterior mitral leaflet and (3) appearance of abnormal echoes in the left atrium during systole, representing prolapse of the tumor through the mitral orifice. Dillon and co-workers' in describing the appearance of three infected mitral valves called attention to the fact that the abnormal echoes appear to be "attached" directly to the mitral valve leaflet(s), whereas in the case of a

Summary

Echocardiographic examination of a 30-yearold man with endocarditis involving the mitral valve showed a pattern suggestive of left atrial

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Figure 5.-A, echocardiogram from the same patient as in Figure 4 showing block of echoes in the left atrium representing a myxoma. B, echocardiogram from 35-year-old woman with ruptured chordae to anterior mitral leaflet confirmed at surgical operation. Arrow points to echoes in early systole thought to represent mitral valve or chordae projected into left atrium during ventricular contraction, or both. Note similarity to Figure 2. See Figures 1, 2 and 4 for abbreviations.

422

MAY 1976 *

124 *

5

LA

CASE REPORTS

myxoma. The abnormal echoes diminished dramatically during antibiotic therapy over the ensuing six weeks and probably represented a vegetation prolapsing through the mitral orifice. Echographic features considered pathognomonic for left atrial tumor may be shared by mitral valve vegetations. REFERENCES 1. Dillon JC, Feigenbaum H, Konecke LL, et al: Echocardiographic manifestations of valvular vegetations. Am Heart 3 86: 698-704, 1973 2. Hirschfeld D, Schiller NB: Echocardiographic localization of aortic valve vegetations. Circulation 50 (Suppl III) :IlI-143, 1974 3. Gottlieb S, Khuddus SA, Balooki H, et al: Echocardiographic diagnosis of aortic valve vegetations in Candida endocarditis. Circulation 50:826-830, 1974 4. Martinez EC, Burch GE, Giles TD: Echocardiographic diagnosis of vegetative aortic bacterial endocarditis. Am J Cardiol 34: 845-849, 1974 5. Dick HJ, Mullin EW: Myxoma of the heart complicated by bloodstream infection by Staphylococcus aureus and Candida parapsilosis. NY State J Med 56:856-859, 1956 6. Rae A: Two patients with cardiac myxoma-One presenting as bacterial endocarditis, and one as congestive cardiac failure. Postgrad Med J 41:644 648, 1965

Refer to: Bellville JW, Swanson GD, Miyake T, et al: Respiratory stimulation observed following ethanol ingestion. West J Med 124:423425, May 1976

Respiratory Stimulation Observed Following Ethanol Ingestion J. WELDON BELLVILLE, MD GEORGE D. SWANSON, PhD TOSHIYUKI MIYAKE, MD KAMEL A. AQLEH, BS Los Angeles ETHANOL IS REGARDED as a central nervous system depressant. Its sedative effect and its effects on psychomotor performance have been studied in detail. Recently its respiratory depressant effects have been shown to be dose related, that is, as the dose of ethanol is increased its respiratory depressant properties are enhanced.",2 This report summarizes our findings in a young man who has an unusual response to ethanol in that it produces respiratory stimulation and the degree of respiratory stimulation appears to be poorly related to the dose of ethanol ingested. From the Department of Anesthesiology, University of California, Los Angeles, School of Medicine. This study was supported in part by Grant No. DA00138, National Institutes of Health. Submitted July 18, 1975. Reprint requests to: J. Weldon Bellville, MD, Department of Anesthesiology, UCLA School of Medicine, Los Angeles, CA 90024.

7. Malloch CI, Abbott JA, Rapaport E: Left atrial myxoma with bacteremia-Report of a case with a bifid systolic apical impulse. Am J Cardiol 25:353-358, 1970 8. Popp RL, Harrison DC: Ultrasound for the diagnosis of atrial tumor. Ann Intern Med 71:785-787, 1969 9. Wolfe SB, Popp RL, Feigenbaum H: Diagnosis of atrial tumors by ultrasound. Circulation 39:615-622, 1969 10. Nasser WK, Davis RH, Dillon JC, et al: Atrial myxomaII. Phonocardiographic, echocardiographic, hemodynamic, and angiographic features in nine cases. Am Heart J 83:810-824, 1972 11. Spencer WH III, Peter RH, Orgain ES: Detection of a left atrial myxoma by echocardiography. Arch Intern Med 128:787-789, 1971 12. Schattenberg TT: Echocardiographic diagnosis of left atrial myxoma. Mayo Clin Proc 43:620-627, 1968 13. Martinez EC, Giles TD, Burch GE: Echocardiographic diagnosis of left atrial myxoma. Am J Cardiol 33:281-285, 1974 14. Kostis JB, Moghadam AN: Echocardiographic diagnosis of left atrial myxoma. Chest 58:550-552, 1970 15. Johnson ML, Sieker HO, Behar VS, et al: Echocardiographic diagnosis of a left atrial myxoma found attached to the free left atrial wall. J Clin Ultrasound 1:75-81, 1973 16. Srivastava TN, Fletcher E: The echocardiogram in left atrial myxoma. Am J Med 54:136-139, 1973 17. Finegan RE, Harrison DC: Diagnosis of left atrial myxoma by echocardiography. N Engl J Med 282:1022-1023, 1970 18. Spangler RD, Johnson ML, Holmes JH, et al: Echocardiographic demonstration of bacterigl vegetations in active infective endocarditis. J Clin Ultrasound 1:126-128, 1973

Methods These studies were done in a 25-year-old healthy man during the course of an investigation of the respiratory effects of pentobarbital, A9-tetrahydrocannabinol and ethanol. The basic method for measuring respiratory depression or stimulation has already been described by us previously3 and represents a modification of the rebreathing technique of Eckenhoff and co-workers4 except that the rebreathing reservoir is charged with 5 percent carbon dioxide in oxygen as suggested by Read.5 The volunteer subject was allowed his usual meals, but it was requested that coffee and tea as well as alcoholic beverages be omitted for the preceding 12 hours. The subject was fitted with a rubber metabolic mouthpiece and nose clip. While in a semireclining position, he breathed through the apparatus for from three to five minutes to become accustomed to it. Then the system was closed and rebreathing begun, continuing for 10 to 12 minutes. At the end of this time, his minute ventilation was at least 30 liters per minute. Simultaneously respiratory response curves were plotted automatically by an analog computer, and a point was plotted for each breath representing the end-expiratory carbon dioxide rate and the ventilation for that breath (Figure 1). This value was also stored in a PDP/8 computer (Digital Equipment Corporation) for analysis. Following the completion of the response curve determination, a least mean square line was fit and the intercept at 20 liters per minute and slope were printed out as well as THE WESTERN JOURNAL OF MEDICINE

423

Mitral valve vegetation simulating left atrial myxoma.

CASE REPORTS Summary The three cases reported are similar radiographically and confirm the uniformly dismal prognosis of bilateral eventration when s...
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