DISCUSSION

Phrenomediastinal echinococcosis is the term coined to describe hydatid cysts that arise in the subphrenic space" or diaphragm• and expand into the mediastinum . Although Praderi et al3 described such a transdiaphragmatic transgression of an echinococcal cyst 33 years ago, this entity has not since been reported. Even large series on hydatid disease do not make any mention of such a presentation of posterior mediastinal echinococcosis. 5 •6 Hydatid disease most frequently involves the liver and lungs and one of these organs is usually involved if a cyst is present anywhere else in the body. 5 Retroperitoneal cysts are almost always secondary to traumatic or surgical rupture of a hepatic cyst. 7 Primary retroperitoneal echinococcal cysts without involvement of any other organ are extremely rare.• It has been suggested that such a cyst may arise in the liver, but by the time the daughter cyst is discovered, the parent cyst may collapse completely leaving no trace in the primary organ of involvement. 7 Primary mediastinal echinococcosis is also uncommonly reported and its existence has even been questioned. 9 Differential diagnosis in such a case would include diaphragmatic, dermoid, neurenteric or enterogenous duplication cyst or pararenal pseudocyst. Although characteristic appearances of hydatid cyst have been described on US , Cf and MR,uo this diagnosis was not entertained as the cyst was unilocular, showed no characteristic features, and there was no associated hepatic or pulmonary involvement. The object of this communication is to highlight this rare entity and to illustrate the value of MR in demonstrating the extent of the cyst. It is suggested that hydatid disease must be considered in the diagnosis of a cyst, irrespective of its location, especially in endemic areas. REFERENCES

1 Ismail MA, Aida Bagh MA, Aljanabi TA. The use of

cr

in

diagnosis of hydatid cysts. Clin Radiol1980; 31:287-92 2 Bonie J, Shaw JFH. Hepatobronchial fistula caused by hydatid disease. Thorax 1981; 36:25 3 Praderi LA. Evolution ascendente mediastinal de quistos hidaticos subfrenicos. Bol Soc Cir urugi 1956; 27:573 4 Guedj P. A propos des kystes hydatiques du mediastin. Diagnostic des tumeurs mediastinales. J Chir 1958; 75:417 5 Beggs I . The radiology of hydatid disease. AJR 1985; 145:63945 6 A Bonalcarpour. Echinococcal disease. Report of 112 cases from Iran and a review o£611 cases from the United States. AJR 1967; 99:660-67

7 Saidi F. Abdominal and pelvic hydatid cysts. In: Saidi F, ed. Surgery of hydatid disease. London: W.B. Saunders, 1976: 282 8 Singh RS, Sahay S. Retroperitoneal primary hydatid cyst of the pelvis. J Indian Med Assoc 1985; 83:64-5

9 Rokower J, Milwidsky H . Primary mediastinal echinococcosis. Am J Med 1960; 29:73-83 10 Morris DL, Buckley

J, Gregson R,

Worthington BS . Magnetic

Pneumomediastinum, Pneumothorax and Subcutaneous Emphysema Following the Measurement of Maximal Expiratory Pressure in a Normal Subject* jose C. MaRfO, M .D .; ]oao Terra-Filho, M.D.; and Geruza A. Silva , M .D .

Mediastinal and subcutaneous emphysema have been reported as a consequence of deliberate manipulations of the breathing pattern producing a Valsalva-like maneuver in healthy subjects. We present a case of pneumomediastinum, pneumothorax and subcutaneous emphysema occurring in a normal volunteer after repeated measurements of the PEmax. (Chest 1990; 98:1530-32) PEmax =maximum static expiratory pressure; P1max = maximum static inspiratory pressure; RV =residual volume; TLC =total lung capacity; Pes= esophageal pressure; Po= oral pressure

T

he simplest noninvasive and widely applied method for respiratory muscle strength assessment is the measurement of Plmax and PEmax pressures generated at the mouth during static efforts.'" The P1max usually is measured at RV after full expiration and PEmax at TLC after maximal inspiration. We report the findings in a normal subject who presented with pneumomediastinum, pneumothorax and subcutaneous emphysema following repeated measurements ofPEmax. CASE REPORT

As a part of the investigation to establish the normal values for some pulmonary function tests in our laboratory, a healthy 25-yearold male physician, who was a nonsmoker, was studied after informed consent. The study protocol was approved by the Ethical Committee of our institution. After standard spirometric procedures, the PEmax was measured while the subject was seated and wearing nose clips. We used an apparatus similar to that described by Black and Hyatt• and all the determinations of PEmax were repeated after full in8ation to TLC. Pressures were measured with a direct reading dial gauge having a pressure range of 0 to 300 em H,O and simultaneously, through a lateral line, with a differential pressure transducer (Statham PM 131 TC±5-350). During the expiratory efforts we also measured the esophageal pressure using a 10-cm long balloon containing I ml of air and connected to another Statham differential pressure transducer of the same model. The signals of both transducers were recorded on a direct writing recorder (Beckman R-611). In a series of five measurements of expiratory pressures the highest values achieved were 330 em H 20 for PEmax at the mouth and 308 em H20 for the Pes (Fig 1}; the pressure difference between the Po and the Pes corresponded to the recoil pressure of the lungs at the volume where the measurement was carried out. A few minutes after PEmax measurements were done, the subject reported discomfort in the neck, where the

44

*From the Pneumology Section, Department oflnternal Medicine, School of Medicine of Ribeirao Preto, University of Sao Paulo, Ribeirlio Preto, Sao Paulo, Brazil.

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Compllc:a1ions following Measurement ol Maximal Expiratory Pressure (Manco, Term, Silva)

resonance imaging of hydatid disease. Clin Radioll987; 38:141-

1s

400 0

N

:r::

E

Pes

(,)

400 0

N

:r:: E

Po

(,)

0 FIGURE 1. Simultaneous recordings of the Po and the Pes during PEmax measurement. physical examination had revealed crepitations. A roentgenogram of the neck confirmed the diagnosis of subcutaneous emphysema and the chest roentgenogram demonstrated small mediastinal emphysema and bilateral pneumothorax (Fig 2). During the next 24 h the subject complained of mild dysphagia and dysphonia, but denied any kind of pain. All the symptoms and abnormal signs disappeared in about three days and the subject remained asymptomatic during the next eight weeks of follow-up. DISCUSSION

Previous results from our laboratory< and the results reported by Black and Hyatt2 are in agreement in that values of PEmax higher than 300 em H20 are not uncommon for normal male adults. In fact, four subjects in our series of 30 normal men generated mouth pressures in excess of 300 em H.O during maximal static expiratory efforts.• In spite of the high levels of pressures attained during the test, we did not observe any complications with extensive use of this technique in normal subjects and patients until we studied the case herein reported. Ringqvist, 1 in his classic study on maximal respiratory pressures, stated that "no complications of any importance were observed during the whole study." In other studies,...,. we could find no previous reports wherein complications such as pneumomediastinum, pneumothorax and subcutaneous emphysema have occurred in association with PEmax measurements. Most cases of pneumomediastinum probably are the result of alveolar rupture into the bronchovascular sheath, from momentary shearing force due to a sudden pressure discrepancy between them, mainly in the presence of alveolar overdistention. ,..11 From the bronchovascular sheath, the extra-alveolar air moves centripetally to the mediastinum and eventually may reach the pleural cavities and the soft tissue components of the neck. 11 Pneumomediastinum some-

FIGURE 2. Top, Posteroanterior view of neck roentgenogram showing subcutaneous emphysema (arrowhead). Bottom, Posteroanterior view of chest roentgenogram showing small pneumomediastinum

(arrowheads).

times can be induced by acute changes in the breathing pattern so that the lung volume is increased or sudden pressure changes occur. 11 Deliberate manipulations of the breathing pattern producing a Valsalva-like maneuver can result in mediastinal and subcutaneous emphysema in healthy subjects. Mediastinal emphysema has been reported following marijuana smoking, •• probably as a consequence of the straining against a closed glottis after inhalation to TLC. Other circumstances in which normal subjects developed pneumomediastinum, presumably related to ventilaCHEST I 98 I 6 I DECEMBER, 1990

1531

tory maneuvers, include blowing wind instruments, 9 •13 and the "voluntary pressure breathing" during mountain climbing.1• This so-called "voluntary pressure breathing" or "emergency breathing procedure" consists of slow, deep inhalations fOllowed by forced exhalations through tightly pursed lips. Mediastinum and subcutaneous emphysema also occurred in a healthy medical student following a standard spirometric test. 15 Our report illustrates another circumstance in which a ventilatory maneuver applied with diagnostic purpose, that is, the measurement of PEmax, may result in pneumomediastinum, pneumothorax and subcutaneous emphysema. However, it is important to emphasize that the risk of this kind of complication associated with maximal respiratory pressure measurements is certainly small compared with the useful physiologic information provided by this technique of respiratory muscle assessment. REFERENCES 1 Ringqvist T. The ventiJatory capacity in healthy subjects: an analysis of causal factors with special reference to the respiratory forces. Scand J Clin Lab Invest 1966; 18(suppl88):1-179 2 Blaclc LF, Hyatt RE. Maximal respiratory pressures: normal values and relationship to age and sex. Am Rev Respir Dis 1969;

Management of Brucella Endocarditis with Aortic Root Abscess* Saad AI Kasob , M.D., F.C.C.P.; Mohamed Al Fagih, M.D.; Abdul Al Rasheed, M.B ., B.S., M.Sc.; B. Khan, M.D.; lssam Bitar, M.D.; Maie Shahed, M.D.; and Willtam Sawyer; M.B ., Ch.B.

Three cases of Brucella endocarditis with aortic root abscess are reported. Two patients were successfully managed by a combination of medical therapy and surgery. The third patient died suddenly 36 hours after admission to hospital. (Chest 1990; 98:1532-34)

E

ndocarditis is an uncommon, but serious, complication of brucellosis; 1 the aortic is the most frequently affected cardiac valve. 2 We report the successful management of two complex cases of Brucella endocarditis with aortic root abscess, using a combination of medical therapy and surgery; athird seriously ill patient died within 36 hours of admission to hospital.

99:696-702

3 Blaclc LF, Hyatt RE. Maximal respiratory pressures in generalised neuromuscular disease. Am Rev Respir Dis 1971; 103:641-

50

4 CameJo JS Jr, Terra-Filho J, Man~ JC. Pressaes respiratorias miximas em adultos normais. J Pneumol1985; 11:181-84 5 Wilson SH, Cooke NT, Edwards RHT, Spiro SG. Predicted normal values for maximal respiratory pressures in caucasian adults and children. Thorax 1984; 39:535-38 6 Szeinberg A, Marcotte JE, Roizin H, Mindorff C, England S, Thbacbnik E, et al. Normal values of maximal inspiratory and expiratory pressures with a portable apparatus in children, adolescents, and young adults. Pediatr Pulmonol1987; 3:255-58 7 Koulouris N, Mulvey DA, Laroche CM, Green M, Moxham J. Comparison of two different mouthpieces for the measurement of Plmax and PEmax in normal and weak subjects. Eur Respir J 1988; 1:863-67 8 McEivaney G, Blackie S, Morrison NJ, Wilcox PG, Fairbarn MS, Pardy RL. Maximal static respiratory pressures in normal elderly. Am Rev Respir Dis 1989; 139:277-81 9 Macklin MT, Macklin CC. Malignant interstitial emphysema of the lungs and mediastinum as an important occult complication in many respiratory diseases and other conditions: an interpretation of the clinical literature in the light of laboratory experiment. Medicine 1944; 23:281-358 10 Maunder RJ, Pierson DJ, Hudson LD. Subcutaneous and mediastinal emphysema: pathophysiology, diagnosis, and management. Arch Intern Med 1984; 144:1447-53 11 Pierson DJ. Pneumomediastinum. In: Murray JF, Nadel JA, eds. Textbook of respiratory medicine. Philadelphia: Saunders, 1988; 1795-1808 12 Miller WE, Spiekerman RE, Hepper NG. Pneumomediastinum resulting from performing Valsalva maneuvers during marijuana smoking. Chest 1972; 62:233-34 13 Aisner M, Franco JE. Mediastinal emphysema. N Eng! J Med 1949; 241:818-25 14 Vosk A, Houston CS. Mediastinal emphysema in mountain climbers: report of two cases and review. Heart Lung 1977;

6:799-805 15 Varkey 8 , Kory RC . Mediastinal and subcutaneous emphysema following pulmonary function tests. Am Rev Respir Dis 1973; 108:1393-96

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CASE REPORTS

CASE1 A 45-yeaNJid man was admitted to another hospital for seven weeks in 1987 with clinical and bacteriologically proven Brucella melitensis endocarditis involving the aortic valve; two-dimensional echocardiography revealed a 3 x 4 mm vegetation on the valve. His Brucella agglutination titer was originally > 1:20,000, but fell to 1:1,280 within six weeks of medical treatment with rifampicin, tetracycline, and ceftizoxime, all continued for two months. He was readmitted to the same hospital nine months later with a two- to three-week history of recurrence of the symptoms and signs suggesting Brucella infection (fable 1). Table 2 contains the results of the investigations. The chest x-ray film showed mild cardiomegaly with clear lung fields. Two-dimensional echocardiographic findings are listed in Table 1 and illustrated in Figure 1. On transfer to this hospital, medical therapy with gentamicin, 3 *From the Riyadh Cardiac Center, Armed Forces Hospital, Riyadh, Saudi Arabia. Reprl~ req,~ats: Dr. Al Kasob, Armed rorces Hospital, PO Box 7897, J:Uyaah 11159, Saudi Arabia

FIGURE 1. Two-dimensional echocardiography; left parasternal view, showing the aortic root abscess (Ab), and vegetation (V) on the right coronary cusp. LA is left atrium; LV, left ventricle; RV, right ventricle; and S, septum . Management of Brucella Endocarditis (AI K888b eta/)

Pneumomediastinum, pneumothorax and subcutaneous emphysema following the measurement of maximal expiratory pressure in a normal subject.

Mediastinal and subcutaneous emphysema have been reported as a consequence of deliberate manipulations of the breathing pattern producing a Valsalva-l...
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