underlying cardiomyopathy in conjunction with the generalized skeletal muscle involvement. The literature would seem devoid of microscopic evaluation of cardiac muscle in this disorder. Ionasescu et al14 have demonstrated low calcium-binding activity in the sarcoplasmic reticulum of skeletal muscle in this disorder. In view of the similarities in the structure of the sarcoplasmic reticulum in the two types of muscle, the possibility of a similar cardiac muscle dysfunction is intriguing. The characteristic myopathic vacuoles are present in the diaphragm.t! yet the number of cases of respiratory distress are few. Campa and Sanders'" have demonstrated that the postjunctional membrane, at the motor end-plate, retains at least some potential toward depolarization, and that through repetitive nerve stimulation, a temporary recovery in muscle function can be obtained. The recovery in muscle function occurring during the repetitive stimulation may explain not only the infrequency of respiratory failure but also the lack of cardiac dysfunction that is observed. The etiology of this apparent cardiomyopathy is not clear at this time, and the prospect of more widespread involvement in this disorder requires further evaluation. ACKNOWLEDGMENT: We wish to thank Joseph Lacy, M.D.~ and Richard Dauben, M.D., for their critical comments. We aiso wish to thank Fritzie Cohn for the preparation of the manuscript. REFERENCES

1 Pearson CM, Kalyanaraman K: The Metabolic Basis of Inherited Disease. (Stanbury JB, Wyngaarden JB, Fredrickson DS, eds). New York, McGraw Hill, 1972, p 1182 2 Shinosaki T: Klinische studien uher die periodische extremitatenlahmung. Z Neurol Psychol 100:564, 1926 3 Oppenheim H: Neve mittheilungen uber den von professor westphal beschreibenen fall von periodischer lahmung aller vier extremitaten. Charite-ann 16:350, 1891 4 Neel AV: Zwei von einander unabhangige falle von myoplegia paroxysmatica (periodica familiaris). Z Neurol PsychoI118:269, 1928 5 Gardener HW: A case of periodic paralysis. Brain 35: 143, 1912 6 Singer HD, Goodbody FW: A case of family periodic paralysis with a critical digest of the literature. Brain 24:257, 1901 7 Prudenz RH, McIntosh JF, McEachren D: The role of potassium in familial periodic paralysis. JAMA 111:2253, 1938 8 Gass H, Cherkasky M, Savitsky N: Potassium and periodic paralysis, metabolic study and physiological considerations. 27: 105, 1948 9 Coppen AJ, Reynolds EH: Electrolyte and water distribution in familial hypokalemic periodic paralysis. J Neurol Neurosurg Psychiatr 29:107, 1966 10 Pearson CM: The periodic paralysis: Differential features and pathological observations in permanent myopathy in weakness. Brain 87 :341, 1964 11 Van Hom G, Diori JB, Schwartz FD: Hypokalemic myopathy and elevation of serum enzymes. Arch Neurol 33:335, 1970 12 Sobel BE, Shell WE: Serum enzyme determinations in

192 MAHAJAN, SIMON, HUBER

the diagnosis and assessment of myocardial infarction. Circulation 45:471, 1972 13 Wieme RJ, Herpol JE: Origin of the lactate dehydrogenase isoenzyme pattern found in the serum of patients having primary muscular dystrophy. Nature 194:287, 1962 14 Ionasescu V, Schochet SS, Powers JM, et al: Hypokalemic periodic paralysis. J Neurol Sci 21:419, 1974 15 Campa JF, Sanders DB: Familial hypokalemic periodic paralysis. Arch Neurol 31: 110, 1974

Reexpansion Pulmonary Edema * Vi;ay K. Mohaian, M.D., F.C.C.P.; Morris Simon, M.D.; and Gary L. Huber, M.D., F.C.C.P

A case of pulmonary edema foUowing reexpansion of a coUapsed lung due to pneumothorax is described and illustrated. The importance of recognizing this relatively uncommon phenomenon is stressed. The development of such edema can be prevented by avoiding application of sudden and excessive negative pleural pressures during the evacuation of a pneumothorax or a pleural etfusion. The edema generally occurs in a lung that has been collapsed for more than three days. The importance of the duration of pulmonary collapse in the causation of edema is demonstrated in this patient. number of noncardiac causes of pulmonary edema A have been recognized.' A rare noncardiac type of

pulmonary edema is its occurrence following reexpansion of a lung after collapse due to pneumothorax- or pleural effusion." Because of the rarity of its occurrence, we report the findings in a patient who on one occasion developed radiologically evident pulmonary edema in the reexpanded lung following the evacuation of a prolonged pneumothorax, while after another briefer episode of pneumothorax, edema did not develop. CASE REPORT

An 1S-year-old white woman experienced a sudden sharp pain in her right hemithorax three days prior to the present admission. There was clinical and radiologic evidence of rightsided pneumothorax. The underlying lung was completely collapsed (Fig 1) . No other significant abnonnality was detected on clinical and laboratory examination. A No. 22F Foley catheter was introduced into the right pleural cavity through the second intercostal space, and the pneumothorax was evacuated by applying suction at a negative pressure of 15 cm H 2 0 . A repeat chest x-ray film obtained one-half hour later demonstrated complete reexpansion of the previously collapsed right lung; however, at this time, there was an ill-defined patchy consolidation in the lower zone of the right lung that extended throughout the right lung over the next 72 hours (Fig 2). The pulmonary in6ltra.te progressively decreased within the next 48 to 72 hours (Fig °From the Departments of Medicine and Radiology, Thorndike Laboratory and Beth Israel Hospital, Harvard Medical School, Boston. Reprint requests: Dr. Huber, 330 Brookline Avenue, Bolton 02215

CHEST, 75: 2, FEBRUARY, 1979

DISCUSSION

FiCURE 1. Complete right-sided pneumothorax and collapse of underlyinglung.

FiCURE 2. Development of edema in right lung following expansion of collapsed lung due to pneumothorax.

FicURE 3.

Rapidclearingof edema.

3). The patient was not treated with either diuretics or antibiotics during this period. The patient remained well for two days after the clearing of the infiltrate and then developed a second pneumothorax, with partial collapse of the right lung. She was observed for 17 hours, and because the lung showed no spontaneow expansion, a chest tube was again inserted, and the lung was slowly reexpanded under negative pressure of 15 em H20. This resulted in complete reexpansion of the right lung without recurrence of edema.

CHEST, 75: 2, FEBRUARY, 1979

This case demonstrates the appearance of patchy consolidation in the reexpanded lung following rapid evacuation of a pneumothorax. The appearance of coalescent, ill-defined, and fluffy opacities and their evolution over a period of five to six days fulfill the radiologic criteria for the diagnosis of pulmonary edema.s Absence of cardiomegaly or any other known cause of pulmonary edema suggests, by exclusion, a relationship between the process of pulmonary reexpansion and the development of pulmonary edema. Similar radiologic appearances could result from a rapidly evolving pneumonitis, especially in an immunocompromised host, such as this patient; however, the absence of any clinical features of pulmonary infection (fever, toxemia, cough, purulent sputum, and leukocytosis) makes the diagnosis of pneumonia unlikely. Development of unilateral pulmonary edema in the reexpanded lung is a rare but recognized complication of evacuation of a pneumothorax and rapid pulmonary reexpansion.v" This complication is also known to occur following drainage of pleural effusion.S The first welldocumented report of pulmonary edema following thoracocentesis was by Foucart" in 1875. In their review in 1975, Rogaly and Mervitz 7 could find only eight additional reported cases in the literature, thus emphasizing the relative rarity of this complication. The course of reexpan.sion pulmonary edema appears to be variable. In most instances, it has been described as a radiologic diagnosis without major clinical consequences; however, its recognition is important because in some patients, it may be severe enough to result in a catastrophic respiratory failure that may prove fatal." More likely, lack of awareness of this complication may lead to a mistaken diagnosis of pneumonia and result in unnecessary and at times potentially dangerous diagnostic procedures and therapeutic regimens. When it occurs, the edema is usually evident within two hours after expansion of the collapsed lung and may progress for 24 to 48 hours. It usually disappears in five to seven days without any sequelae. The development of pulmonary edema or its potentially dangerous sequelae cannot be predicted in any patient treated for pneumothorax or pleural effusion; however, prolonged duration of pulmonary collapses and application of excessive negative pressures" during evacuation of air or fluid from the pleural space have been cited as the two most significant factors that predispose to formation of edema. Our review of the reported cases indicated that, with one exception, all patients had prolonged pulmonary collapse ranging in duration from three days to several weeks. In the majority of patients, negative pressure had been employed during rapid reexpansion . The importance of the prolonged duration of pulmonary collapse is particularly well demonstrated in this patient, as the edema did not occur when the lung was reexpanded within 17 hours after the second episode, contrasting with the first episode, when the lung was collapsed for three days. Studies in animals in which pneumothorax was produced

REEXPANSION PULMONARY EDEMA 193

experimentally support the importance of these points." These observations suggest several clinically important guidelines for prevention of reexpansion pulmonary edema. First, on the basis of the available information we recommend that negative pressures of more than em H 20 should not be employed during the evacuation of either a pneumothorax or a massive pleural effusion. 2 Secondly, we suggest that the onset of persistent cough during evacuation of air or fluid from the pleural space may be an early sign of the development of pulmonary edema and should indicate termination of the procedure." Thirdly, in patients with massive effusion, we recommend that the amount of aspirated :8uid should be no more than 1,500 ml at one time. These precautions are especially important in patients with prolonged pulmonary collapse. In some patients with life-threatening edema, recoIlapse of the lung by introducing air into the pleural space may prove helpful. In the occasional patient who is dangerously hypoxic, it has been suggested that transient occlusion of the ipsilateral pulmonary arterial circulation with a balloon-tipped catheter might prove helpful in reducing massive right-to-left intrapulmonary shunt." The pathophysiologic mechanisms responsible for reexpansion pulmonary edema are poorly understood. The most plausible mechanism of edema may be a combined effect of the increased permeability of the pulmonary capillaries due to hypoxic damage and the sudden increase in pulmonary capillary blood flow during reexpansion of the lung under negative pressure. It is tempting to implicate increased alveolar surface tension in the collapsed lung as an important factor contributing to the development of pulmonary edema;" however, in the absence of definitive experimental data, the role of impaired surface activity in reexpansion edema needs further clarification. In addition to these potential pathogenic factors, increases in pulmonary interstitial pressure and bronchial obstruction have also been mentioned to promote the development of edema." In conclusion, we believe that it is important to increase awareness of the entity of reexpansion pulmonary edema and its potentially serious complications, as well as the implications for prevention and management.

10

REFERENCES

1 Phillips IF, Neiman HL, Reeder MM: Non cardiac causes of pulmonary edema. JAMA 234:531-532, 1975 2 Ziskind MM, Weill H, George RA: Acute pulmonary edema following the treatment of spontaneous pneumothorax with excessive negative intrapleural pressure. Am Rev Respir Dis 92: 632-636, 1965 3 Trapnell DH, Thurston JGB: Unilateral pulmonary edema after pleural aspiration. Lancet 1: 1367-1369, 1970 4 Fraser RG, Parei JAP: Diagnosis of Diseases of the Chest. Philadelphia, WB Saunders Co, 1970, p 853 5 Sautter RD, Dreher WH, MacIndoe JH, et al: Fatal pulmonary edema and pneumonitis after reexpansion of chronic pneumothorax. Chest 60:399-401, 1971 6 Foucart EJ: De la Morte Subite on Rapide Apres la Thoracocentese. Paris, A Parent, 1875 7 Rogaly E, Mervitz MD: Unilateral pulmonary edema after

194 SCHULMAN ET AL

drainage of a spontaneous pneumothorax. S Mr Med J 49:1611-1613, 1975 8 Miller we, Toon R, Palat H, et al: Experimental pulmonary edema following re-expansion of pneumothorax. Am Rev Respir Dis 108:664-666, 1973 9 Childress ME, Moy G, Mottram M: Unilateral pulmonary edema resulting from treatment of spontaneous pneumothorax. Am Rev Respir Dis 104:119-121, 1971

Spontaneous Pneumothorax as a Result of Intensive Cytotoxic Chemotherapy* Philip Schulman, M.D.; Edgar Cheng, M.D.; Esteban Cvitkovic, M.D.; and Robert Golbey, M.D.

Spontaneous pneumothorax occurring as a compHcation of the antitumor effect of cytotoxic chemotherapy has been reported in occasional cases of osteogenic sarcoma. Its occurrence in other tumors has not been described. This report describes two cases of this complication in patients with germinal tumors and discusses possible pathophysiologic mechanisms. Spontaneous pneumothorax may occur in various clinical settings. It is seen in the asthenic young smoker who is otherwise healthy or in the older smoker who has chronic obstructive pulmonary disease with fibrosis and localized emphysema. Spontaneous pneumothorax has also been observed as an initial feature in bronchogenic carcinoma;' teratoma," and metastatic pulmonary neoplasms, such as osteogenic sarcoma.s'" lymphosarcoma, 5 and other soft tissue sarcomas. 5 Of 1,143 cases of spontaneous pneumothorax seen at the Mayo Clinic between 1953 and 1973, ten were attributed to malignant pulmonary neoplasms." Spontaneous pneumothorax may also have iatrogenic causes, such as therapy with continuous positive-pressure ventilation, closed-chest cardiac massage, positive end-expiratory pressure, catheterization of the subclavian vein, and tracheostomy. 6 Although the occurrence of spontaneous pneumothorax in the setting of rapid regression of a tumor following chemotherapy has been witnessed in patients with osteogenic sarcoma," we have not found any reference to its occurrence in other tumors. We have seen this phenomenon in recent months in two patients with otherwise uncomplicated germinal tumors and in four o~hers in whom other factors in addition to rapid regression of the tumor may have been involved. CASE REPORTS CASE

1

A 19-year-old white woman developed abdominal pain in the summer of 1976. At exploratory laparotomy a large right .,From ~e Solid Tumor Service, Department of Medicine, Memonal Sloan-Kettering Cancer Center, New York.

CHEST, 75: 2, FEBRUARY, 1979

Reexpansion pulmonary edema.

underlying cardiomyopathy in conjunction with the generalized skeletal muscle involvement. The literature would seem devoid of microscopic evaluation...
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