Acute Supine Respiratory Failure due to Bilateral Diaphragmatic Paralysis

Acute Supine Respiratory Failure due to Bilateral Diaphragmatic Paralysis* }. D. Sandhom, M.D.; D. T.Shaw, M.D.; and Clarence A. Guenter, M.D., F.C.C.P. . This report documents the first known case of bilateral diaphragmatic paralysis foUowing blunt trauma to the chest. The important role of diaphragmatic function in maintaining ventilation, particularly with the patient in the supine position, is illustrated by the reduced total lung capacity, functional residual capacity, and vital capacity. Severe hypoxemia with the patient in the supine position, was markedly improved by elevation of the patient to Jo• and was further improved by sitting the patient upright. FoUowing an initial period of acute respiratory failure, the patient was managed acceptably by maintaining an elevated position for sleeping until diaphragmatic function returned, about nine months after theinjnry.

R espiratory failure aggravated by the supine position most commonly results from left ventricular failure, obesity, severe puhnonary disease, or intra-abdominal disease inhibiting diaphragmatic motion. Rarely, disorders primarily affecting the diaphragm may be responsible. We believe this report documents the first case of transient bilateral diaphragmatic paralysis following blunt trauma to the chest. CASE REPORT

A 55-year-old man was admitted to the hospital following a traffic accident. His multiple injuries included fractures of the left ileum, acetabulum, pubic ramus, femur, humerus, left maxilla, bilateral fractures of the calcanea, and probable basal slcull fractures, as well as a right anterior flail chest. Subsequently, evidence of mild injury to the left brachial plexus was apparent. A chest x-ray film initially demonstrated widening of the superior mediastinum, with relatively clear pulmonary parenchyma ( Fig 1 ) . An angiogram did not demonstrate an aortic tear. Initial management consisted of intubation, ventilation and oxygenation, and stabilization of fractures. Laparotomy demonstrated a large pelvic hematoma, multiple mesenteric lacerations, and a perinephric hematoma, but no other major organ injury. Although requiring a tracheostomy, the patient slowly improved in the intensive care unit and was being weaned from the ventilator three weeks following admission. A chest x-ray film at this time showed elevation of the right hemidiaphragm. Six weekS after admission, the patient began to complain of progressive shortness of breath. Detailed reassessment indicated that he did not have pulmonary emboli, myocardial infarction, infection, or left ventricular failure. An ultrasonic examination of his abdomen did not demonstrate subphrenic °From the University of Calgary Faculty of Medicine and Foothills Hospital, Calgary, Alberta, Canada. Reprint requests: Dr. Guenter, Department of Medicine, Foothills Hospital, Calgary, Alberta, Canada T2N 2T9

96 SANDHAM, SHAW, GUENTER

FIGURE 1. Initial chest x-ray film demonstrated widened superior mediastinum and relatively clear pulmonary fields.

fluid. The patient's condition rapidly deteriorated, and his vital capacity ( VC) decreased to approximately 0.4 L. At this time, he was noted clinically to have elevation of both diaphragms, paradoxic movement of his abdomen on respiration, and increased dyspnea when in the supine position, which was relieved somewhat by sitting. A clinical diagnosis of bilateral paralysis of the diaphragm was confirmed by fluoroscopic examination during stimulation of the phrenic nerve ( Fig 2). The patient's respiratory failure again required support with a ventilator. Three months after admission, the patient was finally weaned from the ventilator, using a rocking bed and a cuirass respirator. When supine, he was severely dyspneic, but when elevated to 30• or greater, he was able to maintain adequate ventilation. Data from studies of pulmonary function performed with the patient in the sitting position, 30• elevated, and supine position are listed in Table 1. The VC, inspiratory capacity ( IC), total lung capacity

FIGURE 2. This chest x-ray film, taken six weeks after injury, reveals bilateral elevation of hemidiaphragms, with small right pleural effusion. Fluoroscopic examination during stimulation of the phrenic nerve demonstrated no motion of either hemidiaphragm.

CHEST, 72: 1, JULY, 1977

Table 1---l»ulmorurrr Funedon Data

Measured. (Percent Predicted) Ventilatory Function •

Sitting

VC,L FEV.,L FEF50%, L/sec FIF50%, L/sec MVV, L/min TLC, Lt FRC,L Helium dilution Body plethysmography IC, L

Supine Predicted**

1.87 (43) 1.52 (45) 2.11 (41) 2.11 (43) 43 (35) 2.27 (34)

0.66 (15)

4.31 3.42 5.17 4.85 124 6.47

1.61 (44) 2.47 (67) 0.66

1.14 (31)

3.70 3.70 2.72

0.39

*FEV., forced expiratory volume in one second; FEF50%, forced expiratory flow after 50 percent of, FVC has been exhaled; FIF50%, forced inspiratory flow, aftei 50 percent of FVC has been inhaled; and MVV, maximal voluntary ventilation. **Predicted values by height and age, sitting or standing. tMeasured by helium dilution. ( TLC) and functional residual capacity ( FRC) were all severely reduced while the patient was sitting and were decreased when he was in the supine position. The maximal expiratory flow rates were reduced as might be anticipated for the low lung volumes, but the maximal inspiratory flow rates were also very low, coosist.ent with weak inspiratOI'Y effort. A significant discrepancy between the functional residual volume measured by helium and by body plethysmographic studies suggested uneven distribution of ventilation. These findings may indicate airway disease due to the patient's previous cigarette smoking or may ·be related to the pulmonary trauma. Analysis of arterial blood gas levels ( Table 2) demonstrated hypoxemia, with progressive increases in the alveolar-arterial oxygen pressure difference (P[A-a]02) when the patient assumed the supine position. This was partially reversed when the patient was lying on a rocking bed or receiving assisted ventilation with an external thoracic negative-pressure ventilator (cuirass). Five months after admission, the patient was beginning to ambulate and was able to feed and dress himself; and he was discharged. Fluoroscopic examination of his diaphragm at that time showed only paradoxic diaphragmatic movement on inspiration. and cervical stimulation of the phrenic nerve during fluoroscopic examination while the patient was holding his breath revealed no diaphragmatic motion. Assessment of abdominal and intrathoracic pressures by esophageal balloon revealed a decrease in abdominal pressure on inspiration. Nine months after the accident, the patient was much improved symptomatically, and fluoroscopic examination demonstrated partial return of motion of both halves of the diaphragm. DISCUSSION

Bilateral paralysis of the diaphragm is rare. Paralysis can occur due to lesions of the spinal cord, nerve roots within the cord, the phrenic nerves themselves, or neuromuscular disease. Processes lcnown to affect the nerve itself include space-occupying lesions in the mediastinum, polyneuropathy ( eg, diphtheria, postinfectious · polyneuropathy, lead toxicity, beriberi), inadvertent sur-

CHEST, 72: 1, JULY, 1977

gical trauma, local infection with herpes zoster, and poliomyelitis. We have been unable to identify reports of palsy of the phrenic nerve secondary to blunt trauma to the chest. The exact reason for the paralysis of this patient's diaphragm is obscure. Possibilities that were considered included mediastinal hematoma, fibrosis, trauma to the spinal cord or nerve roots, t trauma from subclavian venous puncture, 2 reaction to drugs or tetanus toxoid, 8 " 6 infectious polyneuropathy,41 and herpes zoster. 7 No supportive evidence for any of these causes could be found in this patient. Pulmonary function has been measured in several reports of patients with bilateral diaphragmatic paralysis. The VC was decreased to an average of 54 percent of the predicted value, FRC to 61 percent of predicted, and TLC to about 67 percent of predicted; and these changes were accentuated by placing the patient in the supine position. Maximal expiratory flow rates were reduced to about 42 percent of predicted, as a result of low lung volumes, and inspiratory flow rates were comparably reduced to about 35 percent of predicted, due to paralysis of major inspiratory muscles. The P (A-a) 0 2 was increased in recumbency, resulting in more severe hypoxemia.7 • 11 In general, then, these patients exhibit a restrictive pattern of disease with hypoxemia. all of which is more severe in the supine position. Hypoventilation has been reported when diaphragmatic paralysis is associated with generalized neuromuscular disease. 11 The functional derangements of bilateral diaphragmatic paralysis are entirely predictable, on the basis of studies which documented the normal diaphragmatic contribution to ventilation. 11•18 The diaphragmatic contribution .to ventilation was more prominent while the subject was in the supine position, and the contribution of the intercostal muscles was proportionately greater when the subject was in the upright position. Ventilatory abnormalities associated with unilateral diaphragmatic paralysis are measurable but rarely cause dyspnea. Several approaches to therapy of diaphragmatic paralysis have been suggested. Diaphragmatie pacing, using a cuH around the phrenic nerve in the neck, has had remarlcable success in selected cases16 but requires an intact phrenic nerve. Transvenous stimulation of the phrenic nerve16 and direct diaphragmatic stimulation1 ' have not had adequate clinical trials. Recent experimentTable 2-..4raerial Blood Ga. Lewb

pH Sitting 30° recumbent Supine Rocking bed, asleep Cuirass respiration, asleep Mild upright exercise

7.43 7.39 7.40 7.43 7.43 7.43

PaCOs, PaOt, P(A-a)Os, mmHg mmHg mm Hg* 43 39 39 37 37 34

58

20

44 39 51 57 55

39 44 35 29 34

• Alveolar oxygen pressure calculated as follows: P A 0t PlOt - 1.20 Pas. Note that average barometric pressure in Calgary is 660 mm Hg.

ACUTE SUPINE RESPIRATORY FAILURE 97

al evidence suggests that anastomosis of the vagal trunk to the distal end of a damaged phrenic nerve will allow regeneration of the phrenic fibers.H Under these circumstances, stimulation of the vagal trunk would permit pacing of the diaphragm. · Plication of the diaphragm improves some aspects of ventilatory function in experimental animals,Is but no data are available from humans. . Since most patients with bilateral diaphragmatic paralysis function reasonably while in the sitting or erect position and can tolerate mild to moderate exercise conservative management is generally the treatment of choice. Regeneration of the phrenic nerve may occur as late as 12 to 15 months after the onset of paralysis. ·

fu:FERENCFS 1 Michaux P, Curtes ]P: Delayed left phrenic paralysis secondary to torsion due to an automobile accident. Acta Med Leg Soc 20:315-321, 1~ 2 Obel IWP: Transient phrenic nerve paralysis following subclavian venipuncture. Anesthesiology 33:369-370, 1970 3 Comroe JH, Wood FC, Kay CF, et al : Motor neuritis after tetanus antitoxin with involvement of the muscles oi respiration. Am J Med 10:7~789, 1951 4 Reactions to tetanus toxoid. Br Med J 1:48, 1974 5 Blumstein Gl, ICreithen H: Peripheral neuropathy following tetanus toxoid administration. JAMA 198:168-169, 1966 6 Spitzer SA, Korczyn AD, ICalaci J: Transient bilateral diaphragmatic paralysis. Chest 64:355-357, 1973 7 Dutt AK : Diaphragm paralysis caused by herpes zoster. Am Rev Respir Dis 10:755-758, 1970 8 McRedie M, Lovejoy FW Jr, ICaltreider NL: Pulmonary function in diaphragmatic paralysis. Thorax 17:213-217, ' 1962 9 Huettemann U, Hudcauf H : Advanced respiratory failure due to disturbances of diaphragmatic movement. Respiration 27:363-376, 1970 10 Eisele JH, Noble MIM, Katz J, et al: Bilateral phrenic nerve block in man. ·Anesthesiology 37:64-69, 1972 11 Davis J, Goldman M, Loh L, et al: Diaphragm function and alveolar hypoventilation. Q J Med 45:87-100, 1976 12 Wang CS, Josenhans WT: Contribution of diaphragmatic/abdominal displacement to ventilation in supine man. J Appl Physiol 31 :576-580, 1971 13 Sharp JT, Goldberg NB, Druz WS, et al: Relative contributions of rib cage and abdomen to breathing in normal subjects. J Appl Physiol39:608-618, 1975 14 Glenn WWL, Holcomb WG, Hogan J, et al : Diaphragm pacing by radiofrequency transmission in the treatment of chronic ventilatory insufficiency. J Thorac Cardiovasc Surg 66:505-519, 1973 15 Furman S, Koerner SK, Escher DJW, et al: Transvenous stimulation of the phrenic nerves. J Thorac Cardiovasc Surg 62:743-751, 1971 16 Johnson V, Eiseman B: Reinforcement of ventilation with electrophrenic pacing of the paralyzed diaphragm. J Thorac Cardiovasc Surg 62:651-657, 1971 17 Wilson AS, Krcek ]P: Restoration of function in the paralyzed diaphragm. Exp Neurol 47 :490-502, 1975 18 Marcos JJ, Grover FL, Trinkle JK: Paralyzed diaphragm: Effect of plication on respiratory mechanics. J Surg Res 16:523-526, 1974 1

98 ROBLES DE MEDINA ET AL

Removal of a Fragment of Catheter Entrapped in a Biork·Shiley Aortic Valvular Prosthesis* Transarterial Approach Using a Modified Loop-Snare Teehnique Etienne 0 . Robles de Medina, M.D.;•• Pieter W. Westerhof, M .D.; 00 Gerl ]. van Mill, M.D.;•• Hans A. Huysmm'IS, M.D.;t and Alerander L.E.M.S . Schaepkefl8 van Riempst, M.D.t

Fncture of a left atrial catheter made of polyethylene and Its subsequent embollzadon and entrapment in a Bjork-Shiley aortic l'Bivular prosthesis is reported in a 62-year-old woman. 'Ibe entrapped fragment of the catheter was retrieved by a transarterial approach using a homemade modified snare-loop device. Measures to prevent such a complication should be directed towards avoidance of force when removing such catheten. In addition, their length should be determined prior to insertion and again after removal, in order to detect their poesible fracture and loss at once.

Jntravascular severing of cathetersl-4 and guide wires, dislodgment of Pudenz-Holter drains,5,8 and 5 -1

incompletely removed pacing catheters,11 with subsequent embolization to more central parts of the cardiovascular system, have been the subject of quite a number of recent rep(>rts. It is the purpose of this communication to report the accidental loss and subsequent transluminal retrieval of a left atrial pressure-monitoring catheter which was embolized and entrapped in a Bjork-Shiley aortic valvular prosthesis. CASE REPoRT

A 62-year-old woman with severe rheumatic aortic regurgitation and dyspnea on effort had her aortic valve replaced by a Bjork-Shiley valvular prosthesis. During the operation a polyethylene catheter was inserted into the left atrium for postoperative pressure monitoring. No abnormal Shortening was reported when the catheter was removed a few days later. Recovery was uneventful, and the patient was discharged from the hospital. She was seen at the outpatient department two months later and was doing well; however, fluoroscopic control showed a fragment of the catheter, with a long and a short limb, swinging from the prosthetic valve in the ascending aorta (Fig 1 ) . The patient was readmitted to the hospital, and after consent had been obtained, a successful attempt was made at transarterial retrieval. Under local anesthesia, a brachial arterial cutdown was performed in the right arm, and heparin was administered to the patient. A Dormia ureteral stone catcher was first advanced to the ascending aorta but failed to catch the object. Subsequently, a snare-loop device was constructed using a °From the Department of Cardiology, University Hospital, and the Department of Cardiothoracic Surgery, St. Antonius Hospital, Utrecht, the Netherlands. • • Department of Cardiology. tDepartment of Cardiothoracic Surgery. Reprint requests: Dr. Robles de Medina, Department of Medidne, University Hospital, Utrecht, Netherlands

CHEST, 72: 1, JULY, 1977