saecree RePORTS Repair of Tetralogy of Fallot after Catheterization-induced Complete

Heart Block* Michael LaCorte, M.D.;oO Macdonald Dick, M.D.;t Amnon Rosenthal, M.D.;t and Aido Castaneda, M.D., F.C.C.P.§

The case of a four-year-old boy with tetralogy of Fallot who developed complete heart block during cardiac catheterization and subsequently underwent surgical repair in the presence of heart block is presentecL Return to normal sinus rhythm occurred on the 11th postperative day, and the manner in which increased atrioventricular conduction appeared was documented with the use of a continuous Holter monitor. Placement of a temporary transvenous pacemaker, systemic heparinization in the preoperative period, and the insertion of a permanent epicardial wire during surgery were performed. heart block (CHB) developing during carC omplete diac catheterization and persisting for more than

one week has been reported in two children. This is the For editorial comment, see page 482

third such case but the first to undergo repair of the intracardiac defect (tetralogy of Fallot) during the presence of heart block with subsequent return to normal sinus rhythm during the postoperative period. CASE REPORT

A four-year-old boy with tetralogy of Fallot was admitted to the Children's Hospital Medical Center, Boston, on March 4, 1974 for cardiac evaluation. He was the product of a fulltenn pregnancy. Birth weight was 3,572 gm (7 lb 14 oz), and cyanosis was first noted at six months of age. At one year a right-pulmonary-artery-to-ascending-aorta shunt operation was performed because of cyanotic spells. Nine days after surgery, right hemiparesis developed which subsequently improved. At four years of age, he was referred to the Children's Hospital Medical Center because of severe cyanosis and exercise intolerance. At admission physical examination revealed a markedly cyanotic child with no audible shunt murmur. The electrocardiogram showed right axis deviation, and right atrial and ventricular hypertrophy. There was normal sinus rhythm with a P-R interval of 0.16 second (Fig la). o From

the Deparbnents of Cardiology and Cardiovascular Surgery, the Children's Hospital Medical Center, and the Deparbnents of Pediatrics and Surgery, Harvard Medical School, Boston. Supported in part by grants HLI0436 and H11)5855 from the National Institutes of Health and by a grant-in-aid from the American Heart Association. o o Fellow in Cardiology. t Instructor in Pediatrics. tAssociate Professor of Pediatrics. §Professor of Surgery. Reprint requests: Dr. Rosenthal, 300 Longwood Avenue,

Boston 02135

CHEST, 68: 4, OCTOBER, 1975

On March 6, 1974, cardiac catheterization revealed severe tetralogy of FaIlot, a systemic saturation of 72 percent, a nonfunctioning Waterston shunt, and a right aortic arch. Soon after passage of the catheter from the right ventricle to the aorta, atriovenlricular (A V) dissociation was noted on the monitor. The ECG immediately after catheterization revealed complete A-V dissociation with a ventricular rate of 150 and an atrial rate of ISO (Fig Ib ). A 48-hour course of prednisone was given without improvement. Occasional sinus captures were recorded, but a 24-hour Holter monitor placed on the sixth day after catheterization revealed no sinus captures and a ventricular rate of 55 to 78 (Fig lc). The patient remained asymptomatic during this period. On March 13, 1974, one week after catheterization, a transvenous demand pacemaker was placed in the right ventricle, and the patient was heparinized (100 units/kg). Due to the patient's marked cyanosis, nonfunctioning shunt, and severe outflow obstruction, surgical repair was undertaken despite the persistent block. On March 14, 1974 under cardiopulmonary bypass, the patient underwent repair of tetralogy of Fallot. During the procedure, a temporary epicardial pacemaker wire was sewn into the right ventricular myocardium and connected to an external demand pacemaker. In addition, a permanent unipolar pacemaker wire was sewn into the left ventricular myocardium and left embedded within the subfascial layer of the abdominal wall. Attempts to obtain intraoperative His bundle electrograms were unsuccessful. Immediately postoperatively he continued to have eHB with a ventricular rate of 100 (Fig Id). Complete heart block persisted for the next ten days with a ventricular rate of 75 to 90. During this time, the patient was successfully treated for moderate congestive heart failure with digoxin and diuretics. On the tenth postoperative day, 18 days after the onset of CUB, second-degree AV block was documented (Fig Ie ). On the 11th postoperative day a 12-hour Holter monitor tape revealed Mobitz type 1 second-degree A V block with a progressive increase in AV conduction, which was followed by the onset of normal sinus rhythm with first-degree AV block (Fig If and Ig). For the next three days the patient continued in normal sinus rhythm with occasional seconddegree AV block. Thereafter, the patient remained exclusively in normal sinus rhythm with first-degree AV block (Fig Ih ). The temporary epicardial wire was removed, the permanent epicardial wire was buried deep in the rectus sheath, and on the 22nd postoperative day the patient was discharged from the hospital. DISCUSSION

Transient CHB is a known but uncommon complication of cardiac catheterization. In one large series, seven of 4,413 adult patients developed transient CUB during catheterization.' In one series of children, transient second-degree heart block or CHB developed in 4.3 percent of patients undergoing catheterization." In the cooperative study on cardiac catheterization, there was a tenfold increase in the incidence of transient CUB in children as compared to adults." The frequency of catheterizationinduced CUB appears to be inversely related to age and is especially common in the sick infant." Catheterizationinduced CUB is usually short lived, and persistence of CUB for more than one week after catheterization is quite rare.t-" The three children (including the present case) whose CUB has persisted for more than one week have all had tetralogy of Fallot. This may be explained

REPAIR OF TETRALOGY OF FALLOT 575

a

b

c

d



FIGURE 1. (a) Lead 2 prior to catheterization showing normal sinus rhythm (P-R = 0.14 second). (b) Rhythm strip (lead 2) shortly after catheterization showing CHB. 185; ventricular rate 145. Atrial rate ( c) Rhythm strip from Holter tape sixth day after catheterization demonstrating complete heart block. Atrial rate = 118; ventricular rate = 60. (d) Rhythm strip immediately after surgery, There is CHB with ventricular rate of 100 and atrial rate of 135. (e) Rhythm strip (lead 2) obtained on tenth postoperative day showing second-degree AV block with capture-escape bigeminy. (f) and (g) Rhythm strips from Holter tape on 11th postoperative day demonstrating 4 :3 Weckebach (f) followed shortly thereafter (g ) by 6 :5 Wenckebach sequences. ( h ) Rhythm stri p (lead 2) immediately prior to discharge showing normal sinus rhythm with firstdegree AV block .

=

f

D

h by variations in the pathway of the specialized conduction tissue in tetralogy of Fallot making it more vulnerable to catheter trauma." The site of trauma to the conduction tissue in patients developing CHB during catheterization is of interest. Freed and Rosenthal' demonstrated by His bundle recording that the block in their patient was proximal to the His bundle, suggesting injury to the AV node. Catheter-induced right bundlebranch block in adults with preexisting left bundlebranch block has been reported. r-" Our patient is unique because successful repair of tetralogy of Fallot was performed in the presence of catheter-induced CHB, and AV conduction returned II days after surgery. In addition, there are a number of important aspects in the management of this patient prior to, during, and after surgery. A temporary transvenous pacing wire was placed preoperatively in this

576 LA CORTE ET AL

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patient, and heparinization was started after insertion of the pacing wire . The risk of asystole during induction of anesthesia is considerable in the presence of CHB, and the placement of a temporary pacemaker preoperatively" is therefore advised. To our knowledge, paradoxic embolization after transvenous pacemaker insertion has not been reported; however, it has been observed in our institution in a 22-year-old woman with single ventricle and corrected transposition who experienced a mild transient cerebrovascular accident after insertion of a transvenous pacemaker. \Ve, therefore, feel that systemic heparinization is indicated following pacemaker insertion in patients with intracardiac right-to-left shunts. Because heart block persisted for one week after catheterization, permanent epicardial pacemaker wires were placed in the operating room. Intraoperative His bundle electrograms, although not obtained in this pa-

CHEST, 68: 4, OCTOBER, 1975

tient because of technical problems, would be useful in localizing the site of eHB and in preventing further injury to the specialized conduction tissue during surgery. During the postoperative management of this patient, the possibility of inserting a permanent pacing unit was repeatedly considered. On the tenth postoperative day the first signs of AV conduction were noted, and pacemaker installation was deferred. A 12-hour Holter monitor was useful in determining the extent of AV conduction. The Holter record demonstrated gradual progression from 3:2 Wenckebach sequences to 6:5 Wenckebach sequences and then finally to intervals of normal sinus rhythm with first-degree AV block. The return of AV conduction by this mechanism, the rapid ventricular response, and the presence of a narrow QRS complex during CHB in the preoperative period strongly suggest that the block was proximal to the His bundle. 1 0 - 1 2 We believe that the presence of CHB after catheterization is not a contraindication to surgical intervention. The insertion of a temporary transvenous pacemaker preoperatively, systemic heparinization if an intracardiac right-to-left shunt exists, and the insertion of permanent epicardial wires at the time of surgery are recommended. REFERENCES

1 Wennevold A, Christiansen I, Lindeneg 0: Complications in 4,413 catheterizations of the right side of the heart. Am Heart J 69:173-180, 1965 2 Black IFS, Moore AAD, Cortes S, et al: Atrioventricular block as a complication of cardiac catheterization. Circulation 30 (suppl 3) :48, 1964 3 McIntosch HD: Arrhythmias. Circulation 37 (suppl 3): 27-35, 1968 4 Gault JH, Ross J Ir, Braunwald E: Persistent atrioventricular dissociation with block and nodal rhythm after cardiac catheterization. Am Heart J 71: 690-694, 1966 5 Freed MD, Rosenthal A: Complete heart block after cardiac catheterization: A rare complication. Pediatrics 51:935-937, 1973 6 Lev M: The architecture of the conduction system in congenital heart disease: Tetralogy of Fallot. Arch Path 67:572-587,1959 7 Gupta PK, Haft JI: Complete heart block complicating cardiac catheterization. Chest 61: 185-187, 1972 8 Cheng TO, Bashour T, KeIser G Jr: Complete heart block occurring during right heart catheterization in a patient with left bundle branch block and prolonged P-R interval showed by bundle of His recording and atrial pacing. Med Ann DC 41:742-743, 1972 9 Scott DL: Cardiac pacemaker as an anesthetic problem. Anaesthesia 25:87-104, 1970 10 Haft JI: The His bundle electrogram. Circulation 47:897909, 1973 11 Kelley DT, Brodsky SJ, Mirowski M, et al: Bundle of His recordings in congenital complete heart block. Circulation 45:277~1, 1972 12 Damato AN, Lau SH, Helfant It, et al: A study of heart block in man using His bundle recordings. Circulation 39:297-305, 1969

CHEST, 68: 4, OCTOBER, 1975

Idiopathic Pulmonary Hemosiderosis* Electron Microscopic, Immunofluorescent, and Iron Kinetic Studies Charles J. Donlan, Jr., LCDR, MC, USN; Charles H. Srodes, LCDR, MC, USNR; and F. Daniel Duffy, LCDR, Me, USNR

The clinical coune of a 37-year-old white man with idiopathic pulmonary hemosiderosis is presented. This patient is UDusuailn that be bas bad repeated exacerbations and remissions over a period of seven years and remains currently iD spoDbmeous remBsion with DO therapy. Routine sections of the Julll biopsy revealed characteristic findlDgs. Immunofluorescence staining of the lung was negative, and electron microscopic studies showed only nonspecific findiap. While in remission, 51chromiumlabelled red-blood-ceD survival studies and 59jroD kinetic studies were performed; the results were nonaaL

thic pulmonary hemosiderosis (IPH) is an unI diopa common disease characterized by hemoptysis, pul-

monary infiltrates, and iron deficiency anemia. The course of the disease is unpredictable, and patients may die shortly after onset of symptoms or live for many For editorial comment, see page 483

years. 1 , 2 The patient reported herein has had repeated relapses and spontaneous remissions over a period of approximately seven years. CASE

REPoRT

A 37-year-old white man experienced episodes of hemoptysis in 1967 and 1968 not associated with anemia or radiologic pulmonary abnormalities. In 1970 he was hospitalized with hemoptysis and dyspnea. A chest x-ray fUm revealed extensive bilateral infiltrates, and the hematocrit reading was 31 percent. The patient gradually improved, and the anemia responded to oral administration of ferrous sulfate. In 1971 he was hospitalized with hemoptysis, anemia, and an abnormal chest x-ray film and again improved spontaneously. He then remained asymptomatic until August 1973 when he was admitted for the first time to the Portsmouth (Va) Naval Hospital with a history of fatigue, fever, and hemoptysis of four weeks' duration. He has been a 1~ pack-per-day smoker for many years. Physical examination was normal except for pallor. The stool was negative for occult blood. The hematocrit reading was 27 percent, and the mean corpuscular volume was 82 cu p.. The peripheral blood smear showed striking hypochromia, microcytosis, anisocytosis, poikilocytosis, and polychromasia. The results of the white blood cell count and the differential leukocyte count were normal, and the platelet count was 500,000/cu mm. The reticulocyte count was 6 percent. The serum iron level was 46 °From the Divisions of Pulmonary Disease and Hematology, Department of Medicine, Naval Regional Metical Center, Portsmouth, Va. Reprint requests: LCDR Donlan, US Naval Hospital, Box 535, Portsmouth, Virginia 23708

IDIOPATHIC PULMONARY HEMOSIDEROSIS 511

Repair of tetralogy of Fallot after catheterization-induced complete heart block.

The case of a four-year-old boy with tetralogy of Fallot who developed complete heart block during cardiac catheterization and subsequently underwent ...
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