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Bronchial Atresia Associated with a Bronchogenic Cyst
mold growth, and safe safe handling, if necessary, by using respiration filters.' filters. Q All molds of suitable size (0.5-8 ,...m)4 size (0.5-8 pm)' can probably cause cause the disease if exposure is massive and long enough enough..
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ACKNOWLEDGMENT: The authors thank Tarja Ojanen (UniverUniversity of Kuopio) Kuopio) for Penicillium for performing the precipitin tests on Penicil ium sp. REFERENCES
1 Solley Solley GO, GO, Hyatt RE. Hypersensitivity pneumonitis induced by Penicillium species. J Allerg Clin Immunol 1980; 1980; 65:65-70 6565-70 A, Forsen K-O, K-0, Keskinen H, Alanko K. Humidi2 van Assendelft A, fier-associated fier-associated extrinsic extrinsic allergic alveolitis. Scand J Work Environ Health 1979; 1979; 5:35-41 535-41 33 Pellikka Pellikka M, Kotimaa M. The mould dust concentration caused by the handling of fuel chips and its modifying factors. Folia Forestalia 1983; 1983; 563:1-18 563:l-18 4 Terho EO. Extrinsic allergic alveoli tis-the state of the art. Eur J alveolitis-the Respir Dis 1982; 63 (suppl (suppl 124): 124): 10-26 10-26 1982; 63 55 Ahlbom Ahlborn G, Skaaning Jensen JC, JC, Gravesen S. An outbreak of allergic alveolitis 63(suppI124): (suppl124): alveolitis in a library. library. Eur J Respir Dis 1982; 1982;63 32 6 Campbell JA, JJ Jr, JA, Kryda MJ, MJ,Treuhaft Treuhaft MW, Marx JJ Jr, Roberts RC. Cheese worker's workers hypersensitivity penumonitis. Am Rev Respir 1983; 127:495-96 Dis 1983; 127:495-96 7 Fergusson RJ. RJ, Milne LJR, Crompton GK. PeniciUium allergic GK. Penidium alveolitis: alveolitis: faulty installation of central heating. Thorax 1984; 39:294-98 8 Lundgren R, Rosenhall L. Fuel chips disease-another disease-another allergic alveolitis. Lakartidningen alveolitis. W i d n i n g e n 1979; 1979; 76:4730 9 Silvennoinen L, van Assendelft Assendelf? A, Luojus E. Socio-economic Socio-economic aspects of farmer's 41 63 (suppll24): (suppl124): 41 farmer's lung. Eur JJ Respir Dis 1982; 1982; 63
Bronchial Associatedwith with Bronchial Atresia Associated a Bronchogenic Cyst Cyst Evidence of Early Early Appearance of Atretic Segments J. Williams, M.B., EC.C.P;* F.C.C.P ;- and Adrian J. Williams, M.B., Samuel R. R. Schuster, M.D . t Schuster, M.D.t We report a case case of congenital lobar emphysema in an adult due to bronchial atresia and presenting characteristically characteristically with a solitary pulmonary pulmonary nodule due to a mucous plug. In the same bronchogenic cyst leads same patient the presence of a bmnchogenic to a hypothesis that the atretic a h t i c segment is the result of an insult occurring during the 5th or 6th week of intrauterine life rather than one occurring after airway development is complete.
developmental disorders of the respiratory system M Manypresent in the postnatal period because of respiratory
any developmental disorders of the respiratory system present in the postnatal period because of respiratory distress and may require surgical surgical intervention. intervention. Such Such anomalies include congenital congenital diaphragmatic diaphragmatic herniae, tracheoesophageal fistulae, asphyxiating asphyxiating thoracic thoracicdystrophy, dystrophy,and and esophageal fistulae, some examples of congenital lobar emphysema. emphysema. However, congenital lobar others are are known to persist into adult adult life and are associated associated
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VA *Director, Chest Unit, Wadsworth V A Medical Center, and Univer· University sitv of California. Los Angeles. tPresently Naples. Florida. Florida. t~rksentlyat Naples, Reprint reauests: requests: Dr. Pulmonary Division. Division, Wadsworth Revrint Dr: Williams, Williams, Pulmonaru WadsworthVA VA Hospital. Los Angeles 90073
396
FIGURE FIGUREl. 1. Posteroanterior Postemanterior chest radiograph shows shows a solitary pulmonary nodule in the left upper zone. This area is seen to be relatively relatively radiolucent. A lucency is also also seen below the left hilum. with a variable variable degree of disordered function function.. The spectrum spans spans the innocuous, innocuous, such as as an azygous azygous lobe, lobe, to the potentially troublesome, such as a bronchogenic cyst or sequestration. tion. Characteristically, these lesions are discovered incidentally with recognition recognition of abnormal findings on the radiograph congenital lobar emphysema in the diograph.. Presentation of ofcongenital adult has been described in this way. When due to bronchial atresia, mucous plug is characteristically atresia, the associated associated mucous characteristically visible on the chest radiograph radiograph and may divert attention from from the true pathology. The purpose of this report is to highlight this this phenomenon. In addition, this case records, for for the first first mediastinal time, the coexistence coexistence of bronchial atresia atresia and a mediastinal bronchogenic cyst, bronchogenic cyst, an experiment of nature which allows allows more precise timing of the development of atretic atretic segments segments.. CASE REPORT
A A 28-year-old woman was was admitted to UCLA Center for for Health Sciences for cone biopsy of the uterine cervix follOWing following the report of abnormal findings smear. She was asymptomatic. An findings on cervical smear. admission radiograph (Fig 1) was reported as as showing a solitary (Fig 1) pulmonary nodule in the left mid-zone. mid-zone, and she was referred for for further evaluation. tobacco evaluation. Direct questioning revealed no history of oftobacco smoking. was no previous radiograph. smoking, cough or dyspnea. There was Physical examination System review was unremarkable. unremarkable. Physical examination showed no abnormal physical physical signs. There was no appreciable alteration of breath sounds over the chest, chest. and no local local abnormality of the chest. Review of the chest film film showed the left mid-zone density. density. The left upper zone was noted to be comparatively radiolucent due to reducjudged by the presence of a normal number of tion in blood flow flow as judged vessels was noted below the vessels of reduced size. size. In addition. addition, a lucency was left hilum. All these features were highlighted by tomography Bronchial Atresia Atresia (WIlliams. (Wlliams, Schuster)
FIGURE 2. Antemposterior Anteroposterior tomographic cut of ofthe chest showing the FIGURE mediastinal cystic lucency. lucency. left mid-zone density and mediastinal (Fig 2). A A selective selective bronchogram defined the extent of the hilar (Fig bronchogenic cyst arising from radiolucency and confirmed it as a bronchogenic (Fig 3). 3). The left upper lobe could not be the left main stem bronchus (Fig filled. The bronchial bronchial anatomy of the remainder of the left lung was was filled. normal. normal. DISCUSSION
of The radiographic features of this case are characteristic of lobar emphysema due to bronchial atresia. A predilection for noted. l The atretic bronchus, the left upper lobe has been noted.' filled with mucus, subtends a segment (or (or lobe) lobe) which is venfilled Lam bert or tilated through collateral routes, the channels of Lambert pores of Kohn. This mode of ventilation favors favors inspiration rather than expiration. expiration. The collateral routes are distended during inspiration, but collapse expirations leading to collapse during expirations air-trapping and hyperinflation. Alveolar development is largely postnatal,' postnatal,2 and in these patients the lack of normal ventilatory stimulus to growth growth leads to a reduction in alveolar number.3 number.= At the same time, because of hyperinflation, air spaces are dilated leading to the designation emphysema. emphysema. The atretic bronchus in such cases lacks lacks a central communication with the bronchial tree so so that bronchial secretions accumulate. accumulate. This This leads to the development of a mucocele m u m e l e which is seen radiographically radiographically as as a solitary pulmonary nodule. nodule. The pathogenesis of the bronchial the blood chial atresia is uncertain, though an interruption of ofthe supply to the affected affected part of the bronchus has been postulated. lated. On repeated occasions, occasions, the bronchial tree (distal (distal to the atresia) Since atresia) has been found found to branch normally.3.4 n~rmally.~.' Since the process of airway airway development is is complete by the 16th 16th week of intrauterine life,2 life,' it has has been said said that the insult which leads leads to to this localized localized abnormality is is likely likely to have have occurred after this. 3 However, the coexistence in this patient of another this.3 However, coexistence in congenital bronchial abnormality, abnormality, a mediastinal bronchial cyst, cyst, supports a different hypothesis. hypothesis. Bronchial development development
FIGURE 3. Selective Selective bronchogram demonstrates the extent of the FIGURE bronchogenic hilar and mediastinal radiolucency radiolucencv and confirms confirms it as a bronchogenic cyst arising arising from the left main-stem bronchus. bronchus. In this procedure, the bronchus could not be filled. filled. The remainder of the left upper lobe bronchus bronchial tree appeared normal. bronchial
has been described in detail by O'Rahilly and Boyden. Boyden.'' The lung bud appears on day 26 after ovulation. ovulation. It divides and elongates into two lung sacs sacs (32 (32 days), days), followed followed by the appearance of lobar buds (36 (36 days). days). All bronchopulmonary segments are started by day 42. 42. Considering Considering the time course of this bronchial development, development, it has been argued, logically logically and appropriately, appropriately, that mediastinal bronchial cysts cysts may appear during the 5th (and (and 6th) 6th) weeks of intrauterine intrauterine life life when the central airways airways are being formed. formed. The left upper lobe segmental bronchi are also also first first seen at the end of this this time and we believe it would would be reasonable to to postulate an an insult at this early stage stage as as the cause of both abnormalities. The advancing advancing growth growth plates are are under local trophic controls controls and could form form a normal peripheral bronchial tree tree beyond the atresia in spite of the interruption in the airway. airway. The The lack lack of pathologic pathologic examination examination in this case, case, however, however, makes other causes necessary. Commakes consideration of ofother causes ofCLE of CLEnecessary. pression of the left left main-stem bronchus with hyperinflation hyperinflation of that lung has has been reported in association association with a bronchogenic cyst cyst in newborn newborn infants,6 infants,=but was not demonstrated on on bronchography in this case. case. Among Among other anomalies anomalies of the respiratory tract implicated as CLE are are abnormal or as causing causing CLE deficient bronchial bronchial cartilages,1.8 cartilages,'.' bronchial mucosal folds, folds,'9 mucous mucous plugs plugs and polyalveolar 10be.1O lobe." Only bronchial bronchial carcarCHEST CHEST I/ 87 I/ 33 I/ MARCH, MARCH. 1985 1985
397
tilage defects have been described with any frequency. BronCLE, but the late chial atresia is relatively rare as a cause of CLE, asymptomatic presentation with typical radiographic features of of a coin lesion makes this diagnosis more likely. The of additional finding of a bronchogenic cyst is an experiment of nature which, assuming both anomalies develop at the same time, allows us to time the appearance of the atretic segment more accurately than has been possible before. REFERENCES }O, Rubin S, Heard BE. Congenital lobar 1 Warner JO, lobar emphysema: emphysema:a bronchial atresia atresia and abnormal case with bronchial abnormal bronchial bronchialcartilages. cartilages.Br 76:177-84 1982;76:177-& J} Dis Chest 1982; development of the respiratory 2 Hislop A. Reid L. Growth and development jA, Dobbing Dobbing}J (eds). (eds). system-anatomical development. development. In: system-anatomical In: Davis JA, Scienti6c foundations foundations of pediatrics. London: Heinemann Scientific Schuster SR. Hanis Harris GBC. GBC, Williams Williams AJ, AJ. Kirkpatrick J, J, Reid L. 3 Schuster Bronchial atresia: atresia: a recognizable recognizable entity in the pediatric pediatric age group. J Ped Surg 1978; 1978; 13:682-89 13:682-89 emphysema. London: London : Lloyd-Luke, Lloyd-Luke. 4 Reid L. The pathology of emphysema. 1967 1967 5 O'Rahilly R, R. Boyden EA. The timing and sequence of events events in development of the human respiratory system during the the development embryonic period proper. Z Anat Entwicklungsgesh Entwicklungsgesh 1973; 141: 1973; 141: embryonic 237-50 237-50 Erakus AJ, AJ, Griscom NT, McGovern JB. J8. Bronchogenic cysts of 6 Erakus infancy. N Engl Eng! J Med 1964; 281:1150-53 1964; 281:1150-53 the mediastinum in infancy. 77 Binet JP, JP, Nezelof C, Fredet J. J. Five cases of lobar tension emphysema in infancy: infancy: importance importance of of bronchial malformation malformation emphysema and value of post-operative steroid 1962; stemid therapy. Dis Chest 1962; 4l:l26-32 41: 126-32 Campbell PE. etiolOgical studies. studies. 8 Campbell PE. Congenital lobar lobar emphysema; etiological 5:226-31 1960; 5:226-31 Aust Pediatr J 1960; ShaSr R, R. JJaffe Bronchiectasis: a muse cause of infantile infantile 9 Shafir d e R. Kalter Y. Bronchiectasis: emphysema. J Pediat Surg 1976; 1976; 11:107-12 11:107-12 lobar emphysema. A. Reid L. New pathological patholOgical findings 6ndings in emphysema of 10 10 Hislop A, 1.. Polyalveolar Polyalveolar lobe with emphysema. emphysema. Thorax 1970; childhood. 1 1970; childhood. 25:682-85 25682-85
IatrogeniC Iatrogenic Fistula Fistula from the Aorta to the Left Coronary Vein* Left Marginal Marginal Coronary P. john lohn Ross, Ross. M.D.; M.D. ; and Geun C.. jang, lang. M.D. M.D. Z? CwnC
We report tthe h e first documented case caseof iatrogenic aortocoroaortocoronary fistula to the left marginal coronary vein following coronary bypass surgery. Unique clinical data, findings findings from catheterization, aand n d angiographic features aare r e prethose in in the seven previously sented and compared with those reported cases of iatrogenic aortocoronary venous fistulae fistulae after coronary bypass operations. atter
A.t
rare but recognized complication of surgery for coronary revascularization rcvascularization isis an aortocoronary venous venous fistula causcd by the inadvertent distal anastomosis of caused of a saphenous vcin bypass graft graft to a coronary vein instead of to a coronary vein artcry. The Thc present patient paticnt had undergone undcrgonc an intended intcndcd byartery. pass operation opcration to the left circumflex circumflcx or intermediate intermcdiatc artery artcry
1"1
-From the Cardiovascular Cardiovascular Laboratories, Laboratories. Loma Lorna Linda University *From Medical Center. Lorna Linda, Linda. CA. CA. Medical Center, Loma Reprint requests: Dr Dr: long. lang. Cardiovascular Laboratories, LaboratOries. Room 2434. Lomo Lorna Lindo Linda University Center. Loma Lorna Linda, Linda. CaliforCalifor2434, Uniwrsity Medical Center, nia 92354 92354
398
FIGURE FIGURE 1.l. Frame from from 35-mm cineangiogram on 70° i O O left anterior projection showing oblique projection showing "numeral-3" appearance appearance of opaci6cation opacification graft (SVG) prox(SVG) anastomosed anastomosed to proxof aortocoronary aortocoronary saphenous saphenous vein graft imalleft (LMY)near its junction with great imal left marginal coronary coronary vein (LMV) cardiac vein (GCV) (GCV)and andso communicating con~municatingvia coronary coronary sinus sinus (CS) (CS) to right atrium (RA). (RA). A. A, distal anastomosis anastomosis of saphenous saphenous vein graft. graft. and is thc aortocoronary the first first dcscribcd described with an iatrogenic aortocoronarv venous fistula fistula to the left marginal coronary vein. Observed featurt>s features of this case are the absence of a continuous murmur and the presence of a small shunt by oximetric studies. studies. CASE REPORT
A 44-year-old A @-year-oldman with recurrent pain in the ('hest chest had a history of myocardial infarction. procedure in 1963. lW,and tripletriplemyocardial infarction. a Vineberg Vineberg prvcedure 1982. On 011 vessel coronary coronary bypass surgery at another hospital in May hlay 1982. physical examination. examination, there was a short. short, soft systolic murmur mllrmur at the murmurs were were deupper left sternal sternal border. border, but no continuous continuous nlurrnilrs tected. The electrocardiogram electrocardiogram showed an "old" "old" inferior inferiormyocardial mycwardial infarcinfarction. A roentgenogram showed persistent postoperative elevaA chest roentgenogram elevation. tion of the left hemidiaphragm. hemidiaphragm. The left ventriculo)(ram ventriculogram was normal. nor~nal. percent. and the left cardiac pressures 50 percent, cardiac pressures The ejection fraction was 50 were normal. normal. The coronary coronary arteriogram demonstrated demonstrated a proximal ocdusion c~vlusionof the dominant right coronary cwronary artery. artery, of whit-h which the posterior descenddescendjump vein ing and and posterolateral posterolateral branches brancheswere supplied supplied by a patent jurnp graft. was a severe proximal stenosis stenosis of the anterior descenddescendgraft. There was second patent bypass ing coronary cwronary artery. with good gvod distal How via a second graft. A moderately moderately large large unbypassed unbypassed intermediate coronary coronary artery graft. A left drcumHex mrohad a signi6cant significant proximal stenosis. The small left circumflex curonary artery had a severe origin. Contrast medium severe stenosis at its origin. Contrast ~nediurn injected down a third bypass graft graft entered the left marginal marginal vein injected close to its junction close junction with the great cardiac cardiac vein and drained directly 1). into the right atrium (Fig (Fig 1). Right cardiae catheterization and andoximetric oximetric studies cardiac catheterization studies (two (two runs) runs) pressures and a small showed normal pressures small hut but definite definite left-to-right left-to-rightshunt shunt at low right atrial level. level. The QP:QS shunt ratio was 1.4:1.0 . 1.4:l.O. DISCUSSION Five published cases of aortocoronary venous fistulae were 'who added another. Sub1982 by Przybojewski. Przybojewski,'who reviewed in 1982 scquently, has appeared. of sequently, onc one more case has appeared,'2 making a total of seven. All were men aged 43 43 to 66 66 years. years, with angina (except (except one who had intractable ventricular tachycardia). Angina was rclieved patients. The sites of graft relieved after surgery in four four of six patients. patient). two (two patients). insertion numbered one (one (one patient), (hvo patients), thrcc (three three (three patients), and four (one (one patient). A continuous Iatrogenic Aorta (Ross. Iatrogenic Fistula Fistula from Aorta (Ross. Jang) Jeng)
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ACKNOWLEDGMENT: The authors thank Tarja Ojanen (UniverUniversity of Kuopio) Kuopio) for Penicillium for performing the precipitin tests on Penicil ium sp. REFERENCES
1 Solley Solley GO, GO, Hyatt RE. Hypersensitivity pneumonitis induced by Penicillium species. J Allerg Clin Immunol 1980; 1980; 65:65-70 6565-70 A, Forsen K-O, K-0, Keskinen H, Alanko K. Humidi2 van Assendelft A, fier-associated fier-associated extrinsic extrinsic allergic alveolitis. Scand J Work Environ Health 1979; 1979; 5:35-41 535-41 33 Pellikka Pellikka M, Kotimaa M. The mould dust concentration caused by the handling of fuel chips and its modifying factors. Folia Forestalia 1983; 1983; 563:1-18 563:l-18 4 Terho EO. Extrinsic allergic alveoli tis-the state of the art. Eur J alveolitis-the Respir Dis 1982; 63 (suppl (suppl 124): 124): 10-26 10-26 1982; 63 55 Ahlbom Ahlborn G, Skaaning Jensen JC, JC, Gravesen S. An outbreak of allergic alveolitis 63(suppI124): (suppl124): alveolitis in a library. library. Eur J Respir Dis 1982; 1982;63 32 6 Campbell JA, JJ Jr, JA, Kryda MJ, MJ,Treuhaft Treuhaft MW, Marx JJ Jr, Roberts RC. Cheese worker's workers hypersensitivity penumonitis. Am Rev Respir 1983; 127:495-96 Dis 1983; 127:495-96 7 Fergusson RJ. RJ, Milne LJR, Crompton GK. PeniciUium allergic GK. Penidium alveolitis: alveolitis: faulty installation of central heating. Thorax 1984; 39:294-98 8 Lundgren R, Rosenhall L. Fuel chips disease-another disease-another allergic alveolitis. Lakartidningen alveolitis. W i d n i n g e n 1979; 1979; 76:4730 9 Silvennoinen L, van Assendelft Assendelf? A, Luojus E. Socio-economic Socio-economic aspects of farmer's 41 63 (suppll24): (suppl124): 41 farmer's lung. Eur JJ Respir Dis 1982; 1982; 63
Bronchial Associatedwith with Bronchial Atresia Associated a Bronchogenic Cyst Cyst Evidence of Early Early Appearance of Atretic Segments J. Williams, M.B., EC.C.P;* F.C.C.P ;- and Adrian J. Williams, M.B., Samuel R. R. Schuster, M.D . t Schuster, M.D.t We report a case case of congenital lobar emphysema in an adult due to bronchial atresia and presenting characteristically characteristically with a solitary pulmonary pulmonary nodule due to a mucous plug. In the same bronchogenic cyst leads same patient the presence of a bmnchogenic to a hypothesis that the atretic a h t i c segment is the result of an insult occurring during the 5th or 6th week of intrauterine life rather than one occurring after airway development is complete.
developmental disorders of the respiratory system M Manypresent in the postnatal period because of respiratory
any developmental disorders of the respiratory system present in the postnatal period because of respiratory distress and may require surgical surgical intervention. intervention. Such Such anomalies include congenital congenital diaphragmatic diaphragmatic herniae, tracheoesophageal fistulae, asphyxiating asphyxiating thoracic thoracicdystrophy, dystrophy,and and esophageal fistulae, some examples of congenital lobar emphysema. emphysema. However, congenital lobar others are are known to persist into adult adult life and are associated associated
-
-
VA *Director, Chest Unit, Wadsworth V A Medical Center, and Univer· University sitv of California. Los Angeles. tPresently Naples. Florida. Florida. t~rksentlyat Naples, Reprint reauests: requests: Dr. Pulmonary Division. Division, Wadsworth Revrint Dr: Williams, Williams, Pulmonaru WadsworthVA VA Hospital. Los Angeles 90073
396
FIGURE FIGUREl. 1. Posteroanterior Postemanterior chest radiograph shows shows a solitary pulmonary nodule in the left upper zone. This area is seen to be relatively relatively radiolucent. A lucency is also also seen below the left hilum. with a variable variable degree of disordered function function.. The spectrum spans spans the innocuous, innocuous, such as as an azygous azygous lobe, lobe, to the potentially troublesome, such as a bronchogenic cyst or sequestration. tion. Characteristically, these lesions are discovered incidentally with recognition recognition of abnormal findings on the radiograph congenital lobar emphysema in the diograph.. Presentation of ofcongenital adult has been described in this way. When due to bronchial atresia, mucous plug is characteristically atresia, the associated associated mucous characteristically visible on the chest radiograph radiograph and may divert attention from from the true pathology. The purpose of this report is to highlight this this phenomenon. In addition, this case records, for for the first first mediastinal time, the coexistence coexistence of bronchial atresia atresia and a mediastinal bronchogenic cyst, bronchogenic cyst, an experiment of nature which allows allows more precise timing of the development of atretic atretic segments segments.. CASE REPORT
A A 28-year-old woman was was admitted to UCLA Center for for Health Sciences for cone biopsy of the uterine cervix follOWing following the report of abnormal findings smear. She was asymptomatic. An findings on cervical smear. admission radiograph (Fig 1) was reported as as showing a solitary (Fig 1) pulmonary nodule in the left mid-zone. mid-zone, and she was referred for for further evaluation. tobacco evaluation. Direct questioning revealed no history of oftobacco smoking. was no previous radiograph. smoking, cough or dyspnea. There was Physical examination System review was unremarkable. unremarkable. Physical examination showed no abnormal physical physical signs. There was no appreciable alteration of breath sounds over the chest, chest. and no local local abnormality of the chest. Review of the chest film film showed the left mid-zone density. density. The left upper zone was noted to be comparatively radiolucent due to reducjudged by the presence of a normal number of tion in blood flow flow as judged vessels was noted below the vessels of reduced size. size. In addition. addition, a lucency was left hilum. All these features were highlighted by tomography Bronchial Atresia Atresia (WIlliams. (Wlliams, Schuster)
FIGURE 2. Antemposterior Anteroposterior tomographic cut of ofthe chest showing the FIGURE mediastinal cystic lucency. lucency. left mid-zone density and mediastinal (Fig 2). A A selective selective bronchogram defined the extent of the hilar (Fig bronchogenic cyst arising from radiolucency and confirmed it as a bronchogenic (Fig 3). 3). The left upper lobe could not be the left main stem bronchus (Fig filled. The bronchial bronchial anatomy of the remainder of the left lung was was filled. normal. normal. DISCUSSION
of The radiographic features of this case are characteristic of lobar emphysema due to bronchial atresia. A predilection for noted. l The atretic bronchus, the left upper lobe has been noted.' filled with mucus, subtends a segment (or (or lobe) lobe) which is venfilled Lam bert or tilated through collateral routes, the channels of Lambert pores of Kohn. This mode of ventilation favors favors inspiration rather than expiration. expiration. The collateral routes are distended during inspiration, but collapse expirations leading to collapse during expirations air-trapping and hyperinflation. Alveolar development is largely postnatal,' postnatal,2 and in these patients the lack of normal ventilatory stimulus to growth growth leads to a reduction in alveolar number.3 number.= At the same time, because of hyperinflation, air spaces are dilated leading to the designation emphysema. emphysema. The atretic bronchus in such cases lacks lacks a central communication with the bronchial tree so so that bronchial secretions accumulate. accumulate. This This leads to the development of a mucocele m u m e l e which is seen radiographically radiographically as as a solitary pulmonary nodule. nodule. The pathogenesis of the bronchial the blood chial atresia is uncertain, though an interruption of ofthe supply to the affected affected part of the bronchus has been postulated. lated. On repeated occasions, occasions, the bronchial tree (distal (distal to the atresia) Since atresia) has been found found to branch normally.3.4 n~rmally.~.' Since the process of airway airway development is is complete by the 16th 16th week of intrauterine life,2 life,' it has has been said said that the insult which leads leads to to this localized localized abnormality is is likely likely to have have occurred after this. 3 However, the coexistence in this patient of another this.3 However, coexistence in congenital bronchial abnormality, abnormality, a mediastinal bronchial cyst, cyst, supports a different hypothesis. hypothesis. Bronchial development development
FIGURE 3. Selective Selective bronchogram demonstrates the extent of the FIGURE bronchogenic hilar and mediastinal radiolucency radiolucencv and confirms confirms it as a bronchogenic cyst arising arising from the left main-stem bronchus. bronchus. In this procedure, the bronchus could not be filled. filled. The remainder of the left upper lobe bronchus bronchial tree appeared normal. bronchial
has been described in detail by O'Rahilly and Boyden. Boyden.'' The lung bud appears on day 26 after ovulation. ovulation. It divides and elongates into two lung sacs sacs (32 (32 days), days), followed followed by the appearance of lobar buds (36 (36 days). days). All bronchopulmonary segments are started by day 42. 42. Considering Considering the time course of this bronchial development, development, it has been argued, logically logically and appropriately, appropriately, that mediastinal bronchial cysts cysts may appear during the 5th (and (and 6th) 6th) weeks of intrauterine intrauterine life life when the central airways airways are being formed. formed. The left upper lobe segmental bronchi are also also first first seen at the end of this this time and we believe it would would be reasonable to to postulate an an insult at this early stage stage as as the cause of both abnormalities. The advancing advancing growth growth plates are are under local trophic controls controls and could form form a normal peripheral bronchial tree tree beyond the atresia in spite of the interruption in the airway. airway. The The lack lack of pathologic pathologic examination examination in this case, case, however, however, makes other causes necessary. Commakes consideration of ofother causes ofCLE of CLEnecessary. pression of the left left main-stem bronchus with hyperinflation hyperinflation of that lung has has been reported in association association with a bronchogenic cyst cyst in newborn newborn infants,6 infants,=but was not demonstrated on on bronchography in this case. case. Among Among other anomalies anomalies of the respiratory tract implicated as CLE are are abnormal or as causing causing CLE deficient bronchial bronchial cartilages,1.8 cartilages,'.' bronchial mucosal folds, folds,'9 mucous mucous plugs plugs and polyalveolar 10be.1O lobe." Only bronchial bronchial carcarCHEST CHEST I/ 87 I/ 33 I/ MARCH, MARCH. 1985 1985
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tilage defects have been described with any frequency. BronCLE, but the late chial atresia is relatively rare as a cause of CLE, asymptomatic presentation with typical radiographic features of of a coin lesion makes this diagnosis more likely. The of additional finding of a bronchogenic cyst is an experiment of nature which, assuming both anomalies develop at the same time, allows us to time the appearance of the atretic segment more accurately than has been possible before. REFERENCES }O, Rubin S, Heard BE. Congenital lobar 1 Warner JO, lobar emphysema: emphysema:a bronchial atresia atresia and abnormal case with bronchial abnormal bronchial bronchialcartilages. cartilages.Br 76:177-84 1982;76:177-& J} Dis Chest 1982; development of the respiratory 2 Hislop A. Reid L. Growth and development jA, Dobbing Dobbing}J (eds). (eds). system-anatomical development. development. In: system-anatomical In: Davis JA, Scienti6c foundations foundations of pediatrics. London: Heinemann Scientific Schuster SR. Hanis Harris GBC. GBC, Williams Williams AJ, AJ. Kirkpatrick J, J, Reid L. 3 Schuster Bronchial atresia: atresia: a recognizable recognizable entity in the pediatric pediatric age group. J Ped Surg 1978; 1978; 13:682-89 13:682-89 emphysema. London: London : Lloyd-Luke, Lloyd-Luke. 4 Reid L. The pathology of emphysema. 1967 1967 5 O'Rahilly R, R. Boyden EA. The timing and sequence of events events in development of the human respiratory system during the the development embryonic period proper. Z Anat Entwicklungsgesh Entwicklungsgesh 1973; 141: 1973; 141: embryonic 237-50 237-50 Erakus AJ, AJ, Griscom NT, McGovern JB. J8. Bronchogenic cysts of 6 Erakus infancy. N Engl Eng! J Med 1964; 281:1150-53 1964; 281:1150-53 the mediastinum in infancy. 77 Binet JP, JP, Nezelof C, Fredet J. J. Five cases of lobar tension emphysema in infancy: infancy: importance importance of of bronchial malformation malformation emphysema and value of post-operative steroid 1962; stemid therapy. Dis Chest 1962; 4l:l26-32 41: 126-32 Campbell PE. etiolOgical studies. studies. 8 Campbell PE. Congenital lobar lobar emphysema; etiological 5:226-31 1960; 5:226-31 Aust Pediatr J 1960; ShaSr R, R. JJaffe Bronchiectasis: a muse cause of infantile infantile 9 Shafir d e R. Kalter Y. Bronchiectasis: emphysema. J Pediat Surg 1976; 1976; 11:107-12 11:107-12 lobar emphysema. A. Reid L. New pathological patholOgical findings 6ndings in emphysema of 10 10 Hislop A, 1.. Polyalveolar Polyalveolar lobe with emphysema. emphysema. Thorax 1970; childhood. 1 1970; childhood. 25:682-85 25682-85
IatrogeniC Iatrogenic Fistula Fistula from the Aorta to the Left Coronary Vein* Left Marginal Marginal Coronary P. john lohn Ross, Ross. M.D.; M.D. ; and Geun C.. jang, lang. M.D. M.D. Z? CwnC
We report tthe h e first documented case caseof iatrogenic aortocoroaortocoronary fistula to the left marginal coronary vein following coronary bypass surgery. Unique clinical data, findings findings from catheterization, aand n d angiographic features aare r e prethose in in the seven previously sented and compared with those reported cases of iatrogenic aortocoronary venous fistulae fistulae after coronary bypass operations. atter
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rare but recognized complication of surgery for coronary revascularization rcvascularization isis an aortocoronary venous venous fistula causcd by the inadvertent distal anastomosis of caused of a saphenous vcin bypass graft graft to a coronary vein instead of to a coronary vein artcry. The Thc present patient paticnt had undergone undcrgonc an intended intcndcd byartery. pass operation opcration to the left circumflex circumflcx or intermediate intermcdiatc artery artcry
1"1
-From the Cardiovascular Cardiovascular Laboratories, Laboratories. Loma Lorna Linda University *From Medical Center. Lorna Linda, Linda. CA. CA. Medical Center, Loma Reprint requests: Dr Dr: long. lang. Cardiovascular Laboratories, LaboratOries. Room 2434. Lomo Lorna Lindo Linda University Center. Loma Lorna Linda, Linda. CaliforCalifor2434, Uniwrsity Medical Center, nia 92354 92354
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FIGURE FIGURE 1.l. Frame from from 35-mm cineangiogram on 70° i O O left anterior projection showing oblique projection showing "numeral-3" appearance appearance of opaci6cation opacification graft (SVG) prox(SVG) anastomosed anastomosed to proxof aortocoronary aortocoronary saphenous saphenous vein graft imalleft (LMY)near its junction with great imal left marginal coronary coronary vein (LMV) cardiac vein (GCV) (GCV)and andso communicating con~municatingvia coronary coronary sinus sinus (CS) (CS) to right atrium (RA). (RA). A. A, distal anastomosis anastomosis of saphenous saphenous vein graft. graft. and is thc aortocoronary the first first dcscribcd described with an iatrogenic aortocoronarv venous fistula fistula to the left marginal coronary vein. Observed featurt>s features of this case are the absence of a continuous murmur and the presence of a small shunt by oximetric studies. studies. CASE REPORT
A 44-year-old A @-year-oldman with recurrent pain in the ('hest chest had a history of myocardial infarction. procedure in 1963. lW,and tripletriplemyocardial infarction. a Vineberg Vineberg prvcedure 1982. On 011 vessel coronary coronary bypass surgery at another hospital in May hlay 1982. physical examination. examination, there was a short. short, soft systolic murmur mllrmur at the murmurs were were deupper left sternal sternal border. border, but no continuous continuous nlurrnilrs tected. The electrocardiogram electrocardiogram showed an "old" "old" inferior inferiormyocardial mycwardial infarcinfarction. A roentgenogram showed persistent postoperative elevaA chest roentgenogram elevation. tion of the left hemidiaphragm. hemidiaphragm. The left ventriculo)(ram ventriculogram was normal. nor~nal. percent. and the left cardiac pressures 50 percent, cardiac pressures The ejection fraction was 50 were normal. normal. The coronary coronary arteriogram demonstrated demonstrated a proximal ocdusion c~vlusionof the dominant right coronary cwronary artery. artery, of whit-h which the posterior descenddescendjump vein ing and and posterolateral posterolateral branches brancheswere supplied supplied by a patent jurnp graft. was a severe proximal stenosis stenosis of the anterior descenddescendgraft. There was second patent bypass ing coronary cwronary artery. with good gvod distal How via a second graft. A moderately moderately large large unbypassed unbypassed intermediate coronary coronary artery graft. A left drcumHex mrohad a signi6cant significant proximal stenosis. The small left circumflex curonary artery had a severe origin. Contrast medium severe stenosis at its origin. Contrast ~nediurn injected down a third bypass graft graft entered the left marginal marginal vein injected close to its junction close junction with the great cardiac cardiac vein and drained directly 1). into the right atrium (Fig (Fig 1). Right cardiae catheterization and andoximetric oximetric studies cardiac catheterization studies (two (two runs) runs) pressures and a small showed normal pressures small hut but definite definite left-to-right left-to-rightshunt shunt at low right atrial level. level. The QP:QS shunt ratio was 1.4:1.0 . 1.4:l.O. DISCUSSION Five published cases of aortocoronary venous fistulae were 'who added another. Sub1982 by Przybojewski. Przybojewski,'who reviewed in 1982 scquently, has appeared. of sequently, onc one more case has appeared,'2 making a total of seven. All were men aged 43 43 to 66 66 years. years, with angina (except (except one who had intractable ventricular tachycardia). Angina was rclieved patients. The sites of graft relieved after surgery in four four of six patients. patient). two (two patients). insertion numbered one (one (one patient), (hvo patients), thrcc (three three (three patients), and four (one (one patient). A continuous Iatrogenic Aorta (Ross. Iatrogenic Fistula Fistula from Aorta (Ross. Jang) Jeng)