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ANOMALIES OF UPPER LOBE BRONCHIAL DISTRIBUTION
ANOMALIES OF UPPER LOBE BRONCHIAL DISTRIBUTION Eugene G. Laforet, MD.,* George W. B. Starkey, MD.,** and Saul Scheff, MD., *** Boston, Mass. the province principally of anatomists, anomalies of bronchial distribution have assumed clinical importance because of improved methods for their detection and the ever-increasing use of pulmonary resection. While the pattern of bronchial arborization on the whole is remarkably constant, the gross variations that do occur are largely confined to the upper lobes. It is the purpose of this paper to describe the significant clinical and roentgenographic features in 10 such cases. ,
L
ONG
CASE REPORTS
CASE I.-On March 11, 1947, this 5-day-old girl was, admitted to the Children's Hospital Medical Center, Boston, because of choking and cyanosis following feedings. A Lipiodol esophagram demonstrated a tracheoesophageal fistula and outlined the tracheobronchial tree. The adventitious bronchogram (Fig. 1) showed the right upper lobe bronchus to originate from the trachea. Operative closure of a high tracheoesophageal fistula was performed on the second hospital day but the patient did poorly and died 5 days after operation. Autopsy showed the right upper lobe bronchus to arise directly from the trachea but there were no gross abnormalities of the pulmonary vessels. CASE 2.-This 65-year-old man was seen at Massachusetts Memorial Hospitals in June, 1953, because routine x-ray studies had demonstrated two rounded densities at the right hilus. He was free of respiratory symptoms. Bronchoscopy showed that the trachea was normal to a point 1.5 em, above the main carina, at which level a trifurcate right upper lobe bronchus was seen with the right-angle lens. The remainder of the endoscopy was unremarkable except for redness and edema of the left upper lobe orifice. Laminagraphy confirmed the diagnosis of abnormal origin of the right upper lobe bronchus from the trachea but showed no tumor. The patient was discharged. CASE 3.-The patient was a 37·year-old woman who was admitted to the Sanatorium Division, Boston City Hospital, because of right upper lobe tuberculosis. Neither bronchoscopy nor laminagraphy showed a bronchial abnormality. After appropriate drug therapy, right upper lobectomy was performed on Jan. 15, 1957, for residual cavitary disease. At operation an anomalous small bronchus was found which arose from the right lateral aspect From the Department of Surgery, Boston University School of Medicine, and the Thoracic Surgery Services of the Boston City Hospital and Massachusetts Memorial Hospitals. Received for publication May 12, 1961. 'Clinical Instructor in Sur-ger-y, Boston University School of Medicine; Associate Visiting Surgeon for Thoracic Surgery, Boston City Hospital; Assistant in Surgery. Massachusetts Memorial Hospitals. '-Clinical Associate in Surgery, Harvard Medical School, and Instructor in Surgery, Boston University School of Medicine; Associate Visiting Surgeon for Thoracic Surgery, Boston City Hospital; Surgeon, Children's Hospital, Boston. - _. Assistant Professor of Radiology, Boston University School of Medicine; Associate Radiologist, Massachusetts Memorial Hospitals.
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of the trachea slightly above the main carina. The right upper lobe bronchus was trifurcate and in normal position. The cavity in the resected specim en yielded acid-fast bacilli and was positive for Mycobact erium tuber culosis on culture. CASE 4.-This 51-year-old male alcoholic entered the San atorium Division, Boston City Hospital, in .I uly, 1955, becau se of pulmonary tuberculosis. Bronchoscopy and laminagraphy demonstrated no bronchial abnormality. On Dec. ll, 1956, right upper lobectomy was done becau se of persi sting ca vitary disease. Thoracotom y disclosed that pleural symphysis involved th e entire upp er lob e. Dissection revealed an azygos lobe which was complet ely destroyed and replaced by large cysts and bullae. The apical and posterior segments showed ext ensive fibrotic changes. With considerable difficulty the a zygos lobe was mobilized and passed beneath th e azygo s vein . It was th en found that th er e was a sepa rate artery to the
Fig. I.- Ca se 1.
Esophagram and adventitious bronchogram which shews entire r ight upp er lobe br onchus to or iginate trom the trachea.
azygos lob e from the main pulmonary artery and tha t the venous drainage was als o by way of a distinct vessel. In addition, it was served by a separate bronchus arising f rom the r ight main-stem bronchus proximal to the origin of an apparently trifurcate upper lobe bronchus which occupied the normal position. Th e operati ve specimen showed a destroyed upper lobe with num erous cavities and cysts, smear and cult ure of which were negative for tuberculosis. CASE 5.-0n May 13, 1960, this 43-year-old man und erwent br onchoscopy at the Soldiers ' Home , Chelsea, Mass., because of hemoptysis of und etermined etiology. There was diffuse endobronchitis, more marked on the left. In addition, there was a small bronchial orifice on the right lateral aspect of the trachea at approximately the level of the ca ri na, an d a larger distal upp er lobe orifice originating from the right main- st em bronchus. Unfortunately bronchography was not performed. CASE 6.-This 62-year-old man was an inv et erate smoker who was admitted to the Soldiers' Home, Chelsea, Mass ., for inve stigation of a productive cough. Lingular pn eumonitis had occurred twice during the preceding 12 months. On March 2, 1959, the findings at bronchoscopy were unremarkable except for acute endobronchitis of the basal bronchi on the left sid e. No abnormal bronchus was noted on the right but a right-angle lens was not available. A bilateral bronchogram was then performed and showed normal lingular fiIIing. As an incidental finding, there was a small tracheal bron chus on the right slightly above the level of the main carina. CASE 7.-A 47-year-old man was seen because of rec ent worsening of a chronic product ive cough. A chest roentgenogram was interpreted as showing post-pneumonic atelectasis of
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the left lower lobe. Findings at bronchoscopy on April 10, 1958, at the Carney Hospital, Boston, wer e unremarkabl e except for an a bnorm al right upper lob e bronchial orifi ce just below th e level of t he car ina, with th e orifice to the r emainder of the upper lobe being visualized in the expect ed location. The diagnosis of anom alous r ight upper lobe br onchus was confirmed by a bronchogram done on May 13, 1958, at Quincy Cit y Hospital, Quincy, Mass. (Fig. 2). In addition, there was saccular bronchi ectasis of th e left lower lobe for which res ection was succes sf ull y undertak en. CASE 8.-Brisk hemoptysis prompted investigation of this 56-yea r-old man. A ches t roentgenogram was interpreted as sho wing tuberculosis of the right upper lobe . In an effor t to locali ze the source of the hemorrhage , emergency bronchoscopy was p erformed at N ewton 'Wellesley Hospital, Newton, Mass., on July 28, 1955. Th e profuse ble eding appeared t o
F ig. 2.- Case 7.- Br onchogr am which shows anomalous or igin of a segmental bronchus of th e right upp er lobe ( indicate d by arrow). The position of th e horizontal fissu re wa s clearly seen and is marked on the illustration. originate from a markedly contracted ri g ht upper lob e ori fice. Th e hemop ty sis ceas ed spontaneously and the diagnosis of pulmonary tuberculosis was confirmed by th e results of guinea pig inoculat ion . Appropr iate antimicrobial therapy was instituted. Bronchography was attempted on 2 separate occasions but was unsuccessful because of intractable cough. Laminagrams were performed at Massachusetts Memorial Hospitals on Sept. 22, 1955, to delineate th e extent of the r esidu al disease. In addit ion to persisting fibrocaseous tuberculosis in the right upper lob e, these st udies demonstrated an a nomalous bronchu s t o t his lobe from t he trachea, slightly proximal to th e level of th e main car ina . It th en becam e apparent that t he endoscopic interpretation of marked ste nosis of the right upper lobe bronchial orifi ce had been in er ror, sin ce th e orifice visua liz ed r epresent ed only part of th e upper lob e supply. Ri ght upper lob ectomy was perform ed a t Mas sachusetts Memorial H ospitals on March 14, 1956, f or r esidual fibrocaseous tubercul osis. At oper ation ther e was a separ ate bronchus to t he apical segment which a rose from th e trachea at a pprox ima tely th e level of tile main car ina. Th e bronchus to t he r ema ining upper lob e seg ments was found a t th e norm al site . CASE 9.-This patient was a 79-year-old man who was a dmi t te d to Ma ssachus etts Memorial Hospitals on May 20, 1957, because of dyspn ea and a cough productive of bloody sputum. Nephrectomy had been pe rfor med 4 years 'Previously for carc inoma . X -ra y st udies
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of the chest showed an infiltrate at the bases of both lungs and prominence of the right hilus. Two therapeutic bronchoscopies were done because of the patient's inability to raise secretions, but no abnormal bronchus was seen on either occasion. Laminagrams showed no metastatic deposits in the lungs, but demonstrated an anomalous right upper lobe bronchus arising from the distal trachea (Fig. 3). The patient died and permission for autopsy was withheld.
Fig. 3.--Case 9. Laminagram demonstrating ectopic segmental right upper lobe bronchus arising above main carina. (From Scheff, Kaufman, and Levene, Radiology 70: 82-83, 1958.) Ar'row indicates the anomalous bronchus. The significant features of the laminagram have been emphasized for improved photographic reproduction. CASE 10.-This 40-year-old woman was admitted to the Sanatorium Division, Boston City Hospital, in November, 1955, because of active tuberculosis involving both upper lobes. On March 1, 1956, laminagrams were secured to evaluate the results of therapy. Bilateral apical cavities were present and, in addition, there was a suggestion of an anomalous bronchus originating from the left main stem. Findings at bronchoscopy on May 23, 1956, were unremarkable and the bronchial anomaly was not detected. However, a bronchogram performed immediately thereafter demonstrated that the apical-posterior segment of the left upper lobe was supplied by an individual bronchus arising proximal to the bronchus serving the remaining upper lobe segments (Fig. 4). On June 7, 1956, resection of the apical-posterior segment of the left upper lobe was accomplished, the procedure being technically facilitated by the independent origin of the segmental bronchus.
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In Table I are recorded the salient features of the 10 preceding cases. Case 4, however, warrants special consideration since, as far as can be determined, it is the first recorded instance of an azygos lobe being supplied by an anomalous separate bronchus. According to Boyden,' certain fundamental conceptions are of importance concerning the azygos lobe, namely, "that the lobus is not a supernumerary structure but merely a segregated portion of the right upper lobe, that it is aerated by a variable number of bronchi from one or more segments, and that the descending wedge of the azygos bends certain bronchi, thereby favoring such sequelae as atelectasis, bronchiectasis, and the like." These points are emphasized by Gerson and Rothstein," who state that an anomalous
Fig. 4.-Case 10.
Bronchogram which demonstrates separate origin of apical-posterior segmental bronchus proximal to left upper lobe bronchus.
tracheal bronchus to the right upper lobe" is never related to another anomaly, the azygos lobe, also located in this portion of the chest. The azygos lobe is an anomaly of the azygos vein rather than of the lung and the separated azygos lobe is supplied by the usual bronchial branches. Foster-Carter presents detailed anatomical studies of the bronchial and vascular supply to the azygos lobe, but in none was any bronchial anomaly or tracheal bronchus discovered." Does Case 4 modify or nullify these concepts of the azygos lobe ~ We believe not, since it seems most likely that the association of anomalous bronchus and azygos lobe in this patient was merely coincidental. Indeed, on a statistical basis such an occurrence might even have been predictable. ETIOLOGY
As with most developmental defects the etiology of upper lobe bronchial abnormalities is not known. Har-ris" has summarized present knowledge concerning the evolutionary development, embryology, and comparative anatomy of the lung as related to the occurrence of these anomalies. Phylogenetic features have been emphasized by Schaff and Baum.'
Separate proximal bronchus to apical segment; distal bifurcate bronchus to remaining RUL segments
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Right
43
5
Normally positioned trifurcate RUL bronchus; proximally, a separate small bronchus from right main stem to azygos lobe
Right
M
51
4
Normally positioned trifurcate RUL bronchus; small tracheal bronchus to apical segment
Right
F
37
3
Trifurcate RUL bronchus arising from trachea
Trifurcate RUL bronchus arising from trachea
M
65
2
Right
X
(X)
(X)
X
-
-
X
-
-
(X)
(X)
-
X'"
-
x
No
Yes
Yes
-
X
No
No
X
-
(X)
DISEASE IN TISSUE SERVED MEANS OF DIAGNOSIS BY ABNORMAL BRONCHOS-/ BRONCHOG-ILAMINAG-IOPERA-I AUCOPY RAPHY RAPHY TION TOPSY BRONCHUS
Routine chest xray reported ' , abnormal, ' , with" 2 rounded hilar densities"
Died 5 davs after operation
REMARKS
Diffuse endobronchitis
Hemoptysis
Right upper 10Fibrocaseous tubectomy berculosis, RUL
Fibrocaseous tuRight upper 10bcrculosis, RUL bectomy
None
Tracheoesophageal fistula
PRIMARY DISEASE
SUMMARY OF PERTINENT DATA IN TEN PATIENTS WITH UPPER LOBE BRONCHIAL ANOMALIES
TYPE OF ANOMALY
1.
Right
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SIDE OF SEX ANOMALY
1
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TABLE
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Right
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Right
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(X)
X
(X)t
Separate proximal bronchus to apicalposterior segIllent (X)
Separate proximal (X)§ (X) bronchus to apical segment; distal bifurcate bronchus to remaining RUL segments
Separate proximal bronchus to apical segment; distal bifurcate bronchus to remaining RUL segments
Separate proximal bronchus to apical segment; distal bifurcate bronchus to remaining RUL segments
Separate proximal bronchus to apical segment; distal bifurcate bronchus to remaining RUL segments
X
-
=
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X
X
X
X
X
-
X
X
-
-
-
-
-
-
-
Yes
No
Yes
No
No
:j:Two attempts at bronchography unsuccessful because of intractable cough. §Therapeutic bronchoscopies.
Profuse bleeding at time of bronchoscopy; right upper lobectomy
Left lower lobectomy
Productive cough
not per-
Resection of A-P segment LUL
- = Examination
Cavitary tubercnIosis, A-P segment LUL
Died Arteriosclerotic heart disease and metastatic renal carcinoma
Cavitary tubercuIosis, RUL
Bronchiectasis, LLL
Bronchitis, LLL
Legend: X == Examination performed and diagnostic. (X) Examination performed but not diagnostic. formed. The Iamlnagraphtc features of Cases 2, 8, and 9 have been previouslv reported.'. °Lipiodol swallow given to demonstrate tracheoesophageal fistula. tRight-angle lens not available.
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62
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LAFORET, STARKEY, SCHEFF
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j. Thoracic and Cardiovas, Surg.
INCIDENCE
Although the 10 cases reported here have been garnered over a period of 2 years from several sources, chronologically they extend over a longer interval. For this and other reasons it is impossible to estimate the total experience from which these examples have been culled. Consequently little can be said about the clinical incidence of such anomalies and even less about their absolute incidence. As is true generally, the number of cases diagnosed bears a direct relation to the prevailing level of awareness and interest. In a review of the Japanese literature, lnada and Kiahimoto" found that the reported incidence of anomalous tracheal bronchus to the right upper lobe ranged from 0.1 to 0.6 per cent. It is of interest that on an embryologic basis one might expect a greater frequency than occurs clinically. Bremer," for example, demonstrated abnormal bronchial buds from the right side of the trachea in 4 of 80 embryos but believed that in most instances these are resorbed, thus accounting for the lower incidence in adults.
Fig. 5.-DiagrammaNo repreeentatum. oj the anomalies o] upper lobe bronohial supply encountered in the present series. A, The usual bronchial distribution. B, Origin of the entire right upper lobe bronchus from the trachea (2 patients). 0, A small tracheal bronchus coexisting with an apparently normal right upper lobe bronchus (l patient). D, Normal origin and distribution of right upper lobe bronchus with a separate small bronchus from the right main-stem bronchus to an azygos lobe (1 patient). E, The most common anomaly, separate origin of the apical segmental bronchus proximal to the common origin of anterior and posterior segmental bronchi (5 patients). F, The only left-sided abnormality encountered in the present series but the commonest variant on this side, separate origin of the apicalposterior segmental bronchus proximal to the origin of the remainder of the superior division (1 patient). CLASSIFICA'l'ION
Upper lobe bronchial anomalies have been the subject of considerable taxonomic study. Anatomically"- 8 a valid distinction has been made between displaced and supernumerary bronchi. If intralobar segmentation is normal but the mode of bronchial origin is not, the abnormal bronchus is termed displaced. A true supernumerary bronchus is much less common and implies that the bronchus serves pulmonary parenchyma that is definitely in excess of the normal.
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This classification necessarily involves detailed anatomic analysis. Thus, for example, the mere demonstration of a trifurcate upper lobe bronchus in conjunction with a tracheal bronchus on this side is not sufficient to permit designation of the latter as supernumerary, unless it can be shown that it supplies lung parenchyma in excess of that normally encountered in the right upper lobe." 8 'When only clinical data are available, efforts to compress upper lobe bronchial anomalies into this anatomic classification may be inaccurate and confusing. As a result, several systems related chiefly to the chinical aspects of right upper lobe bronchial abnormalities have been proposed.v " In order to encompass anomalies of the left upper lobe as well, the following descriptive classification is offered, based on the present material and reports in the literature: Type I.-The upper lobe is anatomically complete but its bronchus arises from the trachea (entire upper lobe bronchus displaced onto trachea). Type Il.-An accessory bronchus arises proximal to an apparently normal upper lobe bronchus, either (A) from the trachea or (B) from the main stem. Type IIl.-A segmental bronchus (usually the apical or apical-posterior) is "split" from the lobar bronchus and arises proximal to it, either ,(A) from the trachea or (B) from the main stem. Fig. 5 presents a diagrammatic representation of the present cases on the basis of this classification. DIAGNOSIS
Bronchography is the most precise and accurate means of establishing the clinical diagnosis of upper lobe bronchial anomalies and, in the present series, was conclusive in all 4 instances in which it was successfully employed. It should be noted, however, that the abnormal bronchus may not be outlined unless careful attention is given to the trachea during the examination. Laminagraphy affords a reliable technique for the demonstration of upper lobe bronchial anomalies"? and was diagnostic on 4 of 6 occasions (cf. Table I). Conventional radiographic technique, however, has not been helpful. Bronchoscopy was performed in all but one case and was diagnostic in only 3. One patient underwent 2 separate endoscopic examinations without detection of the abnormality but the primary indication for the procedures was therapeutic and not diagnostic. In Case 8 there was profuse bleeding at the time of bronchoscopy and the anomalous orifice was not visualized, although the source of bleeding was correctly established as the right upper lobe. In Case 6, a right-angle lens was not available at the time of bronchoscopy and diagnosis was not accomplished. Thus, while precise interpretation of the findings may be difficult, bronchoscopy is an additional diagnostic measure that may be rewarding. Awareness of the possibility of bronchial anomalies and use of the right-angle lens to inspect the trachea as well as the major bronchi should improve chances for diagnosis." Operation disclosed the abnormal bronchus in 4 of 5 patients, In 2 of those, its presence had been unsuspected preoperatively. Since the operation that dill not disclose the abnormality was done for tracheoesophageal fistula, demonstration of the abnormality could not have been expected. The only autopsy in the
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series confirmed the clinical diagnosis of anomalous right upper lobe bronchus (Case 1). However, routine necropsy technique may be inadequate to demonstrate such anomalies and increased awareness of these variants is as necessary for the pathologist as for the clinician. CLINICAL SIGNIFICANCE
Whether lung parenchyma served by an abnormal bronchus is intrinsically more susceptible to disease is uncertain. According to Harris," "Once identified, the tracheal bronchus has no particular clinical significance." However, others 4 , 12 believe that such bronchi are associated with an unusually high incidence of disease, particularly bronchiectasis, in the area of lung to which they are distributed. In one recorded instance," the anomalous bronchus was the seat of an adenoma which was successfully resected. Much of the difficulty in assessing the clinical significance of these variations stems from indefinite information about their true incidence, since there undoubtedly are individuals with such defects who remain asymptomatic and never come to medical attention. Disease was absent in tissue served by the abnormal bronchus in 6 of the 10 patients comprising the present series. The etiology of the disease in the 4 remaining patients was tuberculosis and not obviously related to the bronchial abnormality. In Case 4, the additional finding of cystic changes in the azygos lobe was almost certainly the result of the abnormal vein rather than the malpositioned bronchus. In cases of the type here presented, then, it seems likely that the bronchial abnormality is principally one of position and is not otherwise associated with altered form or function of either the bronchus or its subtended parenchyma. An exception might exist if the abnormal bronchus is so situated that its drainage is inadequate. Prescinding from the question of intrinsic vulnerability to disease, however, anomalous bronchi by their mere presence may be of considerable clinical importance. In some instances" a bronchoscopic diagnosis of marked stenosis of the upper lobe bronchus has resulted from erroneous interpretation of an accessory bronchial orifice as that of the entire upper lobe. In addition, operative demonstration of the segmental anatomy may be confused in the presence of a bronchial abnormality unless this possibility is considered. As a further point of surgical importance, anomalous vessels may be associated with such bronchial variations." SUMMARY AND CONCLUSIONS
While uncommon, anomalies of upper lobe bronchial distribution are of considerable clinical interest. The right side is affected far more frequently than the left, there being only one left-sided abnormality among the 10 cases here reported. The present series includes the first recorded instance of an azygos lobe being supplied by a separate anomalous bronchus. The incidence of upper lobe bronchial anomalies is probably in the range of 0.1 to 0.6 per cent. Since a strict anatomic classification of such variants is not generally adaptable to clinical use, several major types have been defined on a descriptive basis. Bron-
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chography is the most precise and accurate means of establishing the clinical diagnosis but important information may be obtained from bronchoscopy and laminagraphy. Lung parenchyma served by an abnormal bronchus is probably not intrinsically more susceptible to disease. However, by th eir mere presence, such bronchi may assume dia gnosti c and surgical significance.
F ig. G.-Probable bronchial bud from the right side of t h e trachea In a 70-year-old m an.
ADDENDUM
Two additional cases of upper lobe bronchial abnormalities have been encountered sin ce t his paper was accepted f or publication and are here presented f or completeness. In one patient, a 70'y ear-old man with hemoptysis, br onchoscopy showed chronic endo· bro nchit is but no abnormali t y of br onchial origin was det ected. Ho wever, the bro nchogr am (Fig. 6) demonstrated a probable bronchial bud from th e r ight side of th e trachea. It is lik ely t hat t his represen ts persistence into adult life of th e embryoni c out pouching descri bed by Bremer.e Although its conge nital na tu re ca nnot be established conclusively, th e ab sence of additiona l def ects in t he traeheohrone hial tree strongl y sug ges ts t hat it is not an acquir ed div erticul ar lesio n of t he type occasiona lly seen with severe cndobrouchit.is. Th e second an omaly occur red in a 49-year-old woman wit h scle roder ma. L am iuagr a ms of t he chest obt ained at Mas sa chusetts Memorial Hospi tal s mad e on Aug. 29, 1961, showell a b ronchus, sufficiently large to be considered seg mental, arising fro m the left side of th e trnehea well ab ove the carina .
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Permission to report the following cases is gratefully acknowledged: Case 1, Department of Radiology, Children's Hospital Medical Center, Boston; Case 7, Dr. Joseph P. Lynch; and Case 8, Dr. John W. Strieder. REFERENCES
1. Boyden, E. A.: Segmental Anatomy of the Lungs. A study of the Patterns of the Segmental Bronchi and Related Pulmonary Vessels, New York, 1955, McGraw-Hill Book Co., Inc., p. 224. 2. Gerson, C. E., and Rothstein, E.: An Anomalous Tracheal Bronchus to the Right Upper Lobe, Am. Rev. Tuberc. 64: 686-690, 1951. 3. Harris, J. H., J'r.: The Clinical Significance of the Tracheal Bronchus, Am. J. Roentgenol. 79: 228-234, 1958. 4. Schaff, B., and Baum, G.: The Tracheal Bronchus, J. THORACIC SURG. 33: 282-286, 1957. 5. Inada, K., and Kishimoto, S.: An Anomalous Tracheal Bronchus to the Right Upper Lobe. Report of Two Cases, Dis. Chest 31: 109-112, 1957. 6. Bremer, J. L.: Accessory Bronehi in Embryos. Their Occurrence and Probable Fate, Anat. Rec. 54: 361-374, 1932. 7. Foster-Carter, A. F.: Broncho-pulmonary Abnormalities, Brit. J. Tuberc. 40: 111-124, 1946. 8. Boyden, E. A.: The Distribution of Bronchi in Gross Anomalies of the Right Upper Lobe, Particularly Lobes Subdivided by the Azygos Vein and Those Containing Pre-eparterial Bronchi, Radiology 58: 797-807, 1952. 9. Holinger, P. H., and Johnston, K. C.: Clinical Aspects of Congenital Anomalies of the Trachea and Bronchi, Dis. Chest 31: 613-621, 1957. 10. Scheff, S., Kaufman, S. A., and Levene, G.: The Laminagraphic Appearance of Ectopic Right Upper Lobe Bronchi, Radiology 70: 82-83, 1958. 11. Knight, A. C.: Supernumerary Tracheal Lobe, Dis. Chest 37: 696, 1960. 12. Golding, A. M. B.: Supernumerary Tracheal Bronchus, Thorax 15: 174-176,1960. 13. Epstein, T.: Bronchial Adenoma in a Supernumerary Tracheal Lobe. Report of an Unusual Case, J. THORACIC SURG. 21: 362-369, 1951.
L
ONG
CASE REPORTS
CASE I.-On March 11, 1947, this 5-day-old girl was, admitted to the Children's Hospital Medical Center, Boston, because of choking and cyanosis following feedings. A Lipiodol esophagram demonstrated a tracheoesophageal fistula and outlined the tracheobronchial tree. The adventitious bronchogram (Fig. 1) showed the right upper lobe bronchus to originate from the trachea. Operative closure of a high tracheoesophageal fistula was performed on the second hospital day but the patient did poorly and died 5 days after operation. Autopsy showed the right upper lobe bronchus to arise directly from the trachea but there were no gross abnormalities of the pulmonary vessels. CASE 2.-This 65-year-old man was seen at Massachusetts Memorial Hospitals in June, 1953, because routine x-ray studies had demonstrated two rounded densities at the right hilus. He was free of respiratory symptoms. Bronchoscopy showed that the trachea was normal to a point 1.5 em, above the main carina, at which level a trifurcate right upper lobe bronchus was seen with the right-angle lens. The remainder of the endoscopy was unremarkable except for redness and edema of the left upper lobe orifice. Laminagraphy confirmed the diagnosis of abnormal origin of the right upper lobe bronchus from the trachea but showed no tumor. The patient was discharged. CASE 3.-The patient was a 37·year-old woman who was admitted to the Sanatorium Division, Boston City Hospital, because of right upper lobe tuberculosis. Neither bronchoscopy nor laminagraphy showed a bronchial abnormality. After appropriate drug therapy, right upper lobectomy was performed on Jan. 15, 1957, for residual cavitary disease. At operation an anomalous small bronchus was found which arose from the right lateral aspect From the Department of Surgery, Boston University School of Medicine, and the Thoracic Surgery Services of the Boston City Hospital and Massachusetts Memorial Hospitals. Received for publication May 12, 1961. 'Clinical Instructor in Sur-ger-y, Boston University School of Medicine; Associate Visiting Surgeon for Thoracic Surgery, Boston City Hospital; Assistant in Surgery. Massachusetts Memorial Hospitals. '-Clinical Associate in Surgery, Harvard Medical School, and Instructor in Surgery, Boston University School of Medicine; Associate Visiting Surgeon for Thoracic Surgery, Boston City Hospital; Surgeon, Children's Hospital, Boston. - _. Assistant Professor of Radiology, Boston University School of Medicine; Associate Radiologist, Massachusetts Memorial Hospitals.
595
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of the trachea slightly above the main carina. The right upper lobe bronchus was trifurcate and in normal position. The cavity in the resected specim en yielded acid-fast bacilli and was positive for Mycobact erium tuber culosis on culture. CASE 4.-This 51-year-old male alcoholic entered the San atorium Division, Boston City Hospital, in .I uly, 1955, becau se of pulmonary tuberculosis. Bronchoscopy and laminagraphy demonstrated no bronchial abnormality. On Dec. ll, 1956, right upper lobectomy was done becau se of persi sting ca vitary disease. Thoracotom y disclosed that pleural symphysis involved th e entire upp er lob e. Dissection revealed an azygos lobe which was complet ely destroyed and replaced by large cysts and bullae. The apical and posterior segments showed ext ensive fibrotic changes. With considerable difficulty the a zygos lobe was mobilized and passed beneath th e azygo s vein . It was th en found that th er e was a sepa rate artery to the
Fig. I.- Ca se 1.
Esophagram and adventitious bronchogram which shews entire r ight upp er lobe br onchus to or iginate trom the trachea.
azygos lob e from the main pulmonary artery and tha t the venous drainage was als o by way of a distinct vessel. In addition, it was served by a separate bronchus arising f rom the r ight main-stem bronchus proximal to the origin of an apparently trifurcate upper lobe bronchus which occupied the normal position. Th e operati ve specimen showed a destroyed upper lobe with num erous cavities and cysts, smear and cult ure of which were negative for tuberculosis. CASE 5.-0n May 13, 1960, this 43-year-old man und erwent br onchoscopy at the Soldiers ' Home , Chelsea, Mass., because of hemoptysis of und etermined etiology. There was diffuse endobronchitis, more marked on the left. In addition, there was a small bronchial orifice on the right lateral aspect of the trachea at approximately the level of the ca ri na, an d a larger distal upp er lobe orifice originating from the right main- st em bronchus. Unfortunately bronchography was not performed. CASE 6.-This 62-year-old man was an inv et erate smoker who was admitted to the Soldiers' Home, Chelsea, Mass ., for inve stigation of a productive cough. Lingular pn eumonitis had occurred twice during the preceding 12 months. On March 2, 1959, the findings at bronchoscopy were unremarkable except for acute endobronchitis of the basal bronchi on the left sid e. No abnormal bronchus was noted on the right but a right-angle lens was not available. A bilateral bronchogram was then performed and showed normal lingular fiIIing. As an incidental finding, there was a small tracheal bron chus on the right slightly above the level of the main carina. CASE 7.-A 47-year-old man was seen because of rec ent worsening of a chronic product ive cough. A chest roentgenogram was interpreted as showing post-pneumonic atelectasis of
Vol. 43, No. 5 May, 1962
ANOMALIES OF UPPER LOBE BRONCHIAL DISTRIBUTION
597
the left lower lobe. Findings at bronchoscopy on April 10, 1958, at the Carney Hospital, Boston, wer e unremarkabl e except for an a bnorm al right upper lob e bronchial orifi ce just below th e level of t he car ina, with th e orifice to the r emainder of the upper lobe being visualized in the expect ed location. The diagnosis of anom alous r ight upper lobe br onchus was confirmed by a bronchogram done on May 13, 1958, at Quincy Cit y Hospital, Quincy, Mass. (Fig. 2). In addition, there was saccular bronchi ectasis of th e left lower lobe for which res ection was succes sf ull y undertak en. CASE 8.-Brisk hemoptysis prompted investigation of this 56-yea r-old man. A ches t roentgenogram was interpreted as sho wing tuberculosis of the right upper lobe . In an effor t to locali ze the source of the hemorrhage , emergency bronchoscopy was p erformed at N ewton 'Wellesley Hospital, Newton, Mass., on July 28, 1955. Th e profuse ble eding appeared t o
F ig. 2.- Case 7.- Br onchogr am which shows anomalous or igin of a segmental bronchus of th e right upp er lobe ( indicate d by arrow). The position of th e horizontal fissu re wa s clearly seen and is marked on the illustration. originate from a markedly contracted ri g ht upper lob e ori fice. Th e hemop ty sis ceas ed spontaneously and the diagnosis of pulmonary tuberculosis was confirmed by th e results of guinea pig inoculat ion . Appropr iate antimicrobial therapy was instituted. Bronchography was attempted on 2 separate occasions but was unsuccessful because of intractable cough. Laminagrams were performed at Massachusetts Memorial Hospitals on Sept. 22, 1955, to delineate th e extent of the r esidu al disease. In addit ion to persisting fibrocaseous tuberculosis in the right upper lob e, these st udies demonstrated an a nomalous bronchu s t o t his lobe from t he trachea, slightly proximal to th e level of th e main car ina . It th en becam e apparent that t he endoscopic interpretation of marked ste nosis of the right upper lobe bronchial orifi ce had been in er ror, sin ce th e orifice visua liz ed r epresent ed only part of th e upper lob e supply. Ri ght upper lob ectomy was perform ed a t Mas sachusetts Memorial H ospitals on March 14, 1956, f or r esidual fibrocaseous tubercul osis. At oper ation ther e was a separ ate bronchus to t he apical segment which a rose from th e trachea at a pprox ima tely th e level of tile main car ina. Th e bronchus to t he r ema ining upper lob e seg ments was found a t th e norm al site . CASE 9.-This patient was a 79-year-old man who was a dmi t te d to Ma ssachus etts Memorial Hospitals on May 20, 1957, because of dyspn ea and a cough productive of bloody sputum. Nephrectomy had been pe rfor med 4 years 'Previously for carc inoma . X -ra y st udies
598
LAFORET, STARKEY, SCHEFF
J. Thoracic and Cardiovas, Surg.
of the chest showed an infiltrate at the bases of both lungs and prominence of the right hilus. Two therapeutic bronchoscopies were done because of the patient's inability to raise secretions, but no abnormal bronchus was seen on either occasion. Laminagrams showed no metastatic deposits in the lungs, but demonstrated an anomalous right upper lobe bronchus arising from the distal trachea (Fig. 3). The patient died and permission for autopsy was withheld.
Fig. 3.--Case 9. Laminagram demonstrating ectopic segmental right upper lobe bronchus arising above main carina. (From Scheff, Kaufman, and Levene, Radiology 70: 82-83, 1958.) Ar'row indicates the anomalous bronchus. The significant features of the laminagram have been emphasized for improved photographic reproduction. CASE 10.-This 40-year-old woman was admitted to the Sanatorium Division, Boston City Hospital, in November, 1955, because of active tuberculosis involving both upper lobes. On March 1, 1956, laminagrams were secured to evaluate the results of therapy. Bilateral apical cavities were present and, in addition, there was a suggestion of an anomalous bronchus originating from the left main stem. Findings at bronchoscopy on May 23, 1956, were unremarkable and the bronchial anomaly was not detected. However, a bronchogram performed immediately thereafter demonstrated that the apical-posterior segment of the left upper lobe was supplied by an individual bronchus arising proximal to the bronchus serving the remaining upper lobe segments (Fig. 4). On June 7, 1956, resection of the apical-posterior segment of the left upper lobe was accomplished, the procedure being technically facilitated by the independent origin of the segmental bronchus.
Vol. 43, No.5 May, 1962
ANOMALIES OF UPPER LOBE BRONCHIAL DIS'l'RIBUTION
599
In Table I are recorded the salient features of the 10 preceding cases. Case 4, however, warrants special consideration since, as far as can be determined, it is the first recorded instance of an azygos lobe being supplied by an anomalous separate bronchus. According to Boyden,' certain fundamental conceptions are of importance concerning the azygos lobe, namely, "that the lobus is not a supernumerary structure but merely a segregated portion of the right upper lobe, that it is aerated by a variable number of bronchi from one or more segments, and that the descending wedge of the azygos bends certain bronchi, thereby favoring such sequelae as atelectasis, bronchiectasis, and the like." These points are emphasized by Gerson and Rothstein," who state that an anomalous
Fig. 4.-Case 10.
Bronchogram which demonstrates separate origin of apical-posterior segmental bronchus proximal to left upper lobe bronchus.
tracheal bronchus to the right upper lobe" is never related to another anomaly, the azygos lobe, also located in this portion of the chest. The azygos lobe is an anomaly of the azygos vein rather than of the lung and the separated azygos lobe is supplied by the usual bronchial branches. Foster-Carter presents detailed anatomical studies of the bronchial and vascular supply to the azygos lobe, but in none was any bronchial anomaly or tracheal bronchus discovered." Does Case 4 modify or nullify these concepts of the azygos lobe ~ We believe not, since it seems most likely that the association of anomalous bronchus and azygos lobe in this patient was merely coincidental. Indeed, on a statistical basis such an occurrence might even have been predictable. ETIOLOGY
As with most developmental defects the etiology of upper lobe bronchial abnormalities is not known. Har-ris" has summarized present knowledge concerning the evolutionary development, embryology, and comparative anatomy of the lung as related to the occurrence of these anomalies. Phylogenetic features have been emphasized by Schaff and Baum.'
Separate proximal bronchus to apical segment; distal bifurcate bronchus to remaining RUL segments
]I,{
Right
43
5
Normally positioned trifurcate RUL bronchus; proximally, a separate small bronchus from right main stem to azygos lobe
Right
M
51
4
Normally positioned trifurcate RUL bronchus; small tracheal bronchus to apical segment
Right
F
37
3
Trifurcate RUL bronchus arising from trachea
Trifurcate RUL bronchus arising from trachea
M
65
2
Right
X
(X)
(X)
X
-
-
X
-
-
(X)
(X)
-
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-
x
No
Yes
Yes
-
X
No
No
X
-
(X)
DISEASE IN TISSUE SERVED MEANS OF DIAGNOSIS BY ABNORMAL BRONCHOS-/ BRONCHOG-ILAMINAG-IOPERA-I AUCOPY RAPHY RAPHY TION TOPSY BRONCHUS
Routine chest xray reported ' , abnormal, ' , with" 2 rounded hilar densities"
Died 5 davs after operation
REMARKS
Diffuse endobronchitis
Hemoptysis
Right upper 10Fibrocaseous tubectomy berculosis, RUL
Fibrocaseous tuRight upper 10bcrculosis, RUL bectomy
None
Tracheoesophageal fistula
PRIMARY DISEASE
SUMMARY OF PERTINENT DATA IN TEN PATIENTS WITH UPPER LOBE BRONCHIAL ANOMALIES
TYPE OF ANOMALY
1.
Right
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Separate proximal (X)§ (X) bronchus to apical segment; distal bifurcate bronchus to remaining RUL segments
Separate proximal bronchus to apical segment; distal bifurcate bronchus to remaining RUL segments
Separate proximal bronchus to apical segment; distal bifurcate bronchus to remaining RUL segments
Separate proximal bronchus to apical segment; distal bifurcate bronchus to remaining RUL segments
X
-
=
(X)t (X)
X
X
X
X
X
-
X
X
-
-
-
-
-
-
-
Yes
No
Yes
No
No
:j:Two attempts at bronchography unsuccessful because of intractable cough. §Therapeutic bronchoscopies.
Profuse bleeding at time of bronchoscopy; right upper lobectomy
Left lower lobectomy
Productive cough
not per-
Resection of A-P segment LUL
- = Examination
Cavitary tubercnIosis, A-P segment LUL
Died Arteriosclerotic heart disease and metastatic renal carcinoma
Cavitary tubercuIosis, RUL
Bronchiectasis, LLL
Bronchitis, LLL
Legend: X == Examination performed and diagnostic. (X) Examination performed but not diagnostic. formed. The Iamlnagraphtc features of Cases 2, 8, and 9 have been previouslv reported.'. °Lipiodol swallow given to demonstrate tracheoesophageal fistula. tRight-angle lens not available.
M
62
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LAFORET, STARKEY, SCHEFF
602
j. Thoracic and Cardiovas, Surg.
INCIDENCE
Although the 10 cases reported here have been garnered over a period of 2 years from several sources, chronologically they extend over a longer interval. For this and other reasons it is impossible to estimate the total experience from which these examples have been culled. Consequently little can be said about the clinical incidence of such anomalies and even less about their absolute incidence. As is true generally, the number of cases diagnosed bears a direct relation to the prevailing level of awareness and interest. In a review of the Japanese literature, lnada and Kiahimoto" found that the reported incidence of anomalous tracheal bronchus to the right upper lobe ranged from 0.1 to 0.6 per cent. It is of interest that on an embryologic basis one might expect a greater frequency than occurs clinically. Bremer," for example, demonstrated abnormal bronchial buds from the right side of the trachea in 4 of 80 embryos but believed that in most instances these are resorbed, thus accounting for the lower incidence in adults.
Fig. 5.-DiagrammaNo repreeentatum. oj the anomalies o] upper lobe bronohial supply encountered in the present series. A, The usual bronchial distribution. B, Origin of the entire right upper lobe bronchus from the trachea (2 patients). 0, A small tracheal bronchus coexisting with an apparently normal right upper lobe bronchus (l patient). D, Normal origin and distribution of right upper lobe bronchus with a separate small bronchus from the right main-stem bronchus to an azygos lobe (1 patient). E, The most common anomaly, separate origin of the apical segmental bronchus proximal to the common origin of anterior and posterior segmental bronchi (5 patients). F, The only left-sided abnormality encountered in the present series but the commonest variant on this side, separate origin of the apicalposterior segmental bronchus proximal to the origin of the remainder of the superior division (1 patient). CLASSIFICA'l'ION
Upper lobe bronchial anomalies have been the subject of considerable taxonomic study. Anatomically"- 8 a valid distinction has been made between displaced and supernumerary bronchi. If intralobar segmentation is normal but the mode of bronchial origin is not, the abnormal bronchus is termed displaced. A true supernumerary bronchus is much less common and implies that the bronchus serves pulmonary parenchyma that is definitely in excess of the normal.
VoL 43, No.5 May, 1962
ANOMALIES OF UPPER LOBE BRONCHIAL DISTRIBUTION
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This classification necessarily involves detailed anatomic analysis. Thus, for example, the mere demonstration of a trifurcate upper lobe bronchus in conjunction with a tracheal bronchus on this side is not sufficient to permit designation of the latter as supernumerary, unless it can be shown that it supplies lung parenchyma in excess of that normally encountered in the right upper lobe." 8 'When only clinical data are available, efforts to compress upper lobe bronchial anomalies into this anatomic classification may be inaccurate and confusing. As a result, several systems related chiefly to the chinical aspects of right upper lobe bronchial abnormalities have been proposed.v " In order to encompass anomalies of the left upper lobe as well, the following descriptive classification is offered, based on the present material and reports in the literature: Type I.-The upper lobe is anatomically complete but its bronchus arises from the trachea (entire upper lobe bronchus displaced onto trachea). Type Il.-An accessory bronchus arises proximal to an apparently normal upper lobe bronchus, either (A) from the trachea or (B) from the main stem. Type IIl.-A segmental bronchus (usually the apical or apical-posterior) is "split" from the lobar bronchus and arises proximal to it, either ,(A) from the trachea or (B) from the main stem. Fig. 5 presents a diagrammatic representation of the present cases on the basis of this classification. DIAGNOSIS
Bronchography is the most precise and accurate means of establishing the clinical diagnosis of upper lobe bronchial anomalies and, in the present series, was conclusive in all 4 instances in which it was successfully employed. It should be noted, however, that the abnormal bronchus may not be outlined unless careful attention is given to the trachea during the examination. Laminagraphy affords a reliable technique for the demonstration of upper lobe bronchial anomalies"? and was diagnostic on 4 of 6 occasions (cf. Table I). Conventional radiographic technique, however, has not been helpful. Bronchoscopy was performed in all but one case and was diagnostic in only 3. One patient underwent 2 separate endoscopic examinations without detection of the abnormality but the primary indication for the procedures was therapeutic and not diagnostic. In Case 8 there was profuse bleeding at the time of bronchoscopy and the anomalous orifice was not visualized, although the source of bleeding was correctly established as the right upper lobe. In Case 6, a right-angle lens was not available at the time of bronchoscopy and diagnosis was not accomplished. Thus, while precise interpretation of the findings may be difficult, bronchoscopy is an additional diagnostic measure that may be rewarding. Awareness of the possibility of bronchial anomalies and use of the right-angle lens to inspect the trachea as well as the major bronchi should improve chances for diagnosis." Operation disclosed the abnormal bronchus in 4 of 5 patients, In 2 of those, its presence had been unsuspected preoperatively. Since the operation that dill not disclose the abnormality was done for tracheoesophageal fistula, demonstration of the abnormality could not have been expected. The only autopsy in the
604
LAFORET, STARKEY, SCHEFF
J. Thoracic and Cardiovas. Surg.
series confirmed the clinical diagnosis of anomalous right upper lobe bronchus (Case 1). However, routine necropsy technique may be inadequate to demonstrate such anomalies and increased awareness of these variants is as necessary for the pathologist as for the clinician. CLINICAL SIGNIFICANCE
Whether lung parenchyma served by an abnormal bronchus is intrinsically more susceptible to disease is uncertain. According to Harris," "Once identified, the tracheal bronchus has no particular clinical significance." However, others 4 , 12 believe that such bronchi are associated with an unusually high incidence of disease, particularly bronchiectasis, in the area of lung to which they are distributed. In one recorded instance," the anomalous bronchus was the seat of an adenoma which was successfully resected. Much of the difficulty in assessing the clinical significance of these variations stems from indefinite information about their true incidence, since there undoubtedly are individuals with such defects who remain asymptomatic and never come to medical attention. Disease was absent in tissue served by the abnormal bronchus in 6 of the 10 patients comprising the present series. The etiology of the disease in the 4 remaining patients was tuberculosis and not obviously related to the bronchial abnormality. In Case 4, the additional finding of cystic changes in the azygos lobe was almost certainly the result of the abnormal vein rather than the malpositioned bronchus. In cases of the type here presented, then, it seems likely that the bronchial abnormality is principally one of position and is not otherwise associated with altered form or function of either the bronchus or its subtended parenchyma. An exception might exist if the abnormal bronchus is so situated that its drainage is inadequate. Prescinding from the question of intrinsic vulnerability to disease, however, anomalous bronchi by their mere presence may be of considerable clinical importance. In some instances" a bronchoscopic diagnosis of marked stenosis of the upper lobe bronchus has resulted from erroneous interpretation of an accessory bronchial orifice as that of the entire upper lobe. In addition, operative demonstration of the segmental anatomy may be confused in the presence of a bronchial abnormality unless this possibility is considered. As a further point of surgical importance, anomalous vessels may be associated with such bronchial variations." SUMMARY AND CONCLUSIONS
While uncommon, anomalies of upper lobe bronchial distribution are of considerable clinical interest. The right side is affected far more frequently than the left, there being only one left-sided abnormality among the 10 cases here reported. The present series includes the first recorded instance of an azygos lobe being supplied by a separate anomalous bronchus. The incidence of upper lobe bronchial anomalies is probably in the range of 0.1 to 0.6 per cent. Since a strict anatomic classification of such variants is not generally adaptable to clinical use, several major types have been defined on a descriptive basis. Bron-
Vol. 43, No.5 Ma y, 196 2
AN OMALI E S OF UPPER LOBE BRONCHIAL DISTRIBUTION
605
chography is the most precise and accurate means of establishing the clinical diagnosis but important information may be obtained from bronchoscopy and laminagraphy. Lung parenchyma served by an abnormal bronchus is probably not intrinsically more susceptible to disease. However, by th eir mere presence, such bronchi may assume dia gnosti c and surgical significance.
F ig. G.-Probable bronchial bud from the right side of t h e trachea In a 70-year-old m an.
ADDENDUM
Two additional cases of upper lobe bronchial abnormalities have been encountered sin ce t his paper was accepted f or publication and are here presented f or completeness. In one patient, a 70'y ear-old man with hemoptysis, br onchoscopy showed chronic endo· bro nchit is but no abnormali t y of br onchial origin was det ected. Ho wever, the bro nchogr am (Fig. 6) demonstrated a probable bronchial bud from th e r ight side of th e trachea. It is lik ely t hat t his represen ts persistence into adult life of th e embryoni c out pouching descri bed by Bremer.e Although its conge nital na tu re ca nnot be established conclusively, th e ab sence of additiona l def ects in t he traeheohrone hial tree strongl y sug ges ts t hat it is not an acquir ed div erticul ar lesio n of t he type occasiona lly seen with severe cndobrouchit.is. Th e second an omaly occur red in a 49-year-old woman wit h scle roder ma. L am iuagr a ms of t he chest obt ained at Mas sa chusetts Memorial Hospi tal s mad e on Aug. 29, 1961, showell a b ronchus, sufficiently large to be considered seg mental, arising fro m the left side of th e trnehea well ab ove the carina .
fiOfi
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J. Thoracic and Cardiovas, Surg.
Permission to report the following cases is gratefully acknowledged: Case 1, Department of Radiology, Children's Hospital Medical Center, Boston; Case 7, Dr. Joseph P. Lynch; and Case 8, Dr. John W. Strieder. REFERENCES
1. Boyden, E. A.: Segmental Anatomy of the Lungs. A study of the Patterns of the Segmental Bronchi and Related Pulmonary Vessels, New York, 1955, McGraw-Hill Book Co., Inc., p. 224. 2. Gerson, C. E., and Rothstein, E.: An Anomalous Tracheal Bronchus to the Right Upper Lobe, Am. Rev. Tuberc. 64: 686-690, 1951. 3. Harris, J. H., J'r.: The Clinical Significance of the Tracheal Bronchus, Am. J. Roentgenol. 79: 228-234, 1958. 4. Schaff, B., and Baum, G.: The Tracheal Bronchus, J. THORACIC SURG. 33: 282-286, 1957. 5. Inada, K., and Kishimoto, S.: An Anomalous Tracheal Bronchus to the Right Upper Lobe. Report of Two Cases, Dis. Chest 31: 109-112, 1957. 6. Bremer, J. L.: Accessory Bronehi in Embryos. Their Occurrence and Probable Fate, Anat. Rec. 54: 361-374, 1932. 7. Foster-Carter, A. F.: Broncho-pulmonary Abnormalities, Brit. J. Tuberc. 40: 111-124, 1946. 8. Boyden, E. A.: The Distribution of Bronchi in Gross Anomalies of the Right Upper Lobe, Particularly Lobes Subdivided by the Azygos Vein and Those Containing Pre-eparterial Bronchi, Radiology 58: 797-807, 1952. 9. Holinger, P. H., and Johnston, K. C.: Clinical Aspects of Congenital Anomalies of the Trachea and Bronchi, Dis. Chest 31: 613-621, 1957. 10. Scheff, S., Kaufman, S. A., and Levene, G.: The Laminagraphic Appearance of Ectopic Right Upper Lobe Bronchi, Radiology 70: 82-83, 1958. 11. Knight, A. C.: Supernumerary Tracheal Lobe, Dis. Chest 37: 696, 1960. 12. Golding, A. M. B.: Supernumerary Tracheal Bronchus, Thorax 15: 174-176,1960. 13. Epstein, T.: Bronchial Adenoma in a Supernumerary Tracheal Lobe. Report of an Unusual Case, J. THORACIC SURG. 21: 362-369, 1951.