Two cases of pulmonary intralobar sequestration are presented, with aortography and selective arteriography. These examinations are prerequisites for the diagnosis and management of a patient with suspected sequestration. There should be a high degree of suspicion in a case of a persistent left lower-lobe tumour.S. Afr. Med. J., 48, 171 (1974)

Gerst, P.H.

Miller, H.L.

Puljic, S.

Suarez, F.

English

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2 February 1974 S.A. MEDICAL JOURNAL 171of suicide, however, is more common in the elderly, yetsuicide is the cause of death ranking third in youth, andin South Africa it appears to be increasing, particularlyin the Indian teenage group.Depressive patients frequently drink in order to alleviatetheir symptoms, but become subsequently more depressed.'Depression is common in the unemployed, the separatedand the divorced, as is a high rate of alcohol consumption.A large percentage of drivers who consume excessivequantities of alcohol fall into one or both categories andan inference can be drawn that driving under the influenceand related accidents may be psychologically motivated andare metaphors for sef-destruction or even suicidal actions.'"Wyckert" quotes Tabachnick et al. who studied 3 groupsof hospitalised patients, car accident driver victims, po-tential suicides and appendectomy patients, and whostated 'The results did not bear out the accident-suicidehypothesis but significantly the accident driver victimsresembled those who attempted suicide only in one areaof behaviour, that of drinking!' Although these findingsevoke strong criticism from certain psychoanalytically-orientated investigators, a number of other writers, inclu-ding Wold," stated: 'Most interviewers felt that self-destructive components are to be found among accidentsubjects', postulating that under certain circumstancesunderlying characteristics may present themselves. Further-more, it is widely accepted that a person. subsequent toa sudden and severe loss of a loved object. may experienceself-destructive tendencies.(To be continued)Pulmonary Intralobar SequestrationA REVIEW OF THE LITERATURE WITH TWO CASE REPORTSH. L. MILLER, S. PULJIC, P. H. GERST, F. SUAREZSUMMARYTwo cases of pulmonary intralobar sequestrationare presented, with aortography and selective arterio-graphy. These examinations are prerequisites for the diag-nosis and management of a patient with suspectedsequestration. There should be a high degree of suspicionin a case of a persistent left lower-lobe tumour.S. Afr. Med. J., 48, 171 (1974).Department of Radiology, Bronx-Lebanon Hospital Center,BroDx, New York, USAH. L. MILLER, M.D.S. PULJIC, M.D.Department of Surgery, Bronx-Lebanon Hospital Center,Bronx, New York, USAP. H. GERST, M.D.F. SUAREZ, M.D.Date received: I August 1973.6Pulmonary sequestration is a well-recognised developmen-tal anomaly, in which a portion of the lung in itsprimitive form has become separated from the rest ofthe lung.' It receives its blood supply from the aorta viaan abnormal aberrant vessel.There are two types of sequestration-an extra- and anintralobar variety. In the extra lobar type, ectopic lungtissue is situated below or above the diaphragm and isenveloped in its own pleura, with venous drainage intothe systemic circulation, that is, the inferior or superiorvena cava, the azygos, or portal veinY The intralobartype differs in that the venous drainage is invariably intothe pulmonary veins' and is without its own pleuralcovering.The first report of pulmonary sequestration by Rokit-ansky and Rektorzik appeared in 1861;·6 but it was notuntil after the Harris-Lewis publication in 1940' andthe classical description by Price et al.'" that this entitybecame better known.172 S.-A. MEDIESE TYDSKRIF 2 Februarie 1974More than 250 cases of intralobar sequestration havebeen published.' However, failure to recognise this con-dition may result in the inadvertent incision of the ano-malous arterial supply, with subsequent exsanguination.Fortunately, surgeons keep sequestration in mind, butaccording to Bruwer et al.,1JJ 3% of the patients undergoingoperation have died because of a failure to recognise theentity.Findlay and Maier" mention that surgeons should palpatethe pulmonary ligament as a routine before any pulmonaryresection, as anomalous vessels frequently arise in thisstructure, even without sequestration. In view of thesurgical implication, a diagnosis of intralobar sequestrationshould be considered in any persistent inflammatory pro-cess in a lower lobe (usually the left), especially in ayoung patient.CASE REPORTSCase 1A 12-year-old girl was admitted to hospital with recur-rent and persistent infiltration in the right lower lobe. Twomonths before this admission, the patient had been hospi-talised in another institution for recurrent right lower-lobepneumonitis.The chest X-ray examination, including tomograms,showed a density in the posterior basal segment of theright lower lobe (Fig. 1), and a subsequent bronchogramrevealed no filling of this region with contrastmaterial. Bronchoscopy failed to show the posterior basalsegment bronchus, and in view of the above findings,Fig. 1. Case 1. Infiltration in .the posterior basal segmentof the right lower lobe.sequestration was considered. She was treated for pneu-monia and discharged in excellent condition.On a second admission, aortography and a selectivearteriogram established the diagnosis by demonstratingan aberrant vessel from the aorta (Fig. 2), supplying theFig. 2. Case 1. Aortography and selective sequesteredarteriogram demonstrates an aberrant vessel from the;lOrt" supplying the sequestered portion of the lung.sequestered portion of the lung with venous drainagevia the pulmonary vein. Resection of the diseased segmentwas undertaken, and the aberrant artery and pulmonaryvein were divided. A specimen showed no bronchi, butan ovoid, cystic tumour in the centre of the specimen, con-taining homogeneous, gelatinous, pale yellow material,was demonstrated. The patient was discharged withoutcomplications. .Case 2A 15-year-old girl was admitted to hospital with a historyof left lower chest pain, coughing, high fever, and chills,of 2 days' duration. The history indicated 4 previoussimilar episodes over several years. The chest radiographshowed an infiltrate in the left lower lobe (Fig. 3), whichwas treated with antibiotics, but 2 weeks later no significantchanges in the X-ray film were demonstrated. However,an air-fluid level in the same area was noted. On tomo-graphy, air-fluid levels with a lobulated tumour were dis-tinctly shown. Pulmonary sequestration was thereforeconsidered.2 February 1974 S.A. MEDICAL JOURNAL 173Fig. 3. Case 2. Postero-anterior chest film demonstratingan infiltration in the left lower lobe.Subsequent aortography and a selective arteriogramshowed a large aberrant vessel arising at the level ofTlO and TII (Fig. 4), from the abdominal aorta whichsupplied the lobulated left lower lobe tumour. Venousdrainage was by a pulmonary vein draining into the leftatrium. Another smaller vein drained downward andmedially into the hemiazygous system (Fig. 5).Fig 4. Case 2. Aortogram and selective sequestered arterio-gram showing a large aberrant vessel arising at the levelof TIO and TU from the abdominal aorta and supply-ing the left lower lobe tumour.Fig. S. Case 2. Late film of arteriogram demonstratingthe small vein draining into the hemiazygous system.Bronchography demonstrated marked diminution in thenumber of bronchioles, and very few tertiary and smallerbronchioles which showed no connection with the cysticstructure. Normal bronchi were stretched around thisarea.At operation, a large aberrant artery arising from theaorta, and a smaller vessel from the pulmonary artery,were encountered, and noted to be supplying the seques-tered region. The arteries were clamped and ligated.Segmental branches arising from the left lower lobe,and veins, were identified, ligated, and the sequesteredsegment resected. A specimen showed a well-circumscribedmultilocular structure filled with pale yellow, gelatinousmaterial. Bronchi of the normal lung were stretchedaround the cystic structure.DISCUSSIONThe symptomatology of pulmonary intralobar sequestra-tion is non-specific. It includes cough, fever, and chestpain. Infrequently patients are asymptomatic. Clinicallyit may be confused with pneumonia, bronchiectasis, em-pyema, or bronchitis. Treatment is usually by antibiotics,but only temporary relief is achieved, with recurrence ofsymptoms being quite characteristic. Definitive therapy isresection of the segment. The sexes are equaliy involved.More than 50% of patients are below the age of 20 years,"with the posterior segment of the left lower lobe beinginvolved in two-thirds of all reported cases, and theposterior segment of the right lower lobe being next infrequency." The upper lobes are rarely involved.The chest X-ray film usually shows a tumour suggestingbronchiwctasis, and when infection supervenes, infiltrationwith air-fluid levels may be demonstrated (case 2). Tomo-graphy of this area may show the aberrant arterial supply."The bronchogram fails to show filling of the bronchi inthe region of the tumour, but an arcuate displacement ofthe bronchi around this area.'• The latter differs from the174 S.-A. MEDIESE TYDSKRIF 2 Februarie 1974findings in bronchiectasis, where contrast-filled dilatedbronchi are readily seen. Zelefsky et al.'· reported on 6l:ases in the literature (2 of which were their own), where,on bronchography, the contrast medium entered the cys-tic area, suggesting cystic bronchiectasis, and in view ofthis the importance of arteriography was stressed inmaking a diagnosis. Arteriography confirms the diagnosisby showing an aberrant vessel which usually arises fromthe lower thoracic or abdominal aorta and is usuallysingle. Turk and Lindskog," in their review of the litera-ture consisting of 114 surgically-proved cases, reported theaberrant vessel to be arising from the thoracic aorta in74 cases; the abdominal aorta in 25; the intercostalarteries in 5; and to be of multiple origin in 17 cases.Selective studies of the aberrant vessel have severaladvantages; in that the venous drainage may be morereadily demonstrated, and therefore helpful in the diagnosisof intra- or extralobar sequestration; and an adequateextent of the region supplied by the abnormal vessel maybe shown, so helping to assess the amount of resectionto be undertaken, as this depends greatly on the amountof aberrant vessel supply.In their report, Turk and Lindskog" noted 6 cases ashaving venous drainage to the right side of the heart viathe inferior vena cava, instead of the much more usualdrainage to the pulmonary veins. These veins were shownto be smaller than the supplying artery, suggesting thatonly a portion of the venous blood returns to the rightheart and that some must enter the pulmonary circulation.Venous drainage to the portal system is rarely encounteredand only one such case has been reported."The second case reported in this article is ratheratypical, as the venous drainage passed into the hemiazy-gous vein (systemic circulation) and a small vein drainedinto the pulmonary vein (pulmunary circulation). Only oneother such case appears in the literature!Gross pathology of intralobar sequestration has beenclassified by Price' as follows: (a) a large cyst in thesequestered region in 50% of the cases; (b) a polycystictumour in 40% of cases; and (c) a sequestered tumour con-taining bronchi which branch parallel to the artery.The cystic or bronchial elements are usually completelydissociated from the normal bronchial tree, with theanomalous vessel lying in the centre of the sequesteredtumour. The vessel has a thick wall and is elastic, similarto the pulmonary artery,IQ and is frequently arteriosclerotic.The cysts are lined with respiratory epithelium, andusually filled with thick, brown, mucoid material.Different theories on the origin and embryologicalsignificance of the sequestration have been presented byvarious authors. The anomalous vessel is believed to bea persistent branch from the dorsal or ventral aorta, tothe splanchnic plexus, although the exact manner of itspersistence has not as yet been satisfactorily established. 14,11As the lung bud develops ventrally from the primitiveforegut, one of its developing branches apparently be-comes detached and acquires a systemic arterial bloodsupply from the persistent branch of the splanchnicplexus." A relatively complete survey of the varioustheories of the aetiology was presented by Nielsen' andGallagher."Find1ay and Maier" found that when the origin of ananomalous pulmonary vessel was from the systemic sys-tem above the level of the lung roots, there were associatedanomalies of the heart and great vessels in 75% of thecases. If the origin was below this level, 16% of casesshowed such abnormalities.CONCLUSIONIt is obvious that aortography and selective arteriographyare absolute prerequisites for the diagnosis and propermanagement of a patient with suspected sequestration.A high degree of suspicion should be present in cases ofa left, lower-lobe, persistent tumour. This should be anindication for angiography.REFERENCES1. Brower, A. J. (1950): J. Thorac. Surg., 19, 957.2. Nielsen, P. B. (1964): Amer. J. Roentgeno!.. 92. 547.3. Ranninger, K. and Valvassor, G. E. (1964): Ibid., 92, 540.4. Wyman, S. M. and Eyler, W. R. (1952): Radiology, 59, 658.5. Rektorzik, E. (1861): Wbl. Z. K.K. Ges. Artze Wien, p. 17.6. Rokitansky, C. (1861): Lehrbuch der Parhologischen Ana/omie.vol. 3, p. 44. Vienna: Braumiiller & Seidel.7. Hams, H. A. and Lewis, I. (1940): J. Thorac. Surg., 9, 667.8. Price, D. M., Sellors, T. J. and Blair, L. G. (1947): Brit. J. Surg.,35, 18.9. Price, D. M. (1946): J. Path. Bact., 58, 457.10. Brower, A. J., Clagett, O. T. and McDonald, J. R. (1950): J.Thorac. Surg., 19, 957.11. Findlay, C. W. jun. and Maier, H. C. (1951): Surgery, 29, 604.12. Turk, L. N. and Lindskog, G. E. (1961): J. Thorac. Cardiovasc.Surg., 41, 299.13. Krishnan M. and Snelling, M. R. t. (1970): J. Surg., 39, 362.14. Zelefsky, M. N., Janis, M., Bernstein, R., Blatt, C., Lin, A. andMeng. C. H. (1971): Chest, 59, 266.15. Shufford, W. H. and Sybers, R. G. (1969): Amer. J. Roentgenol.,lOll, 118.16. Davies, D. V. and Gunz, F. W. (1944): J. Path. Bact., 56, 417.17. Smith, R. A. (1956): Thorax, 11, ID.18. Gallagher, P. G. (1957): New Engl. J. Med., 257, 643.

Pulmonary intralobar sequestration

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Pulmonary intralobar sequestration

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