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• W2472454676 abstract "Case ReportSubdiaphragmatic Extralobar Pulmonary Sequestration with Granulomatous Necrotizing Reaction Simulating a Tuberculous Infection: A Case Report Bashar Abou-Touk, MD, Facharzt Atilio Baez-Giangreco, MD, FCAP Mohammed Afzal, MD, FCAP Kumar Vel, and MSMCR Al Sowailem AbdulmohsenMD, Facharzt Bashar Abou-Touk Search for more papers by this author , Atilio Baez-Giangreco Search for more papers by this author , Mohammed Afzal Search for more papers by this author , Kumar Vel Search for more papers by this author , and Al Sowailem Abdulmohsen Search for more papers by this author Published Online:1 Nov 1995https://doi.org/10.5144/0256-4947.1995.648SectionsPDF ToolsAdd to favoritesDownload citationTrack citations ShareShare onFacebookTwitterLinked InRedditEmail AboutIntroductionA one-year-old boy presented with a left retroperitoneal subdiaphragmatic mass accidentally discovered during admission for bronchopneumonia. The clinical impression was neuroblastoma. Histologically, the mass revealed pulmonary sequestration with cystic adenomatoid pattern associated with severe granulomatous epithelioid necrotizing inflammation simulating tuberculous infection.Pulmonary sequestration represents 1.1% to 6.8% of lung malformations.1 It consists of a cystic or solid mass of nonfunctioning lung tissue. Such pulmonary tissue does not communicate with the tracheobronchial tree and in most cases retains its systemic arterial supply.Sequestration may occur within the normal lung tissue (intralobar) or outside the normal lung (extralobar). The anterior portion of the endodermis or foregut is considered to be the origin of the esophagus and the lungs. If the accessory lung bud arises early in embryonic life before development of the pleura, an intralobar sequestration (ILS) will be formed; if the accessory bud arises after the pleura has developed, an extralobar sequestration (ELS) will be the result.2–4Twelve to 25% of the pulmonary sequestrations are extralobar1,4 and can be located above or below the diaphragm. ELS are found in 90% of the cases above the diaphragm and 10% are subdiaphragmatic. ELS are associated in 40% to 60% of the cases with other congenital malformations.1,6,7Concurrent malformation affecting the lungs and the gastrointestinal system prompted Gerle and colleagues2 to propose a unifying etiologic concept to this abnormal development of the foregut with the term of congenital bronchopulmonary foregut malformation2,6 to include the accessory lung tissue, whether intralobar or extraolobar in location, with or without communication with the esophagus or stomach.Subdiaphragmatic extralobar pulmonary sequestrations (SEPS) are uncommon. They can be located in the retroperitoneum or intraperitoneally. Most of the ELPS are asymptomatic and discovered accidentally during investigation of other clinical symptoms or during repair of congenital anomalies.8 Only 13 cases of retroperitoneal location have been previously reported in the literature,3,4,7–14 but to our knowledge, none with bronchocentric granulomatosis mimicking tuberculous granulomatous reaction as in the present case.Case ReportA one-year-old Yemeni male was transferred to the Maternity and Children’s Hospital in Riyadh because of an incidental finding of a left suprarenal mass during ultrasound examination for evaluation of the unresponsiveness of his bronchopneumonia and left pleural effusion in spite of one month of intravenous antibiotic administration. Bacteriologic cultures from an aspirated xanthochromic pleural fluid were negative.On admission, the child was febrile. A computed tomography (CT) scan showed a cystic mass in the left suprarenal area associated with left pleural effusion (Figure 1). With a possible diagnosis of neuroblastoma, a 24-hour urine VMA was done and proved to be negative. Past history was unremarkable. No vaccination was administered after delivery.FIGURE 1 CT scan showing a cystic mass in the left suprarenal area.Download FigureThe mass was approached through the left 11th intercostal space without opening the pleura. A solid cystic mass measuring 8 × 6 × 3 cm was found. It was adherent to the diaphragmatic peritoneum and adrenal gland. The mass was partially cystic with solid areas showing caseous-appearing granular material. With a presumptive diagnosis of tuberculosis, cultures for routine and acid-fast bacilli were done and liver biopsy was taken. The diaphragm was intact and the abdominal and pelvic organs were normal. The patient was treated with antituberculous therapy that was interrupted when the pathology report and bacteriologic cultures were reported to be negative. The child recovered uneventfully and the left bronchopneumonia and pleural effusion were resolved.HistopathologySections from the mass revealed adenomatous bronchopulmonary malformation associated with bronchocentric palisading granulomatous reaction. Characteristically, the lesion showed numerous microcystic cavities separated by fibrous connective tissue with inflammatory reaction. Some of the microcysts were lined by a cylindrical ciliated bronchiolar epithelium (Figure 2). No cartilaginous or alveolar parenchyma was identified. The lesion was surrounded by dense fibrous tissue with large blood vessels suggestive of pleura. Some of the microcysts were filled with mucinous material, others with macrophages and polymorphonuclear (PMN) cells. Most of the microcysts were ulcerated and lined by epithelioid palisading histiocytic granulomatous reaction in areas associated with extensive necrosis (Figure 3). No Langerhan’s or multinucleated giant cells were observed. Elastic fibers were mildly increased around the preserved dilated bronchiolar spaces. No elastic blood vessels were seen, although one showed atherosclerotic changes. Special stains for fungi and acid-fast microorganisms were negative. The liver biopsy was normal. No inflammatory reactions or granulomata were present.FIGURE 2 Microphotograph showing cystic spaces lined by ciliated bronchiolar epithelium. Hematoxylin and eosin 200×.Download FigureFIGURE 3 Microphotograph showing granulomatous changes with central necrosis. Hematoxylin and eosin 200×.Download FigureDiscussionSubdiaphragmatic extralobar pulmonary sequestrations are uncommon. They can be located in the retroperitoneum or intraperitoneally. They are most frequently observed in the left side although two cases have been reported in the right side.4,12 In his review of the literature from 1862 to 1975, Savic4 observed 12 cases of EPS, only two in the retroperitoneum. Since that time, another 11 cases have been described2,7–14 to our knowledge. Most of the cases have been described in adults. Four showed histologic changes of bronchogenic cysts.11 Very few cases of retroperitoneal location have been described in children. They were accidentally found when a roentgenogram of the chest was taken for another reason or during evaluation of co-existing congenital anomalies.7,8,16 One case has been discovered on maternal ultrasound at 23 weeks’ gestation.8In the case reported here, the mass was subdiaphragmatic and adherent to the left adrenal gland and diaphragm. No diaphragmatic hernia or communication with the gastrointestinal tract was observed. Histologically, a necrotizing palisading granulomatous reaction intermixing with the dysplastic lung tissue with a pattern similar to the one observed in cystic adenomatous malformation of the lung was present. Lung with cystic adenomatous malformation type II associated with ELPS16 has been reported previously, and in two cases the dysplastic histologic changes were found in the EPS.8,15 This is not surprising, since both entities are part of the same embryologic malformation,6 one inside the lung, another in sequestrated lung tissue.In review of the literature of 130 cases of ELPS, Savic observed six cases associated with tuberculous changes in the pulmonary parenchyma, but neither the age nor the fact that the microorganisms were isolated had been reported.Congenital tuberculosis is thought to be acquired transplacentally through the umbilical vein with subsequent primary liver complex and secondary hematogenous spread, or through aspiration in utero or during delivery, resulting in primary disease of either the lung or gastrointestinal tract.17During the surgical procedure, this possibility was considered. However, a complete workup of the patient and the mother failed to reveal any clinical, bacteriological or histological presence of tuberculosis or other infection. The tuberculin skin test was negative. The patient did not receive BCG vaccination. The ELPS did not communicate with the gastrointestinal tract or with the lung. Furthermore, the liver biopsy obtained during the surgical procedure was essentially normal. One of Beitzke’s19 criteria for the diagnosis of congenital tuberculosis is the demonstration of a primary complex in the liver, indicating passage of the organism through the umbilical vein.To our knowledge, this gross and histologic pattern of adenomatous cystic lung with necrotizing epithelioid granulomata mimicking tuberculosis has not been reported in the literature of ELPS. Necrotic granulomatous reaction is not only observed in tuberculosis, fungal infection, rheumatoid arthritits, bronchocentric granulomatosus, but also in necrotic tumor.2,3,6 In this particular case, the granulomatous reaction is probably an inflammatory response to the mucus and necrotic material in the sequestrated lung spaces after destruction of the lining epithelium as observed in bronchocentric granulomatosis.We report this case of ELPS because of its unusual presentation, the clinical confusion with a possible adrenal gland tumor, and the histologic picture of necrotizing palisading granulomatous reaction mimicking tuberculosis.ARTICLE REFERENCES:1. Carter R. Pulmonary sequestration . Ann Thorac Surg. 1969; 7:68-88. Google Scholar2. Gerle RD, Jartzki A, Ashley CA, Berne AS. Congenital bronchopulmonary foregut malformation . N Engl J Med. 1968; 278:1413-9. Google Scholar3. Katzenstein ALA, Askin FB. Surgical pathology of nonneoplastic lung disease. Pediatric Disorders. I. Congenital Malformations, n the series . Major Problems In Pathology. Philadelphia: W.B. Saunders Co; 1982; 13:314-32. Google Scholar4. Savic B, Birtel FJ, Tholen W, et al. Lung sequestration: report of seven cases and review of 540 published cases . Thorax. 1979; 34:96-101. Google Scholar5. de Pareded CG, Pierce WS, Johnson DG, et al. Pulmonary sequestration in infants and children: a 20-year experience and review of the literature . J Pediatr Surg. 1970; 5:136-46. Google Scholar6. Hruba RH, Shumway SJ, Orel SB, Dunler JS, Baker R, Hutchins GM. Congenital bronchopulmonary foregut malformation . Am J Clin Path. 1989; 91:403-9. Google Scholar7. Buntain WK, Wolley MM, Mahour GH, et al. Pulmonary sequestration in children: twenty-five years’ experience . Surg. 1977; 81:413-20. Google Scholar8. Lager DJ, Kupper KA, Haake GK. Subdiaphragmatic extralobar sequestration . Arch Path Lab Med. 1991; 115:536-8. Google Scholar9. Baker EL, Gore RM, Moss AA. Retroperitoneal pulmonary sequestration: computed tomographic findings . Am J Roentgenol. 1982; 136:965-77. Google Scholar10. Coselli MP, De Ipolyi P, Blose PS, et al. Bronchogenic cyst above and below the diaphragm. Report of eight cases . Ann Thorac Surg. 1987; 44:491-4. Google Scholar11. Foerster HM, Sengupta EE, Montag AG, et al. Retroperitoneal bronchogenic cyst presenting as an adrenal mass . Arch Path Lab Med. 1991; 115:1057-9. Google Scholar12. McClelland RR, Kapsner AL, Ueker HH. Pulmonary sequestration associated with a gastric duplicated cyst . Radiol. 1977; 124:13-4. Google Scholar13. O’Mara CS, Baeker RR, Jeyasingham K. Pulmonary sequestration . Surg Gynecol Obstet. 1978; 147:609-16. Google Scholar14. Rogers BM, Harnab PK, Johnson AM. Bronchopulmonary foregut malformation . Ann Surg. 1986; 203:517-24. Google Scholar15. Stocker JT, Kagan-Hallet K. Extralobar pulmonary sequestration. Analysis of 15 cases . Am J Clin Pathol. 1979; 72:917-25. Google Scholar16. Miller RF, Graub M, Pashuick ET. Bronchogenic cystic anomalies resulting from maldevelopment of the primitive foregut and midgut . Am J Roentgenol. 1953; 70:771-85. Google Scholar17. Sallworth JT, Brasfield DM, Tiller RE. Congenital miliary tuberculosis proved by open lung biopsy specimen and successfully treated . Am J Dis Child. 1980; 134:320-1. Google Scholar18. Hageman J, Shulman S, Schreiber M, et al. Congenital tuberculosis: critical reappraisal for clinical findings and diagnostic procedures . Pediatr. 1980; 66:980-4. Google Scholar19. Beitzke H. Uber die Angioborene Tuberkulose Infektion . Ergebn Tuberk Forsch. 1935; 7:1. Google Scholar20. Kolin A, Hiruki T. Palisading granulomas associated with lung cancer . Arch Pathol Lab Med. 1990; 114:667-99. Google Scholar Previous article Next article FiguresReferencesRelatedDetails Volume 15, Issue 6November-December 1995 Metrics History Accepted14 March 1995Published online1 November 1995 InformationCopyright © 1995, Annals of Saudi MedicineThis work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License.PDF download" @default.
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