smokers. Seven patients had single nodes and two pa-tients had multiple nodes. Nodules were detected inthree patients by screening chest x-ray, and others weredetected by preoperative or follow-up CT scan for coex-isting lung diseases. The diameters ranged from 3 to13 mm (mean, 6.5 � 3.0 mm). The characteristics of theresected nodules on high-resolution CT scan were (a) theborder was sharp and clear (13 of 13, 100%); (b) the shapewas oval (12 of 13, 92.3%); (c) they were located in thesubpleural region (range, 0 to 13 mm; mean, 4.0 � 4.0 mmfrom visceral pleura), and (d) internal density was highand homogeneous (12 of 13, 92.3%). Five of 13 nodules(38.5%) had some short spicules around them, but nonodules had notches, cavities, or calcifications. Threepatients had transbronchial biopsy or CT-guided needleaspiration biopsy, but these techniques did not preciselydiagnose the nodules as intrapulmonary LNs.

Nodule growth has been documented in two previousreports [3, 4], over 8 months and 4 years, respectively.The shapes were both oval and did not change sodramatically. In the present case, the shape of the in-trapulmonary LNs changed remarkably as they grewfrom a thin stick-like shape to oval in a period as short as3 months. Although the reason for the growth of theintrapulmonary LNs is not known in the present case, wesuspected that the operation was a triggering mechanismfor growth for the following reasons. First, an obstructionof lymphatic ducts by mediastinal lymph node dissectioncan cause congestion of lymph fluid in the peripheralarea, resulting in enlargement of intrapulmonary LNs.Second, postoperative nonspecific inflammatory reactionin the thoracic cavity can extend into lung parenchyma,causing enlargement of intrapulmonary LNs. Interest-ingly, the same thin sticklike shadows in the right upperlobe, which could also be intrapulmonary LNs, did notchange in size and shape, thereby supporting thishypothesis.

Intrapulmonary LNs should be included in the differ-ential diagnosis of small peripheral nodules, becausethey will be encountered more frequently as the preci-sion of diagnostic systems increases. Although somecharacteristics of intrapulmonary LNs can be seen onx-rays, it is still hard to distinguish them from small lungcancers; therefore, we should not hesitate to performthoracoscopic or open biopsy.

References

1. Yokomise H, Mizuno H, Ike O, Wada H, Hitomi S, Itoh H.Importance of intrapulmonary lymph nodes in the differentialdiagnosis of small pulmonary nodular shadows. Chest 1998;113:703–6.

2. Kradin RL, Spirn PW, Mark EJ. Intrapulmonary lymph nodes:clinical, radiologic, and pathologic features. Chest 1985;87:662–7.

3. Houk ZN, Osborne DP. Subvisceral pleural lymph nodepresenting as an expanding intrapulmonary nodule. Am RevRespir Dis 1965;91:596–9.

4. Ehrenstein FI. Pulmonary lymph node presenting as anenlarging coin lesion. Am Rev Respir Dis 1970;101:595–9.

Adenocarcinoma Arising in aRetained Esophageal RemnantOnur Genc, MD, Ronald K. Knight, FRCP, Andrew G.Nicholson, DM, and Peter Goldstraw, FRCS

Departments of Thoracic Surgery and Histopathology, RoyalBrompton Hospital, London and Frimley Park Hospital,Surrey, United Kingdom

Fistulation between the trachea and an infected mucocelearising from an excluded retained esophageal segment isan interesting and unusual complication of esophagealbypass surgery. We present such a case in which therewas also malignant transformation in the cyst wall,manifested by invasive adenocarcinoma and widespreadhigh-grade glandular dysplasia.

(Ann Thorac Surg 2001;72:2117–9© 2001 by The Society of Thoracic Surgeons

Bypass of the diseased esophagus has been under-taken in the treatment of varied esophageal condi-

tions, including benign strictures, perforations and rup-ture, unresectable esophageal cancer, and congenitaltracheoesophageal fistula. Mucoceles may form in theexcluded esophageal segment, but they reportedly re-main small and asymptomatic [1]. We describe a patientwho presented with malignant transformation in an ex-cluded esophageal segment, presenting through fistulationinto the trachea of an enlarged and infected mucocele.

A male patient underwent attempted closure of a con-genital tracheoesophageal fistula during the first year oflife, but because of complications, he eventually requireda colonic conduit and subtotal esophagectomy, withexclusion of a small portion of the distal esophagus. Hesubsequently remained well and asymptomatic until 23years of age, when he presented with recurrent episodesof chest infection, expectorating large quantities of pus. Acomputed tomographic scan showed a 90-mm � 40-mmthick-walled abscess cavity in the mediastinum, extend-ing from the carina to the diaphragm and lying to theright of the vertebral bodies (Fig 1). On bronchoscopy, alarge diverticulum was seen in the posterior wall of thetrachea above the carina, although no fistula was seen atthis time. A diagnosis of an infected mucocele was made.To drain the abscess cavity, a transcutaneous pigtailcatheter was inserted, and the patient’s immediate con-dition subsequently improved.

However, the patient continued to drain smallamounts of pus through the pigtail catheter, and he wastherefore referred for surgical excision of the infectedmucocele in the excluded portion of distal esophagus. Atright thoracotomy, the tracheal diverticulum was foundto communicate with the distal esophageal remnant, witha blind-ending mucocele extending through the hiatus

Accepted for publication March 27, 2001.

Address reprint requests to Mr Goldstraw, Royal Brompton Hospital,Sydney St SW 3 6 NP, London, England; e-mail: pgoldstraw@rbh.nthnames.nhs.uk.

2117Ann Thorac Surg CASE REPORT GENC ET AL2001;72:2117-9 ADENOCARCINOMA IN ESOPHAGEAL REMNANT

© 2001 by The Society of Thoracic Surgeons 0003-4975/01/$20.00Published by Elsevier Science Inc PII S0003-4975(01)02721-7

and adhering to the upper surface of stomach. Theesophageal remnant was mobilized and excised, and thetracheal orifice was repaired with continuous polypro-pylene sutures. Adjacent soft tissues were suturedagainst the fistula repair. The opened sac had a focallyulcerated pale mucosal surface and a diffusely thickenedfibrotic wall. A perforation in the wall, relating to thepigtail catheter, was noted (Fig 2). Microscopy revealedthe majority of the epithelium to be of gastric type and toshow widespread high-grade glandular dysplasia, al-though squamous epithelium without dysplasia waspresent at the fistula margin. In addition, there wereseveral foci of invasive adenocarcinoma, one of whichextended into the muscularis propria. Both invasive anddysplastic components were completely excised (Fig 3).No Helicobacter pylori organisms were seen. The patient’spostoperative course was uneventful, and he remainswell and free of disease at 6 months.

CommentPrimary repair of a congenital tracheoesophageal fistulacan be undertaken in most cases [2], but esophagealreplacement is sometimes required, either followingbreakdown of the primary anastomosis or in patientswith associated “long-gap” esophageal atresia. In such

cases, the esophagus is usually resected and restored bya colon interposition or gastric tube [3, 4]. However, thishas not always been the case, as seen in this report, inthat the distal esophagus had not been resected when thecolonic graft was placed through the left chest to theneck. Experimental studies in dogs have shown that anexcluded esophageal segment may develop into a cystwithin a matter of weeks, with the growth of the cystregulated by its internal pressure [5], and series ofmucoceles arising post–esophageal replacement havebeen reported, in cysts lined by both squamous andglandular epithelium [1, 6]. However, infection within anesophageal mucocele is very rare, with only one previousreport [6]. Furthermore, although premalignant changeshave been described in an esophageal mucocele post-surgery for achalasia [6], development of an invasiveadenocarcinoma is a unique finding. This may reflect the20-year history, with unknown potential carcinogenicfactors in the cyst contributing to the eventual develop-ment of neoplasia. It is also interesting that most esoph-ageal adenocarcinomas arise on a background of meta-plasia secondary to gastroesophageal reflux. In thispatient, it is most unlikely that metaplasia developedwithin the first year of life; therefore, either the columnarepithelium from which this tumor developed was heter-otopic in nature, or, more likely, it arose in associationwith chronic inflammation within the enclosed cyst.

Fig 1. Computed tomographic scan of the thorax shows a thick-walled cystic mass in the right posterior mediastinum; the cyst isfilled with fluidlike material.

Fig 2. Opened esophageal remnant with tracheal fistula. The wall isthickened, and a pigtail catheter is noted in situ.

Fig 3. A section from the cyst wall shows a polypoid area of glan-dular dysplasia with a focus of invasive adenocarcinoma extendingto infiltrate the superficial aspect of the muscularis propria.

2118 CASE REPORT GENC ET AL Ann Thorac SurgADENOCARCINOMA IN ESOPHAGEAL REMNANT 2001;72:2117-9

The presence of an adenocarcinoma may also havecontributed to the patient’s presentation through over-production of mucus leading to increased pressure andfistulation. However, given the history of recent infectionwithin the mucocele, it is likely that a combination ofthese factors led to increased pressure and recanalizationof the fistula. We conclude that, if there is a risk ofmalignant transformation within these remnants, sur-gery should perhaps be considered even if patients areasymptomatic.

References1. Olsen CO, Hopkins RA, Postlethwait RW. Management of an

infected mucocele occurring in a bypassed excluded esopha-geal segment. Ann Thorac Surg 1985;40:73–5.

2. Moores DWO, Campbell DP. Congenital anomalies of theoesophagus. In: Shields TW, ed. General thoracic surgery.Philadelphia: Lippincott, Williams and Wilkins, 1994;118:1529–40.

3. Kelly JP, Shackelford GD, Roper CL. Esophageal replacementwith colon in children: functional results and long-termgrowth. Ann Thorac Surg 1983;36:634–43.

4. Richardson JV, Heintz SE, Rossi NP, et al. Esophageal atresiaand tracheoesophageal fistula. Ann Thorac Surg 1980;29:364–8.

5. Deaton WR, Bradshaw HH. The fate of an isolated segment ofthe oesophagus. J Thorac Surg 1952;23:570–4.

6. Kamath MV, Ellison GR, Rubin WJ, Moore HV, Pai GP.Esophageal mucocele: a complication of blind loop esopha-gus. Ann Thorac Surg 1987;43:263–9.

Double Switch Operation forSuperior-Inferior VentriclesNarutoshi Hibino, MD, Yasuharu Imai, MD,Mitsuru Aoki, MD, Toshiharu Shin’oka, MD, andTakeshi Hiramatsu, MD

Department of Pediatric Cardiovascular Surgery, The HeartInstitute of Japan, Tokyo Women’s Medical College, Tokyo,Japan

Superior-inferior ventricles are a rare cardiac malforma-tion characterized by the two ventricles lying one abovethe other instead of side by side. Consequently, theinterventricular septum that separates such ventricles ishorizontal, and anomalies of the atrioventricular valvesand the ventriculoarterial relations are almost alwayspresent. This complex anomaly is difficult to managewith an operation, so few cases have been reported. Wedescribe a successful experience in which we performeda double switch operation, consisting of the Senning andJatene procedures, for this rare malformation accompa-nied by double-outlet right ventricle {S,L,L}. This is thefirst report we have been able to locate of a double switchoperation for superior-inferior ventricles.

(Ann Thorac Surg 2001;72:2119–21)© 2001 by The Society of Thoracic Surgeons

Superior-inferior ventricles are a rare anomaly charac-terized by a horizontal ventricular septum with large

defect and a hypoplastic right ventricular sinus localizedanterosuperiorly to the left ventricle. The results of thiscomplex lesion have been poor. We report a successfulexperience for the repair of such a malformation usingdouble switch operation.

The patient was a 14-year-old boy who was born withdouble-outlet right ventricle {S,L,L}, superior-inferiorheart, ventricular septal defect (VSD), and coarctation ofthe aorta. At 3 months of age, he underwent subclavianflap and pulmonary artery banding for coarctation of theaorta. At 13 years of age, cardiac catheterization revealeda pulmonary vascular resistance (Rpu) of 3.7 w.u:m2, andtransvenous atrial septostomy was performed. Medica-tion with prostacycline was commenced. At 14 years ofage, repeated cardiac catheterization reproduced theabove diagnosis, as well as tricuspid regurgitation (2 of 4),Rpu of 2.7, flow ratio of pulmonary blood flow/systemicblood flow (Qp/Qs) of 1.2, right ventricular end diastolicvolume of 72% with an ejection fraction of 55%, and leftventricular end diastolic volume of 113% with an ejectionfraction of 59% (Fig 1). Because tricuspid regurgitationwas worsening and Rpu was slightly high, we decided toperform a double switch operation consisting of theSenning and Jatene procedures, despite moderately lowright-ventricular volume.

A median sternotomy was performed under generalanesthesia. After dissection of the great arteries anddivision of the previously ligated ductus, pump perfusionwas established through the ascending aortic and bicavalcannulation. The main pulmonary artery was divided atthe banded site. During cold fibrillation, the right atriumwas opened and the Senning procedure was performed.The tricuspid valve showed prolapse of the anteriorleaflet, and a Reed’s annuloplasty was performed. Aftercardiac arrest was induced, the aorta was divided and

Accepted for publication Feb 3, 2001.

Address reprint requests to Dr Hibino, Department of Pediatric Cardio-vascular Surgery, The Heart Institute of Japan, Tokyo Women’s MedicalCollege, 8-1 Kawada-cho Shinjuku-ku, Tokyo, 162-8666 Japan.

Fig 1. Preoperative angiograms (lateral projection). (A) Injectioninto the right ventricle (RV). (B) Injection into the left ventricle(LV). These figures show both great arteries (Ao � aorta; PA �pulmonary artery) and the right ventricle through the ventricularseptal defect (arrow) in the horizontal septum.

2119Ann Thorac Surg CASE REPORT HIBINO ET AL2001;72:2119-21 DOUBLE SWITCH OPERATION

© 2001 by The Society of Thoracic Surgeons 0003-4975/01/$20.00Published by Elsevier Science Inc PII S0003-4975(01)02651-0