Colobronchial fistula due to Crohn\'s disease

446

CASEREPORT KARMY-J()N[!SEI AI COLOBRONCHIAL FISTUI.A

the course of Legionella p n e u m o n i a , and pericardial involvement usually is found incidentally [1-8] (Table 1). It is interesting to note that most patients with Legionella pericarditis have been y o u n g e r than those previously thought to be susceptible to Legionella p n e u m o n i a . As o p p o s e d to a m e a n age of 60 years seen in patients with Legionnaires' pneumonitis, the m e a n age of those with Legionella pericarditis was 35 years. Furthermore, patients presenting with acute pericarditis also were healthier than those typically afflicted with Legionnaires' disease, with 6 of 9 patients r e p o r t e d in the literature having no prior health problems. The diagnosis of Legionella infections can be difficult. Direct fluorescent antibody staining has b e e n successful in making the diagnosis of pericardial fluid in prior reports [8]. Direct fluorescent antibody staining on nonpericardial tissue has a sensitivi~, of 25% to 80% a n d a specificity of 96% to 99% in biopsy specimens obtained from sources other than pericardial tissue. Direct fluorescent antibody staining on pericardial tissue may i m p r o v e the diagnosis of this elusive organism, especially when other testing is negative or if the diagnosis has been m a s k e d by prior antibiotic treatment. According to our review of the literature, complications related to Legionella pericarditis include relapsing infection, recurrent pericardial effusion [5, 6] (20%), and late constrictive pericarditis [2, 6] (20%). Ve D, few patients were treated early in the course of infection because of difficulties in diagnosis. Three of the patients reviewed h a d treatment initiated within 5 days of admission and recovered completely without complications. This underscores the importance of early diagnosis of acute pericarditis not only to abort a potentially debilitating infection but also to prevent constrictive pericarditis, which can result from misdiagnosis. The role of video-assisted thoracoscopic pericardiectomy in this setting a p p e a r s promising, particularly in obtaining sufficient tissue specimens for pathologic and bacteriologic examination. Further experience with this technique in this setting is needed. In conclusion, Legionella pericarditis should be considered in the differential diagnosis of acute pericarditis, especially when there is associated pneumonia. Unlike Legionella p n e u m o n i a , young and relatively healthy subjects may be more p r e d i s p o s e d to pericardial involvement. Pericardial fibrosis and constrictive pericarditis, as well as recurrent infection, are c o m m o n and serious sequelae that occur in almost one third of patients with Legionella pericarditis. Early accurate diagnosis and appropriate t r e a t m e n t seem essential to prevent late complications. Direct fluorescent antibody staining of pericardial tissue m a y prove to be valuable in establishing the diagnosis of Legionella infection, especially when the diagnosis remains unclear.

References

1. Harris LF. Legionnaires' disease associated with massive pericardial effusion [Letter]. Arch Intern Med 1981;141:1385. 2. Landes BW, Pogson W, Beauchamp GD, Skillman RK, Brewer © 1995 by The Society of Thoracic Surgeons

Ann Thorac Surg 1995;60:446-8

3. 4. 5. 6. 7. 8.

JH. Pericarditis in a patient with Legionnaires' disease. Arch Intern Med 1982;142:1234-5. Maycock R, Skale B, Kohler RB. Legionella pneumophila pericarditis proved by culture of pericardial fluid. Am J Med 1983;75:534-6. Reyes RR, Noble RC. Legionnaires' pericarditis. J Ky Med Assoc 1983:757-8. Friedland L, Snydman DR, Weingarden AS, Hedges TR, Brown R, Busky M. Ocular and pericardial involvement in Legionnaires' disease. Am J Med 1984;77:1105-7. Nelson DP, Rensimer ER, Raffin TA. Legionella pneumophila pericarditis without pneumonia. Arch Intern Med 1985;145: 926. Svendsen JH, Jonsson V, Niebuhr U. Combined pericarditis and pneumonia caused by Legionella infection. Br Heart J 1987;58:663- 4. Luck PC, Helbig JH, Wunderlich E, et al. Isolation of Legionella pneumophila serogroup 3 from pericardial fluid in a case of pericarditis. Infection 1989;17:388-9.

C o l o b r o n c h i a l Fistula D u e to Crohn's D i s e a s e Riyad Karmy-Jones, MD, Anees Chagpar, BMedSci, Eric Vallieres, MD, and Stuart Hamilton, MD Department of Surgery, University of Alberta, Edmonton, Alberta, Canada Enteropulmonary fistulas may present as recurrent localized pneumonia. A case of one such fistula originating from the colon in a patient with Crohn's disease is discussed.

(Ann Thorac Surg 1995;60:446-8) ecurrent localized p n e u m o n i a , in the absence of endobronchial obstruction or other p r e d i s p o s i n g p u l m o n a r y lesions, should p r o m p t a w o r k u p for a gastrointestinal (GI) cause, including aspiration or fistula. The majority of fistulas originate from the intrathoracic esophagus, but rarely they m a y have a s u b d i a p h r a g m a t i c origin. In this report we describe a rare complication of Crohn's disease, a colobronchial fistula that p r e s e n t e d as recurrent left lower lobe p n e u m o n i a .

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A 33-year-old man p r e s e n t e d with mild h e m o p t y s i s and a 4-month history, of p u r u l e n t s p u t u m production characterized by a feculent odor, left-sided chest pain, and generalized malaise. Thirteen months earlier he had sustained a fall in which he suffered multiple left rib fractures with p u l m o n a r y contusion. His past history was significant only for an episode, 6 years previously, of a b d o m i n a l pain, diarrhea, and cramps, which was diagnosed as giardiasis. A b a r i u m e n e m a at that time was normal. Accepted for publication Jan 11, 1995. Address reprint requests to Dr Karmy-Jones, Division of Trauma and Critical Care, Henry Ford Hospital, 2799 W Grand Blvd, Detroit, Ml 48202.

0003-4975/95/$9.50 0003-4975(95)00207-2

Ann Thorac Surg 1995;60:446-8

Fig 1. Computed tomographic scan of chest demonstrating left lower lobe consolidation and eventration. Arrow points to air-containing density, which subsequently was demonstrated to represent the fistulous tract.

Sputum cultures grew mixed flora. Laboratory work was normal save for mild microcytic anemia. Examination revealed evidence of consolidation at the left base. A chest roentgenogram confirmed left lower lobe consolidation. Computed tomography demonstrated left lower lobe pneumonic changes, eventration of the posterior d i a p h r a g m , an ill-defined i n f r a d i a p h r a g m a t i c aircontaining density posteriorly, and splenomegaly (Fig 1). Magnetic resonance and fluoroscopy suggested that the diaphragm was intact but paralyzed. Bronchoscopy revealed only signs of chronic inflammation. Because of the question of traumatic diaphragmatic injury, a barium

Fig 2. Barium study demonstrating splenic flexure fistula rising above diaphragm (arrow).

CASE REPORT KARMY-JONESET AL COLOBRONCHIALFISTULA

447

enema study was obtained, which demonstrated fistula arising from the areas of the splenic flexure and in the general region of the sigmoid arising to the diaphragm (Fig 2). The patient underwent thoracoabdominal exploration, at which time a complex fistulous network originating from the left colon and involving the distal small bowel and the spleen was found. The diaphragm was penetrated by a 1-cm extension of the tract, and the adjacent segments of the lower lobe appeared chronically inflamed. A pulmonary wedge resection was performed and the tract was closed. A splenectomy, left hemicolectomy, and small bowel resection also were performed. Pathologic examination revealed transmural inflammation of the small and large bowel consistent with Crohn's disease. The spleen showed evidence of chronic inflammation, and the lung parenchyma demonstrated chronic inflammation and extensive interstitial fibrosis. The patient had an uneventful recovery. At 1-year follow-up he has had no recurrent pulmonary or GI symptoms and his anemia has resolved. His diaphragm remains paralyzed.

Comment Fistulous connection between the GI and respiratory tracts result in recurrent localized pneumonia, characterized by foul sputum production, the presence of mixed gastrointestinal flora, and lung necrosis. The esophagus is the most c o m m o n site of origin, and the cause may be congenital or acquired, the latter as a result of malignant, granulomatous, trauma, or diverticular diseases [1]. Subdiaphragmatic origins are very unusual. The most common, bronchobiliary fistula, is characterized by jaundice, cholangitis, and bile-stained sputum. Such fistulas may be due to congenital communications, obstruction of the biliary tree, or inflammatory, infectious, or traumatic hepatic processes [2]. Initially, in our case, the possibility of posttraumatic diaphragmatic hernia with GI/pulmonary fistulous connection was entertained. Although the initial diagnosis of this injury may be overlooked in up to approximately 50% of cases, leading to a significant risk of visceral incarceration, the symptoms, signs, and radiographic findings tend to be those of pulmonary compression, effusion, and, in the case of visceral infarction, pleural sepsis rather than recurrent pneumonia [3, 4]. With Crohn's disease fistulas are usually enteroenteric or enterocutaneous. To our knowledge, only 1 other report documents colobronchial fistulization in this setting [5]. There are a n u m b e r of important similarities between the 2 cases, including the presence of microcytic anemia, recurrent left lower lobe pneumonia with feculent sputum in the absence of an intrapulmonary cause, absence of GI symptomatology at the time of presentation but a history suggesting the possibility of GI disease, diagnosis confirmed by barium enema, left colon involvement, and enteroenteric fistulization. Presumably the left lung was at greater risk because of the higher riding splenic flexure and the absence of interposed liver. In our case, the

448

CASEREPORT SILENET AL VATS FOR BOCHDALEKHERNIA

absence of diaphragmatic m o v e m e n t may have promoted upward tracking of the fistulizing process. In conclusion, persistent p n e u m o n i a without a discernable i n t r a p u l m o n a r y cause should prompt a GI workup. In the presence of left lower lobe p n e u m o n i a , especially if there has been a history of i n t r a a b d o m i n a l inflammatory disease or trauma, a colon workup should be considered.

References 1. Trastek VF. Esophageal diverticula. In: Shields TW, ed. General thoracic surgery. 4th ed. Baltimore: Williams & Wilkins, 1994:1580-1. 2. Gugenhaim J, Ciardullo M, Traynor O, Bismuth H. Bronchobiliary fistulas in adults. Ann Surg 1988;207:90-4. 3. Saber WL, Moore EE, Hopeman AR, Aragon WE. Delayed presentation of traumatic diaphragmatic hernia. J E M 1986; 4:1-7. 4. Symbas PN, Vlasis SA, Hatcher C Jr. Blunt and penetrating diaphragmatic injuries with or without herniation of organs into the chest. Ann Thorac Surg 1986;42:158-62. 5. Domej W, Kullnig P, Petritsch W, et al. Colobronchial fistula: a rare complication of Crohn's colitis. Am Rev Respir Dis 1990;142:1225-7.

Video-Assisted Thoracic Surgical Repair of a Foramen of Bochdalek Hernia Mark L. Silen, MD, David A. Canvasser, MD, Arlet G. Kurkchubasche, MD, Charles H. Andrus, MD, and Keith S. Naunheim, MD Department of Surgery, St. Louis University Health Sciences Center, St. Louis, Missouri A case report of a congenital posterolateral diaphragmatic hernia in an adolescent is presented and a technique for thoracoscopic repair of Bochdalek hernia is described. Postoperative discomfort was minimal and the hospital stay was less than 24 hours. Video-assisted thoracic surgery may be the technique of choice for repair of certain congenital diaphragmatic hernias w h e n identified after infancy.

(Ann Thorac Surg 1995;60:448-50) ideo-assisted thoracic surgery (VATS) provides a u n i q u e set of opportunities and challenges to the thoracic surgeon. The safe and successful m a n a g e m e n t of the presenting clinical problem must not be compromised by the use of this technique, yet the avoidance of the morbidity of a formal thoracotomy must be taken into consideration. Expanding indications for a n d applica-

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Accepted for publicationJan 20, 1995. Address reprint requests to Dr Silen, Departmentof Surgery, St. Louis University Health Sciences Center, 1465 South Grand Blvd, St. Louis, MO 63104. © 1995 by The Society of Thoracic Surgeons

Ann Thorac Surg 1995;60:448-50

tions of VATS test the j u d g m e n t and the creativity of the surgeon. The successful m a n a g e m e n t of a congenital diaphragmatic hernia in an adolescent using thoracoscopic surgery serves to illustrate these points. A 16-year-old boy presented with a 1-year history of episodic, cramping, left flank pain. Each episode lasted between 30 a n d 60 minutes. The pain was not associated with meals or with any specific activity a n d he denied concomitant nausea or vomiting as well as any respiratory or urinary symptoms. The past medical history was notable for an episode of left u p p e r q u a d r a n t pain at age 7 years. His parents were told that a chest radiograph at that time demonstrated a "hole in his diaphragm," but no therapy was advised. Before referral, a chest radiograph a n d a b a r i u m enema were performed a n d interpreted as normal. Physical examination at presentation was unremarkable, as were abdominal a n d chest radiographs taken at this time. The chest roentgenogram from age 7 years was unavailable. A computed tomographic scan of the chest was ordered, a return a p p o i n t m e n t was made, and the patient was instructed to return if his symptoms recurred. O n e week later, the patient experienced an episode of acute pain and chest radiographs at that time demonstrated an air-filled soft tissue mass above the left h e m i d i a p h r a g m (Fig 1A). After resolution of symptoms after 30 minutes, a repeat chest roentgenogram appeared normal (Fig 1B). Further workup included an upper gastrointestinal series a n d a chest c o m p u t e d tomographic scan, both of which were read as normal. At operation, single-lung ventilation was instituted and a thoracoscope was placed through a 2-cm left fifth anterior axillary intercostal incision. A 4-cm long posterolateral diaphragmatic defect was identified with o m e n t u m herniating through the opening. No hernia sac was present. A second trocar site was placed in the posterior axillary line of the fifth intercostal space a n d an additional port was placed in the n i n t h intercostal space immediately overlying the site of the defect. A 30-degree rigid telescope was placed through the posterior axillary trocar and i n s t r u m e n t s were placed through the other two sites. Adhesions between the o m e n t u m a n d the rim of the defect were lysed, and the o m e n t u m was returned to the abdominal cavity. To facilitate the repair, the lateral n i n t h interspace trocar site was enlarged to 4 cm in length, the latissimus dorsi muscle was split in the direction of its fibers, a n d the intercostal muscles were divided for a distance of 3 cm. The ribs were not spread. This allowed introduction of standard i n s t r u m e n t s to close the diaphragmatic hernia. Repair of the hernia was accomplished with seven interrupted n o n a b s o r b a b l e sutures. A single 28F catheter was placed through the anterior axillary trocar site before closure of the r e m a i n i n g incisions. The patient took a regular diet the evening of operation, and analgesia was m a n a g e d with oral medications. The chest tube was removed and the patient was discharged from the hospital on the m o r n i n g after operation. 0003-4975/95/$9.50 0003-4975(95)00100-Y