British Journal of Radiology (2006) 79, 170-172
© 2006 British Institute of Radiology
doi: 10.1259/bjr/27258284
Colobronchial fistula: a late complication of childhood radiotherapy
G C MacKay, MRCP,
J Howells, MRCP, FRCR and
F W Poon, FRCR
Department of Radiology, Glasgow Royal Infirmary, Queen Elizabeth Building, 16 Alexandra Parade, Glasgow G31 2ER, UK
Correspondence: Dr Gillian MacKay, 7 Rosevale Road, Bearsden, Glasgow G61 2RX, Scotland, UK.
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Abstract
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We present the case of a colobronchial fistula in a 41-year-old man who underwent radiotherapy for nephroblastoma as an infant. He attended for barium enema, which demonstrated a fistula between colon and bronchial tree. Following right hemicolectomy and pathological examination of the resected bowel, no active disease process was identified to explain the development of this rare fistula. Radiotherapy was deemed the most probable aetiology. We are unaware of this having been previously described.
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Introduction
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Colobronchial fistulae are rare, and have previously been reported in adults secondary to Crohn's disease [1], colonic malignancy [2], tuberculosis [3] and as complications of gastrointestinal surgery [4–6]. We report the relevant radiological and clinical findings in a case of colobronchial fistula as a likely result of radiotherapy 40 years previously.
To the best of our knowledge, this aetiology has not previously been described.
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Case report
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A 41-year-old man was referred to the surgical out-patient department for investigation of a left sided discharging perianal sinus. Colonoscopy had been normal. 99Tcm-hexamethylpropyleneamineoxime (HMPAO) labelled white cell isotope scan demonstrated increased tracer uptake in the subhepatic space and to the right of the lumbar spine (Figure 1
), the significance of which was initially unclear. Subsequent examination under anaesthetic (EUA) and endoanal ultrasound diagnosed a complex extrasphincteric fistula. A barium enema was arranged to try to demonstrate any fistulous connection which may have been missed by previous colonoscopy. His relevant past medical history included a right nephrectomy and subsequent intensive radiotherapy for nephroblastoma at the age of 1 year.
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Figure 1. 99Tcm hexamethylpropyleneamineoxime (HMPAO) labelled isotope scan demonstrating high uptake in the right paravertebral region (illustrated by arrow).
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On presentation for barium enema, the patient complained of a 6 week history of general malaise, weight loss, right upper quadrant pain, dyspnoea and cough with associated malodorous "chocolate-coloured" sputum.
Barium enema demonstrated a tract of extraluminal barium arising from the proximal transverse colon which extended superiorly towards the right subphrenic space (Figure 2). The examination was immediately terminated, although the patient remained haemodynamically stable with no signs of peritonism. Appearances were consistent with a localized colonic perforation, for which an iatrogenic cause was not thought likely. The leak appeared confined, with no evidence of free intraperitoneal air nor generalized barium contamination of the peritoneal cavity.
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Figure 2. Fistulous tract of barium arising from proximal transverse colon(arrows delineate the extent of the tract). Marked deformity of the vertebral bodies is noted consistent with previous radiotherapy induced damage.
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Contrast-enhanced CT of abdomen and pelvis performed on the same day confirmed a broad tract of barium extending superiorly from the transverse colon, passing behind the liver to enter the right hemithorax. It communicated directly with a thick walled cavity within right posterior hemithorax, measuring 7.5 cm x 4.5 cm, containing air and barium. Barium was also seen to enter adjacent bronchi (Figure 3). Overall, appearances were consistent with an established colobronchial fistula. Marked enlargement of the azygous and hemi-azygous venous systems was noted, together with a reduction in calibre of the inferior vena cava. Deformities of the upper thoracic vertebrae were also present.
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Figure 3. Contrast enhanced CT chest demonstrating a large thick-walled cavity, containing barium and air, within the lower lobe of the right lung. Barium is seen within the right lower lobe bronchial tree.
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The patient was admitted to the general surgical ward on the day of presentation, and underwent extended right hemicolectomy with ileo-transverse anastomosis. The thoracic cavity was not examined or drained by the cardiothoracic surgeons, who were present at the time of surgery. Dense adhesions in the right hypochondrium, between colon and liver, and duodenum and colon were identified. The operative findings were thought to have been radiotherapy related. Pathology of the right hemicolon revealed fibrous adhesions and serosal fibrosis with no evidence of active inflammation or malignancy.
The patient endured a stormy post-operative course, but made a slow recovery. He has been found to be unfit for thoracotomy, and his pulmonary sepsis has therefore been managed conservatively.
A subsequent barium follow-through examination identified a hitherto clinically silent duodenal stenosis (Figure 4). In the absence of other significant history, this series of findings were felt to be consistent with late sequelae of wide-field, high-dose radiotherapy.
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Figure 4. Barium follow-through examination showing a significant duodenal stenosis(illustrated by arrow).
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It is worth noting that the presenting complaint of the ischiorectal fistula was unrelated to the eventual diagnosis.
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Discussion
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Nephroblastoma (Wilms' tumour) is the most common cancer of the urinary tract in children. In the pre-chemotherapy era, post-operative radiotherapy was shown to increase patient survival. Wide-field radiotherapy was administered to the side of the abdomen on which the tumour occurred, concurring with the findings in this patient.
With regard to the patient's symptoms on presentation, it now seems likely that the brown, malodorous sputum he described was actually faeculent. The organisms grown from the endotracheal tube secretions were Coliform bacilli, consistent with this conclusion.
In retrospect, and with the benefit of further imaging, it is likely that the area of high white cell uptake adjacent to the right lumbar area represented inflammation at the site of the fistulous track within the abdomen.
The patient has had a normal colonoscopy, negative Gram-staining of sputum and blood cultures, and non-specific pathology of the right hemicolon (in particular, there is a lack of features to suggest Crohn's disease). The most likely aetiological conclusion has therefore been reached by a process of elimination.
Colobronchial fistula is an uncommon finding. It has been described in relation to Crohn's disease, in which fistula formation is a classical manifestation, as a late complication of appendicitis, and following laparoscopic biliary surgery. Colonic malignancy and tuberculosis are other documented causes. However, we can find no reference to a fistula of this nature ever having been described secondary to abdominal radiotherapy. Not only is the extent of this fistula highly unusual, but the length of time taken for symptoms to develop is exceptional.
Although it is extremely rare, fistula formation many years following radiotherapy has been previously described in a patient who developed an enterocutaneous fistula 27 years after radiotherapy for carcinoma of the penis [7]. Given that the oncological treatment of this patient took place in the early 1960s, the field of radiotherapy would have been right-sided but wide, with larger fractions being administered in comparison with today's treatment doses. Fistulae more localized to the site of disease and area of treatment are known to be potential direct complications of radiotherapy and have been well-documented. However, one would expect the onset of related symptoms to occur within a relatively short timescale of treatment.
In treating this condition, surgical resection of the colon is usual, with subsequent lung resection. Unfortunately, the patient has not, as yet, been deemed fit enough to undergo lobectomy, and so treatment has been suboptimal. Long term clinical outcome is therefore unclear.
Received for publication February 15, 2005.
Revision received May 9, 2005.
Accepted for publication May 11, 2005.
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References
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Abstract
Introduction
