Ann Thorac Surg 2003;76:2085-2087
© 2003 The Society of Thoracic Surgeons
Case report
Bronchial adenoma: an unusual cause of recurrent pneumonia in childhood
Francesco Morini, MDa,
Serena Quattrucci, MDb,
Denis A. Cozzi, MDa,
Giancarlo Tancredi, MDb,
Anna Maria Cicconi, MDc,
Roberto Guidi, MDb,
Fabio Midulla, MDb*
a Department of Pediatric Surgery, Rome, Italy
b Department of Cystic Fibrosis, Rome, Italy
c Department of Thoracic Surgery Policlinico Umberto I, University of Rome "La Sapienza", Rome, Italy
Accepted for publication April 28, 2003.
* Address reprint requests to Dr Midulla, Cystic Fibrosis Service, University of Rome "La Sapienza," 324 Vle Regina Elena, 00161 Rome, Italy
e-mail: midulla{at}uniroma1.it
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Abstract
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Primary lung tumors are rare in childhood and often overlooked owing to the nonspecific presentation. We report the case of a 15-year-old boy with a 3-year history of recurrent pneumonia always involving the right lower lobe due to bronchial mucinous adenoma. After endoscopic removal the tumor recurred locally, necessitating open surgical resection of the tumor. In a child with localized recurrent pneumonia of uncertain pathogenesis, the differential diagnosis should include a primary lung tumor. Bronchial adenomas should not be removed endoscopically; patients must undergo open thoracotomy for surgical excision.
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Introduction
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Recurrent pneumonia, defined as two or more episodes of pneumonia in 1 year or three or more total episodes of pneumonia with resolution of radiographic changes between episodes, accounts for about 8% of all admissions for pneumonia in children [1]. Owing to the variety of possible pathogenetic mechanisms, pediatric pulmonologists find this condition challenging to diagnose and treat. Recurrent pneumonia may be secondary to congenital or acquired disorders, and in some cases the predisposing factor remains unidentified [1]. Congenital causes of recurrent pneumonia include malformations of the respiratory tract and cardiovascular system, primary ciliary dyskinesia, cystic fibrosis, and primary immunodeficiencies. Acquired predisposing disorders include acquired immunodeficiencies, abnormal bronchial clearance secondary to infections, foreign body aspiration, and endobronchial tumors. We report the rare case of a 15-year-old boy with recurrent pneumonia secondary to an adenoma of the right bronchus intermedius.
A 15-year-old boy was referred to our department with a 3-year history of recurrent cough, fever, right lower lobe consolidation, and a clinical diagnosis of Swyer-James disease. Despite antibiotic treatment, the lung consolidation after the last episode of pneumonia failed to resolve. The boy had no history of other pulmonary diseases and between the episodes was in good health.
On admission to our department the patient was afebrile, the respiratory rate was 30 breaths/min, and the heart rate 144 beats/min. Peripheral oxygen saturation was 98% in room air. Chest examination revealed a hypomobile right hemithorax, dullness, and reduced breath sounds over the right lower hemithorax. The remainder of the clinical examination was normal. Laboratory tests revealed mild leukocytosis (white blood cells: 9400/µL) with neutrophilia (84%) and an increased erythrocyte sedimentation rate (59 mm/hour). Swab cultures, tuberculin skin test, and sweat tests were negative. A chest roentgenogram illustrated right lower lobe consolidation.
Because the radiographic abnormalities persisted, the child underwent flexible bronchoscopy under sedation and local anesthesia. This examination revealed a fleshy tumor originating in the bifurcation between the right upper and intermedius bronchi, completely obstructing the bronchi in the middle and lower lobes. The histologic study of endoscopic biopsies diagnosed a mucinous adenoma of the bronchus. Therefore, the child underwent rigid bronchoscopy under general anesthesia and the tumor was resected using a Nd:YAG laser.
Two months later, a follow-up computed tomographic chest scan suggested tumor recurrence (Fig 1),
a finding confirmed by a repeat flexible bronchoscopy. The boy underwent surgery with a sleeve resection of the right upper and intermedius bronchi (Fig 2)
and mediastinal lymph node sampling. The final histologic study of the endobronchial lesion confirmed a mucinous bronchial adenoma with clear resection margins. No metastases were evident in the lymph glands.
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Fig 1. (a) Thoracic computed tomographic scan obtained 2 months after endoscopic removal of the tumor, revealing recurrent tumor in the right intermedius bronchus (arrowheads). No lymph node involvement was seen. (b) Higher magnification demonstrates the recurrent tumor does not extend beyond the bronchial wall (arrow).
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Fig 2. Surgical specimen after sleeve resection of the right upper and intermedius bronchus showing the resected bronchial adenoma (arrowheads).
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Four months after surgery, a repeat flexible bronchoscopy under local anesthesia excluded recurrent disease and characterized a patent bronchial anastomosis.
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Comment
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The case of a 15-year-old boy with localized recurrent pneumonia secondary to a right intermedius bronchus adenoma we report here illustrates three points of importance in these patients' clinical management.
In our patient, referred to us with a 3-year history of recurrent pneumonia and a suspected diagnosis of Swyer-Johnson syndrome, the bronchial tumor was eventually diagnosed only after the child underwent fiberoptic bronchoscopy. Flexible bronchoscopy, considered an invasive test and often used only as a last resort in children, is now a widely used technique in pediatric pulmonology. If experienced physicians in specialized centers do the examination children tolerate it well. Flexible bronchoscopy provides specific information not available with other imaging techniques. It directly visualizes the bronchial lumen and allows biopsy of endobronchial lesions. In their review, Curtis and colleagues [2] suggest that any child who presents with persistent unexplained consolidation, which fails to resolve within 2 weeks of vigorous physiotherapy and antibiotics, should be referred for early bronchoscopy.
In this report we present that the diagnostic workup failed to consider bronchoscopy as a diagnostic tool probably because bronchial tumors were not envisaged in the differential diagnosis of recurrent pneumonia. Identifying the cause of recurrent pneumonia necessitates a specific systematic diagnostic approach including clinical history taking, physical examination, and diagnostic tests. Medical history taking should focus on birth, feeding, physical development, genetic components, and atopy. Additional information should be sought about age at onset and duration of the symptoms, the site of pulmonary disease, and possible trigger factors. Diagnostic imaging and laboratory tests should include immunologic tests, sweat test, lung function testing and chest roentgenograms. In a large series of children with recurrent pneumonia [1], the most frequent underlying illness was aspiration pneumonia secondary to oropharyngeal incoordination, followed by immune disorders, congenital heart disease, asthma, congenital respiratory tract anomalies, gastroesophageal reflux, and sickle cell anemia. Further causes of recurrent pneumonia include foreign body aspiration, tuberculosis and other granulomatous infections, and endobronchial tumors, as in our patient.
A benign bronchial tumor is an unusual finding in a 15-year-old patient. Primary pulmonary neoplasms are rare in childhood, with an approximate ratio to non neoplastic lesions of the lungs of 1:60 [3]. Although rare, most primary pulmonary neoplasms are malignant. In a study investigating a series of 230 primary lung neoplasms, Hartman and Sochat [4] found that 151 (65.6%) were malignant. A subsequent review reported similar results in a series of 383 primary pulmonary neoplasms, in which 76% were malignant [5]. The most common primary pulmonary neoplasms are the so-called bronchial adenomas, a heterogeneous group comprising carcinoid, mucoepidermoid carcinoma, adenoid cystic carcinoma (cylindroma), and mucinous adenomas. Mucinous adenomas are exceedingly rare tumors, considered benign on the basis of their clinical course and immunohistochemical markers [6]. Yet, as our case underlines, even if apparently completely removed, they have the potential for local recurrence. The other histologic types of bronchial adenoma are considered malignant because they recur locally or metastasize to distant site [4–6]. In our patient, because the biopsy revealed a benign lesion, we attempted endoscopic removal. But the tumor recurred. Although bronchoscopy is the diagnostic procedure of choice in endobronchial lesions, their iceberglike appearance makes endoscopic resection inadvisable. Because endoscopy does not guarantee radical excision, the tumor may recur locally. Some investigators suggest that bronchoscopic resection should be reserved only for small lesions that are histologically benign [5]. For all other lesions, the preferred treatment is open thoracotomy for surgical removal with mediastinal lymph node sampling. Nevertheless because all neoplastic lesions that are incompletely resected can recur locally, even for benign lesions we advise open thoracotomy. In the past, many patients underwent pneumonectomy. The current trend favors a more conservative approach to preserve ventilatory function. Patients with no mediastinal lymph node involvement can undergo organ-preserving operative procedures (sleeve resection and circumferential resection), provided that care is taken to ensure histologically negative margins [7]. In our patient the open thoracotomy procedure had a successful outcome and no evident signs of local tumor recurrence 18-months after surgery.
In conclusion, the differential diagnosis of recurrent pneumonia in childhood or adolescence should include endobronchial tumors. These lesions can be diagnosed best by flexible bronchoscopy that will directly visualize the lesion and allow its biopsy. Because endoscopic resection of a benign bronchial adenoma does not assure a sufficiently radical resection, the preferred treatment is open surgery.
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References
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- Owayed A.F., Campbell D.M., Wang E.E.L. Underlying causes of recurrent pneumonia in children. Arch Pediatr Adolesc Med 2000;154:190-194.[Abstract/Free Full Text]
- Curtis J.M., Lacey D., Smyth R., Carty H. Endobronchial tumours in childhood. Eur J Radiol 1998;29:11-20.[Medline]
- Cohen M.C., Kaschula R.O.C. Primary pulmonary tumors in childhood: a review of 31 years' experience and the literature. Pediatr Pulmonol 1992;140:222-232.
- Hartman G.E., Shochat S.J. Primary pulmonary neoplasms of childhood: a review. Ann Thorac Surg 1983;36:108-119.[Abstract]
- Hancock B.J., Di Lorenzo M., Youssef S., Yazbeck S., Marcotte J.-E., Collin P.-P. Childhood primary pulmonary neoplasms. J Pediatr Surg 1993;28:1133-1136.[Medline]
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- Lowe J.E., Bridgman A.H., Sabiston D.C., Jr The role of bronchoplastic procedures in the surgical management of benign and malignant pulmonary lesions. J Thorac Cardiovasc Surg 1982;83:227-234.[Abstract]
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Abstract
Introduction
