Lobectomy in cystic fibrosis - CORE · 2017-01-06 · this patient experienced recurrent pneumonia,...

3
quinolones appear to be a promising agent on the basis of in liit~o results. Anaissie et al. reported that imipenem, ceftazi- dime and ciprofloxacin are effective against P. putida (6). It has also been reported that meropenem shows a good to moderate activity against P. putida (9). Stockley et al. reported that nebulized amoxicillin is effective to control chronic purulent bronchiectasis (10). As this patient experienced recurrent pneumonia, nebulization of antibiotics may be useful to prevent pneumonia. The case described here emphasizes that a mucoid strain of P. putida can be isolated from human clinical sources, and may cause pulmonary infection in an immunocompetent host. References 1. Gudbjerg CE. Roentgenologic diagnosis of brochi- ectasjs. An analysis of 112 cases. Acta Radio1 1955; 43: 209-225. 2. Gilardi CL. Pseudomonas. In: Lennette EH, Balows A, Hausler WJ, Shadomy HJ, eds. Manual of Clinical ,Wicrobiology. Washington: American Society for Microbiology 1985; 350-372. 3. Bullock GL. The identification and separation of Aeromonas liquefaciens from Pseudomonasjuorescens CASE REPORTS 695 and related organisms occurring fish. Microbial 1961; 9: 587-590. 4. Khabbaz RF, Arnow PM, Highsmith AK. Pseudo- monas j?uoyscens bacteremia from blood transfusion. Am J Med 1984; 76: 62-68. 5. Tabor E, Gerety RJ. Five cases of Pseudomonas sepsis transmitted by blood transfusions (letter). Lancet 1984; 1: 1403. 6. Anaissie E, Fainstein V, Miller P et al. Pseudomonas putida. Newly recognized pathogen in patients with cancer. Anz J Med 1987; 82: 1191-I 194. 7. Nicotra MB, Rivera M; Dale AM: Shepherd R, Carter R. Clinical, pathophysiologic, and microbiologic char- acterization of bronchiectasis in an aging cohort. Chest 1995; 108: 955-1061. 8. Oberhofer TR. Use of the API 2OE, OxiiFerm, and Minitek systems to identify nonfermentative and oxidase-positive fermentative bacteria: seven years of experience. Diagn Microbial IIzfect Dis 1983; 1: 241- 256. 9. Kropec A, Lemmen S, Wursthorm M, Daschner FD. Combination effect of meropenem with aminoglyco- sides and teicoplanin on Pseudomonas and entevococci. Infection 1994; 22: 306-308. 10. Stockley RA, Hill SL, Burnett D. Nebulized antibiotics in chronic purulent bronchiectasis. Clin Therapeut 1985: 7: 593-599. Lobectomy in cystic fibrosis R. BRAGONIER+, D. GRIER+, F. CARSWELL”, AND S. CUNNINGHAM* Departments of “Paediatrics, ‘Paediatvic Radiology and *Cavdiothovacic Suvgevy, Bristol Royal _Elospifal fey Sick Children, Bristol, U.K. Introduction Case 1 In cystic fibrosis, (CF) the lungs are usually affected dif- fusely, but in some patients, focal disease may predominate (1). Aggressive medical therapy with intravenous antibiotics and chest physiotherapy is the first-line treatment, but in cases which fail to respond, surgical resection of the affected lobe has been advocated (2). We reviewed the clinical courses, up to 15 months post-operatively, of all three of our patients in whom this procedure had been undertaken. All patients had received prolonged intensive medical treat- ment without significant improvement in their clinical or radiological status prior to surgery. Received 2 May 1996 and accepted in revised form 8 January 1997. A lo-year-old boy had persistent right upper lobe bron- chiectasis. His mean lung function tests (o/ predicted for height) preceding lobectomy, derived from all lung function results recorded over the year, were FEV, 44% (SD 3.7%) and FVC 61% (SD 9.0%), and he required seven hospital admissions. During the post-operative year, his mean lung function tests improved, with FEV, 48% (SD 6.4%) and FVC 68% (SD 9.6%): and he was admitted five times for treatment. One year post- operatively, his weight centile position was unchanged but his height centile position had improved; although he remained pre-pubertal. His chest X-ray Crispin Norman (CN) score had fallen from 28, pre-operatively, to 8 (Plate 1).

Transcript of Lobectomy in cystic fibrosis - CORE · 2017-01-06 · this patient experienced recurrent pneumonia,...

Page 1: Lobectomy in cystic fibrosis - CORE · 2017-01-06 · this patient experienced recurrent pneumonia, nebulization of antibiotics may be useful to prevent pneumonia. The case described

quinolones appear to be a promising agent on the basis of in liit~o results. Anaissie et al. reported that imipenem, ceftazi- dime and ciprofloxacin are effective against P. putida (6). It has also been reported that meropenem shows a good to moderate activity against P. putida (9).

Stockley et al. reported that nebulized amoxicillin is effective to control chronic purulent bronchiectasis (10). As this patient experienced recurrent pneumonia, nebulization of antibiotics may be useful to prevent pneumonia.

The case described here emphasizes that a mucoid strain of P. putida can be isolated from human clinical sources, and may cause pulmonary infection in an immunocompetent host.

References

1. Gudbjerg CE. Roentgenologic diagnosis of brochi- ectasjs. An analysis of 112 cases. Acta Radio1 1955; 43: 209-225.

2. Gilardi CL. Pseudomonas. In: Lennette EH, Balows A, Hausler WJ, Shadomy HJ, eds. Manual of Clinical ,Wicrobiology. Washington: American Society for Microbiology 1985; 350-372.

3. Bullock GL. The identification and separation of Aeromonas liquefaciens from Pseudomonasjuorescens

CASE REPORTS 695

and related organisms occurring fish. Microbial 1961; 9: 587-590.

4. Khabbaz RF, Arnow PM, Highsmith AK. Pseudo- monas j?uoyscens bacteremia from blood transfusion. Am J Med 1984; 76: 62-68.

5. Tabor E, Gerety RJ. Five cases of Pseudomonas sepsis transmitted by blood transfusions (letter). Lancet 1984; 1: 1403.

6. Anaissie E, Fainstein V, Miller P et al. Pseudomonas putida. Newly recognized pathogen in patients with cancer. Anz J Med 1987; 82: 1191-I 194.

7. Nicotra MB, Rivera M; Dale AM: Shepherd R, Carter R. Clinical, pathophysiologic, and microbiologic char- acterization of bronchiectasis in an aging cohort. Chest 1995; 108: 955-1061.

8. Oberhofer TR. Use of the API 2OE, OxiiFerm, and Minitek systems to identify nonfermentative and oxidase-positive fermentative bacteria: seven years of experience. Diagn Microbial IIzfect Dis 1983; 1: 241- 256.

9. Kropec A, Lemmen S, Wursthorm M, Daschner FD. Combination effect of meropenem with aminoglyco- sides and teicoplanin on Pseudomonas and entevococci. Infection 1994; 22: 306-308.

10. Stockley RA, Hill SL, Burnett D. Nebulized antibiotics in chronic purulent bronchiectasis. Clin Therapeut 1985: 7: 593-599.

Lobectomy in cystic fibrosis

R. BRAGONIER+, D. GRIER+, F. CARSWELL”, AND S. CUNNINGHAM*

Departments of “Paediatrics, ‘Paediatvic Radiology and *Cavdiothovacic Suvgevy, Bristol Royal _Elospifal fey Sick Children, Bristol, U.K.

Introduction Case 1

In cystic fibrosis, (CF) the lungs are usually affected dif- fusely, but in some patients, focal disease may predominate (1). Aggressive medical therapy with intravenous antibiotics and chest physiotherapy is the first-line treatment, but in cases which fail to respond, surgical resection of the affected lobe has been advocated (2). We reviewed the clinical courses, up to 15 months post-operatively, of all three of our patients in whom this procedure had been undertaken. All patients had received prolonged intensive medical treat- ment without significant improvement in their clinical or radiological status prior to surgery.

Received 2 May 1996 and accepted in revised form 8 January 1997.

A lo-year-old boy had persistent right upper lobe bron- chiectasis. His mean lung function tests (o/ predicted for height) preceding lobectomy, derived from all lung function results recorded over the year, were FEV, 44% (SD 3.7%) and FVC 61% (SD 9.0%), and he required seven hospital admissions. During the post-operative year, his mean lung function tests improved, with FEV, 48% (SD 6.4%) and FVC 68% (SD 9.6%): and he was admitted five times for treatment. One year post- operatively, his weight centile position was unchanged but his height centile position had improved; although he remained pre-pubertal. His chest X-ray Crispin Norman (CN) score had fallen from 28, pre-operatively, to 8 (Plate 1).

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696 CASEREPORTS

PLATE 1. PA chest radiographs of Case 1 showing air-filled bronchi within the collapsed right upper lobe pre-operatively (a), and appearances following right upper lobectomy and insertion of a totally implantable venous access device (b).

Case 2

Another lo-year-old boy had suffered from left lower lobe collapse for 2 years. Overall, mean lung function during the post-operative year was reduced, with FEV, 32% (SD 5.5%) and FVC 42% (SD 4.8%) compared to 50% (SD 0.0%) and 60% (SD 4.2%), respectively, during the pre-operative year. However the trend in lung function following recovery from surgery showed gains in FEV, of +5% and FVC of + 10% (percentages predicted for height) between 3 and 15 months post-operatively. He was admitted four times in the pre- operative year and the same number of times in the post-operative year, although this was on a new programme of elective 3-monthly antibiotic courses. One year after operation, his weight centile had declined slightly although his height centile was unchanged. His CN score had fallen from 8 to 5.

Case 3

This 7-year-old girl had right lower lobe bronchiectasis. Mean lung function results in the pre-operative year were FEV, 76% (SD 6.5%) and FVC 77% (SD 6.5%) which fell to 53% (SD 10.8%) and 57% (SD 14.10/o), respectively, in the year following her lobectomy. As in Case 2, however, she showed a trend of improving lung function between 3 and 15 months post-operatively, with gains in FEV, and FVC of +6 and +24% (percentages predicted for height), respectively. She required only two admissions in the post- operative year compared to five admissions pre-operatively. Height centile improved although weight centile fell. Her CN score rose from 11 at the time of operation to 16 1 year later.

Discussion

Other centres have reported their experience with CF patients treated by pulmonary resection (l-5). The

observed benefits have been a reduction in symptoms of cough and sputum production, less frequent infective exacerbations and reduced rates of hospital admission (as seen in the study patients). Marmon et al. (2) observed little change in their 11 patients’ lung function post-operatively, Smith et al. (3) noted significant decreases in post-operative FEVi but not FVC in their 14 patients, and Lucas et al. (4) reported both increased and decreased respiratory function among six patients. Two of our patients experienced reduc- tion in mean pulmonary function post-operatively, but one improved. Interestingly, once the patients had recovered from surgery (3 months post-operatively), all three showed increases in FEV, and FVC over the subsequent 12 months, perhaps indicating that the removal of the nidus of infec- tion had allowed a degree of overall lung recovery. Growth was not always commented upon in other series. Mearns et al. (1) observed both increases and decreases in growth centile position post-operatively, as was the case in our patients, whereas Steinkampand et al. (5) recorded improvement in weight centile in all three of their subjects (height centiles not recorded). Reduction in CN score, as occurred in two of our patients, is perhaps not surprising since the areas of lung with the most severe disease had been removed.

The small number of patients in this review precludes any definitive conclusions about the value of lobectomy in CF. However, all three patients had some evidence of benefit from the procedure, particularly Case 1 who improved in almost all parameters examined. Lobectomy may be a useful treatment option for those CF patients in whom vigorous medical therapy for localized pulmonary disease has failed.

Note

Previous thoracic surgery may make subsequent heart-lung transplantation more difficult. However, since heart-lung transplantation began in the U.K. in 1985, the relative and

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CASE REPORTS 697

absolute contraindications have changed. Previous lung resection is no longer an absolute contraindication, and patients treated by lobectomy have successfully undergone subsequent transplantation. Each patient should be assessed individually.

Acknowledgements

The authors are indebted to Mr James Wisheart; FRCS, who performed all three lobectomies. RB was supported by The Cystic Fibrosis Trust (U.K.).

References

1. Mearns MB et al. Pulmonary resection in cystic fibro- sis. Arch Dis Child 1912; 47: 499-508.

2. Marmon L, Schidlow D, Palmer J, Balsara RK, Dunn JM. Pulmonary resection for complications of cystic fibrosis. J Pediutr Swg 1983; 18: 81 l-815.

3. Smith MB, Hardin WD Jr; Dressel DA, Beckerman RC, Monyihan PC. Predicting outcome following pul- monary resection in cystic fibrosis patients. J Pediatv Suvg 1991; 26: 655-659.

4. Lucas J, Connett GJ, Lea R, Rolles CJ, Warner JO. Lung resection in cystic fibrosis patients with localised pulmonary disease. Arch Disc Child 1996; 74: 449-45 1,

5. Steinkamp G, von der Hardt H, Zimmermann HJ. Pulmonary resection for localized bronchiectasis in cystic fibrosis. Acta Paediatr Stand 1988; 77: 569-575.

Cardiac sarcoidosis: the value of magnetic resonance imaging in diagnosis and assessment of response to treatment

M. J. DOHERTY*, S. KISHORE KUMAR*, A. A. NICHOLSON+ AND D. V. MCGIVERN’

“Chest Medical Unit, Castle Hill Hospital, Cotfingham, Neav Hull, U.K. ‘Depavflnenf of Radiology, Hull Royal h$nnavy, Hull, U.K.

Introduction

Sarcoidosis is a multisystem granulomatous disorder of unknown aetiology. The respiratory system is most com- monly involved and therefore the majority of cases come under the care of respiratory physicians. Symptomatic cardiac involvement is found in less than 5% of patients (1); although cardiac granulomata have been noted in as many as 27% at autopsy (2). The diagnosis of cardiac sarcoidosis can be difficult and in established cases the prognosis is poor (3). Corticosteroids reportedly help and are commonly used, although patients are still prone to sudden death and progression to end-stage cardiomyopathy (4). We report a case of cardiac sarcoid diagnosed using magnetic resonance imaging (MRI) which suggests that this form of imaging can lead to an early diagnosis at which stage treatment may be more likely to be beneficial.

Received 25 February 1997 and accepted in revised form 2 June 1997.

Case Report

A 55-year-old factory foreman initially presented following an episode of syncope and two episodes of near syncope. These episodes were all of short duration, occurred over a period of 6 months and were not associated with palpita- tions or chest pain. Clinical examination was completely normal, blood pressure was 130/80, breath sounds were vesicular and heart sounds were normal. He was an ex-smoker who had been previously well but did describe the occurrence of red blotches on his lower legs 18 months earlier, which had been self-limiting.

Electrocardiography showed sinus rhythm with a QRS axis of+45 degrees. Twenty-four hour Holter monitoring showed sinus rhythm with an occasional dropped beat. His chest X-ray revealed bilateral hilar lymphadenopathy, raising the possibility of sarcoidosis. Spirometry was supra- normal. HRCT of the thorax revealed huge paratracheal, subcarinal and parabronchial lymphadenopathy but no evidence of interstitial lung involvement. Fibre-optic bron- choscopy revealed a small nodule in the apical segment of the right lower lobe, biopsy of which showed noncaseating