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CaseReport

Introduction

Broncholithiasis is most often associated with pulmonaryinfections, eg, tuberculosis and histoplasmosis. Stonesoriginate from calcified peribronchial lymph nodes thaterode into the trancheobronchial tree, but lithoptysis oc-

Surgical Management of Tuberculous Broncholithiasiswith Hemoptysis: Experience with 5 Operated Cases

From the Cardiothoracic Department, Quem Hospital, MashhadUniversity of Medical Sciences, Mashhad, Iran

Received March 31, 2006; accepted for publication September 5,2006Address reprint requests to Bagheri Reza, MD: CardiothoracicDepartment, Quem Hospital, Mashhad University of MedicalSciences, Mashhad, Iran.

curs infrequently. The most common symptoms are per-sistent cough and hemoptysis, sometimes followed byfeatures of obstructive pneumonia (fever, chills and pu-rulent sputum). The physical findings are nonspecific andradiologic findings are varied. Complications include fis-tula formation between the respiratory tract and theesophagus or aorta and obstructive pulmonary symptomsand hemoptysis. Treatment ranges from non operativemanagement (simple observation), bronchoscopic re-moval of broncholithiasis and thoracotomy for patientsin whom complications develop, such as hemoptysis orobstructive pneumonia or secondary bronchiectasis.1)

In our country, the most common cause of this prob-

Bagheri Reza, MD and Haghi Ziaollah, MD

Introduction: Broncholithiasis is often seen after chronic granulomatous diseases such as tu-berculosis and hystoplasmosis and leads to a wide spectrum of signs and symptoms; includinghemoptysis which often needs surgical management. The goal of this study is evaluation of sur-gery in patients with tuberculous broncholithiasis who present with hemoptysis.Materials and Methods: In this study all patients with tuberculous broncholithiasis had beenoperated on between 1991 and 2005, followed up at least 6 months and at most 9 years, andstudied relating to age, sex, clinical symptoms, diagnostic methods, type of surgical treatment,complications and mortality.Results: Overall 5 patients have been studied (male:female=2:3, mean=31 years); 2 with severeand 3 mild to moderate and recurring hemoptysis, lesion at left in 80% and at right in 1, in 3patients some degree of bronchiectasia was seen, in 4 the lesion was visible in bronchoscopy andendoscopic removal of the lesion failed in all. Three of patients underwent pulmonary resectionsand in 2 broncholithotomy has been done. In follow-up, patients treated with pulmonary resec-tion have had no subsequent problems, but in patients treated with broncholithotomy due tooccurring late bronchiectasia, re-operation and pulmonary resection was inavoidable. Therewas no mortality.Conclusion: Regarding the dangers of hemoptysis and excellent results of surgery and possibleoccurance of late bronchiectasia after broncholithotomy, the results of our study show that pul-monary resection distal to the lesion and as the retention of lung of parenchyma is preferable.Broncholithotomy should be done only in patients in whom pulmonary resection is not techni-cally possible. Because of the very low occurance of this complication complete studies are needed.(Ann Thorac Cardiovasc Surg 2007; 13: 185–190)

Key words: broncholithiasis, tuberculosis, hemoptysis, surgery

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lem is endemic tuberculosis. The goal of this study is theevaluation of surgical management of patients withbroncholithiasis and hemoptysis due to tuberculosis.

Materials and Methods

In a descriptive (case series) study between 1991 to 2005,all patients with tuberculous broncholithiasis and hemop-tysis in Quaem and Omid hospitals in Mashad, have beenevaluated. Age, sex, the duration between lesion occurenceand diagnosis of tuberculosis, the duration between firsthemoptysis episode and surgery, bronchoscopic and ra-diologic diagnosis of lesion, tests for defining tuberculo-sis bacilli in spectrum, the type of surgery, and complica-tions of surgery and mortality have been studied.

Results

Overall 5 patients were included in this study: 2 malesand 3 females. The mean at admission was 31 years (theyoungest 18 years and the oldest 58 years). The meanduration between occurrence of hemoptysis due tobroncholithiasis and of tuberculosis treatment was 8.5years (6 to 10 years); 2 patients with massive hemoptysisin the last refer have been operated and completion in theother 3 patients with mild to moderate recurring hemop-tysis surgery has been done.

All the cases have been under medical treatment be-fore surgery, but none of them had successful medicaltreatment. Due to severe attack hemorrhage, they werecandidates for surgery. As patients with asymptomatic

broncholithiasis do not present for treatment, we did nothave a comparative group.

In all patients chest X-ray and computed tomography(CT)-scan showed broncholithiasis clearly.

The site of the lesion in 4 patients was left sided. In 3patients there were some degrees of bronchiectasia insegments distal to the lesion. Figures 1 and 2 demonstratechest X-Ray and CT-scan of a 29 years old female withtuberculosis, broncholiths and hemoptysis.

Diagnostic fibroptic bronchoscopy was done in all pa-tients and in 4 patients erosion of broncholithiasis into

Fig. 1. Multiple broncholithiasis (adjacent to pulmonary arteryand lower lobe bronchus).

Fig. 2. Computed tomography (CT)-scan of the same patient bron-chiectatic areas in lower lobe is demonstrated.

Fig. 3. Bronchoscopic image of broncholithiasis.

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specimen.Figure 5 shows the radiographic image of the same

patient after operation.In the other 2 patients who had mild hemoptysis with

no distal bronchiectasia on radiographic studies, onlybroncholithotomy and adjacent bronchial vessel ligationhave been done. Figure 6 shows a large bronchiolith inthe inferior lobe bronchi.

At follow-up 3 patients with pulmonary resection (fol-lowed between 6 months to 9 years) there have been noproblem and they were virtually cured. In 2 patients withbroncholithotomy alone, 1 patient at 3 years and the other

the airway were clearly visible. Figure 3 shows thebroncho-scopic view of the lesion in these patients.

Attempting bronchoscopic removal was unsuccessfulin all patients. In all patients the sputum tests for tubercu-losis bacilli were negative.

All patients underwent open surgery using posterolat-eral thoracotomy approach in the 5th intercostal space.Three patients who had moderate to severe hemoptysisand had some degree of bronchiectasis distal to lesion. In2 patients left lower lobectomy and in 1 patient resectionof 2 segments of the left lower lobe has been done. Fig-ure 4 shows the gross pathologic image of the resected

Fig. 4. In gross pathologic image location of broncholithiasis isclearly visible (arrow).

Fig. 5. Postoperative radiographic image of the same patientafter left lower lobectomy.

Fig. 6. Chest X-ray [lateral (left) and frontal (right) views] of patient with large broncholithiasis (arrows).

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at 6 years reoperation was done due to severe symptom-atic bronchiectasis. In 1 patient left lower lobectomy pluslingulectomy and in the other right lower lobectomy hasbeen done. These 2 patients have had no further prob-lems.

Figure 7 shows a CT scan of the patient in Fig. 6 thathad broncholithotomy due to large broncholithiasis. TheCT scan shows bronchiectasis in left lower lobe and lin-gula.

In all patients histopathologic examinations have dem-onstrated tuberculous broncholithiasis. Figure 8 showsthe microscopic image of tuberculous broncholithiasis.

There has been no mortality in our patients.

Fig. 7. Bronchiectasis in left lower lobe and lingula is obvious.

Fig. 8. In microscopic view of broncholitiasis calcification (blackarrow) fibrotic capsule (white arrow) is seen.

Discussion

Broncholithiasis is ossified or calcified material whichhas penetrated the bronchial lumen. The common causeof this lesion is tuberculosis and histoplastomosis.2) It hasa wide spectrum of clinical manifestations: forms withmild symptoms of cough and sometimes litoptysis to morecomplicated forms.3,4)

The chemical composition of bronchiolith is very simi-lar to bone; it has 85–95% calcium phosphate and 7–10%calcium carbonate.5)

Complications of broncholithiasis are various from amild lithoptysis to more severe symptoms. One of the mostdangerous complications of this lesion which almost needssurgery is hemoptysis.6) In most researches due to havefew patients, and this complication has been reported ascase report. For example Stocia has reported a 51 yearsold man with massive hemoptysis and broncholithiasiswho has been operated on.7) Similar cases have been re-ported by Meyer et al. They mention that the cause ofhemoptysis is erosion of broncholithiasis through adja-cent bronchial vessels which leads to severe and lethalhemoptysis in patients.8) Other complications that lead tosurgery are obstructive pneumonia and secondary bron-chiectasis, which may be present with fever and chillsand purulent sputum.1)

Another complication of broncholithiasis which oftenneeds surgery is bronchoesophagial fistula. This is dan-gerous and the operation may be difficult.9)

Broncholithiasis in some patients presents as middlelobe syndrome, and in all patients with such a presenta-

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tion, one should consider broncholithiasis.10)

Radiographic and CT-scan images of tuberculousbroncholithiasis are various but often sufficient to makethe diagnosis. These lesions are clearly found on chestX-ray and CT-scan. Broncholithiasis is suggested on CT-scan when endobronchial or peribronchial calcified nodesare associated with findings of bronchial obstruction.Volume data acquisition by means of helical CT with sec-tions less than 3 mm in thickness and multiplanar refor-mation along the bronchial tree may be helpful in con-firming the endobronchial location of the calcified mate-rial. Primary endobronchial infection with dystrophiccalcification and hypertrophied bronchial arteries withintramural protrusion and calcified endobronchial tumorsand trachobronchial disease with mural calcification maymimic broncholithiasis.2)

In addition to above radiographic findings, primaryendobronchial actinomycosis rarely has very similar im-ages to broncholithiasis. In the report of Seo et al., theyhave undertaken bronchoscopy and biopsy in 2 patientspresentings as broncholithiasis without a history of granu-lomatous disease and the diagnosis of actinomycosiswasapproved in these.11)

Endoscopic findings consist of tracheobronchial dis-tortion, inflammation, a visible broncholith, bleeding andoccasionally a patient may have the endoscopic findingof a fistula in either the esophagus or the tracheobron-chial tree.12)

Treatment guidelines are controversial due to very fewcase reports. It is usually agreed that in asymptomaticforms or mild forms patients may be observed only orbroncholithiasis can be removed bronchoscopically.Huang et al., have reported successful removal ofbroncholithiasis.13) Similar results have been reported byMinivale et al. which in asymptomatic or mild forms theyremoved these lesions bronchoscopically. This method isconsidered the ideal treatment in this group of patients.14)

In our group, endoscopic removal of lesion was not suc-cessful.

The most report on indications surgery by Trastek etal., discussed 54 patients with broncholithiasis. The indi-cations for surgery are: severe or recurring hemoptysis,symptoms of distal obstruction (infection with fever andpuralent sputum) or esophagobronchial fistula.15) Otherinvestigators also mention the same indication for sur-gery.13,14,16)

There are different opinions about surgical technique,from simple broncholithotomy to pulmonary resection andpublished maximum saving of living parenchyma.

Trastek et al., the most complete study, believe thatoperating on these patients is very dangerous due to se-vere adhesions. The goal of surgery is removal of calci-fied masses with destroyed lung or bronchi because oflate complications of bronchial destruction (pneumonia,pulmonary abscess or bronchiectasia) pulmonary resec-tion and saving maximum lung parenchyma is preferedto broncholithotomy.15)

Cole et al. mention attempting pulmonary resection inthese patients should be with control of proximal pulmo-nary vessels where pulmonary resection is not possibledue to severe adhesions, opening of the mass capsule andcauterizing its contents can prevent lethal complicationduring surgery.16)

In our study, we performed pulmonary resection sav-ing maximum lung parenchyma in 3 of our patients andin 2 simple broncholithotomy was done. In follow-up ofthese patients with secondary bronchiectasia, we had tore-operate and lobectomy was done in both. Simplebroncholithotomy without pulmonary resection isinsuffient treatment and is accompanied with late com-plications leading to re-operation.

In very ill patients unfit for surgery where lesion can-not be removed bronchoscopically use of yttrium-aluminummay have some benefit. Success with this procedure hasbeen reported.17)

Conclusion

Hemoptysis is a dangerous complication in tuberculosisbroncholithiasis. We recommend surgery in all patientsbecause endoscopic removal is often unsuccessful andleads to delay in treatment. Due to chronicity of lesionsand the effects in the a bronchi, prevention of secondarybronchiectasia is by pulmonary resection distal to lesionwith saving of maximum paronelyma. Broncholithotomyalone suitable only in patients in whom pulmonary re-section is techniqually impossible. Due to the very lowoccurance of this complication more complete studies areneeded.

Acknowledgments

Special thanks to Mozhgan Bahadori, MD and MahammadKalantary, MD for their cooperations.

References

1. Haine JD. Coughing up a stone. What to do about

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Ann Thorac Cardiovasc Surg Vol. 13, No. 3 (2007)

broncholithiasis. Postgard Med 1988; 83: 83–4.2. Seo JB, Song KS, Lee JS, et al. Broncholithiasis: re-

view of the causes with radiologic-pathologic correla-tion. Radiographics 2002; 199: 213.

3. Larsen KR, Faurschou P. Broncholiothiasis andlithoptysis. Ugeskr Laeger 1994; 156: 3904–6.

4. Nisar Ahmed R. Lithoptysis. J Coll Physician Surg Pak2002; 12: 558–9.

5. Dixon GF, Donnerberg RL, Schonfeld SA, WhitconbME. Advance in the diagnosis and treatment ofbroncholithiasis. Am Res Dis 1984; 129: 1028–1030.

6. Filippov VP, Ismeilob SH, Shnelev MM, MukhamedovKS. Broncholithiasis complicated by pulmonary hem-orrhage and abscessing pneumonia. Probl Tuberk 1987;15: 72–3.

7. Stocia IG. Broncholithiasis and massive hemoptysis: acase report. Eur Respir J 2003; 22: 153.

8. Meyer M, Regan A. Broncholithiasis, NEJM; 2003;348: 318.

9. Carvajal Balagura J, Mallagray Casas S, Martinez CruzR, Dancausa Onge A. Bronchoesophageal fistula andbroncholithiasis. Arch Bronchoneumol 1995; 31: 184–7.

10. Vandenbos F, Passail G. Middle lobe syndrome. RevPneumol Clin 2005; 61: 279–81.

11. Seo JB, Lee JW, Ha SY, Park JW, Jeong SH, Park JW.Primary endobronchial actinomycosis associated withbroncholithiasis. Respiration 2003; 70: 110–3.

12. Yi Ky, Lee HK, Park SJ, Lee YC, Rhee YK, Lee HB.Two cases of broncholith removal under the guidanceof flexible bronchoscopy. Korean J Intern Med 2005;20: 90–1.

13. Huang CC, Lan RS, Chiang YC, Lee CH, Shieh WB.Broncholithiasis: a neglected bronchial disease in thiscountry. illustration of three cases. Changgeng Yi XueZa Zhi 1992; 15: 44–9.

14. Menivale F, Deslee G, Vallerand H, et al. Therapeuticmanagement of broncholithiasis. Ann Thorac Surg2005; 79: 1774–6.

15. Trastek VF, Pairolero PC, Ceithaml EL, et al: Surgicalmanagement of broncholithiasis. J Thorac CardiovascSurg 1985; 90: 842–5.

16. Cole FH, Cole GR, Khandekar A, Watson DC. Man-agement of broncholithiasis: is thoracotomy neces-sary?. Ann Thorac Surg 1986; 42: 225–8.

17. Shields TW, LoCicero III J, Ponn RB, Rusch VW. Bac-terial infection of the lung and bronchial compressivedisorders. In: General Thoracic Surgery. Philadelphia:Lippincot Williams & Wilkins, 2005; p 1230.