Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 12  |  Issue : 1  |  Page : 129-135

Broncial stump aspergillosis, an unusual cause of hemoptysis, and review of the literature


1 MMI Narayana Multispeciality Hospital, Raipur, Chattishgarh; Formerly Senior Resident, Post Graduate Institute of Medical Education and Research, Sector 12, Chattishgarh, India
2 Professor Department of Pulmonary Medicine [WHO Collaborating Center for Research & Capacity Building in Chronic Respiratory Diseases], Post Graduate Institute of Medical Education and Research Sector-12, Chattishgarh, India

Date of Submission25-Jan-2017
Date of Acceptance03-Apr-2017
Date of Web Publication12-Jan-2018

Correspondence Address:
Dipesh Maskey
Room No. 14, MMI Narayana Multispeciality Hospital, Raipur, Chattishgarh 492001
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ejb.ejb_9_17

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  Abstract 

Aspergillus species are ubiquitous dimorphic molds found in an environment. of the 250 species, few of them are pathogenic to human and can cause various lung involvements, tracheobronchial being one of the variant. Bronchial stumps and anastomosis are prone for fungal infection and can present with cough, hemoptysis, central airway obstruction and bronchopleural fistula with its highest incidence in patients with lung transplantation. Its occurrence in other types of lung surgery is very rare and 20 cases have been described in English medical literature till date since first published in 1969. We present a case of recurrent hemoptysis caused by bronchial stump aspergillosis, first reporting from India, and systematically analyze the literature for all the reported cases of bronchial stump and anastomosis aspergillosis in patients with lung surgery other than transplantation.

Keywords: aspergillosis, bronchial anastomosis, bronchial stump, hemoptysis


How to cite this article:
Maskey D, Agrawal R. Broncial stump aspergillosis, an unusual cause of hemoptysis, and review of the literature. Egypt J Bronchol 2018;12:129-35

How to cite this URL:
Maskey D, Agrawal R. Broncial stump aspergillosis, an unusual cause of hemoptysis, and review of the literature. Egypt J Bronchol [serial online] 2018 [cited 2018 Jul 20];12:129-35. Available from: http://www.ejbronchology.eg.net/text.asp?2018/12/1/129/223088


  Introduction Top


Aspergillus spp. are ubiquitous dimorphic molds found in soil, various types of decaying organic debris, water, stored hay and grains, and indoor environments [1]. Of the 250 species, few of them are pathogenic to humans and can cause various lung involvements, tracheobronchial being one of the variants, albeit low in incidence [2]. Bronchial stumps and anastomosis are prone for fungal infection and can present with cough, hemoptysis, central airway obstruction, and bronchopleural fistula with its highest incidence in patients with lung transplantation. Its occurrence otherwise is rare and 20 cases have been described in English medical literature to date, since first published in 1969. We present a case of recurrent hemoptysis caused by bronchial stump aspergillosis (BSA) and systematically analyze the literature for all reported cases of bronchial stump and anastomosis aspergillosis in patients with lung surgery other than transplantation.


  Case report Top


A 59-year-old housewife underwent enucleation of the right middle lobe with ruptured hydatid cyst in 2003 at our institute and had uneventful recovery. She was doing fine until 3 years ago when she started complaining of intermittent episodes of streaky hemoptysis that used to subside with short course of antibiotics. The complaints lasted for about 2 years, and then she had spontaneous resolution a year back when she again had recurrence of hemoptysis, which was more frequent and large in quantity without relief with short courses of antibiotics. However, she denied fever, chest pain, mucopurulent sputum, loss of appetite, and weight loss. A sputum examination was negative for acid fast bacilli. Chest radiography showed consolidation silhouette right cardiac border, and computed tomography thoracic angiography revealed mildly hypertrophied right bronchial artery and subsegmental consolidation in the right middle lobe and the apical segment of the right lower lobe. Fibreoptic bronchoscopy showed foreign body-like hard material in the subsegment of the right middle lobe. Suture granulation versus organized secretions was considered and biopsy was performed [3] ([Figure 1]). Histopathology revealed multiple fragments of suture material, necrotic tissue, and septated fungal hyphae with acute angle branching resembling the morphology of Aspergillus spp. ([Figure 3] and [Figure 4]). BSA was diagnosed and she was started on oral itraconazole 200 mg twice daily for 8 weeks, followed by 100 mg twice daily for another 8 weeks. She did not have hemoptysis following treatment subsequently, and repeat bronchoscopy performed after 16 weeks of therapy revealed the presence of excess secretion but no evidence of BSA ([Figure 2]).
Figure 1 Bronchoscopic image shows golden brownish foreign body like hard material in subsegment of right middle lobe.

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Figure 2 Post treatment bronchoscopic image showing disappearance of stump aspergillosis.

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Figure 3 H&E stain shows multiple fragments of suture material (prolene), necrotic tissue and septated fungal hyphae.

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Figure 4 Silver stain showing multiple septated fungal hyphae in hpf.

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  Discussion Top


BSA and bronchial anastomosis aspergillosis occurs infrequently in immunocompetent host after lung surgery. Bronchial stump/anastomosis aspergillosis occurs due to colonization of endobronchial suture and infection of the surrounding granulation tissue by Aspergillus spp. It is considered as a saprophytic form of tracheobronchial aspergillosis [2]. We performed systematic search in PubMed using the key words BSA and bronchial anastomosis aspergillosis and came across a total of 20 cases published to date; since first published in 1969. This is the first case reported from India and the first case of BSA after enucleation surgery. The baseline characteristics of these patients are shown in [Table 1]; and details of clinical presentation and bronchoscopy findings are tabulated in [Table 2]. The mean age was 44.67 years. A total of 14 patients had undergone lobectomy. Silk was the most commonly used suture for stump closure. Hemoptysis was the most common presentation after surgery with a mean±SD time of 36.43±26.19 months. Many cases received multimodal treatment; which included removal of infected tissues and sutures; local application or inhalation of antifungal agent; and or use of systemic antifungal agents. Two cases required surgery to cure it. Each case management and outcome is tabulated in [Table 3].
Table 1 Baseline characteristics and treatment of bronchial stump/anastomoses aspergillosis

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Table 2 Clinical presentations and bronchoscopic findings

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Table 3 Treatment and outcome of bronchial stump aspergillosis

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Sawasaki et al. [3] first described BSA as a specific clinical entity in English medical literature. In their landmark study, they demonstrated that the use of silk suture was the main reason for developing BSA. Out of 495 pulmonary resections with silk thread suture, nine (1.6%) cases developed BSA, whereas in 140 pulmonary resections using nylon monofilament, none developed this complication. Silk suture produced more local inflammation (24.6%) compared with nylon monofilament (5.7%), which was also proven experimentally. It was shown that, in addition to its irritative quality, silk thread has inherent capillarity and nutritive value that can act as a nidus for fungal growth. This study led to the replacement of silk as a suture material in sewing bronchial stumps. However, there are few cases described with these new suture materials (nylon [4], prolene [5], synthetic thread [9], and titanium staple [10]).

Patients with BSA usually present with hemoptysis that can mimic recurrence of lung cancer. They can present with cough and expectoration of putrid sputum as well as fungal mass and suture material. Interestingly, there are case reports of BSA presenting with Aspergillus empyemas [6], central airway obstruction [4],[5], and increased fluorodeoxyglucose uptake in positron emission computed tomography in an asymptomatic patient after lung cancer resection [9]. The onset of symptoms is variable, presenting as early as 1 month and as late as 7 years [7],[10]. Bronchoscopic examination of the bronchial stump can be normal or show suture thread with surrounding inflamed tissue or granulation tissue covered in purulent material or fungal mass. Histopathological examination of the bronchial biopsies from the stump reveals inflammatory cells and necrotic areas infiltrated by hyphae with morphological feature suggestive of Aspergillus spp. In addition, suture material may be seen as in our case. Culture of the tissue can be positive for Aspergillus spp.

No concomitant presence of other forms of aspergillosis has been published to date except for the presence of a mycetoma [7] and empyema [6] in two of the cases. However, clinical overlap and progression to other forms of aspergillosis is well known [1],[2]. There is no clear recommendation for the treatment of BSA. However, removal of the suture and granulation tissue if possible remains the most effective therapy and prevents relapse. Endobronchial sutures can be removed by means of bronchoscope using forceps, endoscopic suture scissor, or laser therapy. In one of the reported cases in which polypoidal growth was present with airway narrowing, argon plasma coagulation was used to coagulate the lesion with subsequent forceps debridement [5]. In most of the cases, local instillation of antifungal agents (tincture iodine and phenylacetic quicksilver) and or inhalation of antifungal agent (amphotericin and mycostatin) had been used [3],[4]. In one of the reported cases, only removal of suture led to cure, whereas in other two cases patients developed relapse until the thread was removed [4],[7]. In cases in which suture thread cannot be removed, oral itraconazole and voriconazole had been used with success [8],[9],[10], but the exact dosage and duration of treatment cannot be specified due to case reports and patient had received therapy as long as 20 months depending upon the clinical response [8].

In conclusion, BSA should be considered as a possible differential diagnosis in patients who undergo lung surgery and present with symptoms of cough, purulent sputum, or hemoptysis and early bronchoscopy and biopsy must be performed. Removal of the suture thread is the most effective therapy and oral itraconazole or voriconazole can be used adjunctively or if the relapse occurs.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Al-Alawi A, Ryan CF, Flint JD, Muller NL. Aspergillus-related lung disease. Can Respir J 2005; 12:377–387.  Back to cited text no. 1
    
2.
Krenke R, Grabczak EM. Tracheobronchial manifestations of Aspergillus infections. Scientific World J 2011; 11:2310–2329.  Back to cited text no. 2
[PUBMED]    
3.
Sawasaki H, Horie K, Yamada M, Tajima G, Katsura S, Naito Y et al. Bronchial stump aspergillosis. Experimental and clinical study. J Thorac Cardiovasc Surg 1969; 58:198–208.  Back to cited text no. 3
    
4.
Le Rochais JP, Icard P, Simon T, Poirier P, Evrard C. Bronchial stump aspergillosis. Ann Thorac Surg 2000; 70:302–304.  Back to cited text no. 4
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5.
Rissmiller R, Copley P, Conforti J. Localized bronchopulmonary aspergillosis colonizing a previous sleeve resection for carcinoid tumor resulting in critical airway obstruction. J Bronchol 2004; 11:250–253.  Back to cited text no. 5
    
6.
Parry MF, Coughlin FR, Zambetti FX. Aspergillus empyema. Chest 1982; 81:768–770.  Back to cited text no. 6
[PUBMED]    
7.
Roig J, Ruiz J, Puig X, Carreres A, Morera J. Bronchial stump aspergillosis four years after lobectomy. Chest 1993; 104:295–296.  Back to cited text no. 7
[PUBMED]    
8.
Noppen M, Claes I, Maillet B, Meysman M, Monsieur I, Vincken W. Three cases of bronchial stump aspergillosis: unusual clinical presentations and beneficial effect of oral itraconazole. Eur Respir J 1995; 8:477–480.  Back to cited text no. 8
    
9.
Garcia-Olive I, Andreo F, Rosinal O, Sanz-Santos J, Font A, Monso E. Bronchial stump aspergillosis after lobectomy for lung cancer as an unusual cause of false positive fluorodeoxyglucose positron emission tomography and computed tomography: a case report. J Med Case Rep 2011; 5:72.  Back to cited text no. 9
    
10.
Tokuishi K, Yamashita S, Hashimoto T, Moroga T, Miyawaki M, Chujo M et al. Bronchial stump aspergillosis after stapled lobectomy for lung cancer. Ann Thorac Surg 2012; 94:1324–1326.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]
 
 
    Tables

  [Table 1], [Table 2], [Table 3]



 

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