Clinics and Practice 2011; volume 1:e99 Systemic sclerosis with sputum and bronchoscopic examinations. She has received a diagnosis of systemic sclerosis Correspondence: Attapon Cheepsattayakorn, 10th hemoptysis and a huge lung (SSc) with lung involvement without evidence Zonal Tuberculosis and Chest Disease Center, cavity of family history of SSc and has received pred - Chiang Mai, 10 th Office of Disease Prevention nisolone and plaquenil. Cyclophosphamide and Control, Department of Disease Control, Attapon Cheepsattayakorn, 1 was added between 2000 and 2008 due to par - Ministry of Public Health, 143 Sridornchai Road Changklan Muang Chiang Mai 50100 Thailand. 2 Ruangrong Cheepsattayakorn tially clinical response to prednisolone. Five- E-mail: [email protected] 110 th Zonal Tuberculosis and Chest years follow-up of the chest radiograph and the HRCT of the chest still revealed reticulocystic Disease Center, Chiang Mai, 10 th Office of Key words: systemic sclerosis, huge lung cavity, lesions indicated bronchiectasis and bronchi - hemoptysis. Disease Prevention and Control, olectasis, honeycombing, subpleural bands and Department of Disease Control, Ministry septal thickening at the both lower lobes with Received for publication: 19 September 2011. 2 of Public Health; Department of new patchy-cystic lesion at the right midlung Accepted for publication: 18 October 2011. Pathology, Faculty of Medicine, Chiang field, new small fibronodular infiltration at Mai University, Chiang Mai, Thailand This work is licensed under a Creative Commons periphery of the posterior aspect of the both Attribution NonCommercial 3.0 License (CC BY- upper lobes and lower lobes, and new minimal NC 3.0). right apical pleural thickening without pleural effusion while the mediastinum was within ©Copyright A. Cheepsattayakorn and R. Cheepsat- Abstract normal limits (Figure 3). Five years previous tayakorn, 2011 she was also performed the echocardiography Licensee PAGEPress, Italy due to dysnea on supine position for four years Clinics and Practice 2011; 1:e99 Systemic sclerosis or scleroderma is associ - doi:10.4081/cp.2011.e99 ated with distal vasculitis, Raynaud’s phenom - and revealed moderate mitral valve regurgita - enon, and inflammation of internal organs and tion with progression to severe form 1 year the skin. We present on a 58-year-old Thai later without pleural effusion and pulmonary 2,3 woman with systemic sclerosis who came to arterial hypertension (PAH). She has been culopathy. Numerous known agents induce the 10 th Zonal Tuberculosis and Chest Disease added ferrous sulphate therapy. Other labora - SSc-like pulmonary disease; for examples, Center, Chiang Mai, Thailand in 2009 and pre - tory results were unremarkable. Two years pre - benzene, toluene, trichloroethylene, sented with hemoptysis and a solitary huge- vious she developed sclerodactyly of both bleomycin, pentazocine, trytophan, and D- 4 5 lung cavity as the predominant clinical mani - hands with severe anemia. Increased dosage penicillamine including silica and rapeseed 6 festations which spontaneously resoluted 2 of ferrous sulphate had been prescribed for 2 oil denatured with aniline. The patient’s one months later. This case demonstrates a solitary years. She is a non-smoker. On physical exam - major criterion of thickening of both hands huge-lung cavity with hemoptysis and looked ination, the patient’s vital signs were within and two minor criteria of sclerodactyly of both like from non-tuberculous Mycobacterial infec - normal limits. There was mildly thickening of hands and bilaterally basilar idiopathic pul - tions or malignancy with spontaneous resolu - the skin of the face, neck, and extremities monary fibrosis (IPF) made the diagnosis of tion of hemoptysis and the lung cavity, which without dysnea. Evidence of sclerodactyly of SSc. 7 She has had a history of Raynaud’s phe - does not need invasive investigations. both hands was noted. nomenon which is a common clinical manifes - tation (91%) 8 and more than 10 years of IPF which we hypothesized the early clinical sign which was found between 52%-70%. 8,9 Case Report Investigations Hemoptysis found in this patient without evi - dence of PAH, TB and D-penicillamine 10,11 and Hemoptysis for one year developed in a 58- On the first day of the patient’s presenta - other known-induced agents 4-6 exposure is a year-old Thai woman with medical history of tion, we hypothesized that chronic receiving of rare complication which may complicate thickening of the skin, face, neck, and both prednisolone can allow development of pul - bronchial telangiectasia or carcinoma. 12 upper extremities with flexion deformity of monary TB with presenting hemoptysis due to Wangkaew S. et al. reported their study among both hands (Figure 1), cool-exposure-cyanotic drug-induced immunosuppression. Thus, the Thai SSc patients with 59.1% (39 of 275 skin changes and difficulty in the mouth open - three consecutive sputum examinations for patients) of PAH and 92.3% (36 of 275 ing for 10 years presented to the 10 th Zonal the AFB were performed including sputum cul - patients) of dyspnea on exertion 13 while Tuberculosis and Chest Disease Center, tures for Mycobacterium tuberculosis and Panicheewa S. et al. reported of 43.3% with Chiang Mai, Thailand in 2009. Ten years previ - other Mycobacterium species, which revealed pulmonary involvement among Thai SSc ous, she was performed the chest radiograph, negative results. A chest radiograph showed a patients. 14 Only 26.5% (22 of 83 Thai SSc high-resolution computed tomography (HRCT) huge thin-wall cavity with 8 cm in diameter in patients) were diagnosed PAH by echocardiog - of the chest, three consecutive sputum acid- the right lower lobe, bilaterally diffuse fibrotic raphy. 15 A study reported in 2004 of only one fast bacilli (AFB) examinations, and broncho - infiltration, bilateral pleural effusion, and case of complicated pulmonary TB among 11 scopic examinations which revealed reticular bilateral basal pleural thickening (Figure 4). patients with SSc-associated interstitial lung and small cystic infiltration with fibrotic disease treated with azathioprine. 16 Serum change and some ground glass infiltration at concentrations of insulin-like growth factor the both lower lobes, more at the basal lungs (IGF)-1 17 and IGF binding protein, 17 inter - and periphery of the upper lobes (Figure 2). Discussion leukin-15 18 and chest ultrasonography 19 can Tubular bronchiectasis was seen at bilaterally early and simply predict the development of perihilar regions while small cystic bronchiec - SSc is systemic and complex collagen vascu - pulmonary telangiectasia 17 and IPF, 18,19 respec - tasis was shown at the both lower lobes lar disease of unknown etiology, 1 associated tively which were not early done in this case. (Figure 2) without evidence of tuberculosis with excessive tissue fibrosis of the skin and Fortunately, only 0.2% of cases with pulmonary (TB) or other Mycobacterium species from various internal organs and small-vessel vas - hemorrhage or hemoptysis or both are regis - [Clinics and Practice 2011; 1:e99] [page 215 ] Case Report tered as a leading cause of death. 10 Honeycombing 20 of the lungs on the last chest HRCT indicated more advanced disease and serum anti-topoisomerase autoantibody 21,22 measurement could strongly predict the patient’s 10-year previous IPF before progres - sion to this pathological lung appearance. On the chest radiograph of the first attendance at this Center, it demonstrated a huge thin-wall cavity at the right lower lobe with acute pneu - monitis at the right midlung field and bilater - ally pleural effusion which could be SSc-asso - ciated-infective brochiectasis and this lung Figure 1. Showing the thickening of the skin of both upper extremities with sclerodacty - cavity spontaneously resolved 2 months later ly of both hands (A, B). excepted the bilaterally pleural effusion (Figure 4). This lower lobe huge-solitary cavi - ty may be complication of the SSc-associated- cystic bronchiectasis seen on the patient’s 10- year previous chest radiograph and HRCT of the chest and it can cause hemoptysis which disappeared at the same time of this lung cav - ity resolution. The patient’s 10-year and 5-year previous chest radiographs and HRCTs of the chest showed usual patterns of the disease with previously normal bronchoalveolar lavage examinations and only bilaterally minimal- pleural effusion on the last chest radiograph indicated not necessary to perform lung biop - sy. 23 On the last attendance the patient Figure 2. Posterior-anterior chest radiograph (A) and high-resolution computed tomog - revealed very dramatic clinical responses raphy of the chest (B) at ten years previous showing reticular and small cystic infiltration excepted the difficulty in open her mouth, with fibrotic change and some ground glass infiltration in the both lower lobes. thickening of the skin, and the Raynaud’s phe - nomenon. Conclusions This case does not need invasive investiga - tions for diagnosis. Our case highlights a huge lower-lobe-thin-wall cavity in SSc patient, which has been reported as SSc-associated non-tuberculous Mycobacterial pulmonary infections in the medical literature in Thailand. This case demonstrates the impor - tance of the chest radiographic follow-up of the Figure 3. Posterior-anterior chest radiograph (A) and high-resolution computed tomog - SSc-associated-lung cavity with hemoptysis raphy of the chest (B) at five years previous still showing brochiectasis and bronchiolec