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A Case of Pulmonary Fibrosis Associated with Rheumatoid Arthritis

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A Case of Pulmonary Fibrosis Associated with Rheumatoid Arthritis Anand et al. SpringerPlus 2014, 3:513 http://www.springerplus.com/content/3/1/513 a SpringerOpen Journal CASE STUDY Open Access A case of pulmonary fibrosis associated with rheumatoid arthritis, scleroderma sine scleroderma and ANCA associated vasculitis Amritpal Singh Anand1*, Priya Brian Joseph1 and Ernest Vera-Vazquez2 Abstract Introduction: Diffuse parenchymal lung disease (DPLD) may be idiopathic or may be due to known associations such as autoimmune diseases. The prognosis in cases associated with autoimmune diseases depends on many factors such as histopathology, baseline lung function, auto-antibody level, et cetera. DPLD and its prognosis is worse in patients with overlap syndromes. Case description: We present a rare case of a 71 year old Caucasian lady with gradually worsening pulmonary fibrosis secondary to an overlap syndrome with rheumatoid arthritis (RA), scleroderma sine scleroderma (SSS) and anti neutrophil cytoplasmic antibody (ANCA) associated vasculitis. Discussion and Evaluation: In this paper, we share information from review of literature regarding DPLD associated with RA, systemic sclerosis (SSc) and ANCA associated vasculitis. Details of our discussion include prognostic factors, histology and radiographic features of these individual disease entities. Conclusion: Since pulmonary fibrosis in overlap syndromes has a poor prognosis, extensive work up should be performed even when clinical evidence of only one autoimmune disease is present. Keywords: Diffuse parenchymal lung diseases; Pulmonary fibrosis; Rheumatoid arthritis; Scleroderma; ANCA associated vasculitis Introduction UIP and NSIP have characteristic pattern in high reso- Diffuse parenchymal lung disease (DPLD) is a heteroge- lution computed tomography (HRCT) scan of chest. UIP neous group of disorders characterized by inflammation is characterized by basal dominance, peripheral reticular and/or fibrosis of the parenchymal interstitium of the abnormalities, traction bronchiectasis and honeycomb- lung. Progression of disease results in impaired oxygen ing. Honeycombing, while part of the classic appearance transfer and scarring within the lungs (Morgenthau and of UIP, is absent initially. The major HRCT feature in Padilla 2009). It can be idiopathic or associated with other NSIP is ground glass appearance. However ground glass diseases. Many autoimmune diseases such as rheumatoid opacities may be present as the first manifestation of arthritis (RA), systemic sclerosis (SSc), dermatomyosits/ UIP or during acute exacerbations (Morgenthau and polymyositis and anti neutrophil cytoplasmic antibody Padilla 2009). (ANCA) associated vasculitis can result in DPLD. The prognosis depends on many factors like histopath- DPLD associated with autoimmune diseases can have ology, baseline lung function, auto-antibody level, et cetera. either usual interstitial pneumonia (UIP) or non-specific Although rare, there can be an overlap between various interstitial pneumonia (NSIP) pattern in histology. UIP autoimmune conditions, each of them independently con- pattern has worse prognosis compared to NSIP. Also, tributing to pulmonary fibrosis (PF). For example, ANCA positivity is associated with increased incidence of PF in RA patients (Cambridge et al. 1994). Similarly, increased * Correspondence: [email protected] 1Lakeland Rheumatology, 3950 Hollywood Road, 280, St Joseph, MI 49085, incidence of PF is seen in SSc patients with ANCA positiv- USA ity (Derrett-Smith et al. 2013) as well as in SSc-RA overlap Full list of author information is available at the end of the article © 2014 Anand et al.; licensee Springer. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. Anand et al. SpringerPlus 2014, 3:513 Page 2 of 6 http://www.springerplus.com/content/3/1/513 patients (Szücs et al. 2007). As expected, the prognosis Pulmonary function test (PFT) was done which re- of PF in overlap syndromes is worse than individual en- vealed restrictive pattern with forced expiratory volume tities (Cambridge et al. 1994; Derrett-Smith et al. 2013; in 1 second (FEV1) of 1.72 (89% of the predicted) and a Szücs et al. 2007). Also, ANCA positivity in PF can pre- reduced diffusion capacity for Carbon monoxide (cor- dispose to development of ANCA associated vasculitis rected DLCO) of 62%. Transthoracic echocardiogram (AAV) (Arulkumaran et al. 2011). Here, we present a (TTE) demonstrated normal cardiac structure, function case of PF with RA, scleroderma sine scleroderma and ejection fraction except for pulmonary artery pres- (SSS) and AAV which has never been reported before sure of 37 mm of Hg. Open lung biopsy could not be and has a grave prognosis. performed due to high surgical risk of pulmonary com- plications. Biopsy specimen from lungs obtained through Case report bronchoscopy was suggestive of extensive interstitial fi- A 71 year old Caucasian female, with history of chronic brosis with honey combing. cough and dyspnea on supplemental oxygen was re- Patient was started on Hydroxychloroquine, Rituximab ferred to our clinic with complaints of pain in the wrists, and low dose Prednisone at 10 mg per day. Higher doses proximal interphalangial (PIP) joints and metacarpopha- of Prednisone could not be started due to potential risk langial (MCP) joints of hands bilaterally. Associated of scleroderma renal crisis. Leflunomide and Minocy- symptoms included morning stiffness lasting longer than cline were later added to the treatment regimen to man- one hour as well as dysphagia. The cough and dyspnea age RA. Overtime, Prednisone was tapered off as joint had gradual onset and had been progressively worsening symptoms improved along with down trending inflamma- over the past few years. There was no associated fever, tory parameters. Seven months later, she developed nasal chills, sputum production, lower extremity edema, unin- septal perforation along with worsening of inflammatory tentional weight changes, orthopnea or exposure to in- markers. Prednisone and Trimethoprim-Sulfamethoxazole haled occupational irritants. Her past medical history was were started. Otorhinolaryngologist advised against sur- significant for gastroesophageal reflux disease (GERD) and gical correction and recommended conservative man- hypertension for which she was on Omeprazole and Lisi- agement. Subsequently in time, nasal perforation healed nopril respectively. Lisinopril was discontinued and re- without any complications. placed with Amlodipine due to increased cough. However, Currently, her treatment regimen consists of low dose she reported no change in her symptoms. Family history Prednisone (10 mg per day), Hydroxychloroquine, Ritux- of RA (brother, maternal aunt) and lung cancer (father) imab, Leflunomide and Minocycline with which her joint was noted. She is a former smoker who quit smoking symptoms are under good control. She is still dyspneic 37 years ago. At that time, she used to smoke 0.5 pack per and remains on supplemental oxygen without any clin- day for 14 years. Initial investigations did not reveal any ical worsening. significant abnormality other than mildly elevated erythro- cytic sedimentation rate (ESR) of 26 and C-reactive pro- tein (CRP) of 0.9. Discussion On physical examination, coarse crepitations were heard PF is a well known complication in a number of auto- bilaterally in both lung fields, worse at the base. She could immune diseases (Morgenthau and Padilla 2009). The make only 50% of fist on right and 25% of fist on left and prognosis in these patients depends on several factors had a weak hand grip. Squeeze test was positive at the like histology, baseline lung function, presence of auto- MCP and metatarsophalangial (MTP) joints. The PIP joints antibodies and its titer level (Morgenthau and Padilla of hands and MCP joints were tender, warm and swollen. 2009). This is worse in overlap syndromes (Cambridge Elbow extension was decreased by 20 degrees on the left et al. 1994; Derrett-Smith et al. 2013; Szücs et al. 2007). and by 5 degrees on the right. Squatting was incomplete In our patient, there was evidence of more than one due to bilateral knee discomfort. Skin examination noted autoimmune condition. The joint symptoms, seropositiv- several telangiectasias on her face. Nail fold capillaroscopic ity, presence of anti-cyclic citrullinated protein (CCP) examination showed no significant abnormality. antibodies as well as X-ray evidence of erosions in the Results of laboratory work up are detailed in Table 1. joints pointed towards aggressive form of RA. Factors X-ray of feet showed erosive changes at the PIP, MTP which justified the diagnosis of SSS were positive ANA and distal interphalangial (DIP) joints. Periarticular with centromeric pattern with high titer anti-centromere osteopenia and narrowing of the joint space were also antibody, telangiectasia and GERD without obvious cuta- noted at multiple joints in both hands and feet. HRCT neous involvement. In addition, myeloperoxidase (MPO)- chest showed widespread peripheral interstitial fibrotic ANCA positivity manifesting as nasal septal perforation change and mild honeycombing consistent with UIP and worsening of the inflammatory markers led to the (Figure 1 HRCT scan demonstrating PF with UIP). diagnosis of AAV. Since PF in our patient appears to be Anand et al. SpringerPlus 2014, 3:513 Page 3 of 6 http://www.springerplus.com/content/3/1/513
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