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Case based discussions
Case-based discussion from the Royal Devon and Exeter NHS Foundation Trust: a painful paradox
  1. Charles Sharp1,
  2. Manish Powari2,
  3. Ravik Mascarenhas3,
  4. Alexander Spiers4,
  5. Peter O Froeschle5,
  6. Bipen D Patel6,
  7. David A Silver4,
  8. Toby M Maher7,
  9. Michael A Gibbons6
  1. 1Department of Respiratory Medicine, North Bristol NHS Trust, Bristol, UK
  2. 2Histopathology Department, Royal Devon and Exeter NHS Foundation Trust, Exeter, UK
  3. 3Rheumatology Department, Royal Devon and Exeter NHS Foundation Trust, Exeter, UK
  4. 4Radiology Department, Royal Devon and Exeter NHS Foundation Trust, Exeter, UK
  5. 5Department of Thoracic Surgery, Royal Devon and Exeter NHS Foundation Trust, Exeter, UK
  6. 6Respiratory Department, Royal Devon and Exeter NHS Foundation Trust, Exeter, UK
  7. 7Interstitial Lung Disease Unit, Royal Brompton, London, UK
  1. Correspondence to Dr Charles Sharp, Department of Respiratory Medicine, North Bristol NHS Trust, Southmead Road, Westbury on Trym, Bristol BS10 5NB, UK;{at}

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Tumour necrosis factor (TNF)-α inhibitors, despite being an effective treatment for granulomatous diseases such as sarcoidosis and Crohn's, are recognised to trigger a paradoxical granulomatous response.1 ,2 Reported cases have predominantly described lymph node, pulmonary and skin manifestations. We present a case of a granulomatous thoracic spine lesion, leading to atraumatic fracture, arising as a consequence of etanarcept treatment of connective tissue disease with associated interstitial lung disease (ILD).

Dr C Sharp (StR)

A 50-year-old woman, a retired hairdresser, with rheumatoid arthritis/Jo-1 overlap syndrome, diagnosed 14 years earlier, and with associated but hitherto stable ILD, was referred to the ILD service with progressive dyspnoea and deteriorating lung function. Other relevant medical history included febrile neutrophilic dermatosis. The patient reported smoking 10–20 cigarettes per day, but had had no occupational or animal exposures and no past exposure to tuberculosis. At presentation, there was marked joint destruction though no active synovitis, and systemic symptoms were well controlled with the anti-TNFα monoclonal antibody etanercept which had been commenced in 2006. Prior to commencement of the etanercept, a Heaf test had been negative. There was no history of significant corticosteroid usage. Respiratory examination was remarkable only for fine bi-basal inspiratory crackles.

Pulmonary function tests in January 2006 demonstrated a forced expiratory volume in 1 s (FEV1) of 2.62 L (95% predicted), forced vital capacity (FVC) 3.27 L (105% predicted), with diffusion coefficient for carbon monoxide (DLCO) 5.08 mL CO/Min/mm Hg (60% predicted). At presentation to the ILD service in May 2010, the values had fallen to FEV1 2.02 L (78%), FVC 2.53 L (86%) and DLco 3.73 (44%).

Autoimmune profile demonstrated a positive rheumatoid factor (99 IU/mL), a positive antinuclear antibody and the presence of anti-Ro, anti-La and anti-Jo-1 antibodies. A high-resolution (HR) CT scan of the thorax and an echocardiogram were arranged.

Dr A Spiers (Consultant Thoracic …

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  • Contributors The article was conceived by MAG and CS and written by MAG, CS and TMM. All authors contributed to the care of the patient and approved the final text.

  • Competing interests None.

  • Patient consent Obtained.

  • Provenance and peer review Not commissioned; internally peer reviewed.