Background

This case demonstrates the importance of considering complications in immunocompromised patients, even if young with no such prior history. It is also essential to regularly reassess the situation clinically, even if the symptoms remain consistent. Although case reports of sepsis are well recognised with use of infliximab, this is the first case report documenting such serious complications as a result of an apparently small and innocuous infectious complication contributed to, at least in part, by adalimumab therapy.

Case presentation

An 18-year-old man with a history of Crohn’s disease mainly affecting the colon was admitted for management of his difficult to control Crohn’s disease. He had been diagnosed 3 years previously, and had experienced numerous flare-ups requiring steroid treatment, despite immunosuppression trials with methotrexate and currently azathioprine.

His only past medical history was of acne vulgaris. He was a non-smoker and drank alcohol rarely. His brother also had Crohn’s disease that was difficult to manage, requiring treatment with infliximab.

On this occasion, he had been admitted for further symptom control of his persistent bloody diarrhoea and abdominal pains. He underwent a colonoscopy revealing extensive colonic ulceration. He was started on intravenous steroids in addition to his mesalazine, azathioprine, ciprofloxacin and low molecular weight heparin (LMWH), with partial symptom relief. The anti-tumour necrosis factor (anti-TNF) drug adalimumab was also instituted due to ongoing disease activity.

During his stay the patient developed left sided pleuritic chest pain worsened by deep inspiration. A ventilation/perfusion (V/Q) scan had a low probability for pulmonary embolus; a computed tomography pulmonary angiogram (CTPA) confirmed this, but revealed evidence of a small wedge shaped area of consolidation peripherally in the left lung (fig 1). He was kept on his current antibiotic regimen. Throughout his stay, the patient experienced the same degree of pain.

• Download figure
• Open in new tab
• Download powerpoint

Figure 1

Small cavitating peripheral pneumonia

Ten days later the patient experienced worsening of his chest pain. He had a mild cough which produced green sputum. He denied shortness of breath. His saturation was 98% on room air, his respiratory rate was 16/min, and he had notably diminished breath sounds on the left lung base. A chest x-ray was arranged which showed evidence of a large hydro-pneumothorax (fig 2).

• Download figure
• Open in new tab
• Download powerpoint

Figure 2

Chest x-ray revealing pneumothorax.

Differential diagnosis

Differential diagnoses included pulmonary tuberculosis or other opportunistic/fungal infection. Pulmonary involvement with Crohn’s disease was also considered.

Treatment

Initially a chest drain was inserted, leading to good lung re-inflation. The patient was then started on a 2 week course of intravenous tazocin in addition to intravenous teicoplanin once sputum culture results showed infection with Staphylococcus aureus. The steroids were weaned rapidly and he was discharged home on low dose oral budesonide.

Outcome and follow-up

The patient made a good recovery with improvement of the chest pain once pulmonary re-inflation was achieved (fig 3). The infection responded to the intravenous antibiotics and the consolidation slowly resolved. Despite significant improvement in chest pain he still occasionally experiences intermittent left sided chest discomfort, but the chest x-rays are normal. The patient is due to undergo a sub-total colectomy in the near future.

• Download figure
• Open in new tab
• Download powerpoint

Figure 3

Post-pneumothorax ongoing pneumonia

Discussion

Cavitating pneumonia is a recognised cause for the development of a pneumothorax. If peripheral, it could potentially lead to the development of a broncho-pleural fistula with consequent formation of an air leak and pneumothorax. In the context of using anti-TNF therapies, there have been few case reports of developing complicated pulmonary disease. The use of such agents, particularly in combination with other immunosuppressive agents, can to lead to the development of opportunistic or severe infections. This can consequently lead to more complicated disease as occurred in this case. A German study identified a threefold increase in the rate of infections in patients taking anti-TNF therapy for rheumatoid arthritis.1 In inflammatory bowel disease (IBD) patients, the risk of serious infection was increased threefold if the patients were on steroids or thiopurines, and fourfold if they were on infliximab.2 The combination of the two increases the risk ninefold. However, organisms often implicated in such cases, such as Pneumocystis carnii, Nocardia or Candida, are usually opportunistic. It is worth noting that Sandborn et al reported no severe infectious complications in their series of 276 patients using adalimumab for IBD.3 A recent case report presented a severe and ultimately fatal case of invasive pulmonary aspergillosis.4 As far as can be determined from the literature, our patient is the first reported case of pneumothorax developing due to cavitating pneumonia following adalimumab treatment. There was, however, no evidence of fungal infection in this case.