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Background <p>Anti-glomerular basement membrane (anti-GBM) disease is a rare small-vessel vasculitis typically presenting with rapidly progressive glomerulonephritis and/or pulmonary haemorrhage. Environmental and occupational exposures, particularly inhaled hydrocarbons, are recognised triggers, however, cases with initially isolated pulmonary involvement remain diagnostically challenging.</p> Case presentation <p>We report a 32-year-old man with a congenital solitary kidney and no prior renal disease who presented with a three-week history of exertional dyspnoea, dry cough and intermittent haemoptysis. He had daily occupational exposure to petroleum-based solvents for five months without respiratory protection. Initial evaluation revealed bilateral alveolar infiltrates on chest X-ray and diffuse ground-glass opacities consistent with alveolar haemorrhage on high-resolution computed tomography. Laboratory tests showed severe hypochromic microcytic anaemia, markedly elevated inflammatory markers, and acute kidney injury, with urinalysis demonstrating proteinuria, haematuria and casts. Anti-GBM antibodies were strongly positive, while antineutrophil cytoplasmic antibodies were negative. Because of ongoing alveolar haemorrhage and the presence of a single functioning kidney, renal biopsy was deferred and urgent treatment initiated with pulse methylprednisolone, intravenous cyclophosphamide and high-dose oral prednisone. Pulmonary symptoms and inflammatory markers improved rapidly; however, renal function continued to deteriorate and the patient subsequently required chronic haemodialysis.</p> Conclusions <p>This case illustrates an unusual presentation of anti-GBM disease with initially isolated pulmonary manifestations in the context of substantial occupational hydrocarbon exposure and a congenital solitary kidney. It underscores the importance of maintaining a high index of suspicion for anti-GBM disease in patients with unexplained alveolar haemorrhage and relevant environmental exposures, and highlights the need for prompt immunosuppressive therapy even when histological confirmation is not immediately feasible.</p>

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Alveolar haemorrhage–dominant presentation of anti-GBM disease triggered by hydrocarbon exposure in a patient with a congenital solitary kidney: a case report

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  • 发表日期：

    2026年2月3日

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  • Kais Ebraheem,
  • Hanaen Hamed,
  • Momina Jalal,
  • Areej Alboudy,
  • Beshr Abood,
  • Nawar Albraday

摘要

Background

Anti-glomerular basement membrane (anti-GBM) disease is a rare small-vessel vasculitis typically presenting with rapidly progressive glomerulonephritis and/or pulmonary haemorrhage. Environmental and occupational exposures, particularly inhaled hydrocarbons, are recognised triggers, however, cases with initially isolated pulmonary involvement remain diagnostically challenging.

Case presentation

We report a 32-year-old man with a congenital solitary kidney and no prior renal disease who presented with a three-week history of exertional dyspnoea, dry cough and intermittent haemoptysis. He had daily occupational exposure to petroleum-based solvents for five months without respiratory protection. Initial evaluation revealed bilateral alveolar infiltrates on chest X-ray and diffuse ground-glass opacities consistent with alveolar haemorrhage on high-resolution computed tomography. Laboratory tests showed severe hypochromic microcytic anaemia, markedly elevated inflammatory markers, and acute kidney injury, with urinalysis demonstrating proteinuria, haematuria and casts. Anti-GBM antibodies were strongly positive, while antineutrophil cytoplasmic antibodies were negative. Because of ongoing alveolar haemorrhage and the presence of a single functioning kidney, renal biopsy was deferred and urgent treatment initiated with pulse methylprednisolone, intravenous cyclophosphamide and high-dose oral prednisone. Pulmonary symptoms and inflammatory markers improved rapidly; however, renal function continued to deteriorate and the patient subsequently required chronic haemodialysis.

Conclusions

This case illustrates an unusual presentation of anti-GBM disease with initially isolated pulmonary manifestations in the context of substantial occupational hydrocarbon exposure and a congenital solitary kidney. It underscores the importance of maintaining a high index of suspicion for anti-GBM disease in patients with unexplained alveolar haemorrhage and relevant environmental exposures, and highlights the need for prompt immunosuppressive therapy even when histological confirmation is not immediately feasible.