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

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. 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. 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.

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Publication Details

Journal
Egyptian Journal of Bronchology
Published
2026-02-03
DOI
https://doi.org/10.1186/s43168-026-00525-3
Primary Topic
Vasculitis and related conditions
Type
article
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article

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

K. A. K. Ebraheem, Nawar Albraday, Beshr Abood, Hanean Hamed et al.
Egyptian Journal of Bronchology
Vasculitis and related conditions
article

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

K. A. K. Ebraheem, Nawar Albraday, Beshr Abood, Hanean Hamed, Momina Jalal, Areej Alboudy
article en

Abstract

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. 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. 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.

Egyptian Journal of BronchologyVol. 20(1)
Damascus Hospital (SY), Damascus University (SY)
Openalex Percentile: Top 70%
Vasculitis and related conditions
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