IgG4-Related Disease Strikes the Cervical Spine: First Description of a Rare Cause for C1 Destruction and Tetraparetic Stenosis

Background and Clinical Significance: Immunoglobulin G4-related disease (IgG4-RD) is a systemic immune-mediated fibroinflammatory disorder that can mimic infection or malignancy. Spinal involvement is exceedingly rare and usually limited to pachymeningitis or epidural pseudotumors. True vertebral bone destruction has been reported only sporadically. Case Presentation: A 54-year-old man presented to our emergency department with severe neck pain after a fall. CT and MRI revealed extensive osteolysis of the C1 posterior arch and odontoid process with atlantoaxial subluxation. Following a second inpatient fall, he developed acute tetraparesis. Emergency posterior occipitocervical fusion (C0–C4) with C1–C2 laminectomy and foramen magnum decompression was performed. Histopathology demonstrated dense lymphoplasmacytic infiltration and fibrosis with up to 36 IgG4+ plasma cells per high-power field and an IgG4+/IgG ratio > 40%, confirming IgG4-RD. The patient recovered substantial motor function postoperatively and regained independent ambulation after neurological rehabilitation. Conclusions: IgG4-RD can rarely present as destructive craniovertebral osteolysis with neurological compromise. Unexplained C1–C2 osteolytic lesions should prompt evaluation for IgG4-RD, a rare but treatable cause of cervical instability.

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Journal
Reports — Medical Cases Images and Videos
Published
2026-03-26
DOI
https://doi.org/10.3390/reports9020097
Primary Topic
IgG4-Related and Inflammatory Diseases
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article
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article

IgG4-Related Disease Strikes the Cervical Spine: First Description of a Rare Cause for C1 Destruction and Tetraparetic Stenosis

Joe Mehanna, Christine Schülin, Sascha Gravius, Sven Schulz et al.
Reports — Medical Cases Images and Videos
IgG4-Related and Inflammatory Diseases
article

IgG4-Related Disease Strikes the Cervical Spine: First Description of a Rare Cause for C1 Destruction and Tetraparetic Stenosis

Joe Mehanna, Christine Schülin, Sascha Gravius, Sven Schulz, Frederic Bludau, Franz-Joseph Dally
article en

Abstract

Background and Clinical Significance: Immunoglobulin G4-related disease (IgG4-RD) is a systemic immune-mediated fibroinflammatory disorder that can mimic infection or malignancy. Spinal involvement is exceedingly rare and usually limited to pachymeningitis or epidural pseudotumors. True vertebral bone destruction has been reported only sporadically. Case Presentation: A 54-year-old man presented to our emergency department with severe neck pain after a fall. CT and MRI revealed extensive osteolysis of the C1 posterior arch and odontoid process with atlantoaxial subluxation. Following a second inpatient fall, he developed acute tetraparesis. Emergency posterior occipitocervical fusion (C0–C4) with C1–C2 laminectomy and foramen magnum decompression was performed. Histopathology demonstrated dense lymphoplasmacytic infiltration and fibrosis with up to 36 IgG4+ plasma cells per high-power field and an IgG4+/IgG ratio > 40%, confirming IgG4-RD. The patient recovered substantial motor function postoperatively and regained independent ambulation after neurological rehabilitation. Conclusions: IgG4-RD can rarely present as destructive craniovertebral osteolysis with neurological compromise. Unexplained C1–C2 osteolytic lesions should prompt evaluation for IgG4-RD, a rare but treatable cause of cervical instability.

Reports — Medical Cases Images and VideosVol. 9(2)
Heidelberg University (DE), University Hospital Heidelberg (DE), Kliniken Schmieder (DE), University Medical Centre Mannheim (DE)
Good health and well-being
Openalex Percentile: Top 5%
IgG4-Related and Inflammatory Diseases
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