Enteropathic SAPHO Syndrome in Ulcerative Colitis Responsive to Bisphosphonates

Area of Science:

• Rheumatology
• Gastroenterology
• Dermatology

Background:

• SAPHO (Synovitis, Acne, Pustulosis, Hyperostosis, Osteitis) syndrome is a rare inflammatory disorder affecting bone, joints, and skin.
• While associations with inflammatory bowel disease (IBD), particularly Crohn's disease, are documented, its link with ulcerative colitis (UC) is rarely reported.
• The clinical heterogeneity of SAPHO syndrome and overlap with IBD's extra-intestinal manifestations can lead to underdiagnosis.

Observation:

• A case of SAPHO syndrome in a patient with UC is presented.
• Imaging revealed sternocostal involvement, including rib enlargement and hyperostosis.
• Bone biopsy confirmed reactive bone changes without infection or malignancy.

Findings:

• The patient with UC and SAPHO syndrome experienced complete symptom resolution after 4 months of zoledronic acid treatment.
• Zoledronic acid was well-tolerated, with no significant adverse events reported.
• This case highlights a successful therapeutic approach for a rare SAPHO syndrome presentation in UC.

Implications:

• This case provides valuable data on the diagnosis and management of SAPHO syndrome in UC patients.
• Increased awareness and diagnostic vigilance are crucial for recognizing SAPHO syndrome in IBD patients.
• The findings suggest zoledronic acid as a potential treatment option for SAPHO syndrome associated with UC.