We recently reported a rare and clinically significant case of combined melorheostosis and osteopoikilosis presenting with symptomatic sciatic nerve compression in JBJS Case Connect.

A 27-year-old man presented with an 8-year history of chronic right gluteal and posterior thigh pain that was aggravated by sitting and resistant to conservative management. Advanced imaging revealed widespread sclerosing bone changes consistent with melorheostosis and osteopoikilosis involving the right lower extremity, along with two gluteal soft tissue masses causing direct compression of the sciatic nerve.

The patient underwent surgical excision of both masses using a dual surgical approach, resulting in effective sciatic nerve decompression. Postoperatively, he experienced complete pain relief and regained normal sitting and sleeping tolerance, with sustained improvement at 15-month follow-up.

This case highlights:

• The rare coexistence of melorheostosis and osteopoikilosis
• The potential for associated soft tissue lesions to cause compression neuropathy
• The role of surgical management when conservative treatment fails

Although melorheostosis is typically benign and often managed conservatively, this report emphasizes that surgical intervention may be necessary when lesions produce significant neurovascular compression.

📄 Combined Melorheostosis and Osteopoikilosis: Uncommon Presentation with Sciatic Nerve Neuropathy — A Case Report
JBJS Case Connect, January 2026
DOI: 10.2106/JBJS.CC.25.00546

Article links:

https://pubmed.ncbi.nlm.nih.gov/41505508/

https://journals.lww.com/10.2106/JBJS.CC.25.00546

We hope this case contributes to greater awareness of this rare condition and its potential neurological implications.