Evidence map›Paper›PMID 41938408›Full record

ArticleInternational journal of surgery case reports2026

A case report and literature review of pediatric multifocal melorheostosis in a unilateral limb.

Xia Na, Yu Fuyong, Tang Xiushu, Wang Lijun, Yu Lingjia, Tang Kai, Xu Yisheng

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In one paragraph

Article in International journal of surgery case reports, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

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1citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

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3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

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4 · The record

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5 · Who and what money

Authors and funding

7 authors.

Xia NaQianxinan Autonomous Prefecture Hospital of TCM, Guizhou, China.
Yu FuyongQianxinan Autonomous Prefecture Hospital of TCM, Guizhou, China.
Tang XiushuQianxinan Autonomous Prefecture Hospital of TCM, Guizhou, China.
Wang LijunGuizhou Medical University, Guizhou, China.
Yu LingjiaDepartment of Orthopedics, Beijing Friendship Hospital, Capital Medical University, Beijing, China.
Tang KaiFaculty of Chinese Medicine/State Key Laboratory of Quality Research in Chinese Medicine, Macau University Science & Technology, Macao, China.
Xu YishengQianxinan Autonomous Prefecture Hospital of TCM, Guizhou, China.ORCID https://orcid.org/0000-0001-5328-0446

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Introduction and importance: Melorheostosis (Léri-Joanny syndrome) is a rare, nonhereditary, congenital sclerotic bone dysplasia with an estimated prevalence of 0.9 per million. Its enigmatic etiology and distinctive "melting wax" radiological appearance warrant detailed case reporting to enhance understanding of diagnosis and management, particularly in pediatric populations. Case presentation: A 9-year-old male presented with incidental right upper limb bone abnormalities discovered post-trauma. Asymptomatic without pain/swelling, he had no relevant family history. Laboratory tests (CBC, CRP, ESR) were normal. Imaging revealed unilateral cortical sclerosis with increased density in the right scapular coracoid process, humeral head, distal humerus, ulnar head, wrist, and metacarpals, exhibiting a pathognomonic "melting wax drip" pattern. Histopathology confirmed irregular endosteal bone proliferation, densely arranged lamellar bone, and enlarged/distorted Haversian canals . Clinical discussion: Melorheostosis predominantly affects limbs unilaterally; multifocal involvement (as here) is uncommon. Radiography remains the diagnostic cornerstone, supplemented by CT/MRI for spatial extent. This case was classified as endosteal subtype. Management is symptom-directed: asymptomatic cases require monitoring only. Surgical correction (e.g., osteotomy) is reserved for severe pain/deformity refractory to analgesics/physiotherapy. Long-term follow-up is critical due to theoretical malignant transformation concerns, though unreported clinically. Conclusion: This pediatric case underscores melorheostosis' incidental presentation and diagnostic reliance on classic imaging. The patient remained asymptomatic over 6 months, obviating intervention. Longitudinal surveillance is planned to elucidate natural history, contributing to management paradigms for this rare entity.

Indexed as

bone abnormalitiescase reportLéri-Joanny syndromemelorheostosis

Identifiers

PMID41938408
PMCPMC13046020

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.