Chronic recurrent multifocal osteomyelitis - case report of two patients and review of literature

Abstract

Background Chronic Recurrent Multifocal Osteomyelitis (CRMO) is an autoinflammatory skeletal disease characterized by unifocal or multifocal nonbacterial inflammatory bone lesions in the metaphysis of long bones. Common sites of CRMO are tibia, pelvis, proximal femur, clavicle, calcaneum and vertebrae. However, unifocal presentation or presentation in adults, atypical locations, and absence of recurrence have also been reported. Methods and Findings We describe two cases of female patients with unifocal presentation of nonbacterial inflammatory bone lesions in the upper and lower extremity. Furthermore, a review of literature is presented. Conclusion Although CRMO is seen primarily in children and adolescents, and usually occurs in a multifocal pattern, our cases illustrate that this disease can arise in adult patients and in a single location. The diagnosis of CRMO should be made in a multi-disciplinary approach amongst orthopedic surgeons, radiologists and pathologists.

Mimics of Erdheim–Chester disease

“…In addition to bone involvement, it is associated with a diverse range of clinical manifestations including myalgia, mucocutaneous lesions and gastrointestinal symptoms 20 . Extra‐osseous manifestation in adults is rare and has been reported in up to 6% of all cases 20,21 . Among the clinical features at presentation, pain is a consistent symptom and is usually associated with constitutional symptoms similar to our patient's clinical presentation.…”

“…20 Extra-osseous manifestation in adults is rare and has been reported in up to 6% of all cases. 20,21 Among the clinical features at presentation, pain is a consistent symptom and is usually associated with constitutional symptoms similar to our patient's clinical presentation. Histologic features are usually non-specific and reveal acute as well as chronic inflammation with reparative bone features such as hyperostosis.…”

Clinical and Genetic Features of Chinese Adult Patients With Chronic Non-Bacterial Osteomyelitis: A Single Center Report

“…CNO usually begins in childhood, but adult-onset patients have also been reported ( 16 – 21 ). Two-thirds of the patients in our study were adult-onset, which might be related to the fact that our center mainly admitted adult patients.…”

First Reported Case of Majeed Syndrome From Pakistan

“…Ferguson et al, attributed the condition to homozygous mutation in LPIN. 2 The gene was mapped to the short arm of chromosome 18. LPIN2 was found to be expressed in all body tissues in this study on 6 individuals.…”

“…So far, reported extraskeletal features include Congenital Dyserythropoeitic Anemia (CDA) with a microcytic and hypochromic picture, neutrophilic dermatosis (sweet syndrome), psoariasis, severe acne, crohns disease, ulcerative colitis, palmoplantar pustulosis, granulomatous polyangitis, takayasu arteritis. 2 Early and correct recognition of this syndrome will lead to timely initiation of proper treatment, prevention of futile therapies due to misdiagnosis and reduction in long-term morbidity. Here, we report a case of 9-year-old boy that underwent a below-knee-amputation (BKA) due to suspicion of infective etiology for the non-resolving osteomyelitis.…”