Chronic recurrent multifocal osteomyelitis beginning with a solitary lesion of the ilium

Zhang, Ping; Jia, Xiao Ying; Zhang, Yun; Morelli, John N.; Zhang, Ze Kun

doi:10.1186/s12891-017-1611-4

Cited by 3 publications

(6 citation statements)

References 26 publications

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“…Metaphyseal involvement without periosteal reaction is the typical presentation of CRMO. 6 Significant periosteal reaction and adjacent soft tissue component, as in our case, delayed the diagnosis as the initial impression was either Ewing’s sarcoma or Langerhans cell Histiocytosis. Osteomyelitis was low in clinical suspicion because of the absence of systemic findings, leucocytosis and negative blood culture.…”

Section: Discussionmentioning

confidence: 63%

“…Chronic recurrent multifocal osteomyelitis is an idiopathic inflammatory disease, mostly prevalent in children and adolescents, that results in episodic bone pain. 3 , 4 , 5 , 6 It is an uncommon condition with an estimated prevalence of one in a million. 6 Interestingly, for some unknown reasons, literature indicates that up to 85% of the affected are females.…”

Section: Discussionmentioning

confidence: 99%

“… 3 , 4 , 5 , 6 It is an uncommon condition with an estimated prevalence of one in a million. 6 Interestingly, for some unknown reasons, literature indicates that up to 85% of the affected are females. 6 Chronic recurrent multifocal osteomyelitis is also known as chronic non-bacterial osteomyelitis as no pathogens grow on blood culture and antibiotics do not relieve the symptoms.…”

Section: Discussionmentioning

confidence: 99%

“… 6 Chronic recurrent multifocal osteomyelitis is also known as chronic non-bacterial osteomyelitis as no pathogens grow on blood culture and antibiotics do not relieve the symptoms. 6 , 7 The aetiology remains elusive but has been associated with many dermatological and autoimmune disorders such as psoriasis, Takayasu’s arteritis, Crohn’s disease, ulcerative colitis, SAPHO (synovitis, acne, pustulosis, hyperostosis, osteitis) syndrome and some genetic disorders. 8 , 9 , 10 Clinically, patients present with non-specific symptoms of pain, swelling and limited range of motion.…”

Section: Discussionmentioning

confidence: 99%

“…Involvement of the ilium as a potential site for CRMO has been reported infrequently. 2 , 6 , 12 , 13 Chronic recurrent multifocal osteomyelitis of the pelvic bone itself is rare and the site of predilection in pelvis includes metaphyseal equivalents such as the ischiopubic synchondrosis and sacroiliac joints. 2 , 14 , 15 Pelvic CRMO can rarely manifest as sclerosis of the iliac wings.…”

Section: Discussionmentioning

confidence: 99%

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Rare presentation of chronic recurrent multifocal osteomyelitis of the Iliac wing mimicking Ewing’s sarcoma

Nepal

Alam

Sajid

et al. 2021

South African Journal of Radiology

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This report describes a case of chronic recurrent multifocal osteomyelitis (CRMO) in an 11-year-old girl, involving the iliac bone as an initial, solitary site. Atypical imaging features were suspicious of a bone tumour, such as Ewing’s sarcoma. Chronic recurrent multifocal osteomyelitis is a great masquerader and can present atypically. Radiologists should be familiar with both typical and atypical presentations, to determine an accurate diagnosis and guide appropriate management. Timely diagnosis may avoid invasive bone biopsy and inappropriate long-term antibiotic prescription for children.

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Section: Discussionmentioning

confidence: 63%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

See 3 more Smart Citations

Rare presentation of chronic recurrent multifocal osteomyelitis of the Iliac wing mimicking Ewing’s sarcoma

Nepal

Alam

Sajid

et al. 2021

South African Journal of Radiology

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Bell’s palsy with facial bone involvement: A rare presentation of chronic nonbacterial osteomyelitis with literature review

Ailumerab

Aguiar

2022

Modern Rheumatology Case Reports

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Chronic nonbacterial osteomyelitis (CNO) is a chronic, sterile, inflammatory disease. It primarily presents with nonspecific bone pain and swelling, but ultimately can cause bone destruction and deformities, if left untreated. The involvement of the cranial bones (apart from the mandible) is rare in CNO. In this report, we present a rare case of CNO affecting facial and cranial bones presenting as facial palsy with a review of the literature about similar affection. Summary of the case: A 10-year-old, previously healthy female was initially evaluated for swelling of the left side of her face with slight tenderness on palpation, but no fever. Her complete blood count was unremarkable, her inflammatory markers were elevated (CRP 7.5 mg/dl and ESR 104 mm/hr), and CT/MRI of the face and brain showed a destructive osseous process involving the left maxillary, zygomatic, sphenoid bones, and the clivus (figure 1:a & b). Bone biopsy of the left maxilla showed fibrous dysplasia with abscess formation, most consistent with an infectious etiology (acute osteomyelitis). She was started on oral Clindamycin for a 3 months course. The facial swelling improved after starting Clindamycin, but on her 6th week of treatment, she developed right-sided Bell’s palsy. An MRI of the brain showed hyperenhancement of the right 7th cranial nerve (figure 1:c). A month later, she was evaluated for right wrist and knee swelling, pain, and limitation of movement. Skeletal survey and MRI showed multifocal lesions with mixed sclerosis and lucency. Her inflammatory markers continued to be elevated. Another bone biopsy of the right radius showed similar findings of destruction with no evidence of malignancy. She was ultimately diagnosed with CNO. She was started on non-steroidal anti-inflammatory drugs (NSAIDs) with gastric protection and regular follow up. Over more than a year of follow-up, the patient’s inflammatory markers remain normal and joint swelling/limitation have remained in remission. Results: We found 5 additional cases (supplemental table 1) presented with similar presentation. To our knowledge, our patient is the first reported case in the US involving the cranial/facial bones apart from the mandible presenting with facial palsy. Conclusions: The affection of the facial bones (apart from the mandible) in CNO is very rare, but awareness of such presentation by the clinician is an important aspect of reaching the diagnosis.

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Clinical features and diagnostic approaches to chronic recurrent multifocal osteomyelitis: A retrospective study

Huang,

Zhu,

Zhao

et al. 2023

Preprint

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Background: Chronic recurrent multifocal osteomyelitis (CRMO) is an autoinflammatory bone disease which is difficult to diagnose in the early stages. The aim of this study was to summarize the methods of diagnosis and differential diagnosis of CRMO, study diagnostic methods for early identification and diagnosis of the disease, and improve the ability of clinicians to identify and diagnose the disease at an early stage. Methods: All children diagnosed with CRMO at our hospital between 2013 and 2022 were included. Clinical characteristics, imaging findings, and reasons for delayed diagnoses were summarized. In addition, the literature on CRMO from database establishment to February 1, 2023, was retrieved through Embase, Medline, and PubMed, and the management of CRMO was proposed based on the literature. Results: There were seven patients with CRMO (six males and one female). Mean age at first symptom: 6.3 (IQR 2.3-8.2) years; mean delay in diagnosis (from symptoms): 2.1 (IQR 1.2-2.8) years; mean age at relapse: 8.1 (IQR 3.2-12.1) years; mean disease duration: 3.8 (IQR 2.4-3.9) years. All children had local swelling and bone pain, and three patients had pathological fractures (one in the femur and two in the tibia). A total of 34 anatomical sites were involved in the lesions in seven children; one case had a single lesion, and six had multiple lesions. The blood and puncture tissue bacterial culture results in seven cases were negative, and the bone marrow cytology examination was normal. A total of 119 clinical research reports on CRMO (1202 patients) were included, and it was found that the first symptom in 1142 cases (approximately 95%) was bone pain or local swelling. In all cases in which bacterial cultures of peripheral blood or bone tissue were performed, the results were negative. In addition, bone marrow cytology results were negative in 1178 (98%). Conclusions: This study systematically summarized the clinical characteristics of patients with CRMO in our center and those reported in the literature. In addition, this study proposes primary and secondary diagnostic criteria for the management of CRMO, which requires verification.

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Chronic recurrent multifocal osteomyelitis beginning with a solitary lesion of the ilium

Cited by 3 publications

References 26 publications

Rare presentation of chronic recurrent multifocal osteomyelitis of the Iliac wing mimicking Ewing’s sarcoma

Rare presentation of chronic recurrent multifocal osteomyelitis of the Iliac wing mimicking Ewing’s sarcoma

Bell’s palsy with facial bone involvement: A rare presentation of chronic nonbacterial osteomyelitis with literature review

Clinical features and diagnostic approaches to chronic recurrent multifocal osteomyelitis: A retrospective study

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