This is a case of an 11-year-old boy with left clavicle pain with two months of evolution presented to emergency department. No history of trauma, fever or systemic signs, recent illness and no relevant personal or familiar medical history. No neurological or vascular abnormalities were found in the upper arms. Left shoulder X-ray revealed a hyperdense lesion and increased thickness of left clavicle (Figure 1). In this context a computed tomography (CT) scan and after a magnetic resonance imaging (MRI) scan were ordered. Both scans demonstrated morphological changes of the median half of the clavicle, cortical destruction and periosteal reaction. Also changes in the surrounding soft and muscular tissue and bulky and numerous locoregional ganglion formations (Figure 2) and (Figure 3). Blood analysis, blood cultures and bone biopsy were negative for infection and neoplastic disorder. In this context, by exclusion, and in the presence of two Jansson major diagnostic criteria (radiologically proven osteolytic/osteosclerotic bone lesions and sterile bone biopsy with signs of inflammation) [1], we reached the diagnosis of chronic recurrent multifocal osteomyelitis (CMRO) (Table 1). Patient was treated with NSAIDs for six months and a short course of corticoids without any response. By keeping complaints of marked and recalcitrant pain in clavicle region, we opted for the surgical treatment: resection of the lesion en bloc, about 7 cm of clavicle (Figure 4), maintaining the integrity of periosteum to allow neo-osteogenesis of the clavicle. No bone grafting or another material was interposed in the dead space left by the resection of the clavicle. Post operatory X-ray, showing a radiopaque area corresponding to the excision of the medial portion of the clavicle (Figure 5).

At third month after surgery, there is an image compatible with a neo-clavicle and patient is asymptomatic (Figure 6).

Chronic recurrent multifocal osteomyelitis is a non-bacterial bone inflammation (osteitis), that has symptoms similar to the conventional osteomyelitis, but without infection. It is characterized by multifocal bone lesions with pain and swelling recurring over months to years, accounting of 2 to 5% of all cases of osteomyelitis [2] [3] mostly children and adolescents. The differential diagnosis includes bacterial osteomyelitis, Ewing sarcoma, leukemia, lymphoma, rhabdomyosarcoma, neuroblastoma metastasis, eosinophilic granuloma or Langerhans cell histiocytosis. The tubular long bones, pelvis, hip, sternum and mandible are the most affected bones [4] [5]. The CRMO, in pediatric patients , may be associated with various skin disorders: Pustulosis palmoplantar is syndrome [6] [7] [8], diffuse pustulosis, psoriasis vulgaris, acne [9] [10] [11], Sweet's syndrome and pyoderma gangrenosum [9]. Multiple sites of apparent osteomyelitis with negative pathology and cultures and no response to antibiotherapy characterize it [12]. NSAIDs as naproxen, indomethacin or aspirin are the initial therapy. A short course of corticosteroids can be used in refractory cases. Other treatments such as sulfasalazine or methotrexate [6], bisphosphonates [13] and hyperbaric chamber [14] are also indicated in refractory cases. Antibiotics do not seem to be helpful. Although surgical treatment has been used, its role is not yet clearly defined. We opted for a surgical excision of the clavicle, because of the constant and intense pain with no response to six months of conservative treatment. We think that the good response to surgery makes this case exceptionally unusual. The prognosis for these patients is however good. In one study, 17 of 23 patients had complete resolution of the clinical findings, at an average of 5.6 years after diagnosis. Six patients continue to have active disease, and the other six had intermittent relapses or chronic pain. 78% had no physical impairment [2], but further prospective studies are needed to determine the optimum outcome measures and treatment strategy [15],

The absence of positive microbiologic results, the clinical course and the presence of reactivating and remitting lesions over time suggest the diagnosis of chronic recurrent multifocal osteomyelitis. As the disease is self-limiting, knowledge of its characteristics appearance can lead to the most appropriated treatment, and can prevent overly aggressive medical and surgical evaluation and treatment.

1. Jansson A, Renner ED, Ramser J, et al. Classification of non-bacterial osteitis: retrospective study of clinical, immunological and genetic aspects in 89 patients. Rheumatology (Oxford) 2007 Jan;46(1):154–60.   [CrossRef]   [Pubmed]    
2. Huber AM, Lam PY, Duffy CM, et al. Chronic recurrent multifocal osteomyelitis: Clinical outcomes after more than five years of follow-up. J Pediatr 2002 Aug;141(2):198–203.   [CrossRef]   [Pubmed]    
3. McKenna Vozzelli K, Butman L. Osteomyelitis and suppurative arthritis. In: Gershon. Krugman's Infectious Diseases of Children. St. Louis: Mosby Inc 2004:290–301.    
4. Dabov G. Osteomyelitis. In: Canale T. Cambell\'s operative orthopaedics. St. Louis: Mosby Inc 2003:661–84.    
5. Carr AJ, Cole WG, Roberton DM, Chow CW. Chronic multifocal osteomyelitis. J Bone Joint Surg Br 1993 Jul;75(4):582–91.   [Pubmed]    
6. Job-Deslandre C, Krebs S, Kahan A. Chronic recurrent multifocal osteomyelitis: Five-years outcomes in 14 patients cases. Joint Bone Spine 2001 May;68(3):245–1.   [CrossRef]   [Pubmed]    
7. Schultz C, Holterhus PM, Seidel A, et al. Chronic recurrent multifocal osteomyelitis in children. Pediatr Infect Dis J 1999 Nov;18(11):1008–3.   [CrossRef]   [Pubmed]    
8. Bjorkstén B, Gustavson KH, Eriksson B, Lindholm A, Nordstrom S. Chronic recurrent multifocal osteomyelitis and pustulosis palmoplantaris. J Pediatr 1978 Aug;93(2):227–31.   [Pubmed]    
9. Beretta-Piccoli BC, Sauvain MJ, Gal I, et al. Synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome in childhood: A report of ten cases and review of the literature. Eur J Pediatr 2000 Aug;159(8):594–601.   [CrossRef]   [Pubmed]    
10. Jana H, Pavel K. The SAPHO syndrome in a 16-year-old boy: Coincidence of acne conglobata and osteoarthritis. J Adolesc Health 1993 Mar;14(2):120–3.   [CrossRef]   [Pubmed]    
11. King SM, Laxer RM, Manson D, Gold R. Chronic recurrent multifocal osteomyelitis: A noninfectious inflammatory process. Pediatr Infect Dis J 1987 Oct;6(10):907–11.   [CrossRef]   [Pubmed]    
12. Kotilainen P, Merilahti-Palo R, Lehtonen OP, et al. Propionibacterium acnes isolated from sternal osteitis in a patient with SAPHO syndrome. J Rheumatol 1996 Jul;23(7):1302–4.   [Pubmed]    
13. Akikusa JD, Zacharin M, Shugg AW, Melbourne RCA, Tasmania VC. Bisphosphonates in the treatment of chronic recurrent multifocal osteomyelitis. Arthritis Rheum 2001 44 Suppl 9:171.    
14. van Merkesteyn JP, Groot RH, Bras J, McCarroll RS, Bakker DJ. Diffuse sclerosing osteomyelitis of the mandible: A new concept of its etiology. Oral Surg Oral Med Oral Pathol 1990 Oct;70(4):414–9.   [Pubmed]    
15. Batu E, Gulhan B, Topaloglu R, Ozen S. A proposed treatment scheme for chronic recurrent multifocal osteomyelitis (CRMO): A case series of nine patients. Pediatric Rheumatology 2014;12:239.   [CrossRef]