Chronic Recurrent Multifocal Osteomyelitis of the Hand: A Rare Pediatric Condition

Abstract

Background: Chronic recurrent multifocal osteomyelitis (CRMO) is a rare autoinflammatory condition characterized by bone pain and swelling, secondary to sterile bone osteolytic lesions. Estimated incidence is 1:1 000 000, with ~2% involving the hand. We present a case series of CRMO of the hand and review the available literature, with the aim of educating and raising awareness of this condition. Methods: A retrospective chart review was conducted for all patients presenting to the institution diagnosed with CRMO involving the hands. Subsequently, a literature review was performed. Results/Case Series: Three cases of CRMO of the hand were identified. Age ranged from 20 months to 6 years, with 1 female. Time from presentation to diagnosis ranged from 3 to 15 months. Two had a single lesion, with the remaining patient having a second lesion on full-body magnetic resonance imaging (MRI). Three lesions involved phalanges, and one involved a metacarpal. Two received antibiotics without improvement prior to diagnosis. After diagnosis of CRMO, two received intravenous pamidronate. They both later showed improvement on MRI. Conclusion: CRMO is a rare disease with scarce literature to guide management. We present a series of CRMO specifically of the hand. The cases educate key stakeholders, raise awareness of the diagnosis, and illustrate challenging aspects of managing these patients. This includes the special functional and anatomical consideration essential in managing cases involving the hand.

A Successful Bisphosphonates Monotherapy in Spinal Form of Paediatric Chronic Recurrent Multifocal Osteomyelitis (CRMO)—Case Report

“…Early reports suggest that the bisphosphonates offer promising efficacy in controlling CRMO, as well as safety of use [ 16 , 28 ], though results of recent studies indicated a burden of adverse events (AEs) in pamidronate therapy. In some patients, bisphosphonates were found to worsen bone pain [ 25 ], as well as lead to nausea, headache, fever and phlebitis [ 29 ], which ultimately decrease patients’ compliance [ 30 ]. The presented patient’s therapy was well tolerated and led to no significant AEs.…”

“…As for therapeutic safety, its efficacy was also ambiguously discussed in recent studies. Some reports state that more than 2/3 patients treated with bisphosphonates achieved remission of CRMO symptoms and the progression of their disorder was inhibited, both in spinal and peripheral lesions [ 25 , 26 , 29 , 30 , 31 , 32 , 33 , 34 ]. It was also suggested that pamidronate leads to a rapid improvement in patient’s condition [ 26 ], which is in accordance with the observations of this study—within 1.5 years the reported patient returned to regular height range (50.-75. centile) from the threshold of 10. centile.…”