Multifocal Recurrent Periostitis Responsive to Colchicine

Abstract

A brother and sister with multifocal recurrent periostitis are presented. Their disease started at an early age and manifested itself as an episodic migrating arthropathy. At roentgenography, reversible solid periosteal reactions were visible along large tubular bones. Scintigraphic and histological investigations revealed a sterile osteitis and thickened periosteum, but there was no indication of a viral infection. The girl experienced spontaneous amelioration after puberty; the boy improved markedly on colchicine.

The syndrome of chronic recurrent multifocal osteomyelitis and congenital dyserythropoietic anaemia. Report of a new family and a review

“…However, the age at onset in our two present patients was much earlier (3±8 weeks) and is similar to the four patients of the family reported by us earlier [19] (Table4). It is interesting that the two patients (brother and sister) with CRMO described by Festen et al [9] also had an early onset ranging between 1±2 years. Furthermore, the clinical course of CRMO in our patients seems to be far more aggressive (1±2 relapses/month) than that in reports describing the sporadic form.…”

“…None of our patients had a long remission. The girl described by Festen et al [9] had CRMO for 23 years, although she had spontaneous amelioration after puberty. However, two of the seven patients with the sporadic form of CRMO described by Leisure et al [16] had spontaneous remission after a short follow-up period of 15 and 27 months.…”

“…However, in the presence of a protracted course in some patients with CRMO, we believe that the adverse eects of long-term corticosteroid therapy, including growth retardation, are potentially dangerous. Festen et al [9] reported a remarkable improvement in one of their two patients on 1 mg daily colchicine therapy. This is not in agreement with our experience; we tried 1.5±2 mg daily colchicine therapy for 4 months in our ®rst three patients, initially diagnosed as familial Mediterranean fever, with no response (unpublished observation).…”

A missense mutation in pstpip2 is associated with the murine autoinflammatory disorder chronic multifocal osteomyelitis

“…There are reports of affected siblings (with unaffected parents), concordance in monozygotic twins and a report of child with CRMO whose father had noninfectious osteomyelitis of the sternum [10][11][12][13]. There is an autosomal recessive syndromic form of CRMO (Majeed syndrome) which is caused by mutations in LPIN2 [14][15][16][17].…”

Síndrome de Goldbloom: síndrome febril prolongado con disproteinemia. Caso clínico

“…En pocos casos, se han administrado corticoides o colchicina con buena respuesta. [9][10][11][12][13][14] Se han reportado casos de persistencia de las imágenes radiológicas hasta por 2 años.…”