Cyclic intravenous pamidronate treatment in children with nodulosis, arthropathy and osteolysis syndrome

Al‐Mayouf, Sulaiman M.; Bin-Abbas, Bassam

doi:10.1136/ard.2005.035725

Cited by 15 publications

(14 citation statements)

References 9 publications

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“…Unfortunately, no effective treatment is available for these disorders. Supportive treatment in the form of calcium and vitamin D supplementation in addition to bisphosphonate might increase the bone density but does not change the disease course [18,19].…”

Section: Idiopathic Multicentric Osteolysismentioning

confidence: 99%

Noninflammatory disorders mimic juvenile idiopathic arthritis

Al‐Mayouf

2018

International Journal of Pediatrics and Adolescent Medicine

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Juvenile idiopathic arthritis (JIA) is the most common chronic childhood arthritis; unfortunately, no diagnostic tool is available. Genetic disorders with musculoskeletal involvement that mimic chronic polyarthritis should be considered in the differential diagnostics of JIA. Normal inflammatory markers and characteristic radiological features are able to distinguish these disorders from JIA. Timely diagnosis of these disorders is crucial to offer the family proper genetic counseling and avoid inappropriate therapy. This review highlights selected noninflammatory disorders that often present with articular manifestations and that are often mislabeled as JIA. The focus is on the clinical, biochemical, and imaging features of these disorders.

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Section: Idiopathic Multicentric Osteolysismentioning

confidence: 99%

Noninflammatory disorders mimic juvenile idiopathic arthritis

Al‐Mayouf

2018

International Journal of Pediatrics and Adolescent Medicine

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“…There is no specific therapy for the disorder. Bisphosphonates have been tried by two groups but did not benefit the affected children to a significant extent [Al‐Mayouf et al, ; Phadke et al, ]. There is no data on the benefit of analgesics either.…”

Section: Discussionmentioning

confidence: 99%

Clinical and mutation profile of multicentric osteolysis nodulosis and arthropathy

Bhavani

Shah

Shukla

et al. 2015

American J of Med Genetics Pt A

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Multicentric osteolysis nodulosis and arthropathy (MONA) is an infrequently described autosomal recessive skeletal dysplasia characterized by progressive osteolysis and arthropathy. Inactivating mutations in MMP2, encoding matrix metalloproteinase-2, are known to cause this disorder. Fifteen families with mutations in MMP2 have been reported in literature. In this study we screened thirteen individuals from eleven families for MMP2 mutations and identified eight mutations (five novel and three known variants). We characterize the clinical, radiographic and molecular findings in all individuals with molecularly proven MONA from the present cohort and previous reports, and provide a comprehensive review of the MMP2 related disorders.

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“…The authors also noticed a decrease in limb and joint pain and improvement in functional ability although not significant. However bone mineral density had increase significantly in all patients at the end of treatment 34 .…”

Section: Discussionmentioning

confidence: 82%

“…Similarly to our study they noticed a subjective decrease in pain, tenderness and joint contractures at the wrist as well as increase of bone mineral density of the axial skeleton but no increase at the radius and ulna. Al-Mayouf et al 34 treated seven children with NAO syndrome with intravenous pamidronate. The dosis was 2 mg/kg on each of three consecutive days every three month for one year.…”

Section: Discussionmentioning

confidence: 99%

Bisphosphonates in multicentric osteolysis, nodulosis and arthropathy (MONA) spectrum disorder – an alternative therapeutic approach

Pichler

Kotzot

Steichen-Gersdorf

et al. 2016

Sci Rep

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Multicentric osteolysis, nodulosis and arthropathy (MONA) spectrum disorder is a rare inherited progressive skeletal disorder caused by mutations in the matrix metalloproteinase 2 (MMP2) gene. Treatment options are limited. Herein we present successful bisphosphonate therapy in three affected patients. Patients were treated with bisphosphonates (either pamidronate or zoledronate) for different time periods. The following outcome variables were assessed: skeletal pain, range of motion, bone densitometry, internal medical problems as well as neurocognitive function. Skeletal pain was dramatically reduced in all patients soon after initiation of therapy and bone mineral density increased. Range of motion did not significantly improve. One patient is still able to walk with aids at the age of 14 years. Neurocognitive development was normal in all patients. Bisphosphonate therapy was effective especially in controlling skeletal pain in MONA spectrum disorder. Early initiation of treatment seems to be particularly important in order to achieve the best possible outcome.

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Cyclic intravenous pamidronate treatment in children with nodulosis, arthropathy and osteolysis syndrome

Cited by 15 publications

References 9 publications

Noninflammatory disorders mimic juvenile idiopathic arthritis

Noninflammatory disorders mimic juvenile idiopathic arthritis

Clinical and mutation profile of multicentric osteolysis nodulosis and arthropathy

Bisphosphonates in multicentric osteolysis, nodulosis and arthropathy (MONA) spectrum disorder – an alternative therapeutic approach

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