Mitigating the Denosumab-Induced Rebound Phenomenon with Alternating Short- and Long-Acting Anti-resorptive Therapy in a Young Boy with Severe OI Type VI

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dc.contributor.authorSeale, Emily
dc.contributor.authorMolina, Maria Ochoa
dc.contributor.authorCarsen, Sasha
dc.contributor.authorSheffield, Holden
dc.contributor.authorKoujok, Khaldoun
dc.contributor.authorRobinson, Marie-Eve
dc.contributor.authorFeber, Janusz
dc.contributor.authorSmit, Kevin
dc.contributor.authorPage, Marika
dc.contributor.authorWalker, Scott
dc.contributor.authorKhan, Nasrin
dc.contributor.authorKonji, Victor N.
dc.contributor.authorRauch, Frank
dc.contributor.authorWard, Leanne M.
dc.date.accessioned2025-01-20T20:17:17Z
dc.date.available2025-01-20T20:17:17Z
dc.date.issued2023
dc.description.abstractOsteogenesis imperfecta (OI) type VI, a recessively inherited form of OI caused by mutations in SERPINF1, is a severe form distinguished by osteomalacia on bone histomorphometry. We describe a boy with severe OI type VI who was initially treated with intravenous (IV) zoledronic acid (ZA) at 1.4 years of age; however, a year later he transitioned to denosumab 1 mg/kg sub-cutaneously every three months in an effort to decrease fracture rates. After two years on denosumab, he presented with symptomatic hypercalcemia due to the denosumab-induced, hyper-resorptive rebound phenomenon. Laboratory parameters at the time of the rebound were as follows: elevated serum ionized calcium (1.62 mmol/L, N 1.16-1.36), elevated serum creatinine due to hypercalcemia-induced muscle catabolism (83 mu mol/L, N 9-55), and suppressed parathyroid hormone (PTH) (< 0.7 pmol/L, N 1.3-5.8). The hypercalcemia was responsive to low-dose IV pamidronate, with a rapid decline in serum ionized calcium, and otherwise normalization of the aforementioned parameters within 10 days. To benefit from the powerful, albeit short-term, anti-resorptive effect of denosumab without further rebound episodes, he was treated thereafter with denosumab 1 mg/kg alternating every three months with IV ZA 0.025 mg/kg. Five years later, he remained on dual alternating anti-resorptive therapy without further rebound episodes, and an overall improvement in his clinical status. This novel pharmacological approach of alternating short- and long-term anti-resorptive therapy every three months has not previously been described. Our report suggests this strategy may be an effective method for prevention of the rebound phenomenon in select children for whom denosumab may be beneficial.
dc.fuente.origenWOS
dc.identifier.doi10.1007/s00223-023-01065-4
dc.identifier.eissn1432-0827
dc.identifier.issn0171-967X
dc.identifier.urihttps://doi.org/10.1007/s00223-023-01065-4
dc.identifier.urihttps://repositorio.uc.cl/handle/11534/92387
dc.identifier.wosidWOS:000943182200001
dc.issue.numero5
dc.language.isoen
dc.pagina.final620
dc.pagina.inicio613
dc.revistaCalcified tissue international
dc.rightsacceso restringido
dc.subjectDenosumab
dc.subjectZoledronic acid
dc.subjectOsteogenesis imperfecta
dc.subjectRebound
dc.subjectOI VI
dc.subjectHypercalcemia
dc.subjectHypercalciuria
dc.titleMitigating the Denosumab-Induced Rebound Phenomenon with Alternating Short- and Long-Acting Anti-resorptive Therapy in a Young Boy with Severe OI Type VI
dc.typeartículo
dc.volumen112
sipa.indexWOS
sipa.trazabilidadWOS;2025-01-12
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