Treatment of osteoporotic fractures in alkaptonuria by teriparatide stimulates bone formation and decreases fracture rate – A report of two cases

Two cases of advanced alkaptonuria (AKU) with co-existing osteoporosis are described. Case 1 developed multiple non-vertebral fragility fractures, while Case 2 developed vertebral fragility fractures, both refractory to bisphosphonates. Difficulties in diagnosing osteoporosis in AKU complicated by e...

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Autores principales: L.R. Ranganath, M. Khedr, A. Mistry, S. Vinjamuri, J.A. Gallagher
Formato: article
Lenguaje:EN
Publicado: Elsevier 2021
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Acceso en línea:https://doaj.org/article/1aa76595e0634722a03c4727044dfcaf
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spelling oai:doaj.org-article:1aa76595e0634722a03c4727044dfcaf2021-12-04T04:34:35ZTreatment of osteoporotic fractures in alkaptonuria by teriparatide stimulates bone formation and decreases fracture rate – A report of two cases2352-187210.1016/j.bonr.2021.101151https://doaj.org/article/1aa76595e0634722a03c4727044dfcaf2021-12-01T00:00:00Zhttp://www.sciencedirect.com/science/article/pii/S2352187221004083https://doaj.org/toc/2352-1872Two cases of advanced alkaptonuria (AKU) with co-existing osteoporosis are described. Case 1 developed multiple non-vertebral fragility fractures, while Case 2 developed vertebral fragility fractures, both refractory to bisphosphonates. Difficulties in diagnosing osteoporosis in AKU complicated by extensive calcifying and ossifying spondylosis are discussed. Both patients continued to fracture despite nitisinone therapy for metabolic control of AKU, as well as bisphosphonate antiresorptive therapy for osteoporosis. Subsequently the patients were treated with teriparatide 20 μg subcutaneous injections daily for two years, leading to reduction in fractures soon after commencing therapy in both cases. Markers of bone remodelling P1NP and CTX were stimulated. No complications due hypercalcaemia or calcification were encountered in either case. We conclude that teriparatide is an effective adjunct in the treatment of AKU when bisphosphonates prove ineffective.L.R. RanganathM. KhedrA. MistryS. VinjamuriJ.A. GallagherElsevierarticleAlkaptonuriaNitisinoneHomogentisic acidOchronosisOsteoporosisBisphosphonatesDiseases of the musculoskeletal systemRC925-935ENBone Reports, Vol 15, Iss , Pp 101151- (2021)
institution DOAJ
collection DOAJ
language EN
topic Alkaptonuria
Nitisinone
Homogentisic acid
Ochronosis
Osteoporosis
Bisphosphonates
Diseases of the musculoskeletal system
RC925-935
spellingShingle Alkaptonuria
Nitisinone
Homogentisic acid
Ochronosis
Osteoporosis
Bisphosphonates
Diseases of the musculoskeletal system
RC925-935
L.R. Ranganath
M. Khedr
A. Mistry
S. Vinjamuri
J.A. Gallagher
Treatment of osteoporotic fractures in alkaptonuria by teriparatide stimulates bone formation and decreases fracture rate – A report of two cases
description Two cases of advanced alkaptonuria (AKU) with co-existing osteoporosis are described. Case 1 developed multiple non-vertebral fragility fractures, while Case 2 developed vertebral fragility fractures, both refractory to bisphosphonates. Difficulties in diagnosing osteoporosis in AKU complicated by extensive calcifying and ossifying spondylosis are discussed. Both patients continued to fracture despite nitisinone therapy for metabolic control of AKU, as well as bisphosphonate antiresorptive therapy for osteoporosis. Subsequently the patients were treated with teriparatide 20 μg subcutaneous injections daily for two years, leading to reduction in fractures soon after commencing therapy in both cases. Markers of bone remodelling P1NP and CTX were stimulated. No complications due hypercalcaemia or calcification were encountered in either case. We conclude that teriparatide is an effective adjunct in the treatment of AKU when bisphosphonates prove ineffective.
format article
author L.R. Ranganath
M. Khedr
A. Mistry
S. Vinjamuri
J.A. Gallagher
author_facet L.R. Ranganath
M. Khedr
A. Mistry
S. Vinjamuri
J.A. Gallagher
author_sort L.R. Ranganath
title Treatment of osteoporotic fractures in alkaptonuria by teriparatide stimulates bone formation and decreases fracture rate – A report of two cases
title_short Treatment of osteoporotic fractures in alkaptonuria by teriparatide stimulates bone formation and decreases fracture rate – A report of two cases
title_full Treatment of osteoporotic fractures in alkaptonuria by teriparatide stimulates bone formation and decreases fracture rate – A report of two cases
title_fullStr Treatment of osteoporotic fractures in alkaptonuria by teriparatide stimulates bone formation and decreases fracture rate – A report of two cases
title_full_unstemmed Treatment of osteoporotic fractures in alkaptonuria by teriparatide stimulates bone formation and decreases fracture rate – A report of two cases
title_sort treatment of osteoporotic fractures in alkaptonuria by teriparatide stimulates bone formation and decreases fracture rate – a report of two cases
publisher Elsevier
publishDate 2021
url https://doaj.org/article/1aa76595e0634722a03c4727044dfcaf
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