2012
DOI: 10.1002/jbmr.1565
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“Atypical femoral fractures” during bisphosphonate exposure in adult hypophosphatasia

Abstract: We report a 55-year-old woman who suffered atypical subtrochanteric femoral fractures (ASFFs) after 4 years of exposure to alendronate and then zolendronate given for ''osteoporosis.'' Before alendronate treatment, she had low bone mineral density. After several months of therapy, metatarsal stress fractures began. Bisphosphonate (BP) administration was stopped following the ASFFs, and the adult form of hypophosphatasia (HPP) was diagnosed from low serum alkaline phosphatase (ALP) activity, high endogenous lev… Show more

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Cited by 163 publications
(146 citation statements)
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“…Treatment with biphosphonates has been reported disappointing 19 and even detrimental, 20 whereas teriparatide (the recombinant human parathyroid hormone PTH 1-34) was successfully used to improve and resolve metatarsal stress fractures in adult HP, however, depending on the number of mutations and on the nature of the mutation. [21][22][23][24] Nonsteroidal anti-inflammatory drugs (NSAIDs) have been shown to provide clinical improvement with reduced pain, increased muscle strength and a normalised walking distance 25,26 (see also management section).…”
Section: Yes 2 Therapy (Please Describe)mentioning
confidence: 99%
“…Treatment with biphosphonates has been reported disappointing 19 and even detrimental, 20 whereas teriparatide (the recombinant human parathyroid hormone PTH 1-34) was successfully used to improve and resolve metatarsal stress fractures in adult HP, however, depending on the number of mutations and on the nature of the mutation. [21][22][23][24] Nonsteroidal anti-inflammatory drugs (NSAIDs) have been shown to provide clinical improvement with reduced pain, increased muscle strength and a normalised walking distance 25,26 (see also management section).…”
Section: Yes 2 Therapy (Please Describe)mentioning
confidence: 99%
“…Hypophosphatasia that has radiological and clinical features similar to AFF should be considered in differential diagnosis of AFF. However, hypophosphatasia is associated with tooth losses in early ages, history of osteomalacia, recurrent metatarsal fractures, and delayed fracture healing (13). Hypophosphatasia was excluded due to the absence of these conditions in our patient's medical history and normal levels of parathyroid hormone, alkaline phosphatase and inorganic phosphate.…”
Section: Discussionmentioning
confidence: 99%
“…This risk has been also reported in HPP. One study of Sutton et al (11) described a case of a 55-year-old Caucasian woman manifesting a fragility atypical subtrochanteric femur fracture, occurred after 4 year-therapy with alendronate and zoledronate for suspected osteoporosis; targeted analyses revealed the patient to be an adult HPP form. NBPs are synthetic molecules that mimic the chemical structure of PPi, a natural substrate of TNAP and a potent inhibitor of mineralization.…”
Section: Fragility Atypical Femur Fractures In Hppmentioning
confidence: 99%