Teriparatide treatment in an adult patient with hypophosphatasia exposed to bisphosphonate and revealed by bilateral atypical fractures

Righetti, Morgane; Wach, Jean; Desmarchelier, Romain; Coury, Fabienne

doi:10.1016/j.jbspin.2017.12.001

Cited by 27 publications

(27 citation statements)

References 13 publications

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“…The most recent study also reported some benefit for 2 adult female HPP patients . Interestingly, all patients who have been treated with teriparatide have been woman . Our study is therefore the first to report the effects of teriparatide on male adult patients.…”

Section: Discussionsupporting

confidence: 56%

“…Hence, there is yet no approved medical therapy for adult‐onset HPP patients in Europe and North America, who had no symptoms in childhood or adolescence. Off‐label use of teriparatide has first been reported in adults in 2007, and showed variable clinical effects in further studies . In 2017, the treatment of 8 adult HPP patients with a monoclonal antisclerostin antibody (BPS804) increased lumbar BMD and bone formation markers, but no data on fracture healing or BME were presented.…”

Section: Introductionmentioning

confidence: 99%

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Outcome of Teriparatide Treatment on Fracture Healing Complications and Symptomatic Bone Marrow Edema in Four Adult Patients With Hypophosphatasia

et al. 2019

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The response to teriparatide has been described in very few cases of hypophosphatasia (HPP). In this cross‐sectional study, we report the prevalence of symptomatic bone marrow edema (BME) and fracture healing complications in a large cohort of childhood and adult HPP patients and discuss the results of teriparatide treatment in four cases. From 2016 to 2018, 51 patients with a diagnosis of HPP were seen at our institution. The diagnosis of HPP was established by low serum alkaline phosphatase (ALP), elevated serum pyridoxal‐5‐phosphate (PLP), at least one typical clinical symptom of HPP and supported by ALPL mutation analysis. In this study cohort, 28 (56%) and 14 (27%) patients had a history of fracture or a history of BME, respectively. Four patients, including middle‐aged to elderly women and men who all presented with persistent symptomatic BME or fracture healing complications, were treated with teriparatide. DXA was performed prior to treatment and laboratory values were measured on a regular basis during treatment. Treatment with teriparatide showed variable effects in terms of clinical and biochemical response. Although all four patients displayed a temporary increase in ALP activity, only two patients with a mild form of adult HPP and moderately increased PLP levels showed definite clinical and radiological improvement after teriparatide treatment. In conclusion, fracture healing complications and BME occur frequently in HPP patients. Teriparatide shows variable clinical and biochemical effects depending on the severity of the disease. PLP levels and the number of ALPL alleles might be good parameters to predict treatment outcomes. © 2019 The Authors. JBMR Plus Published by Wiley Periodicals, Inc. on behalf of the American Society for Bone and Mineral Research

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Section: Discussionsupporting

confidence: 56%

Section: Introductionmentioning

confidence: 99%

Outcome of Teriparatide Treatment on Fracture Healing Complications and Symptomatic Bone Marrow Edema in Four Adult Patients With Hypophosphatasia

et al. 2019

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“…A number of the heterozygous cases in Table had received bisphosphonate treatment, but the limited information available suggests the risks of fracture are substantially lower than in those with bi‐allelic mutations. Teriparatide has been used as a treatment for HPP patients with femoral fractures, with varying degrees of success . However, in the context of osteoporosis and fractures, teriparatide treatment is limited to 18 months and its effects on bone density wane thereafter, so antiresorptive treatment is recommended after its use .…”

Section: Discussionmentioning

confidence: 99%

“…Table summarizes data from published case reports and series that have described femoral fractures in adults with HPP confirmed by genetic analysis. The Table also includes data on 3 patients who had multiple fractures (but not including a femoral fracture) . Fifteen of the 34 subjects had been exposed to bisphosphonates.…”

Section: Methodsmentioning

confidence: 99%

“…The prototype bisphosphonate used in humans, etidronate, can cause osteomalacia when taken in high doses . Although the second and third generation bisphosphonates in current clinical use do not have this effect, a number of recent case reports have documented so‐called atypical femoral fractures occurring in adults with HPP who have been treated with bisphosphonates . Review articles and expert opinion now caution against their use in individuals with HPP .…”

Section: Introductionmentioning

confidence: 99%

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Bisphosphonate Use and Fractures in Adults with Hypophosphatasia

et al. 2019

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Adults with hypophosphatasia (HPP) may suffer femoral fractures resembling the atypical femoral fractures that can occur with long‐term bisphosphonate treatment, and there is an emerging consensus that bisphosphonates should not be used in adults with HPP and low bone mass. However, the spectrum of HPP in adults is wide: ranging from the severely affected—who commonly have osteomalacia—through to the minimally affected. The former typically have biallelic and the latter, heterozygous ALPL mutations. We have reviewed reports of fractures in adults with genetically proven HPP which suggest that the risk of fracture is at least 200‐fold greater in those with biallelic mutations. We also discuss two cases of postmenopausal women with heterozygous ALPL mutations. One had fractures and severe osteoporosis, but histology revealed no evidence of osteomalacia. The second had taken alendronate for 8 years, but despite profound suppression of bone turnover, histology again revealed no evidence of osteomalacia. The management of adults with HPP who have coexisting osteoporosis is challenging. More data are clearly needed, but we suggest that the risks of bisphosphonate therapy may be relatively low in patients who have heterozygous mutations and no histological evidence of osteomalacia. © 2019 The Authors. JBMR Plus published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research © 2019 The Authors. JBMR Plus published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research.

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Alkaline Phosphatase Replacement Therapy for Hypophosphatasia in Development and Practice

Bowden

Foster

2019

Advances in Experimental Medicine and Biology

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Teriparatide treatment in an adult patient with hypophosphatasia exposed to bisphosphonate and revealed by bilateral atypical fractures

Cited by 27 publications

References 13 publications

Outcome of Teriparatide Treatment on Fracture Healing Complications and Symptomatic Bone Marrow Edema in Four Adult Patients With Hypophosphatasia

Outcome of Teriparatide Treatment on Fracture Healing Complications and Symptomatic Bone Marrow Edema in Four Adult Patients With Hypophosphatasia

Bisphosphonate Use and Fractures in Adults with Hypophosphatasia

Alkaline Phosphatase Replacement Therapy for Hypophosphatasia in Development and Practice

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