Off-Label Use of Teriparatide for Osteotomy Healing in an Adolescent with Osteogenesis Imperfecta Type VIII: A Case Report

Osteogenesis imperfecta (OI) type VIII is an autosomal recessive skeletal dysplasia caused by P3H1 variants, resulting in defective collagen post-translational modification and increased bone fragility. Teriparatide (TPTD), a recombinant parathyroid hormone analogue, stimulates osteoblast activity and bone formation and is approved for the treatment of osteoporosis in adults. A 16-year-old male with genetically confirmed OI type VIII presented with delayed union with characterized by the absence of radiographic evidence of healing six months after femoral osteotomy and intramedullary rodding surgery. His prior management included intermittent bisphosphonate therapy which had improved bone mineral density. He had no recent history of fractures. Off-label therapy with TPTD (20 µg/day subcutaneously) was initiated and continued for six months. Although systemic markers of bone formation showed no remarkable changes, except osteocalcin, during therapy, serial radiographs confirmed successful bone union. The treatment was well tolerated, with no reported side effects; however, mild hypercalcemia with hypercalciuria, which persisted after discontinuation of TPTD therapy, was detected. Bisphosphonate therapy was resumed upon completion of TPTD treatment. This case represents the first documented use of TPTD to promote osteotomy healing in an adolescent with OI. The favourable clinical and radiologic outcome suggests a potential role for anabolic therapy in managing complex bone healing challenges in paediatric OI. Further research is warranted to evaluate safety and efficacy in this population.

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Publication Details

Journal
Journal of Clinical Research in Pediatric Endocrinology
Published
2026-07-22
DOI
https://doi.org/10.4274/jcrpe.galenos.2026.2026-3-21
Primary Topic
Connective tissue disorders research
Type
article
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article

Off-Label Use of Teriparatide for Osteotomy Healing in an Adolescent with Osteogenesis Imperfecta Type VIII: A Case Report

Sare Betül Kaygusuz, Ahmet Hamdi Akgülle, Didem Helvacıoğlu, Zehra Yavaş Abalı et al.
Journal of Clinical Research in Pediatric Endocrinology
Connective tissue disorders research
article

Off-Label Use of Teriparatide for Osteotomy Healing in an Adolescent with Osteogenesis Imperfecta Type VIII: A Case Report

Sare Betül Kaygusuz, Ahmet Hamdi Akgülle, Didem Helvacıoğlu, Zehra Yavaş Abalı, Aylin Günay, Motasım Khalid Bavaneh, Serap Turan
article en

Abstract

Osteogenesis imperfecta (OI) type VIII is an autosomal recessive skeletal dysplasia caused by P3H1 variants, resulting in defective collagen post-translational modification and increased bone fragility. Teriparatide (TPTD), a recombinant parathyroid hormone analogue, stimulates osteoblast activity and bone formation and is approved for the treatment of osteoporosis in adults. A 16-year-old male with genetically confirmed OI type VIII presented with delayed union with characterized by the absence of radiographic evidence of healing six months after femoral osteotomy and intramedullary rodding surgery. His prior management included intermittent bisphosphonate therapy which had improved bone mineral density. He had no recent history of fractures. Off-label therapy with TPTD (20 µg/day subcutaneously) was initiated and continued for six months. Although systemic markers of bone formation showed no remarkable changes, except osteocalcin, during therapy, serial radiographs confirmed successful bone union. The treatment was well tolerated, with no reported side effects; however, mild hypercalcemia with hypercalciuria, which persisted after discontinuation of TPTD therapy, was detected. Bisphosphonate therapy was resumed upon completion of TPTD treatment. This case represents the first documented use of TPTD to promote osteotomy healing in an adolescent with OI. The favourable clinical and radiologic outcome suggests a potential role for anabolic therapy in managing complex bone healing challenges in paediatric OI. Further research is warranted to evaluate safety and efficacy in this population.

Journal of Clinical Research in Pediatric Endocrinology
Marmara University (TR)
Good health and well-being
Openalex Percentile: Top 8%
Connective tissue disorders research
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