Primary hypertrophic osteoarthropathy presenting as juvenile idiopathic arthritis: a case report

Merwin Samuel; Arul Rajamurugan; Ramesh Subramanian; Ramesh Ramamoorthy

doi:10.18203/2349-3291.ijcp20250361

Authors

Merwin Samuel Department of Rheumatology, Madras Medical College, Chennai, Tamil Nadu, India https://orcid.org/0009-0008-8331-3163
Arul Rajamurugan Department of Rheumatology, Madras Medical College, Chennai, Tamil Nadu, India https://orcid.org/0000-0002-2259-1572
Ramesh Subramanian Department of Rheumatology, Madras Medical College, Chennai, Tamil Nadu, India
Ramesh Ramamoorthy Department of Rheumatology, Madras Medical College, Chennai, Tamil Nadu, India

DOI:

https://doi.org/10.18203/2349-3291.ijcp20250361

Keywords:

Pachydermoperiosteitis, Bisphosphonates, Zolendronic acid, Proliferative bone disease

Abstract

Primary hypertrophic osteoarthropathy (PHO) is an inherited disorder, characterised by skin thickening of face and extremities, and periosteal proliferation of the long bones. It is caused by HPGD or SLCO2A1 gene mutations, inherited in an autosomal dominant or recessive pattern. It presents with characteristic skin changes and musculoskeletal pain. We present the case of an adolescent male patient with hypertrophic osteoarthropathy. We managed him successfully with zolendronic acid.

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References

Martínez-Lavín M. Hypertrophic osteoarthropathy. Best practice & research. Clin Rheumatol. 2020;34(3):101507. DOI: https://doi.org/10.1016/j.berh.2020.101507

Yap FY, Skalski MR, Patel DB, Schein AJ, White EA, Tomasian A, et al. Hypertrophic Osteoarthropathy: Clinical and Imaging Features. Radiographics. 2017;37(1):157-95. DOI: https://doi.org/10.1148/rg.2017160052

Lu Q, Xu Y, Zhang Z, Li S, Zhang Z. Primary hypertrophic osteoarthropathy: genetics, clinical features and management. Front Endocrinol (Lausanne). 2023;14:1235040. DOI: https://doi.org/10.3389/fendo.2023.1235040

Faust B, Parkinson A, Baumrucker SJ. Pharmacology update: pamidronate for hypertrophic pulmonary osteoarthropathy in palliative care. Ther Adv Rare Dis. 2022;3:26330040211070298. DOI: https://doi.org/10.1177/26330040211070298

Jayakar BA, Abelson AG, Yao Q. Treatment of hypertrophic osteoarthropathy with zoledronic acid: case report and review of the literature. Semin Arthritis Rheum. 2011;41(2):291-6. DOI: https://doi.org/10.1016/j.semarthrit.2011.01.007

Gupta M, Lehl SS, Singh R, Sachdev A. Touraine-Solente-Gole' syndrome. BMJ Case Rep. 2011;2011:bcr0820114605. DOI: https://doi.org/10.1136/bcr.08.2011.4605

Ganesan GRG, Ganesan VR. Primary Hypertrophic Osteoarthropathy and successful treatment with Oral Bisphosphonates: A Case Report with Literature Review. Int J Orthop Rheumatol. 2016;2(1):47-51.

Palui R, Sridharan K, Sahoo J, Suryadevara V, Kamalanathan S, Naik D, et al. Role of bisphosphonates in hypertrophic osteoarthropathy: a systematic review. Endocrine. 2024;85(2):883-93. DOI: https://doi.org/10.1007/s12020-024-03804-5