Skip to main content

Advertisement

Log in

Two cases with pachydermoperiostosis and discussion of tamoxifen citrate treatment for arthralgia

  • Original Article
  • Published:
Clinical Rheumatology Aims and scope Submit manuscript

Abstract

Idiopathic hypertrophic osteoarthropathy is a rare syndrome, characterized by clubbing, progressive enlargement of the joints, coarse facial features suggesting acromegaly, palmoplantar hyperhidrosis, fibrous covering at the ends of long bones (periostosis), and acro-osteolysis. It is a self-limiting condition but arthralgia may be as severe as limiting social life and activity, and it is the main complaint to be treated. We report on two siblings with pachydermoperiostosis (PDP), one of whom had arthralgia. Although several drugs have been used, we used tamoxifen for his arthralgia and his complaint disappeared. To the best of our knowledge, this is the second case treated by tamoxifen reported in the literature.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Fig. 1
Fig. 2

Similar content being viewed by others

References

  1. Matsui Y, Nishii Y, Maeda M, Okada N, Yoshikawa K (1991) PDP—report of a case and review of 121 Japanese cases. Nippon Hifuka Gakkai Zasshi 101:461–467

    PubMed  CAS  Google Scholar 

  2. Jajic Z, Jajic I, Nemcic T (2001) Primary hypertrophic osteoarthropathy. Clinical, radiologic, and scintigraphic characteristics. Arch Med Res 32:136–142

    Article  PubMed  CAS  Google Scholar 

  3. Matucci-Cerinic M, Lotti T, Jajic I, Pignone A, Bussani C, Cagnoni M (1991) The clinical spectrum of pachydermoperiostosis (primary hypertrophic osteoarthropathy). Medicine 70:208–214

    Article  PubMed  CAS  Google Scholar 

  4. Fischer DS, Singer DH, Felman SM (1964) Clubbing: a review with emphasis on hereditary acropathy. Medicine 43:459–479

    Article  PubMed  CAS  Google Scholar 

  5. Rimoin DL (1965) Pachydermoperiostosis (idiopathic clubbing and periostosis). Genetic and physiological considerations. N Engl J Med 272:923–931

    Article  PubMed  CAS  Google Scholar 

  6. Pineda C, Martinez Lavin M (1990) The spectrum of soft tissue and skeletal abnormalities of hypertrophic osteoarthropathy. J Rheumatol 17:626–632

    PubMed  CAS  Google Scholar 

  7. Pineda C (1992) Diagnostic imaging in hypertrophic osteoarthropathy. Clin Exp Rheum 10 [Suppl 7]:27–33

    Google Scholar 

  8. Sinha GP, Curtis P, Haigh D, Lealman TG, Dodds W, Bennett CP (1997) Pachydermoperiostosis in childhood. Paediatr Rheumatol 36:1224–1227

    Article  CAS  Google Scholar 

  9. Ansell BM (1992) Hypertrophic osteoarthropathy in the paediatric age. Clin Exp Rheumatol 10 [Suppl 7]:15–18

    PubMed  Google Scholar 

  10. Oikarinen A, Palatsi R, Kylmaniemi M, Keski-Oja J, Ristelli J, Kallioinen M (1994) PDP: analysis of the connective tissue abnormalities in one family. J Am Acad Dermatol 31:947–953

    Article  PubMed  CAS  Google Scholar 

  11. Matucci-Cerinic M, Sacerdoti L, Perrone C, Carossino A, Cagnoni ML, Jagic I et al (1992) Pachydermoperiostosis: in vitro evidence for abnormal fibroblast proliferations. Clin Exp Rheumatol 10 [Suppl 7]:57–60

    PubMed  Google Scholar 

  12. Bianchi L, Lubrano C, Carrozzo AM, Iraci S, Tomassoli M, Spera G et al (1995) Pachydermoperiostosis: study of epidermal growth factor and steroid receptors. Br J Dermatol 132:128–133

    Article  PubMed  CAS  Google Scholar 

  13. Maeda H, Kumagai K, Konnishi F, Katayamma Y, Hiyama K, Ishioka S, Yamakido M (2000) Successful treatment of arthralgia with tamoxifen citrate in a patient with pachydermoperiostosis. Rheumatology 39:1158–1159

    Article  PubMed  CAS  Google Scholar 

Download references

Author information

Authors and Affiliations

Authors

Corresponding author

Correspondence to Ilke Mungan.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Okten, A., Mungan, I., Kalyoncu, M. et al. Two cases with pachydermoperiostosis and discussion of tamoxifen citrate treatment for arthralgia. Clin Rheumatol 26, 8–11 (2007). https://doi.org/10.1007/s10067-005-1161-2

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s10067-005-1161-2

Keywords

Navigation