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A case of synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome complicated by IgA nephropathy with nephrotic syndrome

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Abstract

A 62-year-old man visited our hospital with a mild sore throat, high-grade fever, and clavicular pain. Seven years earlier, he had been diagnosed with synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome. His clavicles were tender and remarkably swollen. Also noted was marked pitting edema in the lower extremities and pustulosis on the palms and soles of the feet. Laboratory studies on admission showed an elevated white cell count (23,400/μl) and serum C-reactive protein level (24.4 mg/dl). Urinalysis revealed proteinuria (2+) and occult blood (3+) with numerous dysmorphic red blood cells and hyalin casts. The patient was diagnosed with recurrence of his SAPHO syndrome and started on oral glucocorticoid therapy. By day 9 after admission, he had gained 16 kg in body weight, and his proteinuria (6.4 g/day) and serum creatinine level (2.3 mg/dl) were elevated. Renal biopsy revealed mesangial proliferative glomerulonephritis with deposition of IgA and C3 in the mesangial area and along the capillary walls. The patient was diagnosed with IgA nephropathy accompanied by nephrotic syndrome. With oral prednisolone therapy, his fever, clavicular pain, and proteinuria were gradually relieved. The clinical course in this case suggests the onset of nephrotic syndrome with IgA nephropathy was associated with the recurrence of the patient’s SAPHO. To our knowledge, this is the first reported case of SAPHO-associated IgA nephropathy.

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References

  1. Chamot AM, Benhamou CL, Kahn MF, Beraneck L, Kaplan G, Prost A. Acne-pustulosis-hyperostosis-osteitis syndrome. Results of a national survey. 85 cases. Rev Rhum Mal Osteoartic. 1987;54:187–96.

    CAS  PubMed  Google Scholar 

  2. Benhamou CL, Chamot AM, Kahn MF. Synovitis-acne-pustulosis hyperostosis-osteomyelitis syndrome (SAPHO). A new syndrome among the spondyloarthropathies? Clin Exp Rheumatol. 1988;6:109–12.

    CAS  PubMed  Google Scholar 

  3. Kahn MF, Khan MA. The SAPHO syndrome. Baillieres Clin Rheumatol. 1994;8:333–62.

    Article  CAS  PubMed  Google Scholar 

  4. Valentin R, Gurtler KF, Schaker A. Renal amyloidosis and renal failure—a novel complication of the SAPHO syndrome. Nephrol Dial Transplant. 1997;12:2420–3.

    Article  CAS  PubMed  Google Scholar 

  5. Colina M, Govoni M, Orzincolo C, Trotta F. Clinical and radiologic evolution of synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome: a single center study of a cohort of 71 subjects. Arthritis Rheum. 2009;61:813–21.

    Article  PubMed  Google Scholar 

  6. Hayem G, Bouchaud-Chabot A, Benali K, Roux S, Palazzo E, Silbermann-Hoffman O, et al. SAPHO syndrome: a long-term follow-up study of 120 cases. Semin Arthritis Rheum. 1999;29:159–71.

    Article  CAS  PubMed  Google Scholar 

  7. Govoni M, Colina M, Massara A, Trotta F. SAPHO syndrome and infections. Autoimmun Rev. 2009;8:256–9.

    Article  CAS  PubMed  Google Scholar 

  8. Colina M, La Monaco A, Khodeir M, Trotta F. Propionibacterium acnes and SAPHO syndrome: a case report and literature review. Clin Exp Rheumatol. 2007;25:457–60.

    CAS  PubMed  Google Scholar 

  9. Kotilainen P, Merilahti-Palo R, Lehtonen OP, Manner I, Helander I, Mottonen T, et al. Propionibacterium acnes isolated from sternal osteitis in a patient with SAPHO syndrome. J Rheumatol. 1996;23:1302–4.

    CAS  PubMed  Google Scholar 

  10. Delyle LG, Vittecoq O, Bourdel A, Duparc F, Michot C, Le Loet X. Chronic destructive oligoarthritis associated with Propionibacterium acnes in a female patient with acne vulgaris: septic-reactive arthritis? Arthritis Rheum. 2000;43:2843–7.

    Article  CAS  PubMed  Google Scholar 

  11. Zhao Z, Li Y, Li Y, Zhao H, Li H. Synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) syndrome with review of the relevant published work. J Dermatol. 2011;38:155–9.

    Article  PubMed  Google Scholar 

  12. Sanders JT, Wyatt RJ. IgA nephropathy and Henoch–Schonlein purpura nephritis. Curr Opin Pediatr. 2008;20:163–70.

    Article  PubMed  Google Scholar 

  13. Roufosse CA, Cook HT. Pathological predictors of prognosis in immunoglobulin A nephropathy: a review. Curr Opin Nephrol Hypertens. 2009;18:212–9.

    Article  CAS  PubMed  Google Scholar 

  14. Suzuki Y, Suzuki H, Nakata J, Sato D, Kajiyama T, Watanabe T, et al. Pathological role of tonsillar B cells in IgA nephropathy. Clin Dev Immunol. 2011;2011:639074.

    Article  PubMed  PubMed Central  Google Scholar 

  15. Kataura A, Tsubota H. Clinical analyses of focus tonsil and related diseases in Japan. Acta Otolaryngol Suppl. 1996;523:161–4.

    CAS  PubMed  Google Scholar 

  16. Krothapalli R, Neeland B, Small S, Duffy WB, Gyorkey F, Senekjian HO. IgA nephropathy in a patient with ankylosing spondylitis and a solitary kidney. Clin Nephrol. 1984;21:134–7.

    CAS  PubMed  Google Scholar 

  17. Dan Y, Iigari T, Ishii S, Nishiyama K, Jitsukawa K, Takemura T. A case of IgA nephropathy associated with psoriaic arthritis (abstract, in Japanese). Jpn J Nephrol. 1986;28:1594

    Google Scholar 

  18. Numata K, Mathumura O, Yumura W, Watanabe Y, Shimamoto Y, Ono A, et al. A case of IgA nephropathy with pustulosis palmaris et plantaris and inetr-sterno-costo-clavicular ossification (ISCCO)(abstract, in Japanese). Jpn J Nephrol. 1991;33:1193

    Google Scholar 

  19. Imai H, Kodama T, Ishino T, Yasuda T, Miura AB, Asakura K, et al. IgA nephropathy associated with hyper IgAnemia, psoriasis or pustulosis and ossification. Clin Nephrol. 1995;44:64–8.

    CAS  PubMed  Google Scholar 

  20. Noda K, Kodama S, Suenaga S, Suzuki M. Tonsillar focal infectious disease involving IgA nephropathy, pustulosis, and ossification. Clin Exp Nephrol. 2007;11:97–101.

    Article  CAS  PubMed  Google Scholar 

  21. Ono T, Jono M, Kito M, Tomoda T, Kageshita T, Egawa K, et al. Evaluation of tonsillectomy as a treatment for pustulosis palmaris et plantaris. Acta Otolaryngol Suppl. 1983;401:12–6.

    Article  CAS  PubMed  Google Scholar 

  22. Hotta O, Miyazaki M, Furuta T, Tomioka S, Chiba S, Horigome I, et al. Tonsillectomy and steroid pulse therapy significantly impact on clinical remission in patients with IgA nephropathy. Am J Kidney Dis. 2001;38:736–43.

    Article  CAS  PubMed  Google Scholar 

  23. Xie Y, Nishi S, Ueno M, Imai N, Sakatsume M, Narita I, et al. The efficacy of tonsillectomy on long-term renal survival in patients with IgA nephropathy. Kidney Int. 2003;63:1861–7.

    Article  PubMed  Google Scholar 

  24. Furukawa M, Miwa T, Kamide M, Sakumoto M, Umeda R. Three cases of inter-sterno-costo-clavicular ossification associated with tonsillar infection. J Laryngol Otol. 1989;103:783–6.

    Article  CAS  PubMed  Google Scholar 

  25. Shimizu S, Yukawa K, Kawaguchi S, Okubo Y, Suzuki M. Fluctuating mixed-type hearing loss associated with synovitis-acne-pustulosis-hyperostosis-osteomyelitis (SAPHO) syndrome. Auris Nasus Larynx. 2010;37:508–10.

    Article  PubMed  Google Scholar 

  26. Shiraishi W, Hayashi S, Iwanaga Y, Murai H, Yamamoto A, Kira J. A case of synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome presenting with hypertrophic pachymeningitis. J Neurol Sci. 2015;349:229–31.

    Article  PubMed  Google Scholar 

  27. Wu W, Debbaneh M, Moslehi H, Koo J, Liao W. Tonsillectomy as a treatment for psoriasis: a review. J Dermatolog Treat. 2014;25:482–6.

    Article  PubMed  PubMed Central  Google Scholar 

  28. Liu LL, Wang LN, Jiang Y, Yao L, Dong LP, Li ZL, et al. Tonsillectomy for IgA nephropathy: a meta-analysis. Am J Kidney Dis. 2015;65:80–7.

    Article  PubMed  Google Scholar 

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Correspondence to Kimihiko Nakatani.

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Morimoto, K., Nakatani, K., Asai, O. et al. A case of synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome complicated by IgA nephropathy with nephrotic syndrome. CEN Case Rep 5, 26–30 (2016). https://doi.org/10.1007/s13730-015-0184-z

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  • DOI: https://doi.org/10.1007/s13730-015-0184-z

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