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A diabetic patient with finger gangrene

  • Sho Osonoi
    Correspondence
    Corresponding author at: 5, Zaifu-cho, Hirosaki, Aomori 036-8562, Japan.
    Affiliations
    Department of Endocrinology and Metabolism, Hirosaki University Graduate School of Medicine, 5 Zaifu-cho, Hirosaki, Aomori 036-8562, Japan

    Department of Diabetes and Endocrinology, Mutsu General Hospital, Aomori 035-0071, Japan
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  • Kiho Hamaura
    Affiliations
    Department of Endocrinology and Metabolism, Hirosaki University Graduate School of Medicine, 5 Zaifu-cho, Hirosaki, Aomori 036-8562, Japan
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  • Hiroshi Murakami
    Affiliations
    Department of Endocrinology and Metabolism, Hirosaki University Graduate School of Medicine, 5 Zaifu-cho, Hirosaki, Aomori 036-8562, Japan

    Department of Diabetes and Endocrinology, Mutsu General Hospital, Aomori 035-0071, Japan
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      Keywords

      1. Main section (case description)

      A 72-year-old male with type 2 diabetes and end-stage renal disease (ESRD) receiving hemodialysis (HD) for 10 months presented to hospital with one week of worsening pain, swelling, and bluish-black discoloration in the second and fourth right, and third left fingers. Preceding trauma to the digits had not occurred. Physical examination revealed dry gangrene in the digits (Fig. A), but no necrotic lesions in the lower limbs. An arteriovenous fistula (AVF) was created in his left forearm. Laboratory examinations showed fairly controlled diabetes (HbA1c 7.3%, post-prandial blood glucose 175 mg/dL), leukocytosis, and elevated C-reactive protein. Serological markers for auto-immune diseases and vasculitis syndromes, including anti-nuclear, rheumatoid factor, anti-Jo1, anti–double-stranded DNA, cytoplasmic–anti-neutrophil cytoplasmic (ANCA), and perinuclear–ANCA antibodies were all negative. A radiograph showed prominent calcification of the digital arteries in both hands (Fig. B). Computed tomography–angiography revealed only incomplete arterial occlusions in the upper limbs. What is your diagnosis for this patient?
      Fig A
      Fig. APhysical and radiological findings of the hands. (A) Physical examination revealed dry gangrene in the digits. (B) A radiograph of the right hand showed prominent calcification of the digital arteries.

      2. Discussion section

      The findings were consistent with calciphylaxis, a rare, life-threatening syndrome involving systemic calcification of the arteries. It typically affects patients with ESRD, with an estimated annualized incidence of 0.35% in the US, and less than 0.01% in Japan, which is lower than that of Western countries [
      • Nigwekar SU
      • Thadhani R
      • Brandenburg VM
      Calciphylaxis.
      ,
      • Hayashi M.
      Calciphylaxis–Diagnosis and clinical features.
      ]. One-year mortality in patients with calciphylaxis and ESRD is 45 to 80%, which is supposed to be related to sepsis, the most common cause of death, originating from infected wounds [
      • Nigwekar SU
      • Thadhani R
      • Brandenburg VM
      Calciphylaxis.
      ]. Despite poorly controlled diabetes that often manifests as acral gangrene of the foot, the upper limbs seldom show necrotic lesions without specific conditions that include calciphylaxis, warfarin-induced skin necrosis, atherosclerotic vascular diseases, arterial thromboembolism, vasculitis, infectious ulceration, and steal syndrome following an AVF creation [
      • Nigwekar SU
      • Thadhani R
      • Brandenburg VM
      Calciphylaxis.
      ]. Known risk factors, other than ESRD, included diabetes and hyperparathyroidism, which were associated with our patient. Earlier studies described how patients treated with peritoneal dialysis (PD) had a higher incidence of calciphylaxis than those treated with HD [
      • Nigwekar SU
      • Thadhani R
      • Brandenburg VM
      Calciphylaxis.
      ,
      • Fine A
      • Zacharias J.
      Calciphylaxis is usually non-ulcerating–Risk factors, outcome and therapy.
      ]. Our patient initially received PD but an intractable tunnel infection led to a change to HD; the preceding influence of PD might have increased the risk for the onset of calciphylaxis. The detailed pathogenesis and radical treatments for calciphylaxis are still not established. Our patient was surgically treated by amputation of the second and fourth right fingers at the metacarpophalangeal joint level.

      Funding statement

      This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

      Informed consent

      We have obtained written consent from the patient for publication of the report.

      CRediT authorship contribution statement

      Sho Osonoi: Conceptualization, Data curation, Investigation, Visualization, Writing – original draft, Writing – review & editing. Kiho Hamaura: Conceptualization, Data curation, Investigation, Visualization, Writing – original draft, Writing – review & editing. Hiroshi Murakami: Conceptualization, Supervision, Writing – review & editing.

      References

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        Calciphylaxis.
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        Calciphylaxis–Diagnosis and clinical features.
        Clin Exp Nephrol. 2013; 17: 498-503
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        Calciphylaxis is usually non-ulcerating–Risk factors, outcome and therapy.
        Kidney Int. 2002; 61: 2210-2217