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Letters: Observations

Resolution of Diabetic Cheiroarthropathy After Pancreatic Transplantation

  1. Samantha L. Hider, MRCP, MSC, BM, BS1,
  2. Dipak K. Roy, MRCP, MSC2,
  3. Titus Augustine, MS, FRCSED3,
  4. Neil Parrott, MD, FRCS3 and
  5. Ian N. Bruce, MD, FRCP12
  1. 1Arc Epidemiology Unit, University of Manchester, Manchester, U.K
  2. 2University of Manchester Rheumatism Research Centre, Central Manchester and Manchester Children’s University Hospitals National Health Service Trust, Manchester, U.K
  3. 3Manchester Institute of Nephrology and Transplantation, Central Manchester and Manchester Children’s University Hospitals National Health Service Trust, Manchester, U.K
  1. Address correspondence to S.L. Hider, Arc Epidemiology Unit, University of Manchester, Stopford Building, Oxford Road, Manchester M13 9PT, U.K. E-mail: sam.hider{at}man.ac.uk
Diabetes Care 2004 Sep; 27(9): 2279-2280. https://doi.org/10.2337/diacare.27.9.2279-a
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A 51-year-old man was referred to our unit with a 12-month history of progressive impairment of hand function. He was unable to make a fist and had difficulty picking up small objects. He did not describe any joint pain, swelling, or morning stiffness, and there were no features to suggest an inflammatory arthropathy. He had been diagnosed with type 1 diabetes at age 7 years, complicated by diabetic nephropathy requiring a renal transplant 20 years previously (for which he was on long-term ciclosporin) and retinopathy.

On examination, his skin appeared slightly thickened. He had contractures evidenced by a positive prayer sign and was unable to flatten his hands completely. The remainder of the physical examination was unremarkable except for a functioning renal transplant; specifically, he had no evidence of synovitis or neuropathy. Laboratory investigations, including erythrocyte sedimentation rate, C-reactive protein, and rheumatoid factor, were normal. His HbA1c was 6.0%. Hand radiographs were unremarkable. He was treated with physiotherapy and wax for a presumed diagnosis of diabetic cheiroarthropathy with little improvement in his symptoms. He had a successful pancreatic transplant 2 months later, and his immunosuppression was changed to mycophenolate mofetil, tacrolimus, and a reducing course of prednisolone. His symptoms began to improve within a few weeks of surgery, and 6 weeks later he had full range of movement in both hands. His HbA1c was reduced to 5.2%. One year later, he remains asymptomatic with normal hand function.

Diabetic cheiroarthropathy is characterized by skin thickening and restriction defined as “the inability to extend the metacarpophalangeal joints fully” (1) and is thought to be caused by collagen abnormalities and increased glycation of connective tissue. Studies suggest that cheiroarthropathy is associated with type 1 diabetes (2), duration of diabetes (3,4), and secondary complications (2,4). Treatment is often unsatisfactory, involving corticosteroid injection or surgery in severe cases. It is suggested that improved glycemic control may improve symptoms, although it is not usually associated with complete resolution.

Pancreatic transplantation is currently the only therapy for type 1 diabetes that re-establishes endogenous insulin secretion, rendering the recipient euglycemic. Follow-up studies of pancreatic transplant patients suggest that complications, including retinopathy, nephropathy, and neuropathy, are stabilized. Several studies report reversal of nephropathy (5,6), although it is suggested this can only be expected after a long observation period (6).

This is the first case we are aware of in which cheiroarthropathy has been noted to resolve posttransplantation and highlights two potential mechanisms of cheiroarthropathy. Firstly, the early improvement on corticosteroids suggests an inflammatory component, which other authors have postulated (7), although the sustained improvement suggests that this is not simply a steroid effect but may reflect other factors such as a change in immunosuppression. Secondly, an alternative hypothesis suggests that the improvement in glycemic control may lead to resolution of symptoms, although the speed of improvement makes this less likely.

In conclusion, we observed the resolution of diabetic cheiroarthropathy after successful pancreatic transplant, which raises interesting potential mechanisms of cheiroarthropathy.

Footnotes

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References

  1. ↵
    Crispin JC, Alcocer-Varela J: Rheumatologic manifestations of diabetes mellitus. Am J Med 114:753–757, 2003
    OpenUrlPubMed
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    Cagliero E, Apruzzese W, Perlmutter GS, Nathan DM: Musculoskeletal disorders of the hand and shoulder in patients with diabetes mellitus. Am J Med 112:487–490, 2002
    OpenUrlCrossRefPubMedWeb of Science
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    Gamstedt A, Holm-Glad J, Ohlson CG, Sundstrom M: Hand abnormalities are strongly associated with the duration of diabetes mellitus. J Intern Med 234:189–193, 1993
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    Frost D, Beischer W: Limited joint mobility in type 1 diabetic patients: associations with microangiopathy and subclinical macroangiopathy are different in men and women. Diabetes Care 24:95–99, 2001
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    Sutherland DE, Gruessner RW, Dunn DL, Matas AJ, Humar A, Kandaswamy R, Mauer SM, Kennedy WR, Goetz FC, Robertson RP, Gruessner AC, Najarian JS: Lessons learned from more than 1,000 pancreas transplants at a single institution. Ann Surg 233:463–501, 2001
    OpenUrlCrossRefPubMedWeb of Science
  6. ↵
    Hopt UT, Drognitz O: Pancreas organ transplantation: short and long-term results in terms of diabetes control. Langenbecks Arch Surg 385:379–389, 2000
    OpenUrlCrossRefPubMed
  7. ↵
    Sibbitt WL Jr, Eaton RP: Corticosteroid responsive tenosynovitis is a common pathway for limited joint mobility in the diabetic hand. J Rheumatol 24:931–936, 1997
    OpenUrlPubMed
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Resolution of Diabetic Cheiroarthropathy After Pancreatic Transplantation
Samantha L. Hider, Dipak K. Roy, Titus Augustine, Neil Parrott, Ian N. Bruce
Diabetes Care Sep 2004, 27 (9) 2279-2280; DOI: 10.2337/diacare.27.9.2279-a

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Resolution of Diabetic Cheiroarthropathy After Pancreatic Transplantation
Samantha L. Hider, Dipak K. Roy, Titus Augustine, Neil Parrott, Ian N. Bruce
Diabetes Care Sep 2004, 27 (9) 2279-2280; DOI: 10.2337/diacare.27.9.2279-a
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