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Undifferentiated spondyloarthritis following allogeneic stem cell transplantation.

Karia VR, Cuchacovich R, Espinoza LR - BMC Musculoskelet Disord (2010)

Bottom Line: The patient suffered from intermittent inflammatory back pain and peripheral joint swelling for several years and did not find relief through multiple emergency room visits at different medical facilities.After a thorough history and physical exam, it was noted that our patient had developed signs of axial disease along with dactylitis and overall that he had been insidiously developing an undifferentiated spondyloarthopathy after allogeneic stem cell transplantation.Thus, larger studies and awareness of this association are needed to delineate the exact underlying mechanism(s).

View Article: PubMed Central - HTML - PubMed

Affiliation: Section of Rheumatology, Department of Internal Medicine, Louisiana State University Health Sciences Center, New Orleans, LA 70112-2822, USA. vrk900@yahoo.com

ABSTRACT

Background: Stem cell transplant has been utilized in the treatment of malignancies and rheumatic disease. Rheumatic disease may be transferred from the donor with active disease or may be developed in a recipient de novo as a late complication of SCT.

Case presentation: We here report the rare case of a 26-year old male patient, who has been diagnosed with undifferentiated spondyloarthropathy after unique circumstance. The patient suffered from intermittent inflammatory back pain and peripheral joint swelling for several years and did not find relief through multiple emergency room visits at different medical facilities. After a thorough history and physical exam, it was noted that our patient had developed signs of axial disease along with dactylitis and overall that he had been insidiously developing an undifferentiated spondyloarthopathy after allogeneic stem cell transplantation.

Conclusion: Our observation supports the hypothesis that de novo rheumatic disease can develop after stem cell transplant for a variety of reasons. Thus, larger studies and awareness of this association are needed to delineate the exact underlying mechanism(s).

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Related in: MedlinePlus

Sacroiliac joints x-ray showing narrowing and irregularity of the left sacroiliac joint.
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Figure 4: Sacroiliac joints x-ray showing narrowing and irregularity of the left sacroiliac joint.

Mentions: A 26-year-old white man diagnosed with acute pre-B lymphocytic leukemia (ALL) at age 10 refractory to chemotherapy for which he underwent a haplo-identical allogeneic stem cell transplantation (SCT) from his father who did not have any rheumatic and/or dermatologic inflammatory diseases including psoriasis. Except for a febrile and convulsive episode that lasted only a few days with a very good response to a short course of oral prednisone, he remained well until about 10 years later when he began to exhibit lower back pain and stiffness. On Christmas day 2003, he woke up with a painful, stiff and swollen right ankle which made walking very difficult. From December 2003 onwards the patient complained of right ankle pain, swelling and stiffness with difficulty in walking and only partial and transient clinical response to indomethacin, colchicine and allopurinol therapy. In January 2008, he presented to a local emergency room with a painful, stiff, and swollen right elbow for which he received a short course of oral prednisone. Over the subsequent 4 months, he continued to have right elbow and ankle pain and swelling accompanied by difficulty using his arm and walking. In May 2008, he was admitted to our University Hospital for left elbow and hand swelling and pain. On physical examination, blood pressure was 131/73, pulse 81, height 5'8", weight 215, BMI 31.6. He was in distress and pertinent findings revealed the presence of mild flaking and dryness of earlobes overlying skin, swelling, redness and tenderness of the second digit distal interphalangeal joint of left hand, swelling, tenderness, and decreased range of motion of right ankle, sausage digit deformity of the middle digit of the left foot, and enthesitis of the right Achilles tendon (Figures 1, 2, 3). In addition, both sacroiliac joints were tender to palpation and a Schober test was positive. Past history: Hypertension, bilateral cataracts, and metabolic syndrome. Family history: Paternal grandfather had psoriasis and also suffered from stiff hands. Maternal grandmother had a history of rheumatoid arthritis for over 30 years. Laboratory findings: ESR: 56 mm/h (n < 20), CRP: 17.02 mg/dL (n < 0.80). Rheumatoid factor, CCP antibodies, ANA, and HLA-B27 were negative and/or normal. Hands and feet radiographs did not show erosions. Sacroiliac joints x-ray showed unilateral sacroiliitis (Figure 4). Follow-up: patient is improved on symptomatic NSAID therapy.


Undifferentiated spondyloarthritis following allogeneic stem cell transplantation.

Karia VR, Cuchacovich R, Espinoza LR - BMC Musculoskelet Disord (2010)

Sacroiliac joints x-ray showing narrowing and irregularity of the left sacroiliac joint.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC2902413&req=5

Figure 4: Sacroiliac joints x-ray showing narrowing and irregularity of the left sacroiliac joint.
Mentions: A 26-year-old white man diagnosed with acute pre-B lymphocytic leukemia (ALL) at age 10 refractory to chemotherapy for which he underwent a haplo-identical allogeneic stem cell transplantation (SCT) from his father who did not have any rheumatic and/or dermatologic inflammatory diseases including psoriasis. Except for a febrile and convulsive episode that lasted only a few days with a very good response to a short course of oral prednisone, he remained well until about 10 years later when he began to exhibit lower back pain and stiffness. On Christmas day 2003, he woke up with a painful, stiff and swollen right ankle which made walking very difficult. From December 2003 onwards the patient complained of right ankle pain, swelling and stiffness with difficulty in walking and only partial and transient clinical response to indomethacin, colchicine and allopurinol therapy. In January 2008, he presented to a local emergency room with a painful, stiff, and swollen right elbow for which he received a short course of oral prednisone. Over the subsequent 4 months, he continued to have right elbow and ankle pain and swelling accompanied by difficulty using his arm and walking. In May 2008, he was admitted to our University Hospital for left elbow and hand swelling and pain. On physical examination, blood pressure was 131/73, pulse 81, height 5'8", weight 215, BMI 31.6. He was in distress and pertinent findings revealed the presence of mild flaking and dryness of earlobes overlying skin, swelling, redness and tenderness of the second digit distal interphalangeal joint of left hand, swelling, tenderness, and decreased range of motion of right ankle, sausage digit deformity of the middle digit of the left foot, and enthesitis of the right Achilles tendon (Figures 1, 2, 3). In addition, both sacroiliac joints were tender to palpation and a Schober test was positive. Past history: Hypertension, bilateral cataracts, and metabolic syndrome. Family history: Paternal grandfather had psoriasis and also suffered from stiff hands. Maternal grandmother had a history of rheumatoid arthritis for over 30 years. Laboratory findings: ESR: 56 mm/h (n < 20), CRP: 17.02 mg/dL (n < 0.80). Rheumatoid factor, CCP antibodies, ANA, and HLA-B27 were negative and/or normal. Hands and feet radiographs did not show erosions. Sacroiliac joints x-ray showed unilateral sacroiliitis (Figure 4). Follow-up: patient is improved on symptomatic NSAID therapy.

Bottom Line: The patient suffered from intermittent inflammatory back pain and peripheral joint swelling for several years and did not find relief through multiple emergency room visits at different medical facilities.After a thorough history and physical exam, it was noted that our patient had developed signs of axial disease along with dactylitis and overall that he had been insidiously developing an undifferentiated spondyloarthopathy after allogeneic stem cell transplantation.Thus, larger studies and awareness of this association are needed to delineate the exact underlying mechanism(s).

View Article: PubMed Central - HTML - PubMed

Affiliation: Section of Rheumatology, Department of Internal Medicine, Louisiana State University Health Sciences Center, New Orleans, LA 70112-2822, USA. vrk900@yahoo.com

ABSTRACT

Background: Stem cell transplant has been utilized in the treatment of malignancies and rheumatic disease. Rheumatic disease may be transferred from the donor with active disease or may be developed in a recipient de novo as a late complication of SCT.

Case presentation: We here report the rare case of a 26-year old male patient, who has been diagnosed with undifferentiated spondyloarthropathy after unique circumstance. The patient suffered from intermittent inflammatory back pain and peripheral joint swelling for several years and did not find relief through multiple emergency room visits at different medical facilities. After a thorough history and physical exam, it was noted that our patient had developed signs of axial disease along with dactylitis and overall that he had been insidiously developing an undifferentiated spondyloarthopathy after allogeneic stem cell transplantation.

Conclusion: Our observation supports the hypothesis that de novo rheumatic disease can develop after stem cell transplant for a variety of reasons. Thus, larger studies and awareness of this association are needed to delineate the exact underlying mechanism(s).

Show MeSH
Related in: MedlinePlus