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Response to rituximab in a case of lupus associated digital ischemia.

Küçükşahin O, Düzgün N, Okoh AK, Kulahçioglu E - Case Rep Rheumatol (2014)

Bottom Line: We report the case of a 38-year-old female patient with systemic lupus erythematosus (SLE) and Jaccoud arthritis (JA) that sequentially developed digital ischemic lesions of the hands.In spite of follow-up treatment with glucocorticoids, immunosuppressant, antiaggregant, and potent vasodilatator agents, a serious progression to digital gangrene over a one-month period was observed.Surprisingly, her nonhealing digital lesions improved after two cycles of rituximab (RTX) administration.

View Article: PubMed Central - PubMed

Affiliation: Department of Rheumatology, Ankara University Medical Faculty, Sihhiye, 06410 Ankara, Turkey.

ABSTRACT
We report the case of a 38-year-old female patient with systemic lupus erythematosus (SLE) and Jaccoud arthritis (JA) that sequentially developed digital ischemic lesions of the hands. In spite of follow-up treatment with glucocorticoids, immunosuppressant, antiaggregant, and potent vasodilatator agents, a serious progression to digital gangrene over a one-month period was observed. Surprisingly, her nonhealing digital lesions improved after two cycles of rituximab (RTX) administration.

No MeSH data available.


Related in: MedlinePlus

(a) Right hand with swan neck and Z deformity of the thumb (before treatment). (b) Radiograph; AP of bilateral hands suggesting Jaccoud radiological features. (c) Digital ulcers of the second to fourth digits of the right hand (2 weeks after initial treatment with cyclophosphamide). (d) Unresponsiveness to initial treatment demonstrated by a serious progression from ischemic lesions to digital gangrene lesions of the second to fourth digits of the right hand (one month after initial treatment regimen; cyclophosphamide, corticosteroids, and prostaglandins). (e) Response to treatment observed after first cycle of rituximab therapy (5 months following the start of RTX treatment). (f) Complete remission of digital gangrenous lesions after 2 cycles of rituximab therapy (10th month of the RTX treatment protocol (rituximab, glucocorticoid, aspirin, azathioprine, and hydroxychloroquine)).
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fig1: (a) Right hand with swan neck and Z deformity of the thumb (before treatment). (b) Radiograph; AP of bilateral hands suggesting Jaccoud radiological features. (c) Digital ulcers of the second to fourth digits of the right hand (2 weeks after initial treatment with cyclophosphamide). (d) Unresponsiveness to initial treatment demonstrated by a serious progression from ischemic lesions to digital gangrene lesions of the second to fourth digits of the right hand (one month after initial treatment regimen; cyclophosphamide, corticosteroids, and prostaglandins). (e) Response to treatment observed after first cycle of rituximab therapy (5 months following the start of RTX treatment). (f) Complete remission of digital gangrenous lesions after 2 cycles of rituximab therapy (10th month of the RTX treatment protocol (rituximab, glucocorticoid, aspirin, azathioprine, and hydroxychloroquine)).

Mentions: Two years following surgical treatment for avascular necrosis, the patient presented to our rheumatologic polyclinic with complaints of pain, swelling, warmth, and cyanosis over the fingers of both hand and toe tips. Physical examination revealed swelling, warmth, and tenderness in the proximal interphalangeal (PIP) joints of both hands, Ulnar deviation, and bilateral swan neck deformities. Right is greater than left. “Z” deformities of thumbs (Figure 1(a)) and cyanotic ulcers were seen on the third to fifth digits (Figure 1(c)) and on both digits of feet. All described deformities were reducible. Peripheral pulses were normal. In addition, she had a history of photosensitivity and two gestations with two uneventful births. On further interrogation, the patient was admitted to stopping medical treatment at her own discretion three years before presentation due to exorbitant side effects.


Response to rituximab in a case of lupus associated digital ischemia.

Küçükşahin O, Düzgün N, Okoh AK, Kulahçioglu E - Case Rep Rheumatol (2014)

(a) Right hand with swan neck and Z deformity of the thumb (before treatment). (b) Radiograph; AP of bilateral hands suggesting Jaccoud radiological features. (c) Digital ulcers of the second to fourth digits of the right hand (2 weeks after initial treatment with cyclophosphamide). (d) Unresponsiveness to initial treatment demonstrated by a serious progression from ischemic lesions to digital gangrene lesions of the second to fourth digits of the right hand (one month after initial treatment regimen; cyclophosphamide, corticosteroids, and prostaglandins). (e) Response to treatment observed after first cycle of rituximab therapy (5 months following the start of RTX treatment). (f) Complete remission of digital gangrenous lesions after 2 cycles of rituximab therapy (10th month of the RTX treatment protocol (rituximab, glucocorticoid, aspirin, azathioprine, and hydroxychloroquine)).
© Copyright Policy
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4120793&req=5

fig1: (a) Right hand with swan neck and Z deformity of the thumb (before treatment). (b) Radiograph; AP of bilateral hands suggesting Jaccoud radiological features. (c) Digital ulcers of the second to fourth digits of the right hand (2 weeks after initial treatment with cyclophosphamide). (d) Unresponsiveness to initial treatment demonstrated by a serious progression from ischemic lesions to digital gangrene lesions of the second to fourth digits of the right hand (one month after initial treatment regimen; cyclophosphamide, corticosteroids, and prostaglandins). (e) Response to treatment observed after first cycle of rituximab therapy (5 months following the start of RTX treatment). (f) Complete remission of digital gangrenous lesions after 2 cycles of rituximab therapy (10th month of the RTX treatment protocol (rituximab, glucocorticoid, aspirin, azathioprine, and hydroxychloroquine)).
Mentions: Two years following surgical treatment for avascular necrosis, the patient presented to our rheumatologic polyclinic with complaints of pain, swelling, warmth, and cyanosis over the fingers of both hand and toe tips. Physical examination revealed swelling, warmth, and tenderness in the proximal interphalangeal (PIP) joints of both hands, Ulnar deviation, and bilateral swan neck deformities. Right is greater than left. “Z” deformities of thumbs (Figure 1(a)) and cyanotic ulcers were seen on the third to fifth digits (Figure 1(c)) and on both digits of feet. All described deformities were reducible. Peripheral pulses were normal. In addition, she had a history of photosensitivity and two gestations with two uneventful births. On further interrogation, the patient was admitted to stopping medical treatment at her own discretion three years before presentation due to exorbitant side effects.

Bottom Line: We report the case of a 38-year-old female patient with systemic lupus erythematosus (SLE) and Jaccoud arthritis (JA) that sequentially developed digital ischemic lesions of the hands.In spite of follow-up treatment with glucocorticoids, immunosuppressant, antiaggregant, and potent vasodilatator agents, a serious progression to digital gangrene over a one-month period was observed.Surprisingly, her nonhealing digital lesions improved after two cycles of rituximab (RTX) administration.

View Article: PubMed Central - PubMed

Affiliation: Department of Rheumatology, Ankara University Medical Faculty, Sihhiye, 06410 Ankara, Turkey.

ABSTRACT
We report the case of a 38-year-old female patient with systemic lupus erythematosus (SLE) and Jaccoud arthritis (JA) that sequentially developed digital ischemic lesions of the hands. In spite of follow-up treatment with glucocorticoids, immunosuppressant, antiaggregant, and potent vasodilatator agents, a serious progression to digital gangrene over a one-month period was observed. Surprisingly, her nonhealing digital lesions improved after two cycles of rituximab (RTX) administration.

No MeSH data available.


Related in: MedlinePlus