Peripheral gangrene during infancy: a rare presentation of systemic lupus erythematosus

Shetty, Varun; Rao, Suchetha; Krishnamurthy, P.; Shenoy, VijendraS

doi:10.1136/adc.85.4.335

Cited by 12 publications

(11 citation statements)

References 2 publications

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“…It is rare to have digital gangrene in children below the age of five years. Yet, it is worth mentioning that there has been one report of digital gangrene in infancy [7].…”

Section: Discussionmentioning

confidence: 99%

Peripheral Gangrene as the Initial Presentation of Systemic Lupus Erythematosus in Emergency Department

Alalawi¹,

Almahroos²,

Albloushi³

2020

Cureus

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Systemic lupus erythematosus (SLE) is a chronic multisystemic autoimmune disease. Among the cutaneous manifestations of SLE, digital gangrene is considered to be very rare. This complication, which may lead to severe ischemic necrosis and amputation, is suggested to be the result of poor perfusion that is usually caused by vasculitis, vasospasm, thromboembolism, or atherosclerosis. Digital gangrene is seen mostly at a late stage of the disease proposing that a long history of SLE is a considered risk factor. Only 0.2% of patients with SLE presented initially as digital necrosis. This is a case report of a 20-year-old Saudi female who presented to the emergency room primarily with acute painful localized dry digital gangrene associated with bilateral lower limbs petechial rash. Her medical history was not suggestive of autoimmune diseases. Serology was positive for SLE. A diagnosis of SLE, lupus nephritis, and vasculitis has been established clinically and serologically. The patient adequately responded to rituximab and steroids as a medical therapy. To our knowledge, cases of acute peripheral gangrene as the initial and only presentation of SLE have rarely been documented in Emergency Medicine.

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“…It is rare to have digital gangrene in children below the age of five years. Yet, it is worth mentioning that there has been one report of digital gangrene in infancy [7].…”

Section: Discussionmentioning

confidence: 99%

Peripheral Gangrene as the Initial Presentation of Systemic Lupus Erythematosus in Emergency Department

Alalawi¹,

Almahroos²,

Albloushi³

2020

Cureus

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“…In SLE a variety of clinical manifestations such as Raynaud’s phenomenon, acrocyanosis, livedo patterns, erythematous or violaceous macules, and papules or necrosis are triggered by heterogeneous pathophysiological mechanisms such as vasospasm, vasculitis, or thromboembolism. Gangrene of the extremities is very rare, occurring in about 1% of SLE patients, and most often affects the upper extremities [8]. Endarteritis, although rare, is an important complication of SLE, in which vasculopathy affects arteries in the extremities.…”

Section: Discussionmentioning

confidence: 99%

“…Shetty et al reported the first case of SLE presenting with foot and hand gangrene in a neonate [8]. …”

Section: Discussionmentioning

confidence: 99%

“…In several studies on SLE in children, gangrene and Raynaud phenomenon have been described as a rare manifestation of SLE [1–5]. In adult patients with SLE, these rare symptoms have been described as the presenting symptoms [6,7], but they have not been described as the first and only presenting symptoms of systemic lupus erythematosus in children, although there is 1 report in the literature showing peripheral gangrene as the presenting symptom in infancy [8]. We present the case of a girl referred to our center, presenting with peripheral gangrene plus Raynaud’s phenomenon, who proved to have SLE.…”

Section: Introductionmentioning

confidence: 99%

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Peripheral gangrene: A rare presentation of systemic lupus erythematosus in a child

2013

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Patient: Female, 12Final Diagnosis: Antiphospholipid positive SLESymptoms: Gangrene • Raynaud’s phenomenon • autoamputation of the terminal phalanx of the second left hand fingerMedication: Prednisolone • mycophenolate mofetil • captoprilClinical Procedure: Renal Biopsy • treatment of lupus nephritis • control of hypertensionSpecialty: Pediatric rheumatologyObjective:Unusual clinical courseBackground:SLE in children has many manifestations. In several studies on SLE in children, gangrene and Raynaud phenomenon have been described as a rare manifestation of SLE during its course in children.Case Report:We present the case of a 12-year-old girl referred to our center, presenting with peripheral gangrene plus Raynaud’s phenomenon, who proved to have SLE. Our patient was treated with steroids and mycophenolate mofetil. She appeared to respond to this combination judging by the disappearance of the digital cyanosis, appearance of extremity pulses, and return of renal function.Conclusions:This case highlights the importance of precise management and awareness of very rare manifestations of a common disease like SLE. Gangrene can be initial symptom of SLE in children. We recommend SLE evolution in all children with gangrene symptom.

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“…However, gangrene is an exceptionally rare form of SLE cutaneous manifestations, accounting for 1.4% of SLE patients [2]. SLE gangrene most often affects the upper extremities and involves no more than the digits [3]. Pathophysiology underlying SLE gangrene includes vasospasm, thromboembolism, and vasculitis [4].…”

Section: Introductionmentioning

confidence: 99%

Systemic Lupus Erythematosus with Multiple Autoimmune Disease Presented with Extensive Peripheral Gangrene

Alzughayyar

Zalloum

Elqadi

et al. 2020

Case Reports in Rheumatology

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Systemic lupus erythematosus (SLE) is an autoimmune disease and can be associated with other autoimmune diseases. SLE usually presents with skin change and rarely presents with gangrene. SLE gangrene usually involves the digits of upper extremities. We report the first case of SLE associated with an extremely rare constellation of neuromyelitis Optica (NMO) and diabetes mellitus type 1, presented with a rare form of the SLE gangrene which involves bilateral lower extremities up to midlegs, a case that has not yet been reported in the literature. Although SLE gangrene may respond to immunosuppressants, it has a high risk of complications that can end up with amputations.

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Peripheral gangrene during infancy: a rare presentation of systemic lupus erythematosus

Cited by 12 publications

References 2 publications

Peripheral Gangrene as the Initial Presentation of Systemic Lupus Erythematosus in Emergency Department

Peripheral Gangrene as the Initial Presentation of Systemic Lupus Erythematosus in Emergency Department

Peripheral gangrene: A rare presentation of systemic lupus erythematosus in a child

Systemic Lupus Erythematosus with Multiple Autoimmune Disease Presented with Extensive Peripheral Gangrene

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