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Vol. 16. Núm. 5. P1.
Páginas 364-365 (septiembre - octubre 2020)
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Vol. 16. Núm. 5. P1.
Páginas 364-365 (septiembre - octubre 2020)
Images in Clinical Rheumatology
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Post-infectious Digital Ischemia Successfully Treated with Iloprost in a Child
Isquemia digital postinfecciosa infantil tratada con éxito con iloprost
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3584
Teresa Martins-Rochaa,
Autor para correspondencia
teresa.asmr@gmail.com

Corresponding author.
, José Pedro Matiasb, Margarida Vicente Ferreirab, Bárbara Motab, Iva Britoa,c
a Rheumatology Department, Centro Hospitalar de São João, Porto, Portugal
b Department of Pediatrics, Centro Hospitalar de São João, Porto, Portugal
c Rheumatology Department, Faculty de Medicine University of Porto (FMUP), Portugal
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We report a case of a 14-month-old girl, with no relevant pathological history, which presented additive bullous lesions on her hands and feet fingertips. She had no previous burn, trauma or other associated events. One week before, she was diagnosed with Acute Otitis Media and was treated with amoxicillin/clavulanic acid for 8 days. Later on, the fingertip lesions progressively evolved into necrotic lesions and, after two months of the initial presentation, she was observed at our hospital. Objectively, she was afebrile and presented digital plaques of dry necrosis, with edema and redness of the distal end of the affected fingers, without fluctuation or drainage of any content (Fig. 1). Peripheral arterial pulses were symmetric and no vascular or heart bruits were audible. As iloprost has been described to be safe and effective for the treatment of ischemic digits in children, intravenous iloprost (0.4ng/kg/min for 5 days followed by 0.6ng/kg/min) and acetylsalicylic acid were started.1-4

Figure 1.

Ischemic lesions at the admission (A), at the 13th (B) and at the 21st day (C) of therapy with intravenous iloprost.

(0.23MB).

Concerning laboratory studies, she had CRP (50mg/L) and ESR (60mm/1st hour) elevation; negative serologies for EBV, Herpes simplex, CMV, parvovirus and Mycoplasma pneumoniae; negative antistreptolysin O titer and blood cultures. Pro-thrombotic and immunological studies (complement levels, rheumatoid factor, ANA, anti-ENA, anti-dsDNA, ANCA and antiphospholipid antibodies) were unremarkable. Echocardiogram, chest and hands X-rays, and abdominal ultrasound were normal.

She completed 21 days of iloprost and gradually presented resolution of the fingertip lesions. Considering the previous infection and the diagnostic work-up, we considered the digital ischemia in a probable post-infectious context.

Digital ischemia is described in meningococcal infection, which can present extrameningeal involvement in up to 19% of patients.5 Additionally, few cases were also reported with other infectious agents.1,6 Among others, treatment with antiplatelet drugs, topical nitrates and sympathetic blockade is described.7 Besides our report, favorable response to iloprost was documented in 4 children with post-infectious digital ischemia.1,4,8

Funding

No specific funding was received from any bodies in the public, commercial or not-for-profit sectors to carry out the work described in this manuscript.

Conflicts of interest

The authors declare they have no conflicts of interest.

References
[1]
P. Garcia Soler, G. Milano Manso, J.M. Camacho Alonso, J. Manuel Gonzalez.
[Iloprost for the treatment of digital ischaemia in pediatric septic shock].
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[2]
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Iloprost treatment for refractory Raynaud's phenomenon in two infants.
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Safety and efficacy of iloprost for the treatment of ischaemic digits in paediatric connective tissue diseases.
Rheumatology (Oxford), 43 (2004), pp. 229-233
[4]
K. Siddiqui, A.R. Razak, B. Kneafsey, N. Delanty.
Use of prostacyclin (iloprost) in digital vasculitis secondary to meningococcaemia.
J Neurol Neurosurg Psychiatry, 75 (2004), pp. 506
[5]
E.A. Kirsch, R.P. Barton, L. Kitchen, B.P. Giroir.
Pathophysiology, treatment and outcome of meningococcemia: a review and recent experience.
Pediatr Infect Dis J, 15 (1996), pp. 967-978
[6]
Y. Singh, T.R. Khan, K.P. Kushwaha.
Spontaneous pneumothorax with symmetrical digital gangrene -a rare complication of chickenpox.
Indian J Pediatr, 80 (2013), pp. 706-707
[7]
F. Leclerc, S. Leteurtre, R. Cremer, C. Fourier, A. Sadik.
Do new strategies in meningococcemia produce better outcomes?.
[8]
R.A. Seaton, D. Nathwani, J. Dick, D. Smith.
Acute meningococcaemia complicated by late onset gastrointestinal vasculitis.
J Infect, 41 (2000), pp. 190-191
Copyright © 2018. Elsevier España, S.L.U. and Sociedad Española de Reumatología y Colegio Mexicano de Reumatología
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