Prolonged antibiotic treatment for infected low flow vascular malformations

Wagner, Katy M.; Lokmic, Zerina; Penington, Anthony

doi:10.1016/j.jpedsurg.2017.05.022

Cited by 9 publications

(5 citation statements)

References 17 publications

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“…6,7 Venous malformations and LMs are usually present at birth, although they are not always apparent at this stage. Apart from macrocystic LMs, which may resolve spontaneously after common infections of infancy, 8 the natural history of VMs and LMs consists mainly of slow progressive increases in size and the occurrence of complications, such as pain, bleeding, and oozing, 9,10 which are associated with functional impairment and have a negative impact on quality of life (QoL). 11 Management of VMs and LMs includes observation, physiotherapy, sclerotherapy, complete or partial resection, and medical therapies, such as mammalian target of rapamycin (mTOR) inhibitors.…”

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confidence: 99%

Sirolimus (Rapamycin) for Slow-Flow Malformations in Children

et al. 2021

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IMPORTANCESirolimus is increasingly being used to treat various vascular anomalies, although evidence of its efficacy is lacking.OBJECTIVE To assess the efficacy and safety of sirolimus for children with slow-flow vascular malformations to better delineate the indications for treatment. DESIGN, SETTING AND PARTICIPANTSThis multicenter, open-label, observational-phase randomized clinical trial included 59 children aged 6 to 18 years with a slow-flow vascular malformation who were recruited between September 28, 2015, and March 22, 2018, in 11 French tertiary hospital centers. Statistical analysis was performed on an intent-to-treat basis from December 4, 2019, to November 10, 2020.INTERVENTIONS Patients underwent an observational period, then switched to an interventional period when they received oral sirolimus (target serum levels, 4-12 ng/mL). The switch time was randomized from month 4 to month 8, and the whole study period lasted 12 months for each patient. MAIN OUTCOMES AND MEASURESThe primary outcome was change in the volume of vascular malformations detected on magnetic resonance imaging scan (with centralized interpretation) per unit of time (ie, between the interventional period and the observational period). Secondary outcomes included subjective end points: pain, bleeding, oozing, quality of life, and safety. RESULTS Among the participants (35 girls [59.3%]; mean [SD] age, 11.6 [3.8] years), 22 (37.3%) had a pure venous malformation, 18 (30.5%) had a cystic lymphatic malformation, and 19 (32.2%) had a combined malformation, including syndromic forms. Variations in the volume of vascular malformations detected on magnetic resonance imaging scans associated with the duration period were not overall significantly different between the interventional period and the observational period (all vascular malformations: mean [SD] difference, -0.001 [0.007]; venous malformations: mean [SD] difference, 0.001 [0.004]; combined malformations: mean [SD] difference, 0.001 [0.009]). However, a significant decrease in volume was observed for children with pure lymphatic malformations (mean [SD] difference, -0.005 [0.005]). Overall, sirolimus had positive effects on pain, especially for combined malformations, and on bleeding, oozing, self-assessed efficacy, and quality of life. During sirolimus treatment, 56 patients experienced 231 adverse events (5 serious adverse events, none life-threatening). The most frequent adverse event was an oral ulcer (29 patients [49.2%]).CONCLUSIONS AND RELEVANCE This observational-phase randomized clinical trial allows for clarifying the goals of patients and families when starting sirolimus therapy for children older than 6 years. Pure lymphatic malformations seem to be the best indication for sirolimus therapy because evidence of decreasing lymphatic malformation volume per unit of time, oozing, and bleeding and increasing quality of life was found. In combined malformations, sirolimus significantly reduced pain, oozing, and bleeding. Benefits seemed lower for pure venous malformation...

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confidence: 99%

Sirolimus (Rapamycin) for Slow-Flow Malformations in Children

et al. 2021

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“…As the blood flow in VMs is slow and the hypoplasia of blood vessels and abnormal structures have been shown to cause blood flow congestion [ 6 ], it may be associated with bacterial growth. The incidence of infections among patients with VM was investigated in 620 cases of low-flow vascular malformations [ 7 ]. Of the 21 patients with infections, 19 had lymphatic malformations and two had VM.…”

Section: Discussionmentioning

confidence: 99%

Streptococcal Toxic Shock Syndrome in a Child With Venous Malformation

Yoshida¹,

Asakura²,

Miura³

et al. 2022

Cureus

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We report the case of a child with a venous malformation (VM), in whom streptococcal toxic shock syndrome (STSS) developed from cellulitis. A six-year-old boy with VM of the left lower limb had a fever and left lower limb pain since the afternoon of the day before admission. He presented with swelling, redness, heat, and tenderness on an area extending from the sole of the foot to the lower leg on the left side. Disturbance of consciousness gradually appeared, and he was admitted to the intensive care unit. We administered intravenous antibiotics and an immunoglobulin. On day two of hospitalization, group A hemolytic streptococci were detected in the blood culture. We managed the patient in coordination with a plastic surgeon for consideration of surgical interventions. The local findings subsequently improved to change the antibiotics promptly without debridement, and he was discharged after 14 days of antibiotic therapy. In this case, the VM may have contributed to the worsening of the infection. In children with VM, soft tissue inflammation with local pain and fever must be treated promptly, with the expectation of prompt surgical intervention, because the condition can progress to sepsis and necrotizing fasciitis.

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“…In a supportive or complementary role, medical therapy can be used for general consequences, such as pain, anemia, and infection, as well as addressing hemostatic complications. In patients with infections, long-term antibiotic prophylaxis can be considered [72]. Patients with localized intravascular coagulopathy have been found to benefit from subcutaneous low-molecular weight heparin or oral anticoagulants [73,74].…”

Section: Medical Therapymentioning

confidence: 99%

Congenital Vascular Malformations in Children: From Historical Perspective to a Multidisciplinary Approach in the Modern Era—A Comprehensive Review

Bouwman,

Verhoeven,

Klein

et al. 2024

Children

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Congenital vascular malformations (CVMs) are the result of an aberrant development during embryogenesis. Although these lesions are present at birth, they are not always visible yet. Once symptomatic, patients suffer from pain, bleeding, ulcers, infections or lymphatic leakage, depending on the subtype of vessels involved. Treatment includes conservative management, surgery, sclerotherapy, embolization and pharmacological therapy. The clinical presentation varies widely and treatment can be challenging due to the rarity of the disease and potential difficulties of treatment. This review gives an overview of the historical developments in diagnosis and classification and exposes the key elements of innovations in the past decades on the identification of genetic mutations and personalized treatment. These advances in the field and a multidisciplinary approach are highly valuable in the optimization of clinical care aimed at both curing or stabilizing the CVM and pursuing physical and psychosocial wellbeing.

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Prolonged antibiotic treatment for infected low flow vascular malformations

Cited by 9 publications

References 17 publications

Sirolimus (Rapamycin) for Slow-Flow Malformations in Children

Sirolimus (Rapamycin) for Slow-Flow Malformations in Children

Streptococcal Toxic Shock Syndrome in a Child With Venous Malformation

Congenital Vascular Malformations in Children: From Historical Perspective to a Multidisciplinary Approach in the Modern Era—A Comprehensive Review

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