Triple therapy of vincristine, bleomycin and etoposide for children with Kaposi sarcoma: Results of a study in Malawian children

Macken, Marita; Dale, Helen; Moyo, Dominic; Chakmata, Eunice; Depani, Sarita; Israëls, Trijn; Niyrenda, Dalida; Bailey, Simon; Chagaluka, George; Molyneux, Elizabeth

doi:10.1002/pbc.26841

Cited by 7 publications

(26 citation statements)

References 21 publications

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“…Clinical presentation with severe cytopenias is unique and relatively common in childhood KS . Statistically significant associations between clinical presentation with severe thrombocytopenia and elevated KSHV VL and IL‐6 levels were not demonstrated, potentially limited by the small sample size.…”

Section: Discussionmentioning

confidence: 99%

“…Whereas adult Kaposi sarcoma (KS) has been intensely studied since its classification as an AIDS defining malignancy, pediatric KS with or without HIV coinfection has been a neglected disease entity. Although contemporary descriptions of pediatric KS have shed light on the unique clinical features of disease in children, the virologic and biological underpinnings have yet to be explored in children . Caused by KS‐associated herpesvirus (KSHV), also known as human herpesvirus‐8, KS is currently among the most common childhood malignancies in KSHV‐endemic regions including central, eastern and southern Africa .…”

Section: Introductionmentioning

confidence: 99%

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KSHV viral load and Interleukin‐6 in HIV‐associated pediatric Kaposi sarcoma—Exploring the role of lytic activation in driving the unique clinical features seen in endemic regions

El‐Mallawany

Mehta

Kamiyango³

et al. 2018

Intl Journal of Cancer

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Kaposi sarcoma (KS) is among the most common childhood malignancies in central, eastern, and southern Africa. Although its unique clinical features have been established, biological mechanisms related to the causative agent, KS-associated herpes-virus (KSHV), have yet to be explored in children. We performed a prospective observational pilot study to explore associations between KSHV viral load (VL), human interleukin-6 (IL-6) and IL-10 levels, and clinical characteristics of 25 children with KS in Lilongwe, Malawi from June 2013-August 2015. The median age was 6.4 years. Lymphadenopathy was the most common site of KS involvement (64%), followed by skin and oral mucosa (44% each), woody edema (12%), and pulmonary (8%). Baseline samples for plasma KSHV VL, IL-6 and IL-10 analyses were available for 18/25 patients (72%) at time of KS diagnosis. KSHV VL was detectable at baseline in 12/18 (67%) patients, the median baseline IL-6 level was 8.53 pg/mL (range 4.31-28.33), and the median baseline IL-10 level was 19.53 pg/mL (range 6.91-419.69). Seven (39%) patients presented with an IL-6 level > 10 pg/mL (exceeding twice the upper limit of normal). Detectable KSHV VL was significantly associated with lymphadenopathic KS (p = 0.004), while having undetectable KSHV VL was associated with a higher likelihood of presenting with hyperpigmented skin lesions (p = 0.01). Detectable KSHV VL and elevated IL-6 levels are present in a subset of children with KS. Lytic activation of KSHV and associated elevation in KSHV VL may contribute to the unique clinical manifestations of pediatric KS in KSHV-endemic regions of Africa.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

KSHV viral load and Interleukin‐6 in HIV‐associated pediatric Kaposi sarcoma—Exploring the role of lytic activation in driving the unique clinical features seen in endemic regions

El‐Mallawany

Mehta

Kamiyango³

et al. 2018

Intl Journal of Cancer

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“… 26 , 34 In HIV negative children, the male-to-female ratio ranges from 2–7:1. 1 , 22 , 24 , 44 The ratio in HIV positive children is tighter, though it still favors males by as much as 2.5:1, and may be closer to 1:1 in some settings. 18 , 21 – 23 The underlying innate risk factor for endemic KS is as yet unknown, but the male predominance suggests a possible sex-linked genetic predisposition.…”

Section: Epidemiologymentioning

confidence: 99%

“…Prevalence of HIV negative patients among pediatric KS cases ranges from 10–14% in Malawi to 22% in Uganda. 7 , 22 , 24 , 43 , 44 Outside of Africa, HIV-unrelated pediatric KS in the form of transplantation-related and classic disease is extremely rare and has been reported in fewer than 50 patients. 45 …”

Section: Epidemiologymentioning

confidence: 99%

“… 12 – 17 In comparison, several reports describing relatively large cohorts of children and adolescents with KS in sub-Saharan Africa have been published in the last decade. 18 – 24 Ultimately, KS has become one of the most common pediatric malignancies in central, eastern, and southern Africa. 8 Increased awareness of its unique clinical characteristics and therapeutic response is vital in the quest to improve overall outcomes for children with cancer throughout the region.…”

Section: Introductionmentioning

confidence: 99%

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Pediatric Kaposi sarcoma in context of the HIV epidemic in sub-Saharan Africa: current perspectives

El‐Mallawany¹,

McAtee²,

Campbell³

et al. 2018

PHMT

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The global experience with pediatric Kaposi sarcoma (KS) has evolved immensely since the onset of HIV (human immunodeficiency virus). In this review, current perspectives on childhood KS are discussed in the context of the HIV epidemic in sub-Saharan Africa. Endemic (HIV-unrelated) KS was first described over 50 years ago in central and eastern Africa, regions where human herpesvirus-8, the causative agent of KS, is endemic. With the alarming rise in HIV prevalence over the past few decades, KS has become not only the most common HIV-related malignancy in Africa, but also one of the most common overall childhood cancers throughout the central, eastern, and southern regions of the continent. The unique clinical features of pediatric KS that were described in those early endemic KS reports have been re-affirmed by the contemporary experience with HIV-related KS. These characteristics include a predilection for primary lymph node involvement, significant proportions of patients lacking prototypical cutaneous lesions, and the potential for fulminant disease progression. Other clinical features that distinguish childhood KS from adult disease include disease presentation with severe cytopenias, and the common occurrence of childhood KS without severe CD4 count suppression. Distinct clinical heterogeneity in disease presentation and treatment response have been demonstrated. Long-term complete remission and event-free survival can be achieved—especially in children with lymphadenopathic KS—utilizing treatment with antiretroviral therapy plus mild–moderate chemotherapy regimens that are well tolerated, even in low-income settings. A pediatric-specific staging classification and risk-stratification platform have been retrospectively validated, and may help guide therapeutic strategies. With expansion of the HIV treatment infrastructure throughout Africa, coupled with recent developments in establishing comprehensive pediatric oncology programs, there is great potential for improving outcomes for children with KS. Increased awareness of the unique clinical nuances and collaborative evaluations of pediatric-specific treatment paradigms are required to optimize survival for children with KS.

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Vascular Tumors Including Kaposi Sarcoma

Rößler

2022

Pediatric Oncology

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Triple therapy of vincristine, bleomycin and etoposide for children with Kaposi sarcoma: Results of a study in Malawian children

Abstract: Overall survival, event-free survival and quality of life appear to have improved with this three-agent combination chemotherapy; however larger, randomized studies are needed to determine optimal management.

Cited by 7 publications

References 21 publications

KSHV viral load and Interleukin‐6 in HIV‐associated pediatric Kaposi sarcoma—Exploring the role of lytic activation in driving the unique clinical features seen in endemic regions

KSHV viral load and Interleukin‐6 in HIV‐associated pediatric Kaposi sarcoma—Exploring the role of lytic activation in driving the unique clinical features seen in endemic regions

Pediatric Kaposi sarcoma in context of the HIV epidemic in sub-Saharan Africa: current perspectives

Vascular Tumors Including Kaposi Sarcoma

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