Partial lipodystrophy in patients who have undergone hematopoietic stem cell transplantation during childhood: an institutional cross-sectional survey

Adachi, Masanori; Oto, Yuji; Muroya, Koji; Hanakawa, Junko; Asakura, Yumi; Goto, Hiroaki

doi:10.1297/cpe.26.99

Cited by 29 publications

(25 citation statements)

References 24 publications

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“…A total of 13,442 subjects were represented by the included studies. The majority of the studies were conducted in the following developed countries ( n = 52, 88%): Japan [ 27 , 29 , 30 , 33 , 35 , 38 – 40 , 42 , 43 , 45 , 49 , 54 , 57 , 59 , 61 , 63 , 64 , 67 – 69 , 78 , 80 ] ( n = 24, 41%), South Korea [ 24 , 26 , 31 , 32 , 34 , 37 , 46 , 53 , 55 , 56 , 58 , 60 , 62 ] ( n = 13, 22%), Hong Kong [ 41 , 50 – 52 , 72 – 75 , 76 , 77 , 79 , 81 , 82 ] ( n = 12, 20%), Taiwan [ 47 , 48 ] ( n = 2, 3%), Singapore [ 36 ] ( n = 1, 1.5%). Only seven studies (12%) were conducted in developing countries [ 25 , 28 , 44 , 65 , 66 , 70 , 75 ] (Thailand and Malaysia).…”

Section: Resultsmentioning

confidence: 99%

Clinical ascertainment of health outcomes in Asian survivors of childhood cancer: a systematic review

Poon

Peng

et al. 2019

J Cancer Surviv

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Purpose Survivorship in children with cancer comes at a cost of developing chronic treatment-related complications. Yet, it is still an under-researched area in Asia, which shares the largest proportion of the global childhood cancer burden given its vast population. This systematic review summarizes existing literature on clinically ascertained health outcomes in Asian survivors of childhood cancer. Methods A search was conducted on Ovid Medline and EMBASE for studies that focused on survivors of childhood cancer from countries in East and Southeast Asia; adopted post-treatment clinical ascertainment of organ-specific toxicities or/and secondary malignancy. Studies were excluded if health outcomes were assessed during the acute treatment. Results Fifty-nine studies, enrolling a total of 13,442 subjects, were conducted on survivors of leukemia (34%), CNS tumor (14%), and cohorts of survivors with heterogeneous cancer diagnoses (52%). The studies used different medical evaluation methods to assess cardiovascular (15%), metabolic and infertility (32%), and neurological/neurocognitive (20%) outcomes in survivors. The collective findings suggest potential differences in the prevalence of certain late effects (e.g., secondary malignancy and obesity) among Asian and non-Asian populations, which may reflect differences in treatment regimens, practice, genetic variations, or/and socioeconomic disparity. Conclusions We recommend developing collaborative initiatives to build a regional repository of systematically assessed health outcomes and biospecimens to investigate treatment, social-environmental and genetic predictors, and interventions for late effects in this population. Implications for Cancer Survivors The existing types of chronic health problems identified in this review suggest the need for active screening, better access to survivorship care, and promotion of protective health behavior in Asia. Keywords Childhood cancer. Late effects. Survivorship. Risk-based. Asian. Organ toxicity The review was presented at the 8th Nursing Symposium on Cancer Care, Hong Kong (24th to 25th May), and was awarded the Best Poster Award.

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Section: Resultsmentioning

confidence: 99%

Clinical ascertainment of health outcomes in Asian survivors of childhood cancer: a systematic review

Poon

Peng

et al. 2019

J Cancer Surviv

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“…A final total of 20 papers were included-seven controlled studies [1,10,[18][19][20][21][22], five uncontrolled studies [23][24][25][26][27] and eight case reports/series [28][29][30][31][32][33][34][35]. Only one study included an intervention [23].…”

Section: Search Resultsmentioning

confidence: 99%

“…Three studies found significantly higher body fat: body fat % compared to short stature controls [19] and healthy controls [20] and whole body fat mass z score compared to reference controls [1]. Data from Adachi et al [26] suggests BMI may be lower than leukaemic controls with no TBI, and, although significance could not be tested, within the normal range for age.…”

Section: Total Body Fatmentioning

confidence: 98%

Body composition after allogeneic haematopoietic cell transplantation/total body irradiation in children and young people: a restricted systematic review

et al. 2020

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Purpose To collate evidence of changes in body composition following treatment of leukaemia in children, teenagers and young adults (CTYA, 0-24 years) with allogeneic haematopoietic stem cell transplant and total body irradiation (HSCT+TBI). Methods Papers were identified by searching Medline and Google Scholar, reference lists/citations and contacting key authors, with no date or language restrictions. Inclusion criteria were as follows: leukaemia, HSCT+TBI, aged ≤ 24 years at HSCT and changes in body composition (total fat, central adiposity, adipose tissue function, muscle mass, muscle function). Quality was assessed using a brief Newcastle-Ottawa scale.Results Of 900 papers, 20 were included: seven controlled, five uncontrolled studies and eight case reports. Study quality appeared good. There was little evidence of differences in total fat/weight for HSCT + TBI groups (compared to healthy controls/population norms/short stature controls). There was some evidence of significantly higher central adiposity and differences in adipose tissue function (compared to leukaemic/non-leukaemic controls). Muscle mass was significantly lower (compared to healthy/obese controls). Muscle function results were inconclusive but suggested impairment. Case reports confirmed a lipodystrophic phenotype. Conclusions Early remodelling of adipose tissue and loss of skeletal muscle are evident following HSCT + TBI for CTYA leukaemia, with extreme phenotype of overt lipodystrophy. There is some evidence for reduced muscle effectiveness. Implications for Cancer Survivors Body composition changes in patients after HSCT + TBI are apparent by early adult life and link with the risk of excess cardiometabolic morbidity seen in adult survivors. Interventions to improve muscle and/or adipose function, perhaps utilizing nutritional manipulation and/or targeted activity, should be investigated.

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“…The development of lipodystrophy after whole body irradiation in preparation for bone marrow transplant (49,50,51,52,53,54), following cranial irradiation and as a result of hypothalamic tumors (55) has been described fat loss to varying extents (56,57). These unusual forms of acquired lipodystrophy will be reviewed in a specific review in the near future.…”

Section: Other Causes Of Acquired Lipodystrophymentioning

confidence: 99%

Current Diagnosis, Treatment and Clinical Challenges in the Management of Lipodystrophy Syndromes in Children and Young People

Özen

Akıncı

Oral

2020

Jcrpe

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Lipodystrophy is a heterogeneous group of disorders characterized by lack of body fat in characteristic patterns, which can be genetic or acquired. Lipodystrophy is associated with insulin resistance that can develop in childhood and adolescence, and usually leads to severe metabolic complications. Diabetes mellitus, hypertriglyceridemia, and hepatic steatosis ordinarily develop in these patients, and most girls suffer from menstrual abnormalities. Severe complications develop at a relatively young age, which include episodes of acute pancreatitis, renal failure, cirrhosis, and complex cardiovascular diseases, and all of these are associated with serious morbidity. Treatment of lipodystrophy consists of medical nutritional therapy, exercise, and the use of anti-hyperglycemic and lipid-lowering agents. New treatment modalities, such as metreleptin replacement, promise much in the treatment of metabolic abnormalities secondary to lipodystrophy. Current challenges in the management of lipodystrophy in children and adolescents include, but are not limited to: (1) establishing specialized centers with experience in providing care for lipodystrophy presenting in childhood and adolescence; (2) optimizing algorithms that can provide some guidance for the use of standard and novel therapies to ensure adequate metabolic control and to prevent complications; (3) educating patients and their parents about lipodystrophy management; (4) improving patient adherence to chronic therapies; (5) reducing barriers to access to novel treatments; and (5) improving the quality of life of these patients and their families.

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Partial lipodystrophy in patients who have undergone hematopoietic stem cell transplantation during childhood: an institutional cross-sectional survey

Cited by 29 publications

References 24 publications

Clinical ascertainment of health outcomes in Asian survivors of childhood cancer: a systematic review

Clinical ascertainment of health outcomes in Asian survivors of childhood cancer: a systematic review

Body composition after allogeneic haematopoietic cell transplantation/total body irradiation in children and young people: a restricted systematic review

Current Diagnosis, Treatment and Clinical Challenges in the Management of Lipodystrophy Syndromes in Children and Young People

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