Shuichi Ozono

Factors related to posttraumatic stress in adolescent survivors of childhood cancer and their parents

Goals of workThe purpose of this study was to investigate factors related to severe posttraumatic stress symptoms (PTSS) in adolescent survivors of childhood cancer and their parents.Materials and methodsEighty-nine families (88 adolescent survivors of childhood cancer, 87 mothers, 72 fathers) completed a self-report questionnaire. Multivariate logistic regression analyses were performed using the following risk factors for severe PTSS: trait anxiety, family functioning, demographic and medical variables.Main resultsSevere PTSS were reported by 10.9% (n=9) of the survivors, 20.7% (n=18) of the mothers, and 22.2% (n=16) of the fathers. Preliminary analyses found significant correlations of PTSS between mother–survivor (Spearman’s γ=0.377, p<0.01) and mother–father (Spearman’s γ=0.483, p<0.01). The results of multivariate analyses indicated that higher trait anxiety [odds ratio (OR):1.16; 95% confidence interval (CI): 1.03–1.31; p<0.05] and having medical sequelae (OR: 5.85; 95% CI:1.02–33.72; p<0.05) were significant factors related to PTSS for survivors. For mothers, the significant PTSS-related factors were: higher trait anxiety (OR:1.13; 95% CI:1.04–1.23; p<0.01); 5- to 9-year interval from the first diagnosis to the present investigation, compared to more than a 10-year interval (OR: 6.45; 95% CI:1.67–24.89; p<0.01); and a relatively lower rating on “roles” of family functioning (OR: 12.34; 95% CI:1.11–136.97; p<0.05). For fathers, trait anxiety was a significant related factor (OR: 1.07; 95% CI:1.01–1.14; p<0.05).ConclusionsSurvivors and their parents suffered from PTSS after long interval from completion of treatment, and PTSS-related factors varied for each family member. Appropriate allocation of responsibility for family functioning may promote the ability to decrease PTSS, especially for mothers.

Social outcomes and quality of life of childhood cancer survivors in Japan: a cross-sectional study on marriage, education, employment and health-related QOL (SF-36).

Social outcomes and quality of life (QOL) of childhood cancer survivors (CCSs) remain unknown in Japan. We investigated these outcomes in young adult CCSs compared to those of their siblings in Japan, and analyzed the association between social outcome and SF-36 health survey subscale scores. Between 2007 and 2009, we performed a cross-sectional survey using self-rating questionnaires. We estimated social outcomes and health-related QOL by performing the SF-36 in each group: CCSs with or without stem cell transplantation (SCT)/radiotherapy (RT) and their siblings. Adjusted odds ratios for outcomes of interest were estimated using logistic regression analysis. Questionnaires from 185 CCSs and 72 CCS’s siblings were analyzed. There were no differences in educational attainment or annual income. The SF-36 subscale scores of CCSs with SCT and RT were significantly lower than those of siblings in physical functioning (PF) (p < 0.001 and 0.003, respectively) and general health (GH) (both p = 0.001). Lower PF scores correlated with recurrence (p = 0.041) and late effects (p = 0.010), and poor GH scores with late effects (p = 0.006). The CCSs had made efforts to attain educational/vocational goals; however, a significant proportion of CCSs who had experienced late effects remain at increased risk of experiencing diminished QOL.

Psychological distress related to patterns of family functioning among Japanese childhood cancer survivors and their parents

Objective: Family functioning appears to be a predictor of psychological distress among childhood cancer survivors and their family members; however, relatively little is known about patterns in those families that are psychologically at‐risk. The purpose of this study was to identify distinct clusters of families that include childhood cancer survivors, and to evaluate differences between the clusters with respect to anxiety, depression, and post‐traumatic stress symptoms (PTSS).

Wiskott-Aldrich syndrome presenting with a clinical picture mimicking juvenile myelomonocytic leukaemia.

Wiskott–Aldrich syndrome (WAS) is a rare X‐linked immunodeficiency caused by defects of the WAS protein (WASP) gene. Patients with WAS typically demonstrate micro‐thrombocytopenia.

Family functioning and psychological distress among Japanese breast cancer patients and families

Goals of workThe purpose of this study was to develop a typology of family functioning in the families of breast cancer patients.Patients and methodsSeventy-four families (189 individuals: 74 patients, 54 spouses, 46 offspring, and 15 other relatives) completed self-report questionnaires. Perception of family functioning was assessed using the family relationship index (FRI) and its three dimensions (cohesiveness, expressiveness, and conflict) and was classified into groups by a cluster analytic approach. Psychological distress was assessed using the Zung self-rating depression scale (SDS) and the Zung self-rating anxiety scale (SAS).Main resultsCluster analysis yielded three groups of patients and their family members: one cluster with high cohesiveness, high expressiveness, and low conflict (“supportive” type: n=64); a second group with low cohesiveness, low expressiveness, and high conflict (“conflictive” type: n=65); and a third group with limited cohesiveness, limited expressiveness, and low conflict (“intermediate” type: n=60). Analysis of variance revealed that conflictive families manifested the highest level of depression and anxiety among these clusters.ConclusionsTypology of family functioning can identify psychologically at-risk families. A family-focused approach can help to reduce psychological distress, especially in conflictive families.

Regional cerebral blood flow changes in early-onset anorexia nervosa before and after weight gain

To investigate the changes of regional cerebral blood flow (rCBF) in early-onset anorexia nervosa (AN) before and after weight gain, we examined resting rCBF using single photon emission computed tomography (SPECT) with [(123)I]iodoamphetamine ((123)I-IMP). Ten female children with AN (mean age 13.2 years old) participated in this study. SPECT examinations were performed in all patients twice at the beginning of treatment and after weight gain. The mean body mass index (BMI) was changed from 13.1 to 16.6 during 4 months treatment period. Automatic voxel-based analysis of the images was carried out using statistical parametric mapping (SPM) software. Relatively increased rCBF in the bilateral parietal lobe and limbic lobe including the posterior cingulate cortex (PCC) were observed after weight gain in early-onset AN. There was no significant decrease in the rCBF after weight gain. A significant positive correlation was observed between BMI and rCBF in the right thalamus, right parietal lobe, and right cerebellum. These results suggested that weight gain during the process of recovery from early-onset AN might activate specific brain regions which are possibly relevant to the pathophysiological aspects of the disorder.

Juvenile myelomonocytic leukemia characterized by cutaneous lesion containing Langerhans cell histiocytosis-like cells

We present a 1-year-old boy who developed a cutaneous lesion on the trunk and hepatosplenomegaly. Laboratory examination showed leukocytosis with peripheral blasts, atypical monocytosis, anemia, hyper IgG, and a mild elevation of C-reactive protein. Clinical features and skin biopsy findings matched the diagnostic criteria of both juvenile myelomonocytic leukemia (JMML) and Langerhans cell histiocytosis (LCH). Histopathology revealed atypical mononuclear cells that had infiltrated around vessels throughout the dermis in a skin biopsy specimen. These cells were CD1a (+), S-100 (+), CD68 (+), CD207 (−), lysozyme (+), and myeloperoxidase (−). The diagnosis of JMML was confirmed by detection of spontaneous colony formation and granulocyte–macrophage colony-stimulating factor hypersensitivity in vitro, and a somatic NRAS point mutation. Transplantation of bone marrow from an HLA-matched unrelated donor was performed, and the marrow was successfully engrafted. The cutaneous lesion and hepatosplenomegaly were improved at the time of discharge. It is often difficult to distinguish between JMML and LCH-like infiltrates by assessing clinical and light microscopic features of various cutaneous lesions. In the current case, molecular biological analysis enabled us to develop a precise diagnosis.

Prefrontal brain function in children with anorexia nervosa: A near-infrared spectroscopy study

To investigate the prefrontal hemodynamic response during a cognitive task in childhood anorexia nervosa (AN), we measured regional cerebral blood volume changes in terms of changes in hemoglobin concentrations [Hb], using near-infrared spectroscopy (NIRS). Sixteen females with AN (mean age 14.2 years old) and 12 age-matched healthy female control subjects (mean age 14.3 years old) participated in this study. Waveform patterns for [Hb] during the word fluency task differed between the two groups, although their task performances showed no significant difference. In the control group, the [total-Hb] and [oxy-Hb] immediately increased and the [deoxy-Hb] immediately decreased after the beginning of the task and gradually reached the baseline level after the end of the task. The patients with AN were consistently characterized by an unchanged or less fluctuating response pattern of [total-Hb], [oxy-Hb] and [deoxy-Hb] during the task and rest periods. In the AN group, subjects with higher Eating Attitudes Test (EAT-26) scores showed higher [oxy-Hb] during the task. On the other hand, in the control group, subjects with higher EAT-26 scores showed lower [oxy-Hb] during the task. The grand waveforms of each [Hb] during a motor activation task, which was applied as a control task, did not differ significantly between two groups. The different prefrontal hemodynamic responses might indicate that AN subjects might apply fewer brain circuits or fewer neurons per circuit during cognitive tasks and might use different brain circuits in relation to their preoccupation with eating behaviors.

Medical visits of childhood cancer survivors in Japan: A cross-sectional survey

Background:  Although more children with cancer continue to be cured, these survivors experience various late effects. Details of the medical visit behaviors of childhood cancer survivors (CCS) in adulthood remain to be elucidated.

Characteristic prefrontal blood volume patterns when imaging body type, high-calorie food, and mother–child attachment in childhood anorexia nervosa: A near infrared spectroscopy study

The aim of this study was to investigate the prefrontal activation in childhood anorexia nervosa (AN) patients when imaging symptom-provocative factors such as body type, high-calorie food, and attachment between mother and child. The prefrontal activations during each task, in terms of blood volume changes, were measured by near infrared spectroscopy. Twelve females with childhood AN (mean age, 14.4 years old) and 13 age-matched healthy female controls (mean age, 14.3 years old) participated in this study. Both groups showed increased prefrontal blood volume when viewing images of each symptom-provocative factor. Unexpectedly, there was no significant difference in the prefrontal blood volume increases between the control group and the childhood AN group when viewing images of slender and obese body types and high-calorie food. On the other hand, images of mother-child attachment resulted in significantly greater increases in prefrontal blood volume in the childhood AN group than in the control group. These results indicated that prefrontal activation in AN might be associated with imaging attachment between mother and child, but not associated with imaging body type or high-calorie food.