[THE CASE REPORT OF A 8 YEARS OLD BOY WITH COCHINEAL ALLERGY].

BACKGROUND: Cochineal dyes are used as additives in various foods for the purpose of red coloration. On the other hand, although it has been reported as a causative agent of immediate allergy, most of them are adult women. We report a case of an 8-year-old boy who developed a cochineal allergy.Current disease history: He has been suffering from atopic dermatitis, bronchial asthma, and food allergies since childhood. At the age of seven, he experienced an unknown anaphylaxis reaction twice. When he was 8 years old, he ate a frankfurter containing hypoallergenic cochineal for the first time; cold sweat, intraoral discomfort, respiratory distress, and urticaria appeared throughout the body. His skin prick tests were positive, with a result of 2+to frankfurter and cochineal dyes (color value 0.1 and 0.01). In the immunoblot assay, binding of IgE antibody was observed with CC38K (the main component of cochineal) and a protein of approximately 80-200 kDa in the high molecular weight region. DISCUSSION: We report a case of anaphylaxis with hypoallergenic cochineal onset in a school-age boy.

Cochineal dye-induced immediate allergy: Review of Japanese cases and proposed new diagnostic chart.

BACKGROUND: Cochineal dye is used worldwide as a red coloring in foods, drinks, cosmetics, quasi-drugs, and drugs. The main component of the red color is carminic acid (CA). Carmine is an aluminum- or calcium-chelated product of CA. CA and carmine usually contain contaminating proteins, including a 38-kDa protein thought to be the primary allergen. Severe allergic reactions manifest as anaphylaxis. The aim of this study was to review all Japanese reported cases and propose useful diagnostic chart. METHODS: All reported Japanese cases of cochineal dye-induced immediate allergy were reviewed, and newly registered cases were examined by skin prick test (SPT) with cochineal extract (CE) and measurement of CE and carmine-specific serum IgE test. Two-dimensional (2D) western blotting using patient serum was conducted to identify the antigen. RESULTS: Twenty-two Japanese cases have been reported. SPT and the level of specific IgE test indicated that six cases should be newly registered as cochineal dye allergy. All cases were adult females, and all cases except three involved anaphylaxis; 13 cases involved past history of local symptoms associated with cosmetics use. Japanese strawberry juice and fish-meat sausage, and European processed foods (especially macarons made in France) and drinks were recent major sources of allergen. 2D western blotting showed that patient IgE reacted to the 38-kDa protein and other proteins. Serum from healthy controls also weakly reacted with these proteins. CONCLUSIONS: SPT with CE and determination of the level of CE and carmine-specific IgE test are useful methods for the diagnosis of cochineal dye allergy.

Cutaneous histopathology of the tick-bite region in severe fever with thrombocytopenia syndrome.

A case of severe fever with thrombocytopenia syndrome (SFTS) in which a skin biopsy from the tick-bite region was analyzed is reported. The patient was a 72-year-old woman who developed fever and thrombocytopenia after a tick bite. SFTS was diagnosed from polymerase chain reaction (PCR) analysis of a blood sample. Histopathological analysis of a skin biopsy specimen from the tick-bite region showed CD20-positive perivascular and interstitial immunoblastic cells, which were positive to anti-SFTS virus (SFTSV) nucleoprotein antibody. In addition, SFTSV RNA was detected by real-time PCR from this biopsy specimen. Moreover, hemophagocytosis was also found in the tick-bite region. To the best of our knowledge, this is the first report to analyze the details of the tick-bite region of skin in SFTS, and the first to detect virus-infected cells in the skin. The present findings may help elucidate the mechanisms of entry of SFTSV.

Related visual impairment to mother-infant interaction and development in infants with bilateral retinoblastoma.

PURPOSE: This study was conducted with infants diagnosed with bilateral retinoblastoma (RB) and their mothers. It explored characteristics of the mother-infant interaction, the infants' developmental characteristics and related risk factors. METHOD: Cross-sectional statistical analysis was performed with 18 dyads of one-year-old infants with bilateral RB and their mothers. RESULTS: Using the Japanese Nursing Child Assessment Teaching Scale (JNCATS) results showed that infants with RB had significantly lower scores compared to normative Japanese scores on all of the infants' subscales and "Child's contingency" (p < 0.01). Five infants with visual impairment at high risk of developmental problems had a pass rate of 0% on six JNCATS items. There were positive correlations between Developmental quotients (DQ) and JNCATS score of "Responsiveness to caregiver" (ρ = 0.50, p < 0.05) and DQ and "Child's contingency" (ρ = 0.47, p < 0.05). CONCLUSIONS: Infants with visual impairment were characterized by high likelihood of developmental delays and problematic behaviors; they tended not to turn their face or eyes toward their mothers, smile in response to their mothers' talking to them or the latter's changing body language or facial expressions, or react in a contingent manner in their interactions. These infant behaviors noted by their mothers shared similarities with developmental characteristics of children with visual impairments. These findings indicated a need to provide support promoting mother-infant interactions consistent with the developmental characteristics of RB infants with visual impairment.

Hereditary lactate dehydrogenase M-subunit deficiency with late-developing pustular psoriasis-like lesions.

Hereditary lactate dehydrogenase (LDH) M-subunit deficiency is very rare and we have found reports of close to a dozen cases in the published work, two of which were associated with pustular psoriasis-like lesions. We report a third case of pustular psoriasis-like eruptions associated with LDH M-subunit deficiency, which occurred 24 years after the diagnosis of LDH M-subunit deficiency. These cases indicate that abnormal activity of LDH can induce pustular psoriatic lesions in the long term. Some patients with symptoms of hereditary LDH M-subunit deficiency have antecedent annular scaly plaque lesions, that resemble psoriatic lesions. We discuss a hypothesis to explain this scenario.

Three cases of pigmented cosmetic dermatitis-like eruptions associated with primary Sjögren's syndrome or anti-SSA antibody.

Pigmented cosmetic dermatitis-like (Riehl's melanosis-like) pigmentation was reported in three of 27 patients with primary Sjögren's syndrome. But case reports of such eruptions are rare. We describe three cases of such eruptions associated with primary Sjögren's syndrome or anti-SSA antibody and possible associations with specific types of human leukocyte antigen (HLA) and infiltrating lymphocytes. These middle-aged Japanese women had reticular facial pigmentation and histopathological examination revealed interface dermatitis, melanophages, and dense lymphocytic infiltration around hair follicles and sweat ducts. HLA typing revealed common antigenic equivalents or genetic typing of HLA-A2, DR52, DPA1(02:02) and DPB1(05:01). Immunohistochemical staining revealed major subsets of T cells to be CD8 and CD45RO. Some Foxp3- and few IL17-positive cells were found in strong contrast to the major CD4 subset of infiltrated T cells in annular erythema associated with Sjögren's syndrome. Apparently, our patients' pigmentation represented a specific etiology associated with primary Sjögren's syndrome or anti-SSA antibody.

Comparative analysis of mamushi (Gloydius blomhoffii) bite patients indicates that creatinine kinase levels/white blood cell count trends reflect severity.

Aim: There has been no indicator that allows an early quantitative evaluation of the severity of a mamushi snake (Gloydius blomhoffii) bite. Because the number of severe mamushi bite cases is much fewer than non-severe cases, a formal case-control study is difficult. Therefore, we tried to generate a preliminary quantitative, real-time index for its severity by referring to published reports of severe mamushi bite cases. Methods: We enrolled patients who presented with a mamushi bite and visited our outpatient clinic. Severe cases were collected from published works. Creatinine kinase levels and white blood cell counts of non-severe and severe cases were compared and analyzed. Results: There was a lag time of 10 h before the creatinine kinase level began to rise. The speed of the increase was higher in severe cases than in non-severe cases, and severe cases were recognized as those showing speeds of above 250 IU/L/h. White blood cell counts increased earlier than creatinine kinase levels without any lag time. Severe cases were recognized as those with the counts of over 1,000 × (h) + 6,000 [/µL] before 5 h and 300 × (h) + 10,000 [/µL] after 5 h. Conclusion: We herein present the creatinine kinase level and white blood cell count trends and demonstrate preliminary cut-off equations. The trends for both parameters serve as quantitative indicators of the severity of a mamushi bite until a large scale case-control study is achieved.

Effect of Early Intervention to Promote Mother - Infant Interaction and Maternal Sensitivity in Japan: A Parenting Support Program based on Infant Mental Health.

This study investigated the effects of the Japanese Early Promotion Program (JEPP), which is based on the Infant Mental Health (IMH) program. The JEPP aims to promote mother-infant interactions by enhancing the mother's ability to respond appropriately her child. Mothers in the JEPP group (n = 15) received support from IMH nurses in a pediatric clinic until their infants reached 12 months of age. The nurses provided positive feedback that emphasized strength of parenting, and assisted the mothers in understanding the construct of their infants. Mother-infant interactions and mother's mental health status were assessed at intake (1-3 months), and at 6, 9, and 12 months of infants' age. The JEPP group data were compared with cross-sectional data of the control group (n = 120). Although JEPP dyads were not found to be significantly different from the control group in general dyadic synchrony, both before and after intervention, JEPP mothers significantly improved their ability to understand their infant's cues and to respond promptly. In the JEPP group, unresponsiveness to infants was reduced in mothers, while infants showed reduced passiveness and enhanced responsiveness to the mother. Furthermore, the intervention reduced the mothers' parenting stress and negative emotions, thereby enhancing their self-esteem.

A Prospective Study of Factors Related to Mother-Infant Interaction in One-year-old Infants with Retinoblastoma.

This study aimed to clarify the relationships among developmental characteristics of retinoblastoma (RB) infants, mother-infant interaction, and mental health of mothers. Prospective studies were conducted twice with 13 dyads of mothers and infants who were between one (Time 1) and two years old (Time 2). Cross-sectional and longitudinal analyses were performed. The mean Developmental Quotients (DQ) significantly decreased between Time 1 and Time 2 (p < .05). Four (30.8%) infants showed DQ p < 85 at Time 1. They showed a DQ below the borderline at Time 2 and their developmental changes were different from RB infants in the normal DQ range. Infant Behavior Checklist-R (IBC-R) scores negatively correlated with DQ (p < .05). The mothers' Japanese Nursing Child Assessment Teaching Scale (JNCATS) scores were significantly higher than the normative mean. The IBC-R scores negatively correlated with mothers' JNCATS scores at Time 1 (ρ = -.66, p < .05) and positively correlated with the child domain scores on the Parenting Stress Index at Time 2 (ρ = .62, p < .05). Some RB infants showed a developmental delay. Mother-child interactions were negatively affected and mothers recognized their infants' problem. The support needs to be provided in early infancy.

Association between HLA-DRB1*0405, -DQB1*0401 and -DQA1*0303 alleles and lamotrigine-induced cutaneous adverse drug reactions. A pilot case-control study from Japan.

BACKGROUND: Human leukocyte antigen (HLA) genotypes in lamotrigine -induced (LTG-induced) cutaneous adverse drug reactions (cADRs) have been described in several reports but controversy remains even for a given ethnic group. We attempted to clarify a possible association between LTG-induced cADRs and HLA alleles in Japanese patients. METHOD: Sixteen subjects, including eight patients with LTG-induced cADRs and eight LTG-tolerant controls were included in this study. All eight patients with LTG-induced cADRs gave positive results in a drug-induced lymphocyte stimulation test (DLST) with LTG. We performed HLA-typing for HLA-A, -B, -C, -DRB1, -DQA1, -DQB1, -DPA1 and -DPB1, using PCR with sequence-specific oligonucleotide probes and multiple analyte profiling (xMAP) technology (Luminex System; Luminex Corporation, Austin, TX). We examined differences between allele frequencies in our two groups of subjects and the allele frequencies in the general Japanese population. RESULTS: The frequencies of HLA-DRB1*0405, and HLA-DQB1*0401 alleles were higher in our LTG-cADRs patients than the reference frequencies in the general Japanese population. We also detected HLA-DQA1*0303 frequently in our LTG-cADRs patients, but data for this allele in the Japanese population was not available. Our observation was presumably due to the linkage disequilibrium among the three alleles. The haplotype frequency of HLA-DRB1*0405, DQB1*0401 and DQA1*0303 in our LTG-cADRs subjects was also different from the corresponding haplotype frequency in the database for the Japanese population and the difference was statistically significant. One patient with the HLA-DRB1*0405, -DQB1*0401 and DQA1*0303 haplotype was safely re-treated with LTG after results of a DLST with LTG ceased to be positive about 4 months after discontinuation of LTG. LIMITATIONS: Our analysis included only 16 patients. Associations between LTG-induced cADRs and specific HLA loci will have to be confirmed in larger studies. CONCLUSIONS: LTG-induced cADRs are associated with HLA-DRB1*0405, -DQB1*0401 and -DQA1*0303.

Pustular-type drug-induced hypersensitivity syndrome/drug reaction with eosinophilia and systemic symptoms due to carbamazepine with systemic muscle involvement.

Drug-induced hypersensitivity syndrome (DIHS)/drug reaction with eosinophilia and systemic symptoms (DRESS) is a severe reaction usually associated with maculopapular eruptions and systemic involvement. Here we report the first case, to our knowledge, of DIHS/DRESS due to carbamazepine with acute generalized pustular bacterid-like (AGPB-like) eruptions and skeletal muscle involvement. Reviewing our case and the published work, we discuss pustular-type DIHS/DRESS which, in most cases, involves acute generalized exanthematous pustulosis (AGEP)-like skin eruptions in response to carbamazepine. Pustular eruptions may appear in relatively few cases of DIHS/DRESS, in particular, when the causative drug is carbamazepine and, even in cases of intractable pustular bacterid-like eruptions, a reaction to a drug should be suspected. Skeletal muscle involvement may be associated with DIHS/DRESS as one of its systemic manifestations.