Displaying publications 1 - 20 of 28 in total

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  1. Lee WS, Chong LA, Begum S, Abdullah WA, Koh MT, Lim EJ
    J Pediatr Hematol Oncol, 2001 May;23(4):244-6.
    PMID: 11846304
    We report a newborn infant girl, born to consanguineous parents, with recurrent intracranial hemorrhage secondary to congenital factor V deficiency with factor V inhibitor. Repeated transfusions of fresh-frozen plasma (FFP) and platelet concentrates, administrations of immunosuppressive therapy (prednisolone and cyclophosphamide), and intravenous immunoglobulin failed to normalize the coagulation profiles. Exchange transfusion followed-up by administrations of activated prothrombin complex and transfusions of FFP and platelet concentrates caused a temporary normalization of coagulation profile, enabling an insertion of ventriculoperitoneal (VP) shunt for progressive hydrocephalus. The treatment was complicated by thrombosis of left brachial artery and ischemia of left middle finger. The child finally died from another episode of intracranial hemorrhage 10 days after insertion of the VP shunt.
    Matched MeSH terms: Consanguinity
  2. Shmukler BE, Kedar PS, Warang P, Desai M, Madkaikar M, Ghosh K, et al.
    Am J Hematol, 2010 Oct;85(10):824-8.
    PMID: 20799361 DOI: 10.1002/ajh.21836
    Familial distal renal tubular acidosis (dRTA) can be caused by mutations in the Cl2/HCO32 exchanger of the renal Type A intercalated cell, kidney AE1/SLC4A1. dRTA-associated AE1 mutations have been reported in families from North America, Europe, Thailand, Malaysia, Papua-New Guinea, Taiwan, and the Philippines, but not India. The dRTA mutation AE1 A858D has been detected only in the context of compound heterozygosity. We report here two unrelated Indian patients with combined hemolytic anemia and dRTA who share homozygous A858D mutations of the AE1/SLC4A1 gene. The mutation creates a novel restriction site that is validated for diagnostic screening.
    Matched MeSH terms: Consanguinity
  3. Khositseth S, Bruce LJ, Walsh SB, Bawazir WM, Ogle GD, Unwin RJ, et al.
    QJM, 2012 Sep;105(9):861-77.
    PMID: 22919024 DOI: 10.1093/qjmed/hcs139
    Distal renal tubular acidosis (dRTA) caused by mutations of the SLC4A1 gene encoding the erythroid and kidney isoforms of anion exchanger 1 (AE1 or band 3) has a high prevalence in some tropical countries, particularly Thailand, Malaysia, the Philippines and Papua New Guinea (PNG). Here the disease is almost invariably recessive and can result from either homozygous or compound heterozygous SLC4A1 mutations.
    Matched MeSH terms: Consanguinity
  4. Tey S, Shahrizaila N, Drew AP, Samulong S, Goh KJ, Battaloglu E, et al.
    Neurogenetics, 2019 08;20(3):117-127.
    PMID: 31011849 DOI: 10.1007/s10048-019-00576-3
    Charcot-Marie-Tooth (CMT) disease is a form of inherited peripheral neuropathy that affects motor and sensory neurons. To identify the causative gene in a consanguineous family with autosomal recessive CMT (AR-CMT), we employed a combination of linkage analysis and whole exome sequencing. After excluding known AR-CMT genes, genome-wide linkage analysis mapped the disease locus to a 7.48-Mb interval on chromosome 14q32.11-q32.33, flanked by the markers rs2124843 and rs4983409. Whole exome sequencing identified two non-synonymous variants (p.T40P and p.H915Y) in the AHNAK2 gene that segregated with the disease in the family. Pathogenic predictions indicated that p.T40P is the likely causative allele. Analysis of AHNAK2 expression in the AR-CMT patient fibroblasts showed significantly reduced mRNA and protein levels. AHNAK2 binds directly to periaxin which is encoded by the PRX gene, and PRX mutations are associated with another form of AR-CMT (CMT4F). The altered expression of mutant AHNAK2 may disrupt the AHNAK2-PRX interaction in which one of its known functions is to regulate myelination.
    Matched MeSH terms: Consanguinity
  5. Bin NJ, Heng HM, Poh R, Noor SM, Subrayan V
    Retina, 2015 Jun;35(6):1266-72.
    PMID: 25549071 DOI: 10.1097/IAE.0000000000000446
    To evaluate the association of phospholipase A2, Group V (PLA2G5), with benign familial fleck retina in a consanguineous family with triplets.
    Matched MeSH terms: Consanguinity
  6. Salemi S, Besson A, Eblé A, Gallati S, Pfäffle RW, Mullis PE
    Growth Horm. IGF Res., 2003 Oct;13(5):264-8.
    PMID: 12932747
    OBJECTIVE: Growth is an inherent property of life. About 10% of the congenital forms of growth retardation and short stature are genetically caused. Beside the gene involved in direct GH-production, there are different candidate genes important for appropriate pituitary development causing combined pituitary hormone deficiency (CPHD). However, severe growth retardation and failure to thrive remain the leading reason for medical assessment in these patients.

    PATIENTS AND METHODS: We report two siblings of a healthy but consanguineous Malaysian family presenting with severe short stature caused by CPHD with a variable phenotype. Importantly, at the beginning the girl presented with isolated GHD, whereas the boy was hypothyroid. As the most common gene alterations responsible for CPHD are within either the PROP-1- or the POU1F1- (PIT-1)-gene these two genes were further studied.

    RESULTS: Subsequent sequencing of the six exons of the POU1F1-gene allowed the identification of a new N-terminal mutation (Q4ter) in these two children. A substitution of C to T induced a change from a glutamine (CAA) to a stop codon (TAA) in exon 1 of the PIT-1 protein. Both affected children were homozygous for the mutation, whereas the mother and father were heterozygous.

    CONCLUSION: We describe two children with autosomal recessive inherited CPHD caused by a new N-terminal located mutation within the PUO1F1-gene. The clinical history of these two children underline the phenotypic variability and support the fact that children with any isolated and/or combined PHD need to be closely followed as at an any time other hormonal deficiencies may occur. In addition, molecular analysis of the possible genes involved might be most helpful for the future follow-up.

    Matched MeSH terms: Consanguinity
  7. Mohamed R, Tan CT, Wong NW
    Med J Malaysia, 1994 Mar;49(1):49-52.
    PMID: 8057991
    The clinical course of 18 patients with Wilson's disease is reported. There were 13 males and five females of whom one is Malay. The prevalence of Wilson's disease in Malaysia is probably the same as elsewhere. Being a genetic syndrome, the genetic carrier rate for Wilson's disease is probably lower amongst the Malays. At diagnosis, the clinical signs were predominantly hepatic in 10 patients, neurological in five patients with three asymptomatic cases. All patients were commenced on penicillamine but poor compliance was observed in many patients. Two patients defaulted follow-up and seven patients died. Out of the nine surviving patients, only four are well with no clinical symptoms.
    Matched MeSH terms: Consanguinity
  8. Lee WS, Boey CC, Goh AY
    Singapore Med J, 1999 Apr;40(4):278-80.
    PMID: 10487085
    Hyperimmunoglobulin E syndrome (HIE) is a rare condition characterised by marked elevation of serum IgE level, chronic dermatitis, intense pruritus, and recurrent serious infection. The major organism is usually S aureus. We report a case of an infant with HIE, who had pulmonary nocardiosis. The clinical features, immunological abnormalities, and radiological features of the condition are described. The child finally succumbed to the complications of pulmonary nocardiosis.
    Matched MeSH terms: Consanguinity
  9. Abdul-Wahab J, Naznin M, Suhaimi A, Amir-Hamzah AR
    Singapore Med J, 2007 Jul;48(7):e206-8.
    PMID: 17609817
    Familial myelodysplastic syndrome occurring at a young age is a very rare childhood haematological malignancy. Two siblings, aged three and 18 years, from a consanguineous marriage, presented with pancytopenia and was subsequently diagnosed to have myelodysplastic syndrome. Both remained clinically stable throughout the illness. Splenectomy appeared to have fully corrected the cytopenia in one of them.
    Matched MeSH terms: Consanguinity
  10. Nathan AM, Thong MK, deBruyne J, Ariffin H
    J Paediatr Child Health, 2011 Aug;47(8):573-5.
    PMID: 21843195 DOI: 10.1111/j.1440-1754.2011.02149.x
    Matched MeSH terms: Consanguinity
  11. Lopez CG, Thiruselvam A, Hutton RA
    Clin Lab Haematol, 1982;4(4):411-5.
    PMID: 7166027 DOI: 10.1111/j.1365-2257.1982.tb00486.x
    Matched MeSH terms: Consanguinity
  12. Al-Herz W
    J Clin Immunol, 2008 Mar;28(2):186-93.
    PMID: 18008151
    Primary immunodeficiency disorders are heterogeneous group of illnesses that predispose patients to serious complications. Registries for these disorders have provided important epidemiological data and shown both racial and geographical variations. The clinical features of 76 patients with primary immunodeficiency disorders registered in Kuwait National Primary Immunodeficiency Registry from 2004 to 2006 were recorded. Ninety-eight percent of the patients presented in childhood. The prevalence of these disorders in children was 11.98 in 100,000 children with an incidence of 10.06 in 100,000 children. The distribution of these patients according to each primary immunodeficiency category is: combined T and B cell immunodeficiencies (21%), predominantly antibody immunodeficiency (30%), other well defined immunodeficiencies (30%), diseases of immune dysregulation (7%), congenital defects of phagocyte number, function or both (8%), and complement deficiencies (4%). The consanguinity rate within the registered patients was 77%. The patients had a wide range of clinical features affecting different body systems. Primary immunodeficiency disorders are prevalent in Kuwait and have a significant impact into the health system.
    Matched MeSH terms: Consanguinity
  13. Thambiah, Subashini Chellapah, Zalinah Ahmad, Zarida Hambali, Malina Osman, Munia Mohd Zain, Fuziah Md Zain, et al.
    MyJurnal
    A clinical descriptive study was done to determine the sociodemographic, laboratory and clinical characteristics of patients with congenital adrenal hyperplasia (CAH) referred to Hospital Putrajaya, a tertiary endocrine centre in Malaysia. Electronic laboratory data of 51 CAH patients were obtained. The demographics and clinical details of the study population were acquired from a questionnaire completed by parents of participants. There were 25 males (49%) and 26 females (51%), of which, 58.8% were Malays. Median age of participants was 4 years whilst median age at diagnosis of CAH was two years. Parental consanguinity was documented in three patients (5.9%). Patients originated from Johor (19.6%), Selangor (19.6%), Negeri Sembilan (17.6%) and Kedah (13.7%). Majority of patients were diagnosed after one week of life (80.4%) although more females were diagnosed under the age of one week compared to males (p=0.041). Most females presented with ambiguous genitalia (42.3%) [p=0.001] whereas 72% of males presented with salt wasting (p=0.003). No significant associations between race and all other variables, though interestingly three Malay patients presented with ambiguous genitalia and hypertension. Equal gender distribution noted as expected in an autosomal recessive condition, although not in keeping with other Asian countries. Early diagnosis in females attributed to obvious genital ambiguity at birth. Varied clinical presentation, although in minority, necessitates genetic studies for prompt diagnosis and treatment. Considering that majority of patients presented with salt wasting and the age at diagnosis was delayed, the introduction of a neonatal screening programme is essential in Malaysia.
    Matched MeSH terms: Consanguinity
  14. El Kishawi RR, Soo KL, Abed YA, Muda WAMW
    BMC Pediatr, 2017 12 21;17(1):210.
    PMID: 29268788 DOI: 10.1186/s12887-017-0957-y
    BACKGROUND: Stunting continues to be a major public health problem in developing countries. It is one of the most important risk factors for morbidity and mortality during childhood. In Palestine, it is another health problem, which adds to the catastrophic issues in the region. This study aimed to determine the prevalence of stunting and its associated factors among preschool children in the Gaza Strip.

    METHODS: A cross-sectional study design was conducted in the Gaza Strip. A total of 357 children aged 2-5 years and their mothers aged 18-50 years were recruited. A multistage cluster sampling was used in the selection of the study participants from three geographical areas in the Gaza Strip: Jabalia refugee camp, El Remal urban area, and Al Qarara rural area. A structured questionnaire was used for face- to -face interviews with the respective child's mother to collect sociodemographic information and feeding practice. Anthropometric measurements for children were taken to classify height-for-age (HAZ), while maternal height was measured as well. Descriptive and binary logistic regression analyses were applied to determine the prevalence and associated factors with stunting.

    RESULTS: The total prevalence of stunting in this study was 19.6%, with the highest prevalence being (22.6%) in Jabalia refugee camp. It turns out that shorter mothers had increased the odds of stunting in preschool children in the Gaza Strip. Children born to mothers whose height was 1.55-1.60 m or <1.55 m were more likely to be stunted (p = 0. 008), or (p 1.60 m. Moreover, parental consanguinity increased the risk of stunted children (p = 0. 015).

    CONCLUSIONS: This study showed the prevalence of stunting was of alarming magnitude in the Gaza Strip. Our results also demonstrated that parental consanguinity and short maternal stature were associated with stunting. Culturally appropriate interventions and appropriate strategies should be implemented to discourage these types of marriages. Policy makers must also raise awareness of the importance of the prevention and control of nutritional problems to combat stunting among children in the Gaza Strip.

    Matched MeSH terms: Consanguinity
  15. Al-Herz W, Chou J, Delmonte OM, Massaad MJ, Bainter W, Castagnoli R, et al.
    Front Immunol, 2018;9:3146.
    PMID: 30697212 DOI: 10.3389/fimmu.2018.03146
    Objective: To present the genetic causes of patients with primary immune deficiencies (PIDs) in Kuwait between 2004 and 2017. Methods: The data was obtained from the Kuwait National Primary Immunodeficiency Disorders Registry. Genomic DNA from patients with clinical and immunological features of PID was sequenced using Sanger sequencing (SS), next generation sequencing (NGS) of targeted genes, whole exome sequencing (WES), and/or whole genome sequencing (WGS). Functional assays were utilized to assess the biologic effect of identified variants. Fluorescence in situ hybridization (FISH) for 22q11.2 deletion and genomic hybridizations arrays were performed when thymic defects were suspected. Results: A total of 264 patients were registered during the study period with predominance of patients with immunodeficiencies affecting cellular and humoral immunity (35.2%), followed by combined immunodeficiencies with associated syndromic features (24%). Parental consanguinity and family history suggestive of PID were reported in 213 (81%) and 145 patients (55%), respectively. Genetic testing of 206 patients resulted in a diagnostic yield of 70%. Mutations were identified in 46 different genes and more than 90% of the reported genetic defects were transmitted by in an autosomal recessive pattern. The majority of the mutations were missense mutations (57%) followed by deletions and frame shift mutations. Five novel disease-causing genes were discovered. Conclusions: Genetic testing should be an integral part in the management of primary immunodeficiency patients. This will help the delivery of precision medicine and facilitate proper genetic counseling. Studying inbred populations using sophisticated diagnostic methods can allow better understanding of the genetics of primary immunodeficiency disorders.
    Matched MeSH terms: Consanguinity*
  16. Lee WS, Tay CG, Nazrul N, Paed M, Chai PF
    Med J Malaysia, 2009 Mar;64(1):83-5.
    PMID: 19852331 MyJurnal
    A five-month-old Indian girl, product of consanguineous marriage, presented with diarrhoea with an onset within two days after birth, severe malnutrition and metabolic acidosis. The diarrhoea persisted even with lactose-free formula, amino acid-based formula and glucose-containing oral rehydration solution, but stopped when fasted. She required prolonged parenteral nutrition. Fructose and glucose tolerance tests were performed, confirming the child was able to absorb and metabolize fructose but not glucose, indicating a diagnosis of glucose-galactose malabsorption. This case illustrate how simple and pertinent clinical observations and laboratory investigations is sufficient to allow a firm diagnosis to be made.
    Matched MeSH terms: Consanguinity
  17. Chan LG, Ting HS
    Clin. Dysmorphol., 2005 Jan;14(1):1-5.
    PMID: 15602085
    We present two siblings from a consanguineous Malaysian family with multiple skeletal abnormalities, anterior segment anomalies of the eye and early lethality. These features are consistent with a syndrome first described by Al-Gazali and we provide further delineation of the syndrome.
    Matched MeSH terms: Consanguinity
  18. Khalili Samani N, Esa Y, Amin SM, Fatin Mohd Ikhsan N
    PeerJ, 2016;4:e1930.
    PMID: 27231645 DOI: 10.7717/peerj.1930
    Plotosus canius (Hamilton, 1822) is a significant marine species in Malaysia from nutritional and commercial perspectives. Despite numerous fundamental research on biological characteristics of P. canius, there are various concerns on the level of population differentiation, genomic structure, and the level of genetic variability among their populations due to deficiency of genetic-based studies. Deficiency on basic contexts such as stock identification, phylogenetic relationship and population genetic structure would negatively impact their sustainable conservation. Hence, this study was conducted to characterize the genetic structure of P. canius for the first time through the application of mitochondrial Cytochrome Oxidase I (COI) gene, cross amplification of Tandanus tandanus microsatellites, and a total of 117 collected specimens across five selected populations of Malaysia. The experimental results of the mitochondrial analysis revealed that the haplotype diversity and nucleotide diversity varied from 0.395-0.771 and 0.033-0.65 respectively. Moreover, the statistical analysis of microsatellites addressed a considerable heterozygote insufficiency in all populations, with average observed heterozygosity (Ho ) value of 0.2168, which was lower than the standard heterozygosity in marine populations (Ho = 0.79). This alongside the high Fis values estimation, high pairwise differentiation among populations and low within population variations are supposed to be associated with small sample size, and inbreeding system. Besides, the significant finding of this study was the sharing of common haplotype KR086940, which reflects a historical genetic connectivity between Peninsular Malaysia and Borneo populations due to the geological history of Southeast Asia during Pleistocene era. Demographic analyses showed that all populations were in an equilibrium state with no significant evidence of population expansion. To put it briefly, the current study has managed to provide an initial genomic database toward understanding of the genetic characterization, phylogenetic, molecular diversification and population structure in P. canius, and should be necessary highlighted for appropriate management and conservation of species. Further studies must be carried out involving more geographical and sampling sites, larger population size per site, and utilization of species specific microsatellites loci.
    Matched MeSH terms: Consanguinity
  19. Monteiro ES
    1. Three cases of situs inversus are described and the clinical and aetiological factors are discussed briefly.
    2. This abnormality occurring in father and son whose family history indicates a great degree of consaguinity seems to support the view held by Cockayne that it is a recessive inheritance.
    3. Situs inversus when complete seems to cause no disability. When incomplete as in examples of isolated dextrocardia, congenital heart diseases are commonly seen in association with it. Other congenital malformations may also co-exist. Recently the syndrome of hypertrophic rhinitis, nasal polyposis and sinusitis and bronchiectasis have been encountered in association with transposition of the viscera.
    Matched MeSH terms: Consanguinity
  20. Leelavathi, M., Lim, J.L., Ahmad, S.
    Medicine & Health, 2014;9(1):74-79.
    MyJurnal
    Cerebral palsy is a common cause of childhood disability. It has a great impact on parents and caregivers, especially when it reoccurs in the same family. Although familial cerebral palsy is relatively uncommon, cases have been reported among children from consanguineous, non-consanguineous marriages and multiple pregnancies suggesting a possible complex genetic mode of inheritance. Physicians need to be aware of the possibility of familial cerebral palsy for early detection and counseling. We describe a rare case of two male siblings from a non-consanguineous marriage affected by cerebral palsy.
    Matched MeSH terms: Consanguinity
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