Successful pregnancy and childbirth without metabolic abnormality in a patient with holocarboxylase synthetase deficiency.

Holocarboxylase synthetase deficiency (HSD), an autosomal recessive biotin cycle disorder, is caused by holocarboxylase synthetase (HLCS) genetic variants, resulting in multiple carboxylase deficiency. Catabolic stress can induce metabolic crises in patients with HSD. Although pharmacological doses of biotin have improved HLCS enzyme activity and HSD prognosis, the prolonged life expectancy has gradually highlighted novel issues in adult patients with HSD. To the best of our knowledge, there is only one report on a case of HSD during pregnancy and childbirth, and the metabolic profile was not well defined. In this report, we present the history and metabolic profile of a woman with HSD who had an uncomplicated pregnancy and childbirth. A high pharmacological dose of biotin, 100 mg/day, had no effect on the fetus. Even during the emergency cesarean section, the detailed metabolic assessments revealed no significant laboratory findings, such as ketolactic acidosis, hyperammonemia, and remarkable acylcarnitine change. This report suggests that a woman with HSD who regularly takes biotin can conceive and give birth safely, and biotin doses of 100 mg/day may not influence the growth and development of the fetus. Further research and case studies on pregnant women with HSD are required to determine an acceptable maximum dosage of biotin for human fetuses.

Factors Associated with Surgical Treatment in Postpartum Women with Mastitis or Breast Abscess: A Retrospective Cohort Study.

Objective: The aim of this study was to identify factors associated with requiring surgical treatment for mastitis or breast abscess in postpartum mothers. Materials and Methods: This was a retrospective cohort study using the Japan Medical Data Center Claims Database. Information on pairs of infants born between April 2012 and December 2016 and their mothers were extracted. Data regarding the baseline characteristics, medical history, medical practice, and prescription drugs of mothers and their infants were collected. Multivariable logistic regression analysis was used to examine factors associated with undergoing surgical treatment. Results: The data of 69,363 eligible mothers were analyzed. The proportion of mothers who were diagnosed with mastitis or breast abscess within 1 year after childbirth was 10.8% (7,516/69,363). There were 114 mothers who underwent surgical treatment within 1 year after childbirth. This was 0.2% of all mothers and 1.5% of those diagnosed with mastitis or breast abscess. Surgical treatment was significantly associated with mothers having their first child (adjusted odds ratio [AOR], 2.58; 95% confidence interval [CI], 1.63-4.07) compared to those with a second or later child; it was also significantly associated with the diagnosis of breast abscess (AOR, 10.38; 95% CI, 5.28-20.40). Conclusions: This was the first report of the prevalence of mastitis or breast abscess and the requirement for surgical treatment among postpartum mothers in Japan. A first diagnosis of breast abscess and having a first child were associated with requiring surgical treatment. Health care professionals providing postpartum care should be aware of these factors.