Gastroschisis and septo-optic-pituitary dysplasia: Is there an association?

AIM: There are several case reports describing patients with both optic nerve hypoplasia/septo-optic-pituitary dysplasia (ONH/SOD) and gastroschisis (GS). Our aim was to investigate whether ONH/SOD is associated with GS. METHODS: A retrospective population-based study was undertaken using the Population Research Data Repository at the Manitoba Center for Health Policy in Manitoba, Canada to investigate if any patient with ONH/SOD also had GS. In addition, Winnipeg's Surgical Database of Outcomes and Management (WiSDOM), a hospital-based paediatric surgical database, was searched to ascertain if any of the patients with GS also have ONH/SOD. RESULTS: Cases were 124 patients with ONH/SOD diagnosed during 1990-2019. None had GS. The surgical database had 188 patients from Manitoba with GS during 1991-2019. None had ONH/SOD. CONCLUSION: There does not appear to be an association between ONH/SOD and GS in our cohorts of patients with these two disorders.

Mental Health Outcomes of Mothers of Children With Congenital Gastrointestinal Anomalies Are Similar to Control Mothers: A Longitudinal Retrospective Cohort Study.

PURPOSE: Mothers of infants born with congenital gastrointestinal surgical anomalies experience a unique caregiving role. Whether these challenges result in more mental health diagnoses than the general population is unknown. This study assessed mental health diagnoses in mothers of children born with congenital surgical anomalies (CSA) compared to mothers of children without CSA. METHODS: A total of 780 mothers of children with CSA (case-mothers) and 6994 control-mothers were included. Mental health diagnoses were obtained using International Classification of Diseases 9 codes from an administrative database. Multivariate hazard ratios (HR) of mental health diagnoses were determined for mothers following childbirth, controlling for socioeconomic status, mental health diagnoses prior to childbirth, and age at childbirth. Subgroup analyses were completed for anxiety, depression, and post-traumatic stress disorder (PTSD). The association between a woman's mental health history and becoming a case-mother was analyzed using risk ratios (RR). RESULTS: Case-mothers were not at increased risk of mental health disorders compared to controls (HR = 1.00; CI95 = 0.92-1.09). In aggregate analysis, no increased risk of anxiety, depression, or PTSD was found. Subgroup analyses revealed that congenital diaphragmatic hernia case-mothers had an increased risk of depression (HR = 1.43; CI95 = 1.08-1.88). No other case-mothers were at increased risk of mental health diagnoses, anxiety, depression, or PTSD. Only omphalocele was associated with mental health diagnoses before childbirth (RR = 1.89; CI95 = 1.58-2.26). CONCLUSIONS: Collectively, mothers of children with CSAs did not have a greater risk of a mental health disorder, anxiety, depression, or PTSD compared to control-mothers. Only mothers of children with congenital diaphragmatic hernia had an increased risk of depression. LEVEL OF EVIDENCE: II.

Prevalence of Mental Health Disorders in Individuals With Gastrointestinal Congenital Surgical Anomalies: A Systematic Review and Meta-analysis.

PURPOSE: Studies examining functional outcomes and health-related quality of life for patients with congenital surgical anomalies (CSA) are increasingly common. However, the prevalence of mental health disorders in this population has not been determined. The purpose of this review is to summarize the reported prevalence of mental health disorders in children born with gastrointestinal CSA. METHODS: A systematic review of the literature was conducted on Medline (OVID), EMBASE (OVID), CINAHL (EbscoHost), and Scopus to identify studies reporting mental health diagnoses in children with a gastrointestinal CSA. A meta-analysis of the prevalence of anxiety disorders and depressive disorders was completed. RESULTS: Of 2546 manuscripts, seven met the inclusion criteria. All included manuscripts looked at patients with anorectal malformation (ARM) and/or Hirschsprung disease (HD). No studies assessed other gastrointestinal CSAs. A total of 183 psychiatric diagnoses were reported in 1167 patients. A pooled analysis of ARM patients revealed a 14.0% prevalence of anxiety disorders (N = 208; CI95 = 4.4-23.4%) and an 18.75% prevalence of depressive disorders (N = 48; CI95 = 7.7-29.8%). A pooled analysis of HD patients revealed a 15.8% prevalence of anxiety disorders (N = 19; CI95 = 0.6-32.2%) and a 4.75% prevalence of depressive disorders (N = 758; CI95 = 3.23-6.26%). CONCLUSIONS: There is a paucity of evidence on mental health outcomes of individuals born with gastrointestinal CSA. However, the available evidence suggested a high prevalence of mental health diagnoses in individuals born with ARM or HD with 1 in 7 having an anxiety disorder. Additionally, 1 in 5 ARM patients and 1 in 20 HD patients were diagnosed with a depressive disorder. TYPE OF STUDY: Systematic review and meta-analysis. LEVEL OF EVIDENCE: IV.

Post-discharge follow-up of congenital duodenal obstruction patients: a systematic review.

PURPOSE: Long-term follow-up of congenital duodenal obstruction patients often falls on care providers with little experience of this condition. We performed a systematic review of the long-term outcomes of duodenal obstruction and provide a summary of sequelae care providers should anticipate. METHODS: In 2022, after registering with PROSPERA, Medline (Ovid), EMBASE, PSYCHINFO, CNAHL and SCOPUS databases were searched using the title keyword 'intestinal atresia'. Abstracts were filtered for inclusion if they included the duodenum. Papers of filtered abstracts were included if they reported post-discharge outcomes. Methodological Index for Non-Randomized Studies was used to grade the papers. RESULTS: Of the 1068 abstracts were screened, 32 papers were reviewed. Eleven studies were included. Thirty additional papers were included after reviewing references, for a total of 41 papers. The average MINORS was 7/16. CONCLUSION: There is good evidence that children with congenital duodenal obstruction do well in terms of survival, growth and general well-being. Associated cardiac, musculoskeletal and renal anomalies should be ruled-out. Care providers should be aware of anastomotic dysfunction, blind loop syndrome, bowel obstruction and reflux. Reflux may be asymptomatic. Laparoscopic repair does not change long-term outcomes, and associated Trisomy 21 worsens neurodevelopmental outcomes.

Effect of trisomy 21 on long-term gastrointestinal outcomes in duodenal atresia.

PURPOSE: We aimed to determine if Trisomy 21 (T21) affected gastrointestinal outcomes for children with duodenal atresia (DA). METHODS: We identified children born with DA between 1991 and 2017. Cases were divided into DA with T21 and DA without T21. Ten healthy controls per case were included. Esophageal, ulcerative, obstructive and stomach complaints were assessed. Risk ratios (RR), rate ratios (RaR) and Cox models were constructed. Analyses were performed for cases versus controls, and for T21 cases versus non-T21 cases. RESULTS: DA cases totaled 52: 22 had T21 and 30 did not. There were 520 controls. DA cases had more gastrointestinal complaints than controls. T21 cases were at greater risk and frequency of esophageal disease than non-T21 cases (RR = 4.08, p = 0.002, RaR = 69.8, p < 0.001). T21 and non-T21 cases were equally likely to present with obstruction (RR = 0.91, p = 1), but T21 cases complained of obstructive symptoms less (RaR = 0.57, p = 0.003). T21 and non-T21 cases had the same risk of stomach diseases, but T21 cases complained more frequently (RaR = 6.20, p < 0.001). Cox models supported these observations. T21 did not affect ulcerative diseases. CONCLUSION: DA cases had more gastrointestinal problems than controls. T21 increased esophageal and gastric complaints in DA cases but did not affect ulcerative and obstructive complaints.