We present a rare neurocutaneous genetic disorder where patients develop a combination of cerebral dysgenesis, neuropathy, ichthyosis, and keratoderma, commonly known as CEDNIK syndrome. It is an autosomal recessive inheritance involving the SNAP29 protein, mapped to the 22q11.2 gene. Phenotypic variation is seen with this disease, with clinical manifestation of developmental milestone delays ranging in severity. With only a handful of documented cases, available research, management of the syndrome, and prognosis are not well established. As CEDNIK syndrome has systemic implications, care coordination between specialists is essential in improving patient outcomes. Particularly important is preventing patients from meeting the criteria of failure to thrive, a commonly reported issue. In this case, we present a four-month-old male with a past medical history of pyloric stenosis status/post pyloromyotomy who has failure to thrive, gastroesophageal reflux disease, profound hypotonia, and delayed progression of developmental milestones. Additionally, the case is complicated by idiopathic pyloric stenosis, further contributing to the patient's failure to thrive. We aim to discuss the pathophysiology of this syndrome, explore the timeline of disease progression, as well as compare our case to the current literature.
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http://dx.doi.org/10.7759/cureus.59475 | DOI Listing |
Int J Surg Case Rep
January 2025
Faculty of Medicine, Damascus University, Damascus, Syria.
Introduction: Congenital pyloric web or diaphragm are rare causes of Gastric Outlet Obstruction (GOO) after infancy, representing approximately 1 % of gastrointestinal obstructions. While it typically presents in the neonatal period with nonbilious vomiting, delayed diagnosis beyond infancy is exceptionally rare.
Presentation Of The Case: An 11-year-old girl with presented with one-month history of postprandial vomiting.
Int J Surg Case Rep
January 2025
Department of Public Health and Infectious Diseases, Faculty of Medicine, Herat University, Herat, Afghanistan; Department of Epidemiology and Biostatistics, Schulich School of Medicine & Dentistry, Western University, London, ON, Canada.
Introduction: This case report presents a rare occurrence of Type III Congenital Paraesophageal Hiatal Hernia (CPEHH) with Infantile Hypertrophic Pyloric Stenosis (IHPS) in a 28-day-old neonate. However, this unusual combination poses significant diagnostic and surgical challenges.
Presentation Of Case: A 28-day-old male presented with respiratory distress and persistent vomiting.
Expert Opin Drug Saf
December 2024
Department of Pharmacy, DaLi University First Affiliated Hospital, DaLi City, Yunnan Province, China.
Aim: To mine and analyze adverse drug events (ADEs) signals of azithromycin use in children in the real world to inform the safety assessment of azithromycin use in children.
Methods: ADE reports from the FDA Adverse Event Reporting System (FAERS) involving children (0-17 years) with azithromycin as the primary suspected drug from 2004 to early 2024 were extracted. ADEs were categorized using MedDRA, and signal detection was conducted using Reporting Odds Ratio (ROR) and Proportional Reporting Ratio (PRR).
J Surg Case Rep
December 2024
Department of Surgery, University of Miami, PO Box 016960 (C203), Miami, FL 33101, United States.
Congenital colonic stenosis (CCS) is a rare cause of intestinal obstruction, most commonly presenting in the neonatal period. We present a case of delayed CCS and describe the diagnostic challenges experienced. A 16-week-old female patient presented with persistent failure to thrive associated with signs of intestinal obstruction.
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