METHOD: A male Chinese neonate, who presented with abdominal distention and constipation on the second day of life, was found to have features of Currarino triad. Colostomy was done in the neonatal period, and the presacral mass was excised by posterior sagittal perineal approach at the age of six months.
RESULTS: The excised presacral mass consisted of an anterior meningocele and a teratoma. The patient continued to have constipation during follow-up and required anorectoplasty to correct residual anorectal stenosis. At the time of this report the patient was three years old and growing normally with normal anorectal function.
DISCUSSION: Of a total of about 200 cases of complete Currarino triad found in the literature, in only 22 patients did the presacral mass contain both meningocele and teratoma. The features of these 22 patients and the current views on the surgical management of Currarino triad are discussed.
CASE PRESENTATION: We report on a case of anterior glottis stenosis with keel aspiration for two weeks, after endoscopic CO2 laser resection of the stenotic section and keel placement. The patient was admitted to our center, where bronchoscopy was performed and the keel was removed. A new custom-made silastic keel was properly placed in raw areas and fixed to the skin with suture through the cricothyroid and thyrohyoid membranes. The keel was removed three weeks later.
CONCLUSIONS: Endoscopic keel placement should be done with heavy suture through cricothyroid and thyrohyoid membranes. Surgeons should suture the keel to the anterior laryngeal wall with specially designed Lichtenberger's needle-carriers to prevent complications such as keel aspiration, adhesion formation and imposing a second trip under general anesthetics, which put the patient at increased risk. The false vocal cord microflaps, as biological keels and a relatively new method may replace silastic keel placement in the future.
OBJECTIVE: To determine the clinical course and outcome of familial congenital laryngotracheal stenosis (FCLS).
METHODS: A literature search was conducted over a period of one month (September 2023) by searching several databases to identify studies published from inception to 31st August 2023.
RESULTS: Of 256 papers identified, five articles met the inclusion criteria. A total of 17 patients with slight female predominance (59 %) were identified. Familial congenital tracheal stenosis was reported in female twins (100 %). A variety of clinical presentations were listed. An endoscopic airway study was performed on all patients. 64.8 % of the included children were managed surgically. Genetic studies performed on 41 % of children could not locate genetic abnormalities.
CONCLUSION: Consanguinity, twin births, and female gender could be predisposing factors for FCLS, although the quality of evidence is low due to the rarity of the condition.