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13 June 2023: Articles  Japan

Preoperative Contrast Examinations Help Determine the Appropriate Cervical Approach for Congenital Gross Type C Esophageal Atresia: A Report of Two Cases

Challenging differential diagnosis, Congenital defects / diseases

Yoichi Nakagawa ORCID logo1E, Hiroo Uchida ORCID logo1F*, Chiyoe Shirota1B, Takahisa Tainaka ORCID logo1B, Wataru Sumida1B, Satoshi Makita ORCID logo1B, Hizuru Amano1B, Aitaro Takimoto1B, Seiya Ogata1B, Shunya Takada ORCID logo1B, Takuya Maeda1B, Yousuke Gohda1B, Yaohui Guo1B, Akinari Hinoki ORCID logo2B

DOI: 10.12659/AJCR.938723

Am J Case Rep 2023; 24:e938723

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Abstract

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BACKGROUND: Kluth demonstrated that esophageal atresia/tracheoesophageal fistula (EA/TEF) has several anatomical variations and thus requires a preoperative imaging study to determine the surgical strategy. We routinely perform a contrast examination with iodixanol to assess the location of the TEF and the upper end of the esophageal pouch to determine the most appropriate approach. We herein present two cases of type C EA/TEF who successfully underwent radical surgery by a cervical approach based on the information from the contrast examination.

CASE REPORT: Case 1 was a Japanese boy suspected of type C EA/TEF after birth. A contrast examination with iodixanol showed that a TEF was at the second thoracic vertebra (Th2), as was the upper end of the esophageal pouch. Thus, the patient underwent esophago-esophageal anastomosis and TEF ligation using a cervical approach; the postoperative course was uneventful. Case 2 was also a Japanese boy suspected of type C EA/TEF. A contrast examination showed that the TEF was at Th1-2, as was the upper end of the esophageal pouch. Thus, the patient underwent esophago-esophageal anastomosis and TEF ligation using a cervical approach. The patient suffered from congenital tracheal stenosis and required tracheoplasty. However, there were no apparent complications after the surgery.

CONCLUSIONS: Here, we used the imaging information to adopt the cervical approach in type C EA/TEF cases and concluded that routine preoperative contrast examinations helped assess the TEF location and upper end of the esophageal pouch without significant complications.

Keywords: Contrast Media, esophageal atresia, Esophageal Atresia with or without Tracheoesophageal Fistula, Male, Humans, Triiodobenzoic Acids

Background

Gross type C esophageal atresia/tracheoesophageal fistula (EA/TEF) accounts for approximately 90% of congenital EA/TEF cases and is accompanied by TEF. TEF is usually located at the middle trachea or within 1 cm from the first tracheal carina. Hence, type C EA/TEF is almost always repaired by thoracotomy or thoracoscopy. However, Kluth demonstrated that EA/TEF has several anatomical variations [1]. To our knowledge, there are six cases with type C EA/TEF eventually repaired using a cervical approach [2–5], indicating the necessity of a preoperative imaging study to determine the appropriate surgical strategy. Herein, we report two cases of type C EA/TEF repaired using a cervical approach, which was selected after a routine preoperative contrast examination of the esophagus that accurately located the TEF.

Case Reports

CASE 1:

A Japanese boy was born at a gestational age of 39 weeks (2327 g) without prenatal abnormalities. However, due to foamy secretion, the patient’s respiratory condition was poor; chest radiography showed the nasogastric tube coiled in the proximal esophagus. Therefore, esophageal atresia/tracheoesophageal fistula (EA/TEF) was suspected, and the patient was transferred to our hospital on the first day of life. A contrast examination showed that a TEF was at the second thoracic vertebra (Th2), as was the upper end of the esophageal pouch (Figure 1). Thus, the patient underwent esophago-esophageal anastomosis and TEF ligation using a cervical approach; the postoperative course was uneventful, except for temporary left recurrent laryngeal nerve palsy (RLNP). A postoperative contrast examination revealed no apparent anastomotic strictures. The left RLNP improved on day 46 postoperatively, and the patient has lived with no significant symptoms for four years since the operation.

CASE 2:

A Japanese boy was born at a gestational age of 40 weeks (2938 g). The patient was diagnosed with VATER syndrome, consisting of type C EA/TEF, a horseshoe kidney, cervical hemivertebra, and left rib partial agenesis. A contrast examination showed that the TEF was at Th1-2, as was the upper end of the esophageal pouch (Figure 2). Thus, the patient underwent esophago-esophageal anastomosis and TEF ligation using a cervical approach. A contrast examination revealed no apparent anastomotic stricture. However, temporary left RLNP was present after the surgery but improved at day 57 of life. The patient suffered from congenital tracheal stenosis and required tracheoplasty. However, there were no apparent complications two years after the EA/TEF surgery.

CONTRAST EXAMINATION:

We routinely perform contrast examinations of the esophagus after birth in patients with suspected EA/TEF since 2013. Unstable patients do not undergo contrast examination. Contrast examinations are performed in a fluoroscopy room under awake, non-intubated conditions. We used iodixanol (Visipaque; iodine: 270 mg/mL; GE Healthcare Pharma, Tokyo, Japan) for the contrast examinations, which is a new, soluble, iso-osmotic (290 mOsm/kg), non-ionic contrast agent. Contrast is injected into the upper pouch via a 6-Fr nasogastric tube. The excess contrast that spills-over into the trachea and bronchial tree is used to assess for the level of the tracheoesophageal fistula. We did not intend to spill the excess contrast into the trachea for all cases. When the level of upper end of the esophagus was Th1 or Th2, we spilled over the contrast agent into the trachea to determine the surgical approach (ie, cervical or thoracoscopic approach). One or two ml of contrast agent was enough to evaluate the Th1-2 level trachea. An intratracheal suction tube was always prepared at the time of contrast study.

We also used and evaluated the contrast examinations from the front and lateral views by fluoroscopy (2–3 shots per second) to check the size and position of the upper esophagus and whether TEF and tracheomalacia existed. Fluoroscopy is reported to be highly specific, but lacks good sensitivity [6,7], but it may be of utility because it can clearly show trachea collapses during exhalation [8]. We simultaneously evaluate for tracheomalacia and esophagus problems.

SURGICAL METHOD:

The patient was positioned with a slight neck extension. Then, a 5-cm transverse incision was made 2 cm above the left clavicle. The platysma muscle was incised, and the left sternocleidomastoid muscle was encircled with vessel tape. Next, the left internal jugular vein was identified and encircled with vessel tape. The sternothyroid and sternohyoid muscles were dissected transversely to obtain a good surgical field, and the upper pouch end of the esophagus was freely mobilized. Here, the TEF was identified and resected by ligation with a 5-0 non-absorbable monofilament, and end-to-end esophago-esophageal anastomosis was performed. Then, a 6-Fr nasogastric trans-anastomotic feeding tube was placed into the stomach, and a Penrose drain was placed at the anastomotic site. Each resected muscle was sutured with an absorbable 5-0 monofilament, and the skin was subcuticularly closed with an interrupted suture using a 6-0 absorbable monofilament.

Discussion

We performed 48 type C EA/TEF surgeries between January 2014 and July 2022, with 4% (2/48) undergoing radical EA/TEF surgery by a cervical approach that achieved good outcomes in both patients.

The general criterion for adopting a cervical approach is based on the TEF location; if it is at Th2 or higher, a cervical approach should be used [9]. Unlike type E EA/TEF, esophago-esophageal anastomosis is required for type C EA/TEF. Hence, simply adopting this criterion for type C EA/TEF is questionable. However, a cervical approach has some advantages over a thoracic approach in managing the patient’s respiratory conditions during surgery. In our cases, the TEFs were above Th2; therefore, we concluded that a cervical approach was the better option.

We used iodixanol for the contrast examinations. In Japan, barium sulfate and diatrizoic acid are approved oral contrast agents. However, barium sulfate can cause granulation tissue formation at aspiration and perforation [10], and diatrizoic acid can cause aspiration pneumonia if accidentally aspired [11]. Conversely, off-label use of iodixanol is not approved for oral contrast examinations in Japan. However, a previous phase III multicenter trial showed that iodixanol is well-suited for gastrointestinal tract examinations in children (age range, 0–15 years) [12]. Iodixanol causes the same, or less, histopathologic lung reactions as normal saline and can be rapidly absorbed from the peritoneal cavity [13]. Additionally, even when accidentally aspirated, non-ionic contrast agents do not cause any significant complications because they can be safely used as contrast agents for tracheobronchoscopy [14]. We have performed this oral contrast examination for EA since 2013 without apparent complications.

Although three-dimensional computed tomography (CT) helps locate the position of the TEF, especially in neonates with EA/TEF, CT examination cannot always locate a small one. Furthermore, bronchoscopy can also assess the TEF location and is performed before the repair surgery in approximately 60% of EA/TEF cases [15]. Therefore, our institution’s first choice of preoperative evaluation is a contrast examination, but we also use bronchoscopy to evaluate the TEF location and tracheomalacia preoperatively, during surgery, and postoperatively. Bronchoscopy can mostly reveal the TEF location, but it can overlook TEF in some cases, even when repeating simple bronchoscopy alone [16]. Preoperative occult TEF can increase intraoperative risk by misrecognition and requires additional TEF repair after EA surgery. Contrast studies are easily performed under awake, non-intubated conditions and can improve the accuracy of preoperative diagnosis combined with bronchoscopy. We believe that accurate knowledge of TEF and other information through a simple and safe preoperative examination is desirable to ensure a reliable radical operation.

Conclusions

Contrast examinations with iodixanol help assess the TEF location and upper end of the esophageal pouch in EA patients with the suspected high location of TEF.

References:

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2.. Ward HC, Brereton RJ, Cervical repair of esophageal atresia: J Pediatr Surg, 1988; 23(9); 802-4

3.. Kemmotsu H, Joe K, Nakamura H, Yamashita M, Cervical approach for the repair of esophageal atresia: J Pediatr Surg, 1995; 30(4); 549-52

4.. Ford WD, Freeman JK, Martin AJ, Supraclavicular approach to cervical esophageal atresia with tracheoesophageal fistula: J Pediatr Surg, 1985; 20(3); 242-43

5.. Kosloske AM, Jewell PF, Cartwright KC, Crucial bronchoscopic findings in esophageal atresia and tracheoesophageal fistula: J Pediatr Surg, 1988; 23(5); 466-70

6.. Sanchez MO, Greer MC, Masters IB, Chang AB, A comparison of fluoroscopic airway screening with flexible bronchoscopy for diagnosing tracheomalacia: Pediatr Pulmonol, 2012; 47(1); 63-67

7.. Snijders D, Barbato A, An update on diagnosis of tracheomalacia in children: Eur J Pediatr Surg, 2015; 25(4); 333-35

8.. Sumida W, Yasui A, Shirota C, Update on aortopexy and posterior tracheopexy for tracheomalacia in patients with esophageal atresia: Surg Today, 2023 [Online ahead of print]

9.. Dai J, Pan Z, Wang Q, Experience of diagnosis and treatment of 31 H-type tracheoesophageal fistula in a single clinical center: Pediatr Surg Int, 2018; 34(7); 715-19

10.. McAlister WH, Askin FB, The effect of some contrast agents in the lung: An experimental study in the rat and dog: Am J Roentgenol, 1983; 140(2); 245-51

11.. Trulzsch DV, Penmetsa A, Karim A, Evans DA, Gastrografin-induced aspiration pneumonia: A lethal complication of computed tomography: South Med J, 1992; 85(12); 1255-56

12.. Wright NB, Carty HML, Sprigg A, Iodixanol in paediatric gastrointestinal imaging: Safety and efficacy comparison with iohexol: Br J Radiol, 2002; 75(890); 127-35

13.. Davenport D, Cohen MD, Hanna MP, Studies of iodixanol in the rabbit lung and peritoneum: Pediatr Radiol, 1999; 29(10); 724-30

14.. Lee T, Lee SK, Upper airway obstruction in infants and children: Evaluation by tracheobronchography with a non-ionic contrast agent: Pediatr Radiol, 1997; 27(3); 276-80

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16.. Levine H, Schonfeld T, Handelsman S, Low flow intermittent bronchoscopic oxygen insufflation to identify occult tracheo-esophageal fistulas: Respir Med, 2021; 186; 106544

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American Journal of Case Reports eISSN: 1941-5923
American Journal of Case Reports eISSN: 1941-5923