Congenital H-type tracheoesophageal fistula is a rare anomaly in infants and the early diagnosis of this disorder is still a challenge to pediatricians due to scarcity, non-specific symptoms and lack of a single diagnostic examination. We report the case of a 3-month-old baby with choking and recurrent aspiration which finally turned out to be a tracheoesophageal fistula without esophageal atresia (H-type) by radionuclide salivagram (Fig. 1).
Fig. 1.
A 3-month-old baby girl with choking and recurrent aspiration was referred to our hospital for the surgical correction of an alleged laryngeal diverticulum. She was a full-term baby born by Cesarean section without any perinatal complication. One month ago, she underwent a chest computed tomography (CT) scan in the local hospital owing to chief complaints above. Her symptoms seemed to originate from a laryngeal diverticulum seen on the chest CT. However, when laser microscopic surgery for the correction of an alleged laryngeal diverticulum was performed in our hospital, no abnormal finding was seen in the larynx. Afterwards, she was consulted to the general pediatric department to work up for other reasons for the aspiration. Then, radionuclide salivagram was performed by a sublingual dropping of 0.1 mCi (3.7 MBq) of Tc-99 m Phytate with 0.3 ml saline. The serial records for a 10-min dynamic image of anterior view (a) and one frame of a delayed 15-min static image of anterior and posterior view (b) are shown, respectively. An abnormal linear radioactivity was observed in right paramedian lung field with atypical aspiration feature (black arrow). On barium esophagography, H-type TEF was confirmed by aspirated contrast material (c, white arrow), and 3-mm fistula tract was ligated by pediatric surgeon in the end. Congenital H-type trahceoesophageal fistula (TEF) is an extremely rare anomaly in infants and the early diagnosis of this disorder is still a challenge to pediatricians due to scarcity, non-specific symptoms and lack of a single diagnostic examination [1–3]. Radionuclide salivagram (RS) is a physiological modality designed to assess the pulmonary aspiration using only a tiny volume of saline sublingually and has been widely used with the advantages of less radiation exposure and minimal patient cooperation [4, 5]. Previously, two cases of H-type TEF detected by scintigraphy technique were reported, and each of the results was accomplished by esophageal transit study, a somewhat different scintigraphic modality compared with RS [6–8]. Even though, the process of procedure and the clinical application is absolutely distinguishable from two studies, considering the challenge of early diagnosis in H-type TEF, radionuclide study could be a considerable option to patients in whom H-type TEF is suspected
References
- 1.Beasley SW, Myers NA. The diagnosis of congenital tracheoesophageal fistula. J Pediatr Surg. 1988;23:415–417. doi: 10.1016/S0022-3468(88)80437-8. [DOI] [PubMed] [Google Scholar]
- 2.Karnak I, Senocak ME, Hicsonmez A, Buyukpamukcu N. The diagnosis and treatment of H type tracheoesophagea fistula. J Pediatr Surg. 1997;32:1670–1674. doi: 10.1016/S0022-3468(97)90503-0. [DOI] [PubMed] [Google Scholar]
- 3.Genty E, Attal P, Nicollas R, Roger G, Triglia JM, Garabedian EN, et al. Congenital tracheoesophagea fistula without esophageal atresia. Int J Pediatr Otorhinolaryngol. 1999;48:231–238. doi: 10.1016/S0165-5876(99)00039-7. [DOI] [PubMed] [Google Scholar]
- 4.Heyman S, Respondek M. Detection of pulmonary aspiration in children by radionuclide “salivagram”. J Nucl Med. 1989;30:697–699. [PubMed] [Google Scholar]
- 5.Akbunar AT, Kiristioglu I, Alper E, Demiray H. Diagnosis of orotracheal aspiration using radionuclide salivagram. Ann Nucl Med. 2003;17:415–416. doi: 10.1007/BF03006612. [DOI] [PubMed] [Google Scholar]
- 6.Kapoor S, Ratan SK, Gathwala G, Mehndiratta S, Kashyap R. Bronchoesophageal fistula in a neonate: radionuclide study as diagnostic modality. Am J Perinatol. 2003;20:341–345. doi: 10.1055/s-2003-45281. [DOI] [PubMed] [Google Scholar]
- 7.Vatansever U, Acunas B, Salman T, Altun G, Duran R. A premature infant with H-type tracheoesophageal fistula demonstrated by scintigraphic technique. Clin Nucl Med. 2006;31:451–453. doi: 10.1097/01.rlu.0000226898.53461.90. [DOI] [PubMed] [Google Scholar]
- 8.Iascone C, Di Giulio E, Maffi C, Ruperto M. Use of radioisotopic esophageal transit in the assessment of patients with symptoms of reflux and non-specific esophageal motor disorders. Dis Esophagus. 2004;17(3):218–222. doi: 10.1111/j.1442-2050.2004.00411.x. [DOI] [PubMed] [Google Scholar]