Right Phrenic Nerve Injury: A Rare but Possible Complication of Oesophageal Atresia and Tracheoesophageal Fistula Repair

Sanathvaran Sagadevan; Venod Nambiar Somoo; Ashok Krishnan; Mohd Fauzi Sharudin; Quincy Lim; Rosdara Masayuni Mohd Sani

doi:10.33102/mjosht.v8i.287

Authors

Sanathvaran Sagadevan School of Medicine, Monash University Malaysia, Clinical School Johor Bahru, No 8, Jalan Masjid Abu Bakar, 80100 Johor Bahru, Johor, Malaysia.
Venod Nambiar Somoo School of Medicine, Monash University Malaysia, Clinical School Johor Bahru, No 8, Jalan Masjid Abu Bakar, 80100 Johor Bahru, Johor, Malaysia.
Ashok Krishnan Paediatric Surgery Department, Hospital Sultanah Aminah Johor Bahru, Jalan Persiaran Abu Bakar Sultan, 80100 Johor Bahru, Johor, Malaysia.
Mohd Fauzi Sharudin Paediatric Surgery Department, Hospital Sultanah Aminah Johor Bahru, Jalan Persiaran Abu Bakar Sultan, 80100 Johor Bahru, Johor, Malaysia.
Quincy Lim Paediatric Surgery Department, Hospital Sultanah Aminah Johor Bahru, Jalan Persiaran Abu Bakar Sultan, 80100 Johor Bahru, Johor, Malaysia.
Rosdara Masayuni Mohd Sani Emergency Medicine Department, Monash University Malaysia, Clinical School Johor Bahru, No 8, Jalan Masjid Abu Bakar, 80100 Johor Bahru, Johor, Malaysia.

DOI:

https://doi.org/10.33102/mjosht.v8i.287

Keywords:

Phrenic Nerve Palsy, Oesophageal Atresia, Tracheoesophageal Fistula, Non-operative management, Thermal Injury

Abstract

Oesophageal atresia (OA) and Tracheoesophageal fistulas (TOF) are congenital anomalies commonly present in between 1 and 4 per 10 000 births, including stillbirths and termination of pregnancies. Currently, patients with these anomalies are are managed surgically, with common complications including oesophageal formation, anastomotic leakage and recurrence of TOF. This case report highlights the possibility of right phrenic nerve injury as a complication of OA and TOF repair. We report a baby boy born at term with worsening respiratory distress at day 15 of life, two weeks after undergoing an operation to repair OA and TOF. Since the surgery on day 2 of life, he could not be weaned off oxygen support successfully and had two episodes of worsening respiratory distress that was treated as pneumonia. Serial Chest X-Rays (CXR) showed a persistent elevation of the right hemidiaphragm. Ultrasound (USG) thorax supported the findings of right diaphragmatic paralysis by revealing minimal right diaphragm excursion with the paradoxical movement of the right diaphragm. He was initially managed non-operatively with non-invasive ventilation (NIV)/continuous positive airway pressure (CPAP) while monitoring for spontaneous recovery of the right diaphragm, which unfortunately did not occur. He was on NIV/CPAP for a total of 25 days and was tachypnoeic with mild subcostal recessions (SCR) and reduced breath sounds on the right throughout. Six weeks after the initial OA/TOF repair surgery, he was posted for Right Diaphragmatic Plication. Afterwards, he recovered well and was weaned off oxygen support within the next seven days and subsequently discharged.

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Author Biography

Rosdara Masayuni Mohd Sani, Emergency Medicine Department, Monash University Malaysia, Clinical School Johor Bahru, No 8, Jalan Masjid Abu Bakar, 80100 Johor Bahru, Johor, Malaysia.

Emergency Physician and Senior Lecturer

References

Brosens, E., Ploeg, M., van Bever, Y., Koopmans, A.E., IJsselstijn, H., Rottier, R.J., Wijnen, R., Tibboel, D. and de Klein, A., 2014. Clinical and etiological heterogeneity in patients with tracheo-esophageal malformations and associated anomalies. European journal of medical genetics, 57(8), pp.440-452. doi.org/10.1016/j.ejmg.2014.05.009

Lal, D.R., Gadepalli, S.K., Downard, C.D., Ostlie, D.J., Minneci, P.C., Swedler, R.M., Chelius, T., Cassidy, L., Rapp, C.T., Deans, K.J. and Fallat, M.E., 2017. Perioperative management and outcomes of esophageal atresia and tracheoesophageal fistula. Journal of pediatric surgery, 52(8), pp.1245-1251. doi.org/10.1016/j.jpedsurg.2016.11.046

Gross, E. R. (1962). The surgery of infancy and chilhood.

Lee, S., & Meeker, T. M. (2018). Basic knowledge of tracheoesophageal fistula and esophageal atresia. Advances in Neonatal Care, 18(1), 14-21. doi.org/10.1097/ANC.0000000000000464

Henderson, P. W., & Spigland, N. A. (2010). Right phrenic nerve injury as a complication of tracheoesophageal fistula repair. Pediatric Critical Care Medicine, 11(5), e52-e54. doi.org/10.1097/PCC.0b013e3181dde5c3

Man, D., Wheildon, M. H., & Eckstein, H. B. (1982). Paralysis of the right hemidiaphragm following primary anastomosis for oesophageal atresia and tracheo-oesophageal fistula. Zeitschrift für Kinderchirurgie, 37(09), 32-33. doi.org/10.1055/s-2008-1059812

Haller Jr, J. A., Pickard, L. R., Tepas, J. J., Rogers, M. C., Robotham, J. L., Shorter, N., & Shermeta, D. W. (1979). Management of diaphragmatic paralysis in infants with special emphasis on selection of patients for operative plication. Journal of pediatric surgery, 14(6), 779-785. doi.org/10.1016/S0022-3468(79)80264-X

Mok, Q., Ross-Russell, R., Mulvey, D., Green, M., & Shinebourne, E. A. (1991). Phrenic nerve injury in infants and children undergoing cardiac surgery. Heart, 65(5), 287-292. dx.doi.org/10.1136/hrt.65.5.287

Watanabe, T., Trusler, G. A., Williams, W. G., Edmonds, J. F., Coles, J. G., & Hosokawa, Y. (1987). Phrenic nerve paralysis after pediatric cardiac surgery: retrospective study of 125 cases. The Journal of Thoracic and Cardiovascular Surgery, 94(3), 383-388. doi.org/10.1016/S0022-5223(19)36251-8

Fraser III, C.D., Ravekes, W., Thibault, D., Scully, B., Chiswell, K., Giuliano, K., Hill, K.D., Jacobs, J.P., Jacobs, M.L., Kutty, S. and Vricella, L., 2021. Diaphragm paralysis after pediatric cardiac surgery: An STS Congenital Heart Surgery Database study. The Annals of Thoracic Surgery, 112(1), pp.139-146. doi.org/10.1016/j.athoracsur.2020.05.175

Parmar, D., Panchal, J., Parmar, N., Garg, P., Mishra, A., Surti, J., & Patel, K. (2021). Early diagnosis of diaphragm palsy after pediatric cardiac surgery and outcome after diaphragm plication–A single-center experience. Annals of Pediatric Cardiology, 14(2), 178. doi.org/10.4103/apc.APC_171_19

Mickell, J. J., Oh, K. S., Siewers, R. D., Galvis, A. G., Fricker, F. J., Mathews, R. A., & Bahnson, H. T. (1978). Clinical implications of postoperative unilateral phrenic nerve paralysis. The Journal of Thoracic and Cardiovascular Surgery, 76(3), 297-304. doi.org/10.1016/S0022-5223(19)39570-4

Iverson, L. I., Mittal, A., Dugan, D. J., & Samson, P. C. (1976). Injuries to the phrenic nerve resulting in diaphragmatic paralysis with special reference to stretch trauma. The American Journal of Surgery, 132(2), 263-269. doi.org/10.1016/0002-9610(76)90058-1

Floh, A. A., Zafurallah, I., MacDonald, C., Honjo, O., Fan, C. P. S., & Laussen, P. C. (2017). The advantage of early plication in children diagnosed with diaphragm paresis. The Journal of Thoracic and Cardiovascular Surgery, 154(5), 1715-1721. doi.org/10.1016/j.jtcvs.2017.05.109

Affatato, A., Villagra, F., De Leon, J.P., Gomez, R., Checa, S.L., Vellibre, D., Sanchez, P., JI, D.B. and Brito, J.M., 1988. Phrenic nerve paralysis following pediatric cardiac surgery. Role of diaphragmatic plication. The Journal of Cardiovascular Surgery, 29(5), pp.606-609.

Hamilton, J. R. L., Tocewicz, K., Elliott, M. J., De Leval, M., & Stark, J. (1990). Paralysed diaphragm after cardiac surgery in children: value of plication. European journal of cardio-thoracic surgery, 4(9), 487-491. doi.org/10.1016/1010-7940(90)90171-U

Baker, C. J., Boulom, V., Reemtsen, B. L., Rollins, R. C., Starnes, V. A., & Wells, W. J. (2008). Hemidiaphragm plication after repair of congenital heart defects in children: quantitative return of diaphragm function over time. The Journal of Thoracic and Cardiovascular Surgery, 135(1), 56-61. doi.org/10.1016/j.jtcvs.2007.09.031

Schwartz, M. Z., & Filler, R. M. (1978). Plication of the diaphragm for symptomatic phrenic nerve paralysis. Journal of pediatric surgery, 13(3), 259-263. doi.org/10.1016/S0022-3468(78)80397-2

Stone, K. S., Brown, J. W., Canal, D. F., & King, H. (1987). Long-term fate of the diaphragm surgically plicated during infancy and early childhood. The Annals of thoracic surgery, 44(1), 62-65. doi.org/10.1016/S0003-4975(10)62359-4

Lemmer, J., Stiller, B., Heise, G., Alexi-Meskishvili, V., Hübler, M., Weng, Y., & Berger, F. (2007). Mid-term follow-up in patients with diaphragmatic plication after surgery for congenital heart disease. Intensive care medicine, 33(11), 1985-1992. doi.org/10.1007/s00134-007-0717-9

Goldberg, L., Krauthammer, A., Ashkenazi, M., Soudack, M., Tokatly Latzer, I., Vardi, A., & Paret, G. (2020). Predictors for plication performance following diaphragmatic paralysis in children. Pediatric Pulmonology, 55(2), 449-454. doi.org/10.1002/ppul.24539

Snyder, C. W., Walford, N. E., Danielson, P. D., & Chandler, N. M. (2014). A simple thoracoscopic plication technique for diaphragmatic eventration in neonates and infants: technical details and initial results. Pediatric surgery international, 30(10), 1013-1016. doi.org/10.1007/s00383-014-3580-0