Abstract
Introduction: Tonsillectomy is a safe and common procedure. It has not been associated with the development of hypercontractile conditions of the esophagus. Jackhammer esophagus is a type of hypercontractile esophagus with vigorous contraction of the esophagus associated with dysphagia and chest pain. Case Presentation: A 42-year-old woman with no previous history of gastroesophageal dysfunction or symptoms developed Jackhammer esophagus with severe dysphagia and chest pain after undergoing tonsillectomy and adenoidectomy. Conclusion: Although Jackhammer esophagus has been reported as a complication of several invasive procedures, this is the first report of Jackhammer esophagus as a complication of tonsillectomy and adenoidectomy.
Introduction
Tonsillectomy is a common and safe procedure with approximately 399,000 performed in the USA per the most recent National Health Statistics Report put out by the CDC [1]. Most tonsillectomies are performed on children but over 100,000 are performed on adults. Complications appear to occur more commonly in adults, but the data vary. Bleeding and readmission for pain control are the most common complications each occurring in about 5% of cases [2, 3]. There are no reported complications of esophageal dysfunction after tonsillectomy. We report a case of Jackhammer esophagus, an esophageal motility disorder characterized by hypertensive progressive peristaltic contractions associated with chest pain and dysphagia, following tonsillectomy and adenoidectomy.
Case
A 42-year-old ER nurse underwent tonsillectomy and adenoidectomy for recurrent tonsilitis in April 2016. Postoperatively, she developed a change in her voice and ongoing right pharyngeal/throat pain, dysphagia, odynophagia, chest pain. She reported these symptoms on her first follow-up appointment on day 15. She had no prior history of gastrointestinal reflux disease, dysphagia, chest pain, or odynophagia. She continued to report these symptoms in follow-up on days 34 and 51; conservative measures and a trial of prednisone were not helpful. Direct laryngoscopy and cervical esophagoscopy in August 2016 were reported as unremarkable. Symptoms persisted for years and could be incapacitating, described as chest pain spasms and “a vice closing around my esophagus” that lasted seconds, minutes, or as long as 4 h. Symptoms awakened her at night and interfered with sleep, eating, and work. Eating difficulties persisted to the point she was no longer able to work as a nurse.
In January 2017, high-resolution esophageal motility showed 10 of 10 swallows with a distal contractile integral (DCI) >8,000; the mean DCI was >15,000, confirming the diagnosis of Jackhammer esophagus (shown in Fig. 1). Repeat manometry in September 2021 again showed Jackhammer esophagus with 4 of 9 swallows greater than 8,000 and a mean DCI of 10,500 (shown in Fig. 2, 3). Her LES pressures were mildly elevated (45) (normal 13–43 mm Hg), and her IRP was normal (6.5 mm Hg). 24-h pH studies showed an upright acid exposure time of 2.3 min and a recumbent acid exposure time of 0.0 min. The DeMeester score was 1.1. In the upright position, there were 56 impedance episodes (14 acid, 42 nonacid) and in the recumbent position there was one impedance episode (nonacid). Video swallowing studies were normal as was her radionucleotide gastric empty study. Three upper endoscopies with dilation and Botox injection proved unhelpful. A POEM procedure (with no reassurance of success) has been discussed; and a G-tube has been offered.
High-resolution esophageal manometry, January 2017. Swallow composite (mean of 10 swallows) displaying elevated DCI 15,214 mm Hg-cm-s.
High-resolution esophageal manometry, January 2017. Swallow composite (mean of 10 swallows) displaying elevated DCI 15,214 mm Hg-cm-s.
High-resolution esophageal manometry, September 2021. Swallow composite displayed elevated DCI 10,500 mm Hg-cm-s.
High-resolution esophageal manometry, September 2021. Swallow composite displayed elevated DCI 10,500 mm Hg-cm-s.
In 2023, she underwent laryngostroboscopy and laryngeal EMG. EMG found injury to the superior laryngeal nerve (SLN), a branch of the vagal nerve, and this was thought to be consistent with her voice changes.
Discussion
Hypercontractile esophageal conditions have long been described with dysphagia and chest pain as the most common symptoms. The term nutcracker esophagus was introduced by Benjamin and Castell in 1980 to describe the disorder. With the advent of high-resolution esophageal manometry (HREM) and the Chicago Classification, nutcracker esophagus has been replaced by the term Jackhammer esophagus and is defined by a DCI >8,000 mm Hg-s-cm in 20% of swallows. Since the average HREM consists of ten swallows, that would be just two swallows to make a firm diagnosis. In an earlier version of the Chicago Classification, only one hypercontractile swallow was required to make a diagnosis of Jackhammer esophagus [4]. In the most recent edition of the Chicago Classification, version 4, the diagnosis of Jackhammer esophagus requires both 20% or more hypercontractile swallows and associated symptoms such as dysphagia or noncardiac chest pain [5]. When our patient was initially evaluated, all ten of her swallows were hypercontractile. The prevalence of Jackhammer esophagus is about 2% of those sent for HREM with atypical chest pain being a common reason to ordering the test [6]. However, it should be noted there was no HREM study prior to her tonsillectomy and therefore we cannot be certain that a hypercontractile condition did not exist prior to her tonsillectomy.
The etiology of Jackhammer esophagus is unknown but is felt to be an imbalance between excitatory input (cholinergic) and inhibitory input via local pathways of the vagal nerve [7]. Jackhammer esophagus has been reported in patients undergoing radiofrequency catheter ablation for atrial fibrillation and gastric bypass surgery [8]. It has been reported in a high number of patients undergoing lung transplantation [9]. Cangemi et al. [10] in a retrospective study of 57 patients who had esophageal manometry before surgery found 15 patients developed jackhammer esophagus after transplantation.
The vagus nerve exits the base of the skull with cranial nerves IX, XI, and XII, then travels in the carotid sheath with the internal jugular vein lateral and the carotid artery medial and somewhat anterior to the vagus nerve [11]. The SLN is a branch of the vagus nerve and divides into an internal and external branch (shown in Fig. 4). The external branch provides motor activity to the cricothyroid muscle. The internal branch delivers sensory input to the supraglottic larynx. Damage to the external branch is associated with decreased vocal pitch, phonation, increased throat clearing, and hoarseness. Damage to the internal branch is associated with lack of sensation and dysphagia. Damage to the SLN occurs most frequently during thyroid or parathyroid surgery and overall little is known about this type of injury [12]. Scerrino et al. [13] prospectively studied 36 patients with esophageal manometry before and after thyroidectomy. Twenty percent reported worsening of dysphagia after surgery; this was associated with “incoordination” of the UES, but esophageal manometry remained normal in all patients. There are anatomic variations in the path of the external SLN and anatomic variation may have played a role in our patients’ injury [14]. Jackhammer esophagus has not been reported with injury to the SLN.
Pathway of the SLN with internal and external branches and the recurrent laryngeal nerve (Netter illustration used with permission of Elsevier Inc., all rights reserved, www.netterimages.com).
Pathway of the SLN with internal and external branches and the recurrent laryngeal nerve (Netter illustration used with permission of Elsevier Inc., all rights reserved, www.netterimages.com).
In our patient, fiberoptic laryngoscope exam on postoperative day 34 showed the right tonsillar fossa was still not healed and “slightly deeper than the left”; we speculate that damage to the vagal nerve or branches of the vagal nerve in the right tonsillar fossa from thermal injury contributed to her esophageal dysmotility.
In conclusion, this is the first case report of Jackhammer esophagus following tonsillectomy. It is unknown what the mechanism of injury is that caused her jackhammer esophagus. The CARE Checklist has been completed by the authors for this case report, attached as online supplementary material (for all online suppl. material, see https://doi.org/10.1159/000542891).
Statement of Ethics
This retrospective review of patient data did not require ethical approval in accordance with local and national guidelines. Written informed consent was obtained from the patient for publication and any accompanying images.
Conflict of Interest Statement
The authors have no conflicts of interest to declare.
Funding Sources
This study was not supported by any sponsor or funder.
Author Contributions
W. Michael McDonnell: data interpretation, manuscript composition, and article guarantor. Alison Tewnion: data collection and manuscript review.
Data Availability Statement
All data generated or analyzed during this study are included in this article and its online supplementary material files. Further inquiries can be directed to the corresponding article.