Abstract

A patient with pica and Lennox Gastaut syndrome suddenly refused oral intake. Neck radiographs revealed no foreign body. Barium swallow identified an irregular filling defect in the cervical esophagus. Esophagoscopy showed a gold ball-like object (half a lemon) 3 cm distal to the cricopharyngeus. This object had to be removed by esophagotomy after failed attempts with flexible and rigid esophagoscopy, laryngoscopy, and a Foley catheter. The charge for her 39-day hospitalization was $282,761. She had ingeniously procured and swallowed the lemon despite a full-face plastic shield and staff surveillance. In nonverbal persons, medically unexplainable abrupt food-refusal is an emergency. Prevention necessitates attention to pica, impulsivity, dentition, food consistency, eating utensils, seizures, environment, adaptive equipment, and surveillance.

Foreign body ingestion is a common problem. Nearly 90% of swallowed objects traverse the gut uneventfully in less than 7 days. Esophageal obstruction, with its potential complications (perforation, fistulization to trachea or heart, sepsis, abscess, mediastinitis, and death) may occur in the remainder (Galera, Morales, & Prado 2003; Mittelman, Perek, & Kolkov, 1985; Oghalai, 2002). The literature contains numerous reports of esophageal obstruction by a plethora of objects (coin, meat, bone, pin, blade, fish bone, nail, mouse, dental bridge, barbecue skewer) (e.g., Hamilton & Polter, 1998). Individuals with developmental disabilities are particularly at risk of foreign body ingestion because of pica, dysphagia, tooth loss, or impulsive swallowing (Dallal, Odum, & Ahluwalia, 1996; Reilly & Carr, 2001). Here we illustrate the difficulty of diagnosing this condition in a nonverbal patient and suggest preventive strategies.

Case Report

Our patient was an ambulatory 41-year-old woman, with fair hearing and vision, who had lived in a developmental center for 38 years. She had Lennox Gastaut syndrome (mixed epilepsy and encephalopathy), profound mental retardation, hypotonic macroglossia, prognathism, and kyphosis. She wore a helmet to prevent self-injury and a full-face plastic shield for pica control. This patient received fluoxetine, trazodone, and olanzapine for control of maladaptive behavior and impulsive eating; levothyroxine for hypothyroidism; and diazepam, valproic acid, levetiracetam, and phenobarbital for mixed seizures. Her seizure frequency was 1 to 23 (M = 9) per year; seizures lasted 10 to 90 seconds. At mealtime, she habitually stuffed her mouth with food and swallowed relatively large pieces without adequate mastication. She had only 18 teeth; all her incisors were missing, probably because of prior injuries. At 28 years of age, she had choked on a 2 × 7 cm piece of bone, which was successfully extricated by the Heimlich maneuver. She had no prior esophageal burn or surgery.

One day, this patient had a tonic–clonic seizure and choked on a tablet. The Heimlich maneuver was ineffective. She vomited and subsequently refused oral intake. She was physically strong (weight 83 kg, height 170 cm) and did not permit an oropharyngeal examination. Her chest radiograph was normal, and an abdominal X-ray showed only fecal impaction. She was hospitalized. A swallowing evaluation by a speech pathologist revealed pharyngeal phase dysphagia. Neck soft-tissue radiographs showed no foreign body (Figure 1). A barium swallow study identified a filling defect (Figure 2). Esophagoscopy revealed a golf-ball-like object that could not be grabbed by a polypectomy snare (Figure 3). An otorhinolaryngologist could not remove this object with sharp, grasping, or rotating prongs of a large, rigid Hessburg esophagoscope, or a Holinger laryngoscope; he could not navigate a Foley catheter around it. Eventually, a thoracic surgeon performed an esophagotomy via a 3 cm incision in the lower anterior neck, and pushed this object towards the mouth where it was grabbed by the anesthesiologist. There was some local necrosis of the esophageal mucosa from the object, which was half of a lemon piece measuring 6 × 3 × 1.5 cm, with its cut surface facing distally; it had not been served at her meals. On the fifth postoperative day, the patient developed right basal pneumonia, which was treated with intravenous ampicillin-sulbactam. On Day 15, a leak was noted in the neck wound, which required surgical exploration and a Penrose drain. The patient was discharged on Day 39. Hospital charges, exclusive of physicians' fees, were $282,761.

Figure 1

Lateral view of neck soft tissues reveals no radio-opaque foreign body or intraluminal free air in the esophagus. However, the lower three cervical vertebrae and the cervical esophagus are obscured by dense soft tissue (arrow)

Figure 1

Lateral view of neck soft tissues reveals no radio-opaque foreign body or intraluminal free air in the esophagus. However, the lower three cervical vertebrae and the cervical esophagus are obscured by dense soft tissue (arrow)

Figure 2

Barium swallow reveals an irregular filling defect in the cervical esophagus suggestive of a foreign body or a tumor (arrows). Some barium passed distally, indicating that the obstruction was not totally complete. This is probably why the patient did not drool

Figure 2

Barium swallow reveals an irregular filling defect in the cervical esophagus suggestive of a foreign body or a tumor (arrows). Some barium passed distally, indicating that the obstruction was not totally complete. This is probably why the patient did not drool

Figure 3

Esophagoscopy revealed a round speckled golf-ball-like object almost completely blocking the cervical esophagus 3 cm distal to the cricopharyngeus

Figure 3

Esophagoscopy revealed a round speckled golf-ball-like object almost completely blocking the cervical esophagus 3 cm distal to the cricopharyngeus

Discussion

An esophageal foreign body should be seriously considered in all nonverbal patients with pica or impulsive swallowing who suddenly refuse oral intake. Early diagnosis is essential to prevent complications and requires a high index of suspicion, detailed communication with carergivers, and consideration of prompt esophagoscopy. The usual historic clues (swallowing something unusual, choking, coughing, gagging, drooling, local foreign body sensation, odynophagia) may be absent in nonverbal patients with developmental disability, especially if the injurious event went unnoticed or if the obstruction is partial (Kozma & Mason, 2003; Nijhawan, Shimp, & Mathur, 2003; Ren, 2002; Roffman, Scharukh, & Hybels, 2002). Physical examination may reveal drooling and agitation; otherwise, it is not very helpful because the trachea, neck muscles, and spine prevent direct esophageal inspection or palpation. Five physicians (attending, emergency, admitting, gastroenterology, otorhinolaryngology), a speech pathologist, and several nurses noted no anomaly in this patient's throat, neck, chest, or abdomen. Although dysphagia affects as many as 22% of people with developmental disabilities (Kozma & Mason, 2003), medically unexplainable sudden dysphagia should not be reflexively assumed to be functional or behavioral. Had the emergency room physician realized the magnitude of this patient's pica and impulsive swallowing and had the patient been able to communicate her symptoms, an esophageal foreign body might have been suspected and promptly removed, obviating her extended suffering, multiple surgeries, and hospitalization.

For suspected esophageal foreign bodies, emergent esophagoscopy (a relatively safe procedure) deserves prompt consideration because it allows simultaneous diagnosis and removal (Lam, Woo, & Hasselt, 2001). No time should be wasted in empiric dysphagia tests, such as a barium swallow or swallowing evaluation. Barium residue may actually make the endoscopist's job more difficult (Athanassiadi, Gerazounis, & Metaxas, 2002). In our patient, coating of the lemon skin-pores by white barium from the preceding barium swallow gave it a golf-ball resemblance. Some endoscopists may become discouraged by such a miscue because it would be difficult to grab a hard round object. This lemon could not be removed esophagoscopically, probably because of mucosal edema caused by the delay in diagnosis. Reddy, Bhatt, and Vaughan (2002) have used a balloon angiographic catheter to extricate an otherwise nonremovable steel ball. However, a somewhat similar device (Foley catheter) could not be navigated beyond the lemon in our patient. Esophagotomy was the treatment of last resort.

Soft-tissue neck radiographs are often used to visualize suspected esophageal foreign bodies. They may show a radioopaque object or intraluminal air (an indirect evidence of a foreign body holding open the normally collapsed esophagus). However, such films are often nondiagnostic or misinterpreted because of silhouettes from the calcified laryngeal cartilages, clavicles, or dense soft tissue of the lower neck (Figure 1). Under such circumstances, Kanagalingam, Georgalas, and Zainal (2002) have used a swimmer's view (one arm in full overhead flexion, other arm beside the trunk in neutral position) to identify a clamshell in the glottis. Unfortunately, it may not be feasible to obtain this view in noncooperative patients.

Persons with developmental disability ingest foreign bodies because of impulsivity, impaired swallowing controls (food–nonfood discrimination, motor skills, hand–mouth activity, protective reflexes, and intraoral sensitivity and control), and their amazing ingenuity. None of our staff members knew that the patient had procured and swallowed the lemon, despite a full-face plastic shield and extensive staff surveillance. This event illustrates the challenges we face in caring for people with developmental disabilities. Many of our 780 residents are predisposed to foreign body ingestion because of epilepsy (n = 375, 48%), maladaptive behavior (n = 366, 47%), dysphagia (n = 191, 24%), and pica (n = 86, 11%). The fact that such accidents are not common is a tribute to our caregivers' vigilance, dedication, and training. In Table 1, we present strategies to help prevent foreign body ingestion.

Table 1

Conditions Predisposing to Foreign Body Ingestion and Preventive Strategies to Manage Them

Conditions Predisposing to Foreign Body Ingestion and Preventive Strategies to Manage Them
Conditions Predisposing to Foreign Body Ingestion and Preventive Strategies to Manage Them

This case was presented at the 21st Quarterly Coastal-Fairview Grand Rounds on April 30, 2003. We thank Timothy Foley, Donna Rodriguez, and personnel at the Coastal Communities Hospital, Santa Ana, California, for providing the records and Barbara Rycroft for providing library services. Digital Presentation International prepared the photographs. This study was funded by the State of California (Department of Developmental Services and Fairview Developmental Center). Opinions expressed herein are those of the authors only.

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Author notes

Authors: Ghan-Shyam Lohiya, MD (glohiya@fdc.dds.ca.gov), Public Health Officer; Lilia Tan-Figueroa, MD, Medical Director; Hung Van Le, MD, Staff Physician; Lucia Rusu, MD, Staff Physician, Fairview Developmental Center, 2501 Harbor Blvd., Costa Mesa, CA 92626