A Variant Form of Nasogastric Tube Syndrome

Abstract

Nasogastric tube syndrome named by Sofferman et alin 1981 is a laryngeal complication presenting with life-threatening vocal cord abductor paralysis derived fromperoforation of the NG tube-induced esophageal ulcer.As compared with the previously reported cases of thissyndrome, our 4 autopsied patients were so peculiar inthe following two points that vocal cord abductor paraly-sis developed repeatedly and no esophageal ulcer waspresent in spite of the presence of the laryngeal abductormuscle injury. We hypothesized that the etiology of sucha variant form was circulatory injury of the laryngeal ab-ductor muscle which was caused by the compression ofthe postcricoid blood vessels perfusing this muscle.Nasogastric tube syndrome, which is treatable by decan-nulation, cannot be ruled out even if no esophageal ulceris detected by fiberoptic laryngoscopy.(Internal Medicine 44: 1286–1290, 2005)

Key words: nasogastric tube syndrome, vocal cord abductorparalysis, posterior cricoarytenoid muscle,stridor

Introduction

In 1939, Iglauer and Molt (1) reported the patients pre-senting with bilateral vocal cord abductor paralysis (VCAP)which was caused by injuries of the laryngeal abductor, theposterior cricoarytenoid muscle (PCA), derived from the per-foration of the nasogastric (NG) tube-induced esophagealulcer. Sofferman et al (2) named such a pathophysiologicalentity as NG tube syndrome, showing the clinical triad: 1)presence of a NG tube, 2) throat pain and stridor, and 3)presence of VCAP. Considering that tube feeding through aNG tube is often put into practice in patients withneurodegenerative disorders, especially in the advancedstage, the possibility of NG tube syndrome, which is a life-

threatening but a treatable complication, should be notedwhen they develop sore throat or stridor.

Four autopsied patients described here are non-diabeticpatients with NG tube syndrome who had characteristic fea-tures including the absence of postcricoid esophageal ulcerand recurrent episodes of VCAP. Similar cases have notbeen reported in the past. We describe the clinical historiesof two representative cases among the four patients and dis-cuss the etiology of such a variant form of NG tube syn-drome.

Case Report

Case 1A 73-year-old man developed bradykinesia and dementia

at the age of 68. Two years later, his parkinsonism deterio-rated rapidly with no response to l-DOPA treatment. He wasalmost bed-ridden requiring NG tube feeding. On July 30,1994, at the age of 72, he developed inspiratory stridor,which was caused by moderately severe VCAP by fiberopticlaryngoscopy. Arterial blood gas analysis under oxygen in-halation showed pH=7.44, pCO2=38 Torr, pO2=89 Torr. Inthe following month under no treatment, his inspiratorystridor gradually decreased, and he was discharged with sub-tle inspiratory stridor. On February 6, 1995, he was readmit-ted to our hospital because of pneumonia. After recoveryfrom pneumonia with antibiotics, mild inspiratory stridorwas still continued and fiberoptic laryngoscopy demon-strated mild to moderately severe VCAP. Arterial blood gasanalysis on room air showed pH=7.495, pCO2 =43 Torr,pO2=76 Torr. On August 4, 1995, he was again admitted toour hospital because of increased stridor and dyspnea.Arterial blood gas analysis at room air showed severehypoxemia; pH=7.445, pCO2=38 Torr, pO2=40 Torr. The eti-ology of acute respiratory failure was thought to be deterio-ration of VCAP since neither inflammatory reaction of theblood sample nor pneumonia shadow on the chest X-ray filmwas found. He died of respiratory failure on September 6,1995.

Postmortem pathological examination confirmed the diag-

Internal Medicine Vol. 44, No. 12 (December 2005)1286

A Variant Form of Nasogastric Tube Syndrome

Eiji ISOZAKI, Shinsuke TOBISAWA, Rie NAITO*, Toshio MIZUTANI** and Hideaki HAYASHI

□ CASE REPORT □

From the Departments of Neurology, *Neuro-otology and **Pathology, Tokyo Metropolitan Neurological Hospital, TokyoReceived for publication May 2, 2005; Accepted for publication July 27, 2005Reprint requests should be addressed to Dr. Eiji Isozaki, the Department of Neurology, Tokyo Metropolitan Neurological Hospital, 2-6-1 Musashidai,

Fuchu-shi, Tokyo 183-0042

nosis of Parkinson’s disease. Cryosections of the PCAshowed a scattering of a small number of muscle fibers andmassive inflammatory cell infiltration with proliferativesmall blood vessels in the interstitial tissues. Endomysialspace was remarkably edematous. No grouped atrophy ortarget fibers suggestive of neurogenic abnormalities was ob-served. No ulcer formation was found in any region of theesophagus.

Case 2A 77-year-old woman with a history of mild left

hemiparesis, which had been caused by cerebral infarction atthe age of 71, was admitted to our hospital because of com-plete right hemiparesis with severe consciousness distur-bance on April 17, 1996. Brain CT scan showed extensivelow density area over the whole region supplied by the leftmiddle cerebral artery. Since she developed apneic respira-tion of Cheyne-Stokes pattern, mechanical ventilationthrough an intratracheal tube was required for three days. OnApril 23, tube feeding through a NG tube was started. OnJune 3, three days after decannulation of the intratrachealtube, fiberoptic laryngoscopy clarified that vocal cord move-ment was normal. On July 7, mild inspiratory stridor

appeared, becoming loud in the following few days. On July10, fiberoptic laryngoscopy showed the presence of bilateralsevere VCAP with slit-like glottal space (Fig. 1). The arterialblood gas analysis at room air demonstrated acute respiratory

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Figure 1. Cryosections of the posterior cricoarytenoid muscle from Case 2 showed sparse muscle fibers (A, HEstain, ×33), associated with severe inflammatory cell infiltration and proliferative capillaries (B, HE stain, ×60).

Figure 2. Fiberoptic laryngoscopy from Case 2 demonstrated amarkedly narrow glottal space because of the abductor paraly-sis of bilateral vocal cords (VC). Arytenoid mucosa (A) wasmarkedly edematous.

ISOZAKI et al

failure; pH=7.35, pCO2 =53 Torr, pO2 =51 Torr. Subse-quently, however, her inspiratory stridor became decreasedgradually in spite of the lack of treatment. The only medicaltherapeutic for this period was exchange of the NG tube,which was regularly done every two weeks. On August 15when the inspiratory stridor almost disappeared, reexamina-tion of fiberoptic laryngoscopy showed normal abductionand adduction movements of the vocal cords. The arterialblood gas analysis at room air became almost normal;pH=7.420, pCO2 =42 Torr, pO2 =80 Torr. However, onSeptember 3 the inspiratory stridor appeared again withgradual exacerbation. Her consciousness level did notchange till her death. She was found in cardiorespiratory ar-rest by a rounding nurse on October 2, 1997.

Postmortem pathological examination revealed bilateralcerebral softening in the areas supplied by the middle cere-bral arteries with moderately severe atherosclerotic changesof the internal carotid arteries and the basilar artery.Cryosections of PCA showed sparse muscle fibers with mas-sive inflammatory cell infiltration with proliferative smallblood vessels (Fig. 2). Endomysial space was remarkablyedematous. No grouped atrophy or target fibers, suggestiveof neurogenic origin was observed. No ulcer formation wasfound in any region of the esophagus including postcricoidarea.

Discussion

Since Iglauer and Molt reported severe laryngeal injuryresulting from an indwelled duodenal tube in 1939 (1), simi-lar cases have been occasionally reported (2–9). Accordingto the recent review of the literature on this syndrome, it issuspected that the clinical spectrum of severity exists withless severe cases going unrecognized (3). The pathophysio-

logy of this critical illness is considered as follows: The an-terior wall of the upper esophagus is pinched between an in-dwelled NG tube and the posterior lamina of the cricoidcartilage, resulting in the development of the esophagealulcer. When a NG tube still remains at the same position, theNG tube-induced ulcer becomes further aggravated and thenperforates into the laryngeal abductor, PCA, which is locatedjust ventral to the upper esophagus. Consequently, PCA de-velops a severe myositis-like inflammatory reaction, result-ing in the development of VCAP due to the weakness of themuscle in abduction. The present four patients shown inTable 1 were diagnosed as having NG tube syndrome fromthe findings that VCAP associated with inspiratory stridordeveloped after the indwelling of a NG tube and that PCAshowed severe myositis-like changes, just like those ofSofferman et al’s cases (6). Compared to the previously re-ported cases of NG tube syndrome, however, our patientswere so peculiar in the following two points: absence of thepostcricoid esophageal ulcer and recurrent episodes ofVCAP. These features indicate that the mechanism of VCAPin our patients, namely a variant form of NG tube syndrome,is different from that in the previously reported NG tube syn-drome having postcricoid esophageal ulcer.

Myopathological findings of PCA seen in our patientshave a striking resemblance to those of the rat skeletal mus-cles after experimental ischemia or ischemia and subsequentvenous occlusion (10). The radiographical studies of thepostmortem laryngeal specimens with barium injection intothe blood vessels clarified that the venous plexus in the eso-phageal wall covered the dorsal surface of the cricoid carti-lage (11–13), as was confirmed by our pathological exami-nation of the autopsied larynx (Fig. 3).

We, therefore, hypothesized that the injuries of PCA inour patients were caused by circulatory failure due to the


Table 1. Clinical and Myopathological Findings of Patients with a Variant Form of NG Tube Syndrome

Patients Age(yr)

Sex Diseaseduration

(yr)

Number ofVCAP

episodes

Throatpain

Period1

Period2

Cause ofdeath

Postcricoidesophageal

ulcer

Histologyof PCA

Pathologicaldiagnosis

1

2

3

4

73

77

79

72

M

F

F

F

6

0.5

5

1.3

2

2

1

2

No

Unknown(consciousness

disturbance)No

Unknown(consciousness

disturbance)

2 y

2 m

2 w

3 m

1 m

1 m

1.6 m

1.5 m

respiratory failuredue to VCAP



respiratory failuredue to VCAP,

pneumonia, andrespiratory center

involvements

No

No

No

No

myositis-like

myositis-like

myositis-like

myositis-like

Parkinson’sdisease

Cerebralinfaction

Progressivesupranuclear

palsyCreutzfeldt-

Jakob disease

VCAP: vocal cord abductor paralysis, Period 1: Period from the first NG tube indwelling to the first VCAP episode, Period 2: Period fromthe last VCAP episode to death, PCA: posterior cricoarytenoid muscle.

compression of the PCA-perfusing veins and arteries by aNG tube. It indicates that postcricoid esophageal ulcer,which is a culprit in typical NG tube syndrome, is not in-volved in the development of VCAP in the variant form ofNG tube syndrome. Concurrently, some of the PCA-innervating nerve branches from the recurrent laryngealnerves may also be injured in the same manner, causing theneurogenic atrophy of PCA (7).

Another interesting finding in our patients is the “sponta-neous recurrence” of bilateral VCAP in spite of the lack oftreatment. A similar case with spontaneous recovery but notspontaneous recurrence was recently reported (9). We specu-lated that recurrence of VCAP was involved in the regularexchanges of a NG tube. Namely, a NG tube is usually ex-changed regularly (every two weeks in our hospital) for itsluminal smudge, while the possibility of the midline place-ment of a NG tube is only 6% (5) or 8% (14). Therefore,even if VCAP might happen after unfortunate midline posi-tion of a NG tube, it would be relieved after the next ex-change of the tube since the possibility of repeated midlineplacement of a NG tube is so rare. This seems to be the rea-son why VCAP can develop spontaneous recurrence in ourpatients and why NG tube syndrome has not been encoun-tered so often despite the popularity of patients receiving NGtube feeding. Considering that no esophageal ulceration wasprovoked, it is not strange that VCAP ameliorated withoutantibiotics and that throat pain, which is one of the triad ofdiagnostic criteria of NG tube syndrome, was lacking in thetwo patients without consciousness disturbance or dementia.Spontaneous and repeated remission and exacerbation ofVCAP is quite unique and different from VCAP observed inthe patients with neurodegenerative disorders where VCAPis, as a rule, progressive once it develops.

We speculated that NG tube syndrome is caused bymultifactorial mechanisms including neurogenic, vasculo-

genic, and myogenic processes (Fig. 4). They correspond tothe injury of the recurrent laryngeal nerve branches, ischemicand congestive myoinjury, and the postcricoid ulcer-inducedmyositis-like injury, respectively. More than two processesmay participate simultaneously in the development ofVCAP.

Since NG tube syndrome can develop in any patient underNG tube feeding, it is of importance to know the existence ofNG tube syndrome and to distinguish this treatable NG tube-induced VCAP from the neurodegenerative disorder-relatedVCAP (15–18). However, the diagnosis of a variant form ofNG tube syndrome described here was difficult, because nei-ther postcricoid esophageal ulcer nor throat pain, which arediagnostic clues in the typical NG tube syndrome, wasfound. At present, midline position of a NG tube on the neckplain X-ray film and recurrent episodes of VCAP, if present,may be some clues to the diagnosis of a variant NG tube syn-drome. In addition, a tentative decannulation under intrave-nous hyperalimentation for a few weeks may be useful as aprocedure of the therapeutic diagnosis. To examine themotor function of the vocal cords, laryngeal needle electro-myography and sleep load test may be useful. The former ex-amination may disclose neurogenic discharges (19) orprolonged bursts of tonic activity suggestive of laryngealdystonia (15). The latter may demonstrate sleep-induced ex-acerbation of VCAP, which is characteristic to VCAP inmultiple system atrophy (20). Of importance is to think ofthe possibility of NG tube syndrome when patients under NGtube feeding develop inspiratory stridor, whatever the under-lying disease is.

In conclusion, NG tube syndrome can appear in the pa-tients under NG-tube feeding even in those whom nopostcricoid esophageal ulcer is found. They may lack throatpain and repeatedly develop VCAP. We speculated that PCAin the patients with such a variant form of NG tube syndromewas injured by the circulatory failure of the PCA-perfusing

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Figure 3. There are a lot of small arteries (A), veins (V), andsecretary glands (G) in the narrow space between the esopha-geal epithelium (E) and the posterior cricoarytenoid muscle (M)in the normal subject (HE stain, ×20).

Figure 4. The pathomechanism of the injuries of the posteriorcricoarytenoid muscle is multifactorial: myogenic, vasculogenic,and neurogenic. They can occur simultaneously.

ISOZAKI et al

veins and arteries compressed by a NG tube.

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A Variant Form of Nasogastric Tube Syndrome