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Case Report

A RARE CASE PRESENTATION OF PSYCHOGENIC DYSPHAGIA IN 7


YEAR OLD BOY : A CASE REPORT

By :
I Gede Kade Dwi Dharma Kayika

Supervisor :
I Gusti Ngurah Sanjaya Putra, MD, PAED, Consultant
I Putu Gede Karyana, MD, PAED, Consultant
Ida Ayu Kusuma Wardani, MD, Psychiatrist, Consultant
Ni Nyoman Metriani Nesa, MD, PAED

PAEDIATRIC DEPARTMENT
FACULTY OF MEDICINE UNIVERSITAS UDAYANA/ PROF.
DR. I.G.N.G NGOERAH HOSPITAL DENPASAR
2023
A Rare Case Presentation Of Psychogenic Dysphagia In 7 Year Old Boy : A
Case Report

I Gede Kade Dwi Dharma Kayika, I Gusti Ngurah Sanjaya Putra, I Putu Gede
Karyana, Ida Ayu Kusuma Wardani, Ni Nyoman Metriani Nesa

Paediatric Department
Faculty Of Medicine Universitas Udayana/ Prof. Dr. I.G.N.G Ngoerah Hospital
Denpasar, Bali

Abstract

Background : Incidence of dysphagia due to psychogenic dysphagia is quite


common in adult yet the incidence at 5-14 year old age is rare. The incidence are
1.3 per 100.000 case of dysphagia. The main problem of dysphagia is innability to
intake which led to dehydration and malnutrition. Various diagnostic test should
be performed to rule out anatomical and neurogenic cause before psychogenic
dysphagia diagnosis established.

Objective : To describe a rare case of dysphagia due to psychogenic dysphagia at


children.

Case : We report a case of 7 year old boy with difficulty to swallow since 12 days
before admission which got worsen in the last 5 days. Patient was suspected with
achalasia and reffered to Otolaryngologyst at RSUP Prof. Ngoerah. Laboratory
tests within normal limit. Head CT scan showed no abnormality. Patient consulted
to psychiatrician and diagnosed with observation of dysphagia due to psychogenic
dysphagia. After diagnosis psychogenic dysphagia was confirmed and treated, the
complain was relieved.
Conclusion : Psychogenic dysphagia should be kept in mind as a differential
diagnosis especially in children presented with difficulty to swallow and normal
diagnostic imaging.

Keywords : dysphagia, psychogenic, children


INTRODUCTION

Swallowing is a natural yet complex process that is taken for granted every
day. When structural anatomy or neurophysiology are affected, this process
becomes dysfunctional. Dysphagia is a feeling of abnormal food transit upon
swallowing. It typically occurs secondary to abnormal anatomy or physiology and
is classified as oropharyngeal or esophageal dysphagia based on its origin. 1,2
Diagnosis of dysphagia is reliant on a detailed history, which can often
delineate oropharyngeal from esophageal causes as well as anatomic from motor
causes. For instance, neurologic symptoms often are clues to oropharyngeal
dysphagia since it originates mainly from central nervous system and
neuromuscular disorders. In addition, dysphagia to solids and liquids typically
reflects an esophageal motility disorder, whereas progressive dysphagia to solids
reflects a structural disorder. Various diagnostic testing is subsequently available
to confirm the cause. 2,3 Videofluoroscopy, transnasal endoscopy, manometry and
fiberoptic endoscopic evaluation of swallowing are used in the diagnosis of
oropharyngeal dysphagia2,3. Real-Time Magnetic Resonance Imaging (MRI) is a
newer modality that may provide a more comprehensive evaluation of
swallowing, and therefore better illustrate where the oropharyngeal dysfunction
occurs.2,4 Esophagogastroduodenoscopy (EGD) and barium swallow are
traditionally used in the initial diagnosis of esophageal dysphagia, evaluating
mainly for structural lesions. In addition, barium swallow as well as esophageal
intraluminal impedance testing and radionuclide transit studies provide
information about esophageal transit and limited data regarding motility. 1,2
Esophageal manometry has been the gold standard for evaluation of motility
disorders. The recent development of high-resolution manometry has offered
potential for greater diagnostic yield.2
Psychogenic swallowing disorders whether associated or not associated
with eating disorders, is an interdisciplinary phenomenon. Rarely these patients go
directly to a psychiatrist. While a gastroenterologist might be the first physician
meeting a patient with eating disorder, an ENT specialist frequently meets patients
with psychogenic swallowing disorders. 3 Psychogenic dysphagia is as a sensation
of abnormal bolus transit through the esophageal body in the absence of
structural, mucosal, or motility disorders to explain symptoms, a form of
somatisation disorder. 2,3 The incidence of Psychogenic dysphagia predominantly
occurred at young adult to middle aged adult with 10.2 per 100.000 encountered
case of dysphagia while incidence in children is quite rare with 1.3 cases per
100.000 encountered case of dysphagia. 4
The objective of this case report was to describe a rare case dysphagia as
manifestation of Psychogenic dysphagia in 7 year old boy.

CASE REPORT
We report a case of 7 year old boy with dysphagia which turned out was
psychogenic dysphagia.
A 7 year old boy patient with chief complain difficulty to swallow come to
hospital. Difficulty to swallow occurred since 12 days before admission and
getting worse 5 days before admission. The parents said their children didn’t
want to swallow food because afraid to vomit at first. Later he didn’t want to
drink like usual because afraid to vomit. Patient also found slightly drooling
which worsen during hospitalization but later improved. Patient already lost 6
kilograms since 2 months before admitted. Patient also complained nausea and
epigastric pain before admitted. No vomiting nor diarrhea occurred before
admission. Patient was referred to hospital at otolaryngologyst Emergency
Department with achalasia suspicion. Patient was planned to esophagoscopy and
consulted to Pediatric Gastro-Hepatology division due to gastritis.
Patient’s physical examination was alert, heart rate and respiratory rate
within normal limit. Patient’s head and face examination were normal, sclera were
not icteric and conjunctiva were not pale. The ear, nose, and throat examinations
were normal and also double-checked by otolaryngologist. There were no lymph
nodes enlargement found on the neck. The chest were symmetrical both on rest
and movement, breath sound were bronchovesicular without rales or wheezing,
the first and second heart sound were normal, regular and no murmur in
auscultation. The abdomen were not distended, pain occurred when epigastric area
slightly pushed, liver and spleen were not palpable. The power, tonus, and reflex
of the superior and inferior extremities were normal.
Laboratory results at ER showed normal peripheral blood count, liver and
renal function test within normal limit, normal coagulation profile. Patient later
took several radiology imaging test. The Cervicothoracal imaging showed no
radioopaque corpus alienum and no compression nor fracture and listhesis was
shown (figure 1). Abdominal imaging showed no ileus nor pneumoperitoneum.
(Figure 2). Patient then planned to took oesophagography imaging to evaluate
stricture or narrowing of esophagus. Considering the patient inability to swallow
and intracranial proccess was suspected at the moment, the oesophagography
procedure postponed and head CT-scan with contrast performed. Head CT-scan
with and without contrast imaging showed there were no infarct, intracranial
hermorrhage nor Space Occupying Lession (SOL) intracerebral or intracerebellar
(figure 3). Esophagoscopy procedure later performed to confirmed anatomical
anomaly in this patient with flexible laryngoscopy performed before
Esophagoscopy. Flexible laryngoscopy shown normal vocal chord, normal
aritenoid, normal epiglotis and no saliva precipitate was found (Figure 4a) .
Esophagoscopy procedure shown no corpus alienum, stricture, mass and
inflammation along the esophagus, esophagoscope enter the esophagus withaout
any resistance (Figure 4b and 4c).

Figure 1. Cervicothoracal imaging: no radioopaque corpus alienum and no


compression nor fracture and listhesis
Figure 2. Abdominal imaging : no ileus nor pneumoperitoneum

Figure 3. Head CT imaging : no infarct, intracranial hermorrhage nor Space


Occupying Lession (SOL) intracerebral or intracerebellar

4a. 4b. 4c.


Figure 4a :Flexible laryngoscopy shown normal vocal chord, normal
aritenoid and epiglotis; 4b and 4c: Esophagoscopy procedure shown no corpus
alienum, stricture, mass and inflammation along the esophagus, esophagoscope
enter the esophagus withaout any resistance.
Patient later consulted to psychiatrycian due to normal finding from
physical examination and diagnostic imaging. The psychiatrist found the episode
of stress which turns to depression from history taking. The depression was
assessed using Beck Depression Inventory (BDI) and Child Depression Inventory
(CDI) with clinical depression and suspicious of depression as the result. This
depression accumulation later manifested to physical symptom which later worsen
due to accumulation of stress. Psychiatrician later diagnosed patient with
observation of dysphagia due to psychogenic dysphagia according to ROME IV
criteria. Initial management were omeprazole, formula milk intake through
feeding tube, aripiprazole and non-pharmacologic therapy (family
psychoeducation and supportive psycotherapy) .
Patient discharged at 12th days of hospitalization since clinical condition
improved and oral intake improved. Patient never been hospitalized since then and
come to psychiatric outpatient clinic.

DISCUSSION

Dysphagia is one of the most common condition which bring children to doctor or
hospital, however dysphagia due to psychogenic dysphagia in children is rare
condition with 1.3 per 100.000 case of dysphagia. 4
One report suggested that
persistent psychogenic dysphagia accounts for approximately 4% of referrals to
otolaryngologists with peak age of onset of 35–54 year and a lifetime prevalence
of 22%.5 Psychiatrists have reported that conversion disorder is not common in
children especially under 10 year old. Psychogenic dysphagia, dysphagia is
symptoms of conversion disoder but psychogenic dysphagia is very rare in
childhood. 7 Our case, a seven year old boy, came with chief complaint difficulty
to swallow. At the beginning patient said he was unable to swallow solid food but
later the symtomp got worse when the patient said he was unable to drink. After
patient’s consulted to psychiatrist, episodes of depression was found which
accumulated and triggered physical manifestation.
The precise nature of psychogenic dysphagia and its etiology remains
unclear. There is no uniform policy of management of this condition, but several
articles stated that the phenomenon is often associated with stress and anxiety,
possibly due to acute stress’ effect on upper esophageal sphincter
hyperresponsiveness.3 Symptoms of psychogenic dysphagia include aphonia, the
sensation of a lump in the throat, difficulty swallowing, the sensation of choking,
dyspnea, or suffocation.9 Pain has also been described.9 The symptoms must be
positively identified as psychologically related to some underlying mental conflict
or need. Patients are unable to easily discuss whether a conflict exists, because if
they could they would not likely have developed the physical symptoms.
Therefore, active investigation of possible stressors may reveal the unknown
anxiety issue. this specific form of conversion disorder. Etiologically, these
persons may have physiologic vulnerability or a “familiarity” with uncomfortable
sensations that do not better account for the development of psychogenic
dysphagia symptoms in light of known internal conflict. 5,6 In support of a biologic
contribution, some studies show a relationship of up to 70% between abnormal
esophageal acid exposure or distal esophageal immobility and the subsequent
development of psychogenic dysphagia.4,5 Our case, patient’s came to hospital
with chief complain inability to swallow since 12 days before admission with
clinical presentation at that time was slightly drooling which worsen during
hospitalization before receive psychiatric treatment (pharmacological and non-
pharmacological teratment). Patient also got supportive treatment during
hospitalization. The symptoms were improved and patient was discharged in good
condition.
Several examination should be performed before diagnosis patient with
psychogenic dysphagia been made, especially radiology imaging such as cervical
imaging, head CT scan, surface EMG oesophagram, modified barium swallow,
and esophageal manometry as gold standard to measure muscle motility when
dysphagia due to muscle motility was suspected. 4,6 The radioimaging diagnostic
goals is to exclude anatomical anomaly or SOL at brain parenchyme. Laboratory
examamination may performed when the patient in dehydration state due to
inability consume food and water, however no laboratory examination could not
established the diagnosis of psychogenic dysphagia.7,8 Our case, the laboratory
finding when the patient first admitted to ENT emergency triage shown normal
results. Radiology Imaging performed shown normal chest x-ray and normal head
CT scan. Patient planned to oesophagography but cancelled due to inability
patients to swallow contrast. Esophagoscopy procedure performed to confirmed
anatomical anomaly in this patient with flexible laryngoscopy performed before
Esophagoscopy. Flexible laryngoscopy shown normal vocal chord, normal
aritenoid, normal epiglotis and no saliva precipitate was found. Esophagoscopy
procedure shown no corpus alienum, stricture, mass and inflammation along the
esophagus, esophagoscope enter the esophagus withaout any resistance.
Treatment for psychogenic dysphagia itself predominantly is emergencies
and supportive therapy. Therefore once organic dysfunction has been ruled out,
the mainstay of treatment is reassurance and psychotherapy. 7,10 A trusting
relationship between the patient and the physician is essential. The clinician must
reassure the patient that the symptoms do not represent a serious underlying
disorder.7,10 Identification of psychological and emotional issues that are
associated with the symptoms lay the foundation for therapy, but little is known
about whether one mode is more likely to meet with success.9,10 Psychoanalysis,
insight-oriented therapy, and hypnotherapy have been described with little or
mixed support.10,11 Behavior modification may be effective in some cases,
particularly if the symptoms are reinforced by secondary gain. Family therapy was
successful in one case with an adolescent.10,11 Antidepressants have been tried but
have not been adequately studied. Unfortunately, no reports were found that
discussed the use of the newer antidepressants or antipsychotics. 9,11 Beside
antipsychotic drugs, in pediatric field supportive therapy should be given during
hospitalization periode such as maintenance fluid, modified food or parenteral
nutrition and supportive medication.9,11 In our case, Patient was referred to ER of
RSUP Prof Ngoerah fortunately without any life threatening condition such as
decreased of conciousness or dehydration. During hospitalization the patient
already given supportive therapy to relieve abdominal pain due to gastritis.
Patient also given intravenous fluid and consulted to Pediatric Nutritions and
Metabolic Division and was given total parenteral nutrition for 6 days. On the 7 th
day patients symptom was improved therefore oral intake was given.

SUMMARY
We reported a case of 7 year old boy with difficulty to swallow since 12 days
before admission which worsen in the last 5 days. Patient was suspected with
achalasia and reffered to Otolaryngologyst at Prof Ngoerah Hospital. Laboratory
examination within normal limit. Cervical imaging and Head CT scan showed no
anatomical abnormality. Patient consulted to psychiatrician and diagnosed with
observation of dysphagia due to Psychogenic dysphagia. Patient received
supportive therapy and total parenteral nutrition during hospitalization periode.
After diagnosis Psychogenic dysphagia was confirmed and treated, the symptom
improved and patient discharged after 12 days of hospitalization.
EVIDENCE BASED CRITICAL APPRAISAL CASE

CASE

7 year old boy with difficulty to swallow since 12 days before admission which
worsen in the last 5 days. Patient was suspected with achalasia and reffered to
Otolaryngologyst at Prof Ngoerah Hospital. Laboratory examination within
normal limit. Cervical imaging and Head CT scan showed no abnormality. Patient
consulted to psychiatrician and diagnosed with observation of dysphagia due to
Psychogenic dysphagia. Patient received supportive therapy, total parenteral
nutrition during hospitalization periode. After diagnosis Psychogenic dysphagia
was confirmed and treated, the symptom improved and patient discharged after 12
days of hospitalization.

PROBLEMS
1. In children with psychiatric problem are there greater risk of psychogenic
dysphagia according to ROME IV criteria occured?
2. How is the quality of life in children with psychogenic dysphagia?
PROBLEM 1

PECO
Based on the first problem , components of PICO can be described as follows:
Population/problem : Pediatric population
Exposure : psychiatric problem
Comparison : without psychiatric problem
Outcome : psychogenic dysphagia

CLINICAL QUESTION:
“Are children who have psychiatric problem compared to children without
psychiatric problem at increased risk for psychogenic dysphagia”

SEARCH STRATEGY:
Keywords : Children AND dysphagia AND diagnosis AND Rome IV

SEARCH RESULT
“Clinical Characteristics and Associated Psychosocial Dysfunction in Patients
With Functional Dysphagia: A Study Based on High-Resolution Impedance
Manometry and Rome IV Criteria” Clinical and Translational Gastroenterology
2022

JOURNAL SUMMARY
Background: Esophageal dysphagia is a common complaint encountered in the
daily clinical practice of physicians. Some patients may suffer from dysphagia as
a result of luminal obstruction. The pathophysiology of functional dysphagia is
complex and mostly unknown. It is challenging to obtain a definitive diagnosis
and adequate treatment of the condition quickly, leading to high health care
utilization and unnecessary financial burden. Despite the introduction of
functional dysphagia in the Rome IV criteria, related studies on the clinical
features of functional dysphagia remain scarce. In addition, we hypothesized that
psychosocial dysfunction and minor esophageal dysmotility might contribute to
the pathophysiology of functional dysphagia. Our secondary aim was to
investigate the role of psychiatric comorbidities and minor esophageal motility
abnormalities on the clinical manifestations of functional dysphagia.
Methods: Consecutive patients referred to our motility laboratory for evaluation
of esophageal dysphagia were identified. All patients were assessed with upper
endoscopy, high-resolution impedance manometry, and validated symptom
questionnaires. Data from those who were diagnosed with functional dysphagia (n
= 96) based on the Rome IV criteria were analyzed. Age- and sex-adjusted healthy
volunteers were also enrolled for comparison. Psychiatric comorbidity and poor
sleep quality were defined as total score of 5-item Brief Symptom Rating Scale ≥
6 and Pittsburgh Sleep Quality Index ≥ 6, respectively.
Result : The age peak of patients with functional dysphagia was at 40–60 year
(47.9%) with females predominant (67%). Forty-four patients (45.8%) had
psychiatric comorbidities, whereas 80 (83.3%) experienced poor sleep quality.
Female patients were more likely to have trouble falling asleep, shorter sleep
duration, and severe bloating. Compared with the healthy volunteers, patients with
functional dysphagia had higher 5-item Brief Symptom Rating Scale and
Pittsburgh Sleep Quality Index scores (5.34 ± 3.91 vs 1.84 ± 2.61, 9.64 ± 4.13 vs
4.77 ± 3.60, both P < 0.001) but similar results on high-resolution impedance
manometry. Those with ineffective esophageal motility (16.7%) had less sleep
efficiency than those with normal motility.
Conclusions: Patients with functional dysphagia were mainly middle-aged
women and had a high prevalence of psychiatric comorbidities and sleep
disturbances, especially in female patients. Patients with functional dysphagia
displayed similar esophageal motility as the healthy volunteers did.
CRITICAL APPRAISAL ON CAUSSATION
“Clinical Characteristics and Associated Psychosocial Dysfunction in
Patients With Functional Dysphagia: A Study Based on High-Resolution
Impedance Manometry and Rome IV Criteria”
Clinical and Translational Gastroenterology 2022

Is the evidence about causation valid?

Is the patient group clearly defined and similar No,


in all important aspects other than the
The patient group was clearly defined
exposure given?
according to the patient criteria and
was not similar, there was difference
in the characteristics of the subjects.

Were exposure and clinical outcome measured Yes, in both groups the same
in the same way in the two groups? measurements were taken.

Was the patient observation long enough (for Yes


the outcome to occur) and complete?
The observation took 6 year from
2014 until 2020

Do the results of this research on causal


aspects fulfill some of the criteria for
causation?

- In this journal we found that


- Is it clear that the exposure took place
people with psychogenyc
before the denouement?
dysphagia had psychiatric
problem but there were no
further data were dysphagia
occurred before psychiatric
problem happened
- Is there a relationship with increasing
the dose? - In this journal there were
explanation about medication
- Is there positive evidence from a de- and doses
challenge and re-challenge study?
- In this journal there were
explanation de-challenge and re-
challenge study
- Is this relationship consistent?

- Yes, this relationship is


consistent with another study
- Does the causal relationship found make
sense biologically?
- Though the exact
pathophysiollogy remain
unclear, several hypothesis
was made by scientist to
explain psychogenic dysphagia

B Is evidence of valid causation important?

1 What is the magnitude and precision In this journal didn’t gave explanation
(judging by the 95% CI value) of the about CI value, only P value was
relationship found between exposure and describe in results section
outcome?
C Can we apply this valid and important causal evidence to our patients?

1 Are our patients so different from those in Yes, the demography in this study is
the study that the results are not adult population meanwhile the
applicable? incidence of psychogenic dysphagia is
rare in children

2 What are our patient's trends and The study could explain the causation
expectations for the results of the study? effect of psychiatric problem with
dysphagia.

Conclusion: valid, important, and applicable


Level of Evidence IIb, Grade of recommendation B
PROBLEM 2

PICO
Based on the second problem , components of PICO can be described as follows:
Population/problem : Children with psychogenic dysphagia
Intervention : none
Comparison : none
Outcome : Quality of life

CLINICAL QUESTION:
“How is the quality of life in children with psychogenic dysphagia?

SEARCH STRATEGY:
Keywords : Children AND Psychogenic dysphagia AND Quality of life

SEARCH RESULT
“Phagophobia: a case report” The Turkish Journal of Pediatrics 2006; 48: 80-84

JOURNAL SUMMARY
Phagophobia is a form of psychogenic dysphagia. Although it is characterized by
a fear and avoidance of swallowing food, fluids, or pills, physical examination
and laboratory findings are normal. Here, we present a case of phagophobia, who
at 13 year of age was brought to our hospital by his family because of his fear and
avoidance of swallowing food and loss of weight. After psychiatric interview, the
patient underwent an oral peripheral examination, stroboscopic laryngeal
evaluation, the Bedside Swallow Evaluation, and the Modified Barium Swallow
Study. His physical examination and all laboratory findings were normal. The
management of this case included the combination of behavior therapy and a
dysphagia management program. After approximately one month of utilizing
these techniques, the case showed considerable improvement.
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