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Chilaiditi Syndrome: A Case of Recurrent Respiratory Distress be
Multiply Misdiagnosed
Gao Yong-shun, Zhang Yun-fei, Feng Hai-lin, Zhang Yu-kun
Keywords
Chilaiditi syndrome, Respiratory distress, Diagnostic errors
A 61-year-old woman was admitted to the First Affiliated Hospital
Vasculocardiology Department of Zhengzhou University with recurrent
respiratory distress. Her symptoms generally worsen at night when the
patient lies supine, and which was partly alleviated when the patient had
a seat or stand a few minutes, without fever, cough, phlegm, chest pain
and hemoptysis, which had persisted during the preceding 2 weeks. 2 weeks
ago, the patient was admitted to the local hospital and diagnosed as
“1.Coronary Disease, 2.Chronic Hepatitis B, 3.Stage Ⅱ Hypertension”
and was administered with conservative treatment. However, her symptoms
were not significantly alleviated. The patient was then referred to our
hospital in September 2012 for further evaluation and treatment. Her past
medical history included hypertension two years earlier, and her regular
medications included compound kendir lenves (one tablets daily) and
reserpine (one tablets daily). Her family history was negative.
The patient’s height 163cm, weight 78kg, BMI 29.35. On physical
examination, the patient had normal vital signs and oxygen saturation of
Yong-Shun Gao, Yun-Fei Zhang, Hai-Lin Feng, Yu-Kun Zhang. Department of gastrointestinal
surgery, The First Affiliated Hospital of Zhengzhou University, Zhengzhou 450052, China.
Correspondence to: Dr. Gao Yong-Shun，Department of gastrointestinal surgery, The First
Affiliated
Hospital
of
Zhengzhou
China.(E-mail:[email protected],13803899721).
University,
Zhengzhou
450052,
95％-100％on 2 litres of oxygen and seat. Body position was orthopnea.
The breath sounds were decreased at the right base compared with the left.
There was no other physical finding. Contagion test showed HBsAg(+),
HBeAg(+), Anti-HBc(+), WBC:12.5×109 /L. Biochemistry measurements were
found to be normal. The patient was conservatively treated with expanding
coronary artery and act blood therapy according to the diagnosis as
“Coronary Disease”, but her symptom wasn’t significantly alleviated.
ECG and Heart Doppler ultrasound were found to be normal, heart disease
was ruled out the possibility. Then the patient went to the Respiratory
Medicine Department, and was conservatively treated with antibiotic
therapy according to the diagnosis as “pulmonary inflammatory”, because
of her WBC:12.5×109 /L and the breath sounds were decreased at the right
base, but her symptom still existed. The CT scan revealed the air below
the right diaphragm accidentally, with the aim of it was to observe the
condition of pulmonary inflammatory (Fig. 1). Afterwards the patient went
to the Department of Gastrointestinal Surgery. The barium enema
examination revealed the hepatodiaphragmatic interposition of the colon
(Fig. 2), the diagnosis of “Chilaiditi syndrome” was made. The patient
underwent reduction of interposed colon and greater omentum companion
with conventional open surgery on October 10, 2012. Intra-operation
finding included liver volume reduced, surface distribution of multiple
nodules varies in size, and its substantial touch was a little hard. The
hepatic flexure of colon and the great mass of greater omentum were in
between the liver and the right hemi diaphragm. Ascending colon fixed good,
and there was no redundant colon with long mesentery. After the operation,
the patient reported complete resolution of her symptoms, and X-ray showed
the colon was fixed in right location on the 4 day after surgery. The
patient was discharged to home on October 22, 2012. She remained symptom
free at 8 months follow up.
Respiratory
distress
was
frequently
found
in
diseases
of
cardiovascular and respiratory system, such as bronchial asthma,
interstitial pneumonia, cardiac tamponade and cardiac failure resulting
from different causes. This patient’s symptoms presented similar to
paroxysmal nocturnal dyspnea caused by acute left heart failure. The
etiology of Chilaiditi syndrome is very complex. Any condition leading
to an enlarged right subdiaphragmatic space, hypermobility of the colon
or higher intra-abdominal pressure can predispose patients to Chilaiditi
syndrome [4-4]. In the case reported here, the patient had an enlarged right
subdiaphragmatic space due to cirrhosis, and a higher intra-abdominal
pressure due to obesity and omentum fatty disposition, which were likely
the etiology of Chilaiditi syndrome.
Most patients with Chilaiditi syndrome are asymptomatic, be
diagnosed with an incidented radiologic finding. The others patients
may present with a wide range of complaints which can be misleading to
the clinician. Symptoms associated with Chilaiditi syndrome always
occur
as
gastrointestinal
discomfort,
cluding
abdominal
pain,
distention, bloating, nausea, vomiting, flatulence, and changes in
intestinal habits as well as more unusual chest manifestations, such
as substernal pain, cardiac arrhythmias, dyspnea, and respiratory
distress. Moreover, chest symptoms are always accompanies with the
emergence of gastrointestinal symptoms and appear. Reviewed of the
literature, the respiratory distress caused by Chilaitidi syndrome
always occurred in children or the elderly [1-4], and accompanies with the
abdominal pain, distention, nausea, and hiccup. To our knowledge, this
is the first case which only is associated with recurrent respiratory
distress without gastrointestinal symptoms. Therefore, physicians
often ignore the possibility of Chilaiditi syndrome. When the patient
underwent CT scan to observe the condition of pulmonary inflammatory,
the CT showed an incidental radiologic finding of the air below the right
diaphragm. After that, the patient had a definite diagnose.
The diagnosis of Chilaiditi syndrome need to be combined with
radiographic findings. The air below the right diaphragm on X-ray or
CT scan appears mostly in Chilaiditi syndrome, ture pneumoperitoneum
and subphrenic abscess. The presence of visible haustral folds adjacent
to the diaphragm, the Chilaiditi syndrome’s characteristic finding,
makes recognition of this phenomenon critical in distinguishing it from
subphrenic abscess and true pneumoperitoneum. If an X-ray or CT scan
doesn’t show visible haustral folds adjacent to the diaphragm, a barium
enema examination is recommended to establish an accurate diagnosis. Due
to the interposed colonic loop moved down obviously, result of the erect
chest X-ray was negative, but the horizontal position CT scan showed
the air below the right diaphragm. The diagnostic value of ultrasound
in Chilaiditi syndrome is limited[5].
After the patient sitting a few minutes, her symptoms of respiratory
distress still cannot relieve completely. Considering the causes as
though the colon pressure was lifted, but Pulmonary reexpansion and
Pulmonary ventilation flow ratio still needs some time to return to
normal.
The treatment of Chilaiditi syndrome is generally conservative.
Surgical treatment may be required in case of intestinal obstruction,
ischemia, or perforation. Because of the respiratory distress wasn’t
significantly alleviated after conservative treatment, this patient
required for surgical treatment. After the operation, the patient
reported complete resolution of her symptoms, and X-ray showed the colon
was fixed in right location.
This syndrome is a rare disorder with recurrent respiratory distress
and we conclude that this entity should be kept in mind in patients with
recurrent respiratory distress.
References：
1.
Keles S, Artac H, Reisli I, et al. Chilaiditi syndrome as a
cause of respiratory distress. Eur J Pediatr, 2006;165:
367–369 PMID:16489467
2.
Dogu F, Reisli I, Ikinciogullari A, et al. Unusual cause of
respiratory distress: Chilaiditi syndrome. Pediatr Int, 2004;
46:188–190 PMID:23530798
3.
Amy X, Gavin H, Gwendolyn M, et al. Chilaiditi Syndrome
Precipitated by Colonoscopy: A Case Report and Review of the
Literature. HAWAI‘I Journal Of Medicine & Public Health, 2012,
71(6): 158-162 PMID:22787564
4.
Sheikh Z, Khan A, Khan S. Chilaiditi’s syndrome: colonic
interposition in a young patient with abdominal pain. Postgrad
Med J, 2011;87: 239 PMID:21257995
5.
Moaven O, Richard A. Chilaiditi Syndrome: A Rare Entity with
Important
Differential
Diagnoses.
Gastroenterology
Hepatology, 2012,8(4):276-278 PMID:22723763
&
Fig 1 The CT scan revealed the air below the right diaphragm.
A
B
Fig 2A The barium enema examination revealed the hepatodiaphragmatic interposition
of the colon; 2B The X-ray showed the colon was fixed in right location on the 4
day after surgery.