Superior mesenteric artery syndrome and acute pancreatitis in a boy with eating disorder: a case report

doi:10.4236/ojped.2011.14022

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Open Journal of Pediatrics, 2011, 1, 94-97 OJPed

doi:10.4236/ojped.2011.14022 Published Online December 2011 (http://www.SciRP.org/journal/ojped/)

Published Online December 2011 in SciRes. http://www.scirp.org/journal/OJPed

Superior mesenteric artery syndrome and acute pancreatitis in

a boy with eating disorder: a case report

Toru Wa tanabe 1, Motoi Fujita2, Yutaka Hirayama3, Yasushi Iinuma3

1Departments of Pediatrics, Niigata City General Hospital, Niigata, Japan;

2Department of Pediatrics, Niigata National Hospital, National Hospital Organization, Kashiwazaki, Japan;

3Pediatric Surgery, Niigata City General Hospital, Niigata, Japan.

Email: [email protected]

Received 2 October 2011; revised 12 November 2011; accepted 20 November 2011.

ABSTRACT

Eating disorders are an important cause of physical

and psychosocial disturbances. Gastrointestinal com-

plications in eating disorders are common and in-

clude pancreatitis and superior mesenteric artery

syndrome (SMAS). However, only two patients who

simultaneously suffered from both SMAS and pan-

creatitis in the setting of an eating disorder have ever

been reported in the literature. We describe here a

patient with eating disorder not otherwise specified

(ED-NOS) who exhibited both SMAS and pancreati-

tis concomitantly. An 11-year-old boy with ED-NOS

had presented with the sudden onset of recurrent

bilious vomiting, abdominal distension and abdomi-

nal pain. Laboratory and imaging studies revealed

that the patient had SMAS and acute pancreatitis.

Gastric decompression and intravenous infusion of

fluids promptly improved his abdominal symptoms

and laboratory data. This is the first case report of a

patient with ED-NOS who simultaneously suffered

from SMAS and pancreatitis in the pediatric litera-

ture. Since SMAS and pancreatitis can both cause

patients to be severely ill, these diseases should be

considered in patients with eating disorders who ex-

hibit abdominal symptoms such as vomiting or ab-

dominal pa in.

Keywords: Eating Disorder Not Otherwise Specified;

Superior Mesenteric Artery Syndrome; Pancreatitis;

Gastrointestinal Complicatio ns

1. INTRODUCTION

Eating disorders are an important cause of physical and

psychosocial disturbances, which primarily affect ado-

lescent females and can have life-threatening medical

consequences [1]. They are categorized into three disor-

ders including anorexia nervosa, bulimia nervosa and

eating disorder not otherwise specified (ED-NOS) in the

Diagnostic and Statistical Manual of Mental Disorders,

fourth edition (DSM-IV) [2]. ED-NOS includes patients

with clearly aberrant eating patterns and weight man-

agement habits who do not meet the criteria for anorexia

nervosa or bulimia nervosa [2].

The physical abnormalities in eating disorders are

secondary to the patients’ disturbed eating habits and

their compromised nutritional state, which may induce

endocrine, cardiovascular, hematological and gastroin-

testinal system dysfunctions as well as other metabolic

abnormalities [2].

Gastrointestinal complications in eating disorders are

common and include parotid enlargement with hypera-

mylasemia, dysphagia, gastric dilatation due to delayed

gastric emptying, constipation, pancreatic abnormalities

[1] and superior mesenteric artery syndrome (SMAS) [3].

However, only two patients who simultaneously suffered

from both SMAS and pancreatitis have ever been re-

ported in eating disorders [4,5]. We describe here a pa-

tient with ED-NOS who exhibited both SMAS and pan-

creatitis concomitantly, the first case report of a patient

with these diseases in the pediatric literature.

2. PATIENT REPORT

An 11-year-old boy was admitted to our hospital with a

one-day history of recurrent bilious vomiting, abdominal

distension and abdominal pain. The patient had been

diagnosed with ED-NOS 1 month before because of

3-month history of excessive exercising and restricted

intake without severe body image disturbance that led to

a 6 kg weight loss; in addition, the patient denied the

serious nature of his illness. He had a past medical his-

tory notable for Kawasaki syndrome complicated by

coronary artery aneurysms at 3 years of age, which was

treated with high-dose gamma globulin therapy and a

duodenal ulcer at 8 years of age. His older sister has also

suffered from anorexia nervosa and selective mutism

T. Watanabe et al. / Open Journal of Pediatrics 1 (2011) 94-97 95

since 10 years of age.

Physical examination on admission revealed extreme

emaciation with a weight of 27 kg (37% below normal

for age/height), height of 150 cm, body mass index

(BMI) of 12 kg/m 2, bradycardia (pulse rate was 40/bpm),

abdominal distention with hypoactive bowel sounds and

diffuse abdominal tenderness to p alpation without guard-

ing or rebound.

Laboratory studies disclosed metabolic alkalosis with

respiratory compensation (pH 7.455, HCO3 36.3 mmHg,

and PCO2 52.8 mmHg) , hypernatremia (Na 15 0 mEq/l),

elevated blood urea nitrogen (51.4 mg/dl) and serum

creatinine (1.10 mg/dl) levels, and hyperamylasenemia

(484 IU/l) with a predominant increase of pancreatic

isozymes. Serum lipase (608 U/l [normal 13 - 49 U/l]),

trypsin (900 ng/ml [normal 110 - 460 ng/ml]) and elas-

tase-1 (1400 ng/ml [no rmal 100 - 400 ng/dl]) levels wer e

also increased. Liver function tests, white blood cell

counts, blood glucose levels and concentrations of C-

reactive protein were within the normal ranges.

Because an abdominal X-ray demonstrated an extre-

mely dilated stomach, the patient underwent nasogastric

tube decompression with drainage of 500 ml of bilious

fluid, which improved his abdominal signs and symp-

toms promptly. A post-contrast abdominal computed

tomography after nasogastric tube insertion revealed loss

of intra-abdominal ad ipose tissue, reductio n of the aorto-

mesenteric distance (4 mm [normal 10 - 28 mm]) and the

aorto-mesenteric angle (13 [normal 38 - 65]) [6] without

any abnormal findings of the other organs including

pancreas (Figure 1). An upper gastrointestinal tract con-

trast-medium study demonstrated an abrupt vertical cut

off of the t hird portion of the du o den um (Figure 2).

The patient was diagnosed as having ED-NOS com-

plicated by SMAS and pancreatitis, and he underwent

Figure 1. Axial post-contrast computed tomography of the

abdomen demonstrating duodenal obstruction between the

superior mesenteric artery and the aorta (arrow).

Figure 2. Upper gastrointestinal tract contrast-medium study

showing an abrupt cut off the third portion of duodenum (ar-

row).

intravenous fluid infusions and enteral feedings by naso-

jejunal tube. His serum levels of amylase and lipase de-

creased into the normal range one week following the

initiation of treatment, although his weight and nutri-

tional status did not improved. The other abnormal

laboratory tests also normalized within two weeks.

Because the patient had exhibited psychiatric symp-

toms including irritability and profound anxiety, he was

transferred to the eating disorders unit. He underwent

psychiatric care in addition to the nutritional therapy,

which improved his condition gradually. The patient

became well with body weight of 37 Kg (14% below

normal for age/height) two months after being trans-

ferred to the unit and was discharged.

3. DISCUSSION

SMAS and pancreatitis are well-known gastrointestinal

complications of eating disorders [1]. However, only two

patients who concurrently suffered from both SMAS and

pancreatitis similarly to our patient have been repo rted in

eating disord ers.

Keane et al. presented a 16-year-old girl with anorexia

nervosa who developed acute gastric dilation and duo-

denal ileus due to SMAS and acute pancreatitis follow-

ing rapid refeeding [4]. Gwee et al. recently described a

17-year-old girl with anorexia nervosa who exhibited

acute abdominal symptoms following rapid refeeding,

and was diagnosed with SMAS and acute pancreatitis [5].

T. Watanabe et al. / Open Journal of Pediatrics 1 (2011) 94-97

Unlike these two cases, our patient with ED-NOS de-

veloped SMAS and pancreatitis simultaneously without

rapid refeeding.

SMAS involves the entrap ment and obstru ction of the

third part of the duodenum between the SMA and aorta

due to the narrowing of the angle between both vessels

[6]. The cause of this narrowing is related to loss of in-

tra-abdominal adipose tissue, which may result from

eating disorders [6]. Symptoms of SMAS can be acute or

chronic, with intermittent exacerbation [6]. Our patient

developed acute-onset SMAS. Although SMAS has

commonly been reported to present as a chronic condi-

tion in adulthood, one pediatric study reported that

SMAS was more likely to present acutely, with sudden

onset of symptoms related to bowel obstruction [7]. It

may be difficult to distinguish whether gastrointestinal

symptoms of patients with eating disorders result from

the eating disorders themselves or from chronic SMAS

associated with the eating disorders [3]. SMAS can pre-

cipitate and exacerbate anorexia nervosa because of

nausea associated with a small bowel obstruction and

conversely, anorexia nervosa prevents the patient from

being willing or able to ingest adequ ate calories to allow

the SMAS to resolve [8]. Moreover, some fatal cases of

SMAS in eating disorders have been reported due to

duodenal obstruction or gastric perforation [9]. There-

fore patients with eating disorders should undergo im-

aging studies to rule out SMAS, if they exhibit abdomi-

nal symptoms such as vomiting or abdominal pain.

Pancreatitis is a potentially life-threatening illness

with significant morbidity and mortality, and can occur

in eating disorders [5]. Although the exact mechanism

underling the development of pancreatitis in eating dis-

orders remains unclear, two mechanisms have been sug-

gested: pancreatic injury caused by chronic protein en-

ergy malnutrition and pancreatic regurgitation from in-

creased duodenal pressure resulting from SMAS [4,5].

Protein energy malnutrition causes acinar cell atrophy,

epithelial metaplasia and cystic d ilatation of the ducts in

the pancreas [10]. It is also associated with a depleted

antioxidant status and subsequent susceptibility to oxi-

dative stress and damage, and with an increase in in-

flammatory mediators such as TNF-alpha, IL-1 and IL-6,

which have been proposed to play a role in pancreatitis

[10]. Meanwhile SMAS can cause duodenal ileus and

dilatation, which leads to high pancreatic duct pressure

or reflux of duodenal contents into the pancreatic duct

[5]. This process leads to pancreatic cell damage th rough

autolysis thus causing the patients to develop pancrea-

titis [5]. Because abdominal symptoms and elevated

serum amylase levels of our patient improved rapidly

following only gastric decompression, an increased

duodenal pressure resulting from SMAS was the most

likely cause of the pancreatitis in our patient.

Our patient had a past medical history of Kawasaki

syndrome complicated with coronary aneurysm and a

family history of anorexia nervosa. Long-term disease

condition of Kawasaki syndrome affected on the psy-

chological condition of the patient with a family history

of ED and might develop ED-NOS in this patient.

In summary, we reported a patient with ED-NOS who

simultaneously suffered from SMAS and pancreatitis.

Since SMAS and pancreatitis can cause patients to be-

come severely ill, these diseases should be con sidered in

patients with eating disorders who exhibit abdominal

symptoms such as vomiting or abdominal pain.

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