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Journal of Pediatric Surgery Case Reports 37 (2018) 16–18

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Journal of Pediatric Surgery Case Reports


journal homepage: www.elsevier.com/locate/epsc

A large hiatal hernia with cameron ulcer presenting as refractory sever iron T
deficiency anemia: A case report
Ayanaw Tamene∗, Masresha Mela
Bahir Dar University, College of Medicine and Health Sciences, Department of Pediatrics and Child Health, Ethiopia

A R T I C LE I N FO A B S T R A C T

Keywords: Background: Hiatal hernia is an abnormal protrusion of the stomach into thoracic cavity via esophageal hiatus.
Anemia The prevalence is reported as 0.8–5.2% in upper gastrointestinal endoscopy. Patients with large diaphragmatic
Cameron lesions hiatal hernia occasionally manifest as sever iron deficiency anemia. The etiology is chronic bleeding from a
Cameron ulcer gastric ulcer where a hiatal hernia passes through a diaphragmatic hiatus.
Hiatal hernia
Case presentation: A 4-year-old female patient presented with easily fatigability, tinnitus, light headedness,
epigastric abdominal pain, blacking of stool and geophagia of 1 year duration. She was transfused 03 times at
different time at different hospital for congestive heart failure secondary to sever iron deficiency anemia while
she was taking therapeutic iron therapy. The patient recovered completely after surgical repair of the hernia.
Conclusion: Cameron ulcer is a rare cause of iron deficiency anemia from chronic blood loss. It is fatal but easily
treatable ulcer at hiatal hernia. It very rare in pediatrics age particularly under five children and high index of
suspicion is lifesaving.

1. Introduction child health with easily fatigability of 1 year duration associated with
light headedness, tinnitus, geophagia, epigastric abdominal pain and
Hiatal hernia is mostly an asymptomatic incidental finding during intermittent blacking of stool. She had also history bilateral leg swel-
upper gastrointestinal endoscopy. This hernia is mostly associated with ling, shortness of breath and fast breathing which was relieved by
gastro esophageal reflux disease and Cameron ulcer. Cameron ulcer was transfusion of 20 ml/kg of whole blood 02 times at 2 different district
first described by Cameron and Higgins in 1986 [1]. Cameron lesions hospitals over 11 months period and referred to our hospital after 1
are a linear ulcers or erosions on the gastric mucosal folds at the level of month of last transfusion. She had a total of 7 visits of health institution
the diaphragm impressions from mechanical trauma secondary to dia- and was give iron for 1 year till arrival of emergency outpatient de-
phragmatic contraction from respiratory excursions in combination partment (EOPD). She had no history nasal bleeding, malaria attack,
with acid injury [1,2]. It causes mainly iron deficiency anemia due to fever or body rash. On our evaluation at EOPD, she was tachycardic and
chronic gastrointestinal bleeding and dyspepsia [2]. Other complica- tachypnic with sever palmar pallor, systolic ejection murmur, S3 gallop
tions are mucosal prolapse, incarceration, volvulus and esophageal and tender hepatomegaly despite her adherence for iron therapy.
shortening [3]. On laboratory evaluation the following result were found:
Cameron erosion and ulcers represent the mild and sever form of the WBC = 8600/mm3, Hgb = 4.7 g/gl, Hct = 18.2%(8.1% drop within a
same disease spectrum, respectively [2]. The exact prevalence of ero- month while on iron therapy), Plt = 529,000/mm3, MCV = 68.3 fl,
sions and ulcers is not known. The prevalence rates of hiatal hernia RDWcv = 18.1%. Occult blood test was positive. Peripheral mor-
ranges from 0.8 to 5.2 in all patients undergoing upper gastrointestinal phology showed hypochromic normocytic red blood cells. Helicobacter
endoscopy [3]. There are no case reports of Cameron lesions from pylori stool antigen, stool examination, bone marrow aspiration, PT,
African continent. PTT, INR, serum albumin and organ function test revealed normal re-
sult. Chest x-ray (Fig. 1) and chest ultrasound suggest diaphragmatic
2. Case Presentation hernia. Barium meal study (Fig. 2) was conclusive for hiatal hernia.
Congestive heart failure secondary to sever anemia was corrected with
A 4 years old female child presented to department of pediatrics and transfusion. Proton pump inhibitor with anti-acid syrup was given for


Corresponding author.
E-mail address: zeelias04@gmail.com (A. Tamene).

https://doi.org/10.1016/j.epsc.2018.07.008
Received 23 June 2018; Received in revised form 9 July 2018; Accepted 9 July 2018
Available online 12 July 2018
2213-5766/ © 2018 Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license
(http://creativecommons.org/licenses/BY-NC-ND/4.0/).
A. Tamene, M. Mela Journal of Pediatric Surgery Case Reports 37 (2018) 16–18

3. Discussion

Hiatal hernia is an abnormal protrusion of stomach with or without


another intra-abdominal organ through esophageal hiatus into the
thoracic cavity [1,3]. The prevalence rates of hiatal hernia ranges from
0.8 to 2.9 in all patients undergoing upper gastrointestinal endoscopy
[3]. It is classified into type I and type II [4]. Type I (sliding type) is the
most commonly observed type associated with sliding of gastro-
esophageal junction with the stomach [5]. The incidence of hiatal
hernia increases by advancing age due to relaxation at the diaphrag-
matic crura and esophageal shortening induced by short-term in-
traluminal acid perfusion in opossum resulting from aging process
Fig. 1. Air fluid level over right lower chest and mediastinum on poster- [6–8]. Hiatal hernia is commonly incidental asymptomatic find but may
ioanterior chest x ray.
be associated with gastro esophageal reflux disease and Cameron ulcer.
Cameron lesions are a linear ulcers or erosions on the gastric mu-
cosal folds at the level of the diaphragm impressions from mechanical
trauma secondary to diaphragmatic contraction from respiratory ex-
cursions in combination with acid injury and ischemia [1,2,9]. Ca-
meron erosion and ulcers represent the mild and sever form of the same
disease spectrum, respectively [2]. Cameron ulcer was first described
by Cameron and Higgins in 1986 [1]. The prevalence of Cameron ulcer
is dependent on the size of hiatal hernia and increase as the size of
hernia increases [10]. Cameron ulcers are seen in 5.2% of patients with
hiatal hernia who undergo endoscopic gastroduodenoscopy [3]. There
are no case reports of Cameron lesions from African continent accessed
by the author likely not reporting or undiagnosed cases rather than
epidemiological absence of this disease as there is no known genetic or
environmental contributing factor.
It can be single, multiple, small or large. It causes mainly iron de-
ficiency anemia due to chronic gastrointestinal bleeding and dyspepsia
Fig. 2. Posteroanterior barrium meal study shows air fluid level at mediastinu
[2]. Other complications are mucosal prolapse, incarceration, volvulus
on right side and contrast agent seen above the diaphram.
and esophageal shortening [3]. Acute and chronic gastrointestinal
bleeding from lineal ulcer or erosion is the commonest manifestation of
Cameron lesion casing iron deficiency anemia [11,12]. Chronic gastritis
associated with hiatal hernia also contributed to the development of
anemia. Even though hiatal hernia is very rare in pediatrics age, there
are few case reports of Cameron ulcer as early as 5 [13] years 7 [14]
years old children. Our patient 4 years old patient whose symptoms
started at the age of 3 years was the youngest age case report accessed
by the author.
Clinical diagnosis of hiatal hernia and Cameron lesion is often dif-
ficult since they often present with gastroesophageal reflux disease and
anemia [15]. Particularly in developing countries in a place where
nutritional deficiency iron deficiency anemia, malaria, intestinal para-
site and whole cows are commonest cause of anemia in young age, high
index of suspicion has a pivotal role for the diagnosis of Cameron lesion
in refractory iron deficiency anemia with gastrointestinal bleeding.
Chest x ray and barium meal study can be used for first screening of
hiatal hernia. It shows air fluid level in the mediastinum. Upper gas-
Fig. 3. Lateral barrium meal study shows air fluid level at mediastinu on right trointestinal endoscopy is the gold standard of diagnostic modality but
side and contrast agent seen above the diaphram. it may miss the lesion and re look may be needed [16]. CT scan and MRI
also may be diagnostic.
The treatment is mainly medical with acid suppressants and anti-
01 month prior to surgery. Intra-operatively there was 4 cm by 5 cm
ulcers for the ulcer and iron for anemia. If it is refractory, surgery is the
hiatal defect in which stomach herniated without volvulus and the
definitive treatment [9]. No reports of recurrence of ulcer or hemor-
defect repaired by interrupted stich. No mesh applied and fundoplica-
rhage after surgical management on long term follow up [17].
tion was not done. Hematocrit became 30% after transfusion and
In our case she sign and symptoms of anemia for 01 year while she
reached 40% after 01 month of surgery and anti-ulcer treatment. Anti-
was on adequate dose of therapeutic iron. She was transfused whole
ulcer drugs were discontinued after a total 02 months treatment fol-
blood 03 times for severe anemia secondary to congestive heart failure.
lowing resolution of epigastric pain, negative occult blood test and
Additionally, she was also complaining abdominal pain and blacking of
normalization of hematocrit. Endoscopy was not done to check healing
stool. We reach to the diagnosis of a large hiatal hernia with Cameron
of ulcer (no pediatric endoscopy). Chest x ray done after 02 month of
ulcer presenting as refractory iron deficiency anemia made by chest x
surgical management showed normal. She had uneventful course after
ray(gave the first clue for hernia), positive occult blood test and barium
management and discharged from follow up after 6 month of surgical
meal study.
management (see Fig. 3).
Intra-operatively one-third of the stomach herniated to the thoracic
cavity through 4 cm by 5 cm hiatal defect. After surgical correction,

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A. Tamene, M. Mela Journal of Pediatric Surgery Case Reports 37 (2018) 16–18

omeprazole and ant-acid syrup she recovered fully. In areas where Wubeshet Esayas who commented the chest x ray and barium study, Mr
pediatric sized endoscopy probe is not available, barium meal study, CT Alemnew who did the barium meal study and all those involved in the
scan, MRI and adequate response to acid suppressant drugs may be management of the patient.
diagnostic for severe anemia from Cameron ulcer.
Appendix A. Supplementary data
4. Conclusion
Supplementary data related to this article can be found at https://
In conclusion, patients developing symptoms and signs of anemia doi.org/10.1016/j.epsc.2018.07.008.
with dyspeptic symptoms and blackening of stool with persistent iron
deficiency anemia requiring frequent transfusion and uncorrected by References
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operated the patient and repaired the diaphragmatic defect, Dr

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