Journal of Pediatric Surgery Case Reports 59 (2020) 101515
Available online 7 June 2020
2213-5766/© 2020 The Author(s). Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license
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Journal of Pediatric Surgery Case Reports
Whipple surgery for eosinophilic gastroenteritis in a child
Farzaneh Motamed
a, Shokoufeh Ahmadipour
b , c , *a Pediatrics Gastroenterology and Hepatology Research Center, Tehran University of Medical Sciences, Tehran, Iran
b Department of Pediatrics, Faculty of Medicine, Lorestan University of Medical Sciences, Khorramabad, Iran
c Hepatitis Research Center, Lorestan University of Medical Sciences, Khorramabad, Iran
A R T I C L E I N F O Keywords :
Eosinophilia Gastrointestinal Adhesion Surgery
A B S T R A C T
Herein, we pre sent an un usual case of the in volve ment of pan creas and duo de num in eosinophilic gas troen - teri tis (EoG), re vealed in esoph a gogas tro duo denoscopy (EGD). Ow ing to pa tients’ con di tion and find ings from en doscopy, Whip ple surgery was per formed to re move the part of pan creas and duo de num. Long - term ther a - peu tic ef fi cacy was achieved through this in ter ven tion.
1 . Introduction
Eosinophilic gas troen teri tis (EoG) is a rare di ges tive tract dis ease char ac ter ized by patho logic in fil tra tion of eosinophils in the gas troin - testi nal tract [ 10 ]. EoG af fects pa tients of all ages but is more preva - lent in chil dren and adults aged 20 – 25 years. Dis tri b u tion of the dis or - der along sex lines places males at 1.4 times higher risk than fe males [ 5 , 9 ]. Clin i cal man i fes ta tion is de pen dent on the lo ca tion of EoG in gas troin testi nal tract and depth of the in testi nal wall af fected; on the ba sis of which EoG has been clas si fied into 3 types: mu cosal, mus cu lar and sub serosal [ 12 ]. Mu cosal is re port edly the most com mon type with ap prox i mately 57.7% of cases in lit er a ture whereas, mus cu lar and sub serosal are re spec tively marked by 30% and 12.5% of the to tal cases. Symp toms of mu cosal EoG in clude vom it ing, di ar rhea, GI bleed ing as so ci ated with iron de fi ciency ane mia, pro tein loss and mal - ab sorp tion. The mus cu lar type of ten causes ab dom i nal pain, vom it ing par tial or to tal bowel ob struc tion eosinophilic in fil tra tion but with out as cites whereas, the sub serosal type of the dis ease in volves eosinophilic in fil tra tion in the gut and eosinophilic as cites while pre - sent ing as cites, eosinophilia and pos si bly peri toni tis as the com mon symp toms [ 3 , 14 , 20 ]. In rare cases, EoG may cause stric ture, ul cer and steno sis in gas troin testi nal tract [ 11 ]. Case stud ies have shown that eosinophilic in fil tra tion can also be pre sented in stom ach, small in tes - tine and pan creases [ 17 ]. Whip ple pro ce dure (pan cre ati co duo denec - tomy) is the most rec om mended sur gi cal pro ce dure where, head of the pan creas is re moved with the part of duo de num, fol lowed by anas to mo sis of the ream ing part of the pan creas to small in tes tine and at tach ment of small in tes tine to the stom ach. In pe di atric pa tients, it
is a safe pro ce dure and no mor tal ity has been re ported till the date.
Rarely, hy per glycemia or ke toaci do sis might be seen, how ever, it is man age able [ 15 , 19 ]. Long - term side ef fects of this pro ce dure in clude;
di ar rhea, ab dom i nal pain, pan cre atic en zyme sup ple ments and con sti - pa tion. How ever, these com pli ca tions vary ac cord ing to the type of the pathol ogy [ 7 ].
2 . Case presentation
A 5 - year - old boy was ad mit ted to our health care fa cil ity re port ing ab dom i nal pain and vom it ing for 5 months from his re fer ral. He did not pre sent fever, rash or loss of weight. Dur ing esoph a gogas tro duo - denoscopy (EGD) a large tu mor (2 * 2 cm) was ob served in the duo de - nal bulb and the pathol o gist re ported a se vere in fil tra tion of eosinophil (80 – 100/HPF) ( Fig. 1 ). Ad di tion ally, pa tient was neg a tive for any type of par a sitic dis ease (by stool ex am i na tion for par a site and ova) and sys temic in volve ment for the in fil tra tion of eosinophil (un in - volve ment of esoph a gus, eosinophils less than 1500/ HPF, rul ing out hy per e o sionophilic syn drome).
The pa tient was placed on a hy poal ler genic diet and was ad min is - tered pan to pra zole (1mg/ kg/ BID/ dose) and cro molyn sodium. How - ever, he failed to re spond within 2 months, and the pain was in ten si - fied. An other en doscopy was per formed where se vere esophagi tis ac - com pa nied with ul cer in the pre - pyloric area and patchy ery the ma - tous mu cosal le sions in duo de nal bulb were ob served with out any ma - lig nan cies. In the patho log i cal ex am i na tion of esoph a gus and bulb, more than 80 eosinophil/ HPF was de tected where, sheets of eosinophils were seen over lam ina pro pria, mus cu laris mu cosa and
* Corresponding author. Associate Professor of Pediatrics, Lorestan University of Medical Sciences, Khorramabad, Iran.
E - mail address: shokoufeh. ahmadipour@ gmail. com , sh. ahmadipour95@ gmail. com (S. Ahmadipour).
https://doi.org/10.1016/j.epsc.2020.101515
Received 12 May 2020; Received in revised form 28 May 2020; Accepted 1 June 2020
Journal of Pediatric Surgery Case Reports 59 (2020) 101515
2 F. Motamed and S. Ahmadipour
Fig. 1 . Bulb tu moral le sion.
sub mu cosa along with ex cess eosinophils in in traep ithe lial layer ( Fig.
2 ). Fe cal pro tein of the pa tient was seen to be 64mg/ dL with the el e - va tion of al pha1 - antitrypsin. Pred nisone was pre scribed for the pa - tient (1 – 2mg/kg/day). Five months later, he was pre sented with se - vere ab dom i nal pain, tremors, vom it ing thereby, show ing the signs of in testi nal ob struc tion. Ab dom i nal CT re vealed se vere ob struc tion and ad he sion in the ar eas of duo de num, pan creas and lym phatic sys tem there fore, pan cre ati co duo denec tomy (Whip ple pro ce dure) was per - formed. Three - months af ter the surgery, the symp toms were com - pletely re solved, and pred nisone was dis con tin ued, grad u ally. Fol low - up EGD and the pathol ogy was com pletely nor mal. The eosinophilia in the pe riph eral blood at tained nor mal level. It has been 8 years since the sur gi cal in ter ven tion that he is com pletely healthy with out any di ges tive com plaints ( Table 1 ).
3 . Discussion
Ex act cause of EoG is not clearly known but it has been at trib uted to the hy per sen si tiv ity to food al ler gen, and in some re ports, it has been linked to atopies such as asthma, hay fever or eczema. Man age - ment of EoG of ten in cludes di etary elim i na tion of the al ler gen con - tain ing food items fol lowed by ther a peu tic reg i men. Cor ti cos teroids have been known with ef fec tive ther a peu tic out comes in non - obstructive cases [ 1 , 13 , 18 ]. In this case, di etary elim i na tion failed due to the de vel op ment of the ob struc tion hence sur gi cal pro ce dure was un der taken. Over all gen eral prog no sis af ter the surgery is good with
Fig. 2 . Medium power view of a gas tric mu cosal biopsy of a pa tient with eosinophilic gas troen teri tis.
Table 1
Sum ma rizes CBC of the pa tient at the time of ad mis sion fol low ing di ag no sis of for eosinophilic gas troen teri tis and af ter whip ple surgery.
Intervals of
lab findings Normal values Patient's values
At the time of admission
Hb: 11.5 – 13.5 g/dLRBC: 3.9 – 5.3 (x10 12 /l) Hct: 0.34 – 0.40Platelets:
150 – 400 (x10 9 /l) WBC: 5.0 – 17.0 (x10 9 /l) Neutrophils: 1.5 – 8.5 (x10 9 /l) Lymphocytes: 1.5 – 9.5 (x10 9 /l) Monocytes: 0.2 – 1.2 (x10 9 /l) Basophils: <0.11 (x10 9 /l) Eosinophils: <0.81 (x10 9 /l)
Hb: 11 g/dLRBC: 4.3 (x10 12 /l) Hct: 37%Platelets:
250 (x10 9 /l) WBC: 15 (x10 9 /l) Neutrophils: 5 (x10 9 /l) Lymphocytes: 3 (x10 9 /l) Monocytes: 0.8 (x10 9 /l) Basophils: <0.11 (x10 9 /l) Eosinophils: 3.6 (x10 9 /l)
After
surgery Eosinophils: 0.1 (x109/l)
com plete re cov ery es pe cially in chil dren thus al low ing to re store nor - mal di etary in take with out any re stric tions. The gas tric antrum and small in tes tine are the mostly com monly af fected by ob struc tion whereas, ul cer a tion and he m or rhage are not re ported of ten. Stud ies have shown that pathol ogy of EoG in clude ob struc tion of bil iary duct and acute pan cre ati tis [ 8 ]. To the date, very few re ports have men - tioned the in volve ment of the head of pan creas. In a geri atric pa tient, eosinophilic gas troen teri tis was re ported to man i fest as cites, mu cosal le sions and acute pan cre ati tis. How ever, phar ma co log i cal ther apy was suc cess ful to treat the pa tient in this case [ 2 ]. In this case, drug - based treat ment failed and with time, sever ity in the symp toms were seen.
Whip ple surgery was per formed in this case where the head of the pan creas and part of duo de num was re moved ow ing to the ul cer a tion.
Re cent analy sis con cluded that pan cre ati co duo denec tomy is safe and ef fec tive in pe di atric pa tients with min i mal com pli ca tions [ 16 ].
Nonethe less, long - term fol low - up with the pa tient can il lus trate ef fec - tive ness of this pro ce dure for EoG in chil dren.
4 . Conclusion
Eosinophilic gas troen teri tis is a rare di ges tive dis ease in chil dren that is char ac ter ized by the in fil tra tion of eosinophils in any part of GI tract and eosinophilia in blood [ 4 , 6 ]. Clin i cal pre sen ta tion may vary from mild to se vere, but in ex tremely rare cases, it causes ex ten sive ad he sion in the di ges tive sys tem that re quires surgery, such as Whip - ple pro ce dure in this case.
Funding source
No fund ing was se cured for this study.
Contributors’ statement page
Dr. Shok oufeh Ah madipour: con cep tu al ized and de signed the study, drafted the ini tial man u script, and re viewed and re vised the man u script.
Dr. Farzaneh Mo tamed: de signed the data col lec tion in stru ments, col lected data, car ried out the ini tial analy ses, and re viewed and re - vised the man u script.
Co or di nated and su per vised data col lec tion, and crit i cally re - viewed the man u script for im por tant in tel lec tual con tent.
All au thors ap proved the fi nal man u script as sub mit ted and agree to be ac count able for all as pects of the work.
Declaration of competing interest
The other au thors have no con flicts of in ter est to dis close.
Journal of Pediatric Surgery Case Reports 59 (2020) 101515
3 F. Motamed and S. Ahmadipour
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