Unusual Presentation of Pancreatic Insulinoma:a Case Report

ISSN: 2320-5407 Int. J. Adv. Res.
10(02), 974-978
Journal Homepage: -www.journalijar.com
Article DOI:10.21474/IJAR01/14301
DOI URL: http://dx.doi.org/10.21474/IJAR01/14301
RESEARCH ARTICLE
UNUSUAL PRESENTATION OF PANCREATIC INSULINOMA:A CASE REPORT
Zineb El Azime, Fatimazahra Melki, Houda Salhi and Hanane ElOuahabi

Department of Endocrinology, Diabetology and Nutrition - Hassan II University Hospital.Fez, Morocco
Epidemiology and Health Sciences Research Laboratory.
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Manuscript Info Abstract
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Manuscript History Insulinomas are pancreatic neuroendocrine tumors (PNETs)
Received: 18 December 2021 responsible for inappropriate insulin release, leading to episodes of
Final Accepted: 20 January 2022 hypoglycemia. The diagnosis of insulinoma is suspected clinicaly
Published: February 2022 andbiochemically made with low blood glucose, elevated
insulin, proinsulin, and C-peptide levels, and confirmed by localizing
Key words:-
Insulinoma, Endoscopic,Hypoglycemia, the tumor ,usually, by non-invasive imaging techniques and for
Enucleation, Peptide C, Neuro- difficult cases invasive modalities could be needed.Herein, we present
Psychiatric Disorder the case of a 15 years old female who had a 1 year historyof episodic
and repetitive neuroglycopenicsymptoms without weight gain, and loss
of consciousness resolved with ingestion of sugary foods. Initially a
neuro-psychiatric pathology was suspected, the patient was on Sodium
Valproate and Antidepressants (SSRIs)with no improvement.After
thorough examinations, a pancreatic insulinoma was diagnosed. The
patient had a successful resection of the tumor, and her symptoms
disappeared entirely.
Copy Right, IJAR, 2022,. All rights reserved.

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Introduction:-
Among pancreatic endocrine tumors, the most common type is insulinoma. This tumor was reported in 1–4 people
per one million person years[1].Insulinomasare rare tumors of the pancreas that producelarge amounts of insulin and
are characterized by hyperinsulinemic hypoglycemia. 90% are benign, 90% are well-demarcated, solitary lesions,
and 90% are < 2cm in diameter[2].
This unusual tumors presents with episodes of neuroglycopenic signs that can be preceded by sympathetic
symptoms, misleading, sometimes, to a neuropsychiatric disorder. That was the case of a 15-years- old female with a
year history of episodic and repetitive neuroglycopenicsymptoms. Initially a neuro-psychiatric pathology was
suspected, the patient was on Sodium Valproate and Antidepressants (SSRIs) with no improvement.To find out after
thorough investigations that all her symtoms where due to a pancreatic insulinoma.
Case Presentation:
We report the case of a 15 years old female, who presented episodic symptoms of diaphoresis, dizziness, fatigue,
palpitations, tremulousness, hyperphagia without weight gain, and loss of consciousness.Thus, the patient was
misdiagnosed as neuro-psychological case and was treated by Sodium Valproate and Antidepressants (SSRIs)with
no improvement. The patient was admitted to the emergency room, capillary glycemia wasat 40 mg/dL and a
biological assessment was carried out in parallel with anintravenous infusion of 10% glucose that normalized her
Corresponding Author:-Zineb El Azime

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Address:-Department of Endocrinology, Diabetology and Nutrition - Hassan II University
Hospital.Fez, Morocco Epidemiology and Health Sciences Research Laboratory.
ISSN: 2320-5407 Int. J. Adv. Res. 10(02), 974-978
blood sugar levels and her symptoms resolvedafter stabilization then referred to our departmentfor further care and
investigations. She had a family history of diabetes type 2, and no family history of thyroid or pituitary disease.
The physical examination revealed a young well-nourished and well-developed woman without any obvious
developmental abnormality, witha weight of 59 Kg and BMI of 23 Kg/m2. Neurological and cardiorespiratory
examination was normal. The rest of the physical exam was normal.
The blood analysis revealed a serum glucose level of 0.3 g/L. The serum cortisol level was 18.9ug/dl, TSHus was
2.29uUI/ml and LT4 was 1.14ng/l, as well as HbA1c 4.6%, SGOT was 19ui/ml and SGPT 20 ui/ml and serum
creatinine was 4.4 mg/l. However the insulin level was 30,1uIU/L (2.5-25.0), C-peptide level was 4.46 ng/mL (>0.6)
and Turner index 300 (>150).
A computed tomography (CT) with contrast and magnetic resonance imaging (MRI) of the abdomen were also
normal.
Thus, endoscopic ultrasound (EUS) was performed showing a no vascularized,hypoechoic mass of pancreatic body
with 14 mm diameter (figure 1).
Figure 1:- Endoscopic ultrasound (EUS) : hypoechoic lesion of the pancreatic body.
Hormonal and radiological studies, including a brain MRI, were normal thus eliminating MEN-1.
The results were reviewed with the patient, and surgical management was planned. A preoperative EUS was
performed allowing locating preciselythe lesion in the Uncinate process of pancreas. Total enucleation of the tumor
was achieved.
The histopathological examination of the obtained piece was correspondent to a neuroendocrine pancreatic tumor,
well differentiated, and positively stained for synaptophysin, chromogranin and Ki 67 was 2%. (figure2, 3, 4)
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ISSN: 2320-5407 Int. J. Adv. Res. 10(02), 974-978
Figure 2:- Well-differentiated neuroendocrine tumor of the pancreas arranged in nidus and nodes.
Figure 3:- High and diffuse cytoplasmic staining for synaptophysin.
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ISSN: 2320-5407 Int. J. Adv. Res. 10(02), 974-978
Figure 4:- High and diffuse cytoplasmic marking of chromogranin.
After surgery, the patient’s glucose levels remained in normal during the subsequent 3-month follow-up period.
Discussion:-
The clinical diagnosis of insulinomais Based on Whipple's triad:neuroglycopenic and sympathetic symptoms in the
context of low serum glucose levels (under 50 mg/dL) that disappear quickly after glucose
intake[1].Sympathoadrenal and neurological symptoms may be highly suggestive of hypoglycemia, but they cannot
be ascribed to hypoglycemia unless the serum glucose is verified to be low at the same time as the symptoms[3].In
this case, the diagnosis of insulinoma was suggested because all three elements of the triad were present.
In the differential diagnosis in children with hypoglycemia, it is essential to determine whether ketonemia is also
present. It is common for ketonemia to be present in hypoglycemia caused by a spectrum of conditions, such as
substrate deficiency, deficiency of counterregulatory hormones, abnormal liver glycogen metabolism, or
ketotichypoglycemia [4].
The diagnosis of insulinoma can be challenging, it is generally diagnosed within a year and a half of symptom
onset[1]. Besides, in certain cases it can be mistaken as a seizure or psychiatric disorder, which was the case of our
patient who was misdiagnosed for nearly one year as a psychiatric patient.
Insulinoma diagnosis, in order to be absolutely established, requires compatible clinical presentation and the
presence of the following 6 criteria: blood glucose levels ≤ 40mg/dl, insulin ≥36 pmol/L, C-peptide ≥200 pmol/L,
proinsulin level ≥5.0 pmol/L, β-hydroxybutyrate ≤ 2.7 mmol/l and absence of plasma or urine sulfonylurea
metabolites[5].
The 72-h fasting test, considered as the gold standard for confirmation of insulinoma diagnosis, consists of
consecutive blood glucose and insulin levels tests until the patient becomes symptomatic, within 24h in most cases,
and then undergo a serial tests of insulin, proinsulin, C-peptide and insulin/glucose ratio [6] ,[11].
In some cases, insulinoma might occur in conjunction of multiple endocrine neoplasia type 1 (MEN-1 syndrome) an
autosomal dominant disorder due to a mutation in the MEN1 gene on chromosome 11. MEN1-associated
insulinomas are frequently multicentric and occur earlier compared with sporadic ones. Nevertheless the insulinoma
in our patient occured at an earlier age, hormonal and radiological studies, including a brain MRI, were normal.
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ISSN: 2320-5407 Int. J. Adv. Res. 10(02), 974-978
After biological diagnosis, conventional imaging to locate Insulinoma includes high-resolution CT, and MRI.In
some reported cases, abdominal ultrasonography identified an Insulinoma [7], but not in our case. Occasionally,
when non-invasive imaging studies fail to localize the tumor, unconventional imaging such endoscopic ultrasound
(EUS), octreotide scintigraphy, and arterial calcium stimulation test may be needed to localize insulinoma and guide
surgical treatment[2]. In our case CT and MRI failed to locate the tumor, only Endoscopic Ultra Sound was allowed
to detect the lesion and to localize it precisely pre-operatively.
The primary treatment for insulinoma is surgical excision. In most cases, enucleation of the tumor is performed
since insulinomas aresolitary, benign tumors in the majority of cases. Endoscopic-ultrasound-guided ethanol
ablation of pancreatic neuroendocrine tumors has proven safe and efficacious in adult patients in whom surgery is
contraindicated [8], a distal pancreatectomy,Whippleprocedure or subtotal pancreatectomyare also performed
instead impossible enucleation[9].Laparoscopic resection is becoming more popular[5].After operative treatment,
most patients are recovered from the disease. For patients not candidates for surgery, medical treatment may be used
such asdiazoxide or octreotide[10],[12].
Conclusions:-
Insulinoma is a rare tumor. The clinician should have a high suspicion index for this disease in patients with
whipple’striad even without a weight gain and should be careful to not misdiagnose it as neuro-psychological
disorder. They should also be aware of the importance of non conventional imaging such as Endoscopic Ultra Sound
that could be a very effective tool to diagnose and localize the tumors in case of a normal conventional imaging.
Bibliographie:-
[1] Alobaydun, Murtadha A., Albayat, Ali H., Al-nasif, Anwer A., et al. Pancreatic Insulinoma: Case Report of Rare
Tumor. Cureus, 2019, vol. 11, no 12.
[2] Okabayashi, Takehiro, Shima, Yasuo, Sumiyoshi, Tatsuaki, et al. Diagnosis and management of
insulinoma. World journal of gastroenterology: WJG, 2013, vol. 19, no 6, p. 829.
[3] Shreenivas, Aditya. et Leung, Vivien. A rare case of insulinoma presenting with postprandial hypoglycemia. The
American journal of case reports, 2014, vol. 15, p. 488.
[4] Escartín R, Brun N, García Monforte MN, Ferreres JC, Corripio R. Insulinoma: A Rare Cause of Hypoglycemia
in Childhood. Am J Case Rep. 2018;19:1121-1125. Published 2018 Sep 21.
[5] Giannis, Dimitrios, MORIS, Dimitrios, KARACHALIOU, Georgia-Sofia, et al. Insulinomas: from diagnosis to
treatment. A review of the literature. Journal of BU ON.: Official Journal of the Balkan Union of Oncology, 2020,
vol. 25, no 3, p. 1302-1314.
[6] Zhao, Yu‐ pei, Zhan, Han‐ xiang, ZHANG, Tai‐ ping, et al. Surgical management of patients with insulinomas:
result of 292 cases in a single institution. Journal of surgical oncology, 2011, vol. 103, no 2, p. 169-174.
[7]Diaconescu S, Aprodu G, Ioniuc I, Diaconescu MR, Miron L. Diagnostic Difficulties in a Pediatric Insulinoma: A
Case Report.Miron I, Medicine (Baltimore). 2016 Mar; 95(11):e3045.
[8] Qin SY, Lu XP, Jiang HX. EUS-guided ethanol ablation of insulinomas: case series and literature review.
Medicine (Baltimore). 2014 Sep; 93(14):e85
[9] Mehrabi A, Fischer L, Hafezi M, et al. A systematic review of localization, surgical treatment options, and
outcome of insulinoma. Pancreas. 2014;43(5):675-686.
[10] GROVER, Amelia C, SKARULIS, Monica, ALEXANDER, H. Richard, et al. A prospective evaluation of
laparoscopic exploration with intraoperative ultrasound as a technique for localizing sporadic insulinomas. Surgery,
2005, vol. 138, no 6, p. 1003-1008.
[11]DE MESTIER, Louis, LEPAGE, Come, BAUDIN, Eric, et al. Digestive neuroendocrine neoplasms (NEN):
French intergroup clinical practice guidelines for diagnosis, treatment and follow-up (SNFGE, GTE, Renaten,
Tenpath, FFCD, Gercor, Unicancer, SFCD, SFED, SFRO, SFR). Digestive and Liver Disease, 2020, vol. 52, no 5,
p. 473-492.
[12] HIRSHBERG, Boaz, COCHRAN, Craig, SKARULIS, Monica C., et al. Malignant insulinoma: spectrum of
unusual clinical features. Cancer: Interdisciplinary International Journal of the American Cancer Society, 2005, vol.
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ISSN: 2320-5407 Int. J. Adv. Res.

Journal Homepage: -www.journalijar.com

Zineb El Azime, Fatimazahra Melki, Houda Salhi and Hanane ElOuahabi

Copy Right, IJAR, 2022,. All rights reserved.

Corresponding Author:-Zineb El Azime

Figure 3:- High and diffuse cytoplasmic staining for synaptophysin.

Figure 4:- High and diffuse cytoplasmic marking of chromogranin.

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