해외 증례Autoimmune gastritis as an unexpected cause of diarrhea in a young adult with type I diabetes: a case report

관리자
2023-09-20
조회수 79

J Med Case Rep. 2023 Jul 29;17(1):342. doi: 10.1186/s13256-023-04039-0. 


Background

* Type 1 diabetes mellitus (T1DM): immune mediated damage of pancreatic beta cells and subsequent hyperglycemia

* Self-management (important component): close serum glucose monitoring & aggressive insulin therapy

* Maintaining tight glycemic control: prevent complications of chronic hyperglycemia

* Annual foot and dilated ocular exams, quarterly Hemoglobin A1c (HgbA1c), annual creatinine and urine albumin-to-creatinine ratio

* Other autoimmune conditions: most commonly thyroid or celiac disease

- Thyroid stimulating hormone testing (thyroid antibody test)

- Celiac disease: tissue transglutaminase Immunoglobulin (IgA) level, total IgA level (IgA deficiency가 celiac dis. risk를 증가시킴)


Case presentation 

* 20 year old African American female with T1DM (diagnosed at age 10, last HgbA1c=7.8%)

* C/Cx: 2 months of nocturnal, non-bloody diarrhea, left lower quadrant pain, and nausea (daytime: no symptoms)

* P/Hx: T1DM without history of diabetic ketoacidosis (Anti-glutamic acid decarboxylase antibody (13.1 U/mL), Islet Antigen-2 Autoantibody: positive), IgA defciency high risk for celiac disease with HLA DQ2 heterozygosity 

* F/Hx: Father (hyperlipidemia, hypertension), maternal grandfather (Type 2 DM)

* P/Ex: slightly diminished sensation to vibration and pinprick in bilateral toes

* Initial Lab-workup: low total IgA of < 6.7  mg/dL

* Elevated fasting gastrin level and an elevated parietal cell antibody level 

Table 1. Patient’s clinical course and lab evaluation


* Upper and lower endoscopy: unremarkable, however histology revealed a pattern consistent with autoimmune gastritis (Figure 1), Biopsies were negative for Heliobacter pylori 

* Small bowel biopsy: did not demonstrate the typical pathologic pattern of celiac disease (i.e. damage to or fattening of villi, increased intraepithelial lymphocytes, crypt hyperplasia)

Fig. 1 A) Duodenal biopsy demonstrating preserved villous architecture with long villous structures. There is no increase in intra-epithelial lymphocytes. B) Gastric body biopsy with chronic lymphoplasmacytic infammation in the lamina propria (lower left). ×50 magnifcation. C) Gastric body biopsy at ×100, demonstrating increased chronic lamina propria infammation and lack of parietal cells.


* Treatment: vitamin B12 supplementation therapy (administered as a 1000 mcg injection once a week for 8 weeks)

-> resolution of lower extremity symptoms, improvement in her gastrointestinal symptoms, and continued normal Vitamin B12 levels and complete blood counts 

* Further management : patient’s symptomatology and regular lab screening for vitamin B12 defciency and anemia 


Discussion and conclusions

Autoimmune gastritis must be considered in pediatric patients with pre-existing autoimmune conditions and new gastrointestinal or neurologic symptoms, as significant morbidity, and even mortality can occur if undetected, with potential for development of symptomatic macrocytic anemia from Vitamin B12 and iron depletion, as well as risk of ongoing infammation resulting in preneoplastic and neoplastic changes within the gastrointestinal tract.


<Abstract>

Background 

 Type 1 diabetes mellitus (T1DM) is a lifelong diagnosis that involves immune-mediated damage of pancreatic beta cells and subsequent hyperglycemia, manifesting as: polyuria, polydipsia, polyphagia, and weight loss. Treatment of type 1 diabetes centers on insulin administration to replace or supplement the body’s own insulin with the goal of achieving euglycemia and preventing or minimizing complications. Patients with T1DM are at risk for developing other autoimmune conditions, most commonly thyroid or celiac disease. 

Case presentation 

 A 20-year-old African American female with T1DM was referred by her endocrinologist to pediatric gastroenterology for 2 months of nocturnal, non-bloody diarrhea, left lower quadrant pain, and nausea; she was also being followed by neurology for complaints of lower extremity paresthesias and pain. The patient’s initial lab-workup was remarkable for a low total Immunoglobulin A (IgA) level of<6.7 mg/dL. As IgA defciency is associated with an increased risk of celiac disease, the patient underwent upper and lower endoscopy, which was grossly unremarkable; however, histology revealed a pattern consistent with autoimmune gastritis. Subsequent serum evaluation was remarkable for an elevated fasting gastrin level and an elevated parietal cell antibody level without macrocytic anemia, iron defciency, or vitamin B12 depletion. The patient was diagnosed with autoimmune gastritis (AIG) and subsequently initiated on parenteral B12 supplementation therapy with improvement in her neurologic and gastrointestinal symptoms. 

Conclusion 

 This case illustrates the importance of recognition of red fag fndings in a patient with known autoimmune disease. Following well-established health maintenance recommendations for individuals with T1DM ensures that common comorbidities will be detected. Autoimmune gastritis, while a rarer pathology in the pediatric population, deserves consideration in patients with pre-existing autoimmune conditions and new gastrointestinal or neurologic symptoms, as AIG can be associated with poor outcomes and risk of malignancy. Initial lab fndings associated with an eventual diagnosis of AIG typically include anemia, iron defciency, or Vitamin B12 defciency. However, as demonstrated in this case, symptoms of AIG can rarely present before anemia or Vitamin B12 defciency develops. To prevent permanent neurological damage, parenteral Vitamin B12 therapy must be considered even in the absence of Vitamin B12 defciency, especially in those patients already experiencing neurological symptoms.

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