Treatment with the anti-inflammatory drug colchicine resolved recurrent type 2 autoimmune pancreatitis (AIP) in patients who previously received other drugs, a case series in Italy found.
Two patients with type 2 AIP experienced clinical improvement and normalization in serological markers following administration of colchicine years after initially presenting to the emergency department and being treated for symptoms, reported Emanuel Della-Torre, MD, PhD, of the Universita Vita-Salute San Raffaele in Milan, and colleagues in a research letter in the .
These are the first recurrent type 2 AIP patients successfully treated by colchicine, which was chosen "because of its inhibitory effect on neutrophils, the most characteristic immune cells infiltrating type 2 AIP," the authors added.
"Type 2 autoimmune pancreatitis is an uncommon disorder, very challenging to diagnose because there is not an accurate biomarker to do that," said Philip Hart, MD, of the Ohio State University Wexner Medical Center in Columbus, who was not involved in this study. "Patients with type 2 AIP are quite unlikely to have relapse and this is an uncommon scenario that does come up."
AIP is histologically defined by the presence of granulocytic epithelial lesions (GELs) housing infiltrating neutrophils, and is often treated with corticosteroids to induce remission, while maintenance therapy is sustained by azathioprine, Della-Torre and colleagues noted.
The patients were two young men, a 19-year-old, who first presented with symptoms in November 2017, and a 25-year-old, who first presented with symptoms in April 2015. Neither had a history of alcohol or recreational drug misuse. Both presented to the emergency department with abdominal pain, and each had elevated levels of serum amylase and serum lipase, but serum IgG4, chromogranin A, neuron-specific enolase, and carbohydrate antigen 19-9 were all normal. Diagnostic imaging excluded gallstones and showed diffuse enlargement in the pancreas of both patients.
"We made the diagnosis of type 2 AIP by confirming that these patients had pancreatitis with GELs and excluding toxic, stone-related, IgG4-related, and genetic forms of pancreatitis," the authors noted.
Following an endoscopic ultrasound-guided fine needle biopsy to confirm their diagnosis, both patients were started on oral prednisone and had their prednisone dosage tapered over the course of 6 months.
No genetic mutations (CTRC, CFTR, PRSS1, or SPINK1) were found in either patient, the authors added.
The prednisone therapy alleviated pancreatic gland swelling in the 19-year-old patient, and the 25-year-old patient experienced serologic remission, both after a month of treatment.
Five months later, the 19-year-old patient started azathioprine, and was also treated with subcutaneous methotrexate and prednisone, tapered over 4 months, each time following increases in his amylase and lipase levels. In June 2019, imaging showed "diffuse enlargement" of the pancreas, and the patient refused glucocorticoids. His methotrexate was tapered and withdrawn when he started oral colchicine.
The 25-year-old patient experienced a recurrence of abdominal pain in June 2020, as well as increases in amylase and lipase levels and enlargement of the pancreas on imaging.
After starting oral colchicine, serologic and radiographic findings normalized within 2 weeks for the 19-year-old patient and 20 days later for the 25-year-old patient. As of August 2021, neither patient experienced additional flares.
The researchers acknowledged that formal clinical studies are needed to confirm these "preliminary findings," but concluded that based on these results, colchicine could be a promising treatment for type 2 AIP.
Hart characterized their hypothesis as "novel and interesting," but told 鶹ý he also wanted to see more studies "before adopting this into my personal clinical practice."
"It's always a challenge when multiple drugs are used together and it is hard to tell which drug contributed to the effect," he said.
Disclosures
No conflicts of interest were reported.
Primary Source
Annals of Internal Medicine
Chiabrando F, et al "Treating type 2 autoimmune pancreatitis with colchicine: A case series" Ann Intern Med 2021; DOI: 10.7326/L21-0281.