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Pancreaticopleural fistula in a Thai boy with SPINK1 c.101A>G substitution variant-related chronic pancreatitis: a case report and literature review

Chomanad Chittchang
Chomanad Chittchang
, 
Nisa Netinatsunton
Nisa Netinatsunton
 and 
Supika Kritsaneepaiboon
Supika Kritsaneepaiboon
   | Apr 29, 2022
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Article Category: Clinical report
Published Online: Apr 29, 2022
Page range: 99 - 107
DOI: https://doi.org/10.2478/abm-2022-0012
Keywords
© 2022 Chomanad Chittchang et al., published by Sciendo
This work is licensed under the Creative Commons Attribution 4.0 International License.

Background

Chronic pancreatitis is the most common etiology of pancreaticopleural fistula (PPF) in children, and underlying genetic variations are now widely known, accounting for most chronic pediatric pancreatitis.

Case report

We describe a case of previously undetected chronic pancreatitis and PPF with a SPINK1 variation in a 10-year-old Thai boy who presented with massive left pleural effusion. Magnetic resonance cholangiopancreatography (MRCP) revealed disruption of the pancreatic duct, which was communicating with a large pancreatic pseudocyst with mediastinal extension. The patient subsequently underwent endoscopic intervention with improved clinical symptoms. We also reviewed the imaging findings of 12 other reported cases of pediatric PPF.

Conclusions

Massive pleural effusion due to PPF can be an atypical manifestation in children with chronic pancreatitis. MRCP is the preferable imaging study for PPF due to the production of highly detailed images of pancreatic duct disruptions and anatomy, and the imaging is helpful to guide for appropriate treatment. Tests for genetic variation are also recommended in a child with chronic pancreatitis.
eISSN:
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Language:
English
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Journal Subjects:
Medicine, Assistive Professions, Nursing, Basic Medical Science, other, Clinical Medicine
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