CASE REPORT Jejunal overexpression of peptide YY in celiac disease complicated with pneumatosis cystoides intestinalis Angela Gurrado • Simone Giungato • Ivana Catacchio • Domenico Piscitelli • Graziana Arborea • Giuseppe Piccinni • Mario Testini • Angelo Vacca Received: 25 July 2014 / Accepted: 16 September 2014 Ó Springer-Verlag Italia 2014 Abstract A 61-year old man with coeliac disease and chronic lack of appetite, malabsorption and weight loss, despite the gluten-free diet, was operated because of a sub- diaphragmatic free air due to a small-bowel pneumatosis cystoides intestinalis (PCI). The jejunum showed granu- lomatous lesions with a honeycombed appearance of air cysts in the submucosa/subserosa. We found overexpres- sion of peptide YY (PYY) into only the jejunum with PCI, while the expression was very weak or absent in the tissue without cysts. One year after surgery, he had no abdominal pain or PCI recurrence. The above chronic symptoms were plausibly attributable to the PYY. Keywords Pneumatosis cystoides intestinalis (PCI) Á Coeliac disease (CD) Á Peptide YY Á Small bowel Á Surgery Introduction Pneumatosis cystoides intestinalis (PCI) is an uncommon condition characterized by multiple gas-filled cysts within the wall of the gastrointestinal tract which are easily detectable on radiological examination [1]. Clinical mani- festations are abdominal pain and distention, diarrhoea and mucous and/or blood stool. The main PCI feature is the pneumoperitoneum without peritoneal irritation as a result of the cyst rupture [2]. However, Pp or bowel obstruction are uncommon life-threatening complications [3]. PCI is frequently associated to several pathologies including coeliac disease (CD) [4–6]. Peptide YY (PYY) is a 36-amino-acid peptide synthe- sized and released within the distal small bowel and colon from enteroendocrine cells (so called L-cells). It is an anorectic neurohormonal factor that plays a key role in the regulation of energy homoeostasis, gastrointestinal motility and water/electrolytes absorption [7, 8]. Previous studies demonstrated abnormal cellular density and plasma levels of PYY in patients with CD who were not on gluten-free diet (GFD); the PYY expression probably represented a response to diarrhoea and produced a slowdown of the jejunal and colic motility [8–10]. Here, we show a patient with CD who, despite a GFD, was chronically suffering of inappetence, severe weight loss, recurrent diarrhoea and abdominal pain. He also had a PCI in the jejunum which displayed a diffuse overexpression of PYY. Since the patient was fully recovered after surgical removal of the PCI, we hypothesize that PYY was responsible of the above chronic symptoms. The patient gave the informed consent, and the work was performed following all the guidelines for experimental investigation with human subjects. Case report A 61-year old man with a 4-year history of CD on GFD was admitted to our department because of a bowel A. Gurrado Á S. Giungato Á G. Piccinni Á M. Testini (&) Unit of Endocrine, Digestive and Emergency Surgery, Department of Biomedical Sciences and Human Oncology, University of Bari Medical School, Policlinico, Piazza Giulio Cesare, 11, 70124 Bari, Italy e-mail: mario.testini@uniba.it I. Catacchio Á A. Vacca Internal Medicine Units, Department of Biomedical Sciences and Human Oncology, University of Bari Medical School, 70124 Bari, Italy D. Piscitelli Á G. Arborea Unit of Pathology, Department of Emergency Surgery and Organ Transplantation, University Medical School, 70124 Bari, Italy 123 Clin Exp Med DOI 10.1007/s10238-014-0314-5