Case Report
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World J Gastroenterol. Sep 14, 2011; 17(34): 3948-3952
Published online Sep 14, 2011. doi: 10.3748/wjg.v17.i34.3948
Severe chronic diarrhea and maculopapular rash: A case report
Alessandra Elvevi, Federica Grifoni, Federica Branchi, Umberto Gianelli, Dario Conte
Alessandra Elvevi, Federica Branchi, Dario Conte, Gastrointestinal Unit 2, Fondazione IRCCS Cà Granda, Ospedale Maggiore Policlinico and University of Milan, 20121 Milan, Italy
Federica Grifoni, Hematology Unit, Fondazione IRCCS Cà Granda, Ospedale Maggiore Policlinico and University of Milan, 20121 Milan, Italy
Umberto Gianelli, Department of Pathology, Fondazione IRCCS Cà Granda, Ospedale Maggiore Policlinico and University of Milan, 20121 Milan, Italy
Author contributions: Elvevi A, Grifoni F, Branchi F, Gianelli U and Conte D contributed equally to this work; Elvevi A and Conte D wrote the paper.
Correspondence to: Dario Conte, MD, Gastrointestinal Unit 2, Fondazione IRCCS Cà Granda, Ospedale Maggiore Policlinico and University of Milan, 20121 Milan, Italy. dario.conte@unimi.it
Telephone: +39-02-55033418 Fax: +39-02-55033644
Received: September 23, 2010
Revised: February 26, 2011
Accepted: March 5, 2011
Published online: September 14, 2011
Abstract

Systemic mastocytosis (SM) is a heterogeneous disease of the bone marrow characterized by abnormal growth, accumulation and activation of clonal mast cells (MCs). We report a case of SM with multi-organ involvement. A 30-year-old man presented with diarrhea, flushing, maculopapular rash with itching and weight loss. The upper and lower gastrointestinal endoscopies showed macroscopic involvement of stomach and duodenum; mucosal samples from stomach, duodenum, colon and distal ileum showed mucosal infiltration by large, spindle-shaped MCs with abnormal surface molecule expression (CD2 and CD25), a picture fully consistent with SM, according to the World Health Organization diagnostic criteria. A computed tomography scan showed diffuse lymphadenopathy, hepatosplenomegaly and diffuse small bowel involvement. Bone marrow aspirate and biopsy were diagnostic for SM; serum tryptase levels were increased (209 ng/mL, normal values < 20 ng/mL). The conclusive diagnosis was smouldering SM. There were no therapeutic indications except for treatment of symptoms. The patient was strictly followed up because of the risk of aggressive evolution.

Keywords: Mast cells; Systemic mastocytosis; Bone marrow; Tryptase