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Vol. 4, No. 3, 2010
Issue release date: September – December
Section title: Published: October 2010
Open Access Gateway
Case Rep Gastroenterol 2010;4:452–456
(DOI:10.1159/000321573)

Solitary Peutz-Jeghers Polyp in a Paediatric Patient

Retrosi G.a · Nanni L.a · Vecchio F.M.b · Manzoni C.a · Canali R.a · Busato G.a · Pintus C.a
aDepartment of Paediatrics, Division of Paediatric Surgery, and bDepartment of Human Pathology, Università Cattolica del Sacro Cuore, Policlinico ‘A. Gemelli’, Rome, Italy
email Corresponding Author

Abstract

Hamartomatous polyps of Peutz-Jeghers are mostly found in patients affected by Peutz-Jeghers syndrome (PJS), but they can be rarely encountered in the general population. It is unclear whether a solitary Peutz-Jeghers polyp (PJP) is an incomplete form of PJS or a separate entity. We report a case of solitary PJP in a paediatric patient in whom the other features of PJS were absent. The patient underwent laparotomy due to small bowel intussusception secondary to an ileac polyp. Histological examination showed the characteristic features of PJP, but the patient did not fulfill the WHO criteria for PJS diagnosis (negative family history for PJS and absence of mucocutaneous pigmentation); moreover analysis of the STK11/LKB1 gene did not reveal any genomic abnormality. The clinical and investigative findings in our case suggest that the solitary PJP can be considered a different clinical entity from PJS.

© 2011 S. Karger AG, Basel


  

Key Words

  • Peutz-Jeghers syndrome
  • Solitary Peutz-Jeghers polyp
  • STK11/LKB1 gene

  

Author Contacts

Giuseppe Retrosi, MD
Department of Paediatrics, Division of Paediatric SurgeryUniversità Cattolica del Sacro Cuore, Policlinico ‘A. Gemelli’
Largo A. Gemelli 8, IT–00168 Rome (Italy)
Tel. +39 063 015 4032, Fax +39 063 051 162, E-Mail retrosi@alice.it

  

Article Information

Published online: October 18, 2010
Number of Print Pages : 5
Number of Figures : 3,

  

Publication Details

Case Reports in Gastroenterology

Vol. 4, No. 3, Year 2010 (Cover Date: September - December)

Journal Editor: Urrutia R. (Rochester, Minn.)
ISSN: NIL (Print), eISSN: 1662-0631 (Online)

For additional information: http://www.karger.com/CRG


Open Access License / Drug Dosage / Disclaimer

Open Access License: This is an Open Access article licensed under the terms of the Creative Commons Attribution-NonCommercial 3.0 Unported license (CC BY-NC) (www.karger.com/OA-license), applicable to the online version of the article only. Distribution permitted for non-commercial purposes only.
Drug Dosage: The authors and the publisher have exerted every effort to ensure that drug selection and dosage set forth in this text are in accord with current recommendations and practice at the time of publication. However, in view of ongoing research, changes in goverment regulations, and the constant flow of information relating to drug therapy and drug reactions, the reader is urged to check the package insert for each drug for any changes in indications and dosage and for added warnings and precautions. This is particularly important when the recommended agent is a new and/or infrequently employed drug.
Disclaimer: The statements, opinions and data contained in this publication are solely those of the individual authors and contributors and not of the publishers and the editor(s). The appearance of advertisements or/and product references in the publication is not a warranty, endorsement, or approval of the products or services advertised or of their effectiveness, quality or safety. The publisher and the editor(s) disclaim responsibility for any injury to persons or property resulting from any ideas, methods, instructions or products referred to in the content or advertisements.

Abstract

Hamartomatous polyps of Peutz-Jeghers are mostly found in patients affected by Peutz-Jeghers syndrome (PJS), but they can be rarely encountered in the general population. It is unclear whether a solitary Peutz-Jeghers polyp (PJP) is an incomplete form of PJS or a separate entity. We report a case of solitary PJP in a paediatric patient in whom the other features of PJS were absent. The patient underwent laparotomy due to small bowel intussusception secondary to an ileac polyp. Histological examination showed the characteristic features of PJP, but the patient did not fulfill the WHO criteria for PJS diagnosis (negative family history for PJS and absence of mucocutaneous pigmentation); moreover analysis of the STK11/LKB1 gene did not reveal any genomic abnormality. The clinical and investigative findings in our case suggest that the solitary PJP can be considered a different clinical entity from PJS.

© 2011 S. Karger AG, Basel


  

Author Contacts

Giuseppe Retrosi, MD
Department of Paediatrics, Division of Paediatric SurgeryUniversità Cattolica del Sacro Cuore, Policlinico ‘A. Gemelli’
Largo A. Gemelli 8, IT–00168 Rome (Italy)
Tel. +39 063 015 4032, Fax +39 063 051 162, E-Mail retrosi@alice.it

  

Article Information

Published online: October 18, 2010
Number of Print Pages : 5
Number of Figures : 3,

  

Publication Details

Case Reports in Gastroenterology

Vol. 4, No. 3, Year 2010 (Cover Date: September - December)

Journal Editor: Urrutia R. (Rochester, Minn.)
ISSN: NIL (Print), eISSN: 1662-0631 (Online)

For additional information: http://www.karger.com/CRG


Article / Publication Details

First-Page Preview
Abstract of Published: October 2010

Published online: 10/18/2010
Issue release date: September – December

Number of Print Pages: 5
Number of Figures: 3
Number of Tables: 0

ISSN: (Print)
eISSN: 1662-0631 (Online)

For additional information: http://www.karger.com/CRG


Open Access License / Drug Dosage

Open Access License: This is an Open Access article licensed under the terms of the Creative Commons Attribution-NonCommercial 3.0 Unported license (CC BY-NC) (www.karger.com/OA-license), applicable to the online version of the article only. Distribution permitted for non-commercial purposes only.
Drug Dosage: The authors and the publisher have exerted every effort to ensure that drug selection and dosage set forth in this text are in accord with current recommendations and practice at the time of publication. However, in view of ongoing research, changes in goverment regulations, and the constant flow of information relating to drug therapy and drug reactions, the reader is urged to check the package insert for each drug for any changes in indications and dosage and for added warnings and precautions. This is particularly important when the recommended agent is a new and/or infrequently employed drug.
Disclaimer: The statements, opinions and data contained in this publication are solely those of the individual authors and contributors and not of the publishers and the editor(s). The appearance of advertisements or/and product references in the publication is not a warranty, endorsement, or approval of the products or services advertised or of their effectiveness, quality or safety. The publisher and the editor(s) disclaim responsibility for any injury to persons or property resulting from any ideas, methods, instructions or products referred to in the content or advertisements.