Literature DB >> 16808642

Multiple endocrine neoplasia type 2B diagnosed on suction rectal biopsy in infancy: a report of 2 cases.

Minzhi Yin1, Sebastian K King, John M Hutson, Chung Wo Chow.   

Abstract

Suction rectal biopsies in a newborn and a 10-month-old infant presenting with intestinal obstruction showed marked increase in neurons and nerve bundles in the submucosa. Although there were no syndromic features or a positive family history, mutation analysis of the RET proto-oncogene showed a de novo germline Met918Thr mutation in both patients, confirming the diagnosis of multiple endocrine neoplasia type 2B (MEN 2B). Thyroidectomy was performed at 9 and 14 months, showing medullary carcinoma and focal prominent C-cell hyperplasia, respectively. These 2 cases are presented to emphasize that when the submucosal plexus is obviously and prominently increased in suction rectal biopsies, diffuse intestinal ganglioneuromatosis should be considered. As this can be associated with genetic conditions, especially MEN 2B, it is crucial that further investigations be performed to ensure proper patient management, such as early thyroidectomy.

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Year:  2006        PMID: 16808642     DOI: 10.2350/06-05-0072.1

Source DB:  PubMed          Journal:  Pediatr Dev Pathol        ISSN: 1093-5266


  10 in total

1.  Review.

Authors:  Sujal Rangwalla; Cheryl E Gariepy
Journal:  Gastroenterol Hepatol (N Y)       Date:  2007-03

2.  [Genetic bases of Hirschsprung's disease].

Authors:  E Passarge; E Bruder
Journal:  Pathologe       Date:  2007-03       Impact factor: 1.011

Review 3.  Simple rules for a "simple" nervous system? Molecular and biomathematical approaches to enteric nervous system formation and malformation.

Authors:  Donald F Newgreen; Sylvie Dufour; Marthe J Howard; Kerry A Landman
Journal:  Dev Biol       Date:  2013-07-06       Impact factor: 3.582

Review 4.  Classification and diagnostic criteria of variants of Hirschsprung's disease.

Authors:  Florian Friedmacher; Prem Puri
Journal:  Pediatr Surg Int       Date:  2013-09       Impact factor: 1.827

5.  Diffuse intestinal ganglioneuromatosis in a child.

Authors:  Mika A B Matthews; Brent H Adler; Michael A Arnold; Soma Kumar; Ryan Carvalho; Gail E Besner
Journal:  J Pediatr Surg       Date:  2013-05       Impact factor: 2.545

Review 6.  Multiple Endocrine Neoplasia: A Genetically Diverse Group of Familial Tumor Syndromes.

Authors:  M Cristina Pacheco
Journal:  J Pediatr Genet       Date:  2016-03-09

7.  Timely diagnosis of multiple endocrine neoplasia 2B by identification of intestinal ganglioneuromatosis: a case series.

Authors:  Medard F M van den Broek; Ester B G Rijks; Peter G J Nikkels; Victorien M Wolters; Robert J J van Es; Hanneke M van Santen; Bernadette P M van Nesselrooij; Menno R Vriens; Rachel S van Leeuwaarde; Gerlof D Valk; Annemarie A Verrijn Stuart
Journal:  Endocrine       Date:  2021-01-21       Impact factor: 3.633

8.  Retinoic acid upregulates ret and induces chain migration and population expansion in vagal neural crest cells to colonise the embryonic gut.

Authors:  Johanna E Simkin; Dongcheng Zhang; Benjamin N Rollo; Donald F Newgreen
Journal:  PLoS One       Date:  2013-05-22       Impact factor: 3.240

Review 9.  Development of enteric neuron diversity.

Authors:  Marlene M Hao; Heather M Young
Journal:  J Cell Mol Med       Date:  2009-06-16       Impact factor: 5.310

10.  Multiple endocrine neoplasia 2B: Differential increase in enteric nerve subgroups in muscle and mucosa.

Authors:  John M Hutson; Pam J Farmer; Cristal J Peck; Chung W Chow; Bridget R Southwell
Journal:  World J Gastrointest Pathophysiol       Date:  2017-08-15
  10 in total

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