A case of single incision laparoscopic total colectomy for intestinal neuronal dysplasia type B

Masuda, Taro; Nonaka, Takashi; Adachi, Toshiyuki; Hisanaga, Makoto; Nagayoshi, Shigeki; Tokunaga, Takayuki; Taniguchi, Koichi; Kurohama, Hirokazu; Ito, Masahiro; Fujioka, Hikaru

doi:10.1016/j.ijscr.2017.07.026

Cited by 3 publications

(1 citation statement)

References 23 publications

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“…IND-B is regarded as a phenotype of relative enteric neural immaturity that can only be recognized with confidence after the age of 1 year and often spontaneously disappears by the age of 4 years [8]. It is rarely reported in adult patients [9] and is characterized by hyperplasia of the myenteric nerves accompanied by giant ganglia [10]. IND remains surrounded by controversies related to its definition, etiopathogenesis, diagnostic criteria, and therapeutic possibilities [11].…”

Section: Introductionmentioning

confidence: 99%

Diagnostic utility of Bcl-2 immunohistochemical expression in pediatric functional bowel obstruction cases with ganglionated specimens

et al. 2022

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Background Functional disturbances of the gastrointestinal tract are caused by a number of neurodysplastic conditions, including diseases that are rarer than Hirschsprung’s disease (HSCR), such as ganglion cell immaturity and intestinal neuronal dysplasia (IND). Bcl-2 shows positive immunoreactivity in degenerative and immature ganglion cells (IGCs). This work evaluates the implication of the immunohistochemical expression of Bcl-2 in cases of pediatric intestinal pseudo-obstruction (IPO) with ganglionated specimens. This 2-year prospective observational study was conducted from January 2017 to December 2019 on all intestinal specimens for cases referred from a pediatric surgery department with clinical data suggesting IPO (abdominal distension > 3 months, intermittent bilious vomiting, no radiological evidence of the transitional zone of HSCR or evidence of mechanical obstruction). The exclusion criteria were patients with IPO related to myopathic disorders, aganglionic biopsies, and specimens with inadequate tissue blocks. The same number of intestinal specimens of cases without IPO was used as a control group. All specimens underwent a histologic examination of ganglion cells and nerve bundles and were also evaluated for Bcl-2 immunohistochemical expression. Results Twenty-one specimens were analyzed, including six colonic resection specimens and 15 intestinal biopsies taken by full-thickness transanal biopsy (n = 12) or incisional biopsy from inadequately functioning stomas (n = 3). The mean ± standard deviation age of the patient cohort was 22 ± 7.4 months (range, 19–153 months), and there were 13 (61.9%) male patients. Bcl-2 protein was strongly positive in 57.1% of the cases, weakly positive in 19%, and negative in 23.8% as well as in all 21 control specimens. A highly significant correlation was observed between Bcl-2 expression and ganglion cell number (P < 0.001), where all hyperganglionic specimens showed strong positivity compared with the hypoganglionic and adequately ganglionic specimens. A positive association was also found between Bcl-2 expression and IND compared with other disorders of dysganglionosis (P = 0.04) and post-HSCR cases (P = 0.002). Conclusion Bcl-2 immunohistochemistry is a valuable tool to diagnose allied disorders of HSCR through its expression in IGCs, which are difficult to identify by conventional hematoxylin and eosin staining.

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Section: Introductionmentioning

confidence: 99%

Diagnostic utility of Bcl-2 immunohistochemical expression in pediatric functional bowel obstruction cases with ganglionated specimens

et al. 2022

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Single-incision laparoscopic total colectomy using an organ retractor: A case report

Ishimaru

Tominaga

Nonaka

et al. 2020

Annals of Medicine and Surgery

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Intestinal Neuronal Dysplasia Type B: An Updated Review of a Problematic Diagnosis

Kapur

Reyes‐Múgica

2018

Archives of Pathology &Amp; Laboratory Medicine

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Context.-Intestinal neuronal dysplasia type B (IND B) is a controversial histopathologic phenotype that has been associated with intestinal dysmotility, either as an isolated condition or in conjunction with established pathologic disorders (eg, Hirschsprung disease). Many factors contribute to the debate over the existence and/or clinical significance of IND B, including a large body of published data based on inconsistent diagnostic criteria and methods, which have fostered many unwarranted conclusions that lack sufficient scientific basis.Objective.-To critically analyze existing published data regarding IND B to provide supporting evidence-based diagnostic practice and to stimulate necessary and scientifically sound research.Data Sources.-This update focuses on published literature related to the pathology of IND B because without a reliable pathologic diagnosis, studies of epidemiology, pathogenesis, natural history, management, and outcome are all suspect. Problems with existing data are identified explicitly with suggestions as to how future investigations should be designed and evaluated to better understand this entity.Conclusions.-Inconsistencies in diagnostic criteria and methods used to define IND B justifiably encumber the universal acceptance of IND B as a neuropathologic etiology for intestinal dysmotility. IND B will remain a controversial diagnosis until rigorous, well-controlled scientific studies are conducted to establish reproducible and reliable diagnostic criteria that reliably translate from one laboratory to another.

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A case of single incision laparoscopic total colectomy for intestinal neuronal dysplasia type B

Cited by 3 publications

References 23 publications

Diagnostic utility of Bcl-2 immunohistochemical expression in pediatric functional bowel obstruction cases with ganglionated specimens

Diagnostic utility of Bcl-2 immunohistochemical expression in pediatric functional bowel obstruction cases with ganglionated specimens

Single-incision laparoscopic total colectomy using an organ retractor: A case report

Intestinal Neuronal Dysplasia Type B: An Updated Review of a Problematic Diagnosis

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