Erratum to “Counting myenteric ganglion cells in histologic sections: an empirical approach” [Hum Pathol 2010;41:1097-1108]

Swaminathan, Maya; Kapur, Raj P.

doi:10.1016/j.humpath.2012.01.001

Cited by 3 publications

(4 citation statements)

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“…19,20 Formal counts of Hu C/D-immunopositive neurons can be useful to confirm the diagnosis but should follow well-defined criteria for ganglion cell discrimination and may benefit from internally collected control data to reduce the effect of observer bias on ganglion cell counts. 11 We applied the criteria proposed by Yoshimaru et al, 21 and our results were remarkably similar to theirs with mean values ~3 ganglion cells and ~15 ganglion cells per linear mm for CMH patients and controls, respectively. In addition, alternative diagnostic criteria have been published for CMH, some of which rely on enzyme histochemistry (eg, lactate dehydrogenase) or whole-mount preparations, [22][23][24] which are not practical in most clinical settings.…”

Section: Discussionsupporting

confidence: 83%

“…Immunostained neuronal cell bodies were quantified along a contiguous length of myenteric plexus, which measured at least 10 linear millimeters. As described in detail elsewhere, 11 a ganglion cell body was only counted if granular brown staining covered the nucleus or encircled at least 50% of the nuclear circumference; ambiguous nuclei were not counted. Neighboring, but not necessarily immediately adjacent, sections from the same tissue blocks were immunolabelled with each of the antibodies described above.…”

Section: Quantification Of Immunolabelled Neuronsmentioning

confidence: 99%

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Congenital Myenteric Hypoganglionosis

Kapur

Bellizzi

Bond

et al. 2021

American Journal of Surgical Pathology

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Congenital myenteric hypoganglionosis is a rare developmental disorder characterized clinically by severe and persistent neonatal intestinal pseudoobstruction. The diagnosis is established by the prevalence of small myenteric ganglia composed of closely spaced ganglion cells with sparse surrounding neuropil. In practice, the diagnosis entails familiarity with the normal appearance of myenteric ganglia in young infants and the ability to confidently recognize significant deviations in ganglion size and morphology. We review clinical, histologic, and immunohistochemical findings from 12 patients with congenital myenteric hypoganglionosis in comparison with similar data from age-matched controls and clearly delineate the diagnostic features of the condition. Practical guidelines are provided to assist surgical pathologists, who are likely to encounter this condition only infrequently. The diagnosis typically requires full-thickness intestinal biopsy as the abnormality is confined to the myenteric plexus in many patients. Immunohistochemistry for Hu C/D may be used to confirm hypoganglionosis. Reduced staining for calretinin and NeuN implicates a selective deficiency of intrinsic primary afferent neurons in this disease.

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Section: Discussionsupporting

confidence: 83%

Section: Quantification Of Immunolabelled Neuronsmentioning

confidence: 99%

Congenital Myenteric Hypoganglionosis

Kapur

Bellizzi

Bond

et al. 2021

American Journal of Surgical Pathology

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“…Previous reports could not be considered as reliable control data for children due to lack of objectivity, reproducibility, and clinical usefulness in the following aspects: the data were obtained from patients with a large age range, including adult cases; [13][14][15]20,21 evaluations were performed with HE staining alone 8,13 or with staining methods that are no longer in use; 7,14,16,19,22 the use of whole-mount preparations; 15,19 and no definition of the myenteric plexus components to be counted. 7,8,15,[17][18][19][20][21][22][23][24][25][26][27][28][29] Therefore, to collect reliable data, we quantified myenteric plexus components by selecting pediatric 13.9 (9.9-17.7) 0.7 (0.3-1.7) < 0.001* FIGURE 5. Comparison of each parameter in autopsy and IHG cases.…”

Section: Discussionmentioning

confidence: 99%

“…Morphologic measurements of myenteric ganglion cells and myenteric ganglia were carried out with HuC/HuD-immunostained sections and CD56-immunostained sections, which are useful for histologic evaluation. 1,9,[24][25][26] The measured myenteric ganglion cells were defined as cells (1) located between the inner circular and outer longitudinal layers of the muscularis externa, (2) immunostained by HuC/HuD, and (3) showing a nucleus with a visible contour of at least a semicircular shape (Fig. 3).…”

Section: Histopathologic Assessment and Measurementsmentioning

confidence: 99%

A Novel Objective Pathologic Criterion for Isolated Hypoganglionosis

Tamaki,

Kohashi,

Yoshimaru

et al. 2024

American Journal of Surgical Pathology

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Isolated hypoganglionosis (IHG) is histologically characterized by small numbers of myenteric ganglion cells and small myenteric ganglia; however, no numerical diagnostic criteria for IHG have been established. Therefore, this study aimed to develop quantitative pathologic criteria for IHG. We evaluated 160 resected intestinal tissue specimens from 29 pediatric autopsies and 10 IHG cases. These specimens were obtained from the jejunum, ileum, ascending colon, transverse colon, and rectum. Morphologic features of the myenteric ganglion cells and myenteric ganglia were quantified and analyzed in digitized HuC/HuD-immunostained and CD56-immunostained sections, respectively. Quantitative criteria were developed with a scoring system that used parameters with the area under the receiver operating characteristic curve (AUC) values >0.7 and sensitivity and specificity exceeding 70%. The selected parameters were the number of myenteric ganglion cells per cm and the number of myenteric ganglia with an area >2500 µm2per cm. The score for each parameter ranged from −1 to 2, and the total score of the scoring system ranged from −2 to 4. With a cutoff value of ≥2 (AUC, 0.98; 95% CI: 0.96-1.00), the scoring system had a sensitivity of 96% (95% CI: 0.82-1.00) and a specificity of 99% (95% CI: 0.95-1.00). We devised a novel pathologic criterion based on the quantification of the number of myenteric ganglion cells and ganglia. Furthermore, this criterion showed high diagnostic accuracy and could lead to a definitive diagnosis of IHG in clinical practice.

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Dental pulp stem cells as a therapy for congenital entero-neuropathy

Yoshimaru

Yamaza

Kajioka

et al. 2022

Sci Rep

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Hirschsprung’s disease is a congenital entero-neuropathy that causes chronic constipation and intestinal obstruction. New treatments for entero-neuropathy are needed because current surgical strategies have limitations5. Entero-neuropathy results from enteric nervous system dysfunction due to incomplete colonization of the distal intestine by neural crest-derived cells. Impaired cooperation between the enteric nervous system and intestinal pacemaker cells may also contribute to entero-neuropathy. Stem cell therapy to repair these multiple defects represents a novel treatment approach. Dental pulp stem cells derived from deciduous teeth (dDPSCs) are multipotent cranial neural crest-derived cells, but it remains unknown whether dDPSCs have potential as a new therapy for entero-neuropathy. Here we show that intravenous transplantation of dDPSCs into the Japanese Fancy-1 mouse, an established model of hypoganglionosis and entero-neuropathy, improves large intestinal structure and function and prolongs survival. Intravenously injected dDPSCs migrate to affected regions of the intestine through interactions between stromal cell-derived factor-1α and C-X-C chemokine receptor type-4. Transplanted dDPSCs differentiate into both pacemaker cells and enteric neurons in the proximal colon to improve electrical and peristaltic activity, in addition to their paracrine effects. Our findings indicate that transplanted dDPSCs can differentiate into different cell types to correct entero-neuropathy-associated defects.

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Erratum to “Counting myenteric ganglion cells in histologic sections: an empirical approach” [Hum Pathol 2010;41:1097-1108]

Cited by 3 publications

References 0 publications

Congenital Myenteric Hypoganglionosis

Congenital Myenteric Hypoganglionosis

A Novel Objective Pathologic Criterion for Isolated Hypoganglionosis

Dental pulp stem cells as a therapy for congenital entero-neuropathy

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