Sporadic Viscoral Myopathy with Inclusion Bodies

Fogel, S P; deTar, Michael; Shimada, Hideaki; Chandrasoma, Para

doi:10.1097/00000478-199305000-00006

Cited by 21 publications

(8 citation statements)

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“…On the basis that PAS-positive inclusions were only found in cases of gastrointestinal dysmotility in this study, and that such inclusions have only previously been reported by others [4,20,21] in similar conditions, it seems likely that they would be of some significance. On the basis that PAS-positive inclusions were only found in cases of gastrointestinal dysmotility in this study, and that such inclusions have only previously been reported by others [4,20,21] in similar conditions, it seems likely that they would be of some significance.…”

Section: Discussionsupporting

confidence: 79%

“…The characteristic pathological features of this disorder were highlighted by PAS staining, with ovoid inclusion bodies, 2-30 µm in length, demonstrated. Similar or perhaps identical inclusion bodies have been reported in three patients with intestinal pseudo-obstruction and visceral myopathy, which was either idiopathic (two patients: colonic inclusions) [4] or found (a single case) in association with a probable diagnosis of scleroderma (ileal inclusions) [20,21]. Similar or perhaps identical inclusion bodies have been reported in three patients with intestinal pseudo-obstruction and visceral myopathy, which was either idiopathic (two patients: colonic inclusions) [4] or found (a single case) in association with a probable diagnosis of scleroderma (ileal inclusions) [20,21].…”

Section: Introductionsupporting

confidence: 80%

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A systematic analysis of polyglucosan bodies in the human gastrointestinal tract in health and disease

et al. 2003

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Polyglucosan inclusion bodies have been described in smooth muscle of the gastrointestinal tract of aged dogs, and rarely in association with enteric dysmotility in humans. We have systematically examined the human small and large bowel for the presence of such inclusions in health and motility disorders. Systematic, blinded, dual observer analysis of colonic and ileal tissue from patients (n=80, age 20-92 years) undergoing large bowel resections for non-dysmotile conditions, principally neoplasia was performed, as well as retrospective review of all intestinal tissues referred for specialist histochemistry from patients undergoing surgery for motility disorders. All sections were stained with haematoxylin and eosin and periodic acid-Schiff stains. No polyglucosan bodies were identified in any specimen without dysmotility, regardless of age, but were a feature of 4/104 patients with diverse severe gastrointestinal motility disorders. In contrast to dogs, polyglucosan bodies are not a feature of normal ageing in human gastrointestinal smooth muscle but, in accord with previous suggestions, are seen in rare cases of human gut dysmotility. The significance of this difference is unclear.

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Section: Discussionsupporting

confidence: 79%

Section: Introductionsupporting

confidence: 80%

A systematic analysis of polyglucosan bodies in the human gastrointestinal tract in health and disease

et al. 2003

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“…There are multiple known conditions resulting in chronic intestinal pseudoobstruction (CIP) [1][2][3][4][5][6]. Pseudoobstruction implies a pathologic state with signs and symptoms of bowel obstruction without a mechanical cause.…”

Section: Introductionmentioning

confidence: 99%

“…Regardless of the inheritance pattern, VM is overwhelmingly attributed to an atrophic muscularis propria. The atrophy is variable but described as a marked vacuolar degeneration of myocytes, loss of muscle fibers [6], and with or without a highly characteristic honeycomb fibrosis [1][2][3][4][5]. The recognition of the atrophic pattern of injury is the chief diagnostic clue that a visceral myopathy is likely and other causes of CIP are unlikely.…”

Section: Introductionmentioning

confidence: 99%

“…A hypertrophic pattern in VM, dominating the atrophic pattern, is exceedingly rare. As a pattern of injury, hypertrophy is almost absent from the description of visceral myopathy in much of the literature [1,2,[6][7][8][9][10][11] or is mentioned only as a minor feature [2,5]. Much of literature on CIP and VM is contained within the clinical and surgical medical specialties and is less well represented in anatomical pathology, and it is possible that the hypertrophic patterns are underreported.…”

Section: Introductionmentioning

confidence: 99%

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Congenital visceral myopathy with a predominantly hypertrophic pattern treated by multivisceral transplantation

Koh¹,

Bradley²,

French³

et al. 2008

Human Pathology

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Smooth muscle inclusion bodies in slow transit constipation

et al. 2001

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Slow transit constipation (STC) is a disorder of intestinal motility of unknown aetiology. Myopathies, including those characterized by the finding of inclusion bodies, have been described in enteric disorders. Amphophilic inclusion bodies have been reported in the muscularis externa of the colon of STC patients. This study formally tested the hypothesis that these represent a primary muscle disorder, specific to STC. In a systematic, blinded, dual observer qualitative and quantitative analysis, colonic and ileal tissue from patients with STC (n=36) were compared with selected control populations: total colonic aganglionosis (n=10), Chagas' disease (n=6), isolated rectal evacuation disorders (n=6), and a control population of a range of ages (n=80). All sections were stained with haematoxylin and eosin and periodic acid Schiff. Further immunostains were used in an attempt to determine inclusion body composition. Round or ovoid (4-22 microm diameter) amphophilic inclusions increased in number in normal subjects with age. Inclusions were more frequent in idiopathic STC than in age-matched controls or rectal evacuation disorders [ileum (33% vs. 9%), ascending (50% vs. 19%, p<0.05), and sigmoid colon (43% vs. 20%)] and were very frequent in the sigmoid (71%) of patients with STC arising after pelvic surgery. The number of inclusions per unit area was significantly higher in patients with STC (p<0.001). Inclusions were found in all Chagas' patients, but not with aganglionosis. It was not possible to determine inclusion body composition, despite the use of a wide range of conventional and immunostains. This study demonstrates that inclusion body myopathy is identifiable in patients with STC and that it may arise secondary to denervation.

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Sporadic Viscoral Myopathy with Inclusion Bodies

Cited by 21 publications

References 0 publications

A systematic analysis of polyglucosan bodies in the human gastrointestinal tract in health and disease

A systematic analysis of polyglucosan bodies in the human gastrointestinal tract in health and disease

Congenital visceral myopathy with a predominantly hypertrophic pattern treated by multivisceral transplantation

Smooth muscle inclusion bodies in slow transit constipation

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