Segmental muscular defects of the intestine: a possible cause of spontaneous perforation of the bowel in adults

Tamai, Masamitsu; Satoh, Michiko; Tsujimoto, Akio

doi:10.1016/j.humpath.2013.07.004

Cited by 18 publications

(23 citation statements)

References 26 publications

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“…In 1997, Darcha et al described the first case of SAIM in adults in a 64-year-old female, who had iatrogenic sigmoid colon perforation during endoscopic removal of a rectal polyp [ 1 ]. A study published by Tamai et al described seven cases of SAIM in a retrospective study involving patients who presented with spontaneous intestinal perforation [ 2 ]. The true incidence of SAIM is not known, as this entity is segmental and the diagnosis requires a high index of suspicion.…”

Section: Discussionmentioning

confidence: 99%

“…No loss of mucosa, submucosa, and serosa is associated with this condition. Inflammation and mesenteric vessel thrombosis are unusual [ 2 – 6 ].…”

Section: Discussionmentioning

confidence: 99%

“…However, no recurrences of intestinal perforation have been reported. Mortality was reported in only one case, in which the patient died two days postoperatively as a result of pulmonary edema [ 2 ].…”

Section: Discussionmentioning

confidence: 99%

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Segmental Absence of Intestinal Musculature in a 64-Year-Old Female: Case Report and Literature Review

Nawar

Sawyer

2016

Am J Case Rep

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Patient: Female, 64Final Diagnosis: Segmental absence of intestinal musculatureSymptoms: Abdominal discomfortMedication: —Clinical Procedure: ColectomySpecialty: Diagnostics, LaboratoryObjective:Rare diseaseBackground:Segmental absence of intestinal musculature is a well described entity in premature infants. It presents with peritonitis, bowel perforation, and obstruction. The diagnosis is based on pathologic observation of absence of intestinal musculature. Researchers hypothesized that this entity is a result of a vascular accident during embryogenesis. However, segmental absence of intestinal musculature is no longer limited to the pediatric population. Recently, a few cases have been described in adults with and without significant vascular diseases. This change in the age of the affected population with segmental absence of intestinal musculature makes the understanding of the pathogenesis of this entity even more challenging.Case Report:Here, we report a case of segmental absence of intestinal musculature in a 64-year-old female. The patient presented to the emergency room with sudden onset of abdominal pain and signs of peritonitis. Abdominal computed tomography showed free air in the abdomen. Laparotomy was performed, and a perforation involving the descending colon was identified. Left hemicolectomy was performed. Pathologic examination of the resected colon showed segmental absence of intestinal musculature.Conclusions:Although the pathologic diagnosis of segmental absence of intestinal musculature is straightforward, the assumption that this condition is limited to the pediatric population is a major player in overlooking this diagnosis in adults. Pathologists should be aware that this condition can present in adults and is segmental. Gross and microscopic examination of perforated intestine is required to reach the correct diagnosis. To our knowledge, twelve cases of this entity have been described in adults. Here we present the thirteenth case of segmental absence of intestinal musculature in an adult, and we discuss the clinical and pathologic findings of this entity as well as its pathogenesis.

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Section: Discussionmentioning

confidence: 99%

“…No loss of mucosa, submucosa, and serosa is associated with this condition. Inflammation and mesenteric vessel thrombosis are unusual [ 2 – 6 ].…”

Section: Discussionmentioning

confidence: 99%

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Segmental Absence of Intestinal Musculature in a 64-Year-Old Female: Case Report and Literature Review

Nawar

Sawyer

2016

Am J Case Rep

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“…Histolojik olarak perforasyon çevresinde segmental müskülaris propria defektleri saptanır. Erişkinlerde etiyoloji belirsizdir, fokal konjenital anomali neden olabilir (26). Ehlers-Danlos sendromu da tüm vücutta kollojen fiberleri etkileyen konjenital bir defekttir ve spontan perforasyon ve massif kanama gibi ciddi intestinal komplikasyonular ile karşımıza çıkabilir (12).…”

Section: Segmenter Müsküler Distrofide Histomorfolojiunclassified

Kolon perforasyonunda histomorfolojik tanı güçlükleri

Baş

Şahin²

2018

Journal of Health Sciences and Medicine

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Bu derlemede, kolon perforasyonu saptanan ve parsiyel/ total kolektomi uygulanan olgularda histomorfolojik tanı güçlükleri değerlendirildi. Patoloji pratiğinde klinik bilgiler dâhilinde kolon perforasyonunun histomorfolojik bulgularını rapor etmek güç olmamakla birlikte bazı vakalarda karakteristik bulguların olmayışı tanı güçlükleri doğurabilmektedir. Her ne kadar histolojik tanı için klinik bilgiler çoğu zaman yeterli olsa da, Crohn, amibik kolit ve intestinal Behçet hastalığı gibi vakalarda ayırıcı tanı güçlükleri patoloji pratiğinde yaşanmaktadır.

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“…Since then, several reports about infants and neonates with various clinical manifestations such as intestinal obstruction, volvulus, intussusception, and perforation have been published worldwide [ 2 ]. The first adult case of SAMP manifesting as colonic perforation was published in 1997 [ 3 ] and several have been reported since then [ 4 5 6 7 8 9 10 ].…”

Section: Introductionmentioning

confidence: 99%

Spontaneous colonic rupture related to the segmental absence of muscularis propria in an adult

Tseng¹,

Lin

Huang³

et al. 2018

Tzu Chi Med J

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Colonic perforation is a medical emergency that may be fatal if surgery cannot be performed in a timely manner. Colonic rupture in adults is caused by primary (idiopathic) and secondary factors. Although the segmental absence of muscularis propria (SAMP) is a recognized cause of secondary colonic rupture in neonates and infants, few cases have been reported in adults. Here, we present the case of a large colonic rupture caused by SAMP in a 60-year-old woman and a review of the literature. We suggest that SAMP should be included in the differential diagnosis of large perforation and/or periperforation membranous thinning of the colonic wall in adults.

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Segmental muscular defects of the intestine: a possible cause of spontaneous perforation of the bowel in adults

Cited by 18 publications

References 26 publications

Segmental Absence of Intestinal Musculature in a 64-Year-Old Female: Case Report and Literature Review

Segmental Absence of Intestinal Musculature in a 64-Year-Old Female: Case Report and Literature Review

Kolon perforasyonunda histomorfolojik tanı güçlükleri

Spontaneous colonic rupture related to the segmental absence of muscularis propria in an adult

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