Idiopathic megacolon complicated by life-threatening giant megacolon and respiratory failure due to diaphragmatic eventration: A case report

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Abstract

Introduction and importance

Giant megacolon requiring emergency surgery is rare. Eventration of the diaphragm associated with giant megacolon is also uncommon.

Case presentation

We report a 66-year-old male who presented with abdominal distention and progressive dyspnea. After resuscitation following cardiopulmonary arrest, the patient underwent emergent subtotal abdominal colectomy. Eventration of the diaphragm was found postoperatively and his respiratory condition was insufficient to allow liberation. Plication of both diaphragms was performed through left and right thoracotomy via the 8th intercostal space. Postoperatively the patient made a full recovery.

Clinical discussions

Chronic constipation is a common health condition. A life-threatening condition secondary to chronic constipation is a rarely documented complication. Diaphragmatic eventration that was caused due to chronic megacolon in symptomatic patients requires surgical treatment.

Conclusions

We describe a patient with giant megacolon and diaphragmatic eventration secondary to idiopathic megacolon. The patient underwent subtotal colectomy and diaphragmatic plication and recovered fully.

Highlights

•

A life-threatening condition secondary to chronic megacolon is extremely rare.
•

Abdominal compartment syndrome due to giant megacolon resolved by subtotal abdominal colectomy.
•

Chronic megacolon from childhood induced eventration of the diaphragm.
•

Plication of both diaphragms recovered fully the patient from respiratory dysfunction.

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Most cited references 14

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The SCARE 2020 Guideline: Updating Consensus Surgical CAse REport (SCARE) Guidelines

Riaz Agha, Thomas Franchi, Catrin Sohrabi … (2020)

The SCARE Guidelines were first published in 2016 and were last updated in 2018. They provide a structure for reporting surgical case reports and are used and endorsed by authors, journal editors and reviewers, in order to increase robustness and transparency in reporting surgical cases. They must be kept up to date in order to drive forwards reporting quality. As such, we have updated these guidelines via a DELPHI consensus exercise.

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The enteric nervous system in chagasic and idiopathic megacolon.

Gabrio Bassotti, Z Kogan, R Bilder … (2007)

Chagas disease frequently causes megacolon. We investigated the enteric nervous systems in patients with chagasic megacolon compared to idiopathic megacolon and controls. Surgical specimens were obtained from 12 patients with chagasic megacolon (1 woman, 11 men, age range 41 to 72 y) and 9 patients with idiopathic megacolon (3 women, 6 men, age range 39 to 68 y), undergoing surgery for intractable constipation. A control group of 10 patients (9 women, 1 man, age range 43 to 75 y) undergoing left hemicolectomy for nonobstructing colorectal cancer was also studied. Colonic sections were investigated by conventional and immunohistochemical methods, also taking into consideration the presence of lymphocytes. Compared to controls, the 2 megacolon groups showed a decrease of enteric neurons (not due to increased apoptosis) and of enteric glial cells (all more important in chagasic patients). The interstitial cells of Cajal subtypes were decreased but not absent in megacolons, although an increase of the intramuscular subtype was found, suggesting a possible compensative mechanism. An increased amount of fibrosis was found in the smooth muscle and the myenteric plexus of chagasic patients compared to the idiopathic megacolon and the control group. A mild lymphocytic infiltration of the enteric plexuses (more evident in Chagas disease) was also found in megacolons but not in controls. Patients with chagasic megacolon display important abnormalities of several components of the enteric nervous system. Similar alterations, although of lesser severity, may be found in patients with idiopathic megacolon.

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Clinical features of idiopathic megarectum and idiopathic megacolon.

M. Kamm, J Gattuso (1997)

Dilatation of the rectum and/or colon, in the absence of demonstrable organic disease, is an uncommon and poorly characterised condition. To characterise the clinical and diagnostic features, and response to treatment, of patients with idiopathic megarectum (IMR) and idiopathic megacolon (IMC). A retrospective review was undertaken of all patients operated on for these conditions over a 23 year period. In addition all patients treated over a three year period were prospectively studied by means of a questionnaire, contrast studies of the upper and lower intestine, spine x rays to exclude spinal dysraphism, anorectal physiological studies, and assessment of clinical outcome. Patients with Hirschsprung's disease and other known causes of gut dilatation were excluded. (i) Retrospective study: Of 63 operated patients, 22 had IMR, 23 had IMR and IMC, and 18 had IMC only. Five patients with IMC had previous sigmoid volvulus, and three had associated non-gastrointestinal congenital abnormalities. Faecal incontinence was always associated with rectal impaction and 14 patients (82%) with IMR alone had had manual disimpaction. (ii) Prospective study: Twenty two patients had IMR, with a median rectal diameter of 10 cm (normal < 6.5 cm). Six patients had IMC and one patient had IMR and IMC. Patients with IMR were significantly (p = 0.0007) younger than patients with IMC. All patients with IMR became symptomatic in childhood, compared with half the patients with IMC who developed symptoms as adults. Patients with IMR all presented with soiling and impaction, compared with patients with IMC whose symptoms were variable and included constipation or increased bowel frequency, pain, and variable need for laxatives. No upper gut dilatation was seen in either group of patients. Spinal dysraphism was seen in two of 18 patients with IMR and two of four with IMC, suggesting extrinsic denervation as a possible cause in a minority. Twelve of 22 patients with IMR had a maximum anal resting pressure below normal, indicating sphincter damage or inhibition. Both IMR and IMC patients had altered rectal sensitivity to distension, suggesting that despite lack of dilatation the rectum in IMC has altered viscoelasticity, tone, or sensory function. Fifteen of 22 patients with IMR were successfully managed with laxatives or enemas, but seven required surgery. Two of seven patients with IMC required surgery, including one for sigmoid volvulus. Patients with IMR differ clinically, diagnostically, and in their outcome from patients with IMC. These conditions demand specific investigation, and intensive treatment, to achieve optimum care.

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Author and article information

Contributors

Ai Sadatomo

Journal

Journal ID (nlm-ta): Int J Surg Case Rep

Journal ID (iso-abbrev): Int J Surg Case Rep

Title: International Journal of Surgery Case Reports

Publisher: Elsevier

ISSN (Electronic): 2210-2612

Publication date PMC-release: 30 June 2022

Publication date Collection: July 2022

Publication date (Electronic): 30 June 2022

Volume: 96

Electronic Location Identifier: 107372

Affiliations

[a ]Department of Surgery, Division of Gastroenterological, General and Transplant Surgery, Jichi Medical University, 3311 Yakushiji, Shimotsuke, Tochigi 329-0498, Japan

[b ]Division of General Thoracic Surgery, Department of Surgery, Jichi Medical University, 3311 Yakushiji, Shimotsuke, Tochigi 329-0498, Japan

Author notes

[* ]Corresponding author at: Department of Surgery, Jichi Medical University, 3311-1, Yakushiji, Shimotsuke, Tochigi 329-0498, Japan. r0626as@ 123456jichi.ac.jp

Article

Publisher Item ID: S2210-2612(22)00618-6 Publisher ID: 107372

DOI: 10.1016/j.ijscr.2022.107372

PMC ID: 9284071

PubMed ID: 35809478

SO-VID: 6df99c58-d69d-4cb7-9a6b-d3eccd01d06e

License:

This is an open access article under the CC BY license (http://creativecommons.org/licenses/by/4.0/).

History

Date received : 4 May 2022

Date revision received : 26 June 2022

Date accepted : 26 June 2022

Idiopathic megacolon complicated by life-threatening giant megacolon and respiratory failure due to diaphragmatic eventration: A case report

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Abstract

Introduction and importance

Case presentation

Clinical discussions

Conclusions

Highlights

Related collections

European Respiratory Society: COVID-19

Most cited references 14

The SCARE 2020 Guideline: Updating Consensus Surgical CAse REport (SCARE) Guidelines

The enteric nervous system in chagasic and idiopathic megacolon.

Clinical features of idiopathic megarectum and idiopathic megacolon.

Author and article information

Contributors

Journal

Affiliations

Author notes

Article

History

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