Initial case of spontaneous gastrocutaneous fistula following nonsteroidal antiinflammatory drug treatment: a case report

Abstract

Background

Spontaneous fistulization of a gastric perforation to the skin is an extremely rare condition whose pathophysiology and etiology remain difficult to explain, especially since histological examination did not find any cancer cells.

Case presentation

A-45 year-old male of Black African descent, residing in a rural area, was admitted to the General Reference Hospital with a spontaneous gastrocutaneous fistula. The history revealed the intake of nonsteroidal anti-¡inflammatory drugs for pain from a chronic foot wound. After imaging exploration (ultrasound, then computed tomography scan) proved noncontributory, an exploratory laparotomy confirmed a crater on the anterior surface of the gastric antrum leading to the skin, resembling a surgical gastrostomy; there was no mass or sign of peritoneal carcinosis. An atypical gastrectomy confirmed the benign clinical picture.

Conclusion

The interest of this observation lies in its unusual character and encourages etiopathogenic research through the literature.

Introduction

Gastric perforation in general is responsible for generalized acute peritonitis; it is a surgical emergency and remains very common in our regions [1]. The most common etiology is perforation of a gastroduodenal ulcer due to the proliferation of Helicobacter pylori (HP) or uncontrolled intake of antiinflammatory drugs [2]. Underlying gastric cancer remains a major concern for surgeons in this context. However, the gastrocutaneous fistula without causing peritonitis remains an exceptional, atypical, and unusual phenomenon; no case is found in the literature. We report a rare case of gastric perforation after taking a nonsteroidal antiinflammatory drug, which fistulated to the skin in a young patient referred to the Niamey General Reference Hospital in a cachectic state. Gastric fistulization is diagnosed clinically by the direct passage of unprocessed food to the skin. The pathophysiology of this skin fistulization remains difficult to explain. Histological examination did not find any cancer cells. The objective of this case report is to conduct a review of the literature that will allow us to search for similar clinical cases and explain the pathophysiology of this phenomenon.

Case presentation

We report a 45-year-old patient of Black African origin, referred from an integrated health center hundreds of kilometers away to the GRH for a complicated abdominal wound with a digestive fistula.

On questioning, the onset was about a week earlier after taking a nonsteroidal antiinflammatory for pain from a chronic wound on the foot. This was followed by the onset of painful epigastric swelling, which eventually abscessed and fistulated to the skin 72 hours later. The meal was undigested and came out of an epigastric wound with pus. He had no pain in the other abdominal quadrants. There was no medical–surgical or family history.

The clinical examination showed a patient in poor general condition [stage III of the World Health Organization (WHO)], a body mass index (BMI) of 17.64 kg/m², and blood pressure of 100/60 mmHg. Epigastric pain was noted, no vomiting, and no melena or rectorrhagia was reported; bowel transit was preserved with passage of gas. Physical examination showed a flat abdomen and an epigastric perforation with sharp edges about 2 cm in diameter that produced pus and gastric fluid mixed with unprocessed food (Fig. 1). There were skin burn lesions around the perforation. The abdomen was soft in the flanks and pelvis; there was no palpable mass. Rectal examination was unremarkable. A colostomy bag, difficult to fit due to skin lesions, collected about 1 L of greenish gastric fluid mixed with pus and food debris. The patient was fasted, and biological testing noted a microcytic normochromic anemia with 10.1 g/dL of hemoglobin, hyperleukocytosis at 12,200/µL, and no ionic disorder or impaired renal function. Transaminases were not unusual. The ultrasound and then the contrast-enhanced abdominopelvic computed tomography (CT) were normal: no abdominal mass or intraabdominal fluid effusion. The chest X-ray was normal.

Fig. 1 .

Skin fistula at the epigastric region (red arrow) producing undigested digestive fluid; note skin burn lesions

The patient was hospitalized and received parenteral feeding and rehydration, antibiotic therapy with C3G, and proton pump inhibitors. A pre-anesthetic consultation classified the patient as ASA II after 48 hours of resuscitation. Exploratory laparotomy was indicated for suspected gastric perforation fistulated after misuse of a nonsteroidal antiinflammatory drug. Surgical exploration noted that the skin perforation communicated with the gastric mucosa; no communication was noted with the abdominal cavity (Fig. 2A), and there were no signs of peritonitis. After complete release of the perforation edges, it was noted that it affected the distal part of the stomach (Fig. 2B) with irregular inflammatory margins. The liver was smooth, and the rest of the exploration was unremarkable. An atypical gastrectomy (Fig. 3) was performed, and a jejunal loop mounted in Y, 60 cm from the duodenojejunal angle, restored digestive continuity. The postoperative course was uneventful. Oral refeeding was started with fluids on the third day after surgery. Histological examination of the specimen showed a gastric wall remodeled by scar tissue in some areas and in others a largely ulcerated area with significant inflammatory changes. There was no evidence of HP or malignancy.

Fig. 2.

A Beginning of skin perforation release (green arrow) showing below the abdominal cavity (blue arrow). B Total release of the stomach (yellow arrow) and coloploid detachment showing transverse colon (red arrow), before gastric stapling

Fig. 3 .

Atypical gastrectomy specimen showing the perforation (green arrow)

A contrast-enhanced thoracoabdominopelvic CT scan (Fig. 4) performed 1 month postoperatively was normal. The patient was discharged and showed a very favorable clinical outcome, with a normal diet and weight gain at the 1-month follow-up.

Fig. 4 .

Axial abdominal computed tomography scan performed 1 month postoperatively; the white arrow indicates the stomach with a normal wall appearance

Discussion

We report an atypical case of gastric perforation fistulated to the abdominal wall without causing peritonitis after nonsteroidal antiinflammatory drugs taken by a 45-year-old patient. This clinical finding remains an extremely rare phenomenon. The pathophysiology of this parietal fistulization also remains a very rare clinical context. The literature review was poor. Surgical exploration was the only appropriate therapy and allowed for a gastrectomy. Histological examination showed chronic ulcerated gastritis without HP or malignancy. The interest of this study is to search through the literature for similar cases and to understand the physiopathological mechanism of direct fistulization to the wall. However, the bibliographical search remains fruitless. However, spontaneous gastric perforation is common in adults, and the main etiology reported is perforation of ulcerative disease most often associated with HP infection, tobacco, alcohol, and/or antiinflammatory drug use [1]. It is becoming increasingly common in sub-Saharan Africa due to poor nutrition and the intake of high-dose, uncontrolled antiinflammatory drugs available to the population from street vendors [2]. Toxicity of antiinflammatory drugs to the gastric and duodenal mucosa is common and responsible for complications such as perforation [3]. The direct contact of antiinflammatory drugs with the gastric mucosa appears to be an important factor in the pathogenic process; it results in intestinal permeability disorders and secondary inflammation. The breakdown of the mucous barrier is mainly due to the inhibition of the enzyme cyclo-oxygenase II [4]. This enzyme is responsible for the synthesis of prostaglandins involved in many physiological functions, including protection of the gastric mucosa [5]. Studies in healthy volunteers showed that increased permeability appeared rapidly within 12 hours of antiinflammatory drug administration [5]. For people who have to take antiinflammatories on a chronic basis, gastrointestinal protection is recommended with proton pump inhibitor (PPI) and antihistamine 2 (anti-H2) [2]. Perforation of gastric tumors is also reported but is less frequent and poorly documented in Africa, at 3.3% according to Dembélé et al. [6]. Some phenomena, such as gastric perforation by ingested fish bone complicated by pyogenic hepatic abscess, have been reported by Ayonga et al. [7]. However, fistulization of a gastric perforation to the wall without creating peritonitis after ingestion of an antiinflammatory is little described in the literature.

Several theories have been mentioned to explain this phenomenon: the first is the ingestion of a foreign body, the underlying presence of a gastric tumor, or a chronic intestinal inflammatory disease such as Crohn’s disease, but the most likely pathophysiological explanation we can give is that the stomach, affected by an ongoing ulcerative process with a Helicobacter pylori infection, became adherent to the abdominal wall and then developed fistulization toward the skin. The clinical and histological examination of the surgical specimen (with rereading) ruled out all these hypotheses. The diagnosis remains gastric perforation from taking a nonsteroidal antiinflammatory, whose nature undoubtedly avoided acute peritonitis by fistulizing the infection to the wall. The patient was living at the time of illness in an area far from hospitals capable of managing him urgently.

Limitations of the study

Impossibility of performing an intraoperative frozen section.

Conclusion

We report an unusual case, which was the first of a gastric perforation due to antiinflammatory drugs, fistulated to the skin by a natural phenomenon. This fistulization undoubtedly saved the life of the patient, who was several kilometers from a hospital with an operating room and a surgeon. The treatment was a gastrectomy.

Abbreviations

HP

Helicobacter pylori

GRH

General Reference Hospital

WHO

World Health Organization

BMI

Body mass index

CT

Computed tomography

C3G

Third-generation cephalosporin

ASA

American Society of Anesthesiologists

PPI

Proton pump inhibitor

Anti-H2

Antihistamine 2

Author contributions

AS, YBT, ABMLA, AZO, and IK participated in literature search and manuscript drafting. ABMLA participated in the anatomical and pathological study of the surgical room. RS participated in the correction of the manuscript. All authors read and approved the final version of the manuscript.

Funding

None.

Data availability

All data generated or analyzed during this study are included in this article.

Declarations

Ethics approval and consent to participate

Not applicable.

Consent for publication

Written informed consent was obtained from the patient for the publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

Competing interests

The authors declare that they have no competing interests.