Gastric Volvulus Following Left Pneumonectomy in an Adolescent Patient

Abstract

Gastric volvulus is a rare post-pneumonectomy complication. Although it has been described previously, published cases are limited to an older patient population. We report the youngest case of postpneumonectomy gastric volvulus to date, occurring in an 18-year-old male with a history of inflammatory myofibroblastic pseudotumor who underwent left intrapericardial pneumonectomy, and presented 13 years later with chronic intermittent mesenteroaxial gastric volvulus. While postpneumonectomy gastric volvulus is a rare occurrence, it should remain in the differential diagnosis in postoperative thoracic surgical patients presenting with chest pain.

1. Introduction

Gastric volvulus is uncommon among pediatric and adolescent patients [1–4], and is even more rare as a sequela of pneumonectomy, even when broadening the population to include adults. Volvulus of the stomach is defined as twisting or turning of the stomach over 180° and can be organoaxial, mesenteroaxial, or a combination of the two, depending on the axis of rotation of the stomach (Figure 1) [5, 6]. Gastric volvulus has been associated with diaphragmatic eventration, congenital diaphragmatic hernia, intestinal malrotation, wandering spleen, asplenism, and hiatal hernia [5]. Although gastric volvulus following pneumonectomy has been described in the literature, the reports are limited to patients over 30 years old [7–11]. This report describes the history, treatment, and outcome of an adolescent patient who presented with mesenteroaxial gastric volvulus 13 years after undergoing left pneumonectomy.

Figure 1.

Organoaxial volvulus (left) occurs when the stomach rotates along its long axis, placing the greater curvature anteriorly and the lesser curvature posteriorly. Mesenteroaxial volvulus (right) occurs when the stomach rotates along its short axis placing the antrum anteriorly and superiorly. Figure courtesy of Memorial Sloan Kettering Cancer Center. Used with permission.

2. Case report

Our patient is an 18-year-old male with a history of inflammatory myofibroblastic tumor (IMT) that was diagnosed and surgically treated during childhood. At 5 years of age, the patient presented with left-sided pain and nausea and was seen by his pediatrician who noted diminished breath sounds on the left. A chest X-ray revealed a mass, and a confirmatory computed tomography (CT) scan showed a 7-cm mediastinal mass occupying the upper third of the left hemithorax. The mass was subsequently excised through a left thoracotomy, and pathologic analysis supported a diagnosis of IMT. On follow-up imaging, a 2.7 × 2 cm left upper-lobe density was found and the patient was referred to our institution for further care. After consultation with our institution’s tumor board, the patient underwent a thoracotomy and left intrapericardial pneumonectomy with placement of a McGhan tissue expander, initially inflated to 300 cc with saline. Follow-up magnetic resonance imaging (MRI) showed no evidence of recurrent disease.

Thirteen years later, the patient presented to an outside emergency department with left-sided chest pain. The patient had an extensive workup of his pain, including blood work, a cardiac workup, and CT of the chest, all of which were initially unremarkable. The patient’s chest pain recurred, at which time he was referred to our institution. Upon review of the outside CT scan at our institution, the diagnosis of mesenteroaxial gastric volvulus was suspected. An upper gastrointestinal imaging series was performed and revealed mesenteroaxial position of the stomach without obstruction (Figure 2). Additionally, an abdominal ultrasound showed cholelithiasis. The patient was prepared for surgery during which a laparoscopic gastropexy, Stamm gastrostomy, and cholecystectomy were performed.

Figure 2.

(A) Reformatted coronal CT image shows elevation of the left hemidiaphragm. The gastric outlet (arrowheads) is cephalad to the esophagogastric junction (arrow), consistent with mesenteroaxial volvulus. (B) Upper gastrointestinal radiographic series confirms mesenteroaxial volvulus with gastric outlet (arrowheads) cephalad to the esophagogastric junction (arrow).

At the time of laparoscopy, the stomach was located in the left upper quadrant due to the high eventration of the left hemidiaphragm secondary to prior left pneumonectomy. The stomach was easily reduced and no hiatal hernia was found. A gastropexy was carried out by suturing the lesser curvature of the stomach to the round ligament, and a lateral Stamm gastrostomy high on the greater curvature of the stomach was performed. Intraoperative esophagogastroduodenoscopy showed a normal esophagus without evidence of reflux or inflammation. The patient tolerated the procedure well, but suffered postoperative left chest and axillary pain, for which he has been followed by neurology and pain teams. Postoperative clinical assessments were undertaken by the surgeon, who removed the gastrostomy tube and has noted no further evidence of gastric volvulus. No subsequent surgeries have been attempted to remove the deflated tissue expander.

3. Discussion

Gastric volvulus is an uncommon diagnosis as the stomach is held in place by multiple structures, including its attachments to the fixed esophagus and duodenum, the gastrophrenic and gastrosplenic ligaments, the short gastric vessels, and the gastrocolic ligament. Cases of gastric volvulus in pediatric and adolescent populations are even more uncommon as illustrated in a review by Cribbs et al. in which 581 cases of gastric volvulus in infants and children were identified over a 78-year period [5]. Regardless of age, the most common type of gastric volvulus described is organoaxial volvulus; however, mesenteroaxial or a combination of the two is also possible [2, 6, 10, 12]. Gastric volvulus is commonly seen secondary to hiatal hernia, laxity or absence of gastric ligaments, postoperative adhesions, congenital and acquired diaphragmatic defects, and anomalies of adjacent organs [5, 10, 13]. Intrathoracic and intra-abdominal gastric volvulus have both been seen; however, they are usually secondary to varied pathologies. Intrathoracic herniation of the stomach followed by volvulus has been described with patients who have hiatal hernia [6, 9]. However, in the absence of hiatal hernia, gastric volvulus in the abdominal cavity is more likely and may be the result of phrenic nerve dissection and/or elevation of the left hemidiaphragm subsequently moving the tethered fundus of the stomach via its attachment to the gastrosplenic ligament [8, 11].

While multiple instances of gastric volvulus from these various causes have been observed, to our knowledge, only seven published cases (in English) of gastric volvulus have been identified after pulmonary resection (see Table) [6–11, 14]. The median age of these patients was 61 years (range, 33–75 years) with a median time between pneumonectomy and presentation of gastric volvulus of 1 year (range, 1 day – 33 years). The majority of these case reports describe recognition of gastric volvulus and prompt surgical correction without complication; however, a delay of surgery can result in progression to devastating morbidities including gastric ischemia, small bowel ischemia, return visits to the operating room, and mortality in some cases [8, 11].

Table.

Patient characteristics in published reports of postpneumonectomy gastric volvulus.

Author/Year	Sex	Age	Time From Pneumonectomy	Laterality of Pneumonectomy	Thorax/Abdomen	Volvulus Type	Hiatal Hernia	Management
Young 1957 [14]	F	33	1 year	Left	Abdomen	Not described	Unknown	Gastrostomy
Creedon 1965 [11]	F	38	7 years	Bilateral partial	Abdomen	Organoaxial	No	Gastrostomy, gastropexy
Carlisle 1967 [7]	M	65	2 years	Left	Abdomen	Mesenteroaxial	No	Gastropexy
Simoens 1994 [10]	M	75	17 days	Left	Thorax	Not described	Yes	Gastropexy & hiatal hernia repair
Blum 2006 [9]	M	54	1 day	Left	Thorax	Not described	Yes	Hernia reduction, Nissen repair
Batirel 2007 [6]	M	61	33 years	Left	Abdomen	Mesenteroaxial	No	Laparoscopic Toupet fundoplication and gastropexy
Thorpe 2007 [8]	F	69	5 months	Left	Thorax	Not described	Yes	Trans-mesocolic gastrojejunostomy, hiatal hernia repair

Upon recognition of the chronic intermittent gastric volvulus, our patient was admitted to the hospital for further workup, monitoring and preparation for surgery. Gastropexy and gastrostomy, along with possible Nissen fundoplication, were considered as possible corrective measures. Gastropexy alone has been described previously for the treatment of gastric volvulus in pediatric patients [1, 3, 5]. Ultimately, our decision was to treat the gastric volvulus with gastropexy and Stamm gastrostomy. Intraoperative assessment revealed a normal esophagogastric junction along with a good intra-abdominal segment of the esophagus; therefore, a Nissen fundoplication was not deemed necessary. With no history of gastroesophageal reflux disease, the patient was a suitable candidate for gastropexy, and because he had a history of difficulty in gaining weight, the gastrostomy tube placement would allow the possibility of supplemental alimentation.

To identify the etiology of the gastric volvulus 13 years after pneumonectomy, the CT scan was evaluated again; this secondary review revealed apparent deflation of the intrathoracic tissue expander that had been placed during the intrapericardial left pneumonectomy as a prophylactic measure against post-pneumonectomy syndrome (PPS) (Figure 3) [15, 16]. Recognition of severe mediastinal shift after pneumonectomy leading to PPS has been described in pediatric patients, who are hypothesized to be at greater risk due to greater tissue elasticity [16]. While our patient’s preoperative CT showed no evidence of PPS, the deflation of the tissue expander increased the amount of unoccupied space that an intact lung would have otherwise filled. Thus, deflation of the tissue expander may have contributed to the higher elevation of the left hemidiaphragm and subsequent pulling of the gastrosplenic ligament, which ultimately created the circumstances in which gastric volvulus could develop [8]. The presence of an intrathoracic prosthetic device should prompt clinicians to include gastric volvulus in their differential diagnosis when symptoms of chest or abdominal pain occur. As our patient’s pathology was identified with the assistance of CT imaging, these patients may require modification of standard CT scan protocols to include more proximal images into the lung bases, or distal images into the abdomen, to reveal radiologic evidence of gastric volvulus. Furthermore, evidence of deflation and/or rupture of these prosthetic devices on follow-up imaging may provide earlier clues to the occurrence of this pathology.

Figure 3.

Postpneumonectomy axial CT images of intrathoracic tissue expander (A) Tissue expander (asterisk) is inflated with no evidence of mediastinal shift. (B) Partially calcified tissue expander (arrowheads) is deflated resulting in elevation of the left hemidiaphragm and possibility of gastric volvulus.

4. Conclusion

Gastric volvulus is an uncommon sequela of left pneumonectomy, but should remain part of the differential diagnosis for any patient presenting with abdominal or chest pain and who has undergone a prior lung resection. Appropriate management is prompt surgical correction after recognition, as the consequences are potentially catastrophic, as seen in older patients.

Highlights.

• Gastric volvulus, rare in pediatric patients, is a possible sequela of pneumonectomy

• This diagnosis must be considered in patients with chest pain and prior pneumonectomy

• Diagnosis of gastric volvulus may require CT; prompt surgical correction is essential