Inflammatory Pseudotumor in an Irreducible Inguinal Hernia

Citation: Ayabilah EA, Hagan R, Morna M, Akoh JA (2018) Inflammatory Pseudotumor in an Irreducible Inguinal Hernia. Int J Surg Res Pract 5:089. doi.org/10.23937/2378-3397/1410089 Accepted: November 24, 2018 ; Published: November 26, 2018 Copyright: © 2018 Ayabilah EA, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. International Journal of Surgery Research and Practice Open Access ISSN: 2378-3397


Background
Inflammatory pseudotumor (IPT) is a benign lesion that affects virtually every anatomic site, mostly the lung but gastrointestinal tract rarely.IPT is a rare entity with few retrospective reviews in the paediatric population and small series report in the adult tissue adjacent to but not infiltrating the wall of the bowel (Figure 3).It was a well demarcated fleshy lesion measuring 7 cm across with central cystic/myxoid degenerareas with intense chronic inflammatory changes.The hypocellular areas show stellate cells and wavy spindle cells admixed with foamy macrophages and cells showing signet ring configuration.The densely collagenized areas show intense chronic inflammation with numerous medium sized vessels.There were no mitotic figures and no evidence of malignancy.Changes were reported to be consistent with an IPT.

Discussion
The clinical presentation of IPT is mostly nonspecific but depends on the site involved and relation to neighbouring structures.For example, IPT involving the pulmonary system presents with dyspnoea, pain, fever and cough while intraabdominal or retroperitoneal tumors present with abdominal pain, change in bowel movement, weight loss, symptoms of obstruction and as palpable masses.Furthermore, gastrointestinal IPTs may have features suggestive of malignancy, including ulceration, and infiltration of the wall.Our case presented with insignificant weight loss, mild anaemia and inability to reduce the groin swelling.Though adherent to the small bowel, the tumour did not infiltrate its wall.IPT presenting in an inguinal hernia as in this case is extremely rare as only one case has been reported in the literature -an 88-year-old male with IPT mimicking an incarcerated hernia [4,5].Three histological characteristics have been described by recent studies; the first involves inflammatory cells and has myxoid stroma.The second has compact proliferation of spindle-shaped cells while the third has abundant collagen in a plate-like pattern [6].Our case presented with a small bowel associated mass with histological features consistent with IPT though with mixed histological types.
IPT is similar to neoplastic processes clinically and radiologically hence making accurate diagnosis before surgery difficult.Our case presented with clinical findings consistent with irreducible inguinoscrotal hernia; therefore the decision before surgery with limited radiologic information was to do a mesh repair.deliver the sac and scrotal contents into the wound (Figure 1).As the testicle was normal in appearance, the sac was opened in order to inspect its contents.A bowel segment was firmly attached to a fleshy lesion measuring about 7 cm in diameter adjoining the testicle (Figure 2).The testicular appendage was divided and the small bowel adherent to the sac via fibrinoid adhesions was resected with end-to-end bowel anastomosis in two layers using 2 0 Vicryl and 4 0 PDS respectively.The testis along with the hernia sac was resected and the hernia defect closed using 2/0 Vicryl.A modified Shouldice repair was performed using 2/0 Nylon.He was well enough to self-discharge two days later and was well when seen two weeks postoperatively.
The resected specimen consisted of a fibrofatty

Conclusion
This report adds another case of IPT to the literature and raises awareness about its occurrence.Surgical resection remains the first line of treatment of IPT and medium to long term follow up is essential to ensure prompt management of recurrence.
As it turned out, imaging prior to surgery would have ensured a definite planned procedure with informed patient consent.Not surprisingly there are no typical radiological features of IPT.Ultrasonography reveals diverse pattern of echogenicity while contrast enhanced CT scans may reveal images compatible with the diagnosis [4].On MRI, the appearance of these tumors is also variable; relative to skeletal muscle on T1-weighted images they are usually hypointense, and hyperintense on T2-weighted images, and heterogeneously enhanced after administration of contrast material [5].CT scan and MRI were not available and would not have been affordable by the patient.This demonstrates one of the key challenges of surgery in low to middle income economies.
At surgery the hernia sac was found to have content suspicious of a tumour of testicular or bowel origin.This led to resection of small bowel with good margins and radical orchidectomy performed from an oncological point of view.A modified Shouldice repair of the hernia became the logical choice of technique as the wound was considered as contaminated by virtue of the bowel resection.
The first line of treatment of IPT is surgical resection which was done in our case with good margins.Options for non resectable IPTs include: use of steroids particularly for orbital IPTs; chemotherapy (such as cyclosporine, azathioprine, methotrexate, and cyclophosphamide); and COX II inhibitors [2,4,5,7].Response to ancillary therapy is unpredictable.IPT has a tendency toward local aggressive behavior and recurrence.Rare cases of distant metastases and spontaneous remissions have also been described [5].In a study of 53 cases of extrapulmonary tumors, 13 patients (25%) developed one or more recurrences at one to 24 months post resection.Nine of the patients that developed recurrence were found to have intraabdominal or retroperitoneal tumors with two undergoing histologic transformation into a higher grade sarcoma [7].In another study of four cases involving the alimentary canal, one patient had two recurrences occurring at two (managed by surgical resection) and six (treated with oral anti-inflammatory drugs) months respectively [8].These reported incidences of recurrence raises the need for medium to long term follow up with imaging after surgical resection of IPT.

Figure 1 :
Figure 1: Delivery of hernia sac with scrotal content into wound.

Figure 2 :
Figure 2: Opening of the hernia sac revealing the small bowel with the tumour.

Figure 3 :
Figure 3: Gross photograph of paratesticular lesion with a central cystic/myxoid degeneration.