Xanthogranulomatous Appendicitis in a Child: Report of a Case and Review of the Literature

Abstract

Xanthogranulomatous inflammation is a well-described inflammatory process, which may involve any organ but is most frequently encountered in the gall bladder and the kidney. There are rare reports of xanthogranulomatous appendicitis (XA) in the adult population, but only one brief mention of such a diagnosis in a child. In this report, we describe the case of an 11-year-old boy who presented with clinical signs and symptoms of acute appendicitis necessitating appendectomy. Upon microscopic examination, the appendix showed the typical features of XA. To the best of our knowledge, this is the first well-described case XA in a noninterval appendix in a child. We also reviewed the limited medical literature on the subject.

Cytological diagnosis of xanthogranulomatous appendicitis

“…Though initially described in kidney in 1944, it occurs in other organs including fallopian tubes, endometrium, testicle, female and male genital tract, gallbladder, pituitary, colon, retroperitoneum, adrenal, and appendix. [35]…”

“…[3] Of these three cases presented as mass lesion, Chuang et al . in 2005 reported a case of a 39-year-old man who was admitted with fever, lower abdominal pain, and a mass in the right iliac fossa.…”

“…[1] The particular mechanism eliciting XA are organ obstruction, suppurative infection, hemorrhage, defective lipid transport, and local hypoxia. [3] Histopathology of delayed (interval) appendectomy specimens shows higher incidence of XA. [278]…”

“…[1] This type of tissue reaction can involve any organ but the most common sites are kidney and gallbladder. [23] Appendix is a very uncommon site for xanthogranulomatous reaction. Only a few case reports are available in the literature.…”

Abdominal manifestations of histiocytic disorders in adults: imaging perspective

“…35 On ultrasound, hypoechoic nodules may be seen within the thickened gallbladder wall (Figure 7), and CT may demonstrate correlative hypodense foci in the gallbladder wall. 36 Xanthogranulomatous appendicitis is a very unusual type of appendicitis with around 10 cases reported in the literature 37 with a strong association with interval appendectomy. 38 On imaging, it may show features similar to an appendiceal mucocele or tumour-like thickening with or without involvement of the caecum 39 ( Figure 8a,b).…”