Xanthogranulomatous appendicitis presenting asymptomatically 3 years after surgery for hilar cholangiocarcinoma: A case report.

INTRODUCTION: Xanthogranulomatous appendicitis (XGA) is a rare condition involving chronic inflammation of the appendix that is often difficult to distinguish from malignancy using imaging because of the formation of a heterogeneous mass with indistinct borders. Herein, we present a case of XGA with unusual clinical manifestations. PRESENTATION OF CASE: A 78-year-old female patient underwent radical resection of hilar cholangiocarcinoma with extended right hepatic lobectomy and biliary reconstruction. Three years postoperatively, she presented with an irregular mass in the right lateral pelvis, which was observed on computed tomography. The patient had not experienced recent clinical symptoms and did not present with abdominal tenderness. Routine blood tests did not indicate an increased inflammatory response; however, carcinoembryonic antigen levels continued to increase. Although disseminated recurrence of hilar cholangiocarcinoma and appendiceal carcinoma were suspected, XGA was diagnosed via laparoscopic appendectomy. DISCUSSION: XGA generally presents with symptoms of acute or chronic appendicitis, and is diagnosed incidentally during surgery. Hilar cholangiocarcinoma has a high recurrence rate, even after radical resection, and disseminated recurrence usually requires chemotherapy. In the present case, XGA was not suspected preoperatively because of the lack of physical symptoms and increased levels of tumor markers during follow-up for hilar cholangiocarcinoma. There have been no reports of XGA with such a confusing clinical course, thus confirming the difficulty in preoperatively diagnosing XGA. CONCLUSION: The preoperative diagnosis of XGA is difficult to differentiate from malignancy because of its clinical and imaging findings. We diagnosed the patient with XGA using laparoscopic surgery.

Xanthogranulomatous Orchitis With Testicular Atrophy in a Case of Chronic Pyocele: A Rare Case.

Xanthogranulomatous orchitis (XGO) is a benign non-inflammatory condition predominantly affecting the testicular tissue and characterized by the infiltration of lipid-laden macrophages leading to substantial tissue damage. We present the case of an 80-year-old gentleman with chronic pyocele and concurrent testicular atrophy secondary to XGO, a seldom-reported manifestation in the testicular milieu. The patient presented with a protracted history of left-sided scrotal swelling and underwent left orchidectomy subsequent to preoperative diagnosis via ultrasonography. Intraoperative exploration revealed the presence of purulent fluid, and histopathological analysis confirmed characteristic features of XGO, including seminiferous tubule destruction and infiltration of fibroconnective tissue by histiocytes and dilated blood vessels. Differential diagnosis with testicular neoplasms posed a challenge, accentuating the pivotal role of histopathological scrutiny in achieving precise diagnosis. Orchidectomy remains the cornerstone of treatment for XGO. This case underscores the imperative of considering XGO in the diagnostic spectrum of testicular masses and the indispensable role of histopathology in confirming the diagnosis and guiding optimal therapeutic interventions.

Xanthogranulomatous inflammation of the female genital tract: An 11-year single institutional study of 40 cases.

BACKGROUND AND OBJECTIVES: Xanthogranulomatous inflammation (XGI) is a rare form of chronic inflammation that affects the female genital tract (FGT). The absence of a standard lexicon in the literature has contributed to the relative obscurity of this condition. We attempt to study this lesion with its various clinicopathological associations. METHODOLOGY: We conducted an 11-year retrospective study of cases diagnosed with XGI of the FGT, analyzing relevant clinical and pathological parameters. RESULTS: Our study identified 40 cases reported as XGI. The mean age was 43.8 (+/- 11.8 SD) years. The most common clinical presentation was abdominal pain (27.5 %). Abdominal mass was seen in 37.5 % of which 22.5 % was primarily attributed to XGI and was not associated with malignancies. The most common site involved was adnexa(87.5 %), with rare involvement of myometrium(7.5 %) and endometrium(5 %). Adnexal involvement was either as tubo-ovarian masses or isolated ovary/fallopian tube involvement. XGI was also seen associated with other primary lesions of FGT like high-grade serous carcinoma(7.5 %) and mature cystic teratoma (7.5 %), while non-neoplastic associations included tubal gestation, foreign body, and Aspergillus infection in the ovary. Histologically, the infiltrate comprised of chronic inflammatory cells in all cases with additional acute inflammatory cells(60 %). Multinucleated giant cells were seen in 40 % of cases. Urine culture showed bacterial colonies in 23 % of cases. CONCLUSIONS: XGI of FGT is an extremely rare lesion and can present as isolated lesions or in association with other primary lesions of FGT. Adnexal involvement was more common than uterine XGI. It is essential to recognize this lesion because it can mimic malignancy and has a destructive nature.

Xanthogranulomatous epithelial tumor: a novel entity of uncertain biologic potential.

Xanthogranulomatous epithelial tumor (XGET) is an extremely rare and recently described mesenchymal neoplasm characterized by a distinctive histological appearance and clinical presentation. This case report describes a unique case of XGET in a 66-year-old female patient who presented with a 5 cm mass in the dorsal distal left thigh. The clinical, radiological, and pathological findings, as well as the management and follow-up, are discussed.

Xanthogranulomatous Endometritis: A Report of Two Cases.

Xanthogranulomatous endometritis (XGE) or histiocytic endometritis is a chronic inflammatory pathology of rare presentation, characterized by an exaggerated inflammatory infiltrate that can mimic an endometrial carcinoma. We report two cases of this disease, one of them with a classic presentation of endometritis and the other one with a severe compromise in which the clinical presentation and imaging findings suggested a possible endometrial carcinoma. Knowledge of this unusual and rare pathology, including its etiopathogenesis, is important since it can be included in the differential diagnosis of endometrial carcinoma and, therefore, whenever it is found, to avoid excessive treatment.

Contrast-enhanced Ultrasound of Xanthogranulomatous Endometritis: A Case Report and Literature Review.

Xanthogranulomatous endometritis (XGE) is a rare inflammatory disease, which can easily misdiagnose as cancer in imaging diagnosis. Diagnosis of XGE relies on histopathological examination and immunohistochemistry. In this study, a case of a 72-year-old female with XGE and elevated CA125 is presented, which was misdiagnosed as endometrial cancer in transvaginal ultrasonography and ovarian cystadenocarcinoma in CT. However, the features of XGE on the contrast-enhanced ultrasound (CEUS) were different from that of endometrial cancer. The patient finally underwent laparoscopic hysterectomy and bilateral adnexectomy. The histopathological examination and immunohistochemistry suggested xanthogranulomatous endometritis (histiocytic endometritis). This case report manifests that CEUS may be a new noninvasive diagnostic method for XGE, which may reduce extensive tissue sampling and unnecessary hysterectomies for patients.

Xanthogranulomatous inflammation requiring small bowel anastomosis revision: A case report.

BACKGROUND: Xanthogranulomatous inflammation (XGI) is an uncommon process involving an accumulation of inflammatory cells, commonly lipid-laden macrophages. XGI has been described to occur throughout the body but only rarely in the lower gastrointestinal tract. We describe a case of XGI contributing to chronic obstructive symptoms in the terminal ileum, in which the patient had an initial diagnostic laparoscopy, continued to have symptoms, then proceeded to have the definitive treatment. To our knowledge, this is the first report of XGI associated with a prior small bowel anastomosis. CASE SUMMARY: We report the case of a 42-year-old female who presented with intermittent epigastric pain and subjective fevers. She had undergone a laparoscopic small bowel resection for Meckel's diverticulum five years prior. Her workup was notable for computed tomography scan demonstrating mild inflammation and surrounding stranding at the level of the prior anastomosis. She underwent a laparotomy, resection of the prior anastomosis and re-anastomosis, with final histopathological examination findings consistent with mural XGI. CONCLUSION: XGI can occur at the site of a prior bowel anastomosis and cause chronic obstructive symptoms.

Ooforosalpingitis xantogranulomatosa. Comunicación de caso

La inflamación xantogranulomatosa del tracto genital femenino es infrecuente y es aún más rara en las trompas de Falopio y ovarios. Se presenta un caso de ooforosalpingitis xantogranulomatosa en una paciente femenina de 45 años quien asistió a consulta por presentar dolor en fosa iliaca izquierda acompañado de fiebre. La exploración bimanual mostró útero ligeramente aumentado de tamaño con masa anexial izquierda firme, no dolorosa, adherida al útero y con limitada movilidad. La evaluación ecográfica transvaginal determinó tumoración ovárica izquierda, heterogénea con paredes gruesas e irregulares con múltiples septos y ecos internos sin visualizar el ovario. Durante la cirugía, se encontraron adherencias densas desde la masa hacia la pared lateral pélvica, fosa ovárica y asas intestinales. El útero estaba desplazado por tumoración anexial quística izquierda, de color blanco grisáceo y paredes gruesas que drenaba líquido purulento fétido. El diagnóstico definitivo fue ooforosalpingitis xantogranulomatosa. Esta condición es un proceso inflamatorio poco frecuente que plantea dilemas diagnósticos. Sus manifestaciones clínicas y características de estudios por imágenes pueden simular una neoplasia pélvica maligna, por lo que es necesario un alto índice de sospecha para su diagnóstico, como diagnóstico diferencial en pacientes con tumoraciones ováricas quísticas complejas. El examen histopatológico es el estándar de oro para el diagnóstico.

Xanthogranulomatous inflammation of the female genital tract is infrequent and is even rarer in fallopian tubes and ovaries. We present a case of xanthogranulomatous oophorosalpingitis in a 45-year-old female patient who consulted for left iliac fossa pain accompanied by fever. Bimanual examination revealed a slightly enlarged uterus with a firm, non-painful left adnexal mass, adherent to the uterus and with limited mobility. Transvaginal ultrasound evaluation showed a heterogeneous left ovarian tumor with thick and irregular walls, multiple septa and internal echoes without visualization of the ovary. During surgery, dense adhesions were found from the mass to the pelvic lateral wall, ovarian fossa, and bowel loops. The uterus was displaced by a thick-walled, grayish-white, cystic left adnexal tumor draining foul-smelling purulent fluid. The definitive diagnosis was xanthogranulomatous oophorosalpingitis. This condition is a rare inflammatory process that poses diagnostic dilemmas. Its clinical manifestations and imaging features may mimic a malignant pelvic neoplasm, so a high index of suspicion is necessary for its diagnosis, as a differential diagnosis in patients with complex cystic ovarian tumors. Histopathological examination is the gold standard for diagnosis.

Xanthogranulomatous Inflammation of the Aortic Aneurysm Wall after Endovascular Aortic Repair for Abdominal Aortic Aneurysm / 日本心臓血管外科学会雑誌

A 77-year-old woman underwent endovascular abdominal aortic repair (EVAR) for an abdominal aortic aneurysm (AAA)．Five years after surgery, she visited the hospital with the chief complaint of a fever. Enhanced computed tomography (CT) showed enlargement of the AAA around the stent-graft and a mass, which was suspected to be an abscess, outside the aneurysm. A blood test revealed a high level of inflammatory response. The patient was diagnosed with infectious AAA. She received antibiotics; however, the inflammatory response did not completely improve. A second CT scan revealed that the suspected abscess had a spreading tendency. The patient was referred to our hospital for a highly suspected stent-graft infection. We performed Y-graft replacement using a rifampicin-immersed graft, and as much as possible of the wall around the aortic aneurysm was removed. The inflammatory response improved rapidly after the operation, and the patient was discharged 15 days later. According to the results of a pathological examination, a diagnosis of xanthogranulomatous inflammation and fibrosis was made. Here, we report a rare case of xanthogranulomatous inflammation of the aortic aneurysm wall after EVAR.

Branchial Cleft Cyst Associated with Xanthogranulomatous Inflammation - An Unusual Case.

Rationale: Branchial cleft cysts are benign lesions that result from developmental defects arising from primitive branchial arches, cleft, and pouches. Xanthogranulomatous inflammation (XGI) is a mass forming lesion and its association with branchial cleft cyst is rare. Patient Concerns: A 23-year-old male presented with a soft, partially mobile, nontender swelling on the left side of submandibular area. His main concern was removal of pathology. Diagnosis: Computed tomography scan showed a well-circumscribed rounded cystic lesion suggestive of 2nd branchial cleft cyst, which was histopathologically confirmed as branchial cleft cyst with XGI. Treatment: Complete surgical excision was done under general anaesthesia and the lesion was removed in toto. Outcomes: Patient's postoperative course was uneventful and he was discharged from the hospital on the 4th postoperative day. Take-away Lessons: Diagnosis of branchial anomalies should always be considered while dealing with lateral neck swellings and XGI associated with branchial cleft cyst should be carefully investigated and treated.

Xanthogranulomatous inflammation caused by K. pneumonia and nocardiosis mimicking a uterine tumor and invading the ureter and colon: A case report and review of the literature.

OBJECTIVE: To present a rare case of xanthogranulomatous inflammation (XI) mimicking a uterine sarcoma and invading the ureter and colon. CASE REPORT: A 66-year-old woman presented with lower abdominal pain. Pelvic examination showed tenderness over the lower abdominal region without cervical discharge. Per-rectal examination showed a hard tumor on the posterior uterine wall, while ultrasonography showed a tumor-like mass extending from the posterior uterine wall to the rectum. Magnetic resonance imaging showed signs of endometrial cancer invading the rectum. However, the tumor markers carbohydrate antigen (CA) 125, CA199, and carcinoembryonic antigen were in the normal range. Cystoscopy, panendoscopy, and colonoscopy showed no significant findings. On performing exploratory laparotomy, we observed pus and severe adhesion on the posterior uterine wall and rectum. Hysterectomy, bilateral adnexectomy, colectomy, and partial left ureter resection were performed. The final pathology showed XI. The pus culture revealed Klebsiella pneumonia and PCR revealed nocardiosis. The patient received 2 weeks of antibiotic treatment and was discharged thereafter. CONCLUSION: XI in elderly women is rare, and hence, differential diagnoses should be carefully considered.

Xanthogranulomatous orchitis: A rare case of testicular mass.

Xanthogranulomatous orchitis is a rare non-malignant condition. This study reports a new case of xanthogranulomatous orchitis in a 77-year-old male, who presented with acute right scrotal mass. Ultrasound and computed tomography showed a heterogenous lesion involving the testis, with a loculated collection in the overlying scrotal wall. Scrotal exploration revealed an abscess involving the scrotal skin, with hard mass involving the testis, with histological evidence of infiltration and replacement of most testicular parenchyma. This case highlights this important consideration for xanthogranulomatous orchitis in the differential of testicular mass, particularly when associated with a purulent collection. Section headings: •Inflammation and Infection•General Urology.

Xanthogranulomatous appendicitis: A comprehensive literature review.

BACKGROUND: Xanthogranulomatous inflammation is characterized histologically by a collection of lipid-laden macrophages admixed with lymphocytes, plasma cells, neutrophils, and often multinucleated giant cells with or without cholesterol clefts. AIM: To review the medical literature on xanthogranulomatous appendicitis (XGA). METHODS: We present a patient with XGA and review published articles on XGA accessed via the PubMed, MEDLINE, Google Scholar, and Google databases. Keywords used were "appendix vermiformis," "appendectomy," "acute appendicitis," and "XGA." The search included articles published before May 2020, and the publication language was not restricted. The search included letters to the editor, case reports, review articles, original articles, and meeting presentations. Articles or abstracts containing adequate information about age, sex, clinical presentation, white blood cells, initial diagnosis, surgical approach, histopathological and immunohistochemical features of appendectomy specimens were included in the study. RESULTS: A total of 29 articles involving 38 patients with XGA, were retrospectively analyzed. Twenty (52.6%) of the 38 patients, aged 3 to 78 years (median: 34; IQR: 31) were female, and the remaining 18 (47.4%) were male. Twenty-five patients were diagnosed with acute appendicitis, ruptured appendicitis, or subacute appendicitis, and the remaining 13 patients underwent surgery for tumoral lesions of the ileocecal region. Twenty-two of the patients underwent urgent or semi-urgent surgery, and the remaining 16 patients underwent interval appendectomy. CONCLUSION: Xanthogranulomatous inflammation rarely affects the appendix vermiformis. It is associated with significant diagnostic and therapeutic dilemmas due to its variable presentation. It is often associated with interval appendectomies, and a significant number of patients require bowel resection due to the common presentation of a tumoral lesion. XGA is usually identified retrospectively on surgical pathology and has no unique features in preoperative diagnostic studies.

A Rare Case Report on Xanthogranulomatous Osteomyelitis of Hip Mimicking Tuberculosis and Review of Literature.

Xanthogranulomatous osteomyelitis (XO) is a rare chronic inflammatory process characterized by the presence of a large number of lipid-containing macrophages with lymphocytes and plasma cells. We present a case of XO of the hip in a 50-year-old woman with pain in the left hip for 28 months. The patient had a history of taking anti-tuberculosis chemotherapy for five months. Laboratory data revealed an increased erythrocyte sedimentation rate and C-reactive protein (CRP) level. Plain radiographs showed the destruction of the femoral head with arthritis and subluxation. Magnetic resonance imaging (MRI) was suggestive of tubercular infection of the left hip and a benign lesion in the left ilium. The histopathologic examination of the specimen demonstrated the presence of dead bone surrounded by lymph-plasma cells, foamy cells, and histocytes, which was consistent with XO, and culture was positive for Staphylococcus aureus infection. The patient was successfully treated with resection arthroplasty and antibiotics. It is important for the surgeons to keep XO in the list of differentials in cases with lytic lesions of bone and assessment should include microbiological culture along with the biopsy.

A case of neck xanthogranulomatous inflammation-suspected malignant tumor.

Xanthogranulomatous inflammation (XGI) is an uncommon chronic inflammatory disease. A 59-year-old male presented with a neck mass which had been diagnosed as an undifferentiated carcinoma. From CD68 staining, XGI was confirmed. It is important to consider the possibility of XGI for a neck mass mimicking a malignant tumor.

Xanthogranulomatous orchitis: case report of a rare condition.

Xanthogranulomatous orchitis is a rare, non-neoplastic inflammatory condition. We present a case of a 55-year-old diabetic male with an increase in volume, pain and edema in right testicle with a one-year history. The examination involved the investigation of inflammatory markers and ultrasound. Exploration of the scrotum revealed important destruction of the tissue architecture. This case underscores the importance of including xanthogranulomatous orchitis in the differential diagnosis of a testicular lump.

Xanthogranulomatous Orchitis Presenting as a Scrotal Mass in an Elderly Male: Malignancy or Mimicker?

Xanthogranulomatous inflammation is a rare, destructive pattern of inflammation, affecting different organs, that often produces a mass-like lesion, simulating malignancy. Although benign, it can coexist with malignancy. We, herein, report the case of a 60-year-old male patient, who presented with a testicular mass that was partly necrotic and showed heterogeneous enhancement on contrast study. Testicular tumor markers were normal. In view of clinicoradiological suspicion of a malignant cause, high inguinal orchidectomy was performed. However, histopathology showed features of a xanthogranulomatous orchitis with no evidence of any malignancy. Pathologists must be aware of this entity as definitive diagnosis requires histopathologic examination and adequate sampling must be done to rule out a coexistent malignancy.

Xanthogranulomatous endometritis mimicking endometrial carcinoma: A case report and review of literature.

Xanthogranulomatous endometritis (XGE) is an extremely rare chronic inflammatory condition, which may be associated with endometrial hyperplasia, endometrial carcinoma, or cervical stenosis. Imaging features can be easily misdiagnosed as an aggressive malignancy. We present a case of XGE, which is the first case of XGE with serial multimodality imaging examinations, in addition to clinical, surgical and pathologic correlations. As such, this unique case illustrates the evolution of this rare disease.

Xantogranulomatous Salpingo Oophritis, Lessons Learnt: Report of Two Cases With Unusual Presentation.

Xanthogranulomatous inflammation is a rare form of chronic inflammatory response consisting of macrophages, lymphocytes, plasma cells and neutrophils too. Due to its locally destructive nature and mass forming capacity as a result of adhesions, this type of inflammation may mimic malignancy or tuberculosis both clinically and radiologically. We present a report of two such cases, one mimicking tuberculosis and the other mimicking malignancy clinically. Awareness of this condition and a higher index of suspicion among clinicians, radiologists and pathologists can help in early diagnosis and more appropriate treatment of this potentially destructive disorder.