The 490th case: arthralgia, amenorrhea, aphasia / 中华内科杂志

A 25-year-old woman was admitted to Peking Union Medical Hospital presented with arthralgia for 5 years, amenorrhea for 16 months, and speech disorder for 3 months. This patient has been afflicted by intermittent pain in metacarpophalangeal and proximal interphalangeal joints of both hands for 5 years. Her menstruation has been irregular 1 year ago and rapidly progressed to amenorrhea. Laboratory tests revealed postmenopausal sex hormones levels (estradiol<5 ng/L, follicle-stimulating hormone 62.5 IU/L, luteinizing hormone 58.71 IU/L) and no antral follicles were seen in gynecologic ultrasound. She was diagnosed with premature ovarian failure and treated with hormone replacement therapy, still with no ovulation. Numbness and weakness of right arm has recurrently occurred to her 4 months ago, and persistent weakness of right limbs combined with motor speech disorder occurred 1 month later. Magnetic resonance angiography was suggestive of ischemic stroke. Hormone replacement therapy was discontinued. Comprehensive laboratory tests revealed positive anti-dsDNA, anti-SSA/SSB, anticardiolipin and anti-β 2GPⅠ antibodies. Systemic lupus erythematosus (SLE), antiphospholipid syndrome (APS) was diagnosed. Since no drug with gonadal toxicity had been applied to the patient before, her amenorrhea was considered to be due to autoimmune oophoritis secondary to SLE. After treated with high-dose glucocorticoid, mycophenolate mofetil and hydroxychloroquine for 4 months, her menstruation recurred and regularly occurred till now. In some cases, amenorrhea in SLE patient might be resulted from autoimmune oophoritis associated with lupus flare, instead of use of drug with gonadal toxicity.

Xanthogranulomatous salpingo-oophoritis: a case report and review of literature

Xanthogranulomatous inflammation of ovary is a rare disease that is characterized by presence of large number of lipid laden macrophages with an admixture of neutrophils, lymphocytes, plasma cells and multinucleated giant cells. It is misdiagnosed as ovarian tumour that leads to extensive surgery including hysterectomy. In this report we describe a case of Xanthogranulomatous salpingo-oophoritis along with review of literature.

Xanthomatous oophoritis, a rare pathology: case report with review of literature

Xanthomatous oophoritis is a rare chronic inflammation of ovary characterized histologically with infiltration of lipid laden foamy macrophages, lymphocytes, plasma cells leading to tissue destruction. Though exact cause is not known, uterine artery embolization, gloves dusting powder and altered lipid metabolism are hypothesized to cause the pathology. A 28-year-old parous lady with history of multiple laparotomies, known case of hypothyroidism under treatment and history of adequately treated pulmonary tuberculosis was diagnosed to have right ovarian dermoid cyst, while undergoing investigation for secondary infertility. On examination she had pallor, healthy abdominal scar, and small tender fixed mass in right fornix on internal examination. She was subjected to laparotomy and right salpingo oophorectomy with left salpingectomy was performed. Histopathological examination of the resected specimen revealed to be xanthomatous oophoritis of right ovary. As this condition mimics tuberculosis and malignancy, clinical assessment, investigation and intra operative findings with the suspicion of inflammatory aetiology help to limit the extent of surgery. Hence, this case is presented for its rare occurrence as well as its need to be differentiated from other conditions like tuberculosis and malignancy of ovary.

Xanthogranulomatous salpingo-oophoritis mimicking an ovarian malignancy: a clinical dilemma

Background: Xanthogranulomatoussalpingo-oophoritis is an uncommon form of chronic inflammation in the genitourinary tract. Its symptoms and radiological findings mimic ovarian malignancy. Aim of this study was to evaluate the clinicopathological pattern of xanthogranulomatous salphingo oophoritis.Methods: This study was conducted in the department of pathology, Government Medical College, Srinagar. It was a retrospective study done over a period of 5 years, November 2014 to November 2019.Results: The study was done to evaluate the clinicopathological pattern of 6 cases of rare entity xantogranulomatous salphingo oophoritis diagnosed at a tertiary care hospital. All patients, presented with pain abdomen. All patients were operated due to radiological suspicion of ovarian malignancy. Histopathological examination proved the lesion as xanthogranulomatous salphingo oophoritis.Conclusions: Xanthogranulomatoussalpingo-oophoritis is a rare condition that is often mistaken for ovarian malignancy clinically and radiologically. Oophorectomy is the recommended treatment but most women are “over treated” with staging laparotomies and hysterectomies that render them infertile.

Fatal disseminated cytomegalovirus infection with necrotizing oophoritis in a patient with acquired immunodeficiency syndrome

Disseminated human cytomegalovirus (CMV) disease occurs mainly as a congenital infection and among immunocompromised hosts. Patients with acquired immunodeficiency syndrome (AIDS) are at increased risk for CMV infection, and the most prevalent clinical manifestation is retinitis, followed by colitis, esophagitis, pneumonitis, and encephalitis. CMV oophoritis is poorly described in the literature with some cases reported in patients with hematological or solid malignancies, bone marrow or solid organ transplantation, immunosuppressive therapy, and advanced AIDS cases. We report the case of a 61-year-old woman with a recent diagnosis of AIDS, which was associated with a wasting syndrome. The patient presented with abdominal pain, headache, cutaneous vesicular lesions on the abdomen, anemia, lymphopenia, and hyponatremia; she died suddenly on the fourth day of hospitalization. The autopsy was performed and demonstrated disseminated CMV infection with hemorrhagic encephalitis as the immediate cause of death. Additionally, pneumonitis, extensive adrenalitis, ulcerated enteritis, focal hepatitis, and necrotizing oophoritis were found.

Abdominal and pelvic actinomycosis due to longstanding intrauterine device: a slow and devastating infection

Actinomycosis is a chronic or subacute bacterial infection characterized by large abscess formation, caused mainly by the gram-positive non-acid-fast, anaerobic, or microaerophilic/capnophilic, obligate parasites bacteria from the genus. Although pelvic inflammatory disease is an entity associated with the longstanding use of intrauterine devices (IUDs), actinomycosis is not one of the most frequent infections associated with IUDs. We present the case of a 43-year-old female patient who was referred to the emergency facility because of a 20-day history of abdominal pain with signs of peritoneal irritation. Imaging exams revealed collections confined to the pelvis, plus the presence of an IUD and evidence of sepsis, which was consistent with diffuse peritonitis. An exploratory laparotomy was undertaken, and a ruptured left tubal abscess was found along with peritonitis, and a huge amount of purulent secretion in the pelvis and abdominal cavity. Extensive lavage of the cavities with saline, a left salpingo-oophorectomy, and drainage of the cavities were performed. The histopathological examination of the surgical specimen revealed an acute salpingitis with abscesses containing sulfur granules. Therefore, the diagnosis of abdominal and pelvic actinomycosis was made. The postoperative outcome was troublesome and complicated with a colocutaneous fistula, which drained through the surgical wound. A second surgical approach was needed, requiring another extensive lavage and drainage of the recto-uterine pouch, plus the performance of a colostomy. Broad-spectrum antibiotics added to ampicillin were the first antimicrobial regimen followed by 4 weeks of amoxicillin during the outpatient follow-up. The patient satisfactorily recovered and is already scheduled for the intestinal transit reconstitution.

Update on 46,XX primary ovarian insufficiency in adolescents / 中华实用儿科临床杂志

46,XX primary ovarian insufficiency(POI)is a clinical syndrome defined by loss of ovarian activity before the age of 40 years old with a karyotype 46,XX,characterized by menstrual disturbance(amenorrhea or oligomenorrhea)in association with hypergonadotropic hypogonadism.46,XX POI is a rare disease with the prevalence lower than 1％,of whom 2.5％are adolescents.Potential etiologies for 46,XX POI can be divided into genetic,autoimmune,and iatrogenic categories.Unfortunately,for most patients presenting with POI,the cause will remain unexplained.Once,POI is diagnosed,clinical indicated tests are needed to identify the mechanism causing POI.POI is a complex condition appearing with a strong genetic basis.Large-scale genomic sequencing had recently identified new mechanisms of POI.The management of the condition should address both of physical and emotional well-being health,including health education,hormone develoment treatment,prevention and treament of lower-estrogen associated diseases,with the support from a multidisciplinary team.

Autoimmune oophoritis: A rarely encountered ovarian lesion.

Autoimmune oophoritis is a rare disorder causing ovarian failure clinically characterized by amenorrhea and infertility. It often occurs in a setting of autoimmune polyendocrine syndromes. A 38-year-old female presented with a 3 years history of secondary amenorrhea. She was on treatment for Hashimoto’s thyroiditis and Addison’s disease. The ovaries were cystic and histologically featured by folliculotropic lymphoplasmacytic infl ammatory infi ltrate concentrated in the theca interna layer of developing follicles, but sparing the primordial follicles.

Clinical analysis of 12 cases of gynecological infections acute abdomen misdiagnosed as acute suppurative appendicitis / 中国基层医药

Objective To explore the reasons of gynecological infections acute abdomen misdiagnosed as acute suppurative appendicitis and put forward methods to reduce the misdiagnosis.Methods To review and analyze the clinical data of 12 patients with gynecological infectious who had been misdiagnosed as acute suppurative appendicitis.Results The acute pyogenic pelvic inflammatory disease in 6 cases,right salpingitis and empyema in 3 cases,ovarian cysts in 3 cases.Conclusion The lack of clinical experience and examination,omissions and misleading relevant gynecological history,over-reliance on secondary inspections,and lack of consultation are the main reasons of gynecological infectious diseases misdiagnosis.

A case of xanthogranulomatous oophoritis developed in a virgin / 대한산부인과학회지

Xanthogranulomatous inflammation is an uncommon form of chronic granulomatous inflammation that is characterized by the presence of lipid-filled histiocytes with lymphocytes, plasma cells and neutrophils. It is best known to occur in the kidney. Other organs in which xanthogranulomatous inflammation has been reported are the gallbladder, stomach, anorectal area, bone, urinary bladder, testis, epididymis and female genital tract. Only a few cases of xanthogranulomatous oophoritis have been reported to date. Infection has been thought to be the main etiologic factor in the pathogenesis of xanthogranulomatous lesion. The pathogenesis of xanthogranulomatous inflammation remains still unclear. Other proposed causes are abnormality in lipid metabolism, endometriosis and ineffective clearance of bacteria by phagocytes. We describe a case of a xanthogranulomatous oophoritis in a 23-year-old virgin which associates with hemorrhagic ovarian cyst and minimal association of pelvic inflammatory disease.

A Case of Xanthogranulomatous Oophoritis / 대한산부인과학회잡지

Xanthogranulomatous oophoritis is uncommon forms of chronic granulomatous inflammation in the female genital tract. It is characterized by the presence of foamy histiocytes with admixed lymphocytes, plasma cells, and neutrophils. We describe a case of xanthogranulomatous oophoritis in a 75-year-old woman, which was misdiagnosed as uterine sarcoma preoperatively.

A Case of Xanthogranulomatous Oophoritis / 대한산부인과학회잡지

Xanthogranulomatous oophoritis is uncommon forms of chronic granulomatous inflammation in the female genital tract. It is characterized by the presence of foamy histiocytes with admixed lymphocytes, plasma cells, and neutrophils. We describe a case of xanthogranulomatous oophoritis in a 75-year-old woman, which was misdiagnosed as uterine sarcoma preoperatively.

Autoimmune oophoritis: a case report

A case is described of an autoimmune oophoritis that was diagnosed unexpectedly after a hysterectomy and bilateral salpingo-oophorectomy had been performed on the suspicion of ovarian cysts. The patient was a 43-year-old multiparous woman who presented with vaginal bleeding and lower abdominal pain which she had had for one month. Grossly, the ovaries were enlarged and multicystic. The cysts measured up to 3.0 cm. The major histological change was a lymphoplasmacytic infiltrate in close relation to the theca interna of developing, cystic and atretic follicles, but sparing the primordial follicles. The infiltrate increased in density with the follicular maturation and culminated against the corpus luteum. With involution of the developing follicles, the inflammatory infiltrate subsided to some extent. The proportion of the plasma cells increased with the density of the infiltrates. Immunohistochemical study of the ovarian mononuclear cell infiltrate revealed a mixture of B- and T-lymphocytes. The plasma cells were polyclonal. These histological features of the present case are typical of autoimmune oophoritis although the presence of autoantibodies and hormonal level in the patient's serum were unknown. This case may be identified as in the early active stage of autoimmune oophoritis.