Adenoma malignum. Report of a case with cytologic and colposcopic findings and immunohistochemical staining with antimucin monoclonal antibody HIK-1083

BACKGROUND: Adenoma malignum of the uterine cervix was first described by Gusserow. We report here a case with cytologic, histologic and colposcopic findings and immunohistochemistry for HIK-1083. CASE: A 42-year-old female was noted to have a probable adenoma malignum due to the detection of atypical cells classified as V. On colposcopy, comma-shaped, atypical vessels spread over the entire cervical area. Histologic findings were characteristic of tumor invasion beyond the layer of cervical glandular ducts. Immunohistochemical detection of CEA was negative, but HIK-1083, which recognizes gastric glandular mucous cells, was positive. CONCLUSION: For a definitive diagnosis of adenoma malignum of the cervix, immunohistochemical examination for an appropriate marker, such as HIK-1083, should be added to the routine gynecologic examination, cytologic and histopathologic examination, and colposcopy.     

Double cervix with bilateral and synchronous HSIL associated with different high-risk HPV types. A case report

BACKGROUND: High grade squamous intraepithelial lesion (HSIL) of the cervix is well known to be associated with human papillomavirus (HPV) infection. HSIL and invasive carcinomas occurring synchronously in genital malformations, such as a double cervix, have been reported. It has been postulated that the field effect phenomenon of HPV infection is responsible for this synchronous infection. However, there is no information in the literature on the specific types of HPV causing the concomitant lesions in cases with a double cervix. CASE: A 33-year-old nulligravida with a double cervix and a single uterine corpus was diagnosed with bilateral HSIL on Papanicolaou-stained ThinPrep slides (Cytyc Corp., Boxborough, Massachusetts, U.S.A.). A bilateral loop electrosurgical excision procedure cone biopsy revealed HSIL involving both cervices. DNA extracted from the HSIL lesions was analyzed by a polymerase chain reaction-based assay for the presence of HPV. High-risk HPV type 33 was identified in the right cervix, while HPV type 35 was present in the left. CONCLUSION: Demonstration of high-risk HPV types bilaterally supported the etiologic role of HPV infection in the synchronous and bilateral occurrence of HSIL in this case of a double cervix. The HPV types were different in the right and left cervices.     

<Emphasis Type="Italic">Trichophyton rubrum</Emphasis> Infection Characterized by Majocchi’s Granuloma and Deeper Dermatophytosis: Case Report and Review of Published Literature

Infections caused by Trichophyton rubrum are very common in dermatological disease. It most often appears as superficial cutaneous mycosis, such as tinea manuum, tinea pedis, and tinea corporis. However, deep infection caused by T. rubrum was rarely reported. We describe a case of mixed type of deep infection caused by T. rubrum in a 45-year-old man with no significant immunodeficiency. This patient had a history of onychomycosis on the toenails without regular treatment for nearly 6 years. And, he had erythema, papule, and nodules on the submandibular area, neck, and chest for almost 1 year. After treated with intravenous infusion of cefotiam for 2 weeks, the lesion aggravated. The fungal direct microscopic examination of pyogenic fluid was positive, and the fungal cultures that produced reddish-brown and yellow pigment showed cottony, wooly, and white colony. After the DNA sequencing, it was identified as T. rubrum. We gave the patient oral terbinafine 250 mg per day and bifonazole cream for external use. Six months later, the patient’s skin lesion was disappeared, and healthy nail growth was seen in two-thirds of nail bed. The terbinafine is effective against deep infection caused by T. rubrum.     

Acantholytic cells exfoliated from pemphigus vulgaris of the uterine cervix. A case report

BACKGROUND: Pemphigus vulgaris of the uterine cervix is rare and almost always associated with cutaneous or mucosal lesions elsewhere on the body. Without a history of pemphigus, acantholytic cells in cervical smears may be misdiagnosed as malignant ones. CASE: A 52-year-old female presented with a vaginal discharge, and a routine cervical smear was collected for cytology. The smear displayed atypical cells lying singly and in loose clusters, having vesicular nuclei, a thin nuclear membrane, prominent nucleoli and well-defined cytoplasmic margins. These were labeled atypical glandular cells of undetermined significance, and colposcopic examination and biopsy were advised. On colposcopy vesicular lesions and erosions were noticed on the cervix. The biopsy revealed typical intact as well as denuded suprabasal bullae of pemphigus vulgaris. On reevaluation of the cytologic smear, the cytomorphologic features correlated well with the acantholytic cells of pemphigus. Thorough reexamination of the patient revealed 2 tiny vesicles on the oral mucosa that, on biopsy, confirmed the diagnosis of pemphigus vulgaris. CONCLUSION: Cytopathologists should be aware of the typical cytomorphologic features of pemphigus vulgaris and, in an appropriate clinical setting, should be able to diagnose or at least suspect this entity in even rare sites like the cervix. A false positive diagnosis of malignancy can be avoided if the cytologic findings are judiciously correlated with the history and with clinical and colposcopic examinations.     

Fungal Olecranon Bursitis in an Immunocompetent Patient by <Emphasis Type="Italic">Knoxdaviesia dimorphospora</Emphasis> sp. nov.: Case Report and Review

Bursitis is a common medical condition that can occur either with or without infection. We present a case of fungal olecranon bursitis in an immunocompetent individual caused by the new species Knoxdaviesia dimorphospora. It is a dematiaceous filamentous fungus characterized by the production of two different conidia: hyaline and cylindrical, which rise up from phialidic conidiogenous cells located in the upper part of differentiated and unbranched conidiophores, and pale brown and ellipsoidal conidia produced by phialidic conidiogenous cells which are born directly on hyphae. In addition to its morphological peculiarities, the novelty of the fungus was confirmed by sequence analysis of the internal transcribed spacer (ITS) regions and D1/D2 domains of the 28S of the nuclear rRNA gene. The fungal infection was confirmed by cytological examination and repeated cultures. The infection was resolved by surgical debridement and drainage, and the patient presented a complete functional recovery 3 months later. The in vitro antifungal susceptibility to this new human opportunist is provided, terbinafine being the drug with the most potent activity.     

Subcutaneous Mycotic Cyst Caused by <Emphasis Type="Italic">Roussoella percutanea</Emphasis> in a UK Renal Transplant Patient

Fungi from more than 100 genera have been implicated in subcutaneous fungal infections, usually following traumatic inoculation of the etiologic agent. With the advent of molecular approaches to fungal identification and taxonomy, novel agents of subcutaneous mycoses are increasingly reported. In this manner, Roussoella percutanea, a novel species in Pleosporales, was described in 2014 from a subcutaneous mass in an immunocompetent male adult. Two additional cases involving renal transplant recipients were recently reported from patients resident in France and Germany, with several further cases discovered after analyses of historical culture collection isolates. Here, we describe a new case of subcutaneous R. percutanea infection, causing a mycotic cyst in a renal transplant patient resident in the UK. Although fungal infection was confirmed histologically, viable fungal isolates could not be recovered in culture from biopsy material and identification of the causative agent relied upon PCR amplification and sequencing of fungal rDNA genes. This is the fourth well-documented case of infection with R. percutanea in renal transplant patients, and the first reported from a patient resident in the UK. The current case illustrates the importance of molecular approaches for the identification of emerging fungal pathogens in culture-negative subcutaneous fungal infections.     

Acute myelogenous leukemia relapsing as granulocytic sarcoma of the cervix. A case report

BACKGROUND: Granulocytic sarcoma of the uterine cervix is an unusual manifestation of acute myeloid leukemia, representing soft tissue masses of leukemic myeloblasts. An often misdiagnosed entity, it is often confused with other inflammatory or neoplastic conditions, including large cell lymphoma. CASE: A 67-year-old female presented with acute myelogenous leukemia and a normal karyotype. After eight years in complete remission, abdominal pain and an ulcerated mass in the uterine cervix developed, with a normal peripheral blood smear. Vaginal cytology examination revealed myeloid blasts, which, on subsequent cervical biopsy, stained positive for leukocyte common antigen, Kp-1 (CD68), antimyeloperoxidase, lysozyme and chloroacetate esterase, confirming the cytologic diagnosis. K-ras was not mutated at codon 12 or 13. Chemotherapy induced a complete remission, followed nine months later by central nervous system and then systemic relapse. The patient died 13 months after being diagnosed with granulocytic sarcoma of the cervix. CONCLUSION: This case illustrates the value of vaginal cytology and histologic biopsy evaluation in patients with acute myelogenous leukemia, including those without evidence of systemic disease. The characteristic cytologic features of granulocytic sarcoma led to the correct diagnosis. Histologic biopsy evaluation, including immunohistochemistry for myeloid markers, proved of value in confirming the diagnosis.     

Exfoliative cytology of atypical polypoid adenomyoma. A case report.

BACKGROUND: An atypical polypoid adenomyoma (APA) is a well-defined entity. It occurs in the endometrium, lower uterine segment and endocervix. It is usually composed of atypical complex glands with squamous metaplasia admixed with myofibromatous stroma. CASE: A 35-year-old female presented with one-year history of irregular menstrual periods. A diagnosis of adenocarcinoma in situ was rendered on her cervicovaginal smear. Pelvic examination revealed an enlarged uterus due to a leiomyoma. Colposcopic examination revealed a 0.6-cm, sessile, polypoid mass at the junction of the endocervix and ectocervix. A cone biopsy of the lesion showed irregular, endometrial-type glands embedded in a prominent myofibromatous stroma. The atypical glandular component of the mass demonstrated varying degrees of architectural complexity, ranging from simple to complex hyperplasia. In tissue sections the lesion was diagnosed as APA of the cervix. The patient underwent a hysterectomy for the leiomyoma. The hysterectomy specimen showed an 8.5-cm leiomyoma. The cervix and uterine corpus revealed no residual APA. CONCLUSION: APA of the cervix should be considered among the differential diagnoses of atypical glandular cells of undetermined significance. The diagnosis of APA cannot be made on cytology; the final diagnosis requires histologic confirmation.     

Pneumocystis carinii in pleural fluid. The cytologic appearance

We describe the cytologic appearance of Pneumocystis carinii in pleural fluid of a patient with acquired immunodeficiency syndrome and a rapidly accumulating pleural effusion. The diagnosis of P carinii infection was made by examination of air-dried, Diff-Quik-stained Cytospin preparations of the pleural fluid. The diagnostic appearances of P carinii organisms stained by this method and by the Papanicolaou stain are reviewed. The unusual predominance of the trophozoite forms of the organism in this case made Diff-Quik an ideal special stain for identifying the organisms. Furthermore, this case illustrates a novel presentation of P carinii infection and suggests that P carinii should be considered an etiologic agent in the differential diagnosis of pleural effusion in an immunocompromised host.     

Evidence of a <Emphasis Type="Italic">Prototheca Zopfii</Emphasis> Genotype 2 Disseminated Infection in a Dog with Cutaneous Lesions

Protothecosis is a disease caused by saprophyte aerobic unicellular algae belonging to the genus Prototheca. In dogs, it mainly occurs as a disseminated form, with initial clinical manifestations often referable to the gastrointestinal tract, followed by typical ocular and neurological signs. So far, Prototheca zopfii genotype 2 infection has been reported in severe forms of disseminated protothecosis, while in dogs has never been associated with cutaneous forms. In this study, we describe a case of Prototheca zopfii genotype 2 infection in a dog characterized by nodular and ulcerative dermatitis and with evidence of dissemination. In December 2015, a 5-year-old unneutered male English Setter dog was presented with a 4-month history of footpads ulcerations and multifocal nodular lesions (3–5 cm diameter) on both front limbs. Cytological examination of the aspirated fluid collected from all nodules revealed the presence of sporangic forms compatible with Prototheca spp. organisms. Suspected Prototheca spp. colonies were isolated from the aspirated fluid and identified as Prototheca zopfii genotype 2 by molecular methods. Few days after the visit, the patient developed serious neurological and ocular signs, and the owners elected humane euthanasia. To the authors’ knowledge, this case could represent the first report of a disseminated Prototheca zopfii genotype 2 infection associated with cutaneous lesions in a dog. This study underlines the importance of considering Prototheca zopfii genotype 2 infection in the differential etiological diagnosis of nodular and ulcerative dermatitis in dogs.     

被误诊为环状肉芽肿的石膏样小孢子菌引起体癣1例

报告1例被误诊为环状肉芽肿的由石膏样小孢子菌引起的体癣。患者女,60岁,左腕部红斑2个月。外院曾诊断为"环状肉芽肿"治疗不佳,经真菌学检查确诊为石膏样小孢子菌导致的体癣。给予伊曲康唑200 mg/d,共1周,硝酸舍他康唑软膏外用,治疗1周后皮损痊愈。     

1例热带念珠菌脓肿误诊分析

报告1例热带念珠菌引起的腹腔脓肿瘘管真菌感染。患者女,78岁,因十二指肠球部穿孔并发腹膜炎,行腹腔镜术,术后伤口反复未完全愈合,有少量分泌物溢出。发现腹壁瘘管及腹腔脓肿入院,两次细菌培养均无菌生长,再次取分泌物直接镜检阳性,真菌培养鉴定为热带念珠菌。手术清理组织,病理示感染性肉芽肿改变,查见大量假菌丝及孢子。经口服伊曲康唑和两性霉素B溶液纱条引流治疗3个月后基本痊愈。     

Fungal Keratitis Secondary to <Emphasis Type="Italic">Trametes betulina</Emphasis>: A Case Report and Review of Literature

Purpose

We report the first case of human infection and keratitis secondary to Trametes betulina, a rare filamentous fungus.

Methods

Clinical examination including external and slit-lamp examination and corneal scrapings with microbiologic evaluation were performed on a patient with chronic allergic conjunctivitis, entropion and a long-standing corneal ulcer resistant to treatment.

Results

The culture from the corneal scraping revealed a basidiomycetous fungus which was submitted for identification. DNA extraction with sequencing and analysis of the ITS and D1/D2 regions were performed on the isolate and demonstrated 100% similarity to Lenzites betulina/Trametes betulina. Susceptibility testing demonstrated potent in vitro activity of voriconazole (MIC < 0.03 μg/ml). The patient was treated with voriconazole, and the corneal ulcer and infiltrate resolved. The infection resulted in corneal thinning and a dense central corneal scar. Penetrating keratoplasty was performed 5 months after diagnosis and treatment and revealed stromal scarring without fungal elements.

Conclusion

This is the first reported case of keratitis caused by Trametes betulina. This organism should be considered in the differential diagnosis for rare filamentous fungal keratitis and its treatment with voriconazole also noted.

     

21例淋巴瘤合并肺部侵袭性真菌感染的回顾性临床分析

摘要 目的：探讨淋巴瘤合并肺部侵袭性真菌感染( IPFI ) 的高危因素、临床特征和诊疗方案。方法：回顾性分析2019-2020年血液科收治的淋巴瘤合并IPFI患者的临床资料。结果：化疗后出现IPFI的淋巴瘤患者共计21例,总发病率为2.7%,平均年龄60岁,男性占77.8%,其中确诊5例、临床诊断7例、拟诊8例,确诊患者的病原菌为白色念珠菌（80%）和曲霉菌（20%）。肺部CT影像特征不典型,大多表现为弥漫、散在的斑片状絮样密度增高影、结节影,双肺受累多见,在接受一线抗真菌治疗后有18例患者缓解,总有效率为90.4%,其中原发病终末期患者及合并细菌感染患者死亡率高,预后差。结论：老年、男性、原发病控制不佳、高强度或大剂量化疗以及糖皮质激素的使用可能是淋巴瘤IPFI的高危因素,早期临床症状和影像学检查缺乏特异性,常规病原学检出率仍较低,1-3-β-D-葡聚糖试验（G试验）、半乳甘露聚糖试验（GM试验）结合肺部CT检查有助于早期诊断,一线抗真菌药物的使用可显著改善患者预后。     

Endogenous Fusarium Endophthalmitis During Treatment for Acute Myeloid Leukemia,Successfully Treated with 25-Gauge Vitrectomy and Antifungal Medications

Endogenous fungal endophthalmitis (EFE) caused by disseminated fusariosis is a rare condition that generally has a poor outcome, even with intensive therapy. Here, we describe a case in which this type of EFE was diagnosed with vitreous sampling and was successfully treated with 25-gauge vitrectomy and antifungals, including liposomal amphotericin B and voriconazole. A 16-year-old male patient undergoing treatment for acute myeloid leukemia complained of eye pain and blurred vision in his right eye. Treatment was initiated for a vitreous opacity, possibly associated with herpetic retinitis, but the patient worsened and he was referred to us. Right-eye visual acuity was limited to light perception. We suspected endogenous endophthalmitis and performed 25-gauge vitrectomy with antibiotic perfusion of ceftazidime, vancomycin, and voriconazole. Vitreous culturing revealed the presence of Fusarium solani species complex, and enhanced computed tomography revealed disseminated fusariosis lesions in the lung, spleen, and the soft tissue of the left upper arm. The patient received antifungal treatment with liposomal amphotericin B and voriconazole, and these conditions were eliminated. Visual acuity recovered to 20/400 after additional vitrectomy for tractional retinal detachment and was maintained at this level during the 6-month follow-up period. The success of our treatment allowed the capture of optical coherence tomography images of the retina during fusarium-associated endogenous endophthalmitis and the follow-up period. Furthermore, this case showed that immediate vitrectomy for suspected EFE and intensive treatment can lead to a good clinical outcome.     

面癣误诊1例

报道由须癣毛癣菌感染引起并被长期误诊的面癣1例。患者女,37岁,因"面部反复红斑、丘疹、水疱伴瘙痒半年"就诊。多次被误诊为单纯疱疹、脓疱疮、湿疹、皮肤感染、脂溢性皮炎及玫瑰痤疮等,先后应用抗病毒、抗感染和抗过敏治疗,皮损未能完全消退并逐渐扩大。再次就诊,真菌学直接镜检阳性,真菌培养为须癣毛癣菌。给予口服特比萘芬片、外用联苯苄唑乳膏治疗后痊愈。     

Disseminated histoplasmosis in a SIV-infected rhesus monkey

Disseminated histoplasmosis due to Histoplasma capsulatum was diagnosed in a rhesus monkey inoculated with simian immunodeficiency virus (SIV). Immunohistochemical staining of tissues for viral core antigens revealed that those macrophages that expressed viral antigen contained few or no fungal organisms, while those that were filled with fungal organisms did not express viral antigen. This is a previously undocumented condition in a SIV-infected macaque, and suggests that SIV infection of individual macrophages is not the cause of macrophage dysfunction in SIV infections.     

Eumycetoma of the Hand Caused by <Emphasis Type="Italic">Leptosphaeria tompkinsii</Emphasis> and Refractory to Medical Therapy with Voriconazole

We report on the first case of eumycetoma caused by the organism Leptosphaeria tompkinsii to be diagnosed and possibly acquired within the United Kingdom. Conventional culture of fungal grains and surgical tissue specimens was negative and the diagnosis was achieved using panfungal polymerase chain reaction and sequencing technology. Despite limited surgical resection and prolonged antifungal therapy with voriconazole, the patient developed progressive disease with mycetoma bone involvement. This case highlights the usefulness of molecular diagnostic techniques in eumycetoma where organisms may fail to grow with conventional culture or be difficult to identify morphologically. It also reminds us that eumycetoma is a difficult infection to treat and despite optimism regarding the efficacy of the newer triazole antifungals in this condition, treatment failures may still occur.     

Fatal <Emphasis Type="Italic">Talaromyces marneffei</Emphasis> Infection in a Patient with Autoimmune Hepatitis

Talaromyces marneffei, previously known as Penicillium marneffei, is the most important pathogenic thermally dimorphic fungus causing systemic mycosis in Southeast Asia. Traditionally, T. marneffei infection in human was mainly associated with acquired immunodeficiency syndrome caused by HIV infection. In recent years, there has been an increasing number of T. marneffei infections reported in non-HIV-infected patients with other immunocompromised conditions, including autoantibodies against interferon-gamma, systemic lupus erythematosis, solid organ transplantation, Job’s syndrome, hematological malignancies, and use of novel targeted therapies. In this article, we describe the first case of fatal T. marneffei infection in a patient with underlying autoimmune hepatitis, presented as fever without localizing features. The diagnosis of talaromycosis was confirmed with the identification of the fungi isolated from the blood culture specimen by conventional methods and using matrix-assisted laser desorption–ionization time-of-flight mass spectrometer. This case shows the importance of a high index of suspicion, particularly for such a highly fatal but potentially treatable fungal infection.     

Subungual Exostosis in an Osteoporotic Patient Treated with Teriparatide

ObjectiveTo report an unusual case of subungual exostosis in a patient on teriparatide.MethodsWe describe the presentation and symptoms of the patient and review of the relevant literature.ResultsTeriparatide is used for the treatment of osteoporosis. Rat studies using 3-60x the approved human dose have shown an association between teriparatide and an increased risk of osteosarcoma. Subungual exostosis, to our knowledge, has not been reported. We report the case of a 54-year-old female who presented with a 4-month history of pain and swelling in the medial side of her right thumb with no preceding trauma. The patient had history of severe osteoporosis with multiple fractures and was on teriparatide for 16 months. On examination, the right thumb was swollen and tender with no superficial erythema or signs of an infection. X-ray imaging revealed a trabecular bony overgrowth consistent with subungual exostosis. The patient was treated with subungual excision. Pathology showed endochondral bone formation with reactive atypia, consistent with osteocartilaginous exostosis.ConclusionTo our knowledge, this is the first case hypothesizing an association between teriparatide and subungual exostosis. Subungual exostosis is a benign growth of bone that arises in the distal phalanx, under or adjacent to the nail bed. The pathophysiology is not clearly understood, but the lesion has base of trabecular bone with a proliferating fibrocartilaginous cap. Teriparatide can stimulate the trabecular bone formation. Hence, an association between the use of teriparatide and the development of subungal exostosis cannot be excluded. Further studies delineating this relationship are needed. (Endocr. Pract. 2013;19:e115-e117)