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Annals of Diagnostic Pathology Aug 2020Anogenital mammary-like glands, formerly described as ectopic breast tissue, are currently considered to be normal histologic components of the anogenital region....
Anogenital mammary-like glands, formerly described as ectopic breast tissue, are currently considered to be normal histologic components of the anogenital region. Anogenital mammary-like glands can give rise to many lesions identical to counterparts in the native female breast. We describe four cases of such lesions, including fibroadenoma, gynecomastia-like hyperplasia, and ectopic mammary-type tissue with a spectrum of usual ductal hyperplasia, apocrine metaplasia, adenosis, and pseudolactational change. All four cases occurred in young women (ages 29-38) who presented with vulvar or perianal masses. Similar to previously reported cases, these lesions shared histologic and immunohistochemical characteristics identical to native female breast lesions. Novel findings in our cases included (1) the first case of gynecomastia-like change to be reported in the perianal area of a female, (2) Immunohistochemical staining identifying a 3-layered epithelium characterized by a population of CK14 and CK5/6 positive and hormone receptor negative superficial luminal cells, and (3) diffuse, strong positivity for GATA3 in all cases. Our study adds to the literature on these rare lesions and highlights findings which may be useful in understanding the pathogenesis and improving the diagnosis of anogenital mammary-like gland lesions.
Topics: Adult; Anus Neoplasms; Breast; Breast Diseases; Breast Neoplasms; Choristoma; Female; Fibroadenoma; Fibrocystic Breast Disease; GATA3 Transcription Factor; Humans; Immunohistochemistry; Mammary Glands, Human; Metrorrhagia; Perineum; Vulvar Neoplasms
PubMed: 32592993
DOI: 10.1016/j.anndiagpath.2020.151551 -
Veterinary Pathology May 2020Pediatric patients receiving solid organ transplants may develop lymphoproliferative diseases, including graft-versus-host disease (GvHD) and posttransplant...
Pediatric patients receiving solid organ transplants may develop lymphoproliferative diseases, including graft-versus-host disease (GvHD) and posttransplant lymphoproliferative diseases (PTLDs). We characterized lesions in 11 clinically ill NOD.Cg-/SzJ (NSG) mice that received pediatric-patient-derived solid tumors (PDXs) and developed immunodeficiency-associated lymphoproliferations comparable to GvHD and PTLDs over a period of 46 to 283 days after implantation. Lymphoproliferations were diffusely positive for human-specific biomarkers, including NUMA1, CD45, and CD43, but lacked immunoreactivity for murine CD45. Human immune cells were CD3-positive, with subsets having immunoreactivity for CD4 and CD8 as well as PAX5, CD79a, and IRF4, resulting from populations of human T and B cells present within the xenotransplants. Tissues and organs infiltrated included mucocutaneous zones (oral cavity and perigenital and perianal regions), haired skin, tongue, esophagus, forestomach, thyroid, salivary glands, lungs, liver, kidneys, spleen, lymph nodes, bone marrow, and brain. In 4 of 5 mice with PTLD, Epstein-Barr virus (EBV)-encoded small RNAs (EBERs) were detected by hybridization in PAX5 human B cells associated with the PDX ( = 1/4) or with engrafted human immune cells at other anatomic locations ( = 4/11). One of the 4 mice had an EBV-associated human large B-cell lymphoma. NSG mice receiving xenotransplants can develop combinations of GvHD, EBV-driven PTLD, and B-cell lymphoma similar to those occurring in human pediatric patients. Therefore, pediatric xenotransplants should undergo histopathologic and immunohistochemical assessment upon collection to ensure that the specimen is not a lymphoma and does not contain lymphoma cells because these neoplasms can morphologically mimic small round blue cell pediatric solid tumors.
Topics: Animals; B-Lymphocytes; Biomarkers, Tumor; Cell Cycle Proteins; Epstein-Barr Virus Infections; Graft vs Host Disease; Heterografts; Humans; Leukocyte Common Antigens; Leukosialin; Lymphoma; Lymphoproliferative Disorders; Mice; Mice, Inbred NOD; Neoplasm Transplantation; T-Lymphocytes; Transplantation, Heterologous
PubMed: 32202225
DOI: 10.1177/0300985820913265 -
Alimentary Pharmacology & Therapeutics Apr 2020The management of Crohn's disease patients with perianal lesions and anti-TNF failure is challenging. (Observational Study)
Observational Study
BACKGROUND
The management of Crohn's disease patients with perianal lesions and anti-TNF failure is challenging.
AIMS
To assess the effectiveness of vedolizumab in perianal Crohn's disease and the predictors of success in a real-life cohort.
METHODS
We conducted a nationwide multicentre cohort study in patients with perianal Crohn's disease who received vedolizumab. In patients with active perianal Crohn's disease, the success of vedolizumab was defined by clinical success (no draining fistula at clinical examination and no anal ulcers for primary lesions) at 6 months without medical or surgical treatment for perianal Crohn's disease. Logistic regression analyses were performed to identify predictors of success. In patients with inactive perianal Crohn's disease, recurrence was defined by the occurrence of lesions and/or the need for medical or surgical treatments.
RESULTS
One hundred and fifty-one patients were included. Among them 102 patients had active perianal disease, 33 (32.4%) males, mean age 39.8 years, mean Crohn's disease duration 14.6 years; 101 (99%) had received at least one anti-TNF. The median follow-up time was 52 weeks. Sixty-eight per cent of patients discontinued therapy after a median time of 33 weeks. Vedolizumab success was reached in 23/102 (22.5%). Among patients with setons at initiation, 9/61(15%) had a successful removal. In multivariable analysis, factors associated with success were the number of prior biologic agents (≥3, odds ratio, OR: 0.20, 95% CI 0.04-0.98) and no antibiotics at initiation (OR: 4.76, 95% CI 1.25-18.19). In 49 patients with inactive perianal Crohn's disease, perianal disease recurred in 15/49 (30.6%), 11/49 (22.4%) needed dedicated treatments. Median time to recurrence was 22 weeks.
CONCLUSIONS
We identified a low rate of success of vedolizumab in patients with active perianal Crohn's disease, and nearly one third of patients with inactive perianal Crohn's disease had perianal recurrence. Further evaluation is warranted in prospective studies.
Topics: Adult; Animals; Antibodies, Monoclonal, Humanized; Anus Diseases; Cohort Studies; Crohn Disease; Female; France; Humans; Male; Middle Aged; Perianal Glands; Rectal Fistula; Recurrence; Treatment Outcome; Young Adult
PubMed: 32080886
DOI: 10.1111/apt.15665 -
Journal of the American Academy of... Oct 2020
Topics: Adolescent; Adult; Anus Diseases; Drug Eruptions; Dyspareunia; Female; Genital Diseases, Female; Genital Diseases, Male; Hemorrhage; Humans; Isotretinoin; Male; Middle Aged; Mucositis; Sebaceous Glands; Young Adult
PubMed: 32032695
DOI: 10.1016/j.jaad.2020.01.071 -
Annals of Coloproctology Dec 2019Hidradenoma papilliferum is a rare benign cystic tumor that originates from apocrine glands or anogenital mammary glands. Here, we describe 2 cases of hidradenoma...
Hidradenoma papilliferum is a rare benign cystic tumor that originates from apocrine glands or anogenital mammary glands. Here, we describe 2 cases of hidradenoma papilliferum of the anus. Two female patients aged 39 and 35 presented with perianal masses with hemorrhoids. The patients underwent hemorrhoidectomy and excision of the lesion. Histopathology confirmed the masses as hidradenoma papilliferum. The postoperative course was uneventful for both patients, and there were no recurrences after 18 and 12 months of follow-up, respectively. Proctologists should consider hidradenoma papilliferum in their differential diagnosis of benign anal tumors. Surgical excision is necessary for diagnosis and treatment of hidradenoma papilliferum.
PubMed: 31937076
DOI: 10.3393/ac.2018.08.03 -
Diseases of the Colon and Rectum Nov 2019A 66-year-old obese (BMI 34) diabetic man, a former smoker who had quit 4 months ago, had a 20-year history of perianal, inguinal, and gluteal hidradenitis suppurativa.... (Review)
Review
A 66-year-old obese (BMI 34) diabetic man, a former smoker who had quit 4 months ago, had a 20-year history of perianal, inguinal, and gluteal hidradenitis suppurativa. He presented to the office with persistent perianal and gluteal inflammation and drainage. He had had various surgical excisions of the affected area over the preceding 10 years. The patient was also following up with dermatology and was previously on doxycycline and infliximab with little improvement in his symptoms. On physical examination of his gluteal and perianal region, he had multiple areas of scarring and draining sinus tracts with significant induration ( and ). The patient underwent extensive unroofing of the sinus tracts and excision of active disease ().(Figure is included in full-text article.)(Figure is included in full-text article.)(Figure is included in full-text article.).
Topics: Aged; Anal Canal; Anti-Bacterial Agents; Apocrine Glands; Body Mass Index; Buttocks; Conservative Treatment; Diabetes Mellitus, Type 2; Diagnosis, Differential; Hidradenitis Suppurativa; Humans; Male; Obesity; Patient Care Management; Patient Selection; Surgical Procedures, Operative
PubMed: 31596759
DOI: 10.1097/DCR.0000000000001497 -
The Journal of Small Animal Practice Oct 2019To evaluate the diagnostic utility of individual cytological criteria and their best combination to differentiate benign from malignant perianal gland proliferative...
OBJECTIVES
To evaluate the diagnostic utility of individual cytological criteria and their best combination to differentiate benign from malignant perianal gland proliferative lesions in dogs.
MATERIALS AND METHODS
Retrospective study of cytological samples of canine perianal gland proliferative lesions that had subsequent histopathological confirmation.
RESULTS
Seventy-seven perianal gland nodules from 56 dogs were included. Histologically, lesions were diagnosed as hyperplasia (n=2), adenoma (n=53), epithelioma (n=6) and carcinoma (n=16). Of the 28 cytological criteria assessed, 13 showed promise for distinguishing benign from malignant lesions. A diagnostic algorithm with an 87% accuracy (sensitivity, 90.9%; specificity, 85.4%) was developed from these data.
CLINICAL SIGNIFICANCE
Cytological evaluation can provide useful information for presurgical differentiation between benign and malignant hepatoid gland proliferative lesions. The proposed algorithm must be validated and tested for reproducibility in further, preferably larger, series of cases.
Topics: Anal Gland Neoplasms; Animals; Carcinoma; Dog Diseases; Dogs; Perianal Glands; Reproducibility of Results; Retrospective Studies
PubMed: 31368201
DOI: 10.1111/jsap.13062 -
Annales de Dermatologie Et de... Sep 2019To specify the clinical, histopathological and immunochemical features in a large series of hidradenoma papilliferum patients and to point out similarities with breast...
AIM
To specify the clinical, histopathological and immunochemical features in a large series of hidradenoma papilliferum patients and to point out similarities with breast disease.
PATIENTS AND METHODS
We performed a monocentric retrospective histopathological study. Cases of hidradenoma papilliferum seen at the dermatopathology laboratory of Strasbourg (1977-2017) were included and analyzed with hematoxylin-eosin staining and immunostaining. Diagnosis was confirmed where tubular formations and papillary proliferations were noted and were lined by a luminal layer of columnar cells surrounded by a basal layer of myoepithelial cells.
RESULTS
We included 32 cases solely involving women of a mean age of 49 years. Most cases involved the vulva (67 %) or the perianal region (25 %). The lesions ranged in size from 3 to 25mm. They occurred as solitary, non-ulcerated, skin-colored and cyst-like lesions. They were mostly localized in the mid-dermis. We identified several peculiar morphological variants. Three HP had a structure resembling tubular apocrine papillary adenoma, 3 were similar to apocrine cystadenoma, while 9 HP, of which 3 involved epidermal hyperplasia, were connected with the overlying epidermis and had dense stromal infiltrate in plasma cells, closely resembling syringocystadenoma papilliferum. Sixteen tumors had no features of note. Similarities with breast tumors were revealed through columnar cell hyperplasia (6 cases), high mitotic rates (4 cases), and atypical nuclei (3 cases). Anogenital mammary-like glands were found in the immediate vicinity of 10 HP. Myoepithelial cells were immunoreactive for smooth muscle actin and p63, while luminal cells were immunoreactive for CK7, CK8, EMA, and, in a more focused and less consistent manner, for Ber-EP4, CD117, PHLDA1 and androgen receptors.
CONCLUSION
Our study highlighted a variety of patterns and cell morphologies, on occasion within a single tumor, identical to breast disease. Histological and immunochemical examination revealed that anogenital mammary-like glands and HP share common features, which militates in favor of a common histogenesis.
Topics: Anal Canal; Female; Humans; Immunohistochemistry; Mammary Glands, Human; Middle Aged; Perineum; Retrospective Studies; Sweat Gland Neoplasms; Tubular Sweat Gland Adenomas; Vulvar Neoplasms
PubMed: 31285043
DOI: 10.1016/j.annder.2019.03.008 -
Frontiers in Immunology 2019Adipose mesenchymal stem cells (ASC) are considered minimally immunogenic. This is due to the low expression of human leukocyte antigens I (HLA-I), lack of HLA-II...
Adipose mesenchymal stem cells (ASC) are considered minimally immunogenic. This is due to the low expression of human leukocyte antigens I (HLA-I), lack of HLA-II expression and low expression of co-stimulatory molecules such as CD40 and CD80. The low rate of observed immunological rejection as well as the immunomodulatory qualities, position ASC as a promising cell-based therapy for the treatment of a variety of inflammatory indications. Yet, few studies have addressed relevant aspects of immunogenicity such as ASC donor-to-patient HLA histocompatibility or assessment of immune response triggered by ASC administration, particularly in the cases of presensitization. The present study aims to assess allo-immune responses in a cohort of Crohn's disease patients administered with allogeneic ASC (darvadstrocel formerly Cx601) for the treatment of complex perianal fistulas. We identified donor-specific antibodies (DSA) generation in a proportion of patients and observed that patients showing preexisting immunity were prone to generating DSA after allogeneic therapy. Noteworthy, naïve patients generating DSA at week 12 (W12) showed a significant reduction in DSA titer at week 52 (W52), whereas DSA titer was reduced in pre-sensitized patients only with no specificities against the donor administered. Remarkably, we did not observe any correlation of DSA generation with ASC therapeutic efficacy. complement-dependent cytotoxicity (CDC) studies have revealed limited cytotoxic levels based upon HLA-I expression and binding capacity even in pro-inflammatory conditions. We sought to identify CDC coping mechanisms contributing to the limited cytotoxic killing observed in ASC . We found that ASC express membrane-bound complement regulatory proteins (mCRPs) CD55, CD46, and CD59 at basal levels, with CD46 more actively expressed in pro-inflammatory conditions. We demonstrated that CD46 is a main driver of CDC signaling; its depletion significantly enhances sensitivity of ASC to CDC. In summary, despite relatively high clearance, DSA generation may represent a major challenge for allogeneic cell therapy management. Sensitization may be a significant concern when evaluating re-treatment or multi-donor trials. It is still unknown whether DSA generation could potentially be the consequence of donor-to-patient interaction and, therefore, subsequently link to efficacy or biological activity. Lastly, we propose that CDC modulators such as CD46 could be used to ultimately link CDC specificity with allogeneic cell therapy efficacy.
Topics: Adipose Tissue; Adult; Animals; Cells, Cultured; Cohort Studies; Complement Activation; Crohn Disease; Female; Fistula; Graft Rejection; HLA Antigens; Humans; Immunity, Humoral; Immunization; Isoantigens; Male; Membrane Cofactor Protein; Mesenchymal Stem Cell Transplantation; Mesenchymal Stem Cells; Perianal Glands; Postoperative Complications; Transplantation, Homologous
PubMed: 31258526
DOI: 10.3389/fimmu.2019.01244 -
BMJ Case Reports Apr 2019Gynaecological infections are frequent in women, particularly in young women during their reproductive time. Anatomophysiologically, Bartholin's gland is greatly...
Gynaecological infections are frequent in women, particularly in young women during their reproductive time. Anatomophysiologically, Bartholin's gland is greatly susceptible to infections and is characterised by an inherent polymicrobial population. In fact, gynaecological microbiota has a set of agents coming from the perianal region that colonise the vagina and, in particular, Bartholin's gland. Taking this into account, infections caused by agents that do not belong to the genital microbiota are less frequent. Here, we describe a case of a 23-year-old young woman with an abscess in Bartholin's gland caused by .
Topics: Abscess; Anti-Bacterial Agents; Bartholin's Glands; Diagnosis, Differential; Drainage; Female; Humans; Pneumococcal Infections; Streptococcus pneumoniae; Treatment Outcome; Vulvar Diseases; Young Adult
PubMed: 31023734
DOI: 10.1136/bcr-2018-228492