Abstract：
:Patients with primary immunodeficiency are at increased risk for malignancy, especially hematologic neoplasms. This paper reports a unique case of a 47-year-old man with X-linked agammaglobulinemia who presented with progressive asymptomatic violaceous papules and plaques on his face, hands, and trunk for 1 year. Skin biopsies revealed deep, nodular infiltrates of histiocytes and CD8-positive lymphocytes, with a CD4:CD8 ratio of 1:10. Laboratory studies showed cytopenias. Flow cytometry in the skin, blood, and bone marrow (BM) showed a CD3+/CD8+/CD57+ large granular lymphocyte population. BM biopsy showed 30% involvement with these atypical T-cells. T-cell gene rearrangement studies of skin, blood, and BM revealed identical T-cell clones. He was diagnosed with T-large granular lymphocyte leukemia (T-LGLL) with an associated CD8+ cutaneous lymphoproliferation. Skin involvement was suspected to represent infiltration by T-LGLL. However, co-existence of two lymphoproliferative disorders (LPDs), T-LGLL and CD8+ granulomatous LPD, remains a possibility. In general, cutaneous infiltrates associated with LGLL are rare and poorly understood. It has been suggested that they are markers of poor prognosis. Our case report describes skin, blood, and BM findings in an immunosuppressed patient with T-LGLL in detail. These findings have not yet been reported and their significance requires further investigation.
journal_name
J Cutan Patholjournal_title
Journal of cutaneous pathologyauthors
Daou H,Hatch LA,Weinkle A,Morey GE,Messina J,Zhang X,Sokol L,Seminario-Vidal Ldoi
10.1111/cup.13860subject
Has Abstractpub_date
2020-09-03 00:00:00eissn
0303-6987issn
1600-0560pub_type
abstract：BACKGROUND:Even though only 33 cases of Erythromelanosis follicularis faciei et colli (EFFC) have been reported around the world, there have been no quantitative clinicopathological studies yet. The purpose of this study is to examine quantitatively clinical and histopathological evaluations of EFFC using image analysi...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1034/j.1600-0560.2001.028003160.x
更新日期：2001-03-01 00:00:00
abstract：:Penicillamine-induced cutaneous elastosis perforans serpiginosa associated with a large air-cyst in the right lung is described in a 29-year-old female patient with Wilson disease. Identical light and electron-microscopic changes were present in both dermal and pulmonary elastic tissue, suggesting a disseminated drug-...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/j.1600-0560.1979.tb01131.x
更新日期：1979-08-01 00:00:00
abstract：:Dermal non-neural granular cell tumor (NNGCT) was first described in 1991 as an S100-negative polypoid non-melanocytic tumor. Although originally introduced in the literature as a primary cutaneous tumor, it was later emphasized that such qualification could not be held until the line of differentiation was clarified....
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/cup.12926
更新日期：2017-06-01 00:00:00
abstract：BACKGROUND:p16 immunostaining has been used to aid and improve the histopathologic evaluation of equivocal cervical lesions with associated low-grade or high-grade dysplasia. However, the utility of p16 immunostaining in the diagnosis of atypical genital skin lesions remains debatable. METHODS:We conducted a cross-sec...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/cup.12525
更新日期：2015-08-01 00:00:00
abstract：:Oral melanoacanthoma is a rarely-reported, benign pigmented lesion of oral mucosa. Spongiosis was only recently mentioned as a light-microscopical finding occasionally seen in oral melanoacanthoma, and the formation of spongiotic intraepithelial vesicles to our knowledge has never been reported. The authors discuss a ...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/j.1600-0560.1989.tb00587.x
更新日期：1989-12-01 00:00:00
abstract：BACKGROUND:The distinction between ocular sebaceous carcinoma, poorly differentiated ocular squamous cell carcinoma and ocular basal cell carcinoma can be challenging. An appropriate immunohistochemical panel may help to differentiate these lesions. METHODS:To determine the distribution and use of several immunostains...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/j.1600-0560.2007.00871.x
更新日期：2008-07-01 00:00:00
abstract：:A 46-year-old man had a cystic mass on the right side of his scalp. Histological examination revealed a cystic dermal nodule composed of relatively circumscribed lobules of proliferating squamous epithelium, with atypical mitoses and dyskeratotic cells of invasive structure, which was diagnosed as proliferating tricho...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/j.1600-0560.2007.00960.x
更新日期：2008-10-01 00:00:00
abstract：BACKGROUND:The diagnosis of 'cellular dermal nodule in melanocytic nevi' is challenging, as it can simulate invasive and tumorigenic melanoma. Because cellular nodules are presumed benign, it is essential to differentiate them from true malignant melanoma arising within a nevus, which portends a poor prognosis. Althoug...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/j.0303-6987.2004.00156.x
更新日期：2004-02-01 00:00:00
abstract：:Pigmented extramammary Paget disease (PEMPD) is a rare intraepithelial carcinoma which can clinically resemble other pigmented neoplasms. Similarities to melanoma on dermoscopy, histopathology, and reflectance confocal microscopy (RCM) increase the risk of misdiagnosis and, consequently, mismanagement. Here, we descri...
journal_title：Journal of cutaneous pathology
pub_type：
doi：10.1111/cup.13804
更新日期：2020-12-01 00:00:00
abstract：:Post-transplant lymphoproliferative disorder (PTLD) is a lymphoid proliferation that develops as a complication of solid organ or bone marrow transplants. PTLD limited to the skin is very rare. Plasmacytoma-like PTLD is an uncommon variant of monomorphic PTLD. Its presentation in the skin is extraordinary with very fe...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章,评审
doi：10.1111/j.1600-0560.2012.01919.x
更新日期：2012-07-01 00:00:00
abstract：:We report on two patients with chronic myeloid leukemia (CML) who presented blastic transformation involving the skin, with leukemic infiltrates showing unusual morphologic and immunohistologic characteristics. Both patients were elderly men with a 36-month and a 40-month history of CML, respectively. They presented w...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/j.1600-0560.1999.tb01796.x
更新日期：1999-11-01 00:00:00
abstract：:Post-vaccinial non-viral folliculitis has been recognized in the past decade as a new adverse cutaneous reaction to smallpox vaccination. Contrary to more serious smallpox vaccine reactions, post-vaccinial non-viral folliculitis has a benign course and resolves spontaneously within approximately 7 days. We describe ad...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/cup.12064
更新日期：2013-03-01 00:00:00
abstract：:This is the first report of spinous and follicular lichen nitidus associated with perifollicular granuloma formation. Clinicians and pathologists need to distinguish this stage of the disease from lichen scrofulosorum. ...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/j.1600-0560.1988.tb00553.x
更新日期：1988-08-01 00:00:00
abstract：BACKGROUND:The early stages of follicular mycosis fungoides (FMF) have not been described previously in the literature. OBJECTIVE:Our goal was to better categorize the clinicopathologic features of early stages of FMF. METHODS:The clinical notes of patients with a diagnosis of FMF seen during the previous 5 years wer...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/cup.12399
更新日期：2015-03-01 00:00:00
abstract：BACKGROUND:The importance of changes in the supporting tumor stroma for cancer initiation and progression is well established. The characteristics of an activated tumor stroma, however, are not completely understood. In an effort to better characterize the desmoplastic response to human skin tumors, we evaluated the ex...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/j.0303-6987.2006.00446.x
更新日期：2006-02-01 00:00:00
abstract：:Proliferation of the eccrine sweat duct epithelium has been associated with skin tumors, especially keratoacanthomas and basal cell carcinomas. We report our observations on the extensive sweat gland changes in a patient who had idiopathic follicular mucinosis. ...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/j.1600-0560.1987.tb00495.x
更新日期：1987-06-01 00:00:00
abstract：:The lesions of nine patients with early striae distensae (SD) during puberty were examined by light and electron microscopy. Specific changes were seen in very early stage SD, and in clinically uninvolved skin 0.5 to 3 cm remote from the edge of the long axis of the SD lesions. Sequential changes of elastolysis accomp...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/j.1600-0560.1991.tb01376.x
更新日期：1991-12-01 00:00:00
abstract：:The characteristic histologic change seen at 2 h in the Rumpel-Leede (Hess) test is, in addition to focal hemorrhage in the upper dermis, a perivascular lymphocyte infiltrate with focal areas of lymphocytic epidermal invasion. The histologic findings, even with the liquefaction degeneration, are similar to those of pu...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/j.1600-0560.1979.tb00300.x
更新日期：1979-02-01 00:00:00
abstract：:The low-fat and fat-free spindle cell lipomas (SCLs) are rare and often mistaken for other benign and malignant morphological mimics, because of the fact that the diagnosis relies on its non-lipogenic component analysis. Here, we report the clinicopathological features of two oral SCLs (low-fat and fat-free variants)....
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章,评审
doi：10.1111/cup.13512
更新日期：2019-10-01 00:00:00
abstract：:Five cases of superficial epithelioma with sebaceous differentiation (SESD) are reported. They occurred as solitary papules on the face of 5 patients, aged 57 to 72. The tumor is characterized by a superficial platelike proliferation of basaloid to squamoid cells with broad attachments to the overlying epidermis. Clus...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/j.1600-0560.1987.tb01331.x
更新日期：1987-08-01 00:00:00
abstract：:Infundibulocystic basal cell carcinoma (IBCC) is a relatively recently described variant of basal cell carcinoma that is controversial and not universally accepted. Excluding cases of nevoid basal cell carcinoma syndrome, IBCC usually presents as a small, solitary, superficial lesion on the face of older persons. Ther...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1034/j.1600-0560.2000.027006316.x
更新日期：2000-07-01 00:00:00
abstract：BACKGROUND:Cardiac angiosarcoma is a rare tumor that has a predilection for middle-aged males and a marked predominance in the right atrium. The tumor may present abruptly with a fulminant clinical course. Initial presentation with metastatic disease is rare. Only one case has been reported of a patient who presented w...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1034/j.1600-0560.2001.028005265.x
更新日期：2001-05-01 00:00:00
abstract：:We carried out a quantitative study of the matrix and dermal papilla of the human hair follicle of the scalp, both normal and in various degrees of androgenetic alopecia. A stereological study showed the measured parameters to decrease with increase in the degree of alopecia, particularly as regards the total volume o...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/j.1600-0560.1993.tb01273.x
更新日期：1993-08-01 00:00:00
abstract：:Malignant transformation occurred in pre-existing sweat gland tumors in 7 patients. Three lesions showed an histologic pattern of eccrine spiradenoma, 2 eccrine poroma, one cylindroma and one papillary eccrine adenoma. Malignant transformation was histologically characterized by the presence of solid tumor areas popul...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/j.1600-0560.1987.tb00122.x
更新日期：1987-02-01 00:00:00
abstract：:Lichen aureus is a rare disease of unknown origin that is classified under the group of pigmented purpuric dermatoses. Its most important differential diagnosis is both clinically and histologically mycosis fungoides, into which the disease can proceed in very rare cases. We describe an unusual multilocular lichen aur...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/j.1600-0560.2007.00918.x
更新日期：2008-10-01 00:00:00
abstract：:Disseminated bacillus Calmette-Guérin infection (BCGitis) is an uncommon condition which is usually associated with primary immunodeficiency. Skin histopathology findings have been described in rare cases only. A retrospective clinicopathological study was performed to assess the potential utility of skin biopsies in ...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/cup.12048
更新日期：2013-01-01 00:00:00
abstract：:We report a 20-year-old male patient with an atypical Spitz tumor, located in the dorsal aspect of his left hand, and a positive sentinel axillary lymph node. After lymphadenectomy, 1 of 23 non-sentinel lymph nodes excised was found to contain small multiple deposits of large spindle atypical melanocytes. Reviewing th...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章,评审
doi：10.1111/j.1600-0560.2008.01080.x
更新日期：2009-05-01 00:00:00
abstract：:Electron microscopy of lentigines was performed to study the pigmentation abnormality in two children with LEOPARD syndrome. Giant melanosomes similar to those seen in café-au-lait spots of neurofibromatosis and nevus spilus were found in a lentigine from one of our cases. Our results show that "spherical granular mel...
journal_title：Journal of cutaneous pathology
pub_type： 杂志文章
doi：10.1111/j.1600-0560.1976.tb00865.x
更新日期：1976-01-01 00:00:00
abstract：BACKGROUND:Cutaneous leishmaniasis displays considerable variation in its histopathological and clinical presentation. Clinically, it progresses from a papule into a painless ulcerated and crusted nodule/papule. Microscopically, it progresses from sheets of amastigote-filled histiocytes to granulomatous inflammation. ...
journal_title：Journal of cutaneous pathology
pub_type： 临床试验,杂志文章,多中心研究
doi：10.1111/j.1600-0560.2011.01844.x
更新日期：2012-02-01 00:00:00
abstract：:Rare epithelial structures in benign nerve sheath tumors are almost always glandular in appearance. We describe a case of intraoral plexiform schwannoma with concurrent squamous epithelial hyperplasia. The lesion occurred as a pigmented nodule on the gingiva of a 35-year-old woman with no systemic involvement. Histolo...
journal_title：Journal of cutaneous pathology
pub_type：
doi：10.1111/cup.12579
更新日期：2015-12-01 00:00:00
