Familial Mottled Pigmentation: Challenge
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Clinical Pathologic Challenge Source Type: research

A Slow-Growing Papule on the Right Shin: Challenge
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Clinical Pathologic Challenge Source Type: research

Superficial Suppurative Granulomatous Folliculitis and Pigmented Infundibular Spores and Hyphae: Challenge
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Clinical Pathologic Challenge Source Type: research

Multiple Comedonal Plaques of Both Ears With Hearing Loss: Challenge
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Clinical Pathologic Challenge Source Type: research

Pautrier Microabscess: A Topic of Great Controversy
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Letters to the Editor Source Type: research

Two Rare Cases of Facial Vitiligo-Like Hypopigmentation With Upper Dermal Elastic Fibers Degeneration
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Letters to the Editor Source Type: research

Aberrant Melan-A Expression in Extramammary Paget Disease
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Letters to the Editor Source Type: research

Familial Mottled Pigmentation: Answer
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Clinical Pathologic Challenge Answer Source Type: research

A Slow-Growing Papule on the Right Shin: Answer
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Clinical Pathologic Challenge Answer Source Type: research

Superficial Suppurative Granulomatous Folliculitis and Pigmented Infundibular Spores and Hyphae: Answer
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Clinical Pathologic Challenge Answer Source Type: research

Multiple Comedonal Plaques of Both Ears With Hearing Loss: Answer
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Clinical Pathologic Challenge Answer Source Type: research

Microcystic Adnexal Adenoma: The Benign Counterpart of Microcystic Adnexal Carcinoma
We present here a lesion in which the histopathologic study showed a well-demarcated nodular lesion involving the mid dermis and composed of small cystic keratinous structures, solid aggregates of pale squamous cells without cytologic atypia and ductal structures. Although these neoplastic components resembled those of MAC, the sharp delimitation of the lesion as well as the absence of deep extension and perineural involvement supported the benign nature of this lesion. We have named this neoplasm microcystic adnexal adenoma, as the benign counterpart of MAC. (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Extraordinary Case Report Source Type: research

Immunohistochemical ALK Expression in Granular Cell Atypical Fibroxanthoma: A Diagnostic Pitfall for ALK-Rearranged Non-neural Granular Cell Tumor
Abstract: Atypical fibroxanthoma (AFX) is a neoplasm that most commonly occurs on sun-damaged skin of the head and neck in elderly patients and that usually exhibits indolent clinical behavior with complete excision. The granular cell variant of AFX demonstrates overlapping histopathologic features with dermal non-neural granular cell tumor (NNGCT), which typically arises on the extremities of young to middle aged adults with rare reports of regional metastasis. A subset of NNGCT harbors ALK rearrangements and expresses ALK by immunohistochemistry. Here, we present 2 cases of granular cell AFX occurring on the scalp of...
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Extraordinary Case Report Source Type: research

Ciliated and Mucinous Adenomatous Syringometaplasia: The Missing Link of the “Ciliated Eccrine Metaplasia” Theory in the Histogenesis of Cutaneous Eccrine Ciliated Cyst?
Abstract: A variety of metaplastic changes has been reported in eccrine ducts and glands with squamous and mucinous syringometaplasia representing the most common histopathologic patterns. In the current case, a previously unreported variant of syringometaplasia was described in a female newborn with a 2-cm occipital cutaneous defect consistent with cutis aplasia. Over a more than 1-year period of local treatment, an enlarging plaque associated with local alopecia developed at the site of the original ulcerated area. A local excision demonstrated a superficial hypertrophic scar associated with a complete loss of hair f...
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Extraordinary Case Report Source Type: research

Occupational Localized Cutaneous Argyria With Pseudo-Ochronosis in a Jeweler
Abstract: A case of localized argyria in a 36-year-old female jeweler is described who presented with 2 discrete and asymptomatic bluish–black pigmented macules on the pulp of her left middle finger. A skin biopsy from both lesions demonstrated deposition of brown/black pigmented granules along the basement membrane zone of eccrine glands, blood vessels, nerves, and the dermo-epidermal junction fully in keeping with silver deposition. In addition, there was yellow–brown deposition seen within the interstitial dermis mimicking an early form of ochronosis, so called “pseudo-ochronosis.” This latte...
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Extraordinary Case Report Source Type: research

Case of Cutaneous Metastatic Prostate Carcinoma as Initial Evidence of Neuroendocrine Differentiation
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Brief Report Source Type: research

Immunophenotypic Differences in Tumor-Infiltrating Lymphocytes and Neovascularization Between Primary Cutaneous Melanoma With and Without Metastasis: An Immunohistochemical Study of 80 Cases
Abstract: The prognostic implications of the immunophenotype of the tumor-infiltrating lymphocytes (TILs) in primary cutaneous melanoma are well known. In recent years, the study of this immunophenotype has also resulted in immunotherapeutic consequences. The aims of this study were to characterize the subpopulations of TILs in primary cutaneous melanoma, in cases with and without metastasis, as well as the neovascularization associated with the primary neoplasm, and its influence on the development of metastasis. To this end, the immunophenotype of TILs and the neovascularization of 80 patients with primary cutaneous ...
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Original Study Source Type: research

Seborrheic Keratosis With Sebaceous Differentiation, a Series of 8 Cases and Critical Review of the Literature
Abstract: Seborrheic keratosis with sebaceous differentiation (SKSD) can sometimes raise uncertainty, confuse with other even malignant entities, and lead to overestimation of this harmless variant. Retrospective analysis of the cases diagnosed as SKSD and a search of the pertaining literature were conducted. Eight cases of SKSD were found. Histologically, these lesions show a flat to plate-like outgrowth of basaloid cells with solitary or clustered sebocytes at the bottom of the rete ridges and variable sebaceous ducts with luminal cuticula. The lesions differed in the outgrowth subpattern: flat/macular, acanthotic, o...
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Original Study Source Type: research

Preferentially Expressed Antigen in Melanoma Immunostaining in a Series of Melanocytic Neoplasms
We report our observations of PRAME staining in 253 melanocytic tumors. Tumors were classified by hematoxylin and eosin sections. The nuclear PRAME staining of neoplastic melanocytes in each case was categorized as absent, focally present, or diffusely present. The results were compared with those of Lezcano et al 105 of 134 (78%) melanocytic nevi were completely PRAME negative. Of the 29 PRAME-positive benign lesions, 28 exhibited focal but not diffuse positivity, including atypical (n = 11) and dysplastic nevi (n = 11). One of 11 Spitz nevi showed diffuse positivity (9%). Thirty-nine of 51 (76%) invasive melanomas, 41 of...
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Original Study Source Type: research

Desmoplastic Trichoepithelioma With Pseudocarcinomatous Hyperplasia: A Folliculosebaceous Neoplasm in Young Persons
Abstract: Pseudocarcinomatous desmoplastic trichoepithelioma (PDTE) features verrucous squamous epidermal hyperplasia with a jagged undersurface overlying cords of follicular germinative cells in a fibrotic stroma. To date, only 5 cases have been reported. We identified 7 new PDTEs from 2 institutions and reviewed their clinical manifestations and immunohistochemical profile. The median age was 14 years (range 8–34 years). New findings included vacuolization of the basal layer of the pseudocarcinomatous surface epithelium, and the frequent presence of singly distributed sebocytes within the cords of basaloid cell...
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Original Study Source Type: research

Comparison of Immunohistochemical Expression of Cytokeratin 19, c-KIT, BerEP4, GATA3, and NUTM1 Between Porocarcinoma and Squamous Cell Carcinoma
This study sought to confirm the diagnostic utility of cytokeratin 19, c-KIT, BerEP4, GATA3, and NUTM1 immunohistochemistry in distinguishing porocarcinoma from SCC. Immunohistochemical analysis of cytokeratin 19, c-KIT, BerEP4, GATA3, and NUTM1 in 14 porocarcinomas and 22 SCCs was performed; the extents and intensities of expression of these markers were recorded. The statistical associations of the immunoexpression between porocarcinoma and SCC were analyzed using the Pearson χ2 test. Cytokeratin 19 was positive in 13 (92.9%) of 14 porocarcinomas, and for all the positive cases, staining was strong and evident in>...
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Original Study Source Type: research

Pure and Mixed Desmoplastic Melanomas: A Retrospective Clinicopathologic Comparison of 33 Cases
Conclusions: Pure and mixed variants of DM were found to have similar clinicopathologic characteristics. Patients with the mixed histologic subtype were more likely to have mitoses, but no difference in the therapeutic management or patient survival was seen between the 2 subtypes. (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Original Study Source Type: research

Chronic Itch of Unknown Origin Is Associated With an Enhanced Th2 Skin Immune Profile
Abstract: Chronic pruritus of unknown origin (CPUO) is described as chronic itch lasting longer than 6 weeks in the absence of a defined skin rash and any known causative disease process. A retrospective study was performed on biopsy samples from patients with CPUO and normal controls to compare the immune profiles of these patients with healthy individuals. We used dual CD3/T-bet and CD3/GATA3 immunohistochemical staining to assess for T-cells expressing Th1 versus Th2 transcription factors, respectively. Our data showed that CD3+ cells of patients with CPUO co-express significantly more GATA3 compared with normal con...
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: Original Study Source Type: research

Sebaceous Carcinomas: A Clinicopathological Comparison of Ocular and Extraocular Variants
In this study we evaluated the clinical and the histopathology of ocular and extraocular SC to confirm the difference between them. We performed a retrospective review of SC in which we studied the clinical and histomorphologic features of 106 cases, including 39 cases of ocular SC and 67 cases of extraocular SC. Only 2/67 cases of extraocular SC had multiple recurrences and none of them metastasized as opposed to our cases of ocular SC wherein 21/39 cases were locally aggressive with multiple recurrences and 5 cases metastasized. Histologically, both neoplasms showed major distinct morphologic features including poor diff...
Source: The American Journal of Dermatopathology - October 25, 2021 Category: Pathology Tags: CME Article Source Type: research

Case Report—Indurated Plaques and Recurrent Fevers in a 37-Year-Old Man: Challenge
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Clinical Pathologic Challenge Source Type: research

Cluster of Pruritic Papules Refractory to Numerous Treatments: Challenge
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Clinical Pathologic Challenge Source Type: research

Disseminated Pruritic Papules in an Adult Male: Challenge
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Clinical Pathologic Challenge Source Type: research

Indolent, Painful, and Dystrophic Nail Unit Tumor in an Elderly Man
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Clinical Pathologic Challenge Source Type: research

Reevaluation of GLI1 Expression in Skin Tumors
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Letters to the Editor Source Type: research

Neurocristic Cutaneous Hamartoma With Perineuriomatous Differentiation: Can It Be Distinguished From Perineuriomatous Melanocytic Nevi?
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Letters to the Editor Source Type: research

Erythema Elevatum Diutinum Appearing Within Old Scar Tissue
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Letters to the Editor Source Type: research

Case Report—Indurated Plaques and Recurrent Fevers in a 37-Year-Old Man: Answer
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Clinical Pathologic Challenge Answer Source Type: research

Cluster of Pruritic Papules Refractory to Numerous Treatments: Answer
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Clinical Pathologic Challenge Answer Source Type: research

Disseminated Pruritic Papules in an Adult Male: Answer
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Clinical Pathologic Challenge Answer Source Type: research

Indolent, Painful, and Dystrophic Nail Unit Tumor in an Elderly Man: Answer
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Clinical Pathologic Challenge Answer Source Type: research

Papillary Dermal Elastolysis Secondary to Combination Nivolumab and Cabiralizumab Therapy: Histiocytes and Dermal Mucin as Clues to the Diagnosis
We report a third patient with distinctive, generalized atrophic macules that developed after a morbilliform eruption during a clinical trial for treatment of metastatic pancreatic adenocarcinoma. Histopathological findings demonstrated diminished elastic fibers in the papillary dermis, associated with a histiocyte-rich infiltrate and increased dermal mucin, features that should clue the dermatopathologist to this condition. (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Extraordinary Case Report Source Type: research

Fortuitous Eradication of an Aggressive Basal Cell Carcinoma Via Foreign Body Reaction to a Polyurethane Vacuum-Assisted Closure Sponge
Abstract: The foreign body reaction (FBR) is a well-documented immune reaction. Much of the literature on FBRs has focused on minimizing this immune response to mitigate the impact on medical implants. Here, we present a case that illustrates a serendipitous oncologic outcome from an FBR. A 54-year-old man presented with an aggressive basal cell carcinoma (BCC). At the first resection, he had broadly positive surgical margins. The surgical wound was temporized with a polyurethane wound vacuum assisted closure (VAC) device. He was lost to follow-up having retained a VAC sponge for a total of 12 weeks. A wide re-resectio...
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Extraordinary Case Report Source Type: research

Combining Reflective Confocal Microscopy and Dynamic Optical Coherence Tomography to Diagnose Melanoacanthoma: Case Report
We present a case of a 38-year-old man with Fitzpatrick skin type IV with an asymmetric black papule showing clinical and dermoscopic features of both melanoma and SK. Reflectance confocal microscopy (RCM) and dynamic optical coherence tomography (d-OCT) were used for further evaluation. RCM revealed acanthotic epidermis with a mixed honeycomb and cobblestone pattern, polycyclic bulbous rete ridges, and bright plump cells within entrapped, edged, dermal papillae, compatible with pigmented SK. Also noted were a population of fairly uniform bright dendritic cells scattered quite evenly at all levels of the epidermis and the ...
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Extraordinary Case Report Source Type: research

Acral Calcified Vascular Leiomyoma: Report of 3 Cases and Literature Review
We report 3 examples of this unusual condition on the feet of 2 male and one female subjects (aged, 68, 69, and 80 years) and make a review of the 31 cases available in the literature. Two of our cases are associated with highly uncommon features, such as transepidermal calcium elimination and concomitant calcaneal spur. (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Extraordinary Case Report Source Type: research

A Case Report of Small Lymphocytic Lymphoma Mimicking Primary Cutaneous Marginal Zone Lymphoma With Plasmacytic Differentiation
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Brief Report Source Type: research

Diagnostic Utility of C4d by Direct Immunofluorescence in Bullous Pemphigoid
In conclusion, the current study shows that C4d is a more sensitive but not a 100% specific marker of BP. We conclude that C4d by DIF could be an interesting diagnostic adjunct for BP, particularly in IgG−/C3c− double negative cases. (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Brief Report Source Type: research

Characteristic Clinicopathological Features of Secondary Extramammary Paget Disease With Underlying Anorectal Adenocarcinoma: Evenly Circumferential Perianal Distribution, Fibroepithelioma of Pinkus-like Changes, and Subepidermal Mucin Deposits Without Invasive Tumor Cells
This study aimed to identify the clinical and histopathological characteristics of secondary extramammary Paget disease (EMPD) with underlying anorectal adenocarcinoma so as to differentiate it from primary cutaneous EMPD. Seventeen and 8 cases of primary and secondary EMPD with anorectal adenocarcinoma, respectively, were retrieved from the pathology archive and the clinical and histopathological features reviewed. The tumor samples from 21 cases were totally resected specimens, whereas 3 and 1 of secondary and primary cases were punch biopsied, respectively. All 8 (100%) cases of secondary EMPD presented evenly distribut...
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Original Study Source Type: research

Evolution of Dupilumab-Associated Cutaneous Atypical Lymphoid Infiltrates
Conclusion: These cases highlight the transformation of lymphoid infiltrates after dupilumab treatment for AD and emphasize the importance of clinical and histopathologic evaluation before and during treatment with dupilumab for treatment-refractory presumed AD. (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Original Study Source Type: research

Histological and Immunohistochemical Changes of Congenital Melanocytic Nevi With Age
Abstract: Clinical but not histological changes of congenital melanocytic nevi (CMN) with age are well characterized. Our objective was to analyze histological changes of CMN with age and discuss possible clinical implications of our findings. We investigated serial excisions of 21 patients with CMN and compared histological and immunohistochemical features over time. The median number of serial excisions was 6 [interquartile range (IQR) 5–7], the median age at the first excision was 12 months (IQR 5–98), and the median time between the first and last analyzed excision was 53 months (IQR 45–64). The p...
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Original Study Source Type: research

Image Morphometric Analysis of B Cells and Plasma Cells in Erythema Nodosum Leprosum With Clinicopathological Correlation
Abstract: Erythema nodosum leprosum (ENL) occurs as an immunological complication of multibacillary leprosy (MBL). The pathogenesis of ENL is long considered to be a T-cell–mediated process. The role of B cells and plasma cells in ENL is not well described in the literature. Therefore, we investigated the B-cell and plasma cell infiltrates in the skin biopsies of biopsy-proven cases of ENL by immunohistochemistry and image morphometry and compared the result with paucibacillary leprosy and MBL. Moreover, we sought a correlation of the B-cell and plasma cell infiltrates with different clinical, hematological, hist...
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Original Study Source Type: research

Immunoglobulin and Complement Immunohistochemistry on Paraffin Sections in Autoimmune Bullous Diseases: A Systematic Review and Meta-analysis
Abstract: Immunohistochemistry (IHC) on formalin-fixed, paraffin-embedded tissue has been proposed as a potential tool in the diagnosis of autoimmune bullous diseases (AIBDs) in lieu of standard direct immunofluorescence (DIF) microscopy. To comprehensively determine the diagnostic accuracy of immunoglobulin and complement IHC for diagnosis of AIBDs, we conducted a systematic review and multivariate Bayesian model-based meta-analysis of the literature. Quality and heterogeneity assessment of studies was performed using the Quality Assessment of Diagnostic Accuracy Studies (QUADAS-2) checklist and the I2 index, respecti...
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: Review Article Source Type: research

In Memoriam: N. Scott McNutt, MD
No abstract available (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: In Memoriam Source Type: research

Characterization of Morphologic and Cytochemical Staining Properties of Exogenous Materials Mimicking Fungal Organisms Encountered in Skin Biopsies
Conclusion: We present a comprehensive characterization of the morphologic and cytochemical staining properties of multiple exogenous materials that may contaminate a skin biopsy. This characterization will aid the pathologist by providing a mechanism to identify potential contaminants in skin biopsies. (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - September 25, 2021 Category: Pathology Tags: CME Article Source Type: research

Nervous Nipple: Pseudopolythelia Caused by a Neurofibroma of the Areola
We report a case of a solitary neurofibroma (by itself rare in the areola) mimicking a second, twin nipple. In this case, these 2 rare conditions merge resulting in pseudopolythelia. The relevant literature on polythelia and neurofibromas of the breast is briefly reviewed. (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - August 28, 2021 Category: Pathology Tags: Extraordinary Case Report Source Type: research

Solitary CD8-Positive Primary Cutaneous Peripheral T-Cell Lymphoma: A Question of Classification
We report a case of an ulcerated solitary cutaneous lesion arising on the forehead of an adult man, which had a CD8(+) cytotoxic phenotype and areas of dot-like CD68 positivity, but with a number of features that significantly detracted from the classically described acral CD8(+) lymphoma. The nosological status of the lesion is discussed with respect to a preferred diagnosis of peripheral T-cell lymphoma, not otherwise specified. (Source: The American Journal of Dermatopathology)
Source: The American Journal of Dermatopathology - August 28, 2021 Category: Pathology Tags: Extraordinary Case Report Source Type: research