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Article ; Online: Oral sirolimus for the treatment of juvenile xanthogranuloma: Report of two pediatric cases.

Toker, Michelle / Hassonjee, Fatema Esaa / Amodio, John / Edelman, Morris C / Emeghebo, Kristina I / Levy, Carolyn Fein / Shaigany, Sheila

Pediatric dermatology

2024

Abstract: Juvenile xanthogranuloma (JXG) with extensive cutaneous or visceral organ involvement is often associated with high morbidity and treatment commonly involves surgical excision, radiotherapy, systemic steroids, or chemotherapy. Sirolimus, a mammalian ... ...

Abstract	Juvenile xanthogranuloma (JXG) with extensive cutaneous or visceral organ involvement is often associated with high morbidity and treatment commonly involves surgical excision, radiotherapy, systemic steroids, or chemotherapy. Sirolimus, a mammalian target of rapamycin (mTOR) inhibitor, is an oral antitumor and immunosuppressive therapy used to treat various neoplastic disorders, including histiocytic disorders. We report two pediatric cases of JXG successfully treated with oral sirolimus monotherapy, and postulate that sirolimus may induce rapid disease resolution and long-term remission for patients with both skin-limited and multisystemic JXG. Our findings warrant further investigation of the relationship between the mTOR pathway and JXG.
Language	English
Publishing date	2024-03-05
Publishing country	United States
Document type	Case Reports
ZDB-ID	605539-4
ISSN	1525-1470 ; 0736-8046
ISSN (online)	1525-1470
ISSN	0736-8046
DOI	10.1111/pde.15579
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Article ; Online: Outcome Measurement Instruments Used to Evaluate Dermatologic Adverse Events in Cancer Trials: A Systematic Review.

Shaigany, Sheila / Mastacouris, Nicole / Tannenbaum, Rachel / Strunk, Andrew / Luan, Christopher / Burshtein, Joshua / Burshtein, Aaron / Carvajal, Richard / Garg, Amit / Alloo, Allireza

JAMA dermatology

2024

Abstract: Importance: Assessment of type, severity, and impact of dermatologic adverse events (DAEs) necessitates well-developed and validated clinician-reported outcome measures (ClinROMs) and patient-reported outcome measures (PROMs) that evaluate concepts ... ...

Abstract	Importance: Assessment of type, severity, and impact of dermatologic adverse events (DAEs) necessitates well-developed and validated clinician-reported outcome measures (ClinROMs) and patient-reported outcome measures (PROMs) that evaluate concepts specific to mucocutaneous toxic effects and that allow appropriate interpretation and comparison of DAEs across trials. Objective: To evaluate heterogeneity and quality of ClinROMs and PROMs used to assess DAEs from systemic cancer therapy. Evidence review: Two systematic reviews were conducted by searching PubMed and Embase databases from inception through March 7, 2023, and April 12, 2023. The first search included randomized clinical trials and observational studies reporting systemic cancer treatment-induced DAEs assessed by a ClinROM or PROM. The second included studies evaluating measurement properties of frequently used ClinROM and PROM instruments. The Consensus-Based Standards for the Selection of Health Measurement Instruments risk of bias tool was used to evaluate methodologic quality of validation assessments. Findings: A total of 395 studies were included. The Common Terminology Criteria for Adverse Events (CTCAE) was utilized in 331 studies meeting inclusion criteria (83.8%). At least 1 skin-related PROM was infrequently utilized in systemic chemotherapy clinical trials (79 studies [20.0%]). Most frequently utilized PROMs were the Dermatology Life Quality Index (DLQI; 34 studies [8.6%]) and Skindex-16 (20 studies [5.1%]). Among studies capturing DAEs, 115 (29.1%) reported a nondescript term (ie, rash) as the only DAE. Eight studies described 44 property assessments of the CTCAE, DLQI, and Skindex. There were no studies evaluating content validity, intrarater reliability, or measurement error for the CTCAE, DLQI, or Skindex. There were no studies evaluating structural validity, internal consistency, and responsiveness of DLQI or Skindex. Interrater reliability and responsiveness were each assessed for 1 DAE-related component of the CTCAE. Construct validity for CTCAE, DLQI, and Skindex was evaluated in 29 (65.9%), 3 (6.8%), and 9 (20.5%) assessments, respectively. Conclusions and relevance: In this systematic review, there was a narrow spectrum of ClinROMs and PROMs with limited validity for the measurement of DAEs in the context of systemic chemotherapy interventions in clinical trials. Report of trial DAEs often had low morphologic specificity and meaning. Based on existing gaps in measurement and report of DAEs, a frequent and impactful adverse event to chemotherapy, the framework for evaluating cutaneous toxic effects in oncology trials may need collaborative reevaluation.
Language	English
Publishing date	2024-03-20
Publishing country	United States
Document type	Journal Article
ZDB-ID	2701761-8
ISSN	2168-6084 ; 2168-6068
ISSN (online)	2168-6084
ISSN	2168-6068
DOI	10.1001/jamadermatol.2024.0053
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Article ; Online: Comparison between organismal staining on histology and tissue culture in the diagnosis of cutaneous infection: A retrospective study.

Shaigany, Sheila / Steuer, Alexa / Seminara, Nicole / Brinster, Nooshin / Femia, Alisa

Journal of the American Academy of Dermatology

2020 Volume 82, Issue 6, Page(s) 1400–1408

Abstract: Background: In instances of suspected cutaneous infection, the standard of care includes obtaining skin biopsy specimens for histology and tissue culture. Few studies have compared the clinical utility of each test.: Objective: To assess the ... ...

Abstract	Background: In instances of suspected cutaneous infection, the standard of care includes obtaining skin biopsy specimens for histology and tissue culture. Few studies have compared the clinical utility of each test. Objective: To assess the concordance of results between tissue culture and histology, as well as the clinicopathologic features that may influence the diagnostic yield of each test. Methods: A retrospective review of all patients who underwent skin biopsy for histology and tissue culture at New York University from 2013 through 2018. Results: Of 179 patients, 10% had positive concordance, 21% had positive tissue culture only, and 7% had positive histology only. We calculated a kappa correlation coefficient of 0.25 between histology and tissue culture (reference, 0.21-0.39 indicates minimal agreement). Histology exhibited higher sensitivity in detecting fungi, whereas tissue culture was more sensitive in identifying Gram-negative bacteria. Antimicrobial use before biopsy led to significantly fewer positive cultures (37.5% vs 71%; P = .023) in patients ultimately diagnosed with infection. Limitations: This study was conducted at a single institution, thereby restricting its broad applicability. The lack of a validated criterion standard to diagnose infection also limits interpretation of the results. Conclusion: Tissue culture and histopathology often yield discordant results. Dermatologists should recognize specific limitations, yet high clinical utility in special circumstances, of tests when approaching cases of suspected infection.
MeSH term(s)	Adult ; Aged ; Biopsy ; Female ; Humans ; Male ; Microbiological Techniques/methods ; Middle Aged ; Retrospective Studies ; Skin Diseases, Infectious/diagnosis ; Skin Diseases, Infectious/microbiology ; Skin Diseases, Infectious/pathology ; Staining and Labeling ; Tissue Culture Techniques
Language	English
Publishing date	2020-01-28
Publishing country	United States
Document type	Comparative Study ; Journal Article
ZDB-ID	603641-7
ISSN	1097-6787 ; 0190-9622
ISSN (online)	1097-6787
ISSN	0190-9622
DOI	10.1016/j.jaad.2020.01.047
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Article ; Online: Lack of distinct molecular profile of Primary Dermal Melanoma.

Shaigany, Sheila / Tessier-Cloutier, Basile / Busam, Klaus J / Horst, Basil A

Human pathology

2020 Volume 106, Page(s) 32–38

Abstract: Primary dermal melanoma (PDM) is a rare variant of melanoma which simulates metastatic melanoma to the skin. Diagnosis of PDM cannot be established on histologic grounds alone but requires absence of evidence of melanoma elsewhere by clinical history and/ ...

Abstract	Primary dermal melanoma (PDM) is a rare variant of melanoma which simulates metastatic melanoma to the skin. Diagnosis of PDM cannot be established on histologic grounds alone but requires absence of evidence of melanoma elsewhere by clinical history and/or imaging studies. Despite this entity being clinically well documented, limited data on molecular characterization are available. We performed comprehensive mutation and copy number variation analysis in a series of PDMs in search for distinctive molecular features.Studies were approved by respective institutional review boards. Six cases fulfilling strict histologic criteria of PDM were identified in patients with absent history of melanoma elsewhere, negative sentinel lymph node biopsies and imaging studies. DNA was extracted from formalin-fixed, paraffin-embedded (FFPE) tissue, and five cases passed quality control measures and were subjected to targeted exon sequencing using the Memorial Sloan Kettering-Integrated Mutation Profiling of Actionable Cancer Targets 410 panel. Two of five PDM carried NRAS hotspot mutations characteristic of cutaneous melanoma (CM) genomic subtypes. One case showed a probable low-activating NRAS mutation in combination with additional aberrations in other mitogen-activated protein kinase (MAPK) pathway effectors. Hotspot mutations were also identified in the TERT promoter region, and one tumor showed a mutation in the SWI/SNF remodeling complex. No oncogenic mutations were identified in one case. Furthermore, none of the tumors analyzed showed activating mutations in Gα subunits, including GNAQ and GNA11. Copy number alterations included deletions of Chr 9p, characteristic of CM.Despite PDM showing mutational heterogeneity, our findings suggest predominance of MAPK pathway aberrations in agreement with the mutational profile of CMs in general. Given the absence of genetic overlap with other distinct primary dermal melanocytic proliferations, mutational profiling will unlikely aid in the difficult differential diagnosis of PDM versus melanoma metastasis. Thorough metastatic workup remains crucial in establishing this rare diagnosis.
MeSH term(s)	Adult ; Aged ; Biomarkers, Tumor/genetics ; Biopsy ; Chromosomal Proteins, Non-Histone/genetics ; DNA Copy Number Variations ; DNA Mutational Analysis ; Female ; GTP Phosphohydrolases/genetics ; Gene Dosage ; Gene Expression Profiling ; Genetic Heterogeneity ; Genetic Predisposition to Disease ; High-Throughput Nucleotide Sequencing ; Humans ; Male ; Melanoma/genetics ; Melanoma/pathology ; Membrane Proteins/genetics ; Molecular Diagnostic Techniques ; Mutation ; Phenotype ; Predictive Value of Tests ; Skin Neoplasms/genetics ; Skin Neoplasms/pathology ; Telomerase/genetics ; Transcription Factors/genetics ; Transcriptome
Chemical Substances	Biomarkers, Tumor ; Chromosomal Proteins, Non-Histone ; Membrane Proteins ; SWI-SNF-B chromatin-remodeling complex ; Transcription Factors ; TERT protein, human (EC 2.7.7.49) ; Telomerase (EC 2.7.7.49) ; GTP Phosphohydrolases (EC 3.6.1.-) ; NRAS protein, human (EC 3.6.1.-)
Language	English
Publishing date	2020-09-15
Publishing country	United States
Document type	Journal Article ; Research Support, Non-U.S. Gov't
ZDB-ID	207657-3
ISSN	1532-8392 ; 0046-8177
ISSN (online)	1532-8392
ISSN	0046-8177
DOI	10.1016/j.humpath.2020.09.002
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Article ; Online: Enlarging red papulonodule on the chest.

Shaigany, Sheila / Simpson, Cory L / Micheletti, Robert G

Cutis

2018 Volume 101, Issue 2, Page(s) 78;117;118

MeSH term(s)	Biopsy ; Carcinoma, Renal Cell/secondary ; Carcinoma, Renal Cell/surgery ; Erythema ; Fatal Outcome ; Humans ; Kidney Neoplasms/pathology ; Kidney Neoplasms/surgery ; Male ; Middle Aged ; Nephrectomy ; Skin Neoplasms/secondary ; Thorax
Language	English
Publishing date	2018-03-19
Publishing country	United States
Document type	Case Reports ; Journal Article
ZDB-ID	391840-3
ISSN	2326-6929 ; 0011-4162 ; 0151-9522
ISSN (online)	2326-6929
ISSN	0011-4162 ; 0151-9522
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Article ; Online: Diagnostic work-up and treatment in patients with pyoderma gangrenosum: retrospective analysis of US insurance claims-based data.

Shaigany, Sheila / Wong, Priscilla W / Caplan, Avrom / Kim, Randie H / Femia, Alisa

Archives of dermatological research

2021

Abstract: Pyoderma gangrenosum (PG) is a rare, and often challenging to diagnose, inflammatory disorder with relatively high rates of morbidity and mortality. Central to the diagnosis of PG is histologic evaluation and exclusion of other entities. Large-scale ... ...

Abstract	Pyoderma gangrenosum (PG) is a rare, and often challenging to diagnose, inflammatory disorder with relatively high rates of morbidity and mortality. Central to the diagnosis of PG is histologic evaluation and exclusion of other entities. Large-scale studies investigating the proportion of patients receiving a thorough diagnostic work-up, as well as prevalence studies regarding comorbidities and systemic treatment in PG using claims-based data, are sparse. Our objective was to identify patients diagnosed with PG and describe the diagnostic work-up and prevalence of common comorbidities and therapies in this population using claims-based data in a retrospective cohort study. In order to better understand practices of diagnostic work-up, we captured rates of skin biopsy, tissue culture, and/or surgical debridement prior to initial diagnosis. We also identified the prevalence of PG-associated comorbidities and initial immunosuppressive therapy given for PG. Of the 565 patients diagnosed with PG, 9.4% underwent skin biopsy, 8% tissue culture, and 1.4% both skin biopsy AND tissue culture prior to diagnosis. Inflammatory bowel disease was the most prevalent comorbidity (16.3%). The most common treatment administered was systemic corticosteroids (17%). Although practice guidelines explicitly delineate histology and exclusion of infection as important diagnostic criteria, only a minority of patients in this study underwent skin biopsy and/or tissue culture prior to receiving a diagnosis of PG, suggesting that patients may receive a diagnosis of PG without having tissue evaluation. Such discordance between practice guidelines and "real-world" practice inevitably increases the risk for misdiagnosis of PG and misdirected treatment with immunosuppressants for presumptive PG in cases of PG mimickers. Moreover, comorbidities associated with PG may occur, or be identified in, a lower proportion of patients as compared with what is reported in the existing literature. Study limitations include a population restricted to < 65 years with commercial insurance and the reliance upon ICD diagnostic coding to capture the population.
Language	English
Publishing date	2021-10-29
Publishing country	Germany
Document type	Journal Article
ZDB-ID	130131-7
ISSN	1432-069X ; 0340-3696
ISSN (online)	1432-069X
ISSN	0340-3696
DOI	10.1007/s00403-021-02278-z
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Article ; Online: Mucocutaneous manifestations among adults hospitalized with multisystem inflammatory syndrome following SARS-CoV-2 infection.

Tannenbaum, Rachel / Rekhtman, Sergey / Strunk, Andrew / Birabaharan, Morgan / Shaigany, Sheila / Burshtein, Joshua / Grbic, Nicole / Nazir, Zaeem / Norden, Alexandra / Godfred-Cato, Shana / Belay, Ermias / Patel, Pragna / Garg, Amit

JAAD international

2022 Volume 6, Page(s) 111–113

Language	English
Publishing date	2022-01-24
Publishing country	United States
Document type	Journal Article
ISSN	2666-3287
ISSN (online)	2666-3287
DOI	10.1016/j.jdin.2022.01.005
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Article ; Online: An adult with Kawasaki-like multisystem inflammatory syndrome associated with COVID-19.

Shaigany, Sheila / Gnirke, Marlis / Guttmann, Allison / Chong, Hong / Meehan, Shane / Raabe, Vanessa / Louie, Eddie / Solitar, Bruce / Femia, Alisa

Lancet (London, England)

2020 Volume 396, Issue 10246, Page(s) e8–e10

MeSH term(s)	COVID-19 ; Coronavirus Infections/complications ; Humans ; Male ; Middle Aged ; Mucocutaneous Lymph Node Syndrome/etiology ; Pandemics ; Pneumonia, Viral/complications ; Systemic Inflammatory Response Syndrome/etiology
Keywords	covid19
Language	English
Publishing date	2020-07-10
Publishing country	England
Document type	Case Reports ; Letter
ZDB-ID	3306-6
ISSN	1474-547X ; 0023-7507 ; 0140-6736
ISSN (online)	1474-547X
ISSN	0023-7507 ; 0140-6736
DOI	10.1016/S0140-6736(20)31526-9
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Article: Herpetic zoster folliculitis in the immunocompromised host.

Shaigany, Sheila / Dabela, Ellen / Husain, Sameera / Grossman, Marc E

JAAD case reports

2015 Volume 1, Issue 1, Page(s) 38–40

Language	English
Publishing date	2015-01-12
Publishing country	United States
Document type	Case Reports
ZDB-ID	2834220-3
ISSN	2352-5126
ISSN	2352-5126
DOI	10.1016/j.jdcr.2014.11.003
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Article: Resident Rounds Part III: Case Report: Fatal Cryptococcal Panniculitis in a Lung Transplant Recipient.

Reddy, Bobby Y / Shaigany, Sheila / Schulman, Lawrence / Grossman, Marc E

Journal of drugs in dermatology : JDD

2015 Volume 14, Issue 5, Page(s) 519–522

Abstract: Cryptococcal panniculitis is a rare entity previously reported in only 13 solid organ transplant (SOT) recipients. Cutaneous cryptococcosis in SOT recipients warrants extensive systemic workup and treatment as if central nervous system (CNS) disease is ... ...

Abstract	Cryptococcal panniculitis is a rare entity previously reported in only 13 solid organ transplant (SOT) recipients. Cutaneous cryptococcosis in SOT recipients warrants extensive systemic workup and treatment as if central nervous system (CNS) disease is present. It should be included in the differential diagnosis of panniculitis in the immunocompromised host, as early diagnosis and treatment are critical. We report a fatal case of cryptococcal panniculitis in a 44-year-old lung transplant recipient.
MeSH term(s)	Adult ; Cryptococcosis/diagnosis ; Cryptococcosis/pathology ; Dermatomycoses/diagnosis ; Dermatomycoses/microbiology ; Dermatomycoses/pathology ; Diagnosis, Differential ; Fatal Outcome ; Humans ; Immunocompromised Host ; Lung Transplantation/methods ; Male ; Panniculitis/diagnosis ; Panniculitis/microbiology ; Panniculitis/pathology
Language	English
Publishing date	2015-05
Publishing country	United States
Document type	Case Reports ; Journal Article
ZDB-ID	2145090-0
ISSN	1545-9616
ISSN	1545-9616
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