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Archives of Dermatological Research May 2024Serpentine supravenous hyperpigmentation (SSH) describes increased skin pigmentation that develops in the area immediately overlying the vessels through which... (Review)
Review
Serpentine supravenous hyperpigmentation (SSH) describes increased skin pigmentation that develops in the area immediately overlying the vessels through which chemotherapeutic drugs are administered. While SSH can be cosmetically distressing and there are no definitive management options, the literature is severely limited and the variations in clinical presentation, risk factors, and histopathology of SSH across patients are not well understood. We aimed to systematically summarize characteristics from current available data, and thus improve SSH awareness and management. A literature search was conducted in PubMed using specific eligibility criteria through the end of December 2022. Included articles focused on patients who experienced SSH after chemotherapy infusion. Study quality was assessed using a modified Oxford Centre for Evidence-Based Medicine quality rating scheme. Of the 41 articles identified by literature search, 24 met eligibility criteria. Two additional articles were identified through the reference sections of retrieved articles, for 26 articles total. All articles were case reports, representing 28 patients total. Locations of SSH were mostly in the forearm near the site of injection (85%), and the most common associated symptom was erythema. Histopathologic analysis was available for half of cases, the majority of which were inflammatory in nature. The most common inflammatory pattern observed was a vacuolar/lichenoid interface dermatitis. Duration of SSH ranged from days to > 1 year after the chemotherapy was stopped. Six (21%) patients were managed with topical steroids and oral vasodilators, six (21%) patients switched to central venous infusion rather than peripheral infusion, five (18%) patients received only supportive care, three (11%) patients received venous washing with chemotherapy, three (11%) patients stopped chemotherapy, and one (4%) patient reduced the chemotherapy dosage. Ten (36%) patients attained complete resolution, seven (25%) had SSH that was near resolution/fading, and three (11%) had persistent hyperpigmentation. Although SSH often spontaneously resolves once the chemotherapeutic agent is stopped, it can persist in some patients and cause significant distress. As the literature is severely limited and there are no definitive treatments, additional research using more standardized definitions and methods of assessments is necessary to improve characterization of SSH and evaluate potential interventions.
Topics: Humans; Hyperpigmentation; Antineoplastic Agents; Skin Pigmentation; Skin; Erythema
PubMed: 38787426
DOI: 10.1007/s00403-024-03057-2 -
Oxford Medical Case Reports May 2024Dyskeratosis congenita (DKC) is a rare genetic disorder characterized by lacy reticular skin hyperpigmentation, bone marrow failure, nail dystrophy, and oral...
Dyskeratosis congenita (DKC) is a rare genetic disorder characterized by lacy reticular skin hyperpigmentation, bone marrow failure, nail dystrophy, and oral leukoplakia. To the best of our knowledge, only around 200 cases were reported in the medical literature, and in this report, we present another distinctive case from Syria. This case report describes a male patient with generalized reticular pigmentation and abnormal nails since childhood. The patient reported a history of recurrent urethral stenosis and corneal density. Dermoscopic examination revealed pigmented lines arranged in a netlike pattern. Histopathological findings were nonspecific. Hematological values were unremarkable. A contrast CT scan revealed changes in the bladder wall. The final diagnosis of Dyskeratosis Congenita was made based on the clinical criteria. This disorder can present with additional cutaneous manifestations and systemic complications. Treatment are generally prescribed to maintain bone marrow function, based on the fact that it is the major cause of death. Regular monitoring and screening for associated conditions are recommended.
PubMed: 38784779
DOI: 10.1093/omcr/omae049 -
BMC Oral Health May 2024The ceramic soft tissue trimming bur (CeraTip™) was initially introduced for use in gingivoplasty but has recently been used for gingival depigmentation. The aim of... (Randomized Controlled Trial)
Randomized Controlled Trial
BACKGROUND
The ceramic soft tissue trimming bur (CeraTip™) was initially introduced for use in gingivoplasty but has recently been used for gingival depigmentation. The aim of this study is to compare the efficacy of depigmentation between the novel CeraTip™ and the gold-standard surgical scalpel technique.
METHODS
Eight healthy, nonsmokers with moderate to severe gingival hyperpigmentation in both arches were randomly assigned for CeraTip™ depigmentation in one arch as the test group (TG) and scalpel depigmentation in the opposite arch as the control group (CG). Pigmentation indices were used to assess clinical performance. Treatment time, pain level, and esthetic satisfaction were the parameters of patient experience. The assessments were performed at baseline, one week, one month, and three months.
RESULTS
At all assessment visits, pigmentation intensity represented by the Dummet oral pigmentation index (DOPI), and pigmentation distribution represented by the Hedin melanin index (MI), were significantly lower than those at baseline (p < 0.001) in both groups. When comparing the two groups, Scalpel depigmentation had better initial clinical outcomes, while CeraTip™ had less visible repigmentation, pain scores, treatment time, and greater esthetic satisfaction. However, none of the differences were statistically significant.
CONCLUSION
Both techniques successfully removed gingival hyperpigmentation with comparable clinical performance. The patients preferred CeraTip™ depigmentation.
TRIAL REGISTRATION
The study protocol was registered on 11/09/2023 on the www.
CLINICALTRIALS
gov database (NCT06031116) after the approval of the Ethics Committee, Faculty of Dentistry, Ain Shams University (FDASU-Rec012124).
Topics: Humans; Female; Adult; Patient Satisfaction; Gingival Diseases; Male; Ceramics; Hyperpigmentation; Esthetics, Dental; Middle Aged; Gingiva; Treatment Outcome; Gingivoplasty
PubMed: 38783312
DOI: 10.1186/s12903-024-04345-z -
Cureus May 2024Hyperpigmentation of the skin can occur due to internal and external causes. This case highlights an unusual presentation of generalized acute hyperpigmentation...
Hyperpigmentation of the skin can occur due to internal and external causes. This case highlights an unusual presentation of generalized acute hyperpigmentation associated with subclinical hypothyroidism in a 42-year-old Indian American woman. After unsuccessful trials of various topical agents, the patient exhibited significant improvement in hyperpigmentation after levothyroxine treatment. Improvements included lightening in the bilateral antecubital fossa, axillae, and neck regions. This case underscores the importance of considering thyroid dysfunction as a potential factor that may contribute to atypical pigmentation disorders.
PubMed: 38779438
DOI: 10.7759/cureus.60708 -
Clinical and Translational Medicine May 2024Melanocyte stem cells (MSCs), melanocyte lineage-specific skin stem cells derived from the neural crest, are observed in the mammalian hair follicle, the epidermis or... (Review)
Review
Melanocyte stem cells (MSCs), melanocyte lineage-specific skin stem cells derived from the neural crest, are observed in the mammalian hair follicle, the epidermis or the sweat gland. MSCs differentiate into mature melanin-producing melanocytes, which confer skin and hair pigmentation and uphold vital skin functions. In controlling and coordinating the homeostasis, repair and regeneration of skin tissue, MSCs play a vital role. Decreased numbers or impaired functions of MSCs are closely associated with the development and therapy of many skin conditions, such as hair graying, vitiligo, wound healing and melanoma. With the advancement of stem cell technology, the relevant features of MSCs have been further elaborated. In this review, we provide an exhaustive overview of cutaneous MSCs and highlight the latest advances in MSC research. A better understanding of the biological characteristics and micro-environmental regulatory mechanisms of MSCs will help to improve clinical applications in regenerative medicine, skin pigmentation disorders and cancer therapy. KEY POINTS: This review provides a concise summary of the origin, biological characteristics, homeostatic maintenance and therapeutic potential of cutaneous MSCs. The role and potential application value of MSCs in skin pigmentation disorders are discussed. The significance of single-cell RNA sequencing, CRISPR-Cas9 technology and practical models in MSCs research is highlighted.
Topics: Humans; Melanocytes; Homeostasis; Skin; Stem Cells; Animals; Cell Differentiation
PubMed: 38778457
DOI: 10.1002/ctm2.1720 -
Archives of Dermatological Research May 2024
Meta-Analysis
Topics: Vitiligo; Humans; Treatment Outcome; Cell- and Tissue-Based Therapy
PubMed: 38775972
DOI: 10.1007/s00403-024-02920-6 -
Archives of Dermatological Research May 2024Many individuals with vitiligo are uncertain about their skin cancer risk, phototherapy risks, and recommended sun protective practices. This study examined the...
Many individuals with vitiligo are uncertain about their skin cancer risk, phototherapy risks, and recommended sun protective practices. This study examined the perceived skin cancer risk and sun protective practices among individuals living with vitiligo. A secondary objective was to understand where participants obtain this information. This was a prospective cross-sectional study. An online survey was distributed to vitiligo support group leaders globally who shared the survey with their members. Individuals over the age of 18 and with vitiligo were included. There were 209 survey respondents, the majority were between the ages 35-54 (45.5%, n = 95), female (70.8%, n = 148), White (66.0%, n = 138). Nearly half of respondents believed they were at increased risk of skin cancer because of their vitiligo (45.5%, n = 95) and nearly a quarter (22.5%, n = 47) believed that phototherapy increased their risk of skin cancer. Having vitiligo affected sun protective practices with less than a quarter (24.4%, n = 51) of respondents using sunscreen daily or often prior to their vitiligo diagnosis in comparison to the majority of respondents (60.3%, n = 126) using it after their vitiligo diagnosis. The three most common sources where patients obtained information were the internet and social media (46.4%, n = 97), vitiligo support groups (23.4%, n = 49), and dermatologists (20.6%, n = 43). Despite evidence indicating a decreased risk of skin cancer in individuals with vitiligo and supporting the safety of narrowband ultraviolet B phototherapy, many participants believed they were at an increased risk of skin cancer. Findings were sub-stratified and showed differences in sunscreen usage based on gender, skin color, and percent depigmentation. This study also found nearly half of respondents obtained information related to vitiligo from the internet and social media. The number of participants may limit the generalizability of the findings. Survey questionnaires are also subject to response bias. The findings from this study highlight demographic variations in sunscreen usage which may help guide the development of targeted interventions to improve sun protective behaviors among diverse populations with vitiligo. In addition, this study suggests certain sun protective practices and skin cancer risk perceptions may vary based on extent of depigmentation. Lastly, this study also demonstrates the internet and social media as a popular source for obtaining information, emphasizing the need for dermatologists to leverage various online communication channels to help disseminate accurate information.
Topics: Humans; Vitiligo; Female; Cross-Sectional Studies; Male; Skin Neoplasms; Adult; Prospective Studies; Middle Aged; Sunscreening Agents; Health Knowledge, Attitudes, Practice; Surveys and Questionnaires; Young Adult; Aged; Sunburn; Risk Factors; Sunlight
PubMed: 38775848
DOI: 10.1007/s00403-024-02942-0 -
Revista Da Associacao Medica Brasileira... 2024Cellular and humoral immunity plays a role in the pathogenesis of vitiligo. T lymphocytes and natural killer cells involved in cellular immunity carry out their...
OBJECTIVE
Cellular and humoral immunity plays a role in the pathogenesis of vitiligo. T lymphocytes and natural killer cells involved in cellular immunity carry out their cytotoxic activities through perforin/granzyme-dependent granule exocytosis, in which granulysin and cathepsin-L are also involved. The aim of this study was to investigate the possible role of serum granulysin and cathepsin-L in the etiopathogenesis of vitiligo and their association with disease activity and severity.
METHODS
This randomized, prospective case-control study was conducted with 46 vitiligo patients admitted to the hospital for vitiligo between January and November 2021 and 46 healthy volunteers of similar age and gender. Serum levels of granulysin and cathepsin-L were measured by the enzyme-linked immunosorbent assay method.
RESULTS
The mean serum levels of granulysin and cathepsin-L were statistically significantly higher in vitiligo patients compared with the control group (p=0.048 and p=0.024, respectively). There was no statistically significant correlation between serum granulysin and serum cathepsin-L levels and disease severity in the patient group (r=0.30, p=0.062 and r=0.268, p=0.071, respectively). Disease activity also showed no significant association with serum granulysin and cathepsin-L levels (p=0.986 and p=0.962, respectively).
CONCLUSION
Although granulysin and cathepsin-L are molecules involved in the pathogenesis of vitiligo, the use of these molecules may not be helpful in assessing disease activity and severity. It may be helpful to conduct comprehensive and prospective studies to find new molecules to fill the gap in this area.
Topics: Humans; Vitiligo; Female; Male; Antigens, Differentiation, T-Lymphocyte; Adult; Case-Control Studies; Prospective Studies; Severity of Illness Index; Young Adult; Middle Aged; Cathepsin L; Enzyme-Linked Immunosorbent Assay; Adolescent; Biomarkers
PubMed: 38775500
DOI: 10.1590/1806-9282.20231107 -
BMC Neuroscience May 2024Incontinentia pigmenti (IP) is a rare neuroectodermal dysplasia caused by a defect in the IKBKG gene. The pathogenesis of central nervous system injury is believed to be...
INTRODUCTION
Incontinentia pigmenti (IP) is a rare neuroectodermal dysplasia caused by a defect in the IKBKG gene. The pathogenesis of central nervous system injury is believed to be related to microvascular ischemia. Currently, few treatment strategies are available for the inflammatory phase.
MATERIALS AND METHODS
This retrospective descriptive analysis included the clinical data of 41 children with IP collected from 2007 to 2021 in Xi'an, China, comprising clinical characteristics, imaging findings, blood cell analysis, skin histopathology, and genetic data.
RESULTS
Fourteen children (34%) aged 4 days to 5 months exhibited clinical signs and symptoms, including convulsions, delayed psychomotor development following neurological damage, and revealed significant MRI abnormalities, including ischemia, hypoxia, cerebral hypoperfusion, hemorrhage, encephalomalacia, and cerebral atrophy. Eight of the 24 patients (33%) presented with retinal vascular tortuosity and telangiectasis, accompanied by neovascularization and hemorrhage. Thirty-eight children (93%) had elevated eosinophils (mean: 3.63 ± 4.46 × 10), and 28 children (68%) had significantly elevated platelets (mean: 420.16 ± 179.43 × 10). Histopathology of skin revealed microvascular extravasation and vasodilation with perivascular and intravascular eosinophilic infiltration.
CONCLUSION
Brain injury in IP occurs during infancy until 5 months of age, which is also the acute dermatitis phase accompanied by eosinophilia and an increased platelet count. This study provides evidence of microvascular damage to the skin and fundus during the inflammatory phase. The mechanism of microvascular damage may be similar to that in the brain.
Topics: Humans; Incontinentia Pigmenti; Infant; Female; Retrospective Studies; Male; China; Infant, Newborn; Magnetic Resonance Imaging; Brain; Child, Preschool; East Asian People
PubMed: 38773385
DOI: 10.1186/s12868-024-00872-1 -
Clinical Case Reports May 2024Unlike most cases, the lesions were localized to the dorsum of the hand, lacked pruritus (itching), and did not exhibit "sperm-like blood vessels," which are typically...
KEY CLINICAL MESSAGE
Unlike most cases, the lesions were localized to the dorsum of the hand, lacked pruritus (itching), and did not exhibit "sperm-like blood vessels," which are typically pathognomonic to classical MF.
ABSTRACT
The study presents a rare case involving a 44-year-old woman who developed a skin condition on the base of her left thumb. Initially misdiagnosed as pigmented purpura, the need for further investigation arose to determine the nature of the condition accurately. The medical evaluation encompassed a comprehensive analysis of the patient's skin ailment. A series of diagnostic examinations were conducted to ascertain the underlying cause. Although routine blood tests yielded unremarkable results, the distinct characteristics of the rash prompted a more thorough investigation. Subsequent assessment revealed that the skin condition was not pigmented purpura, as initially presumed, but rather a manifestation of cutaneous T-cell lymphoma (CTCL) known as mycosis fungoides (MF). MF is an infrequent lymphoma predominantly affecting individuals aged 45-65, exhibiting a male-to-female sex ratio of 2:1. The annual incidence of MF ranges from 0.3 to 0.96 cases per 100,000 individuals. The woman's skin exhibited discrete patches adorned with colored dots, progressively thickening and pigmentation. Notably, the absence of pruritus did not dispel suspicion. This case underscores the significance of accurately diagnosing uncommon dermatological disorders to facilitate appropriate medical intervention. The unique appearance of the rash and its distinctive features, despite normal blood results, enabled the identification of MF. The patient's treatment encompassed a combination of steroids and narrowband UV therapy. Vigilance, continued research, and heightened awareness are paramount for early intervention and improved patient outcomes. Such efforts contribute to an enhanced understanding of the complexities of this condition.
PubMed: 38770416
DOI: 10.1002/ccr3.8847