Carolina Alexander-Savino, Alexandra Boye-Doe, Edith Bowers, Anne Beaven
{"title":"Oral Mycosis Fungoides: Report of 2 Cases and Review of the Literature.","authors":"Carolina Alexander-Savino, Alexandra Boye-Doe, Edith Bowers, Anne Beaven","doi":"10.1159/000530935","DOIUrl":"10.1159/000530935","url":null,"abstract":"<p><p>Oral and laryngeal cutaneous T-cell lymphoma (CTCL) is rare and usually associated with poor prognosis. Here, we discuss 2 cases of oral CTCL that developed in heavily pretreated patients and provide a review of the literature. The first case is of a 46-year-old African American male with rapidly progressive disease, presenting with a lesion on his hard palate 6 months after being diagnosed with a CD4+CD8+ CTCL. His cutaneous disease was widespread with tumors on >80% of his body surface area. Unfortunately, the patient died 2 ½ years after his CTCL diagnosis and 7 months after developing the oral CTCL lesion. The second case is of a 38-year-old African American male with stage IIb CD3+CD4+CD30+ mycosis fungoides (MF), who developed a tumor on the hard palate 6 months after diagnosis. He received palliative radiation to the oral lesion and multiple lines of systemic therapy for pulmonary, laryngeal, esophageal, and gastric involvement. Biopsy of the gastric lesions showed a CD30+ T-cell lymphoma with the same clonal peak as in his skin but with large cell transformation. Brentuximab vendoin was started, and the patient is now in complete remission, 30 months later. From the 76 cases of oral CTCL that have been reported in the English language, six were of transformed MF. The most common sites affected were the tongue and palate, and the most common presentation were erythematous or ulcerated tumors, plaques, or nodules associated with dysphagia and pain. Oral CTCL typically occurs years after the initial diagnosis of CTCL and portend a poor prognosis with an average survival of just over 1 year after development of oral lesions.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":null,"pages":null},"PeriodicalIF":0.9,"publicationDate":"2023-07-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/bc/2c/cde-2023-0015-0001-530935.PMC10368101.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9880219","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"An Uncommon Presentation of Neonatal Lupus Erythematosus: A Case Report.","authors":"Sara Al Janahi, Raghda Al Maashari","doi":"10.1159/000530465","DOIUrl":"10.1159/000530465","url":null,"abstract":"<p><p>Neonatal lupus erythematosus (NLE) is a rare autoimmune disorder of newborns and infants, born to usually asymptomatic mothers with lupus erythematosus. Clinical manifestations include variable cutaneous findings, with possible cardiac or hepatic involvement. We present a case of a 3-month-old baby girl with NLE, born to an asymptomatic mother. Her atypical clinical presentation included hypopigmented atrophic scars on the temples. She improved with topical pimecrolimus cream, with almost complete resolution of the facial lesions and improvement in atrophy noted at the 4-month follow-up visit. Cutaneous findings of hypopigmentation and atrophic scarring are less commonly reported. To our knowledge, no similar cases have been published in the Middle East. We aim to share this interesting case, highlight the different clinical presentations of NLE and raise awareness among physicians about this variable phenotype of NLE for timely diagnosis of this uncommon entity.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":null,"pages":null},"PeriodicalIF":0.9,"publicationDate":"2023-06-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10294211/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9714521","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Granulomatous Pigmented Purpuric Dermatosis in a Patient with (Inactive) Myasthenia Gravis: A Case Report and Review of the Literature.","authors":"Apirada Dhekariyapak, Penpun Wattanakrai","doi":"10.1159/000530034","DOIUrl":"10.1159/000530034","url":null,"abstract":"<p><p>Granulomatous pigmented purpuric dermatosis (GPPD) is a rare histologic variant of pigmented purpuric dermatosis (PPD) characterized by dermal histiocyte-rich interstitial infiltration with or without granuloma formation in addition to the other typical features of PPD. GPPD was previously observed more frequently to affect Asians and was reported to be associated with dyslipidemia. However, our literature search of 45 documented GPPD cases revealed an increasing prevalence in Caucasians in addition to dyslipidemia and associated autoimmune diseases. To date, etiopathogenesis of GPPD is unknown but may involve dyslipidemia, genetic and immunological factors such as autoimmune dysregulation or a sarcoidal reaction associated with <i>C. acnes</i>. GPPD is usually persistent and recalcitrant to treatments. We report a case of GPPD in a 57-year-old Thai woman with underlying myasthenia gravis who presented with a pruritic eruption on both lower legs. After treatment with 0.05% clobetasol propionate cream and oral colchicine, the lesion improved with marked flattening and disappeared with residual postinflammatory hyperpigmentation. We provide a literature review of the epidemiology, etiopathogenesis, concomitant comorbidities, clinical symptoms, dermatoscopic features, and treatments of GPPD.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":null,"pages":null},"PeriodicalIF":0.9,"publicationDate":"2023-05-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10293941/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9719351","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Kasper Køhler Alsing, Ole Olsen, Christian Bender Koch, Rasmus Hvass Hansen, Daniel Pergament Persson, Klaus Qvortrup, Jørgen Serup
{"title":"MRI-Induced Neurosensory Events in Decorative Black Tattoos: Study by Advanced Experimental Methods.","authors":"Kasper Køhler Alsing, Ole Olsen, Christian Bender Koch, Rasmus Hvass Hansen, Daniel Pergament Persson, Klaus Qvortrup, Jørgen Serup","doi":"10.1159/000530220","DOIUrl":"10.1159/000530220","url":null,"abstract":"<p><p>Adverse reactions in tattooed skin during magnetic resonance imaging (MRI) are rare but well known. Previous reports describe sudden burning pain in tattooed skin, sometimes accompanied by mild erythema and oedema when entering MRI scanners. The pathophysiology remains unclear, but simple direct thermal heating can be excluded. It has been hypothesized that MRI-triggered torque and traction create neural sensations from magnetic pigment particles. However, this case enlightens yet another possible mechanism. We present a 35-year-old woman experiencing reoccurring stinging sensations in three decorative black tattoos just seconds after the initiation of the MRI. Single-blind tests with handheld power magnets or a dummy could reproduce painful subjective feelings in her tattooed skin. Similar events were provoked during re-evaluation with MRI. Surprisingly, chemical analyses and electron microscopy of skin samples revealed carbon black as the colouring agent - no iron-based solids were detected. Our case demonstrates that MRI tattoo reactions are not limited to magnetic contaminants alone. More distinct subgroups of MRI-induced reactions may occur. We hypothesize that radiofrequency induction of surface currents in black carbon particles adjacent to sensory axons in the dermis may lead to neurosensations.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":null,"pages":null},"PeriodicalIF":0.9,"publicationDate":"2023-05-25","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10620551/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"48126684","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Almuntsrbellah Almudimeegh, Mohammed Habib, Omar Alsuhaibani, Nawaf Alkhudhayri
{"title":"Unilateral Lichen Planus Pigmentosus with Blaschko's Line Distribution: A Case Report.","authors":"Almuntsrbellah Almudimeegh, Mohammed Habib, Omar Alsuhaibani, Nawaf Alkhudhayri","doi":"10.1159/000529632","DOIUrl":"10.1159/000529632","url":null,"abstract":"<p><p>Lichen planus pigmentosus (LPP) is a distinctive variant of lichen planus described for the first time in 1974. A wide range of presentations and distribution patterns have been reported, mainly presenting as dark brown hyperpigmented macules and patches, typically involving sun-exposed areas such as the face, neck, and flexural folds. It is more common in dark-skinned patients with a female predominance. Infrequently, LPP may present as a Blaschkoid distribution, with only a few cases reported in the medical literature. We report an unusual case of LPP that developed with a unilateral Blaschkoid distribution on the left side of the trunk in a 32-year-old female following weight reduction surgery.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":null,"pages":null},"PeriodicalIF":0.9,"publicationDate":"2023-04-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10134050/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9763050","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Rhiannon C Miller, Cynthia M Magro, Laura E Melnick, Shari R Lipner
{"title":"Acral Fibrochondromyxoid Tumor Presenting as Enlarging Nodule Involving the Distal Fingertip and Hyponychium: A Case Report.","authors":"Rhiannon C Miller, Cynthia M Magro, Laura E Melnick, Shari R Lipner","doi":"10.1159/000529568","DOIUrl":"10.1159/000529568","url":null,"abstract":"<p><p>An acral fibrochondromyxoid tumor is a newly described type of benign soft tissue neoplasm that presents as a single nodular lesion on a finger or toe. There has only been one previous report on this tumor, a case series that described the initial pathologic and clinical findings; however, details on clinical history, physical examination, and outcome are unknown. In this report, we describe a case of a 39-year-old male who presented with a painful enlarging mass involving the distal right 3rd finger and hyponychium. Punch biopsy was performed and the lesion was identified as an acral fibrochondromyxoid tumor on microscopic examination. X-ray showed no bony involvement. The tumor was successfully excised with complete resolution of pain symptoms. We discuss the clinical features and immunohistochemistry findings of our case in the context of the current limited knowledge about this very rare tumor.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":null,"pages":null},"PeriodicalIF":0.9,"publicationDate":"2023-03-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/9c/d9/cde-2023-0015-0001-529568.PMC10020937.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9145209","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Mari Nakanishi, Makoto Kondo, Koji Habe, Akinobu Hayashi, Keiichi Yamanaka
{"title":"Diagnostic Utility of TUNEL Staining for Degenerative Keratoacanthoma Requiring Pathologic Differentiation from Seborrheic Keratosis.","authors":"Mari Nakanishi, Makoto Kondo, Koji Habe, Akinobu Hayashi, Keiichi Yamanaka","doi":"10.1159/000529728","DOIUrl":"10.1159/000529728","url":null,"abstract":"<p><p>Tumors developed in 2 old women presented with pathological findings similar to seborrheic keratosis, although the clinical feature of tumor showed typical keratoacanthoma. In addition to these two cases, we compared the pathological findings of a total of four cases, one case each of keratoacanthoma and seborrheic keratosis, which were clinically and histopathological typical. These two cases and the typical keratoacanthoma showed cell apoptosis by terminal deoxynucleotidyl transferase dUTP nick end labeling (TUNEL) staining and infiltration of cytotoxic T cells. The keratoacanthoma in the decompensated stage may be histologically similar to seborrheic keratosis. TUNEL staining can help in the diagnosis of fading keratoacanthoma.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":null,"pages":null},"PeriodicalIF":0.9,"publicationDate":"2023-03-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/68/a8/cde-2023-0015-0001-529728.PMC10020938.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9152449","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Samir Shrestha, Bashant Regmi, Raksha Pathak, George Kroumpouzos
{"title":"Idiopathic Acroosteolysis: A Novel Cutaneous Sign Can Help Identify the Condition Early.","authors":"Samir Shrestha, Bashant Regmi, Raksha Pathak, George Kroumpouzos","doi":"10.1159/000529727","DOIUrl":"10.1159/000529727","url":null,"abstract":"<p><p>Acroosteolysis (AO) is a rare condition characterized by resorption of the distal phalanges of the fingers and/or toes. It can be familial, idiopathic (IAO), occupational, or secondary. Other authors suggest a classification into primary (genetic disorders, lysosomal storage disorders) or secondary AO. Various skin and nail changes have been reported in this condition. However, the cutaneous change on the affected digit(s)/toe(s) during the natural course of AO has been poorly documented. A 5-year-old girl presented with a 3-month history of a distinct transverse boundary between normal skin proximally and affected crusted skin overlying osteolysis distally (\"split\" sign) on the plantar surface of the third toe. This boundary gradually elongated circumferentially to involve the dorsal surface. The mother gave a similar history of a delimitation line on the 2nd, 4th, and 5th toes of the right foot with durations of 3 months, 1 year, and 2 years, respectively, that disappeared before she noticed a shortening of those toes. X-rays revealed partial resorption of the terminal phalanx of the third toe and several lytic changes in the middle and terminal phalanx of the second, fourth, and fifth toes. The clinical features, radiology findings, and a workup that helped rule out conditions associated with AO (secondary AO) helped establish the diagnosis of IAO in our patient. This case study highlights that the natural course of IAO includes distinct skin findings, such as the \"split\" sign that we describe. This sign can help identify the condition early.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":null,"pages":null},"PeriodicalIF":0.9,"publicationDate":"2023-03-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/39/48/cde-2023-0015-0001-529727.PMC10018418.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9145207","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Seth Bernacki, Paarth Dodia, Mikél Elizabeth Muse, Luke Maxfield
{"title":"Cutaneous Manifestations of Sarcoidosis Seen in a Patient with a History of Tuberous Sclerosis.","authors":"Seth Bernacki, Paarth Dodia, Mikél Elizabeth Muse, Luke Maxfield","doi":"10.1159/000529159","DOIUrl":"10.1159/000529159","url":null,"abstract":"<p><p>This case report details a patient with a history of tuberous sclerosis presenting with new-onset cutaneous lesions that turn out to be sarcoidosis. There may be a shared dysfunction of mTOR present in sarcoidosis and tuberous sclerosis. As a dermatologist, it is worth understanding the cutaneous manifestations of both diseases and maintaining a wide differential when new lesions arise in a patient with a history of either disorder.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":null,"pages":null},"PeriodicalIF":0.9,"publicationDate":"2023-02-15","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/08/e7/cde-2023-0015-0001-529159.PMC9932741.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"10766745","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"A Diagnostically Challenging Case of De Novo Febrile Ulceronecrotic Mucha-Habermann Disease with Fatal Pulmonary Involvement: A Case Report.","authors":"Waseem Alhawsawi, Khalid Al Hawsawi, Bashaer Almahdi, Shahad Alkidaiwi, Khlood Alzubaidy, Reema Alhuthayli, Abdulmohsin Algethami, Alhusain Alshareef","doi":"10.1159/000528500","DOIUrl":"10.1159/000528500","url":null,"abstract":"<p><p>The febrile ulceronecrotic Mucha-Habermann disease is a rare and potentially lethal variant of pityriasis lichenoides et varioliformis acuta (PLEVA). It is characterized by a sudden onset of ulceronecrotic skin lesions associated with high fever and systemic symptoms. Herein, we report a 23-year-old male, not known to have any medical illnesses, presented with a month-long history of persistent fever of unknown origin associated with a sudden onset of progressive diffuse necrotic ulcers and widespread papulosquamous lesions. Pan CT showed enlarged lymph nodes in the cervix, chest, and abdomen. Unfortunately, a skin biopsy was done late, showing features consistent with PLEVA. Few days after admission, despite being on intravenous methylprednisolone, our patient rapidly deteriorated by showing severe acute respiratory symptoms and consequently died. In spite of the continuous addition of new case reports to the literature, no definite diagnostic criteria have been established, leading to late or missed cases, and an optimum treatment is still waiting.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":null,"pages":null},"PeriodicalIF":0.9,"publicationDate":"2023-01-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/d9/9e/cde-2023-0015-0001-528500.PMC9850257.pdf","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"9228132","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}