Introduction: Annular lichenoid dermatitis of youth (ALDY) is a rare and underreported dermatologic condition. It primarily affects children and adolescents. Clinically, ALDY presents as erythematous annular macules or plaques with raised borders and hypopigmented centers, predominantly affecting the trunk and flexural areas. The etiology remains unknown, and the condition poses a diagnostic challenge due to its resemblance to other annular dermatoses.
Case presentation: A 5-year-old female presented with asymptomatic annular erythematous lesions on the trunk, abdomen, lumbar region, and flanks. Initial differential diagnoses included annular erythema, tinea corporis, morphea, and mycosis fungoides. A mycological examination was negative, and symptomatic treatment with hydrocortisone and pimecrolimus led to temporary lesion resolution, but recurrence followed treatment cessation. A skin biopsy revealed histopathological features characteristic of ALDY, including basal layer vacuolization and a prominent lichenoid lymphocytic infiltrate. Immunohistochemical analysis supported the diagnosis.
Conclusion: ALDY remains a diagnostic challenge due to its clinical overlap with other annular dermatoses. Histopathological examination is essential for definitive diagnosis. The chronic and recurrent nature of ALDY underscores the importance of long-term management strategies, including topical corticosteroids and immunomodulators. Awareness of ALDY among dermatologists can aid in early recognition and appropriate management, ultimately improving patient outcomes.
{"title":"The Importance of Histopathology in the Diagnosis of Annular Lichenoid Dermatitis of Youth: A Case Report.","authors":"Leart Bërdica, Teona Bushati, Sindi Shandro, Entela Shkodrani, Sabina Dedej, Ermira Vasili","doi":"10.1159/000546701","DOIUrl":"10.1159/000546701","url":null,"abstract":"<p><strong>Introduction: </strong>Annular lichenoid dermatitis of youth (ALDY) is a rare and underreported dermatologic condition. It primarily affects children and adolescents. Clinically, ALDY presents as erythematous annular macules or plaques with raised borders and hypopigmented centers, predominantly affecting the trunk and flexural areas. The etiology remains unknown, and the condition poses a diagnostic challenge due to its resemblance to other annular dermatoses.</p><p><strong>Case presentation: </strong>A 5-year-old female presented with asymptomatic annular erythematous lesions on the trunk, abdomen, lumbar region, and flanks. Initial differential diagnoses included annular erythema, tinea corporis, morphea, and mycosis fungoides. A mycological examination was negative, and symptomatic treatment with hydrocortisone and pimecrolimus led to temporary lesion resolution, but recurrence followed treatment cessation. A skin biopsy revealed histopathological features characteristic of ALDY, including basal layer vacuolization and a prominent lichenoid lymphocytic infiltrate. Immunohistochemical analysis supported the diagnosis.</p><p><strong>Conclusion: </strong>ALDY remains a diagnostic challenge due to its clinical overlap with other annular dermatoses. Histopathological examination is essential for definitive diagnosis. The chronic and recurrent nature of ALDY underscores the importance of long-term management strategies, including topical corticosteroids and immunomodulators. Awareness of ALDY among dermatologists can aid in early recognition and appropriate management, ultimately improving patient outcomes.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"311-317"},"PeriodicalIF":0.8,"publicationDate":"2025-07-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12327932/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144793594","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}
Pub Date : 2025-07-04eCollection Date: 2025-01-01DOI: 10.1159/000547244
Nicolò Rivetti, Stefania Barruscotti, Valeria Brazzelli, Carlo Francesco Tomasini
Introduction: Ruxolitinib, a Janus kinase (JAK) 1/2 inhibitor approved for the treatment of myelofibrosis and polycythemia vera, has been associated with an increased risk of non-melanoma skin cancer. While squamous cell carcinoma (SCC), basal cell carcinoma, and keratoacanthomas are commonly reported, Bowen's disease (BD), the in situ form of SCC, has been only rarely described.
Case presentation: We report the case of a 53-year-old woman with polycythemia vera, treated with ruxolitinib for over 9 years. Six months after a dose escalation, she developed multiple erythematous, hyperkeratotic plaques on the trunk and limbs. Histopathological examination confirmed the diagnosis of BD. Over the years, she continued to develop new lesions approximately once per year. All lesions were managed successfully with cryotherapy or surgical excision, and no invasive SCC was observed. She has been under close dermatologic surveillance with full-body skin examinations every 6 months.
Conclusion: This is the first fully documented case of eruptive, multiple BD associated with long-term ruxolitinib therapy. The distinctive chronic pattern, histologically confirmed and followed prospectively for nearly a decade, highlights a potentially underrecognized cutaneous adverse effect of JAK inhibition, warranting further research and awareness among prescribing clinicians.
{"title":"Multiple Bowen's Disease in a Patient Treated with Ruxolitinib: A Case Report and Extended Review of the Literature.","authors":"Nicolò Rivetti, Stefania Barruscotti, Valeria Brazzelli, Carlo Francesco Tomasini","doi":"10.1159/000547244","DOIUrl":"10.1159/000547244","url":null,"abstract":"<p><strong>Introduction: </strong>Ruxolitinib, a Janus kinase (JAK) 1/2 inhibitor approved for the treatment of myelofibrosis and polycythemia vera, has been associated with an increased risk of non-melanoma skin cancer. While squamous cell carcinoma (SCC), basal cell carcinoma, and keratoacanthomas are commonly reported, Bowen's disease (BD), the in situ form of SCC, has been only rarely described.</p><p><strong>Case presentation: </strong>We report the case of a 53-year-old woman with polycythemia vera, treated with ruxolitinib for over 9 years. Six months after a dose escalation, she developed multiple erythematous, hyperkeratotic plaques on the trunk and limbs. Histopathological examination confirmed the diagnosis of BD. Over the years, she continued to develop new lesions approximately once per year. All lesions were managed successfully with cryotherapy or surgical excision, and no invasive SCC was observed. She has been under close dermatologic surveillance with full-body skin examinations every 6 months.</p><p><strong>Conclusion: </strong>This is the first fully documented case of eruptive, multiple BD associated with long-term ruxolitinib therapy. The distinctive chronic pattern, histologically confirmed and followed prospectively for nearly a decade, highlights a potentially underrecognized cutaneous adverse effect of JAK inhibition, warranting further research and awareness among prescribing clinicians.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"325-332"},"PeriodicalIF":0.8,"publicationDate":"2025-07-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12503472/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145250013","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}
Introduction: The most severe complication of filler injection is ischemia due to vascular occlusion, causing skin necrosis, scarring, blindness, and even stroke. Among the available treatments, needling therapy has been used in some clinical practices but has been rarely studied and discussed.
Case presentations: Four recent skin ischemia cases with subsequent needling procedure were observed retrospectively and pertinent literature was analyzed. The objective was to evaluate the efficacy and safety of needling procedure for filler-induced skin ischemia, which is unresponsive to standard therapies.
Conclusion: All of the 4 cases recovered from skin ischemia without side effects by receiving needling procedure. The available data demonstrate some potential mechanisms of needling, such as embolus releasing, ischemia reperfusion and revascularization. Although there is a lack of conclusive evidence for improving the hypoperfusion area by needling treatment, the current cases observation and theoretical analysis as well as our cases provide evidence supporting its potential as an efficacious, simple, and secure treatment for vascular complications.
{"title":"Needling as a Potential and Novel Treatment for Skin Ischemia following Filler-Induced Vascular Occlusion: A Case Series.","authors":"Jieyi Wang, Zimo Zhu, Yanfen Zou, Zhuoying Wang, Jingwen Wu, Bo Yu, Xingling Jian","doi":"10.1159/000547162","DOIUrl":"10.1159/000547162","url":null,"abstract":"<p><strong>Introduction: </strong>The most severe complication of filler injection is ischemia due to vascular occlusion, causing skin necrosis, scarring, blindness, and even stroke. Among the available treatments, needling therapy has been used in some clinical practices but has been rarely studied and discussed.</p><p><strong>Case presentations: </strong>Four recent skin ischemia cases with subsequent needling procedure were observed retrospectively and pertinent literature was analyzed. The objective was to evaluate the efficacy and safety of needling procedure for filler-induced skin ischemia, which is unresponsive to standard therapies.</p><p><strong>Conclusion: </strong>All of the 4 cases recovered from skin ischemia without side effects by receiving needling procedure. The available data demonstrate some potential mechanisms of needling, such as embolus releasing, ischemia reperfusion and revascularization. Although there is a lack of conclusive evidence for improving the hypoperfusion area by needling treatment, the current cases observation and theoretical analysis as well as our cases provide evidence supporting its potential as an efficacious, simple, and secure treatment for vascular complications.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"333-341"},"PeriodicalIF":0.8,"publicationDate":"2025-07-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12503475/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145250066","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}
Pub Date : 2025-06-23eCollection Date: 2025-01-01DOI: 10.1159/000546382
Fatemeh Mohaghegh, Mina Saber, Parvin Rajabi, Haniyeh Sohrabi
Introduction: Rosai-Dorfman disease (RDD) is a rare, benign histiocytic disorder that can present as isolated cutaneous RDD (CRDD), often mimicking other dermatologic conditions and complicating diagnosis.
Case presentation: We report two cases of CRDD: one with palms and soles involvement, and another as an exophytic facial mass, initially misdiagnosed and unsuccessfully treated. Dermoscopy revealed distinctive features, including yellow ovoid structures, linear vessels, and cotton-like white globules. Histopathological and immunohistochemical analyses confirmed CRDD with histiocytic infiltration and emperipolesis, key histological markers of CRDD.
Conclusion: A review of 53 cases (2018-2023) highlights CRDD's clinical variability, common anatomical sites, and diverse treatment responses, emphasizing the need for individualized management and early recognition for optimal treatment. These findings contribute to a broader understanding of CRDD and support a multidisciplinary approach to optimizing patient care.
{"title":"Cutaneous Rosai-Dorfman Disease: A Report of 2 Cases and a Review of Recent Literature (2018-2023).","authors":"Fatemeh Mohaghegh, Mina Saber, Parvin Rajabi, Haniyeh Sohrabi","doi":"10.1159/000546382","DOIUrl":"10.1159/000546382","url":null,"abstract":"<p><strong>Introduction: </strong>Rosai-Dorfman disease (RDD) is a rare, benign histiocytic disorder that can present as isolated cutaneous RDD (CRDD), often mimicking other dermatologic conditions and complicating diagnosis.</p><p><strong>Case presentation: </strong>We report two cases of CRDD: one with palms and soles involvement, and another as an exophytic facial mass, initially misdiagnosed and unsuccessfully treated. Dermoscopy revealed distinctive features, including yellow ovoid structures, linear vessels, and cotton-like white globules. Histopathological and immunohistochemical analyses confirmed CRDD with histiocytic infiltration and emperipolesis, key histological markers of CRDD.</p><p><strong>Conclusion: </strong>A review of 53 cases (2018-2023) highlights CRDD's clinical variability, common anatomical sites, and diverse treatment responses, emphasizing the need for individualized management and early recognition for optimal treatment. These findings contribute to a broader understanding of CRDD and support a multidisciplinary approach to optimizing patient care.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"204-223"},"PeriodicalIF":0.9,"publicationDate":"2025-06-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12185066/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144474064","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}
Introduction: Sarcoidosis can present with a wide range of skin manifestations, occurring in approximately 30% of sarcoidosis patients. However, photosensitivity in cutaneous sarcoidosis is exceedingly rare. This case report details a unique presentation of photosensitive cutaneous sarcoidosis, its diagnosis, and treatment.
Case presentation: A 65-year-old woman presented with numerous erythematous lesions on her eyelids, persisting for 5 months despite treatments with fexofenadine hydrochloride and topical steroids. Her medical history included diabetes mellitus and dyslipidemia, managed with rosuvastatin, metformin, and a combination of teneligliptin and canagliflozin. After discontinuation of rosuvastatin due to suspected interstitial granulomatous dermatitis, her lesions remained unchanged. Clinical examination revealed annular plaques on sun-exposed areas, and skin biopsy showed noncaseating granulomas with eosinophilic infiltration. Elevated serum ACE, calcium, and soluble interleukin-2 receptor levels supported a diagnosis of cutaneous sarcoidosis. Lesions were resistant to systemic and topical steroid treatments, prompting suspicion of photosensitivity. Photo testing identified UVB sensitivity during metformin use, which resolved after discontinuation of the drug. The patient's lesions improved 1 week after stopping metformin, enabling prednisone tapering without recurrence.
Conclusion: This case highlights metformin as a potential photosensitizing agent in cutaneous sarcoidosis. While the relationship between diabetes mellitus and sarcoidosis remains unclear, chronic inflammation in diabetes may influence sarcoidosis progression. In cases of refractory cutaneous sarcoidosis despite systemic steroid treatment, it is crucial to consider the coexistence of photosensitivity, particularly drug-induced photosensitivity. Therefore, carefully reviewing the patient's medication history is essential for accurate diagnosis and management.
{"title":"Photo-Induced Sarcoidosis in a Diabetic Patient Receiving Metformin Hydrochloride.","authors":"Hanae Fujita, Yuki Mizutani, Akinobu Hayashi, Keiichi Yamanaka","doi":"10.1159/000546617","DOIUrl":"10.1159/000546617","url":null,"abstract":"<p><strong>Introduction: </strong>Sarcoidosis can present with a wide range of skin manifestations, occurring in approximately 30% of sarcoidosis patients. However, photosensitivity in cutaneous sarcoidosis is exceedingly rare. This case report details a unique presentation of photosensitive cutaneous sarcoidosis, its diagnosis, and treatment.</p><p><strong>Case presentation: </strong>A 65-year-old woman presented with numerous erythematous lesions on her eyelids, persisting for 5 months despite treatments with fexofenadine hydrochloride and topical steroids. Her medical history included diabetes mellitus and dyslipidemia, managed with rosuvastatin, metformin, and a combination of teneligliptin and canagliflozin. After discontinuation of rosuvastatin due to suspected interstitial granulomatous dermatitis, her lesions remained unchanged. Clinical examination revealed annular plaques on sun-exposed areas, and skin biopsy showed noncaseating granulomas with eosinophilic infiltration. Elevated serum ACE, calcium, and soluble interleukin-2 receptor levels supported a diagnosis of cutaneous sarcoidosis. Lesions were resistant to systemic and topical steroid treatments, prompting suspicion of photosensitivity. Photo testing identified UVB sensitivity during metformin use, which resolved after discontinuation of the drug. The patient's lesions improved 1 week after stopping metformin, enabling prednisone tapering without recurrence.</p><p><strong>Conclusion: </strong>This case highlights metformin as a potential photosensitizing agent in cutaneous sarcoidosis. While the relationship between diabetes mellitus and sarcoidosis remains unclear, chronic inflammation in diabetes may influence sarcoidosis progression. In cases of refractory cutaneous sarcoidosis despite systemic steroid treatment, it is crucial to consider the coexistence of photosensitivity, particularly drug-induced photosensitivity. Therefore, carefully reviewing the patient's medication history is essential for accurate diagnosis and management.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"284-290"},"PeriodicalIF":0.9,"publicationDate":"2025-06-18","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12258872/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144636195","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}
Introduction: Amivantamab is a monoclonal antibody against EGFR and MET receptors, indications for certain types of non-small cell lung cancer. Due to its mechanisms, cutaneous adverse effects are frequent and numerous.
Case presentation: A 67-year-old woman with metastatic adenosquamous carcinoma, stage T3N2M1c, was treated with amivantamab after the first-line chemotherapy failed. Despite prophylactic oral tetracyclines, she developed severe erosive pustular dermatosis (EPD) affecting more than 50% of her scalp, forcing to cut short her hair to provide adequate local care.
Conclusion: EPD is an exceptional and severe adverse event of amivantamab, requiring oral steroids, tetracyclines and appropriate local care with antibiotic creams. Clinicians should be aware of this complication as early therapeutic intervention is mandatory to avoid deleterious consequences and spontaneous recurrences.
{"title":"Erosive Pustular Dermatosis and Amivantamab for Lung Cancer: A Case Report.","authors":"Yseult Senterre, Amandine Bouillenne, Murielle Sabatiello, Anne-Sophie Demoulin, Arjen F Nikkels","doi":"10.1159/000546616","DOIUrl":"10.1159/000546616","url":null,"abstract":"<p><strong>Introduction: </strong>Amivantamab is a monoclonal antibody against EGFR and MET receptors, indications for certain types of non-small cell lung cancer. Due to its mechanisms, cutaneous adverse effects are frequent and numerous.</p><p><strong>Case presentation: </strong>A 67-year-old woman with metastatic adenosquamous carcinoma, stage T3N2M1c, was treated with amivantamab after the first-line chemotherapy failed. Despite prophylactic oral tetracyclines, she developed severe erosive pustular dermatosis (EPD) affecting more than 50% of her scalp, forcing to cut short her hair to provide adequate local care.</p><p><strong>Conclusion: </strong>EPD is an exceptional and severe adverse event of amivantamab, requiring oral steroids, tetracyclines and appropriate local care with antibiotic creams. Clinicians should be aware of this complication as early therapeutic intervention is mandatory to avoid deleterious consequences and spontaneous recurrences.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"263-267"},"PeriodicalIF":0.9,"publicationDate":"2025-06-13","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12240574/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144599543","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}
Pub Date : 2025-06-11eCollection Date: 2025-01-01DOI: 10.1159/000545842
Farzad Alinaghi, Jesper Elberling
Introduction: Aquagenic pruritus is a chronic debilitating itchy skin condition triggered by water exposure with limited treatment options.
Case presentation: Here, we present a case of primary aquagenic pruritus in an adult male who was successfully treated with β-alanine. Treatment efficacy was maintained at 20-week follow-up.
Conclusion: This study adds to the body of evidence supporting a role for β-alanine as an efficient and safe treatment option in patients with aquagenic pruritus. Furthermore, it includes a discussion of the pharmacodynamics of β-alanine and provides an insight into the pathophysiology of aquagenic pruritus.
{"title":"Primary Aquagenic Pruritus Successfully Treated by β-Alanine: A Case Report.","authors":"Farzad Alinaghi, Jesper Elberling","doi":"10.1159/000545842","DOIUrl":"10.1159/000545842","url":null,"abstract":"<p><strong>Introduction: </strong>Aquagenic pruritus is a chronic debilitating itchy skin condition triggered by water exposure with limited treatment options.</p><p><strong>Case presentation: </strong>Here, we present a case of primary aquagenic pruritus in an adult male who was successfully treated with β-alanine. Treatment efficacy was maintained at 20-week follow-up.</p><p><strong>Conclusion: </strong>This study adds to the body of evidence supporting a role for β-alanine as an efficient and safe treatment option in patients with aquagenic pruritus. Furthermore, it includes a discussion of the pharmacodynamics of β-alanine and provides an insight into the pathophysiology of aquagenic pruritus.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"252-254"},"PeriodicalIF":0.9,"publicationDate":"2025-06-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12233996/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144583197","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}
Pub Date : 2025-06-11eCollection Date: 2025-01-01DOI: 10.1159/000546384
Nawa Arif, Sylke Schneider-Burrus
Introduction: Hidradenitis suppurativa (HS), or acne inversa, is a chronic inflammatory skin disease that leads to painful nodules, abscesses, and fistulas.
Case presentation: This case describes the treatment of a 49-year-old male patient with progressive and massively inflamed HS for 20 years who also suffered from heart failure (NYHA II-III, EF 35-40%) and hypochromic microcytic anaemia (Hb 7.8 g/dL). Upon presentation, he was suffering from fatigue and severe pain (8-9/10, NRS). Due to prolonged immobility, knee and hip joint contractures with extension deficits were present. At the initial presentation, communicating, and purulent sinus tracts were observed bilaterally from the gluteal to femoral region and perianal area. Hurley score III, HS-PGA score: very severe, Dermatology Life Quality Index (DLQI) score: 19, ISH4 score: severe (16). Under adalimumab s.c. 40 mg/week combined with clindamycin (300 mg bd), the condition had worsened. The patient was admitted to the hospital for i.v. administration of ertapenem 1 g/day for 14 days in preparation for sinus tract resection. With an Hb level of 7.8 g/dL, the patient received iron carboxymaltose and erythropoietin. After a significant reduction in inflammatory markers (leukocytes 17.11/nL to 7.42/nL), a large excision of the sinus tracts was performed bilaterally in the gluteal region and left femoral area. Following 4 weeks of wound granulation, split-thickness skin grafting was performed. The knee and hip joint contractures improved with intensive physiotherapy. After surgery, the patient received antibiotic therapy with clindamycin, rifampicin, and metronidazole (clindamycin 600 mg, rifampicin 300 mg, metronidazole 500 mg, each p.o. 2×/day). In parallel, anti-inflammatory therapy with secukinumab 300 mg s.c. twice a month was initiated. Over the following 3 months, no progression or recurrence occurred. Quality of life and pain levels improved significantly under the therapy (DLQI from 19 to 10, pain from 8 to 9 to 3/10 NRS).
Conclusion: In extensive cases of HS, a combination of surgical and conservative therapy is necessary. We demonstrate that severe cases can be successfully treated with a combination of antibiotic, anti-inflammatory, and surgical therapy.
简介:化脓性汗腺炎(HS),或反之痤疮,是一种慢性炎症性皮肤病,可导致疼痛的结节、脓肿和瘘管。病例介绍:本病例描述了一名49岁男性患者的治疗进展性和大规模炎症HS 20年,同时患有心力衰竭(NYHA II-III, EF 35-40%)和低色性小细胞贫血(Hb 7.8 g/dL)。在就诊时,他感到疲劳和剧烈疼痛(8-9/10,NRS)。由于长时间不活动,膝关节和髋关节挛缩和伸展缺陷存在。在最初的表现,交通,化脓性窦束被观察到双侧从臀到股区域和肛周区域。Hurley评分III, HS-PGA评分:极重度,Dermatology Life Quality Index (DLQI)评分:19分,ISH4评分:重度(16分)。阿达木单抗40 mg/周联合克林霉素300 mg/周,病情恶化。患者入院静脉滴注厄他培南1 g/天,连续14天,为窦道切除术做准备。由于Hb水平为7.8 g/dL,患者接受了羧麦芽糖铁和促红细胞生成素治疗。在炎症标志物(白细胞17.11/nL至7.42/nL)显著降低后,在双侧臀区和左股区行窦束大切除术。创面肉芽化4周后,进行裂厚皮移植。经强化理疗,膝关节和髋关节挛缩得到改善。术后给予克林霉素、利福平、甲硝唑联合抗生素治疗(克林霉素600 mg、利福平300 mg、甲硝唑500 mg,每次口服2次/天)。同时,开始了每月两次的secukinumab 300 mg s.c.抗炎治疗。在接下来的3个月里,没有进展或复发发生。治疗后患者的生活质量和疼痛水平显著改善(DLQI从19到10,疼痛从8到9,NRS从3/10)。结论:在广泛病例中,手术和保守治疗相结合是必要的。我们证明,重症病例可以成功地治疗抗生素,抗炎和手术治疗的组合。
{"title":"A Recalcitrant Case of Hidradenitis Suppurativa: Successful Combination of Biologic, Antibiotic, and Surgical Therapy.","authors":"Nawa Arif, Sylke Schneider-Burrus","doi":"10.1159/000546384","DOIUrl":"10.1159/000546384","url":null,"abstract":"<p><strong>Introduction: </strong>Hidradenitis suppurativa (HS), or acne inversa, is a chronic inflammatory skin disease that leads to painful nodules, abscesses, and fistulas.</p><p><strong>Case presentation: </strong>This case describes the treatment of a 49-year-old male patient with progressive and massively inflamed HS for 20 years who also suffered from heart failure (NYHA II-III, EF 35-40%) and hypochromic microcytic anaemia (Hb 7.8 g/dL). Upon presentation, he was suffering from fatigue and severe pain (8-9/10, NRS). Due to prolonged immobility, knee and hip joint contractures with extension deficits were present. At the initial presentation, communicating, and purulent sinus tracts were observed bilaterally from the gluteal to femoral region and perianal area. Hurley score III, HS-PGA score: very severe, Dermatology Life Quality Index (DLQI) score: 19, ISH4 score: severe (16). Under adalimumab s.c. 40 mg/week combined with clindamycin (300 mg bd), the condition had worsened. The patient was admitted to the hospital for i.v. administration of ertapenem 1 g/day for 14 days in preparation for sinus tract resection. With an Hb level of 7.8 g/dL, the patient received iron carboxymaltose and erythropoietin. After a significant reduction in inflammatory markers (leukocytes 17.11/nL to 7.42/nL), a large excision of the sinus tracts was performed bilaterally in the gluteal region and left femoral area. Following 4 weeks of wound granulation, split-thickness skin grafting was performed. The knee and hip joint contractures improved with intensive physiotherapy. After surgery, the patient received antibiotic therapy with clindamycin, rifampicin, and metronidazole (clindamycin 600 mg, rifampicin 300 mg, metronidazole 500 mg, each p.o. 2×/day). In parallel, anti-inflammatory therapy with secukinumab 300 mg s.c. twice a month was initiated. Over the following 3 months, no progression or recurrence occurred. Quality of life and pain levels improved significantly under the therapy (DLQI from 19 to 10, pain from 8 to 9 to 3/10 NRS).</p><p><strong>Conclusion: </strong>In extensive cases of HS, a combination of surgical and conservative therapy is necessary. We demonstrate that severe cases can be successfully treated with a combination of antibiotic, anti-inflammatory, and surgical therapy.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"268-273"},"PeriodicalIF":0.9,"publicationDate":"2025-06-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12252370/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144625443","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}
Pub Date : 2025-06-11eCollection Date: 2025-01-01DOI: 10.1159/000546702
Laura Brufau, Pablo Russo, Alessandra Zevini, Daniela Martinelli, Riccardo Barini
Introduction: Port-wine stains (PWS), congenital vascular malformations affecting approximately 0.3-0.5% of live births, present as pink to red patches that darken and thicken over time, potentially leading to disfigurement, functional complications, and significant psychosocial distress. While pulsed dye lasers (PDLs) are considered the standard treatment, they often require multiple sessions and exhibit variable clearance rates. Alternative laser modalities, such as the 532-nm potassium titanyl phosphate (KTP) laser, have shown promising results, particularly in PDL-resistant cases. However, managing complex PWS with diverse vessel calibers, hypertrophy, and scarring remains a challenge, highlighting the need for tailored treatment strategies.
Case presentation: A 22-year-old female presented with a complex PWS affecting the V2 dermatome of the left hemiface, including the lower eyelid, inner canthus, and associated lip/gingival hypertrophy and scarring from previous surgical intervention. She underwent five monthly sessions of a novel sequential laser protocol. This protocol combined long-pulsed KTP 532-nm laser treatment with quasi long-pulsed 1,064-nm Nd:YAG laser treatment to target both larger and smaller superficial vascular structures. In the final two sessions, ablative Er:YAG 2,940-nm laser treatment was added to address existing scars. Fluence and pulse duration were dynamically adjusted throughout the treatment course. Post-treatment care consisted of gentle skincare, emollients, and broad-spectrum sunscreen. The patient achieved significant lightening of the lesion, reduced vascular prominence and hypertrophy, improved facial symmetry, and softening of scars, with minimal and transient side effects.
Conclusion: This case demonstrates the potential of a tailored, sequential combination of long-pulsed KTP 532-nm, quasi long-pulsed 1,064-nm Nd:YAG, and ablative Er:YAG 2,940-nm lasers for effectively managing complex PWS with associated hypertrophy and scarring. This multimodal approach, adjusting parameters to target specific lesion characteristics, suggests a promising strategy for achieving superior esthetic and functional outcomes in challenging PWS cases and improving patient quality of life.
{"title":"Efficacy of Sequential Combination of Long-Pulsed Potassium Titanyl Phosphate 532-nm and Quasi Long-Pulsed 1,064-nm Nd:YAG Laser Therapy for Port-Wine Stain Treatment: A Case Report.","authors":"Laura Brufau, Pablo Russo, Alessandra Zevini, Daniela Martinelli, Riccardo Barini","doi":"10.1159/000546702","DOIUrl":"10.1159/000546702","url":null,"abstract":"<p><strong>Introduction: </strong>Port-wine stains (PWS), congenital vascular malformations affecting approximately 0.3-0.5% of live births, present as pink to red patches that darken and thicken over time, potentially leading to disfigurement, functional complications, and significant psychosocial distress. While pulsed dye lasers (PDLs) are considered the standard treatment, they often require multiple sessions and exhibit variable clearance rates. Alternative laser modalities, such as the 532-nm potassium titanyl phosphate (KTP) laser, have shown promising results, particularly in PDL-resistant cases. However, managing complex PWS with diverse vessel calibers, hypertrophy, and scarring remains a challenge, highlighting the need for tailored treatment strategies.</p><p><strong>Case presentation: </strong>A 22-year-old female presented with a complex PWS affecting the V2 dermatome of the left hemiface, including the lower eyelid, inner canthus, and associated lip/gingival hypertrophy and scarring from previous surgical intervention. She underwent five monthly sessions of a novel sequential laser protocol. This protocol combined long-pulsed KTP 532-nm laser treatment with quasi long-pulsed 1,064-nm Nd:YAG laser treatment to target both larger and smaller superficial vascular structures. In the final two sessions, ablative Er:YAG 2,940-nm laser treatment was added to address existing scars. Fluence and pulse duration were dynamically adjusted throughout the treatment course. Post-treatment care consisted of gentle skincare, emollients, and broad-spectrum sunscreen. The patient achieved significant lightening of the lesion, reduced vascular prominence and hypertrophy, improved facial symmetry, and softening of scars, with minimal and transient side effects.</p><p><strong>Conclusion: </strong>This case demonstrates the potential of a tailored, sequential combination of long-pulsed KTP 532-nm, quasi long-pulsed 1,064-nm Nd:YAG, and ablative Er:YAG 2,940-nm lasers for effectively managing complex PWS with associated hypertrophy and scarring. This multimodal approach, adjusting parameters to target specific lesion characteristics, suggests a promising strategy for achieving superior esthetic and functional outcomes in challenging PWS cases and improving patient quality of life.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"274-283"},"PeriodicalIF":0.9,"publicationDate":"2025-06-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12252374/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144625444","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}
Pub Date : 2025-06-11eCollection Date: 2025-01-01DOI: 10.1159/000546700
Xiangxiang Ren, Tianhao Xie, Lingyun Liu, Fan Yang, Meng Zhang
Introduction: Malignant hidroacanthoma simplex (MHS) is an exceedingly rare cutaneous neoplasm with limited documented cases. This report highlights a distinctive case of MHS with prolonged clinical evolution, emphasizing its diagnostic challenges and management outcomes.
Case presentation: A 64-year-old female presented with a right lumbar mass persisting for over 2 decades, exhibiting progressive enlargement in the past 5 years. Clinical examination revealed a solitary reddish-brown proliferative plaque (3.5 cm × 4.0 cm) on the right waist, characterized by irregular borders, a rough surface, and reddish-brown crusts. Histopathological findings included hyperkeratosis, irregular epidermal hyperplasia, hypertrophic stratum spinosum, and tumor cells displaying pale eosinophilic cytoplasm, vacuolated nuclei, small nucleoli, and atypical mitotic figures. Notably, tumor cells were confined to the epidermis without dermal invasion. The patient underwent local extended excision, and postoperative surveillance over 15 months demonstrated no evidence of recurrence or lymph node metastasis.
Conclusion: This case underscores the indolent yet locally persistent nature of MHS. Complete surgical excision remains the cornerstone of management, with favorable outcomes achievable in the absence of dermal infiltration. Long-term follow-up is critical to monitor potential recurrence.
简介:恶性单纯性汗腺瘤(MHS)是一种极为罕见的皮肤肿瘤,文献记载病例有限。本报告强调了一个独特的MHS病例与长期的临床演变,强调其诊断挑战和管理结果。病例介绍:一名64岁女性,右腰椎肿块持续超过20年,在过去5年中表现出进行性扩大。临床检查示右腰一单发红褐色增生性斑块(3.5 cm × 4.0 cm),边界不规则,表面粗糙,结痂呈红褐色。组织病理学结果包括角化过度,不规则表皮增生,棘层肥大,肿瘤细胞表现为苍白嗜酸性细胞质,细胞核空泡化,核仁小,非典型有丝分裂象。值得注意的是,肿瘤细胞局限于表皮,未向真皮浸润。患者接受了局部扩大切除,术后监测超过15个月未发现复发或淋巴结转移的证据。结论:本病例强调了MHS的惰性和局部持续性。完全手术切除仍然是治疗的基石,在没有真皮浸润的情况下可以获得良好的结果。长期随访是监测潜在复发的关键。
{"title":"Malignant Hidroacanthoma Simplex: A Case Report.","authors":"Xiangxiang Ren, Tianhao Xie, Lingyun Liu, Fan Yang, Meng Zhang","doi":"10.1159/000546700","DOIUrl":"10.1159/000546700","url":null,"abstract":"<p><strong>Introduction: </strong>Malignant hidroacanthoma simplex (MHS) is an exceedingly rare cutaneous neoplasm with limited documented cases. This report highlights a distinctive case of MHS with prolonged clinical evolution, emphasizing its diagnostic challenges and management outcomes.</p><p><strong>Case presentation: </strong>A 64-year-old female presented with a right lumbar mass persisting for over 2 decades, exhibiting progressive enlargement in the past 5 years. Clinical examination revealed a solitary reddish-brown proliferative plaque (3.5 cm × 4.0 cm) on the right waist, characterized by irregular borders, a rough surface, and reddish-brown crusts. Histopathological findings included hyperkeratosis, irregular epidermal hyperplasia, hypertrophic stratum spinosum, and tumor cells displaying pale eosinophilic cytoplasm, vacuolated nuclei, small nucleoli, and atypical mitotic figures. Notably, tumor cells were confined to the epidermis without dermal invasion. The patient underwent local extended excision, and postoperative surveillance over 15 months demonstrated no evidence of recurrence or lymph node metastasis.</p><p><strong>Conclusion: </strong>This case underscores the indolent yet locally persistent nature of MHS. Complete surgical excision remains the cornerstone of management, with favorable outcomes achievable in the absence of dermal infiltration. Long-term follow-up is critical to monitor potential recurrence.</p>","PeriodicalId":9619,"journal":{"name":"Case Reports in Dermatology","volume":"17 1","pages":"246-251"},"PeriodicalIF":0.9,"publicationDate":"2025-06-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12233995/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144583196","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}