Autochthonous leishmaniasis has become increasingly recognized in Thailand, with Leishmania (Mundinia) martiniquensis identified as the predominant species, particularly among immunocompromised individuals. Infected immunosuppressed patients often present with complex clinical features, which can delay diagnosis and complicate treatment. Given the limited clinical data available and emerging reports of resistant infections, improved awareness, prompt diagnosis, and optimized management strategies are urgently needed to address this underrecognized pathogen in Thailand. We report two patients with advanced HIV disease (AHD) from Songkhla Province, Southern Thailand, who developed chronic diffuse cutaneous leishmaniasis characterized by widespread non-ulcerative papulonodular lesions that progressed to visceral involvement. Histopathological examination of the skin nodules showed prominent dermal fibrosis with infiltration by macrophages heavily parasitized with kinetoplast-containing amastigotes, consistent with cutaneous leishmaniasis. Molecular analyses identified L. martiniquensis as the causative agent in both cases. The parasite strains (WHO codes: MHOM/TH/2022/CULE7.1 and MHOM/TH/2022/CULE7.2) were successfully isolated from the bone marrow and cutaneous biopsy of the second patient before treatment. Furthermore, the parasite was isolated again from a cutaneous biopsy of the same patient after relapse, designated MHOM/TH/2023/CULE8. Due to the high costs of liposomal amphotericin B and the unavailability of miltefosine in Thailand, contrary to the WHO guideline recommending these as first-line therapy, patients received intravenous amphotericin B deoxycholate (AmB-D) combined with oral itraconazole. Despite repeated treatment with AmB-D and itraconazole, both patients relapsed, and Case 1 died. This raises concerns about drug resistance. These cases illustrate complex cutaneous manifestations and therapeutic challenges of relapsing diffuse cutaneous and visceral leishmaniasis caused by L. martiniquensis in patients with AHD from Southern Thailand. The persistence and relapse despite AmB-D therapy raise concerns about emerging drug-resistant strains and underscore the need for enhanced surveillance, parasite isolation, and optimized treatment strategies for this neglected pathogen. Moreover, this report expands the understanding of the cutaneous spectrum of L. martiniquensis in patients with AHD, emphasizing the importance of including leishmaniasis in the differential diagnosis of complex skin diseases among immunosuppressed individuals, particularly in endemic areas.
Yuesen Zhang,1,* Junyou Zheng,2,* Xiaofang Li,3 Yan Wang,1 Fang Fang,1 Zhiwen Wang,1 Wenbo Bu1 1Department of Dermatologic Surgery, Hospital for Skin Diseases, Institute of Dermatology, Chinese Academy of Medical Sciences and Peking Union Medical College, Nanjing, Jiangsu, People’s Republic of China; 2Hospital for Skin Diseases, Institute of Dermatology, Chinese Academy of Medical Sciences and Peking Union Medical College, Nanjing, Jiangsu, People’s Republic of China; 3Department of Medical Mycology, Hospital for Skin Diseases, Institute of Dermatology, Chinese Academy of Medical Sciences and Peking Union Medical College, Nanjing, Jiangsu, People’s Republic of China*These authors contributed equally to this workCorrespondence: Wenbo Bu; Zhiwen Wang, Department of dermatologic surgery, Hospital for Skin Diseases, Institute of Dermatology, Chinese Academy of Medical Sciences and Peking Union Medical College, Nanjing, Jiangsu, People’s Republic of China, Email buwenbo@163.com; wzwtyr1972@163.comAbstract: Morpheaform basal cell carcinoma (BCC) is a high-risk histologic subtype accounting for approximately 5– 10% of all basal cell carcinoma cases. We report a case of facial morpheaform BCC successfully treated with oral sonidegib in a patient for whom surgery was contraindicated because of severe thrombocytopenia. A 68-year-old woman presented with an indurated sclerotic plaque with papules and crusts on the left cheek. Histopathological examination confirmed the diagnosis of morpheaform BCC. Because of the high bleeding risk associated with thrombocytopenia, surgical treatment was not considered feasible. The patient was treated with oral sonidegib at a dose of 200 mg daily, resulting in progressive regression of the lesion. After approximately two years of therapy, near-complete clinical remission was achieved, and follow-up histopathological examination with five-point sampling showed no residual tumor. This case highlights the potential role of Hedgehog pathway inhibition as an effective tissue-sparing therapeutic option for patients with difficult-to-treat morpheaform BCC.Keywords: basal cell carcinoma, morpheaform basal cell carcinoma, histopathology, Hedgehog pathway, sonidegib
Differentiation between leishmaniasis and histoplasmosis remains a significant diagnostic conundrum, especially in immunosuppressed patients, due to overlapping clinical presentations and histopathological features. We report a case of a 53-year-old Thai man with advanced HIV disease from Southern Thailand who presented in 2020 with a chronic palate ulcer and hard palate perforation, initially diagnosed as histoplasmosis and treated with itraconazole for a year. Three years later (2023), the patient developed systemic symptoms, and biopsies confirmed visceral leishmaniasis. Given the increasing number of autochthonous leishmaniasis cases, the patient's original diagnosis from 2020 was reexamined. Retrospective analysis of the original palate biopsy revealed amastigotes with kinetoplasts, whereas PAS and GMS staining were negative for fungi, excluding histoplasmosis. Molecular diagnostics, which combined PCR for Leishmania spp. and Histoplasma capsulatum with Sanger sequencing, confirmed Leishmania martiniquensis and ruled out Histoplasma capsulatum, leading to a corrected diagnosis of mucocutaneous leishmaniasis. This report documents the second case of mucocutaneous leishmaniasis caused by L. martiniquensis in Thailand, with primary mucocutaneous presentation that progressed to visceral involvement. This highlights diagnostic challenges due to morphological similarities between Leishmania amastigotes and Histoplasma capsulatum yeasts, emphasizing the need for integrative histopathological (including specific staining techniques) and molecular diagnostics for accurate diagnosis and effective treatment.
Chronic kidney disease (CKD) requires long-term management, with treatment success largely depending on patient adherence and self-monitoring. Many patients, however, struggle with consistent medication use and tracking of vital health indicators. Digital tools offer new opportunities to support patients in daily disease management. “Kidney Care Community” is an interactive platform designed to enhance compliance and self-care in CKD. This study evaluates its effectiveness in improving treatment adherence and monitoring behaviors in patients with stage I–III CKD.