{"title":"H综合征组织细胞增生症-淋巴腺病加综合征。文献综述。","authors":"Alaa Hamad, Hadeel Elwaheidi, Farah Salameh, Mossaed Alyahya, Riad El Fakih, Mahmoud Aljurf","doi":"10.4103/hemoncstem.HEMONCSTEM-D-24-00004","DOIUrl":null,"url":null,"abstract":"<p><p>H syndrome is a rare autosomal recessive genodermatosis that falls under the histiocytosis-lymphadenopathy plus syndrome. The term \"H syndrome\" includes manifestations such as hyperpigmentation, hypertrichosis, hepatosplenomegaly, heart anomalies, hearing loss, hypogonadism, low height, and occasionally hyperglycemia. The syndrome is associated with mutations in the SLC29A3 gene, which encodes the human equilibrative transporter 3 present in endosomes, lysosomes, and mitochondria. The generalized and ubiquitous presence of affected lysosomes and mitochondria contributes to the systemic and phenotypically heterogeneous manifestations of the syndrome. H syndrome manifestations are cutaneous, systemic, and organ-specific. The pathognomonic signs are hypertrichosis and hyperpigmentation in the inner thighs and shins. However, not all patients present with these symptoms. H syndrome management involves a multidisciplinary approach to address specific symptoms and complications. The prognosis of H syndrome depends on several factors, including the extent and severity of clinical manifestations, the presence of complications, and timely diagnosis and management. Further studies are needed to explore the association between prognosis and the different mutations encountered in H syndrome.</p>","PeriodicalId":516321,"journal":{"name":"Hematology/oncology and stem cell therapy","volume":"17 3","pages":"159-167"},"PeriodicalIF":0.0000,"publicationDate":"2024-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"H syndrome: A histiocytosis-lymphadenopathy plus syndrome. A comprehensive review of the literature.\",\"authors\":\"Alaa Hamad, Hadeel Elwaheidi, Farah Salameh, Mossaed Alyahya, Riad El Fakih, Mahmoud Aljurf\",\"doi\":\"10.4103/hemoncstem.HEMONCSTEM-D-24-00004\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>H syndrome is a rare autosomal recessive genodermatosis that falls under the histiocytosis-lymphadenopathy plus syndrome. The term \\\"H syndrome\\\" includes manifestations such as hyperpigmentation, hypertrichosis, hepatosplenomegaly, heart anomalies, hearing loss, hypogonadism, low height, and occasionally hyperglycemia. The syndrome is associated with mutations in the SLC29A3 gene, which encodes the human equilibrative transporter 3 present in endosomes, lysosomes, and mitochondria. The generalized and ubiquitous presence of affected lysosomes and mitochondria contributes to the systemic and phenotypically heterogeneous manifestations of the syndrome. H syndrome manifestations are cutaneous, systemic, and organ-specific. The pathognomonic signs are hypertrichosis and hyperpigmentation in the inner thighs and shins. However, not all patients present with these symptoms. H syndrome management involves a multidisciplinary approach to address specific symptoms and complications. The prognosis of H syndrome depends on several factors, including the extent and severity of clinical manifestations, the presence of complications, and timely diagnosis and management. Further studies are needed to explore the association between prognosis and the different mutations encountered in H syndrome.</p>\",\"PeriodicalId\":516321,\"journal\":{\"name\":\"Hematology/oncology and stem cell therapy\",\"volume\":\"17 3\",\"pages\":\"159-167\"},\"PeriodicalIF\":0.0000,\"publicationDate\":\"2024-07-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Hematology/oncology and stem cell therapy\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.4103/hemoncstem.HEMONCSTEM-D-24-00004\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2024/10/4 0:00:00\",\"PubModel\":\"Epub\",\"JCR\":\"\",\"JCRName\":\"\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Hematology/oncology and stem cell therapy","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.4103/hemoncstem.HEMONCSTEM-D-24-00004","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2024/10/4 0:00:00","PubModel":"Epub","JCR":"","JCRName":"","Score":null,"Total":0}
引用次数: 0
摘要
H 综合征是一种罕见的常染色体隐性遗传皮肤病,属于组织细胞增生症-淋巴腺病加综合征。H 综合征 "包括色素沉着、多毛、肝脾肿大、心脏异常、听力损失、性腺功能减退、身高偏低等表现,偶尔也会出现高血糖。这种综合征与 SLC29A3 基因突变有关,该基因编码存在于内体、溶酶体和线粒体中的人类平衡转运体 3。受影响的溶酶体和线粒体普遍且无处不在,这导致了该综合征的全身性和表型异质性表现。H 综合征的表现有皮肤、全身和器官特异性。大腿内侧和胫骨的多毛症和色素沉着是其标志性症状。但并非所有患者都会出现这些症状。H 综合征的治疗涉及多学科方法,以解决特定症状和并发症。H 综合征的预后取决于多个因素,包括临床表现的范围和严重程度、并发症的出现以及及时的诊断和治疗。需要进一步研究探讨预后与 H 综合征中出现的不同突变之间的关系。
H syndrome: A histiocytosis-lymphadenopathy plus syndrome. A comprehensive review of the literature.
H syndrome is a rare autosomal recessive genodermatosis that falls under the histiocytosis-lymphadenopathy plus syndrome. The term "H syndrome" includes manifestations such as hyperpigmentation, hypertrichosis, hepatosplenomegaly, heart anomalies, hearing loss, hypogonadism, low height, and occasionally hyperglycemia. The syndrome is associated with mutations in the SLC29A3 gene, which encodes the human equilibrative transporter 3 present in endosomes, lysosomes, and mitochondria. The generalized and ubiquitous presence of affected lysosomes and mitochondria contributes to the systemic and phenotypically heterogeneous manifestations of the syndrome. H syndrome manifestations are cutaneous, systemic, and organ-specific. The pathognomonic signs are hypertrichosis and hyperpigmentation in the inner thighs and shins. However, not all patients present with these symptoms. H syndrome management involves a multidisciplinary approach to address specific symptoms and complications. The prognosis of H syndrome depends on several factors, including the extent and severity of clinical manifestations, the presence of complications, and timely diagnosis and management. Further studies are needed to explore the association between prognosis and the different mutations encountered in H syndrome.