肺结核:伟大的模仿者。这一次类似朗格汉斯细胞组织细胞增多症

Catalina Arango-ferreira
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Similarities in clinical presentation as well as differences between both conditions that can help distinguish between these diseases and define the diagnosis are shortly discussed, along with the description of treatments and clinical evolution of the reported patient. *Correspondence to: Catalina Arango-Ferreira, University of Antioquia Medellín, Colombia, E-mail: catarango52@hotmail.com Received: February 21, 2019; Accepted: March 05, 2019; Published: March 08, 2019 Introduction LCH mimics many other conditions and a definitive diagnosis requires a combination of clinical manifestations, histological findings and specific immunohistochemistry markers. Tuberculosis is one of the most frequent differential diagnosis, but still, the clinical and radiological manifestations overlap between these two diseases. A high index of suspicion is necessary to clarify the diagnosis and offer timely treatment. Case report An 8-year-old, apparently previously healthy boy, presented to the emergency room with 3 months of a left paracostal abscess and a frontal subcutaneous nodule, accompanied by weight loss and intermittent non quantified fever. Skull radiography revealed osteolytic lesions of the frontal bone (Figure 1) and chest X rays showed osteolytic lesions of the left tenth and eleventh ribs, without pulmonary compromise. A 3-phase bone scintigraphy, reported abnormal cranial, costal and vertebral (T2, T4 and T9) capitation (Figure 2). Suspecting Langerhans cell Histiocytosis (LCH), a biopsy of the frontal bone along with drainage of the paracostal soft tissue abscess were performed. The Ziehl-Neelsen stain of pus drained from the paracostal abscess reported acid-fast bacilli (Figure 3). After growth in culture, a rapid test (SD BIOLINE TB Ag MPT64 Rapid TEST) confirmed M. tuberculosis complex, later identified as M. tuberculosis by PCR (GenoType Direct mycobacterial assay). Frontal bone biopsy revealed multinucleated cells with granulomas. S100 and CDA1 immunohistochemical markers to exclude LCH came back negative. The bone marrow biopsy and aspirate were both normal, as well as a negative 4th generation HIV test. No different organic compromise was identified except the multiple bones previously mentioned. The patient was started on anti TB treatment with HRZE during the first two months of therapy and continued on Isoniazid/Rifampicin for 10 months 3 times weekly. He completed one year of therapy with clinical improvement and resolution of symptoms. He was referred to immunology to exclude primary immunodeficiency due to his unusual clinical presentation with multiple TB osteomyelitis foci and a family history of a sister with previous Pott’s disease. Figure 1. Osteolytic lesions of the frontal bone on skull radiography Discussion LCH of the bones is a relatively rare disorder of unknown etiology, most commonly reported to occur in children from one to three years of age [1]. A single (monostotic) bone lesion is a more frequent presentation than multiple (polyostotic) osseous compromise. In children, the skull is the most commonly affected bone. Conventional radiographs serve as the primary imaging method used to identify LCH lesions, since skull Arango-Ferreira C (2019) Tuberculosis: the great mimicker. This time resembling Langerhans cell histiocytosis Volume 2: 2-2 Med Case Rep Rev, 2019 doi: 10.15761/MCRR.1000128 lesions classically have a punched-out, lytic appearance. For diagnostic confirmation, the histopathology is necessary, since the clinical and imaging characteristics of LCH may overlap with several other diseases, including infections like tuberculosis [2]. Tuberculosis (TB) has always been called the great mimicker due to its various clinical presentations, sometimes with nonspecific symptoms. It can affect almost any organ including bones; of these, the most frequently affected are the vertebrae in 41% of cases [3], Non-weight-bearing bones, such as the skull, clavicle, and mandible, are less often involved. About 50% of children with skeletal TB have abnormal chest radiographs. Confirmation of this diagnosis is based on microbiological identification in stains, cultures, pathology, or molecular methods [4].","PeriodicalId":93315,"journal":{"name":"Journal of medical case reports and reviews","volume":"24 1","pages":""},"PeriodicalIF":0.0000,"publicationDate":"2019-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Tuberculosis: the great mimicker. 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Frontal bone biopsy revealed multinucleated cells with granulomas. S100 and CDA1 immunohistochemical markers to exclude LCH came back negative. The bone marrow biopsy and aspirate were both normal, as well as a negative 4th generation HIV test. No different organic compromise was identified except the multiple bones previously mentioned. The patient was started on anti TB treatment with HRZE during the first two months of therapy and continued on Isoniazid/Rifampicin for 10 months 3 times weekly. He completed one year of therapy with clinical improvement and resolution of symptoms. He was referred to immunology to exclude primary immunodeficiency due to his unusual clinical presentation with multiple TB osteomyelitis foci and a family history of a sister with previous Pott’s disease. Figure 1. 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引用次数: 0

摘要

本文报告了一名健康的8岁男孩的临床病例,他在急诊室就诊时出现了体质症状,并伴有继发于结核病感染的颅骨、肋骨和椎骨的多发性溶骨性病变,类似朗格汉斯细胞组织细胞增多症(LCH)。诊断通过额骨活检明确,发现多核细胞伴肉芽肿。S100和CDA1免疫组化标记阴性有助于排除。此外,在脓的Ziehl-Neelsen染色中发现一肋旁软组织脓肿的引流液中有抗酸杆菌。骨髓活检及穿刺均正常。临床表现的相似之处以及两种情况之间的差异可以帮助区分这两种疾病并确定诊断,并将简要讨论治疗方法的描述和所报告患者的临床进展。*通讯作者:Catalina Arango-Ferreira,哥伦比亚安蒂奥基亚大学Medellín, E-mail: catarango52@hotmail.com录用日期:2019年3月05日;LCH与许多其他疾病相似,明确的诊断需要结合临床表现、组织学发现和特异性免疫组织化学标志物。结核病是最常见的鉴别诊断之一,但这两种疾病的临床和影像学表现仍然重叠。高度的怀疑指数对于明确诊断和提供及时治疗是必要的。病例报告一名8岁男孩,先前明显健康,因3个月的左侧肋旁脓肿和额部皮下结节就诊于急诊室,伴有体重减轻和间歇性非量化发热。颅骨X线片显示额骨溶骨性病变(图1),胸部X线片显示左侧第十和第十一肋骨溶骨性病变,肺部未受损。3期骨显像显示颅骨、肋部和椎体(T2、T4和T9)头颅异常(图2)。怀疑为朗格汉斯细胞组织细胞增生症(LCH),行额骨活检并引流肋旁软组织脓肿。肋旁脓肿排出的脓液的Ziehl-Neelsen染色报告了抗酸杆菌(图3)。培养生长后,快速测试(SD BIOLINE TB Ag MPT64 rapid test)证实了结核分枝杆菌复合体,后来通过PCR(基因型直接分枝杆菌试验)鉴定为结核分枝杆菌。额骨活检显示多核细胞伴肉芽肿。用于排除LCH的S100和CDA1免疫组织化学标志物均为阴性。骨髓活检和抽吸均正常,第四代HIV检测阴性。除了之前提到的多块骨头外,没有发现其他的有机损伤。患者在治疗的前两个月开始使用HRZE进行抗结核治疗,并继续使用异烟肼/利福平10个月,每周3次。他完成了一年的治疗,临床改善,症状缓解。由于其不寻常的临床表现为多发性结核灶性骨髓炎,且其姐姐既往有波特病家族史,因此转介免疫学以排除原发性免疫缺陷。图1所示。骨的LCH是一种病因不明的相对罕见的疾病,最常见于1至3岁的儿童[1]。单一(单骨)骨损伤比多发性(多骨)骨损伤更常见。在儿童中,颅骨是最常受影响的骨骼。传统x线片是用于识别LCH病变的主要成像方法,因为头骨Arango-Ferreira C(2019)结核病:伟大的模仿者。这一次类似于朗格汉斯细胞组织细胞增多症卷2:2-2医学病例报告Rev . 2019 doi: 10.15761/MCRR。1000128病灶典型表现为穿孔、溶解样。由于LCH的临床和影像学特征可能与其他几种疾病重叠,包括结核病等感染[2],因此需要进行组织病理学检查才能确诊。结核病(TB)由于其临床表现多样,有时伴有非特异性症状,一直被称为大模仿者。它几乎可以影响任何器官,包括骨骼;其中,最常见的受累部位是椎骨,占41%[3],非承重骨,如颅骨、锁骨和下颌骨较少受累。约50%的骨性结核儿童胸片异常。这种诊断的确认是基于染色、培养、病理或分子方法中的微生物鉴定[4]。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Tuberculosis: the great mimicker. This time resembling Langerhans cell histiocytosis
The clinical case of a previously healthy 8 year old boy who presented to the emergency room with constitutional symptoms, along with multiple osteolytic lesions of the skull, ribs and vertebrae secondary to Tuberculosis infection resembling Langerhans cells Histiocytosis (LCH) is presented. Diagnosis was clarified with a biopsy of the frontal bone which revealed multinucleated cells with granulomas. Negative S100 and CDA1 immunohistochemical markers helped exclude. Also, drainage of a paracostal soft tissue abscess reported acid-fast bacilli seen in the Ziehl-Neelsen stain of pus. Bone marrow biopsy and aspirate were both normal. Similarities in clinical presentation as well as differences between both conditions that can help distinguish between these diseases and define the diagnosis are shortly discussed, along with the description of treatments and clinical evolution of the reported patient. *Correspondence to: Catalina Arango-Ferreira, University of Antioquia Medellín, Colombia, E-mail: catarango52@hotmail.com Received: February 21, 2019; Accepted: March 05, 2019; Published: March 08, 2019 Introduction LCH mimics many other conditions and a definitive diagnosis requires a combination of clinical manifestations, histological findings and specific immunohistochemistry markers. Tuberculosis is one of the most frequent differential diagnosis, but still, the clinical and radiological manifestations overlap between these two diseases. A high index of suspicion is necessary to clarify the diagnosis and offer timely treatment. Case report An 8-year-old, apparently previously healthy boy, presented to the emergency room with 3 months of a left paracostal abscess and a frontal subcutaneous nodule, accompanied by weight loss and intermittent non quantified fever. Skull radiography revealed osteolytic lesions of the frontal bone (Figure 1) and chest X rays showed osteolytic lesions of the left tenth and eleventh ribs, without pulmonary compromise. A 3-phase bone scintigraphy, reported abnormal cranial, costal and vertebral (T2, T4 and T9) capitation (Figure 2). Suspecting Langerhans cell Histiocytosis (LCH), a biopsy of the frontal bone along with drainage of the paracostal soft tissue abscess were performed. The Ziehl-Neelsen stain of pus drained from the paracostal abscess reported acid-fast bacilli (Figure 3). After growth in culture, a rapid test (SD BIOLINE TB Ag MPT64 Rapid TEST) confirmed M. tuberculosis complex, later identified as M. tuberculosis by PCR (GenoType Direct mycobacterial assay). Frontal bone biopsy revealed multinucleated cells with granulomas. S100 and CDA1 immunohistochemical markers to exclude LCH came back negative. The bone marrow biopsy and aspirate were both normal, as well as a negative 4th generation HIV test. No different organic compromise was identified except the multiple bones previously mentioned. The patient was started on anti TB treatment with HRZE during the first two months of therapy and continued on Isoniazid/Rifampicin for 10 months 3 times weekly. He completed one year of therapy with clinical improvement and resolution of symptoms. He was referred to immunology to exclude primary immunodeficiency due to his unusual clinical presentation with multiple TB osteomyelitis foci and a family history of a sister with previous Pott’s disease. Figure 1. Osteolytic lesions of the frontal bone on skull radiography Discussion LCH of the bones is a relatively rare disorder of unknown etiology, most commonly reported to occur in children from one to three years of age [1]. A single (monostotic) bone lesion is a more frequent presentation than multiple (polyostotic) osseous compromise. In children, the skull is the most commonly affected bone. Conventional radiographs serve as the primary imaging method used to identify LCH lesions, since skull Arango-Ferreira C (2019) Tuberculosis: the great mimicker. This time resembling Langerhans cell histiocytosis Volume 2: 2-2 Med Case Rep Rev, 2019 doi: 10.15761/MCRR.1000128 lesions classically have a punched-out, lytic appearance. For diagnostic confirmation, the histopathology is necessary, since the clinical and imaging characteristics of LCH may overlap with several other diseases, including infections like tuberculosis [2]. Tuberculosis (TB) has always been called the great mimicker due to its various clinical presentations, sometimes with nonspecific symptoms. It can affect almost any organ including bones; of these, the most frequently affected are the vertebrae in 41% of cases [3], Non-weight-bearing bones, such as the skull, clavicle, and mandible, are less often involved. About 50% of children with skeletal TB have abnormal chest radiographs. Confirmation of this diagnosis is based on microbiological identification in stains, cultures, pathology, or molecular methods [4].
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