不同寻常的手臂疣状皮肤病

Luisa Unterluggauer, Philipp Tschandl, Nicole Harrison, Liliane Borik-Heil
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There was no palpable lymphadenopathy and no systemic symptoms like fever were reported. A skin biopsy was made (Figure 2a).</p><p>Tuberculosis verrucosa cutis</p><p>A skin biopsy showed a pseudoepitheliomatous hyperplasia with a dense dermal superficial lympho-histiocytic inflammatory infiltrate including multinucleated giant cells, suggesting an infectious cause (Figure 2a). Immunohistochemistry confirmed the abundance of CD68+ macrophages (Figure 2b). Acid-fast staining (Ziehl-Neelsen) and a broad-spectrum polymerase chain reaction from the tissue specimen were negative for fungal and bacterial species including <i>Mycobacterium tuberculosis</i>. However, the culture obtained from this skin tissue confirmed the presence of <i>M. tuberculosis</i> complex 6 weeks later. The computed tomography (CT) of the thorax showed pleural consolidations, compatible with former tuberculosis (TB), supported by a positive interferon gamma release assay (IGRA). 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引用次数: 0

摘要

一名来自阿富汗的 55 岁妇女在 8 周前出现左前臂多处淡红色病变。值得注意的是,2 年前腕管综合征浸润后,她的左前臂出现了反复发作的痰。临床检查显示,她的左前臂背侧有多处红斑,部分角化过度,面积达 3 厘米(图 1)。这些斑块无疼痛感,按压时有脓液流出。此外,在植皮处和左前臂正常皮肤上还能观察到单发的红斑丘疹。其余皮肤无异常。未触及淋巴结肿大,也未报告发热等全身症状。皮肤活检显示有假上皮瘤样增生,真皮浅层有密集的淋巴-组织细胞炎症浸润,包括多核巨细胞,提示病因是感染(图 2a)。免疫组化证实了大量的 CD68+ 巨噬细胞(图 2b)。组织标本的耐酸染色(Ziehl-Neelsen)和广谱聚合酶链反应结果显示,真菌和细菌(包括结核分枝杆菌)均呈阴性。然而,6 周后从该皮肤组织中提取的培养物证实了结核分枝杆菌复合体的存在。胸腔计算机断层扫描(CT)显示,胸膜有合并症,与以前的结核病(TB)相符,干扰素γ释放测定(IGRA)呈阳性。通过 CT 和核磁共振成像,可以排除其他器官受累的可能性。痰标本中结核杆菌呈阴性。患者接受了标准的抗结核治疗,包括口服利福平、乙胺丁醇、吡嗪酰胺和异烟肼联合治疗 2 个月,随后又接受了利福平和异烟肼治疗 4 个月。皮损缓解,仅残留炎症后色素沉着(图 3)。肺外结核病(TB)约占所有肺结核病例的 10%-15%,通常表现为淋巴结炎、胸膜炎或骨关节结核。3 它们可表现为不同的临床表型,如溃疡、结节性病变、脓肿和疣状病变,这取决于不同的因素,如感染途径、感染者的免疫力和以前对结核杆菌的敏感性。皮肤疣状结核病(Tuberculosis verrucosa cutis,TVC)是一种罕见(4.3%)的皮肤结核病,多发于免疫力良好的既往致敏者。在多脓疱型结核(即结核性软下疳、瘰疬性皮肤病、人工结核、急性粟粒性结核和转移性脓肿/结核性牙龈肿)中,很容易在组织中检测到结核杆菌,而在较少见的贫脓疱型结核(即:TVC、寻常狼疮)中,则很容易在组织中检测到结核杆菌、4 在全球北方国家,感染非常罕见,通常与特定职业有关,如屠夫、农民和病理学家,导致手部病变。7 在我们的病例中,左前臂上的慢性伤口被认为是病原体的侵入点。本病例说明,皮肤结核虽然是肺外结核的一种罕见形式,在全球北方国家更为罕见,但需要在异常溃疡的鉴别诊断中加以考虑。虽然 PCR 对皮肤结核病的总体敏感性为 73%,但在 TVC 等弱碱性结核病中,敏感性则降至 56%。8 因此,检测弱碱性分枝杆菌感染的诊断程序极具挑战性,必须包括组织样本的显微镜检查、PCR,尤其是培养,因为分枝杆菌的载量可能不同,而且可能低于 PCR 的检测阈值。所有三项检查都应在每位患者身上进行,如果最初结果为阴性,但仍怀疑是皮肤结核,则应重复检查:Luisa Unterluggauer、Liliane Borik-Heil、Philipp Tschandl 和 Nicole Harrison。起草手稿:Luisa Unterluggauer 和 Liliane Borik-Heil。对手稿中的重要思想内容进行严格修改:Liliane Borik-Heil、Philipp Tschandl 和 Nicole Harrison。
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Verrucous cutaneous lesions on the arm of unusual origin

A 55-year-old woman from Afghanistan presented with multiple reddish lesions on the left forearm that appeared 8 weeks prior. Of note, after infiltration of a carpal tunnel syndrome 2 years ago, she had developed recurrent phlegmons on the left forearm. This had led to an ulcer which was covered by a split skin graft 1.5 years ago.

The clinical examination showed several livid-erythematous, partially hyperkeratotic plaques, measuring up to 3 cm, on the dorsal left forearm (Figure 1). These non-painful plaques presented with expression of pus on pressure. In addition, solitary erythematous papules on the skin graft as well as on normal skin of the left forearm could be observed. The rest of the skin was unremarkable. There was no palpable lymphadenopathy and no systemic symptoms like fever were reported. A skin biopsy was made (Figure 2a).

Tuberculosis verrucosa cutis

A skin biopsy showed a pseudoepitheliomatous hyperplasia with a dense dermal superficial lympho-histiocytic inflammatory infiltrate including multinucleated giant cells, suggesting an infectious cause (Figure 2a). Immunohistochemistry confirmed the abundance of CD68+ macrophages (Figure 2b). Acid-fast staining (Ziehl-Neelsen) and a broad-spectrum polymerase chain reaction from the tissue specimen were negative for fungal and bacterial species including Mycobacterium tuberculosis. However, the culture obtained from this skin tissue confirmed the presence of M. tuberculosis complex 6 weeks later. The computed tomography (CT) of the thorax showed pleural consolidations, compatible with former tuberculosis (TB), supported by a positive interferon gamma release assay (IGRA). Further organ involvement could be ruled out via CT and MRI. Sputum specimens were negative for M. tuberculosis. The patient's husband and daughter also had a positive IGRA, but there was no active TB in the patient's social environment.

The patient received a standard tuberculostatic therapy which consisted of an oral combination of rifampicin, ethambutol, pyrazinamide, and isoniazid for 2 months, followed by a treatment with rifampicin and isoniazid for 4 months. The lesions showed remission with just remaining post-inflammatory hyperpigmentation (Figure 3). The IGRA-positive relatives were treated with rifampicin for 4 months.

Extrapulmonary tuberculosis (TB) accounts for approximately 10%–15% of all TB cases and usually manifests as lymphadenitis, pleuritis, or osteoarticular TB.1, 2 Cutaneous forms of TB are very rare, comprising approximately 1%–2% of all TB cases.3 They can present as different clinical phenotypes, for example, ulcers, nodular lesions, abscesses, and verrucous lesions, depending on different factors such as the route of infection, immunity of the infected individual and previous sensitisation to M. tuberculosis.4

Tuberculosis verrucosa cutis (TVC) is a rare (4.3%) form of cutaneous TB that occurs in previously sensitised individuals with good immunity.5 The route of infection is exogenous via inoculation through wounds with infectious sputum or contaminated objects.6 Based on this, the bacterial load within the skin varies in these different forms of cutaneous TB. In multibacillary forms (i.e., tuberculous chancre, scrofuloderma, orificial TB, acute miliary TB, and metastatic abscess/tuberculous gumma), bacilli are easily detected in tissue, while in the less common paucibacillary forms (i.e., TVC, lupus vulgaris) it is difficult to prove the organisms.4 In countries of the global north infections are rare and usually associated with specific professions such as butchers, farmers and pathologists resulting in lesions on the hands.7 For the latter this had led to the historic term of ‘prosector's wart’ or ‘postmortem wart’.7

In our case, the chronic wound on the left forearm is considered as the entry point for the pathogen.

This case illustrates that cutaneous TB, though a rare form of extrapulmonary TB and even rarer in countries of the global north, needs to be considered in the differential diagnosis of an unusual ulceration. While the overall sensitivity of PCR in cutaneous TB is 73%, it decreases in cases of pauci-bacillary forms, like TVC, to 56%.8 Therefore diagnostic procedures to detect paucibacillary mycobacterial infections are challenging and must include microscopy from tissue samples, PCR and—in particular—culture since the load of mycobacteria may vary and be below the PCR detection threshold. All three should be performed in every patient and should be repeated in case of initially negative results but persisting suspicion of cutaneous TB.

Substantial contributions to conception and design, or acquisition of data, or analysis and interpretation of data: Luisa Unterluggauer, Liliane Borik-Heil, Philipp Tschandl and Nicole Harrison. Drafting manuscript: Luisa Unterluggauer and Liliane Borik-Heil. Critically revising the manuscript for important intellectual content: Liliane Borik-Heil, Philipp Tschandl and Nicole Harrison. Final approval of the version to be published: all authors.

The authors declare no conflict of interest.

All patients in this manuscript have given written informed consent for participation in the study and the use of their deidentified, anonymized, aggregated data and their case details (including photographs) for publication. Ethical approval: not applicable.

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