Abstract

Kaposi's sarcoma was first described by the Hungarian dermatologist Moritz Kaposi in 1872. This mysterious vascular tumor has since received increasing attention, especially after its association with AIDS was discovered in 1981. Kaposi's sarcoma is an indicative disease of AIDS, which is clinically divided into four forms: the classic (Mediterranean), endemic (African), epidemic (HIV/AIDS-associated), and iatrogenic (transplant-related). All four types share the same causative virus but have distinct epidemiological and clinical presentations [1,2]. Kaposi's sarcoma is now considered as a low-grade vascular tumor and is caused by Kaposi's sarcoma herpesvirus/human herpesvirus-8 (KSHV/HHV-8) infection [3,4]. It usually involves the skin, lymphatic system and viscera, most notably the respiratory and gastrointestinal tracts [5–7], whereas involvement of the glottis alone is rare. In particular, acute laryngeal obstruction occurs when Kaposi's sarcoma completely obstructs the glottis, which is life-threatening. This report summarized the clinical diagnosis and treatment of a case of AIDS complicated with glottic Kaposi's sarcoma. A 31-year-old man with HIV infection, had a history of homosexual sex. Antiviral drugs were stopped 2 years ago after antiretroviral therapy (ART) failure. Hoarseness and dyspnea occurred 3 months before admission without obvious inducement and became significantly worse after exercise. Since then, the symptoms gradually aggravated, and the patient were transferred to the infection department of our hospital for hospitalization. Fiberoptic bronchoscopy: under the microscope, purplish red nodules with different sizes of 0.3–0.8 cm are fused into clusters (Fig. 1a). Laboratory studies showed that antibody testing for HIV type 1 was positive. The viral load was 237 000 copies per milliliter (reference range, <40), and the CD4+ cell count was 2 per cubic millimeter (reference range, 500–1200). Glottic biopsy specimen staining with hematoxylin–eosin (HE) showed that spindle cell tumor (Fig. 1c) and immunohistochemical (IHC) testing for HHV-8 was positive (Fig. 1d). These findings were consistent with Glottic Kaposi's sarcoma. Highly active antiretroviral therapy regimen was treated with Albuvirtide+Twinaqt, and tracheotomy was performed. Two weeks after operation, the metal endotracheal tube was replaced. The patient breathed with tube, and was given liposomal doxorubicin 20 mg/m2 ivgtt D1 q3w chemotherapy for one course of treatment. After 3 months, laryngoscope was rechecked, and the glottic mucosa completely returned to normal (Fig. 1b). The patient's voice returned to normal without dyspnea. The metal endotracheal tube was successfully removed. After the tracheotomy fistula was closed, the patient continued to complete four chemotherapy courses, and then the rechecked viral load was less than 40 copies per milliliter, and the CD4+ cell count was 246 per cubic millimeter.Fig. 1: Fiberoptic bronchoscopy showed that there were multiple purplish red nodules of different sizes of 0.3–0.8 cm.Clinically, Kaposi's sarcoma mainly involves the skin and gastrointestinal mucosa, and only involving the glottis leading to acute respiratory obstruction is extremely rare. There is little clinical diagnosis and treatment experience. At present, there is no unified standard for the treatment of Kaposi's sarcoma. Radiotherapy, chemotherapy, local freezing, laser, surgical resection, and other methods can be selected, and HAART treatment needs to be restarted. For this rare case of AIDS complicated by glottis Kaposi's sarcoma with serious acute laryngeal obstruction and dyspnea, if not treated in time, it will lead to airway obstruction, even life-threatening. The patient had severe immune deficiency and extremely low CD4+ cells. If tracheotomy or thyrocricocentesis is performed according to the treatment principle of conventional patients to relieve airway obstruction, it may lead to incision infection, fistula nonunion, tracheal stenosis, and even sepsis and MODS. Therefore, in this case, we innovated the treatment mode, focusing on minimally invasive treatment, and selected the tracheal tube matching with the narrow airway through nasal tracheal intubation to open the airway to relieve airway obstruction, save lives, and win time for the next treatment; Then the oral cavity was chosen as the treatment path, with a wider field of vision exposure, and the glottis tumor was removed by high-frequency electrosection. To sum up, our treatment method can avoid trachea stenosis, intubation breathing and long-term inability to speak caused by incision infection, which will seriously affect the quality of life. Nasal intubation and oral treatment do not interfere with each other and have no impact on the operation. Furthermore, minimally invasive treatment eliminates new microorganisms, immediately relieves airway obstruction, is more conducive to retaining tissue samples, and is not easy to relapse after extubation. There are also few complications, a short hospitalization time, and a low cost. In conclusion, we summarized the diagnosis and treatment experience of this rare case, hoping to provide reference for the diagnosis and treatment of similar patient. Acknowledgements Conflicts of interest There are no conflicts of interest.

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