Months Of Antituberculous Therapy Research Articles

A patient with Tuberculous para-valvular leak and tuberculous hepatic abscesses treated conservatively with anti-tuberculous medication without the need for surgical intervention. “Case report”

Introduction: We report a case of a young lady with rheumatic heart disease who underwent mechanical prosthetic mitral valve replacement and tricuspid valve repair. The case was complicated after one year by multi system tuberculous abscesses, para-valvular leak and hemolytic anemia. Case summary: The case started one year following mechanical prosthetic mitral valve replacement, and after contact with a tuberculous patient. The lady developed, sterile pyuria, two tuberculous hepatic abscesses, and a big pelvic tuberculous abscess. She first needed percutaneous drainage for the pelvic abscess followed by laparotomy and surgical drainage after recurrence. The case was complicated by Para-valvular leak in the prosthetic mitral valve detected by Transoesophageal echocardiography as well as refractory hemolytic anemia needing multiple transfusions. She required six months of anti-tuberculous therapy for the hemolysis to stop. Treatment continued for a total of one year of anti-tuberculous therapy. The patient restored normal valve dynamics and hematological parameters without the need for surgical intervention for the para-valvular leak. Conclusion: We report this case to show that small tuberculous para-valvular leak and hemolytic anemia as well as tuberculous hepatic abscesses can be treated medically as an alternative option to the well-known and recommended surgical indication. We also confirm delayed healing of the abdominal surgical wound due to tuberculosis.

Cutaneous Tuberculosis in Heart Transplant

Tuberculosis is a disease with a significant global burden in terms of morbidity and mortality. It usually presents as a pulmonary disease but can occasionally have extrapulmonary presentations. Immunosuppressed people are at an increased risk of tuberculosis and more frequently have atypical manifestations of the disease. Cutaneous involvement is estimated to occur in only 2% of extrapulmonary presentations. We report a case of a heart transplant recipient with disseminated tuberculosis who initially presented with cutaneous manifestations in the form of multiple abscesses that were mistaken for a community-acquired bacterial infection. The diagnosis was made after positive nucleic acid amplification testing and cultures for Mycobacterium tuberculosis from the drainage of the abscesses. After initiating antituberculous treatment, the patient had 2 instances of immune reconstitution inflammatory syndrome. A combination of diminished immunosuppression due to discontinuation of mycophenolate mofetil in the setting of acute infection, rifampin drug interactions with cyclosporine, and the beginning of treatment of tuberculosis all contributed to this paradoxical worsening. The patient responded favorably to increased glucocorticoid therapy and showed no signs of treatment failure after 6 months of antituberculous therapy.

Surgical resection of a tuberculoma in the diaphragm: a case report

BackgroundExtrapulmonary tuberculosis commonly affects the lymphatic system, nervous system, and gastrointestinal system. Tuberculous infection in the muscle is very rare. Moreover, tuberculous infection in the diaphragm is extremely rare. We herein report a case of tuberculomas in the diaphragm and posterior mediastinum that were successfully diagnosed and treated.Case presentationWe encountered a 62-year-old woman with a tuberculoma in the diaphragm. The patient presented with mild dyspnea. Computed tomography showed a mass in the left diaphragm, focal thickening of the posterior mediastinum, and multiple nodules in the lungs. Positron emission tomography–computed tomography showed increased uptake in the left diaphragm mass and thickening of the posterior mediastinum; therefore, we considered the masses to be malignant and planned surgical resection. However, the patient was diagnosed with tuberculosis from a sputum culture, and she was treated with anti-tuberculous therapy. The masses in the diaphragm and posterior mediastinum had become enlarged after 6 months of anti-tuberculous therapy; therefore, the patient underwent resection of both masses. Tuberculous infection was histologically confirmed in each lesion. She was pathologically diagnosed with tuberculous abscesses in the diaphragm and posterior mediastinum and began treatment with anti-tuberculosis drugs.ConclusionsPreoperative diagnosis of a tuberculoma in the diaphragm is usually difficult, and surgical intervention is important for both diagnosis and treatment.

Pulmonary tuberculosis and diabetes mellitus: Clinical profile and outcomes.

India is endemic for Tuberculosis (TB), contributing to the world's highest number of active cases. Diabetes (DM), with its increasing burden in India, could contribute to adverse outcomes among patients with TB. Consecutive patients with sputum smear positive pulmonary tuberculosis were included in the study. We defined cases as those patients with diabetes at recruitment. Controls were non diabetics (NDM). Sputum samples for AFB smears, AFB culture and Xpert PCR along with blood samples for glycosylated Haemoglobin and glucose levels were collected at recruitment and at 6 months from patients with sputum positive pulmonary TB. Blood glucose levels and sputum smears were repeated at 2 months and monthly till they tested negative. The primary outcome studied was mortality at 6 month follow-up. The secondary outcomes included the time to conversion of sputum smears and cure rates between cases and controls. We recruited 124 patients of which 68 were cases. Mortality after therapy was 15% in cases and 7% in controls, however, the difference was not statistically significant. Equal proportions in each group (Diabetics: 9% vs. NDM 9%) had persistent smear positivity at 2 months. There was no association between delayed sputum conversion and uncontrolled diabetes. Only about 57% of cases and 50% of controls were documented to have completed treatment or been cured. A significant reduction in HbA1c after 6 months of Antituberculous therapy was noted among the cases. [Mean difference - 1.76, P-value - 0.001, 95% CI of difference - (1.01 - 2.52)]. Diabetes did not have adverse outcomes in the form of increased mortality or delayed sputum conversion rates. The high proportion of loss to follow-up seems to be a trend of concern, which should be addressed emergently.

Infliximab-related tubercular spinal epidural abscess without osseous involvement in a patient with Crohn’s disease: A case report

Tubercular spinal epidural abscess without osseous involvement is an extremely rare presentation of spinal tuberculosis. We report a case of perianal Crohn’s disease, who developed a tubercular epidural abscess in lumbosacral spine without osseous involvement, secondary to infliximab administration, despite being on 6 months of antituberculous therapy for perianal tuberculosis even as the primary focus healed well. This is probably the first case of infliximab-related tubercular spinal epidural abscess without osseous involvement. A combination of surgical treatment and antitubercular therapy gives a good outcome. This case highlights the rare and atypical nature of presentation of spinal tuberculosis in this scenario and emphasizes the importance of vigilant follow-up to prevent delayed diagnosis, due to atypical presentations following anti-tumor necrosis factor therapy.

Multifocal Subcutaneous and Muscular Tuberculosis in a Young Immunocompetent Man: A Case Report and Literature Review

Due to unfavorable conditions for survival and multiplication of Mycobacterium tuberculosis, muscle and soft tissue involvement is probably the rarest form of presentation of musculoskeletal tuberculosis, particularly without a clinically or radiologically apparent primary focus. As in other extrapulmonary forms of tuberculosis, its relative incidence is higher among immunocompromised patients. A progressive swelling is the most common complaint at presentation, which is otherwise mild and nonspecific. We present a case of multifocal (seven) subcutaneous and muscular tuberculous abscesses in a 29-year-old Indian patient who had no apparent underlying risk factors, nor any other organ involvement. Provisional diagnosis was initially made upon Mycobacterium tuberculosis complex nucleic acid detection in an aspirate of one of the collections and later confirmed by a positive culture on Lowenstein-Jensen medium. Two abscesses required aspiration and two others drained spontaneously. The patient responded well to nine months of anti-tuberculous therapy. An extensive review of the literature was made.

Unusual Tuberculosis of the Frontal Bone: A Case Report

Tuberculosis is a chronic infectious disease caused by Mycobacterium tuberculosis bacteria that can affect almost any part of the body, especially in lung. Tuberculosis of the bones is not an unusual condition. However, the occurrence of tuberculosis of the skull is much less compared with the other bones. We report a 19-year-old man exhibiting swelling of the scalp and a bony defect of the skull caused by irregular anti-tuberculosis treatment. He was diagnosed with tuberculosis of right upper lung and underwent the anti-tuberculosis treatment for about 20 days in the local contagious hospital. However, he did not adhere to anti-TB treatment due to feeling better after anti-tuberculosis treatment. He had to receive anti-tuberculous chemotherapy again and underwent surgery after the occurrence of the tuberculosis of frontal bone. Fortunately, the patient completed 9 months of anti-tuberculous therapy and fully recovered. To our knowledge, this is the first report of the tuberculosis of frontal bone caused by irregular anti-tuberculosis treatment.

Characteristics of residual lymph nodes after six months of antituberculous therapy in HIV-negative individuals with cervical tuberculous lymphadenitis

BackgroundThe therapeutic response of cervical tuberculous lymphadenitis (CTBL) may be delayed or paradoxical, with the frequent development of residual lymph nodes (LNs) during and after antituberculous treatment. We investigated the incidence of residual LNs and the clinical, radiological, microbiological, and pathologic responses of patients with CTBL after 6 months of antituberculous therapy.MethodsThe medical records of HIV-negative adult patients with CTBL diagnosed between July 2009 and December 2017 were analyzed. After 6 months of first-line antituberculous treatment, computed tomography (CT) scans were conducted to evaluate for residual LNs. Fine-needle aspiration biopsy (FNAB) was carried out if a patient presented with residual LNs > 10 mm in diameter with central necrosis, peripheral rim enhancement, or perinodal inflammation on CT scan.ResultsResidual LNs were detected in 35 of 157 patients who underwent follow-up CT scans and were more commonly observed in younger patients who completed the treatment (mean years ± standard deviation [SD]: 33 ± 13 vs. 44 ± 16, p < 0.001). The recurrence rate was approximately 5%, which was not significantly different in both groups. Among the 15 patients who underwent FNAB, 3 (30%) presented with granuloma, and 2 of 15 and 10 of 14 patients had positive AFB and TB PCR results, respectively. The TB culture results of 15 patients were negative.ConclusionsResidual LNs may still be observed after 6 months of antituberculous treatment. Although the radiologic and pathologic findings after treatment are still indicative of TB, not all residual LNs indicate recurrence or treatment failure. A six-month therapy may be sufficient for cervical tuberculous lymphadenitis.

Clinical Profile and Treatment Response of Patients with Ocular Inflammation due to Presumed Ocular Tuberculosis: A Retrospective Study

Objectives:Ocular tuberculosis is an extrapulmonary tuberculous infection and has varying manifestations which pose a huge challenge to diagnosis and treatment. The purpose of this study is to describe the various clinical manifestations of ocular inflammations due to tuberculosis and to assess the response to treatment following antituberculous therapy (ATT) and corticosteroids in these patients.Materials and Methods:We performed a retrospective analysis of 29 patients with presumed ocular tuberculosis who were started on ATT and completed follow-up of at least 6 months after ATT was initiated. The data collected were: age at presentation, sex, laterality, presence or absence of pulmonary/extrapulmonary tuberculosis, history of exposure to tuberculosis, site of ocular involvement and duration of illness, visual acuity at presentation and at 6-month follow-up, and response to treatment.Results:Most of the patients were of economically productive age, between 21-60 years. This most common presentation in our study population was unilateral nongranulomatous anterior uveitis. In spite of the delay between symptom onset and start of therapy, favorable response was noted in 79.3% of patients at completion of 6 months of ATT. The various reasons for the delay in start of therapy were also evaluated.Conclusion:In this case series, we presented the various ocular manifestations and the difficulties faced in the diagnosis of presumed ocular tuberculosis. Outcomes of ATT were favorable in most of our patients. Thus, the clinician should exercise a very high degree of suspicion and should not withhold a trial of ATT.

Identification of intestinal abnormalities in patients with active pulmonary tuberculosis using small bowel capsule endoscopy.

Background and study aims Mycobacterium tuberculosis is often detected in the feces of patients with active pulmonary tuberculosis. However, no study has examined the small intestine using small bowel capsule endoscopy (SBCE). This study aimed to investigate intestinal abnormalities in patients with active pulmonary tuberculosis. Patients and methods SBCE was performed in sputum/feces smear-positive pulmonary tuberculosis patients. From December 2013 to November 2016, 15 patients were enrolled from a dedicated tuberculous ward. The primary outcome was intestinal abnormalities identified by SBCE. Results Fourteen patients (median age 81 years, range 29 – 91; 3 female) underwent SBCE to the terminal ileum. The video was not recorded for one patient who was excluded from the analysis. Intestinal lesions, including 5 annular ulcers in 4 patients, were observed in 64 % (9/14). In subgroup analysis, prevalence tended to be higher in patients undergoing SBCE within 1 month of anti-tuberculous therapy ( P = 0.051). Distribution of small intestinal lesions tended to be distal. Four of five annular ulcers were located close to the ileocecal valve. Conclusion Prevalence of intestinal lesions in patients with pulmonary tuberculosis is higher than expected before (UMIN 000017292)

A case of tuberculous peritonitis diagnosed by way of laparoscopy

Objective: Tuberculous peritonitis (TBP) is a rare form of extrapulmonary tuberculosis. The diagnosis of TBP is particularly challenging in children. Herein, we present a child who had been admitted with ascites and diagnosed as TBP by way of laparoscopy. Case report: A previously healthy 3-year-old girl was presented with newly onset fever and abdominal pain. She had abdominal distention and generalized tenderness on palpation. Abdominal percussion revealed dullness on nonspesific locations. Abdominal ultrasonography showed diffuse ascites with multiple septations, and paracenthesis performed detected exudative material with plenty of polmorphonuclear leukocytes. She was started on antibiotherapy. Lymphocytic pleocytosis persisted on control peritoneal lavage sample. Acid- resistant bacteria were not seen in the peritoneal fluid, tuberculous polymerase chain reaction test and tuberculous cultures were negative. Cytopathologic examination was not suggestive of malignancy. Diagnostic laparoscopy was compatible with TBP. Adenosine deaminase (ADA) level in peritoneal fluid was increased. She was started on quadruple antituberculous treatment including isoniazid (INH), rifampicin (RIF), pyrazinamide and ethambutol. On the 2nd month of antituberculous therapy, abdominal circumference showed obvious regression. She continued on INH-RIF therapy and is still being followedup in our outpatient clinic. Conclusion: Explorative laparoscopy and peritoneal fluid ADA level are very helpful in the diagnosis of TBP

Réaction paradoxale à la fin du traitement antituberculeux chez le sujet immunocompétent

The features of paradoxical reactions (PR) that occurred in non-HIV infected patients are rare and not well known. The authors reported the case of a 21years old, non-immunocompromised, and HIV negative patient treated for disseminated tuberculosis. PR occurred after 8months after initiation of antituberculous treatment. PR presented as left cervical lymphadenopathy, pulmonary, pleural, costal and spinal location of the tuberculosis. The antituberculous drugs were prolonged. Patient's clinical symptoms improved initially. However, left inguinal lymphadenopathy appeared after 20months of antituberculous therapy. Inguinal lymph node biopsy revealed tuberculous lymphadenitis. The patient has a good compliance to the treatment. The patient was continued on same antituberculous treatment for a total of 28months. The cervical and inguinal lymphadenopathy disappeared and CT scan showed regression of thoracic, abdominal, costal and spinal lesions. PR during antituberculous treatment must be considered after exclusion of other causes. No consensus on the therapeutic management of this entity has been developed to date.

Six months versus nine months anti-tuberculous therapy for female genital tuberculosis: a randomized controlled trial

ObjectiveTo compare six months versus nine months anti-tuberculous therapy in patients of female genital tuberculosis. Study designIt was a randomized controlled trial in a tertiary referral center teaching institute on 175 women presenting with infertility and found to have female genital tuberculosis on clinical examination and investigations. Group I women (86 women) were given 9 months of intermitted anti-tuberculous therapy under directly observed treatment short course (DOTS) strategy while Group II (89 women) were given 6 months of anti-tuberculous therapy under DOTS. Patients were evaluated for primary end points (complete cure, partial response, no response) and secondary end points (recurrence rate, pregnancy rate) during treatment. All patients were followed up further for one year after completion of therapy to assess recurrence of disease and further pregnancies. ResultsBaseline characteristics were similar between two randomized groups. There was no difference in the complete clinical response rate (95.3% vs 97.7%, p=0.441) between 9-months and 6-months groups. Four patients in 9-months group and two patients in 6-months group had recurrence of disease and required category II anti tuberculous therapy (p=0.441). Pregnancy rate during treatment and up to one year follow up was also similar in the two groups (23.2% vs 21.3%, p=0.762). Side effects occurred in 27(31.4%) and 29(32.6%) in 9-months and 6-months of therapy and were similar (p=0.866). ConclusionsThere was no difference in complete cure rate, recurrent rate and pregnancy rate for either 6-months or 9-months of intermittent directly observed treatment short course anti-tuberculous therapy in female genital tuberculosis. Clinical trial registrationThe trial was registered in clinicaltrials.gov with registration no: CTRI/2009/091/001088.

Serum neopterin level in cases of pulmonary tuberculosis and pneumonia

Pulmonary tuberculosis (TB) sometimes has diagnostic difficulties and a lot of differential diagnosis such as pneumonia. The aim of this work was to assess the role of serum neopterin in differentiating between pulmonary TB and pneumonia and to estimate the effect of antituberculous drugs for 2 months on serum levels of neopterin in patients with pulmonary TB. We measured serum neopterin in patients with TB, pneumonia patients, and controls. Serum neopterin was measured again after 2 months of antituberculous therapy in patients with pulmonary TB. Serum neopterin was significantly higher in tuberculous patients than in pneumonia patients, with a sensitivity of 90% and a specificity of 80% at an optimal cut-off value of 20.5 nmol/l. It decreased significantly after 2 months of antituberculous therapy in tuberculous patients. Serum neopterin levels significantly increase in pulmonary TB and correlate with the radiological extent of the disease. Combined anti-TB treatment decreases the levels of serum neopterin. Measurement of the serum neopterin levels may be useful in following up the drug response in pulmonary TB. Serum neopterin levels may also be helpful in discriminating pulmonary TB from pneumonia.

Clinical Presentation, Diagnosis, and Bacterial Epidemiology of Peritoneal Tuberculosis in Two University Hospitals in France.

IntroductionDiagnosis of peritoneal tuberculosis (pTB) is difficult, even in developed countries, where data are lacking. The aim of the present study was to describe the clinical presentation, diagnosis, and bacterial epidemiology of pTB in France over a 10-year period.MethodsA retrospective study was conducted on pTB in two university hospitals in France, between January 2004 and December 2014.ResultsAmong the 34 patients, 76.5% were migrants from areas of endemic tuberculosis (TB), mainly Africa. The main presentation (85.3%) was a checkup of ascites or suspicion of peritoneal carcinomatosis. On abdominal computed tomography, ascites was found in 90.6% and peritoneal thickening in 75%. Surgery was required for diagnosis in 58.8% of patients. Six of the patients who did not undergo surgery had ultrasound-guided peritoneal biopsy. Bacteriology was positive for ascites in only 58.1% of cases, for peritoneal biopsy in 73.3%, while granuloma was found in 95.5%. TB polymerase chain reaction (PCR) was positive in 25% of peritoneal biopsy. Mycobacterium bovis was isolated in 23.1% of cases and Mycobacterium tuberculosis in 76.9%. Isolates were fully susceptible (except M. bovis naturally resistant to pyrazinamide). Many (38%) belonged to the lineage T (genetic analysis by spoligotyping). Cure rate was high (76.5%), after a 6–9 months of anti-tuberculous therapy.ConclusionIn developed countries, early diagnosis of pTB is still a challenge. Ultrasound-guided peritoneal biopsy may facilitate diagnosis. TB PCR can be useful on peritoneal biopsy. The lineage T was the most prevalent lineage, but more data are required to directly incriminate this lineage in the pathophysiology of pTB.

Intradural Extramedullary Tuberculoma of the Spinal Cord Following Tuberculous Meningitis.

Intradural extramedullary tuberculoma of the spinal cord (IETSC) is an uncommon disease which can occurs secondary to tuberculous meningitis. A 31-year-old woman was diagnosed as tuberculous meningitis after mental disorientation. Her mentality was recovered after antituberculous therapy. After 7 months of antituberculous therapy, paraplegia has developed. Magnetic resonance imaging (MRI) revealed a mass lesion between the T1 and T12 spinal levels with arachnoid thickening which results in the development of tuberculoma. She received surgical resection of IETSC followed by antituberculous therapy and neurological function has been improved. The two years after surgical treatment, spinal MRI showed syringomyelia between T1 to L1. But, her neurological outcome was not aggravated.

Factors Impacting Early Mortality in Tuberculosis/HIV Patients: Differences between Subjects Naïve to and Previously Started on HAART

BackgroundMortality among patients with tuberculosis (TB)/HIV is highest during the first few months of antituberculous therapy. The objective of this study was to assess the factors associated with early mortality among TB/HIV patients and whether these factors are similar for HAART naïve and those with prior HAART initiation.MethodsProspective cohort study including HIV patients with tuberculosis confirmed by culture, cared for at a referral center in Rio de Janeiro, Brazil. Multivariable Cox analysis was used to assess predictors of mortality within 3 months of antituberculous therapy.ResultsAmong 227 patients included, 90 (40%) started HAART before TB diagnosis. The median time to TB diagnosis after ARV initiation was 5.9 months (interquartile range [IQR] 3.0–8.9 months). Fourteen patients (6%) died within the first 3 months. Mortality was not different between patients previously started on HAART and those who were naïve to it. In the overall adjusted analysis, HAART use during TB treatment (hazard ratio [HR] = 0.21, 95% confidential interval [CI] = 0.06–0.72) and CD4 lymphocyte count >100 cells/mm3 (HR = 0.21, 95% CI = 0.04–0.99) were associated with lower mortality, while subjects with unknown baseline CD4 lymphocyte count (HR = 9.39, 95% CI = 2.56–34.5) had higher mortality. In subgroup analysis, among HAART naïve subjects, disseminated TB (HR = 5.32, 95% CI = 1.09–25.8) and unknown baseline CD4 lymphocyte count (HR = 13.2, 95% CI = 2.71–64.5) were associated with significantly higher mortality, while HAART (HR = 0.14, 95% CI = 0.03–0.69) predicted a better outcome. Among subjects previously started on HAART, mortality was significantly associated with duration of TB symptoms >120 days (HR = 6.15, 95% CI = 1.15–32.9).ConclusionsPredictors of early mortality among TB/HIV patients may vary according to the timing of HAART initiation. Among HAART naïve patients, mortality was influenced by baseline clinical severity, HAART use and, possibly, the quality of care preceding TB diagnosis. For patients with prior HAART initiation, longer delays in TB diagnosis predicted a significantly higher mortality.

Primary Thyroid Tuberculosis

Thyroid tuberculosis is a very rare entity and its diagnosis is based on the histopathological findings. In this report, we presented a 57 year-old women with palpable neck mass that finally diagnosed as thyroid tuberculosis. Ultrasonography showed bilaterally enlarged thyroid lobes with multiple hypoechogenic heterogenous solid nodules. Except elevated erythrocyte sedimentation rate, all of other laboratory test results including those of thyroid functions were within normal limits. Computed tomography of thorax revealed no abnormality. Intraoperatively purulent hard and irregular masses were seen. Due to suspicion of malignancy, bilateral total thyroidectomy was performed. Histopthological examination confirmed the diagnosis of tuberculosis. The patient received 6 months of antituberculous therapy and put on continuous thyroid hormone replacement.

Tuberculous arthritis revisited as a forgotten cause of monoarticular arthritis

BACKGROUND AND OBJECTIVES:Tuberculosis (TB) continues to be a major concern for health care workers. The number of reported cases of extrapulmonary tuberculosis, particularly osteoarticular tuberculosis, is increasing. This fact is attributed to different factors such as underestimating the disease and difficulty in diagnosis, which requires tissue sampling and can lead to a delay in the diagnosis, and can result in significant morbidity and mortality. The aim of this study was to highlight the difficulties and delay in diagnosis of articular tuberculosis, raising the need to create awareness about the importance of early diagnosis to avoid major complications of joint destruction.DESIGN AND SETTING:Retrospective review of patients presenting to a tertiary care center between 2003 and 2009.PATIENTS AND METHODS:We reviewed cases who presented with monoarticular joint pain and swelling that failed to respond to treatment elsewhere and were eventually diagnosed as having articular tuberculosis. We collected the demographic data, history, data on clinical examination and the relevant laboratory investigations, in addition to the data on radiological studies. All patients were treated medically with antituberculosis chemotherapy and surgically according to the severity of joint destruction.RESULTS:Thirteen patients had a mean age was 40 years (range, 17-70 years). The average delay in diagnosis was 2 years. Only 1 patient had pulmonary TB. The hip, knee and elbow were the most common joints involved. Bacteriology was positive in 69% of the cases; and histopathology, in 92%. Fifteen percent of the patients had arthrodesis. None showed recurrence after follow-up of 4 years.CONCLUSION:A high level of clinical suspicion is essential for early diagnosis and treatment of osteoarticular TB to reduce its significant morbidity.

Dermal Epstein Barr Virus–Associated Leiomyosarcoma: Tocsin of Acquired Immunodeficiency Syndrome in Two Children

Although rare in childhood, a relatively high incidence of smooth muscle tumors are recognized in patients with AIDS, mainly in association with Epstein Barr virus (EBV) infection. Although EBV-associated smooth muscle tumors have been documented rarely in the subcutis of AIDS patients, dermal involvement has not been described to date. This report describes dermal EBV-associated leiomyosarcomas (EBV-LMS) with a nodular but superficial plaque-like appearance on the lower limbs of 2 males, 9 and 12 years old. Histopathological assessment of the excised lesions demonstrated hypercellular mitotically active dermal tumors with hyperchromatic spindle and round cells, arranged in short fascicles and sheets, with microfoci of necrosis. A smooth muscle immunophenotype, including prominent desmin immunopositivity, and positive EBV-encoded RNA in situ hybridization investigation confirmed a diagnosis of EBV-LMS. Subsequent HIV seropositivity and AIDS were confirmed in both patients. Both patients also had pulmonary tuberculosis and received antituberculous therapy. Patient 1 had a 3 cm re-excision of the prior tumor site. He received highly active antiretroviral therapy, completed 6 months of antituberculous therapy, achieved immune reconstitution and viral suppression and is tumor-free 2 years after tumor excision. Patient 2 died before further therapy. The immune status, presence, and appropriate therapy of co-existent systemic infection and highly active antiretroviral therapy in AIDS patients with EBV-LMS are crucial to a favorable outcome.