Candida albicans meningitis in a premature neonate successfully treated with 5-fluorocytosine and amphotericin B: a case report and review of the literature.

Candida albicans meningitis was diagnosed in a 45-day-old premature infant whose birth weight was 1,616 gm. Symptoms consisted of poor weight gain and poor suckling. The combined use of amphotericin B and 5-fluorocytosine (5-FC) resulted in negative CSF cultures after 12 days of therapy. Amphotericin B was given for 45 days (total 83 mg) and 5-FC for 60 days (total 19 mg). Only one other premature infant has been reported in the literature who had similar treatment. A review of Candida meningitis diagnosed before death in 11 other infants less than 1 year of age is presented.     

Shaken baby syndrome masquerading as apparent life threatening event

Disseminated cryptococcosis is a rare and often fatal disease in children. The majority of cases usually occur in individuals with defective cell-mediated immunity, most commonly due to HIV infection. The authors here in report an 8-year-old girl from Nepal who presented with fever, cough, headache, lymphadenopathy, hepatosplenomegaly and cutaneous lesions. Lymph node biopsy revealed multiple granulomas composed of histiocytes and epitheliold cells along with numerous yeast forms of cryptococcus. Cultures of CSF, sputum and urine yielded cryptococcus neoformans. Surprisingly,the immune function in terms of T-cell number, CD4 : CD8 ratio, serum immunoglobulins and HIV serology was normal. After the diagnosis of disseminated cryptococcosis was established, the patient was treated with 5-fluorocytosine (100 mg/kg/day) for initial two weeks and amphotericin B (1 mg/kg/day) for 13 weeks. Patient responded well to the treatment with disappearance of presenting symptoms, cutaneous lesions, and lymphadenopathy, though she still had hepatosplenomegaly, which also decreased. Unfortunately, she developed loss of vision in 10th week of therapy. The patient was discharged on oral fluconazole (6 mg/kg/day) and no recurrence was found during the follow-up period of more than 9 months. This is the first case of disseminated cryptococcosis with no detectable immune deficit, from India.     

Lethal double infection with Acremonium strictum and Aspergillus fumigatus during induction chemotherapy in a child with ALL

Fungal infections are a major cause of morbidity and mortality in patients during chemotherapeutic treatments and malignant hematologic disease. We present a case of a double fungal infection with disseminated Acremonium strictum (A. strictum) and pulmonary Aspergillus fumigatus (A. fumigatus) and its rapid clinical course. A 17-year-old boy with prolonged neutropenia developed a disseminated fungal infection during induction chemotherapy of his acute lymphoblastic leukemia. The infection was rapidly lethal despite neutrophil recovery and early antifungal combination therapy with amphotericin B and caspofungin. Since there are only a few reports about invasive Acremonium infections, we present this case with regard to differences in the clinic pathologic features of Aspergillosis and other opportunistic fungal infections due to Fusarium or Acremonium species.     

Acquired immunodeficiency syndrome (AIDS) with lymphocytic interstitial pneumonitis (LIP) in a multi transfused child with thalassemia major.

The case of a 10.5-year-old girl, who was diagnosed with a case of thalassemia major at the age of 8 months and had been on regular blood transfusions since then, is related. Donor screening for HIV was started in mid-1988, thus she had received unscreened blood for a number of years. In February 1991, she presented with a dry persistent cough, moderate grade continuous fever, and breathlessness on exertion for over 2 weeks. Chest X-ray showed bilateral infiltrations. She was put on penicillin and chloramphenicol with a provisional diagnosis of bronchopneumonia. In March 1991, she had to be hospitalized for impending respiratory failure. After treatment with intravenous fluids and parenteral antimicrobials, her condition stabilized and she was discharged. In April 1991, she was readmitted because of complaints of difficulty in swallowing and weight loss. Her chest signs had persisted and she had developed oropharyngeal candidiasis with ulcerations. She also had alopecia, a generalized lymphadenopathy, digital clubbing, and bilateral parotid enlargement. Candidiasis responded to vigorous therapy with clotrimazole. Fine needle aspiration of lymph node showed a reactive hyperplasia. HIV antibodies were detected in the serum with ELISA and confirmed by Western blot. Immunologic tests showed evidence of severe immunodeficiency. The Multitest CMI, which simultaneously tests delayed skin hypersensitivity to seven common recall antigens, was totally nonreactive. She was classified as having AIDS according to World Health Organization criteria for children under 13 years of age. The diagnosis of lymphocytic interstitial pneumonitis (LIP) was also made based on the symptoms. Oral prednisolone was given 2 mg/kg/day in 3 divided doses for a month. The cough and dyspnea showed great improvement and the parotid swellings disappeared; lymphadenopathy, clubbing, and alopecia, however, persisted. The child was kept on maintenance therapy of prednisolone and on alternate day co-trimoxazole for prophylaxis against Pneumocystis carinii infection.     

儿童白血病化疗后合并全身性真菌感染诊断及治疗体会

目的 积累和交流白血病化疗病人合并深部真菌感染时临床经验.方法 总结2年中明确诊断深部霉菌感染病人的诊断过程、方法、临床特征、治疗反应和转归.结果确诊深部真菌感染者共5例,均为白血病患儿,感染前或感染中白血病达完全缓解,特征性表现为发热,抗菌素治疗＞5天无效,轻中度压痛的多发性硬性皮下小结（2/5例）和肝脾肾散布性低密度病灶（3/5例）,需四周以上二性霉素治疗,有明显但可以处理的毒性反应.结论 发热、抗菌素治疗无效、硬性皮下小结和肝脾肾散布性低密度病灶时应考虑深部真菌感染,并给予4周以上二性霉素治疗.     

儿童白血病化疗后合并全身性真菌感染诊断及治疗体会

目的积累和交流白血病化疗病人合并深部真菌感染时临床经验。方法总结2年中明确诊断深部霉菌感染病人的诊断过程、方法、临床特征、治疗反应和转归。结果确诊深部真菌感染者共5例,均为白血病患儿,感染前或感染中白血病达完全缓解,特征性表现为发热,抗菌素治疗>5天无效,轻中度压痛的多发性硬性皮下小结(2/5例)和肝脾肾散布性低密度病灶(3/5例),需四周以上二性霉素治疗,有明显但可以处理的毒性反应。结论发热、抗菌素治疗无效、硬性皮下小结和肝脾肾散布性低密度病灶时应考虑深部真菌感染,并给予4周以上二性霉素治疗。     

Candida (amphotericin-sensitive) lens abscess associated with decreasing arterial blood flow in a very low birth weight preterm infant

In this report, we review the case of a candidal lens abscess in a premature infant girl who was 28 weeks' gestational age at birth. The culture obtained from the lens abscess grew Candida albicans sensitive to amphotericin B but resistant to flucytosine. This case is unique in that the infant developed a fungal lens cataract at 34 weeks' postconceptional age during the last week of a 30-day course of amphotericin B. The embryonic hyaloid artery system, which perfuses the developing lens, regresses between 29 and 32 weeks of gestation; thus, the mechanism for an infection of the lens may be inoculation of the lens by Candida before hyaloid artery system regression, followed by developmental loss of this blood supply, which makes the lens inaccessible to antimicrobial penetration. Candidal endophthalmitis with lens abscess is an uncommon morbidity that requires prompt recognition and surgical intervention for effective management.     

Hepatitis B or non-A,non-B virus infection in multitransfused thalassaemic patients.

We undertook a four year study of 128 thalassaemic patients who had undergone several transfusions, to determine the incidence of hepatitis B virus markers and the activities of transaminases in their sera each month. The results showed that the possibility of these patients contracting hepatitis B virus infection is still high, although on only one occasion was a transient antigenaemia found, indicating low viral replication. Furthermore, the probability of contact with hepatitis B virus increases with the number of transfusions and, therefore, with age. About 25% of these patients were positive for hepatitis B markers and 80% for other hepatitis markers including the case of cytomegalovirus hepatitis.     

Fibrosing mediastinitis secondary to zygomycosis in a twenty-two-month-old child

We report the case of a 22-month-old immunocompetent male child with fibrosing mediastinitis secondary to zygomycosis, an unusual presentation of a rare fungal infection. This patient was successfully treated with amphotericin B and itraconazole for 20 weeks. Stenting of the superior vena cava was helpful in relieving the patient's superior vena cava syndrome.     

Vincristine therapy for severe platelet alloimmunization.

A 19-month-old girl with idiopathic severe aplastic anemia refractory to multi-agent immunosuppressive therapy developed severe platelet alloimmunization following several months of platelet transfusions. She became refractory to human leukocyte antigen (HLA)-matched platelet transfusions and experienced frequent episodes of bleeding. She was treated with intravenous vincristine administered weekly for three doses and showed marked improvement in both clinical and laboratory response to platelet transfusions. When vincristine was held for 3 weeks, she again became refractory to HLA-matched platelet transfusion. Reinstitution of vincristine resulted in cessation of clinical bleeding and improved response to platelet transfusion. The mechanism of response likely involves selective delivery of cytotoxic drug to macrophages. To our knowledge this is the first reported case of alloimmune thrombocytopenia responsive to vincristine.     

Disseminated protothecosis

Here is presented, a rare case of disseminated protothecosis in a 10-year-old boy with combined immunodeficiency, hitherto unreported from India. Even though it is difficult to diagnose clinically, observation of the sporangiospores within the sporangium in culture gives the accurate laboratory identification of Prototheca spp. In this patient, failure to eradicate the infection with amphotericin B and recurrence with olecranon bursitis along with skin lesions and splenomegaly was observed. Disseminated protothecosis in a child with combined immunodeficiency and failure to eradicate the infection with amphotericin B is reported.     

实体瘤患儿行自体外周血干细胞移植治疗中真菌感染情况

目的通过对31例实体瘤患儿行自体外周血干细胞移植(auto-PBSCT)治疗中真菌感染的情况进行分析,总结其临床特点、诊断与治疗经验。方法回顾性分析2006年5月-2009年12月本院儿科收治的31例实体瘤患儿行auto-PBSCT治疗过程中防治真菌感染的过程。结果 1.Ⅳ期进展期神经母细胞瘤患儿2例行自体外周血干细胞移植过程中经微生物学检查明确并肺部真菌感染,其中1例并脑、肝真菌感染;2例患儿血培养均为近平滑假丝酵母菌;4例患儿行肺部CT检查,表现为密度增高、渗出炎症阴影;1例进展期神经母细胞瘤患儿明确真菌感染后应用氟康唑、两性霉素B脂质体、伏立康唑静脉滴注抗真菌治疗有效,体温正常,度过骨髓抑制期,原发病获得部分缓解;1例进展期神经母细胞瘤患儿因存在颅内、骨骼、脊髓、肺、肝多发转移肿瘤,于自体外周血造血干细胞移植9 d后骨髓抑制期死亡。2.部分缓解期Ⅳ期神经母细胞瘤1例及进展期肝母细胞瘤1例患儿表现为发热、咳嗽、抽搐,怀疑侵袭性真菌感染,给予氟康唑、两性霉素B、伏立康唑治疗后好转度过骨髓抑制期,原发病获得部分缓解。3.余27例实体瘤患儿auto-PBSCT治疗中应用氟康唑预防真菌感染,临床未发生侵袭性真菌感染,1例进展期Ⅳ期神经母细胞瘤患儿因有多脏器转移,且有原发心脏损害,大剂量化疗后骨髓抑制期免疫耐受差,导致多脏器衰竭死亡。余26例实体瘤患儿顺利度过骨髓抑制期,病情获得缓解。结论实体瘤患儿auto-PBSCT治疗中易并真菌感染,需结合病史、血培养、G试验及CT、MRI等影像学检查做出诊断,经验性应用氟康唑、伏立康唑、两性霉素B等可防治真菌感染,原发病严重未缓解者预后差。     

Mucormycosis and diabetes in acute leukemia

The authors report the case of 5 1/2 year-old boy with insulin-dependent diabetes mellitus revealed during the induction therapy of an acute leukemia of the mixed type, and who presented with an unusual type of pulmonary fungal infection: mucormycosis. It had a favourable outcome with surgical excision preceded and followed by amphotericin B treatment.     

Penicillium marneffei infection in a non-HIV infected child

We report a case of Penicillium marneffei infection in a previously healthy 3.5 years-old Chinese boy and review the literature. The details of the case are described with emphasis on his immune function and treatment outcome. This boy had transient immunodeficiency involving phagocytic and NK cells due to P. marneffei infection which resolved after treatment with gamma interferon and amphotericin B. A prolonged course of fluconazole of 1 year was successful in preventing relapse. Gamma interferon, amphotericin B and a prolonged course of fluconazole may be useful in the treatment of life-threatening infection by P. marneffei.     

Three distinct episodes of GBS septicemia in a healthy newborn during the first month of life

In neonates, recurrent group B streptococcal infection is uncommon, and two relapses are extremely rare. We report a case of three distinct episodes of Streptococcus agalactiae bacteremia in a healthy full-term infant over a 4-week period. The newborn had no apparent predisposing condition or infectious focus, and the recurrences occurred after adequate antimicrobial treatment, the second occurring after withdrawal of colonized breast milk. Finally, oral rifampin was combined to his antibiotic regimen, his colonization cultures turned negative, and no further relapses have occurred during the 11-month follow-up. Conclusion In this report, we describe a newborn with two exceptionally rapid recurrences of group B streptococcal septicemias, and who was successfully treated with combining rifampin to the antibiotic regimen.     

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目的探讨血液病患儿侵袭性真菌感染联合药物治疗组合及疗效。方法 2006年1月至2011年4月在中山大学孙逸仙纪念医院就诊的血液病合并侵袭性真菌感染并予以联合治疗患儿39例,对患儿的临床资料及治疗效果进行回顾性分析。结果联合抗真菌药物治疗者共48例次,其中拟诊11例次,临床诊断32例次,确诊5例次。两性霉素B联合卡泊芬净18次,无效3次;两性霉素B联合伊曲康唑静脉13次,口服4次,均有效;两性霉素B联合伏立康唑9次,无效1次;卡泊芬净联合伊曲康唑6次,无效1次;卡泊芬净联合伏立康唑5次,均有效;伊曲康唑联合制霉菌素1次,有效。完全缓解27例,部分缓解7例,放弃1例,恶化死亡4例。三唑类联合两性霉素B或卡泊芬净与两性霉素B联合卡泊芬净相比,效果差异无统计学意义(P>0.05)。结论 免疫功能明显抑制、既往侵袭性真菌感染且再次接受强化疗或造血干细胞移植、严重或突破性真菌感染、单药治疗失败或毒副反应大、合并移植物抗宿主病患儿的联合治疗疗效较满意。三唑类联合两性霉素B或卡泊芬净与两性霉素B联合卡泊芬净均可作为联合用药选择。     

Rhinocerebral mucormycosis in a child with acute lymphoblastic leukemia: a case report

BACKGROUND: Mucormycosis is a life-threatening fungal infection in patients with predisposing conditions. CASE REPORT: We report the case of a 7-year-old girl with acute lymphoblastic leukemia and rhinocerebral mucormycosis who successfully completed chemotherapy. The infection with Rhizopus genus was mycologically identified from nasal secretions and therapy was based on amphotericin B with surgical debridement. Chemotherapy was safely continued after resolution of the mycosis and the patient remains in continuous complete remission at 30 months. CONCLUSIONS: Rhinocerebral mucormycosis in acute lymphoblastic leukemia is manageable with sequential therapy including, amphotericin B, surgery, and risk-adapted chemotherapy.     

The therapeutic effect of amphotericin in acrodermatitis enteropathica: Hypothesis and implications

A nine year-old girl with acrodermatitis enteropathica developed typical clinical and biochemical features of zinc deficiency on two occasions while on an oral zinc supplement. On both occasions, these features responded immediately when she was treated with amphotericin B lozenges. Studies in vitro showed that amphotericin increases the permeability to zinc of pure lipid membranes containing cholesterol. We suggest that the antibiotic enhanced zinc absorption from the oral supplement thereby effecting resolution of the patient's zinc deficiency.     

Use of amphotericin B colloidal dispersion in children

PURPOSE: To describe the experience with a new lipid-based amphotericin product (amphotericin B colloidal dispersion or ABCD) in children with fever and neutropenia who are at high risk for fungal infection. PATIENTS AND METHODS: Forty-nine children with febrile neutropenia were treated in a prospective, randomized trial comparing ABCD with amphotericin B. An additional 70 children with presumed or proven fungal infection were treated with 5 different open-label studies of ABCD. Patients were registered into these studies for reasons of: 1) failure to respond to amphotericin B; 2) development of nephrotoxicity or preexisting renal impairment; or 3) willingness to participate in a dose-escalation study. Extensive data detailing response and toxicity were collected from each patient. RESULTS: In the randomized trial, there was significantly less renal toxicity in the children receiving ABCD than in those receiving amphotericin B (12.0% vs. 52.4% [P = 0.003]). Other adverse symptoms were not significantly different. In the additional open-label studies, although 80% of patients receiving ABCD reported some adverse symptom, the majority of these were infusion related, and nephrotoxicity was reported in only 12% of these patients. CONCLUSIONS: ABCD was well-tolerated at doses up to 5 times greater then those usually tolerated with amphotericin B. Renal toxicity was markedly less than expected, and there were no other unexpected severe toxicities. Further randomized studies are needed to further define the role of this and other liposomal products in children.     

Mycetoma of the renal pelvis caused by Fusarium species.

A preterm infant, with posterior urethral valves had a mycetoma of the renal pelvis caused by Fusarium species. Prolonged treatment with amphotericin B alone or with flucytosine failed. Combined surgical drainage and medical therapy resulted in full resolution.