Allergic bronchopulmonary aspergillosis with marked peripheral blood eosinophilia and pulmonary eosinophilia:A case report

BACKGROUND Allergic bronchopulmonary aspergillosis(ABPA)is an immune-related pulmonary disease caused by sensitization of airway by Aspergillus fumigatus.The disease manifests as bronchial asthma and recurring pulmonary shadows,which may be associated with bronchiectasis.The diagnosis of ABPA mainly depends on serological,immunological,and imaging findings.Pathological examination is not necessary but may be required in atypical cases to exclude pulmonary tuberculosis,tumor,and other diseases through lung biopsy.CASE SUMMARY An 18-year-old man presented with recurrent wheezing,cough,and peripheral blood eosinophilia.Chest computed tomography showed pulmonary infiltration.There was a significant increase in eosinophils in bronchoalveolar lavage fluid.There was no history of residing in a parasite-endemic area or any evidence of parasitic infection.Pathologic examination of bronchoalveolar lavage fluid excluded fungal and mycobacterial infections.The patient was receiving medication for comorbid diseases,but there was no temporal correlation between medication use and clinical manifestations,which excluded drug-induced etiology.Histopathological examination of lung biopsy specimen showed no signs of eosinophilic granulomatosis with polyangiitis,IgG4-related diseases,or tumors.The diagnosis of ABPA was considered based on the history of asthma and the significant increase in serum Aspergillus fumigatus-specific immunoglobulin(Ig)E.Eosinophil-related diseases were excluded through pathological biopsy,which showed typical pathological manifestations of ABPA.CONCLUSION The possibility of ABPA should be considered in patients with poorly controlled asthma,especially those with eosinophilia,lung infiltration shadows,or bronchiectasis.Screening for serum IgE,Aspergillus fumigatus-specific IgE and IgG,and alveolar lavage can help avoid misdiagnosis.

Chest radiographic and computed tomographic manifestations in allergic bronchopulmonary aspergillosis

AIM: To investigate the chest radiographic and high resolution computed tomography (HRCT) chest manifestations in glucocorticoid-na?ve allergic bronchopulmonary aspergillosis (ABPA) patients. METHODS: This is a prospective observational study and includes 60 consecutive glucocorticoid-na?ve patients with ABPA who underwent chest radiography and HRCT of the chest (1.25 mm every 10 mm) in the routine diagnostic workup for ABPA. RESULTS: Chest radiographs were normal in 50% of cases. Of the remainder, most patients demonstrated permanent findings in the form of parallel line and ring shadows suggesting bronchiectasis. Consolidation was detected in 17 cases but in the majority, the corresponding HRCT chest scan showed mucus-filled bronchiectatic cavities. Chest HRCT was normal in 22 patients, while central bronchiectasis (CB) was demonstrated in the remaining 38 patients. Bronchiectasis extended to the periphery in 33%-43% depending on the criteria used for defining CB. The other findings observed on HRCT were mucoid impaction, centrilobular nodules and high-attenuation mucus in decreasing order of frequency. CONCLUSION: Patients with ABPA can present with normal HRCT chest scans. Central bronchiectasis cannot be considered a characteristic feature of ABPA as peripheral bronchiectasis is commonly observed. Consolidation is an uncommon finding in ABPA.

Value of eosinophil count in bronchoalveolar lavage fluid for diagnosis of allergic bronchopulmonary aspergillosis: A case report

BACKGROUND An immediate hypersensitive immune response to Aspergillus fumigatus antigens is one of the main characteristic features of allergic bronchopulmonary aspergillosis(ABPA).As ABPA is an allergic respiratory disease,immunoglobulin E and peripheral-blood eosinophilia have been used as diagnostic indicators.However,eosinophilia in bronchoalveolar lavage fluid(BALF)has not been considered in the diagnostic criteria for ABPA.CASE SUMMARY We present a case of ABPA in which the eosinophil count in peripheral blood was not increased,whereas the eosinophil percentage in BALF reached 60%.After antifungal and hormone therapy,imaging revealed very good resolution of lung infiltration.CONCLUSION The value of the eosinophil count in BALF for the diagnosis of ABPA is worthy of the clinician's attention,especially when the patient’s clinical features lack specificity and the diagnostic parameters are negative.

Complete withdrawal of glucocorticoids after dupilumab therapy in allergic bronchopulmonary aspergillosis:A case report

BACKGROUND Allergic bronchopulmonary aspergillosis(ABPA)is an allergic reaction to Aspergillus species that aggravates bronchial asthma.Previous studies demonstrated the glucocorticoid-sparing effect of dupilumab in patients with ABPA.There is no report of complete withdrawal of glucocorticoids after dupilumab.CASE SUMMARY The patient was a 54-year-old woman with bronchial asthma treated with inhaled corticosteroids and a long-acting beta-2 agonist.She consulted our institution for productive cough and fever in March 2017.Chest computed tomography scan revealed mucoid impaction,and the bronchial lavage fluid culture was positive for Aspergillus fumigatus.The diagnosis was ABPA.The patient was treated with oral glucocorticoids from April 2017 to November 2017.In January 2019,she had bronchial asthma exacerbation,and a chest computed tomography scan showed recurrent mucoid impaction.She was treated with oral glucocorticoids and itraconazole.In February 2020,during tapering of oral glucocorticoid,she had the third episode of bronchial asthma exacerbation and a mucoid impaction.The patient was treated with dupilumab in addition to oral glucocorticoid and itraconazole.The clinical response improved,and oral glucocorticoid was discontinued in June 2020.CONCLUSION This is the first case of ABPA in which complete withdrawal of glucocorticoid was possible after treatment with dupilumab.

Heterozygous CARD9 mutation favors the development of allergic bronchopulmonary aspergillosis

Background:Previous research demonstrated that a homozygous mutation of g.136372044G>A(S12N)in caspase recruitment domain family member 9(CARD9)is critical for producing Aspergillus fumigatus-induced(Af-induced)T helper 2(T_(H)2)-mediated responses in allergic bronchopulmonary aspergillosis(ABPA).However,it remains unclear whether the CARD9^(S12N)mutation,especially the heterozygous occurrence,predisposes the host to ABPA.Methods:A total of 61 ABPA patients and 264 controls(including 156 healthy controls and 108 asthma patients)were recruited for sequencing the CARD9 locus to clarify whether patients with this heterozygous single-nucleotide polymorphisms are predisposed to the development of ABPA.A series of in vivo and in vitro experiments,such as quantitative real-time polymerase chain reaction,flow cytometry,and RNA isolation and quantification,were used to illuminate the involved mechanism of the disease.Results:The presence of the p.S12N mutation was associated with a significant risk of ABPA in ABPA patients when compared with healthy controls and asthma patients,regardless of Aspergillus sensitivity.Relative to healthy controls without relevant allergies,the mutation of p.S12N was associated with a significant risk of ABPA(OR:2.69 and 4.17 for GA and AA genotypes,P=0.003 and 0.029,respectively).Compared with patients with asthma,ABPA patients had a significantly higher heterozygous mutation(GA genotype),indicating that p.S12N might be a significant ABPA-susceptibility locus(aspergillus sensitized asthma:OR:3.02,P=0.009;aspergillus unsensitized asthma:OR:2.94,P=0.005).The mutant allele was preferentially expressed in ABPA patients with heterozygous CARD9^(S12N),which contributes to its functional alterations to facilitate Af-induced T_(H)2-mediated ABPA development.In terms of mechanism,Card9 wild-type(Card9^(WT))expression levels decreased significantly due to Af-induced decay of its messenger RNA compared to the heterozygous Card9 S12N.In addition,ABPA patients with heterozygous CARD9^(S12N)had increased Af-induced interleukin-5 production.Conclusion:Our study provides the genetic evidence showing that the heterozygous mutation of CARD9^(S12N),followed by allele expression imbalance of CARD9^(S12N),facilitates the development of ABPA.

Aspirin induced asthma accompanied with allergic bronchopulmonary aspergillosis： a case report

In this paper, we describe a patient with a rather severe form of aspirin-induced asthma （AIA） and allergic bronchopulmonary aspergillosis （ABPA）. The patient is a man born in 1948, who first presented with rhinorrhea, nasal congestion and chronic urticaria, and had an episode of asthma after ingestion of non-steroidal anti-inflammatory drugs （NSAIDs） for the further eight years. He was diagnosed as AIA and the symptoms decreased in frequency when avoiding NSAIDs. At the age of 45, he presented with unusual symptoms, such as malaise, fever, and coughing. The diagnosis of ABPA was made based on his clinical features, such as the symptoms of asthma, typical pulmonary imaging, positive skin test, and high levels of the total IgE （T-IgE） and sIgE. Itraconazole accompanied with oral prednisone prevented pulmonary disease from developing.

Allergic bronchopulmonary aspergillosis coexists with hereditary bisalbuminemia

The electrophoresis of a patient＇s serum proteins occasionally reveals two distinct fractions of serum albumin.This rare phenomenon is called bisalbuminemia.Bisalbuminemia can be classified into two types,a rare hereditary one and non-hereditary bisalbuminemia.There have been many reports about non-hereditary bisalbuminemia,which is mainly caused by the excess-use of antibiotics such as penicillin.Hereditary bisalbuminemia （or alloalbuminemia） is a rare,autosomal,and incomplete dominant hereditary condition characterized by the presence of two distinct fractions of serum albumin by electrophoresis.Scheurlen1 reported the first case of this disease in 1955.The first case of hereditary bisalbuminemia in China was reported by Ying et al2 in 1980.Since then,approximately 130 cases of bisalbuminemia from 21 different families have been reported in China.Here,we described a new family with hereditary bisalbuminemia,which was discovered with a patient complained of refractory asthma.Keywords：hereditary bisalbuminemia; allergic bronchopulmonary aspergillosis

Bronchoscopy and Pathology Findings in an Unusual Case of Allergic Bronchopulmonary Mycosis

The diagnosis of allergic bronchopulmonary mycosis(ABPM)is usually made on clinical,serologic,and roentgenographic findings.However,the indicators in such diagnostic criteria still lack specificity,which results in a high rate of misdiagnosis.A 47-year-old woman was diagnosed with“tuberculosis”or“pneumonia”for 10years and after thoracoscopic right upper lung resection the following chest CT showed right lung atelectasis.Brown sputum was seen in the right bronchus via bronchoscopy.Reviewing the surgical pathology specimen,the bronchi were found to be generally cystic dilated,and a large amount of mucus was retained and formed a mucus plug.The final diagnosis of the case was ABPM.The current diagnostic criteria of ABPM do not emphasize the role of bronchoscopy and pathological examination,but bronchoscopy and pathological examination still have important value for some atypical cases with suspected ABPM.

Relationship between chronic rhinosinusitis and lower airway diseases: An extensive review

Signifcant links between allergic rhinitis and asthma have been reported, and the united airway disease hypothesis is supported by numerous findings in epidemiologic, physiologic, pathologic, and immunologic studies. The impact of allergic rhinitis on asthma has been esta-blished. On the other hand, the relationship between chronic rhinosinusitis and lung diseases has been under investigation. Chronic rhinosinusitis is a common disease, and the high prevalence of chronic rhinosinusitis in some kinds of lung diseases has been reported. Recent studies suggest that the treatment of chronic rhinosinusitis has beneficial effects in the management of asthma. Here, we present an overview of the current research on the relationship between chronic rhinosinusitis and lower airway diseases including asthma, chronic obstructive pul-monary disease, cystic fibrosis, diffuse panbronchiolitis, primary ciliary dyskinesia, idiopathic bronchiectasis, and allergic bronchopulmonary aspergillosis.