Hypereosinophilic Syndrome: Hacettepe Experience

Tavil, Betül; Aytaç, Selin; Ünal, Şule; Kuşkonmaz, Barış; Gümrük, Fatma; Çetin, Mualla

doi:10.1097/mph.0000000000000668

Cited by 6 publications

(7 citation statements)

References 18 publications

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“…After removing duplicate and redundant articles, 25 papers were included in the present study for the final review and analysis. A thorough reading of these articles yielded a total of 32 cases of idiopathic HES with cutaneous involvement 5–29. A comprehensive review of these case reports indicated a slight male predominance (male, n = 19; female, n = 13).…”

Section: Discussionmentioning

confidence: 99%

Idiopathic hypereosinophilic syndrome with cutaneous involvement: a comparative review of 32 cases

Inayat¹,

O’Neill

Zafar

et al. 2018

BMJ Case Rep

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Although idiopathic hypereosinophilic syndrome (HES) is uncommon, we studied the clinical characteristics of this disorder in patients with cutaneous involvement. We chronicle the case of a patient with diffuse skin rash due to idiopathic HES from our clinical experience. Furthermore, a systematic literature search of the medical databases PubMed and Google Scholar was conducted. A total of 32 cases fulfilled the inclusion criteria. The data on patients’ characteristics, epidemiology, clinical features, diagnosis, treatment and outcome were collected and analysed. This review illustrates that physicians should maintain a high index of clinical suspicion for idiopathic HES in patients presenting with dermatological lesions and hypereosinophilia, without an obvious cause. Randomised clinical trials are warranted to outline a generalised and efficient therapeutic approach in these patients. Additionally, this paper highlights the need for population-based studies to delineate the magnitude and scope of this association.

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Section: Discussionmentioning

confidence: 99%

Idiopathic hypereosinophilic syndrome with cutaneous involvement: a comparative review of 32 cases

Inayat¹,

O’Neill

Zafar

et al. 2018

BMJ Case Rep

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show abstract

Section: Discussionmentioning

confidence: 99%

“…Idiopathic HES is rare in children and a limited number of cases have been reported ( 55 – 57 ). The true incidence is unknown and data on the efficacy of treatment regarding the response and survival of patients is also insufficient ( 55 ). Pediatric HES has been reported to have a slight male prevalence ( 56 ), though a recent study by Williams et al ( 57 ) observed that the female/male occurrence ratio was increased to 23/9 ( 57 ).…”

Section: Discussionmentioning

confidence: 99%

“…Presentation of HES is variable, though fever, arthralgias and rash were common presentation symptoms ( 57 ). In pediatric idiopathic HES, pulmonary and cardiac involvement may be the most common clinical manifestations ( 55 ). However, in the current patient, there was no specific clinical presentation except painless and enlarged lymphoma.…”

Section: Discussionmentioning

confidence: 99%

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Concurrent eosinophilia and IgG4‑related disease in a child: A case report and review of the literature

Chen

Huang

et al. 2018

Exp Ther Med

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The current study presents the case of a 9-year-old Chinese boy who presented with eosinophilia and elevated serum levels of immunoglobulin G4 (IgG4). A bone marrow puncture identified an elevated eosinophil rate of 23% (normal range, <5%), which indicated eosinophilia. However, gene analysis, fluorescent in situ hybridization and other examinations, including bone marrow aspiration, blood routine, auto-antibody tests and parasitic and allergens screening, contradicted a diagnosis of secondary or clonal eosinophilia. Furthermore, the patient exhibited multiple lymph node swelling and a lymph biopsy strongly indicted a pathological diagnosis of IgG4-related disease (IgG4-RD). His peripheral blood flow cytometry confirmed an elevated count of plasmablasts, which is specific to IgG4-RD. The patient responded well to therapy with prednisone and remained healthy in all follow-ups. By taking all these factors into consideration, the boy was diagnosed with IgG4-RD. It is difficult to distinguish IgG4-RD from hypereosinophilic syndrome and the potential association between the two remains unclear. However, the present case study serves as a reminder that IgG4-RD may occur in children and medical professionals should not neglect this possibility.

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“… 8 Pulmonary involvements during childhood vary from 27.5% to 55.3%. 7 , 9 In the study conducted by Tavil et al 2 half of the pediatric patients had pulmonary involvement presented as cyanosis, crackles on auscultation, and thorax CT findings. 2 In the presented case, the patient had only dry cough and respiratory distress.…”

Section: Discussionmentioning

confidence: 98%

The First Definition of Pulmonary Component of Hypereosinophilic Syndrome: Bronchial Casts

Onay¹,

Ayhan²,

Oksay³

et al. 2023

Thorac Res Pract

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Hypereosinophilic syndrome is a heterogeneous disease characterized by eosinophilic tissue inflammation and eosinophilia. Pulmonary involvement could be seen in up to 55% among children with hypereosinophilic syndrome. A 3-year-old boy with chronic hypereosinophilia and respiratory complaints was diagnosed with idiopathic hypereosinophilic syndrome. Atelectasis was detected in the radiological evaluation, and bronchial casts with eosinophilic structures were removed by bronchoscopy. Steroid, inhaled hypertonic saline, inhaled bronchodilator, inhaled corticosteroid, and leukotriene receptor antagonist were used for 1 year in the management of hypereosinophilic syndrome, and related eosinophilic casts and repetitive bronchoscopies were administered for removal of the casts. The patient was successfully managed with an inhaled N-acetyl cysteine treatment. In children, the long-term prognosis of hypereosinophilic syndrome is uncertain. Comprehensive diagnostic tests are required for the early diagnosis and management of pediatric hypereosinophilic syndrome. In the presented case, the rare occurrence of pulmonary involvement of hypereosinophilic syndrome in a 3 year-old-boy with recurrent hypereosinophilic casts and its management were discussed.

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Hypereosinophilic Syndrome: Hacettepe Experience

Cited by 6 publications

References 18 publications

Idiopathic hypereosinophilic syndrome with cutaneous involvement: a comparative review of 32 cases

Idiopathic hypereosinophilic syndrome with cutaneous involvement: a comparative review of 32 cases

Concurrent eosinophilia and IgG4‑related disease in a child: A case report and review of the literature

The First Definition of Pulmonary Component of Hypereosinophilic Syndrome: Bronchial Casts

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