Clonal Hypereosinophilic Syndrome: Two Cases Report in Black Men  from Sub-Saharan Africa and Literature Reviews

Messie, Kodjovi; Vovor, A.; Kueviakoe, Irénée Messanh Delagnon; Sallah, Levi kankoe; Agbetiafa, Kossi; Segbena, A.

doi:10.5402/2011/974609

Cited by 4 publications

(2 citation statements)

References 22 publications

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“…Furthermore, chronic cough as the first and most prominent manifestation of F/P+ CEL has been previously reported in 4 patients (table 1) [7,8,9,10]. Thus, despite findings arguing against F/P+ CEL in our patient (absence of organomegaly or elevated serum vitamin B 12 or tryptase; normal blood count besides HE; negative FISH and single-step RT-PCR for the F/P rearrangement), we persevered and eventually detected the F/P rearrangement by nested RT-PCR.…”

Section: Discussionmentioning

confidence: 99%

Severe Prolonged Cough as Presenting Manifestation of FIP1L1-PDGFRA+ Chronic Eosinophilic Leukaemia: A Widely Ignored Association

Roufosse

Heimann²,

Lambert

et al. 2016

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Chronic eosinophilic leukaemia associated with the FIP1L1-PDGFRA fusion gene (F/P+ CEL) is a rare cause of marked persistent hypereosinophilia, arising almost exclusively in male patients. Clinical presentations are heterogeneous with a higher incidence of eosinophil-mediated cardiomyopathy than in other hypereosinophilic syndrome variants. Features of chronic myeloproliferative disease are often present, including splenomegaly and elevated serum vitamin B₁₂ levels. The diagnosis is made by fluorescence in situ hybridization (FISH) showing the deletion of the CHIC2 locus and/or RT-PCR showing the FIP1L1-PDGFRA fusion transcript. Treatment with imatinib mesylate, a tyrosine kinase inhibitor, results in rapid and complete resolution of hypereosinophilia and associated symptoms, except for those related to sub-endocardial fibrosis that may be irreversible. We report the case of a male patient in whom isolated intractable cough remained the only clinical manifestation of F/P+ CEL for 4 years. Furthermore, eosinophil autofluorescence, an as yet unreported artefact in this setting, precluded the detection of the CHIC2 deletion and further delayed diagnosis, underlining that both FISH and RT-PCR should be performed when this disease is suspected.

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Section: Discussionmentioning

confidence: 99%

Severe Prolonged Cough as Presenting Manifestation of FIP1L1-PDGFRA+ Chronic Eosinophilic Leukaemia: A Widely Ignored Association

Roufosse

Heimann²,

Lambert

et al. 2016

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“…We found 16 HES cases [7,[16][17][18][19][20][21][22][23][24][25][26][27][28][29][30] with cough as the main or only symptom (Case 3-18, Table 2). The overall clinical information is summarized in Table 2.…”

Section: Literature Search Strategy and Termsmentioning

confidence: 99%

Cough in hypereosinophilic syndrome: case report and literature review

Xie

Zhang

et al. 2020

BMC Pulm Med

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Background: Cough and airway eosinophilic inflammation has not been highlighted in hypereosinophilic syndrome (HES). Case presentation: We report 2 further cases and reviewed the clinical features and treatment of HES present with cough from the literature. Both cases were middle age male, presenting with chronic cough, airway eosinophilic inflammation and hyper eosinophilia who have been previous misdiagnosed as cough-variant asthma and failed anti-asthma treatment. PDGFRA fusion gene was confirmed in one case, but not in the other case. Both had evidence of myeloproliferative features. The tyrosine kinase inhibitor, imatinib, resulted in complete resolution of eosinophilia and cough. By searching PubMed, we found 8 HES cohorts of 411 cases between 1975 and 2013, where the incidence of cough was 23.11%. Sixteen case reports of HES presented with cough as predominant or sole symptom, with nine male patients with positive PDGFRA fusion gene, who responded well to imatinib. Six of seven patients, who tested negative for the PDGFRA, responded to systemic glucocorticoids. Conclusions: Cough and airway eosinophilic inflammation is common in some HES patients. PDGFRA+ HES patients present with chronic cough respond well to imatinib. Our case reports indicate that PDGFRA negative HES patients may respond to imatinib as well.

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Syndrome hyperéosinophilique d’origine myéloproliférative révélé par des ulcérations muqueuses bipolaires

Isvy-Joubert

Hickman

Flageul

et al. 2012

Annales de Dermatologie et de Vénéréologie

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Clonal Hypereosinophilic Syndrome: Two Cases Report in Black Men from Sub-Saharan Africa and Literature Reviews

Cited by 4 publications

References 22 publications

Severe Prolonged Cough as Presenting Manifestation of <b><i>FIP1L1-PDGFRA</i></b>+ Chronic Eosinophilic Leukaemia: A Widely Ignored Association

Severe Prolonged Cough as Presenting Manifestation of <b><i>FIP1L1-PDGFRA</i></b>+ Chronic Eosinophilic Leukaemia: A Widely Ignored Association

Cough in hypereosinophilic syndrome: case report and literature review

Syndrome hyperéosinophilique d’origine myéloproliférative révélé par des ulcérations muqueuses bipolaires

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