Langerhans's cell histiocytosis in old subjects: two rare case reports and review of the literature

Rosario Guiglia, Giuseppina Campisi, Cristiano Littarru, Michele Giuliani, Carlo Lajolo, Giorgio Deli

Risultato della ricerca: Article

10 Citazioni (Scopus)

Abstract

Background: Langerhans cell histiocytosis (LCH) is a proliferative disease of histiocyte-like cells that generally affects children; LCH onset is rare in adults; immunohistochemistry is essential to obtain the correct diagnosis, and treatment protocols are controversial. Objective: To describe two new cases of adult onset oral LCH. Case reports: Case 1: a 71-year-old woman, complaining of diffuse oral pain, presented with erythematous mucosal lesions; the panoramic radiograph and CT scan showed multiple mandible radiolucent areas. Immunohistochemical assay for S-100, CD1a and langerin test was essential in reaching the correct diagnosis. Case 2: a 77-year-old female patient presented with a non-painful, non-bleeding, slightly elevated erythematous palatal lesion of 6 months duration, together with a genital vulvar lesion of uncertain nature. The pathology confirmed the diagnosis of LCH. Many therapies (etoposid, radiotherapy) could induce only a clinical partial remission; Cladribine induced a complete recovery. Conclusion: The first case was difficult to diagnose: the clinical presentation and course of the disease (LCH) in the elderly are multiple and unpredictable. An immunohistochemistry study is often essential to obtain the correct diagnosis. The second case required several therapeutic interventions: even though some cases regress spontaneously, others require systemic chemotherapy.
Lingua originaleEnglish
pagine (da-a)e1207-e1214
Numero di pagine8
RivistaDefault journal
Volume29
Stato di pubblicazionePublished - 2012

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Langerhans Cell Histiocytosis
Immunohistochemistry
Cladribine
Histiocytes
Clinical Protocols
Mandible
Radiotherapy
Pathology
Drug Therapy
Pain
Therapeutics

All Science Journal Classification (ASJC) codes

  • Dentistry(all)
  • Geriatrics and Gerontology

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Langerhans's cell histiocytosis in old subjects: two rare case reports and review of the literature. / Guiglia, Rosario; Campisi, Giuseppina; Littarru, Cristiano; Giuliani, Michele; Lajolo, Carlo; Deli, Giorgio.

In: Default journal, Vol. 29, 2012, pag. e1207-e1214.

Risultato della ricerca: Article

Guiglia, Rosario ; Campisi, Giuseppina ; Littarru, Cristiano ; Giuliani, Michele ; Lajolo, Carlo ; Deli, Giorgio. / Langerhans's cell histiocytosis in old subjects: two rare case reports and review of the literature. In: Default journal. 2012 ; Vol. 29. pagg. e1207-e1214.
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AU - Lajolo, Carlo

AU - Deli, Giorgio

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N2 - Background: Langerhans cell histiocytosis (LCH) is a proliferative disease of histiocyte-like cells that generally affects children; LCH onset is rare in adults; immunohistochemistry is essential to obtain the correct diagnosis, and treatment protocols are controversial. Objective: To describe two new cases of adult onset oral LCH. Case reports: Case 1: a 71-year-old woman, complaining of diffuse oral pain, presented with erythematous mucosal lesions; the panoramic radiograph and CT scan showed multiple mandible radiolucent areas. Immunohistochemical assay for S-100, CD1a and langerin test was essential in reaching the correct diagnosis. Case 2: a 77-year-old female patient presented with a non-painful, non-bleeding, slightly elevated erythematous palatal lesion of 6 months duration, together with a genital vulvar lesion of uncertain nature. The pathology confirmed the diagnosis of LCH. Many therapies (etoposid, radiotherapy) could induce only a clinical partial remission; Cladribine induced a complete recovery. Conclusion: The first case was difficult to diagnose: the clinical presentation and course of the disease (LCH) in the elderly are multiple and unpredictable. An immunohistochemistry study is often essential to obtain the correct diagnosis. The second case required several therapeutic interventions: even though some cases regress spontaneously, others require systemic chemotherapy.

AB - Background: Langerhans cell histiocytosis (LCH) is a proliferative disease of histiocyte-like cells that generally affects children; LCH onset is rare in adults; immunohistochemistry is essential to obtain the correct diagnosis, and treatment protocols are controversial. Objective: To describe two new cases of adult onset oral LCH. Case reports: Case 1: a 71-year-old woman, complaining of diffuse oral pain, presented with erythematous mucosal lesions; the panoramic radiograph and CT scan showed multiple mandible radiolucent areas. Immunohistochemical assay for S-100, CD1a and langerin test was essential in reaching the correct diagnosis. Case 2: a 77-year-old female patient presented with a non-painful, non-bleeding, slightly elevated erythematous palatal lesion of 6 months duration, together with a genital vulvar lesion of uncertain nature. The pathology confirmed the diagnosis of LCH. Many therapies (etoposid, radiotherapy) could induce only a clinical partial remission; Cladribine induced a complete recovery. Conclusion: The first case was difficult to diagnose: the clinical presentation and course of the disease (LCH) in the elderly are multiple and unpredictable. An immunohistochemistry study is often essential to obtain the correct diagnosis. The second case required several therapeutic interventions: even though some cases regress spontaneously, others require systemic chemotherapy.

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