case_images: PMC11286394_01
case_images: Timeline of patients 1 and 2 with LCH.
case_images: fmed-11-1433463-g001.jpg
case_images: PMC11286394_02_fmed-11-1433463-g001.jpg
case_images: fig1
case_images: This led to a positive clinical response, with cessation of fever and an increase in Hb (Figure 1).
case_images: Patient 1 subsequent bone marrow biopsy. Panel of microphotographs from bone marrow trephine biopsy of a case 1 of LCH indicating BM infiltration by Langerhans Cells: (A) Bone marrow trephine biopsy showing a diffuse area comprising of Langerhans cells with a multinucleated giant cell and some eosinophils (Hematoxylin and Eosin stain, magnification 100X). (B) Bone marrow trephine biopsy showing the morphology of Langerhans cells at higher magnification, the cells having grooved nuclei (Hematoxylin and Eosin stain, magnification 400X). (C) Immunohistochemistry for CD68 showing membranous positivity in histiocytes and Langerhans cells (magnification 200X). (D) Immunohistochemistry for S100 showing nuclear positivity in Langerhans cells (magnification 200X). (E) Immunohistochemistry for CD1a showing membranous positivity in Langerhans cells (magnification 200X). (F) Immunohistochemistry for Cyclin D1 showing nuclear positivity in Langerhans cells, and multinucleated Histiocyte (magnification 200X).
case_images: fmed-11-1433463-g002.jpg
case_images: PMC11286394_01_fmed-11-1433463-g002.jpg
case_images: fig2
case_images: A subsequent BM core biopsy was examined 2 weeks later, showing an adequate BM biopsy with diffuse multiple atypical multinucleated cells (Figures 2A,B). An extensive immunophenotype panel was requested, where CD1a, CD68, Cyclin D1, and S100 were positive in atypical multinucleated cells (Figures 2C-F).
case_images: PMC11286394_02
case_images: Timeline of patients 1 and 2 with LCH.
case_images: fmed-11-1433463-g001.jpg
case_images: PMC11286394_02_fmed-11-1433463-g001.jpg
case_images: fig1
case_images: This led to a positive clinical response, with cessation of fever and an increase in Hb (Figure 1).
case_images: Patient 2 lymph node biopsy. Panel of microphotographs from cervical lymph node biopsy of patient 2 indicating LCH: (A) Lymph node biopsy showing a patchy infiltration of Langerhans cells (Hematoxylin and Eosin stain, magnification 40X). (B) Lymph node biopsy showing the morphology of Langerhans cells at higher magnification (Hematoxylin and Eosin stain, magnification 400X). (C) Immunohistochemistry for CD1a showing membranous positivity in Langerhans cells (magnification 100X). (D) Immunohistochemistry for CD68 showing membranous positivity in histiocytes and Langerhans cells (magnification 100X). (E) Immunohistochemistry for KI67 showing nuclear positivity in Langerhans cells with 10-15% KI67 index (magnification 100X). (F) Immunohistochemistry for S100 showing nuclear positivity in Langerhans cells (magnification 100X).
case_images: fmed-11-1433463-g003.jpg
case_images: PMC11286394_02_fmed-11-1433463-g003.jpg
case_images: fig3
case_images: A cervical lymph node biopsy was performed, revealing interstitial infiltrations of histiocytes positive for CD1a, CD68, and S100, leading to the diagnosis of LCH (Figure 3).
case_images: Patient 2 bone marrow biopsy. Panel of microphotographs from bone marrow trephine biopsy of patient 2 of LCH indicating BM infiltration by Langerhans Cells: (A) Bone marrow trephine biopsy showing a patchy infiltration of Langerhans cells and some eosinophils (Hematoxylin and Eosin stain, magnification 100X). (B) Bone marrow trephine biopsy showing the morphology of Langerhans cells at higher magnification, the cells having grooved nuclei (Hematoxylin and Eosin stain, magnification 400X). (C) Immunohistochemistry for CD68 showing membranous positivity in Langerhans cells (magnification 100X). (D) Immunohistochemistry for S100 showing nuclear positivity in Langerhans cells (magnification 100X). (E) Immunohistochemistry for CD1a showing membranous positivity in Langerhans cells (magnification 100X).
case_images: fmed-11-1433463-g004.jpg
case_images: PMC11286394_02_fmed-11-1433463-g004.jpg
case_images: fig4
case_images: Due to the suspicion of bone marrow (BM) infiltration associated with moderate anemia, a BM trephine biopsy was requested, confirming patchy interstitial infiltrations of histiocytes with CD1a, CD68, and S100 positivity in the tumor cells (Figure 4).