Langerhans Cell Histiocytosis in a Pediatric Patient with Simultaneous Gastrointestinal and Skin Involvement: A Case Report and Literature Review

Fang, Junyong; Deng, Chunhua; Li, Jiwei; Huang, Ti-Long; Liu, Ying; Xie, Yucheng; Wang, Mingying

doi:10.1080/15513815.2022.2142488

Cited by 7 publications

(9 citation statements)

References 8 publications

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“…Both of our cases were less than 2 years old. Referring to other 6 literature reports with GIT symptoms as the initial manifestation, all the 7 cases were all younger than 2 years old [3,[5][6][7][8][9]. It seems to be that GIT-LCH was common in children aged < 2 years at diagnosis.…”

Section: Discussionmentioning

confidence: 77%

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Langerhans cell histiocytosis in children with refractory diarrhea and hypoalbuminemia as the initial presentation: two cases report and literature review

Cao,

Wu,

Yan

et al. 2023

Preprint

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Langerhans cell histiocytosis (LCH) with involvement of the gastrointestinal tract is rare and lack of clinical experience. We describe two cases who initially presented with chronic diarrhea, hypoproteinemia, and intermittent fever. These cases suggest that refractory diarrhea accompanied by recurrent hypoalbuminemia, especially with abdominal rash, LCH should be considered. Then gastrointestinal endoscopy, pathology and PET-CT detection need to be perfected for the definitive diagnosis. It might be helpful for the early recognition of the gastrointestinal tract involvement in LCH.

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Section: Discussionmentioning

confidence: 77%

“…The main gastrointestinal symptoms in our two cases were refractory diarrhea and protein-losing enteropathy. Among the above 7 cases, the initial GIT symptoms were diarrhea in 5 cases, abdominal distension in 1 case, and vomiting in 1 case; while hypoalbuminemia was reported in 6 cases [3,[5][6][7][8][9].…”

Section: Discussionmentioning

confidence: 99%

Langerhans cell histiocytosis in children with refractory diarrhea and hypoalbuminemia as the initial presentation: two cases report and literature review

Cao,

Wu,

Yan

et al. 2023

Preprint

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“…LCH is likely a clonal neoplastic illness with extremely varied biologic behavior, according to Willman et al 1 Because many LCH patients exhibit hypergammaglobulinemia, thymic dysfunction, or reduction in suppressor T lymphocytes in the peripheral blood, Shannon et al suggested that LCH is Langerhans cell proliferation and accumulation at a focal area by immunoregulatory dysfunctions. 2 The Histiocytosis Society Writing Group reported LCH to be the preferred word in 1987, 3 replacing Lichtenstein's 1953 term "histiocytosis X." 4 According to their clinical and pathologic characteristics, this condition covers three diseases: Letterer-Siwe syndrome, eosinophilic granuloma (EG), and Hand-Schuller-Christian syndrome.…”

Section: Introductionmentioning

confidence: 99%

“…The Histiocytosis Society Writing Group reported LCH to be the preferred word in 1987, 3 replacing Lichtenstein's 1953 term “histiocytosis X.” 4 According to their clinical and pathologic characteristics, this condition covers three diseases: Letterer‐Siwe syndrome, eosinophilic granuloma (EG), and Hand‐Schuller‐Christian syndrome 5 . The most frequent form of LCH is EG, which accounts for roughly 70% of patients 5 .…”

Section: Introductionmentioning

confidence: 99%

Solitary Langerhans cell histiocytosis of the sternum in a 21‐year‐old woman

Soleimani,

Hosseinzadeh,

Amirian

et al. 2024

Clinical Case Reports

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Key Clinical MessageChildren are more likely to develop Langerhans cell histiocytosis (LCH), a rare disorder with an unknown cause. LCH often invades skeletal systems, while it has occasionally been seen in the sternum or ribs. The best course of treatment for single‐site, skeletal LCH is yet unknown. We present an instance of sternal LCH with adult onset. By fusing and reconstructing chest computed tomography, it was possible to determine the extent of surrounding soft tissue invasion. Because LCH is so uncommon, it could be challenging to recall when we see a sternal lesion. Adult Patients who arrive with anterior chest discomfort and an osteolytic sternal lesion should include LCH on their differential diagnosis list.

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“…In the case of LCH, granulomatous lesions comprising langerin-positive (CD207+) histiocytes with accompanying inflammatory infiltrate can arise in virtually any organ system but have a particular affinity for bones, skin, the lungs, and the pituitary gland [4,5]. Histiocytic diseases are rare diseases with a widely variable clinical presentation, ranging from single indolent lesions to explosive multisystem disease, pathogenesis of which is related to disorder of proliferation and differentiation of cells of the phagocytic system [6,7]. The underlying cause of this disease is the clonal proliferation of histiocytes (Langerhans cells) immunohistochemically and morphologically resembling dendritic cells.…”

mentioning

confidence: 99%

Diagnostic and therapeutic difficulties in the cutaneous form of Langerhans cell histiocytosis in infant

Arasiewicz,

Dec,

Lesniak-Jakubiec

2023

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Introduction: Langerhans cell histiocytosis is an uncommon inflammatory and neoplastic condition of myeloid dendritic cells. Langerhans cell histiocytosis presents with a wide spectrum of clinical manifestations, ranging from single-system involvement to disseminated disease involving multiple organs. Diagnosis is based on a combination of clinical features, radiological findings, and histopathological examination. Positive immunohistochemical staining of lesional cells for CD1a and S100 is necessary for definitive diagnosis of Langerhans cell histiocytosis. Case report: Herein, we describe the case of a male infant who presented with multiple erythematous-papular lesions covered with yellow scales on the scalp, temples and face and oozing erythematous and erosive lesions mainly on the abdomen, upper chest, groins, gluteal cleft and armpits. Conclusions: Further research is needed to unravel the underlying pathogenesis, develop standardized diagnostic criteria, and improve treatment strategies. Collaboration among various specialties, including dermatology, pediatrics, oncology, radiology, and pathology, is essential for optimal management and long-term follow-up of this complex entity.

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Langerhans Cell Histiocytosis in a Pediatric Patient with Simultaneous Gastrointestinal and Skin Involvement: A Case Report and Literature Review

Cited by 7 publications

References 8 publications

Langerhans cell histiocytosis in children with refractory diarrhea and hypoalbuminemia as the initial presentation: two cases report and literature review

Langerhans cell histiocytosis in children with refractory diarrhea and hypoalbuminemia as the initial presentation: two cases report and literature review

Solitary Langerhans cell histiocytosis of the sternum in a 21‐year‐old woman

Diagnostic and therapeutic difficulties in the cutaneous form of Langerhans cell histiocytosis in infant

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