Pulmonary Langerhans Cell Histiocytosis in an Adult Male Presenting with Central Diabetes Insipidus and Diabetes Mellitus: A Case Report

Langerhans' cell histiocytosis (LCH) comprises a rare group of reticuloendothelial system disorders that can produce focal or systemic manifestations. Diabetes insipidus is considered to be an important indicator of serious underlying diseases in children, including LCH. We report the case of a young patient with monostotic LCH confined to the mandibular ramus, who was diagnosed with the disease after presenting symptoms of central diabetes insipidus and was satisfactorily treated with multi-agent chemotherapy. Additionally, we discuss the clinical, radiographic, histological and immunohistochemical findings, as well as the multidisciplinary approach of this important disease, which should receive attention by dental practitioners, especially when it occurs in children.

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Topical Imiquimod for the Treatment of Relapsed Cutaneous Langerhans Cell Histiocytosis after Chemotherapy in an Elderly Patient

Case Reports in Dermatological Medicine ◽

10.1155/2018/1680871 ◽

2018 ◽

Vol 2018 ◽

pp. 1-5

Author(s):

Shinsaku Imashuku ◽

Miyako Kobayashi ◽

Yoichi Nishii ◽

Keisuke Nishimura

Keyword(s):

Elderly Patient ◽

Elderly Patients ◽

Diabetes Insipidus ◽

Cytosine Arabinoside ◽

Langerhans Cell Histiocytosis ◽

Langerhans Cell ◽

Central Diabetes Insipidus ◽

Diagnosis And Treatment ◽

Topical Imiquimod ◽

Multiagent Chemotherapy

Diagnosis and treatment of Langerhans cell histiocytosis (LCH) in elderly patients are often difficult. We report here a 61-year-old female suffering from a refractory axillary ulcer for nearly a year, whose biopsy revealed LCH. It was also noted that the patient had other cutaneous papulovesicular eruptions of LCH as well as central diabetes insipidus. The patient was first successfully treated with multiagent chemotherapy (cytosine arabinoside/vinblastine/prednisolone). DDAVP also well controlled diabetes insipidus; however, the axillary ulcer and cutaneous LCH relapsed. Thereafter, we found topical imiquimod to be effective in the treatment of relapsed cutaneous LCH lesions.

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Langerhans Cell Histiocytosis with Isolated Central Diabetes Insipidus, Low Grade Fever and Sellar Erosion

Trends in Pediatrics ◽

10.5222/tp.2021.86580 ◽

2021 ◽

Author(s):

İclal Okur ◽

Hasan Ari ◽

Semra Çetinkaya ◽

Betül Emine Derinkuyu ◽

Gizem Çağlar ◽

...

Keyword(s):

Diabetes Insipidus ◽

Langerhans Cell Histiocytosis ◽

Bone Structure ◽

Sella Turcica ◽

Langerhans Cell ◽

Central Diabetes Insipidus ◽

Bone Lesions ◽

Warning Sign ◽

Low Grade ◽

First Case

Langerhans cell histiocytosis (LCH) is a rare disease of the monocyte-macrophage system. Although it is known that bone involvement is seen very frequently in cases with LCH, our case is the first case with a lytic-destructive lesion in the bone structure forming sella turcica. A 4-year-old, 5-month-old male patient who applied to our outpatient clinic was diagnosed with Langerhans cell histiocytosis in further examination after the diagnosis of central diabetes insipidus (CDI) was made. On cranial magnetic resonance imaging (MRI), widespread lytic-destructive bone lesions were observed in the bone structure forming the sella (sphenoid bone), sellar destruction not previously described in the literature. Sellar erosion has not been reported before in cases diagnosed with LCH in the literature. The presence of low-grade fever in a patient presenting with isolated CDI is a warning sign for the diagnosis of LCH.

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