History and exam

Key diagnostic factors

common

age <15 years

Disease is more frequent in children. The mean age at diagnosis of patients ages <15 years is 30 months.[6]

bone pain and/or swelling

Indicative of bone involvement.

Bone lesions are present in 80% of patients, and are commonly seen on the scalp.[10] Lytic osseous lesions are seen in 30% to 50% of adults, and can occur as single-system or multisystem disease.[12] 

Most common site of swelling is over the skull bones, and this is usually painless.

skin rash

Indicative of skin involvement. Most common location is the scalp.[29]

Most common lesion in the first few months of life is a papulosquamous rash that affects the scalp, skin folds, and midline of the trunk.[30][31]

A single or multiple violaceous papulonodular, and sometimes vesicular, rash may also occur, but is rare and may disappear spontaneously.[32]

Among adults, isolated skin involvement is less common, and mostly seen as a part of multisystem disease (30% to 50%).[12] Perianal or genital involvement is more common in adults and can be very disabling and resistant to treatment.

polyuria and polydipsia

Diabetes insipidus is the most common endocrinologic manifestation; it is more common in patients with multisystem disease and those with craniofacial bone lesions.[27]

Risk is high in patients when the disease has been active for a long period of time, or when the disease is reactivated.

growth or sexual maturation failure

Indicative of central nervous system involvement (anterior pituitary gland).

vertebra plana

Spondylitis with reduction of the vertebral body to a thin disk. Leads to compression and collapse of the vertebral body.

Neurologic complications may arise from extension of Langerhans cells into the extradural space.

hepatosplenomegaly

Indicative of liver and spleen involvement.

Other diagnostic factors

common

cough, dyspnea

Indicative of lung involvement.

chest pain

May indicate spontaneous pneumothorax, which may be the first sign of lung involvement.

orbital abnormalities

Indicative of eye involvement.

Includes proptosis, periorbital swelling and erythema, diplopia, or ophthalmoplegia.[45][46]

jaundice

Indicative of liver involvement.[42]

ascites

Indicative of liver involvement.[42]

uncommon

cytopenias

Indicative of bone marrow involvement.[2]

fever

A nonspecific, but common, symptom in infants with multisystem disease.[59]

anorexia

Nonspecific symptom.[2]

weight loss

Nonspecific symptom.[2]

irritability

Nonspecific symptom.[2]

ear pain

Presence of otitis externa can be due to extension of the skin rash into the ear canal and secondary infection with Pseudomonas aeruginosa.[47]

ear discharge

Middle ear involvement can occur secondary to lesions in petrous temporal or mastoid bones, causing intermittent ear discharge.[48]

hearing loss

Inner ear involvement can present with deafness, or with behavioral disturbances in younger children.[49]

perforated tympanic membrane

May occur with middle ear involvement.

headache

May indicate central nervous system involvement.[2]

neurologic signs

Indicative of central nervous system involvement.

Patient may present with seizures, focal neurologic deficits, or progressive cerebellar symptoms such as nystagmus, dysarthria, and hypotonia.[27]

A neurodegenerative syndrome can occur in up to 4% of patients, and usually manifests as ataxia, dysmetria, dysarthria, and behavioral changes.[3][60]

bloody diarrhea

Indicative of gastrointestinal involvement.[51]

oral mucosa lesions

Indicative of oral involvement.

Includes gingival hypertrophy, and ulcers of the palate, tongue, or lips.[50]

May precede the onset of disease in other parts of the body.

lymphadenopathy

Usually part of multisystem disease, but can also occur in isolation, or is sometimes associated with a single bone lesion.[38][39]

Superior vena cava syndrome can result from mediastinal lymph node or thymic enlargement.[40] Careful analysis of phenotype and histopathology is recommended to differentiate reactive Langerhans cells in LCH from primary malignant histiocytosis of the lymph node.

thyroid enlargement

Patients may present with massive thyroid enlargement; however, this is rare.[52] Signs of hypothyroidism may be present.

Risk factors

strong

smoking

In adolescents and adults, smoking has been found to be a clear risk factor for the development of lung involvement. Most children with systemic disease who developed pulmonary disease in adolescence or adulthood started smoking cigarettes before this event.[23]

weak

family history of thyroid disease

Multisystem and single-system disease have both been associated with a family history of thyroid disease.[24]

perinatal infections

An increase in infections and the use of antibiotics in the first 6 months of life have been associated with multisystem disease.[24]

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