Congenital self-healing reticulohistiocytosis. A new entity in the differential diagnosis of neonatal papulovesicular eruptions

Herman, Lori E.; Rothman, Karen F.; Harawi, Sami; González‐Serva, Aldo

doi:10.1001/archderm.126.2.210

Cited by 20 publications

(12 citation statements)

References 6 publications

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“…Presentation during the neonatal period and with clearing during the first 2 to 3 months has been termed congenital self-healing reticulohistiocytosis to emphasize the limited involvement and good prognosis in this subset of patients. 2,3,9,11,13 We prefer avoiding the use of this confusing designation as the course of this disease cannot be predicted without a thorough evaluation and follow-up for extracutaneous organ involvement, although our findings suggest that a comprehensive evaluation at the time of diagnosis may be prognostically helpful. In general, LCH seems to exist on a spectrum from benign to severe and localized to disseminated, with all forms demonstrating the same findings on histopathologic and immunohistochemical studies.…”

Section: Commentmentioning

confidence: 92%

“…12 In fact, as we and others have shown, neonates commonly show vesiculopustular lesions that are easily mistaken for an infectious process, and the more classic "seborrheic" and "eczematous" lesions may be observed later in the course. 3,8,13 In addition, congenital skin lesions have been described as papules, macules or nodules, some with central crateriform ulceration, with a red, brown, blue, or yellow color. 9 These patients with nodular lesions, whether congenital or later, have generally had a better prognosis.…”

Section: Commentmentioning

confidence: 99%

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Langerhans Cell Histiocytosis Presenting in the Neonatal Period

Stein

Paller²,

Haut

et al. 2001

Arch Pediatr Adolesc Med

130

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Section: Commentmentioning

confidence: 92%

Section: Commentmentioning

confidence: 99%

Langerhans Cell Histiocytosis Presenting in the Neonatal Period

Stein

Paller²,

Haut

et al. 2001

Arch Pediatr Adolesc Med

130

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“…Congenital self-healing Langerhans cell histiocytosis (CSHLCH) is a rare condition initially seen at birth or in the neonatal period with generalized papules, vesicles, pustules, or nodules (37,38). The characteristic features of CSHLCH appear to be an otherwise healthy infant with no or mild systemic symptoms, histopathology demonstrating a Langerhans cell infiltrate, and spontaneous involution of skin lesions (39). Immunohistochemically Langerhans cells can be identified by demonstration of important markers, such as CDla (0KT6), and tlte S-100 protein, and electron microscopically' by the presence of Birbeck granules (40).…”

Section: Congenital Self-healing Langerhans Cell Histiocytosismentioning

confidence: 99%

“…The uitrastructural findings of CSHLCH differ only in degree from those other forms of LCH by reason of fewer cells with Birbeck granules and tnore with dense bodies, multivesicular bodies, and "comma-like" or ''worm-like" bodies (38,41). At the present time the clearing of skin lesions and failure to develop other organ involvement over the ensuing months is the only definitive way to be sure that an infant has the congenita] self-healing type of LCH rather than a malignant form of LCH (39). Although CSHLCH is usually a benign, self-limited condition, careful evaluation for .systemic disease must be performed.…”

Section: Congenital Self-healing Langerhans Cell Histiocytosismentioning

confidence: 99%

Diagnosis and Treatment of Pustular Disorders in the Neonate

Praag

Rooij

Folkers

et al. 1997

Pediatric Dermatology

106

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The diagnosis of a pustular dermatosis occurring during the first months of life is usually based on clinical findings. However, some cases may require simple investigations including microscopic examination of pustular content, cultures, and skin biopsies. The main benign transient neonatal types of pustulosis include erythema toxicum neonatorum, infantile acropustulosis, transient neonatal pustular melanosis, and neonatal acne. The most common causes of infectious pustular skin lesions include bacterial infections, which may be initially localized (Staphylococcus aureus) or septicemic (with Listeria monocytogenes as the leading causitive agent); viral infections (herpes simplex, varicella-zoster, and cytomegalovirus infections); fungal infections (candidiasis); or parasitic disorders (scabies). The main objective of this article is to propose a systematic approach to pustular eruptions in the neonate. The need for investigating every neonate with pustules for an infectious disease is emphasized. The Tzanck smear, the Gram's stain, and a potassium hydroxide preparation are the most important quick diagnostic tests. The Tzanck smear is a very easy, rapid, and sensitive test for detection of a herpetic infection (multinucleated giant cells) as well as noninfectious pustular eruptions (eosinophils, neutrophils). Therefore the Tzanck smear should be the first test performed. Moreover, a Gram's stain and potassium hydroxide preparation should be performed in cases of neonatal pustular disorders to detect bacterial and fungal infections. The goal of this diagnostic approach is to spare a healthy neonate with a benign transient condition an invasive evaluation for sepsis, potentially harmful antibiotic therapy, and prolonged hospitalization, with its own inherent morbidity.

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“…Since then, more than 100 cases have been reported. CSRH manifests erythematous papules, nodules or vesicles [3, 4, 5]at or shortly after birth. Most patients presented with multiple skin lesions, but a solitary lesion has been reported in up to 25% of the cases [6, 7, 8, 9].…”

Section: Introductionmentioning

confidence: 99%

Congenital Self-Healing Reticulohistiocytosis Mimicking Diffuse Neonatal Hemangiomatosis

Huang

Chao

et al. 2004

Dermatology

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Congenital self-healing reticulohistiocytosis (CSRH), a rare benign variant of Langerhans cell histiocytosis (LCH), is characterized by (a) congenital skin lesions, (b) a healthy infant with no systemic involvement, (c) the histopathologic finding of a Langerhans cell infiltrate and (d) spontaneous involution within the first year of life without sequelae. We report a Taiwanese girl born with widespread hemangioma-like lesions. The diagnosis of LCH was confirmed by finding a diffuse dermal infiltrate of S-100-protein- and OKT6 (CD1a)-positive mononuclear cells and the presence of Birbeck granules in 10% of the mononuclear cells ultrastructurally. The diagnosis of CSRH was further established by rapid and complete involution of the lesions in 3 months. No recurrence was noted for 7 years. Our case illustrates that CSRH can mimic diffuse neonatal hemangiomatosis clinically; thus, it is important to include CSRH in the differential diagnosis of congenital or neonatal hemangiomatosis.

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Congenital self-healing reticulohistiocytosis. A new entity in the differential diagnosis of neonatal papulovesicular eruptions

Cited by 20 publications

References 6 publications

Langerhans Cell Histiocytosis Presenting in the Neonatal Period

Langerhans Cell Histiocytosis Presenting in the Neonatal Period

Diagnosis and Treatment of Pustular Disorders in the Neonate

Congenital Self-Healing Reticulohistiocytosis Mimicking Diffuse Neonatal Hemangiomatosis

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