Infantile generalized hypertrichosis caused by topical minoxidil

Rare cases of hypertrichosis have been associated with topically applied minoxidil. We present the first reported case in the Brazilian literature of generalized hypertrichosis affecting a 5-year-old child, following use of minoxidil 5%, 20 drops a day, for hair loss. The laboratory investigation excluded hyperandrogenism and thyroid dysfunction. Topical minoxidil should be used with caution in children.     

Telogen effluvium related to post severe Sars‐Cov‐2 infection: Clinical aspects and our management experience

Telogen effluvium (TE) is one of the most common form of hair loss in women. Many triggers have been identified, as stress, drugs, trauma, endocrine disease, nutritional deficiencies, and febrile states. We report three cases of TE occurred after severe Sars‐Cov‐2 infection and provide our clinical management, according to Sars‐Cov‐2 hygiene measures. Only one case report has been found in the literature associating anagen effluvium during severe Sars‐Cov‐2 infection. Other studies reported the exacerbation of a preexisting TE, correlated to the stress of lockdown. In our cases, patients never had a TE diagnosis before and did not report previous evident hair loss. TE can be associated with post severe Sars‐Cov‐2 infection. From our revision of the literature, this is the first case‐series describing TE in post severe Sars‐Cov‐2 patients. Further studies are needed to evaluate the relationship between TE and Sars‐Cov‐2 infection.     

Does collapse of immune privilege in the hair‐follicle bulge play a role in the pathogenesis of primary cicatricial alopecia?

Background. The hair‐follicle bulge has recently been added to a growing list of human tissue compartments that exhibit a complex combination of immunosuppressive mechanisms, termed immune privilege (IP), which seem to restrict immune‐mediated injury in specific locations. As epithelial hair‐follicle stem cells (eHFSC) reside in the hair‐follicle bulge region, it is conceivable that these IP mechanisms protect this vital compartment from immune‐mediated damage, thereby ensuring the ongoing growth and cyclic regeneration of the hair follicle. Primary cicatricial alopecias (PCA) are a group of inflammatory hair disorders that result in hair‐follicle destruction and permanent alopecia. Growing evidence suggests that eHFSC destruction is a key factor in the permanent follicle loss seen in these conditions. Aim. To explore the possible role of bulge IP collapse in PCA pathogenesis. Methods. We report three clinically distinct cases of PCA. Immunohistochemical analyses of paired biopsies from lesional and uninvolved scalp skin were compared using recognized markers of IP. Results. Immunohistochemical investigation found increased expression of major histocompatibility complex (MHC) classes I and II and of β2‐microglobulin in the bulge region of lesional follicles compared with uninvolved follicles in each case. Further, expression of the bulge marker keratin 15 was reduced in lesional skin in two of the cases. Conclusions. This small series represents our first preliminary attempts to ascertain whether bulge IP collapse may play a role in PCA pathogenesis. We present standard parameters relating to hair‐follicle IP in the bulge region of three patients with distinct PCA variants, and show the presence of features consistent with bulge IP collapse in each case.     

'A detective look' at hair biopsies from African-American patients

Background A patient’s ethnicity can be an important clue in the diagnosis of scarring alopecia as some disorders such as traction alopecia (TA) and central centrifugal cicatricial alopecia (CCCA) are more prevalent in or exclusive to African‐Americans. Objectives To perform a retrospective review of 60 scalp biopsies from African‐American patients including 25 cases of CCCA, 22 cases of TA, five cases of frontal fibrosing alopecia, three cases of discoid lupus erythematosus, three cases of hair breakage and two cases of alopecia areata. Methods Serial horizontal and vertical sections were examined. Results Features characteristic of the African‐American scalp include: golf club‐shaped bulb, elliptical shape of the hair shaft, asymmetrical outer root sheath and paired grouping of hair follicles. Clues to the diagnosis of CCCA include: premature desquamation of the inner root sheath, goggles and naked hair shafts in fibrous streamers. Diagnosis of TA is suggested by preserved sebaceous glands along with follicular miniaturization and drop‐out. Conclusions The clues reported here aim to help the dermatopathologists to: recognize at a glance that they are dealing with a scalp biopsy from an African‐American patient; make the most probable diagnosis by connecting the clues (even if only vertical sections are present); and understand the morphological basis for the susceptibility of the African hair to damage.     

Congenital Lipedematous Alopecia: Adding to the Differential Diagnosis of Congenital Alopecia

Lipedematous alopecia is a rare condition of unknown etiology characterized by a thick boggy scalp with varying degrees of hair loss. It is usually seen in adult African-American females, and a case in a 9-year-old was the youngest patient reported thus far. We report on the appearance of this condition in two children, a 6-year-old child and a 10-year-old child. Each presented with congenital patchy hair loss on the occipital area and the left temple. A boggy hairless scalp with soft swelling was detected in both patients. Histological examination showed increased thickness of the subcutaneous fat tissue with a decrease in hair follicles. These features were consistent with a diagnosis of lipedematous alopecia. We report two cases of congenital lipedematous alopecia, which has not been reported previously. Although congenital, these distinct clinical features should be kept in mind in the diagnosis of alopecic hair loss.     

Frontal fibrosing alopecia associated with generalized hair loss

We present six cases of frontal fibrosing alopecia, in which generalized hair loss is a feature. Although this variant of lichen planopilaris has been reported clinically in a number of patients, there is very little histological evidence that the condition exists in peripheral sites. We believe this pattern of involvement may be more common than is reported, and have provided histological evidence of lichen planopilaris being present at sites beyond the scalp and eyebrows.     

Plica neuropathica: novel presentation of a rare disease

Plica neuropathica is a rare condition involving complete matting of scalp hair. It typically occurs in otherwise healthy individuals, and in previous cases has been caused by either shampooing or neglect of hair. Cutting off the matted hair is the only solution. We present the case of a 14-year-old girl with a history of chronic illness and an acute infection presenting with plica neuropathica followed by complete hair loss. This unique case illustrates for the first time plica neuropathica in the absence of shampooing, and demonstrates that it can be associated with underlying illnesses and subsequent hair loss.     

Sclerotic Regressing Large Congenital Nevus

Regression of congenital nevi is usually associated with loss of pigment or halo formation. In rare cases, regression is characterized by sclerosis and hair loss. We describe a rare case of a sclerotic hypopigmented large congenital melanocytic nevus in which a localized scleroderma‐like reaction process of regression seemed to have started in utero and progressed throughout early childhood.     

Ultrasound Diagnosis and Staging in Pediatric Hidradenitis Suppurativa

Hidradenitis suppurativa (HS) can affect children, and ultrasound has been proven to be useful in diagnosis and staging. The sonographic characteristics of HS in children have not been reported. We studied color Doppler ultrasound images of children (≤15 years old; n = 12) with clinically and sonographically positive criteria for HS. Sonographic scoring of hidradenitis suppurativa (SOS‐HS) was used to stage the cases sonographically. Subclinical pseudocysts were found in 92% of the cases, fluid collections in 83%, and fistulous tracts in 58%. Retained hair tracts in the fluid collections and fistulous tracts were present in 100% of patients; 67% of cases were SOS‐HS stage II. In 92% of cases, management was modified after the ultrasound examination. In conclusion, ultrasound can be a reliable and safe imaging tool to support diagnosis and staging and may help in the noninvasive monitoring of treatment in children.     

Periocular Granuloma Annulare: A Case Report and Review of Literature

Granuloma annulare (GA) is a granulomatous dermatosis that rarely presents on the face and is extremely uncommon in the periocular region. We report our experience with the presentation and management of GA lesions on the eyelids of a 17‐year‐old girl. We performed a review of published literature and identified 13 cases of pediatric periocular GA. One additional case was identified upon review of all pediatric GA cases at the Cleveland Clinic Foundation. Review of these cases suggests that periocular GA is a benign condition that spontaneously regresses within a few months. GA nodules have a predilection for the upper eyelids. A greater incidence is noted in African American children. Awareness of the self‐resolving nature of this condition can prevent unnecessary surgical excisions in affected children.     

THERAPEUTIC HOTLINE: Effectiveness of the association of cetirizine and topical steroids in lichen planus pilaris – an open‐label clinical trial

Lichen planus is considered a T cell‐mediated immunological disease. Even mast cells may contribute to the pathogenesis of the disease. Keratinocytes of the basal layer of the skin and/or the hair follicle may represent the “target/victim” of an immune aggression, determining the destruction of the hair follicle and thus scarring alopecia. Therefore, there is a compelling urgency for effective treatment of this potentially disfigurating dermatosis. Our data provide a further therapeutic opportunity: the use of an antihistaminic drug – cetirizine (CTZ) – in an “anti‐inflammatory” regimen. We propose the use of CTZ at the dosage of 30 mg/daily. Twenty‐one patients affected by lichen planus pilaris (LPP) of the scalp have been treated. Topical application of steroids has been coadministrated in all cases during the therapy. Clinical effects, in the sense of stabilization with cessation of the inflammation (erythema, follicular hyperkeratosis, loss of anagen hair), were achieved in all patients but three. One patient developed cardiac arrhythmia after 3 months of successful treatment and dropped out. Our cases indicate that a combined therapy of topical steroid with CTZ can be a safe and effective choice even in severe cases of lichen planus pilaris, so often refractory to the therapy.     

Alopecia areata and isotretinoin; coincidence or causal relation

Isotretinoin is a safe and useful medication for acne management. Adverse effects are mostly related to cutaneous and mucous membranes. We present a case of a 24‐year‐old female patient who was on treatment with isotretinoin for acne vulgaris Grade 4. After 6 months of treatment, the patient presented with localized patch of hair loss. Beginning of alopecia areata in patients with acne vulgaris could be regarded as a side effect of retinoids due to anti‐acne therapy. The exact mechanism by which retinoids cause hair loss is not known.     

Intractable Diffuse Alopecia Caused by Multifactorial Side‐Effects in Treatment of Acute Lymphocytic Leukemia: Connection to Iatrogenic Failure of Estrogen Secretion

Abstract: Treatment of infantile acute lymphocytic leukemia (ALL) may cause failure to thrive and hypogonadism due to hypopituitarism induced by chemotherapy and whole‐brain radiotherapy. We report the case of a 22‐year‐old girl with a genetic predisposition to pattern hair loss who developed inveterate diffuse alopecia. The patient had onset of ALL at 8 years old and underwent bone marrow transplantation (BMT). Diffuse alopecia gradually advanced over her whole body. Her vellus scalp hair gradually came out, and hair loss progressed again at 8 years, after BMT. She later developed iatrogenic failure of secretion of estrogen and was treated with estrogen substitution therapy for 14 months from the age of 20. There was a small increase in the volume of hair during therapy, but alopecia returned to the former level after the therapy was suspended. Histopathologic examinations of the scalp performed during estrogen substitution therapy and 2 years after suspension of the therapy showed a 60% decrease in the number of hair follicles and prominent development of vellus hair. We conclude that estrogen influenced hair growth in the context of a genetic predisposition for pattern hair loss in this case.     

The nevoid pili multigemini over the back

Pili multigemini is a rare developmental defect of hair follicles. It usually involves the beard of an adult and the scalp in children, but there have been no reported cases on the back. We report 3 cases of extensive pili multigemini over the entire back of healthy males. All cases were found incidentally. The direct microscopic examination of one hair follicle showed that it consisted of more than twenty shafts, which had rough surfaces and brush-like bulbs. The histological examination revealed that complicated follicular structures formed as many as 23 to even 36 hair shafts. Lots of hairs were surrounded by each inner root sheath and had one common outer root sheath. We suggest this nevoid is pili multigemini over the back.     

Schwere allergische Reaktion auf Para‐Phenylendiamin bei Kindern und Jugendlichen: Muss die Test‐Konzentration von PPD angepasst werden?

Background: Semipermanent henna tattoos containing para‐phenylenediamine (PPD) are a well‐known cause of severe contact dermatitis, mainly in children. Another relevant exposure source to PPD are hair dyes, which are increasingly used by children and have also been reported to cause intense facial and scalp dermatitis. German patch test guidelines for children recommend that PPD should only be tested in patients who have had a henna tattoo, and then at a reduced concentration of PPD 0.5 % for 24 hours. Patients and methods: We report on patch test results in four patients, three children and one adolescent, with a history of contact dermatitis to henna tattoos or hair dye. We used the recommended or even lower patch test concentrations of PPD with 24‐hours exposure in all patients. Results and conclusion: All patients showed very strong patch test reactions to PPD and cross‐reactions to related compounds even after dilution of PPD to as low as 0.05 %. Therefore, we suggest that in children with a history of allergic reactions to this compound, a titration test should be performed beginning at a concentration of maximal 0.05 %. This procedure has also been proposed previously based on a larger cases series in adults.     

Psoriatic alopecia

Alopecia and other hair abnormalities occurring in patients with psoriasis were first recognized over four decades ago, yet psoriatic alopecia is not a well‐known concept among clinicians. Alopecia may be directly related to the psoriasis itself, and can affect both the scalp and other parts of the body. On the scalp, psoriatic alopecia most commonly affects lesional skin, but may present as a generalized telogen effluvium. In most cases, there is regrowth of hair, but in rare cases it can cause scarring alopecia. Histological findings include features of psoriasis in the interfollicular epithelium, along with perifollicular inflammation and atrophy or loss of the sebaceous glands. Late changes include destruction of the hair follicle, with perifollicular fibrosis and ‘naked’ hair shafts lying free in the dermis. In addition to the hair loss caused by the psoriasis itself, data from population and genetic studies reveal that patients with psoriasis are at greater risk of developing alopecia areata. Psoriasis treatments may also contribute to hair loss. Application of topical preparations may cause hair loss through friction, and many of the systemic treatments used for psoriasis can also cause hair problems. Treatment with anti‐tumour necrosis factor‐α agents can precipitate de novo psoriasis and subsequent psoriatic alopecia.     

Angiomatoid and desmoplastic Spitz nevus presenting as a keloidal nodule

Angiomatoid and desmoplastic Spitz nevi are rare histologic variants of Spitz nevi that present most frequently on the extremities of children and young adults. Although Spitz nevi are clinically heterogeneous, one presenting as a keloidal nodule has not been previously published. We present a case of an angiomatoid and desmoplastic Spitz nevus clinically akin to a keloid on an African‐American teenager and describe its unique histopathologic features.     

Marie Unna hypotrichosis in an Asian family

We report on two patients from one family with Marie Unna hypotrichosis who showed a diffuse hair defect which typically occurs as an isolated phenomenon. The ultrastructural hair findings consisted of longitudinal grooving of the hair shafts and a flattening in cross-section. Characteristically, scalp hair had been lost from the scalp margins and a high frontal and nuchal hairline had been present at birth. Eyelashes were absent, and eyebrows, axillary, and pubic hair were very sparse. Other affected cases have been reported in Caucasian families; however, this is the first case in the English medical literature of a family from Asia affected by Marie Unna hypotrichosis.     

Androgenetic alopecia in children: report of 20 cases

Androgenetic alopecia (AGA) is the most common type of hair loss in adults. Although there are differences in the age at onset, the disease starts after puberty when enough testosterone is available to be transformed into dihydrotestosterone. We report 20 prepubertal children with AGA, 12 girls and eight boys, age range 6-10 years, observed over the last 4 years. All had normal physical development. Clinical examination showed hair loss with thinning and widening of the central parting of the scalp, both in boys and girls. In eight cases frontal accentuation and breach of frontal hairline were also present. The clinical diagnosis was confirmed by pull test, trichogram and dermoscopy in all cases, and by scalp biopsy performed in six cases. There was a strong family history of AGA in all patients. The onset of AGA is not expected to be seen in prepubertal patients without abnormal androgen levels. A common feature observed in our series of children with AGA was a strong genetic predisposition to the disease. Although the pathogenesis remains speculative, endocrine evaluation and a strict follow-up are strongly recommended.     

Centrifugal lipodystrophy of the scalp manifesting as centrifugal lipodystrophic alopecia

Centrifugal lipodystrophy (CLD), characterized by a depressed lesion in the abdominal skin, is a chronic disease occurring more often among younger patients of East Asian descent. We present an extremely unusual case of CLD of the scalp associated with reversible hair loss. The patient demonstrated alopecia in the frontal, temporal and occipital areas of the scalp, which connected to form a ring‐shaped area of hair loss. Curiously, the area of hair loss gradually expanded outwards while the central region showed normal hair regrowth. Immunohistochemical analysis demonstrated reduced expression of leptin, an adipokine capable of inducing the anagen phase of the hair cycle, in the adipose tissue, associated with active inflammation. By contrast, recovery of leptin expression was observed at sites of healed inflammatory lesions, suggesting that reversible hair loss might be caused by a change in leptin expression in adipose tissue.