Indian Journal of Dermatology
CASE REPORT
Year
: 2019  |  Volume : 64  |  Issue : 5  |  Page : 404--406

Disseminate and recurrent infundibulofolliculitis


S Rekha, Vineet Kumar, Pankaj Rao, Dilip Kachhawa 
 Department of Dermatology, Venereology and Leprosy, Dr. SN Medical College, Jodhpur, Rajasthan, India

Correspondence Address:
Dilip Kachhawa
Department of Dermatology, Venereology and Leprosy, Dr. SN Medical College, Jodhpur, Rajasthan
India

Abstract

Three patients, one female and two males, aged 18, 45, and 25 years, respectively, presented with the complaint of skin-colored papules interspersed with pustular lesions over the anterior and lateral aspect of the neck and upper chest for 2, 8, and 6 months, respectively. Histopathologically, mononuclear cell infiltrate centered over the infundibulum of the hair follicle was seen which confirmed the diagnosis of disseminate and recurrent infundibulofolliculitis. We report this case due to its rarity, especially in the Indian population in which less than five case reports have been published. We also report a specific pattern of lesions along the neck skin creases which can serve as an aid to increase the index of suspicion for diagnosing this entity.



How to cite this article:
Rekha S, Kumar V, Rao P, Kachhawa D. Disseminate and recurrent infundibulofolliculitis.Indian J Dermatol 2019;64:404-406


How to cite this URL:
Rekha S, Kumar V, Rao P, Kachhawa D. Disseminate and recurrent infundibulofolliculitis. Indian J Dermatol [serial online] 2019 [cited 2019 Dec 13 ];64:404-406
Available from: http://www.e-ijd.org/text.asp?2019/64/5/404/265949


Full Text



 Introduction



Disseminate and recurrent infundibulofolliculitis (DRIF) is a rare, usually asymptomatic folliculitis found predominantly in the Negroid population.[1],[2],[3] The condition is clinically characterized by asymptomatic or mildly pruritic skin-colored follicular papules distributed predominantly on the trunk and extremities. Etiology is unknown. Histopathological changes involve inflammatory infiltrate centered around the infundibulum of the hair follicle. DRIF is a very rare condition in Indian patients. We present this case series due to the rarity of the documented cases of DRIF in the Indian population.

 Case Report



Three patients, one female and two males, aged 18, 45, and 25 years, respectively, presented with the complaint of skin-colored papules interspersed with pustular lesions over the anterior and lateral aspects of the neck and upper chest [Figure 1], [Figure 2], [Figure 3] for 2, 8, and 6 months, respectively. The lesions were asymptomatic in patients 1 and 2, but associated with mild pruritus in patient 3. In all the cases, the lesions started over the anterior aspect of the neck and gradually progressed to involve the lateral neck and upper chest and shoulders over the course of 8–10 weeks. None of the patients had a history of atopy, fever, or drug intake before the onset of the lesions.{Figure 1}{Figure 2}{Figure 3}

On examination, all three cases showed the presence of multiple monomorphic skin-colored follicular papules distributed linearly along the creases on the anterior and lateral part of the neck and upper chest. There were occasional pustules scattered among the papular lesions in all three cases. There was no perilesional erythema or hyper- or hypopigmentation. No lesions were found on other areas of the body. Oral mucosa, hair, and nail examinations were unremarkable. Based on the clinical examination, differential diagnoses of keratosis pilaris, juxtaclavicular beaded lines, acneiform eruptions, lichen spinulosus, disseminate and recurrent infundibulofolliculitis, and alopecia mucinosa were considered in all the cases.

Routine investigations including hemogram, liver and renal function tests, and urine routine and microscopic examination were normal. Gram-staining and culture from the pustules were negative.

The diagnosis was confirmed through distinct histopathological features in all the cases. Histopathological examination from a papule revealed mononuclear cell infiltrate concentrated around the infundibulum of the hair follicle [Figure 4]. There was minimal spongiosis around the follicle [Figure 5]. A few scattered melanophages were seen in the dermis [Figure 6]. Other areas of the epidermis and the dermis were normal. The findings confirmed the diagnosis of DRIF.{Figure 4}{Figure 5}{Figure 6}

 Discussion



DRIF was first described in 1968 by Hitch and Lund; hence, it is also known as Hitch and Lund disease.[1] It is a rare clinical disorder of unknown etiology. The unique combination of clinical and histopathological findings characteristic of the disease enabled us to reach this diagnosis in the patients, ruling out other close differential diagnoses as mentioned before. The characteristic linear distribution of the lesions along the neck creases has previously been reported in only one case.[4]

The only specific and consistent histopathological finding in all cases of DRIF is perifollicular mononuclear infiltrate concentrated around the infundibular part of the follicle. Spongiosis and exocytosis may be very marked,[5] minimal,[6] or absent.[7] Other findings may be dilated follicle,[6] focus of fibrinoid necrosis around the follicle,[7] and dermal lymphocytic infiltration with the presence of melanophages.[5]

While there are no defined histopathological criteria for diagnosis of DRIF, these findings can be used to rule out other close differentials. Follicular eczema is characterized by diffuse dermal inflammatory infiltrate, which is not specifically concentrated around the infundibulum. Furthermore, clinically, it is associated with significant itching, seasonal exacerbation, and a history of atopic dermatitis. Keratosis pilaris displays prominent hyperkeratosis, both clinically and histopathologically, which is absent in this entity.

Malnutrition has been reported to play a role in one case report.[3] However, it has not been substantiated in later reports. Association with atopic dermatitis was described in the past, but this has been attributed to the misdiagnosis of cases of follicular eczema as DRIF.[3] Negroid races are more predisposed to this condition, so the existence of a genetic linkage to this disease has been hypothesized but not substantiated.[5]

DRIF is considered a benign self-limited condition. Treatment of DRIF is largely based on the anecdotal case reports. High-dose Vitamin A and a combination of Vitamins A and E have been used with good effect.[8] Topical potent corticosteroids and isotretinoin therapy have been tried with good results.[6],[7] Psoralen and ultraviolet A therapy and narrow-band ultraviolet B therapy have shown moderate benefit.[9]

Being mostly asymptomatic, this clinical entity appears to be underreported and should be included among the usual suspects in cases of characteristic linear distribution of monomorphic papules along the neck creases.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

References

1Hitch JM, Lund HZ. Disseminate and recurrent infundibulo-folliculitis. Arch Dermatol 1968;97:432-5.
2Thew MA, Wood MG. Disseminate and recurrent infundibulo-folliculitis. Arch Dermatol 1969;100:728-33.
3Soyinka F. Recurrent disseminate infundibulo-folliculitis. Int J Dermatol 1973;12:314.
4Nair SP, Gomathy M, Kumar GN. Disseminate and recurrent infundibulo-folliculitis in an Indian patient: A case report with review of literature. Indian Dermatol Online J 2017;8:39-41.
5Joshi SR, Chavan GR, Phadke V. Disseminate and persistent infundibulo-folliculitis. Indian J Dermatol Venereol Leprol 1996;62:112-3.
6Calka O, Metin A, Ozen S. A case of disseminated and recurrent infundibulofolliculitis responsive to treatment with systemic isotretinoin. J Dermatol 2002;29:431-4.
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8Arnold HL, Odom RB, James WD. Andrews' Diseases of the Skin. 8th ed. Philadelphia, PA: Saunders WB; 1990. p. 906-7.
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