A rare case of congenital, sporadic, true Leukonychia Totalis

 

Dr. Satish Ramteke1, Dr. Sharmila Ramteke2, Saurabh Nagar3, Dr. Mahendra Kumar Jain4

 

1Assistant Professor, Department of Medicine, Shyam Shah Medical College and Associated Sanjay Gandhi Memorial Hospital, Rewa, M.P., India.

2Assistant Professor, Department of Pediatrics, Gandhi Medical College, Bhopal, M.P., India.

3RMO, Department of Medicine, Shyam Shah Medical College and associated Sanjay Gandhi Memorial Hospital, Rewa, M.P., India.

4Professor & Head, Department of Medicine, Shyam Shah Medical College and Associated Sanjay Gandhi Memorial Hospital, Rewa, M.P., India.

 

ABSTRACT:

We report a rare case of a 40 year old patient with sporadic, congenital, true, leukonychia totalis, not associated with any clinical syndromes or any systemic disease. This is the third case to be reported in literature.

 

KEYWORDS: Leukonychia totalis.

 

CASE PRESENTATION:

A 40 year male, known case of hypertension, was admitted with uncontrolled hypertention .On examination total leukonychia of all twenty nails (figure 1) was found. Patient informed that his all twenty nails have been white ever since he could remember. He had trauma over left thumbnail after which it got distorted, which was totally white similar to other nails earlier to the trauma.  There was no evidence of atopy, lichen planus, alopecia areata or psoriasis. There was no history of exposure to any chemical agent and of any drug intake other than antihypertensive. He had no central nervous system, eye, ear, hair, teeth, or skin abnormalities. The patient was born to non consanguineous parents. His sister also had similar white nails in all twenty digits since birth but none of their parents had such a phenotype. Routine blood investigations including USG abdomen were normal.

 

Fig 1

 

 


DISCUSSION:

Leukonychia is whiteness of nails which may occur either in patches or may involve the entire nail1,8. The condition can be either true leukonychia, with involvement of nailplait, or pseudoleukonychia, caused by subungual and nailbed abnormalities. Leukonychia may be acquired or inherited .Aquired leukonychia is associated with trauma, drugs (chemotherapeutic agents2,8) systemic or local infection such as typhoid, hepatic cirrohsis ,ulcerative colitis , leprosy etc .True leukonychia can be inherited as an isolated condition or as a part of some syndromes. There are number of autosomal dominantly inherited leuconychia syndromes, including leukonychia totalis, where it is seen in combination with kidney stone, sebaceous cysts 3,4,8, sensoryneural deafness and knuckle pads5,6,8.

 

Leuconychia is whitening of the nail plate. It was first described by Mees in 1919 as an associated finding in arsenic intoxication7,8 . Abnormal karatinization of nail plate is belived to be responsible for true leuconychia. According to Newton’s theorem, a surface appears white when it reflects the radiation of visible light. Because  true leukonychia  is thought to be due to  abnormal  matrix  keratinisation , with  persistent  parakerosis .  Large karatohyline granules are found in karatinocytes in nail plate. This parakerotosis and dissociation of karatin bundles may play role in the modification of the light reflection by ungueal plates. True leukonychia can be hereditary or acquired8. Hereditary leukonychia can be as an isolated finding or may be associated with some symdromes like Bart-Pumphrey syndrome, Bauer syndrome, Heimler syndrome, Knuckle pads-leukonychia-deafness syndrome, Keratoderma, hypotrichosis and leukonychia totalis syndrome, Lowry-Wood syndrome, FLOTCH syndrome, congenital keratosis palmaris et plantaris, deafness and total leukonychia, LEOPARD syndrome, while acquired leoconychia can be idiopathic or associated with trauma, drug, systemic infection, local infection, and inflamatory disease. Appearance wise leuconychia can be classified as leuconychia punctata , leukonychia striata, leokonychia partialis and totalis.

 

Our case is not associated with any of above mentioned syndromes. Leuconychia totalis either hereditary or acquired is a rare entity.  Only six cases of idiopathic, acquired, true leukonychia totalis without hereditary cause has been found in literature8, whereas   only two cases of sporadic congenital true leuconychia totalis reported in literature9 before our case.  One of which was associated with deafness and vitiligo while the other was isolated, like our case.   It is importance to report this case not only because the entity is rare but also it may have associated abnomalties. If no association is detected, proper counselling of the patient regarding the benign nature of the condition should suffice.

 

REFERENCES:

1        M. Grossman and R.K. Scher, “leuconychia: review and classification, ”International journal of Dermatology,vol.29, no.8, pp.535-541,1990.

2        A. Yoruk and H. Yukselgungor, “ Chemotherapy induced transverse leukonychia in children,” International Journal of Dermatology, vol.42,no.6, pp.468-469,2009.

3        L.L.Bushkell and R.J.Gorlin, “leukonychia totalis, multiple sebaceous cysts,and renal calculi. A syndrome,” Archives of Dermatology,vol.111,no.7,  pp. 899-901,1975.

4        R.S. Medansky and J.M. Fox , “Hereditary Leukonychia totalis,”Archives of Dermatology, vol.82,  no.3, pp.412-414,1960.

5        R.S. Bart and R.E Pumphrey, “knuckle pads,leukonychia and deafness. Adominantly  inherited  syndrome, ” The New England Journal of  Medicine, vol. 276,  no. 4, pp. 202-207,1967.

6        K.R Ong, S. Visram,S. McKaig, and L.A.Brueton, “Sensorineural deafness,enamel abnormalities and nail abnormalities: a case report of Heimler  syndrome in identical twin girls,” European Journal of Medical Genetics,vol.49,no. 2, pp.187-193,2006.

7        R.A. Mee, “Een verschijnsel bij polyneuritis arsenicasa,” Nederlands Tijdschrift voor Geneeskunde,vol.1,pp.391-396,1919

8        Maria Rita Bongiorno and Mario Arico,Idiopathic Acquired Leuconychia in 34-Year Old Patient : Case Report in Medicine , Volume 2009, Article ID 495809, 3pages ,doi:10.1155/2009/495809.

9        Milano A , and Bonifazi E, “Sporodic congenital leukonychia totalis .Two cases,  Eur. J. Pediat .Dermatol.18, 221-224,2008.

 

Received on 29.12.2012

Modified on

Accepted on

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Research J. Pharmacology and Pharmacodynamics. 5(5): September–October 2013, 289-290