• View in gallery

    Ill-defined, edematous, soft, nontender plaque of size 3.75 cm × 1.5 cm over the right side of the upper lip. This figure appears in color at www.ajtmh.org.

  • View in gallery

    Multiple noncaseating epithelioid granulomas with lymphohistiocytic infiltrates and Langhans giant cells, around the adnexa and neural bundles of dermis (H&E, ×10 magnification). This figure appears in color at www.ajtmh.org.

  • View in gallery

    Fite-Faraco stain showing occasional bacilli (arrow), with a bacillary index of 1+ (×100 magnification). This figure appears in color at www.ajtmh.org.

  • 1.

    Rao R, Kaur GJ, Rao AC, Chandrashekar B, Rao L, Handattu S, 2013. Borderline leprosy masquerading as cheilitis granulomatosa: a case report. Lepr Rev 84: 9599.

    • Search Google Scholar
    • Export Citation
  • 2.

    Pallagatti S, Sheikh S, Kaur A, Aggarwal A, Singh R, 2012. Oral cavity and leprosy. Indian Dermatol Online J 3: 101104.

  • 3.

    Van der Waal R, Shulten E, Van de Scheur MR, Wauters I, Starink TM, Van der Waal I, 2001. Cheilitis granulomatosa. J Eur Acad Dermatol Venereol 15: 519523.

    • Search Google Scholar
    • Export Citation
  • 4.

    Błochowiak KJ et al.2018. Selected presentations of lip enlargement: clinical manifestation and differentiation. Adv Dermatol Allergol 35: 1825.

    • Search Google Scholar
    • Export Citation
  • 5.

    Handa S, Saraswat A, Radotra BD, Kumar B, 2003. Chronic macrocheilia: a clinico-pathological study of 28 patients. Clin Exp Dermatol 28: 245250.

 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 
 

 

 

 

Borderline Tuberculoid Hansen Disease Presenting as Chronic Macrocheilia

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  • 1 Department of Dermatology, Venereology and Leprosy, Sri Manakula Vinayagar Medical College and Hospital, Pondicherry, India

A 40-year-old woman presented with an ill-defined, edematous, soft, nontender plaque of size 3.75 cm × 1.5 cm over the right side of upper lip (Figure 1). The patient was asymptomatic, except for mild paresthesia. The lesion had an insidious onset, with gradual progression over 3 years. There was no history of any injections or trauma to the lips. Her past medical and family history was unremarkable. There were no other significant findings in skin and oral mucosa. Sensation over the lesion was preserved. Neurological examination did not reveal any nerve thickening. Systemic examination was found to be normal.

Figure 1.
Figure 1.

Ill-defined, edematous, soft, nontender plaque of size 3.75 cm × 1.5 cm over the right side of the upper lip. This figure appears in color at www.ajtmh.org.

Citation: The American Journal of Tropical Medicine and Hygiene 106, 1; 10.4269/ajtmh.21-0632

A differential diagnosis of leprosy and cutaneous tuberculosis was considered because of their endemicity in our region. Mantoux test was negative. A punch biopsy was taken from the lesion. In the dermis, multiple noncaseating epithelioid granulomas admixed with lymphohistiocytic infiltrates and Langhans giant cells were seen around the adnexa and neural bundles (Figure 2). The Fite-Faraco stain (special stain for Mycobacterium leprae) was positive, with a bacillary index of 1+ (Figure 3). Based on the clinico-histopathological correlation, a diagnosis of Borderline Tuberculoid Hansen was made. The patient was started on WHO multibacillary-multidrug therapy (MB-MDT). The patient showed partial resolution of the swelling after 6 months of treatment and is under follow-up.

Figure 2.
Figure 2.

Multiple noncaseating epithelioid granulomas with lymphohistiocytic infiltrates and Langhans giant cells, around the adnexa and neural bundles of dermis (H&E, ×10 magnification). This figure appears in color at www.ajtmh.org.

Citation: The American Journal of Tropical Medicine and Hygiene 106, 1; 10.4269/ajtmh.21-0632

Figure 3.
Figure 3.

Fite-Faraco stain showing occasional bacilli (arrow), with a bacillary index of 1+ (×100 magnification). This figure appears in color at www.ajtmh.org.

Citation: The American Journal of Tropical Medicine and Hygiene 106, 1; 10.4269/ajtmh.21-0632

The oral cavity involvement is about 20–60% in lepromatous leprosy whereas it is rare in tuberculoid and borderline tuberculoid spectrum of the disease.1 It is usually asymptomatic in nature with slow progression.2 The epithelioid granulomas in leprosy block the lymphatic channels, thereby producing swelling of the lips.1 It can present as microstomia, macrocheilia, or flat-topped nodules thereby producing severe cosmetic impairment.2

The differential diagnosis of chronic macrocheilia includes infections like cutaneous tuberculosis, Hansen disease, syphilis, leishmaniasis, rhinoscleroma, histoplasmosis, and post-odontogenic infections. The noninfectious differential diagnosis such as cheilitis granulomatosa, Melkersson– Rosenthal syndrome, sarcoidosis, Crohn’s disease, amyloidosis, and foreign body reaction should also be considered.3,4

Hansen disease solely presenting as chronic macrocheilia is extremely unusual.5 A high index of suspicion for Hansen disease is needed for any chronic macrocheilia in high-endemic areas.

REFERENCES

  • 1.

    Rao R, Kaur GJ, Rao AC, Chandrashekar B, Rao L, Handattu S, 2013. Borderline leprosy masquerading as cheilitis granulomatosa: a case report. Lepr Rev 84: 9599.

    • Search Google Scholar
    • Export Citation
  • 2.

    Pallagatti S, Sheikh S, Kaur A, Aggarwal A, Singh R, 2012. Oral cavity and leprosy. Indian Dermatol Online J 3: 101104.

  • 3.

    Van der Waal R, Shulten E, Van de Scheur MR, Wauters I, Starink TM, Van der Waal I, 2001. Cheilitis granulomatosa. J Eur Acad Dermatol Venereol 15: 519523.

    • Search Google Scholar
    • Export Citation
  • 4.

    Błochowiak KJ et al.2018. Selected presentations of lip enlargement: clinical manifestation and differentiation. Adv Dermatol Allergol 35: 1825.

    • Search Google Scholar
    • Export Citation
  • 5.

    Handa S, Saraswat A, Radotra BD, Kumar B, 2003. Chronic macrocheilia: a clinico-pathological study of 28 patients. Clin Exp Dermatol 28: 245250.

Author Notes

Address correspondence to Palaniappan Vijayasankar, Department of Dermatology, Venereology and Leprosy, Sri Manakula Vinayagar Medical College and Hospital, Pondicherry 605107, India. E-mail: vijayasankarpalaniappan@gmail.com

Authors’ addresses: Anusuya Sadhasivamohan, Palaniappan Vijayasankar, and Kaliaperumal Karthikeyan, Department of Dermatology, Venereology and Leprosy, Sri Manakula Vinayagar Medical College and Hospital, Pondicherry, India, E-mails: dranu2302@gmail.com, vijayasankarpalaniappan@gmail.com, and karthikderm@gmail.com.

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