Ethics statement

Ethical approval for this study was granted by the ethical review committees of Dr. Soetomo General Hospital (070/91/301.4.2/Litb/II/2019), the Leprosy Mission Trust India (5/vii/C-37), and London School of Hygiene and Tropical Medicine (17007). Participants gave written informed consent before the data collection began. Formal consent was obtained from the parent/guardian (including whether it was verbal or written) for those participants under 18 years old.

Characteristics of study participants

Sixty-six people affected by leprosy reactions were interviewed. Four Indonesian participants were related; they were two pairs of siblings. Most participants were men and married. Indian participants were slightly older than Indonesian participants. In Indonesia, most of the participants lived with their nuclear family, whereas in India half of the participants stayed with their extended family. Compared to Indonesian participants, there were more Indian participants who still worked while receiving treatment for leprosy reactions. All Indonesian participants were Muslim with diverse educational background. In contrast, the majority of Indian participants were Hindu and had not completed primary school. Table 1 provides more detailed demographic information of participants affected by leprosy reactions. The data saturation happened sooner for participants with T1R, than those with ENL which affects more organ systems and exhibits greater chronicity which makes it a more complex condition.

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Table 1. Demographic of participants affected by leprosy reactions in Indonesia and India.

https://doi.org/10.1371/journal.pntd.0010476.t001

Fourteen participants experienced T1R and 52 experienced ENL. For the majority (n = 42) the first episode started during the MDT course. The reactions commonly emerged on the visible body parts, such as hands, legs, or face. Thirty-nine percent of the Indonesian participants had experienced 2–4 episodes of leprosy reactions, whereas 42 percent of the Indian participants had experienced >4 episodes of leprosy reactions (Table 2).

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Table 2. Clinical Features of leprosy reactions of people affected in Indonesia and India.

https://doi.org/10.1371/journal.pntd.0010476.t002

In the FGDs, 16 Indonesian and 22 Indian family members participated representing 15 Indonesian and 17 Indian families, respectively. In Indonesia, the FGD participants were a parent (38%), spouse (31%), sibling (19%) or adult child (13%) of the participants with leprosy reactions. FGD participants in India were spouses (41%), parents (36%), adult child (18%), or sibling (5%) of the participants. In Indonesia, 12 (38%) and in India 5 (16%) FGD participants were women.

Descriptions of illness

In both countries, the participants with leprosy reactions used lay words to illustrate how their body felt when leprosy reactions occurred. Common terms used in Indonesia are “gringgingan” [tingling sensation], “crecep” [burning sensation], “kenyong kenyong” [swollen] and “cekot-cekot” [sore]. Meanwhile, the participants in West Bengal, India, describe the leprosy reactions as “Jinjin” [tingling sensation]. The ideophones from both countries refer to uncomfortable feelings experienced during leprosy reactions. The participants mentioned pain, fever and cutaneous swelling and some individuals in Indonesia reported nausea and vomiting. The range of symptoms experienced was confirmed during FGDs with family members and validation activities. Participants indicated confusion and anxiety associated with the emergence of leprosy reactions. Participants were able to distinguish between leprosy reactions and non-reactional leprosy due to the inflammatory symptoms described as swelling and burning sensation. The tingling sensation and pain motivated participants to either seek medical advice or to try to reduce the discomfort by using alternative treatments.

Timing of onset

Seventeen of sixty-six (25.75%) participants (seven Indonesians and ten Indians) developed leprosy reactions before leprosy was diagnosed and MDT started, 42 during MDT and 7 after MDT completion (Table 2). In two cases, reactions occurred long after they completed treatment; an Indonesian participant developed reactions more than 10 years and an Indian participant more than 14 years after completing MDT. The delay in onset of leprosy reactions is difficult to reconcile with the concept of cure of the disease.

“I have no idea whether I have been fully recovered or not… It has been ten years [since the first leprosy symptoms]. I started to have the reactions since 2017 after completing MDT. They are getting severer”. (Mustofa, ENL, Man, Teacher, 35 years old, Indonesia)

Perceptions of aetiology and risk factors of leprosy reactions

Indonesian and Indian participants in interviews and FGDs made a connection between strenuous physical activity (e.g., cultivating land, construction work but also sports like running) (n = 6), fatigue (n = 11) and stress (in particular worrying) (n = 6) and the onset of reaction symptoms in the interviews and the validation activities. Leprosy reactions were attributed to cold temperature by two Indonesians and junk food consumption by another. Indian participants associated the illness with insect bites (n = 1), bathing in an unhygienic pond (n = 1) or giving birth (n = 1) and two participants linked leprosy reactions with alcoholism.

“My whole body used to have burning sensation. My neighbours said that I had hathura, the local belief on sickness after giving birth.” (Gina, ENL, Woman, Housewife, 25 years old, India)

“If I carried a heavy load in the morning then the pain would start in the night. Then, the fever and tingling sensation will also come.” (Daksh, ENL, Man, Farmer, 21 years old, India)

“When I was exhausted after doing exercise like running, the nodules appeared.” (Arfan, ENL, Man, Civil Servant, 32 years old, Indonesia)

“The doctor said this is a spoiled/lazy [“ngalem”] person disease. It means that the pain relapsed if you were exhausted or thinking too much, but it would not appear if you were happy.” (Arfan, ENL, Man, Civil Servant, 32 years old, Indonesia)

Diagnosis of leprosy reactions

Seven Indonesian and ten Indian participants who developed leprosy reactions prior to receiving the diagnosis of leprosy often sought medical advice for the reactions and then learned they had leprosy complicated by leprosy reactions. An Indonesian participant assumed, in the interview, that delay in diagnosis of leprosy contributed to leprosy reactions. Delay in diagnosis of leprosy was mentioned often in the interviews. Twenty-eight participants (42%) said there was a delay of more than a year (see Table 1). Eleven Indonesian and fifteen Indian participants reported that the medical doctors at their local health care facilities initially made a diagnosis such as dermatitis or dermatophytosis or allergy. Leprosy reactions which developed following the diagnosis of leprosy and during MDT were diagnosed quickly and was used as a confirmation of the diagnosis of leprosy at health facilities.

“At first, I thought it was an allergy due to I like eating junk foods. Then, I had nodules and pus on my legs. A doctor in Kediri suggested me to visit leprosy hospital and I got MDT and a prescription of methylprednisolone [for treating the leprosy and leprosy reactions]”. (Bambang, ENL, Man, Unemployed, 27 years old, Indonesia]

Participants who developed leprosy reactions after completion of MDT reported delays in the diagnosis of the reactions. Health care professionals did not always inform the patients about the possibility of developing leprosy reactions, hence some participants delayed seeking care as they thought the condition was harmless and not related to leprosy. They only sought help when the symptoms worsened. Misdiagnosis at this stage by health professionals also occurred.

Impact on physical sphere

Direct and indirect effects of leprosy reactions on the physical body were identified. Bodily functions were directly affected by leprosy reactions and indirectly affected by the treatment of leprosy reactions. Problems with bodily functions had a negative effect on daily activities and participation and were reported by participants and family members.

The neuritic pain and burning sensations associated with leprosy reactions created difficulty with movement and muscle strength. Difficulties with moving arms, lifting legs, bending knees, or holding things were reported. The participants in both countries, confirmed by family members during the FGDs, said that fatigue contributed to problems with bodily functions. Most of the participants expressed the idea that symptoms worsened when they felt fatigued after performing strenuous activities. In some cases, bodily functions were severely affected.

“It was excruciating like ants bit my legs. I cried. I couldn’t get a good sleep. The pain was persistent” (Sinta, T1R, Woman, Housewife, 50 years old, Indonesia).

The problems with bodily functions created problems with daily activities like self-care, eating and household tasks but once the inflammatory symptoms settled levels of activity normalised.

“It was all swollen on my arms and legs. I could not even comb my hair”. (Lina, T1R, Woman, Student, 19 years old, Indonesia)

“When the pain relieved, I did all kind of work like cultivating the land or picking up water from the well. Yet, it was not possible to do these activities when the nodules come out along with the pain and fever. Due to this, I was not able to eat, sit and bend my knee.” (Chaarun, Woman, ENL, Farmer, 35 years old, India)

The type of leprosy reaction, the severity of the reaction, finance, and support from household members all influenced the extent of the impact within the physical sphere. Participants in both settings described the pain associated with ENL as more severe than that associated with T1R. The majority of participants with ENL in both countries were incapacitated (bed bound) when the ENL was at its most severe, whereas participants affected by T1R did not experience this level of incapacity due to pain.

Indirect effects of leprosy reactions on bodily functions were caused by medical therapy. The drugs to treat leprosy reactions caused adverse effects that influenced bodily functions according to participants. The most important adverse effects reported by participants were anaemia and skin discoloration. Half of the participants in Indonesia and India said in the interviews that they experienced anaemia during MDT and the treatment of leprosy reactions, and many of the individuals were taking iron tablets in addition to the treatment for leprosy reactions. Other adverse effects reported were increased blood glucose levels, abdominal pain and palpitations.

Impact on psychological sphere

Participants from both countries described the upset and surprise they experienced with the sudden changes that appeared on their hands, face, or legs. Some of participants became tearful when they were asked about the experience of having leprosy reactions during the interview. Participants expressed a wide range of negative emotions and feelings including sadness, fear, despair, anxiousness, anger, and confusion. The family members during the FGDs confirmed this initial shock and reported range of feelings. The participants said that they were optimistic only when the treatment reduced the severity of reactional skin lesions and the burning sensations. Participants with ENL reported more challenges and concerns regarding their illness due to the direct and indirect physical effect of leprosy reactions, and there was a bigger impact on their psychological well-being than on those participants with T1R who experienced less physical discomfort and no incapacitating pain. The range of feelings reported were elicited by several factors; the six main ones were appearance of leprosy reactions, limited activities, unpredictability of the reaction episodes, slow progress of treatment, fear of death and concerns about polypharmacy.

The appearance of leprosy reactions frightened those affected. The fear pushed participants to stigmatize themselves and conceal their symptoms. Most participants said that only their nuclear family knew about their leprosy reactions. In general, we saw that Indonesian participants feared the condition more than Indian participants. The majority of those who were frightened discussed this with their healthcare provider, who tried to give medical reassurance by saying that eventually the skin will be normal again. The continuous burning sensation which limits activities and restricts participation predisposed individuals to negative feelings.

“I used to cry with pain, fever and my weakness.” (Baasim, ENL, Man, Hard Labor, 35 years old, India)

The lack of predictability of the condition and its episodes were reported by twelve Indonesian and ten Indian participants. They described that the onset of reactions can happen in the morning, afternoon, or night. The participants postponed their work or school if the reactions happened during the day and had sleep deprivation if the reactions occurred at night. Five Indonesians and two Indians had new episodes of reactions years after they were discharged from outpatient care for their leprosy reactions. Two-thirds of Indonesian and Indian affected by multiple episodes of ENL expressed feelings of apathy.

“Sometimes, I wanted to be angry, but what could I do? To whom should I be angry?” (Bakri, ENL, Man, Unemployed, 24 years old, Indonesia)

Participants were anxious if the response to treatment was slow but if skin lesions and nerve pain improved, they felt optimistic. In India, the severity of ENL and prolonged requirement for treatment caused a participant to consider self-harm.

“Sometimes, I wanted to take poison and finish my life,” (Raghu, ENL, Man, Farmer, 64 years old, India)

From the interviews, we found that three Indonesian participants and five Indian participants expressed a sense of hopelessness and felt that the leprosy reactions affecting them were incurable. Due to the regular episodes of nerve pain, they started to doubt the effectiveness of the treatment.

“I was frustrated of visiting several hospitals and consuming many kinds of medicines. Yet, these gave no improvement in my health.” (Madhavi, ENL, Woman, Housewife, 40 years old, India)

“I am discouraged because I cannot be recovered from the treatment. Could you imagine? I have been treated for more than three years.” (Bima, ENL, Man, Unemployed, 29 years old, Indonesia).

A few participants in both countries were worried that they might die because of leprosy reactions. An Indonesian and two Indian informants explained that fear of death was associated with severe skin lesions.

“Sometimes, I feel that this is my end, and I am not going to live anymore.” (Madhavi, ENL, Woman, Housewife, 40 years old, India)

Although Indian participants worried about loss of income and separation from their family, six were optimistic about their treatment in the specialist leprosy hospital because they experienced improvement and they encountered less challenges due to hospitalisation than Indonesian participants with severe leprosy reactions who were treated on an outpatient basis.

A lack of information, incomplete information, and inconsistent messages from health professionals about the prognosis of leprosy reactions generated anxiety and despair.

“A doctor previously told that this [leprosy reactions] could be cured completely. But the other days, another doctor explained that full recovery is not possible.” (Lina, T1R, Woman, Student, 19 years old, Indonesia)

The anxiety due to lack of information about their illness lead four Indonesian participants to seek information from the internet and five others consulted with health professionals through WhatsApp messaging or phone calls. Only one Indian participant used the internet to find information about leprosy reactions and the others relied on their doctors for information about their illness.

Participants in both countries also perceived that taking multiple medications might be harmful such as causing renal damage. The people affected by T1R or ENL reported being prescribed at least three types of drugs to treat their leprosy reactions. In addition to oral corticosteroids, analgesics, iron, and vitamin supplements were prescribed. High doses of oral corticosteroids would be divided. Participants said they applied emollients to dry or scaly skin. The concern about the number of drugs prescribed had a negative impact on psychological state of the participants in both countries.

Impact on social sphere

Leprosy reactions affected the social sphere, either directly or through physical and psychological changes. Impact in the social sphere also affected the physical and psychological spheres. Three main themes emerged: relationship with the individual’s family, stigmatization and socio-economic situation.

The impact of leprosy reactions was felt by the families of the persons affected. Almost all people affected by leprosy reactions, at some point, were housebound and had no choice but to rely on their spouse, parents, or children. During the FGDs with family members and the validation activities with participants, we learned that the family members did not understand what leprosy reactions were and that they referred to reactions as leprosy. The common response of the family members during the FGDs was “why are you not recovered yet?” and some family members thought the persons affected used reactions as an excuse to not participate in daily activities. In one case, an Indonesian man with leprosy reactions was left by his wife and son due to this issue. Most nevertheless were willing to support their family member affected by leprosy reactions.

From the interviews and FGDs we learned that about one third of the persons affected in our study were dependent on family members with daily activities such as eating, washing, and using the lavatory when the reactions were severe. Family members in both countries also assisted the persons affected while visiting healthcare facilities, purchased medicines, and encouraged them to adhere to the treatment. Five Indonesian individuals used complementary therapies–such as Jamu, a traditional medicine prepared from herbs and may consist of turmeric and/or ginger–in addition to the allopathic treatment prescribed. The five participants reported that Jamu helped them to feel better. Complementary therapies were less commonly used by Indian participants. In some cases, family members also supported persons affected financially. In a few cases, family members did not support the person affected. Bima was asked by his wife, with whom he has a child, to move back to his parents because he could no longer provide financial support for the family.

Interview participants described a difference between leprosy-related stigma and stigma secondary to leprosy reactions and that stigmatization impacted on the physical and psychological sphere. In some cases, leprosy signs had not been noticeable, or the participants had been able to conceal them, but the leprosy reaction associated skin nodules were in most cases obvious to others. Self-stigma related to skin discoloration due to consumption of clofazimine for treating ENL was reported by five Indonesians and three Indians. Participants mentioned the recurrent feelings of inability to discharge their roles and responsibilities in their family and society. The visibility of symptoms and sense of inability to accomplish their work resulted in feelings of shame or being less worthy. In India, a woman with leprosy reactions recounted that her mother-in-law often cursed her because of the visible skin nodules and her incapacity to manage household chores because of her “weak body”. In Indonesia, multiple individuals with visible nodules said that many of their neighbours avoided them and even encouraged others to do the same. Participants were stigmatized when using public transport.

“When you walk, people start to move away from you, that just hurtful… In public transport, sometimes people moved away from me. What else could I do? I had this disease, and it’s just not possible to take public transport by myself with no one around”. (Sinta, T1R, Woman, Housewife, 50 years old, Indonesia)

The stigmatization appeared to be more severe in Indonesia where most participants said they were advised to conceal of the signs of reactions by healthcare providers to reduce stigmatization and its negative effects. Some participants wore long clothes and masks to conceal skin nodules and wounds. One third of participants felt it was advantageous that the signs of leprosy reaction were not associated with leprosy by lay people and that this reduced the negativity they experienced. Affected individuals used words such as “dirty blood” [darah kotor], “black magic” [guna-guna], “allergy”, “diabetes”, “liver problem” to disguise the name of the condition. Eighteen Indonesian participants considered this was an effective concealment strategy because leprosy reactions were rarely found in their neighbourhood. Several participants in Indonesia explained that some caring neighbours and friends attributed the symptoms to food allergy and suggested the avoidance of certain types of food such as chicken and eggs. One person told us “I complied, and then I became anaemic” (Tirta, ENL, Man, Unemployed, 66 years old, Indonesia). In participants’ perception, the disguise and concealment reduced social isolation and rejection by others. The strategy prevented more questions about leprosy and, attributing the problem to allergy or another skin disease, resulted in sympathy and support from their neighbours or colleagues. Stigma was also reported by Indian participants, but some of them had no qualms disclosing their leprosy diagnosis and experience of leprosy reactions because several neighbours had been similarly affected by leprosy. They reported good support from their family members and peers in seeking effective treatment.

The participants in Indonesia and India experienced loss of income and increased expenditure on medication and transportation which created a financial burden for them and their families. This was confirmed during the FGDs and the validation activities. Participants from both countries with income less than 60 USD per month explained that they ate less nutritious food, were late with taking medicine, and forced themselves to work when it felt better not to. This complex situation led to anxiety which, in their point of view, further triggered the leprosy reactions and negatively influence their bodily functions. The financial hardship experienced by the participants affected the physical and psychological sphere. In both countries, most participants lost opportunities to earn money and pursue education due to severe pain. Some male participants in Indonesia and India said that their wives started working because they were not capable of doing so. Others continued working despite the pain and discomfort because they had no alternative. Some participants had to borrow money from their extended family or neighbours.

“Due to this [leprosy reactions], I resigned my job.” (Rutva, ENL, Man, Self-employed, 38 years old, India)

“I had to work with pain. As I am alone to do all of these works, I had no choice. Honestly, I felt very bad and painful.” (Sajan, ENL, Man, Hard Laborer, 35 years old, India)

“I used to borrow money from my neighbour for the treatment.” (Baasim, ENL, Man, Hard Laborer, 35 years old, India)

The treatment of leprosy reactions, especially ENL, lasted for several months or years and required frequent consultations with healthcare providers. Many of those affected by ENL were advised to take bed rest.

Participants were anxious about the direct and indirect expenses of the treatment. Indonesian individuals without health insurance or those who were treated at a non-charitable hospital in India had to pay for treatment. Travel costs were also incurred. The medical care required was often located in tertiary level or a leprosy-specialised hospital increasing travel costs and expenditure on food and beverages for individuals and any accompanying persons. A man affected by ENL from Indonesia and another from India said that treatment was occasionally delayed because of financial hardship.

Ten Indonesian and twenty-three Indian participants reported that leprosy reactions and their treatment impacted their financial independence and ability to fulfil their responsibility as a breadwinner in the household. These participants were unemployed and could barely afford nutritious food while the leprosy reactions were being treated.